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Inoue Y, Kitoh R, Satoh M, Yoshigae Y, Nibe K, Uchida K, Matsunaga S. Epidural Inflammatory Pseudotumor in the Cervical Spine: A Case Report of a Bernese Mountain Dog. Animals (Basel) 2025; 15:1049. [PMID: 40218441 PMCID: PMC11988174 DOI: 10.3390/ani15071049] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/16/2025] [Revised: 03/28/2025] [Accepted: 03/31/2025] [Indexed: 04/14/2025] Open
Abstract
A 3-year, 9-month-old Bernese Mountain dog presented with a 10-day history of acute right hemiparesis. Neurological examination indicated abnormalities in the C1-C5 spinal segments. Magnetic resonance imaging (MRI) revealed an epidural mass at C3-C4, which was surgically removed after dorsal laminectomy. The neurological signs were completely resolved by the 12th postoperative day. The lesion was diagnosed as an inflammatory pseudotumor on histopathological examination. Another MRI performed 63 days after surgery showed no lesion regrowth, and there has been no recurrence for approximately 6 years. Reports of inflammatory pseudotumors are rare, and their treatment remains unclear. Previous reports and the course of this case suggest that a good prognosis may be achieved if complete surgical resection of an inflammatory pseudotumor arising in the epidural region can be accomplished.
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Affiliation(s)
- Yoshiyuki Inoue
- Japan Animal Referral Medical Center, Kawasaki 213-0032, Japan; (R.K.); (M.S.); (Y.Y.); (S.M.)
- Laboratory of Veterinary Surgery, Faculty of Agriculture, University of Miyazaki, Miyazaki 889-2192, Japan
| | - Rie Kitoh
- Japan Animal Referral Medical Center, Kawasaki 213-0032, Japan; (R.K.); (M.S.); (Y.Y.); (S.M.)
| | - Moe Satoh
- Japan Animal Referral Medical Center, Kawasaki 213-0032, Japan; (R.K.); (M.S.); (Y.Y.); (S.M.)
| | - Yuki Yoshigae
- Japan Animal Referral Medical Center, Kawasaki 213-0032, Japan; (R.K.); (M.S.); (Y.Y.); (S.M.)
| | - Kazumi Nibe
- FUJIFILM VET Systems, Tokyo 185-0013, Japan;
| | - Kazuyuki Uchida
- Laboratory of Veterinary Pathology, Graduate School of Agricultural and Life Sciences, The University of Tokyo, Tokyo 113-8657, Japan;
| | - Satoru Matsunaga
- Japan Animal Referral Medical Center, Kawasaki 213-0032, Japan; (R.K.); (M.S.); (Y.Y.); (S.M.)
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Dadson P, Ngum P, Juarez-Orozco LE, Ntodie M, Loba P. The Relevance and Potential Role of Orbital Fat in Inflammatory Orbital Diseases: Implications for Diagnosis and Treatment. Ophthalmol Ther 2025; 14:247-281. [PMID: 39680323 PMCID: PMC11754589 DOI: 10.1007/s40123-024-01079-7] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/12/2024] [Accepted: 11/22/2024] [Indexed: 12/17/2024] Open
Abstract
The orbit is an important structure within the skull that houses the eye, optic nerve, and extraocular muscles. It also contains adipose/fat tissue, which provides a protective cushion for these components. Inflammatory orbital disease can affect any or all components of the orbit, often arising from various underlying pathologic conditions, including autoimmune, infectious, and vascular diseases. Typical signs and symptoms of orbital inflammation include swelling, redness, pain, discomfort, and potential loss of function. The role of orbital fat in the pathogenesis of inflammatory orbital diseases has not been fully explored. This review aims to provide a comprehensive description of orbital fat, its relevance and the potential role in inflammatory diseases of the orbit, and the use of radiologic imaging studies for evaluating this fat depot in cases of as inflammatory orbital diseases. Additionally, this review discusses the various procedures available for the treatment and management of these conditions. A range of interventions, including pharmacotherapy and surgical procedures, will be evaluated as promising therapeutic options. This review also explores the characteristics and potential applications of orbital fat-derived stem cells, with an emphasis on their regenerative abilities and anti-inflammatory effects. Understanding the role of orbital fat and its contribution to inflammatory orbital diseases is essential for optimizing diagnostic and treatment strategies.
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Affiliation(s)
- Prince Dadson
- Turku PET Centre, University of Turku, Kiinamyllynkatu 4-8, 20520, Turku, Finland.
- Turku PET Centre, Turku University Hospital, Kiinamyllynkatu 4-8, 20520, Turku, Finland.
| | - Peter Ngum
- Turku Brain Injury Center, Turku University Hospital and University of Turku, Turku, Finland
| | - Luis Eduardo Juarez-Orozco
- Turku PET Centre, University of Turku, Kiinamyllynkatu 4-8, 20520, Turku, Finland
- Turku PET Centre, Turku University Hospital, Kiinamyllynkatu 4-8, 20520, Turku, Finland
- Division Heart and Lungs, Department of Cardiology, University Medical Center Utrecht, Utrecht University, Utrecht, The Netherlands
| | - Michael Ntodie
- Centre for Optometry and Vision Science, Biomedical Sciences Research Institute, Ulster University, Coleraine, UK
| | - Piotr Loba
- Department of Binocular Vision Pathophysiology and Strabismus, Medical University of Lodz, University Barlicki Hospital No.1, Kopcinskiego Street 22, 90-153, Lodz, Poland.
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Lasrado AS, Chattannavar G, Jakati S, Mohamed A, Kekunnaya R. Orbital Myositis and Strabismus: Clinical Profile, Management, and Predictive Factors for Recurrence. J Pediatr Ophthalmol Strabismus 2025; 62:57-66. [PMID: 39465589 DOI: 10.3928/01913913-20240926-04] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 10/29/2024]
Abstract
PURPOSE To study the clinical profile of patients with orbital myositis and their management modalities and predictive factors for recurrence. METHODS The records of all consecutive patients diagnosed as having orbital myositis from 2010 to 2022 were reviewed. Patient demographics, presenting complaints, visual function, primary gaze deviation, and extraocular muscle involved were studied in the clinical profile. The radiological modalities and their management along with the recovery status and need for surgical intervention were documented. RESULTS Fifty-two patients (55 eyes) (69% women) were diagnosed as having orbital myositis with a mean age of 36 ± 16 years presenting with periocular pain (55%) followed by diplopia (49%). Disease was unilateral in 94%. Sixty percent had ocular misalignment in primary gaze. The medial rectus was the most involved muscle (58%), with abduction limitation in 73%. Computed tomography (42%), magnetic resonance imaging (42%), and combined imaging (16%) were used in localizing the disease. Muscle biopsy was done in 38% of patients. Systemic immunomodulators were required in 16%. Strabismus surgery was planned in 15% of patients and done in 11%. Recurrence was noted in 43%. Abrupt cessation of steroids was a significant risk factor in recurrence of the disease (P = .046). Recovery was partial in 44% of patients and complete in 47%. CONCLUSIONS Systemic steroids with slow taper has satisfactory outcomes in orbital myositis. Patients refractory to treatment ideally should undergo biopsy and begin taking steroid-sparing agents. Once the disease is in remission, residual disease can be surgically treated for better functional outcome. [J Pediatr Ophthalmol Strabismus. 2025;62(1):57-66.].
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Guo S, Wang S, Chen C, He X, Yang B, Huang Z, Ma J, Shi J, Jiang L, Chen FK. Inflammatory Myofibroblastic Tumor of the Orbit: A Case Series and Literature Review. J Inflamm Res 2024; 17:11029-11039. [PMID: 39697794 PMCID: PMC11653865 DOI: 10.2147/jir.s485499] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/18/2024] [Accepted: 11/21/2024] [Indexed: 12/20/2024] Open
Abstract
Purpose Orbital inflammatory myofibroblastic tumors (IMTs) are a rare tumor with intermediate biological potential. We analyzed a series of orbital IMTs to determine their unique features. Methods Records from patients with pathologically confirmed IMT at Beijing Tongren Hospital, Capital Medical University, between January 2004 and August 2022, were reviewed for their ocular presentation and treatment outcomes. Results Thirteen cases of primary orbital IMT with positive anaplastic lymphoma kinase on fluorescent in situ hybridization were included. These consists of five men and eight women with an age of onset ranged from 2 to 54 years. Nearly half of the primary orbital IMTs displayed local invasion into the maxillary sinus, ethmoid sinus, and pterygopalatine fossa. On magnetic resonance imaging, orbital IMT usually appeared as an ill-defined, oval, irregular, or diffuse mass with heterogeneous signals. Nearly half (46.2%) of these lesions caused bone destruction, and 31% developed recurrence, which was more likely to occur in lesions involving multiple tissues (50%). All cases of recurrence occurred within five months after the first surgery. No patient developed distant metastases. Conclusion Orbital IMT is rare and prone to local invasion and recurrence. The histology and behavior of orbital IMT requires further investigation.
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Affiliation(s)
- Sitong Guo
- Department of Ophthalmology, Beijing Friendship Hospital, Capital Medical University, Beijing, 100050, People’s Republic of China
- Beijing Tongren Eye Center, Beijing Tongren Hospital, Capital Medical University; Beijing Ophthalmology and Visual Sciences Key Laboratory, Beijing, 100730, People’s Republic of China
| | - Songze Wang
- Beijing Tongren Eye Center, Beijing Tongren Hospital, Capital Medical University; Beijing Ophthalmology and Visual Sciences Key Laboratory, Beijing, 100730, People’s Republic of China
- Beijing FengTai You’anmen Hospital, Beijing, People’s Republic of China
| | - Chunli Chen
- Beijing Tongren Eye Center, Beijing Tongren Hospital, Capital Medical University; Beijing Ophthalmology and Visual Sciences Key Laboratory, Beijing, 100730, People’s Republic of China
| | - Xiaojin He
- Department of Pathology, Beijing Tongren Hospital, Capital Medical University, Beijing, 100730, People’s Republic of China
| | - Bentao Yang
- Department of Radiology, Beijing Tongren Hospital, Capital Medical University, Beijing, 100730, People’s Republic of China
| | - Zhiqin Huang
- Beijing Tongren Eye Center, Beijing Tongren Hospital, Capital Medical University; Beijing Ophthalmology and Visual Sciences Key Laboratory, Beijing, 100730, People’s Republic of China
| | - Jianmin Ma
- Beijing Tongren Eye Center, Beijing Tongren Hospital, Capital Medical University; Beijing Ophthalmology and Visual Sciences Key Laboratory, Beijing, 100730, People’s Republic of China
| | - Jitong Shi
- Beijing Tongren Eye Center, Beijing Tongren Hospital, Capital Medical University; Beijing Ophthalmology and Visual Sciences Key Laboratory, Beijing, 100730, People’s Republic of China
| | - Libin Jiang
- Beijing Tongren Eye Center, Beijing Tongren Hospital, Capital Medical University; Beijing Ophthalmology and Visual Sciences Key Laboratory, Beijing, 100730, People’s Republic of China
| | - Fred Kuanfu Chen
- Centre for Ophthalmology and Visual Science, The University of Western Australia, Perth, WA, Australia
- Ophthalmology, Department of Surgery, University of Melbourne, Melbourne, VIC, Australia
- Department of Ophthalmology, Royal Perth Hospital, Perth, WA, Australia
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Faraj C, Imrani K, Essetti S, Chait F, Lahlou C, M Billah N, Nassar I, A Babana A, O Touhami Y, El Malki HO, Ifrine L, Belkouchi A, Belkouchi O, El Mohtarim R, Derqaoui S, Bernoussi Z. Mesenteric inflammatory pseudotumor: A rare case report and review of the literature. Radiol Case Rep 2024; 19:4945-4949. [PMID: 39247479 PMCID: PMC11378093 DOI: 10.1016/j.radcr.2024.07.124] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/09/2024] [Revised: 07/18/2024] [Accepted: 07/21/2024] [Indexed: 09/10/2024] Open
Abstract
Inflammatory pseudotumor (IP) is a rare type of benign tumor. Although initially identified in the lung, it has now been identified in a number of somatic and visceral sites, but mesenteric presentation is uncommon and has a variable clinical presentation. As inflammatory pseudotumor mimic malignancy both clinically and radiologically, the radiologist should be familiar with this entity. The only effective treatment is complete surgical resection. We present the case of a 55-year-old woman who presented with chronic abdominal pain and was diagnosed with a mesenteric inflammatory pseudotumor, in an attempt to illustrate the different imaging aspects of this benign condition in ultrasound, computed tomography and magnetic resonance imaging, and to simplify the description of these tumors.
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Affiliation(s)
- Chaymae Faraj
- Department of Radiology, Ibn Sina Hospital, Faculty of medicine and pharmacy, Mohammed V University, Rabat, Morocco
| | - Kaoutar Imrani
- Department of Radiology, Ibn Sina Hospital, Faculty of medicine and pharmacy, Mohammed V University, Rabat, Morocco
| | - Sara Essetti
- Department of Radiology, Ibn Sina Hospital, Faculty of medicine and pharmacy, Mohammed V University, Rabat, Morocco
| | - Fatima Chait
- Department of Radiology, Ibn Sina Hospital, Faculty of medicine and pharmacy, Mohammed V University, Rabat, Morocco
| | - Chaimae Lahlou
- Department of Radiology, Ibn Sina Hospital, Faculty of medicine and pharmacy, Mohammed V University, Rabat, Morocco
| | - Nabil M Billah
- Department of Radiology, Ibn Sina Hospital, Faculty of medicine and pharmacy, Mohammed V University, Rabat, Morocco
| | - Ittimade Nassar
- Department of Radiology, Ibn Sina Hospital, Faculty of medicine and pharmacy, Mohammed V University, Rabat, Morocco
| | - Amina A Babana
- Department of Surgery « A », Ibn Sina Hospital, Faculty of medicine and pharmacy, Mohammed V University, Rabat, Morocco
| | - Youssef O Touhami
- Department of Surgery « A », Ibn Sina Hospital, Faculty of medicine and pharmacy, Mohammed V University, Rabat, Morocco
| | - Hadj Omar El Malki
- Department of Surgery « A », Ibn Sina Hospital, Faculty of medicine and pharmacy, Mohammed V University, Rabat, Morocco
| | - Lahcen Ifrine
- Department of Surgery « A », Ibn Sina Hospital, Faculty of medicine and pharmacy, Mohammed V University, Rabat, Morocco
| | - Abdelkader Belkouchi
- Department of Surgery « A », Ibn Sina Hospital, Faculty of medicine and pharmacy, Mohammed V University, Rabat, Morocco
| | - Omar Belkouchi
- Department of Surgery « A », Ibn Sina Hospital, Faculty of medicine and pharmacy, Mohammed V University, Rabat, Morocco
| | - Rihane El Mohtarim
- Department of Pathology, Ibn Sina Hospital, Faculty of medicine and pharmacy, Mohammed V University, Rabat, Morocco
| | - Sabrine Derqaoui
- Department of Pathology, Ibn Sina Hospital, Faculty of medicine and pharmacy, Mohammed V University, Rabat, Morocco
| | - Zakiya Bernoussi
- Department of Pathology, Ibn Sina Hospital, Faculty of medicine and pharmacy, Mohammed V University, Rabat, Morocco
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Kakuguchi W, Ashikaga Y, Moritani Y, Nakano S, Ogawa N, Yoshitatsu R, Yanagawa-Matsuda A, Maishi N, Kudo A, Okazaki N, Nakamaru Y, Yabe I, Matsuno Y, Ohiro Y. Nonspecific inflammatory pseudotumor of the maxillary and temporal fossa: a study of seven cases. Oral Surg Oral Med Oral Pathol Oral Radiol 2024; 138:494-501. [PMID: 38981814 DOI: 10.1016/j.oooo.2024.06.002] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/03/2024] [Revised: 05/25/2024] [Accepted: 06/01/2024] [Indexed: 07/11/2024]
Abstract
OBJECTIVE Inflammatory pseudotumor (IPT) is a rare, locally aggressive, benign neoplasm of unknown etiology. Because of its aggressive clinical behavior and locally destructive or infiltrative features, it may be mistaken for a malignant tumor. Approximately 5%-44% of extrapulmonary IPT occur in the head and neck, primarily affecting the orbit. STUDY DESIGN Between 2008 and 2021, our hospital received referrals for seven patients (three men and four women, aged 42-73 years) with pain, swelling, mass, and trismus. Computed tomography, magnetic resonance imaging, and biopsy were performed on all patients to diagnose IPT. RESULTS Of the seven patients, four received low-dose prednisolone (PSL), one underwent surgery, and two were left untreated. The IPT disappeared in one of the two untreated cases, whereas it improved and later deteriorated in the other. The surgical patient had no recurrence. Low-dose PSL was effective in two patients; however, high-dose PSL and immunosuppressants were required in the remaining two cases owing to infiltration into each orbit or brain region. CONCLUSIONS Low-dose PSL treatment was applicable in IPT cases affecting the maxillary to temporal fossa region, wherein symptoms did not improve without treatment. However, when low-dose PSL was ineffective, high-dose PSL and immunosuppressants were required.
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Affiliation(s)
- Wataru Kakuguchi
- Department of Oral and Maxillofacial Surgery, Division of Oral Pathobiological Science, Faculty of Dental Medicine and Graduate School of Dental Medicine, Hokkaido University, Sapporo, Japan.
| | - Yuichi Ashikaga
- Department of Oral and Maxillofacial Surgery, Division of Oral Pathobiological Science, Faculty of Dental Medicine and Graduate School of Dental Medicine, Hokkaido University, Sapporo, Japan
| | - Yasuhito Moritani
- Department of Oral and Maxillofacial Surgery, Division of Oral Pathobiological Science, Faculty of Dental Medicine and Graduate School of Dental Medicine, Hokkaido University, Sapporo, Japan; Department of Dentistry and Oral Surgery, Sapporo City General hospital, Sapporo, Japan
| | - Shintaro Nakano
- Department of Oral and Maxillofacial Surgery, Division of Oral Pathobiological Science, Faculty of Dental Medicine and Graduate School of Dental Medicine, Hokkaido University, Sapporo, Japan
| | - Naohiro Ogawa
- Department of Oral and Maxillofacial Surgery, Division of Oral Pathobiological Science, Faculty of Dental Medicine and Graduate School of Dental Medicine, Hokkaido University, Sapporo, Japan; Department of Dentistry and Oral Surgery, Azabu Kitami Triology Hospital, Kitami, Japan
| | - Rieko Yoshitatsu
- Department of Oral and Maxillofacial Surgery, Division of Oral Pathobiological Science, Faculty of Dental Medicine and Graduate School of Dental Medicine, Hokkaido University, Sapporo, Japan
| | - Aya Yanagawa-Matsuda
- Department of Vascular Biology and Molecular Pathology, Division of Oral Pathobiological Science, Faculty of Dental Medicine and Graduate School of Dental Medicine, Hokkaido University, Sapporo, Japan
| | - Nako Maishi
- Department of Vascular Biology and Molecular Pathology, Division of Oral Pathobiological Science, Faculty of Dental Medicine and Graduate School of Dental Medicine, Hokkaido University, Sapporo, Japan
| | - Akihiko Kudo
- Department of Neurology, Faculty of Medicine and Graduate School of Medicine, Hokkaido University, Sapporo, Japan
| | - Nanase Okazaki
- Depaetment of Surgical Pathology, Hokkaido University Hospital, Sapporo, Japan
| | - Yuji Nakamaru
- Department of Otolaryngology-Head and Neck Surgery, Faculty of Medicine and Graduate School of Medicine, Hokkaido University, Sapporo, Japan
| | - Ichiro Yabe
- Department of Neurology, Faculty of Medicine and Graduate School of Medicine, Hokkaido University, Sapporo, Japan
| | - Yoshihiro Matsuno
- Depaetment of Surgical Pathology, Hokkaido University Hospital, Sapporo, Japan
| | - Yoichi Ohiro
- Department of Oral and Maxillofacial Surgery, Division of Oral Pathobiological Science, Faculty of Dental Medicine and Graduate School of Dental Medicine, Hokkaido University, Sapporo, Japan
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Luo Y, Qin X, Liu Y. Hepatic Inflammatory Pseudotumor on 18F-FDG and 18F-FAP-42 PET/CT. Clin Nucl Med 2024:00003072-990000000-01284. [PMID: 39325513 DOI: 10.1097/rlu.0000000000005456] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 09/27/2024]
Abstract
ABSTRACT Hepatic inflammatory pseudotumor is a rare benign disease. We describe the 18F-FDG and 18F-FAPI-42 PET/CT findings of hepatic inflammatory pseudotumor in a 57-year-old man. Compared with 18F-FDG, 18F-FAPI-42 PET/CT identified more lesions and showed more intense uptake within the lesions. This case illustrates that hepatic inflammatory pseudotumor should be contemplated in the differential diagnosis of liver lesions with increased 18F-FDG and 18F-FAPI-42 uptake. 18F-FAPI-42 PET/CT might be used as a helpful tool for evaluating hepatic inflammatory pseudotumor.
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Affiliation(s)
- Yingqi Luo
- From the Department of Nuclear Medicine, The Second Affiliated Hospital, Guangzhou Medical University, Guangzhou, China
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Ozenbas C, Karaarslan S, Gur AS. Coexistence of Pancreatic Neuroendocrine Tumor and Pseudotumor: Two Rare Lesions. Int J Surg Pathol 2024:10668969241272054. [PMID: 39275855 DOI: 10.1177/10668969241272054] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 09/16/2024]
Abstract
Neuroendocrine tumors and pseudotumors of the pancreas are 2 separate rare lesions. Neuroendocrine tumors originate from neuroendocrine cells, but many different factors have been suggested for the origin of pseudotumors. As the first reports of these 2 distinct entities found in a single patient have been published, our aim is to present the imaging and pathological characteristics of these entities.
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Affiliation(s)
- Cemre Ozenbas
- Department of Radiology, Private Buca Hospital, Tınaztepe University, İzmir, Turkey
| | - Serap Karaarslan
- Department of Pathology, Private Buca Hospital, Tınaztepe University, İzmir, Turkey
| | - Akif Serhat Gur
- Department of General Surgery, Private Buca Hospital, Tınaztepe University, İzmir, Turkey
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Reyes HM, Fiorentino E, Matiasek K, Menchetti M. Intradural-extramedullary inflammatory pseudotumour of the cervical spinal cord of a dog. Vet Radiol Ultrasound 2024; 65:513-517. [PMID: 38853369 DOI: 10.1111/vru.13384] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/16/2023] [Revised: 03/14/2024] [Accepted: 05/06/2024] [Indexed: 06/11/2024] Open
Abstract
An 8-year-old mixed-breed dog was presented with cervical hyperesthesia, tetraparesis, and mild proprioceptive ataxia in all four limbs. 3 Tesla MRI showed a dorsal compressive intradural-extramedullary mass at the level of C1-C2, isointense to the gray matter with a hypointense ventral core on T2 weighted images (WI), isointense on T1WI, with a strong and homogeneous contrast enhancement. A C1-C2 partial dorsal laminectomy was performed, and the lesion was removed en bloc. The histopathological and immunohistochemical analysis defined the diagnosis of inflammatory pseudotumor.
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Affiliation(s)
- Henry Mendo Reyes
- Division of Neurology and Neurosurgery, San Marco Veterinary Clinic and Laboratory, Padua, Italy
| | - Erica Fiorentino
- Division of Neurology and Neurosurgery, San Marco Veterinary Clinic and Laboratory, Padua, Italy
| | - Kaspar Matiasek
- Section of Clinical & Comparative Neuropathology, Centre for Clinical Veterinary Medicine, LMU, Munich, Germany
| | - Marika Menchetti
- Division of Neurology and Neurosurgery, San Marco Veterinary Clinic and Laboratory, Padua, Italy
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Ghumman H, Baradaran-Rafii G, Dadabhoy A, Ghumman U, Saad CG. A Rare Case of Adult-Onset Bilateral Nonspecific Orbital Inflammation (NSOI) With Concomitant Unilateral Third Cranial Nerve Palsy. Cureus 2024; 16:e67181. [PMID: 39161552 PMCID: PMC11333032 DOI: 10.7759/cureus.67181] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/30/2024] [Accepted: 08/18/2024] [Indexed: 08/21/2024] Open
Abstract
Nonspecific orbital inflammation (NSOI), also known as orbital pseudotumor, is a condition characterized by inflammation in the tissues around the eye socket (orbit) without a clearly identifiable cause. This inflammatory disorder can affect various structures within the orbit, including muscles, fat, and connective tissues, leading to symptoms such as pain, swelling, and changes in vision. A 74-year-old man with a history of previous orbital trauma presented with acute-onset head and orbital pain, followed by restricted left eye movements in all directions, left ptosis, and a dilated left pupil. Orbital imaging revealed bilateral inflammation of the lateral rectus muscles and orbital fat, suggestive of bilateral NSOI, while brain and laboratory studies ruled out other differential diagnoses. The presence of left ptosis, a dilated pupil, and limited upward, downward, and inward movements in the left eye suggested intraorbital involvement of both the superior and inferior divisions of the left third nerve. The complete resolution of orbital symptoms and third nerve function after systemic corticosteroid therapy supported the inflammatory nature of the nerve involvement in this case. The case is notable in terms of bilateral involvement in adult-onset NSOI, the possible role of previous orbital trauma in the development of the disease, and the inflammatory involvement of third nerve divisions following the extension of inflammation into the orbital tissues. NSOI can mimic other, more serious conditions, making accurate diagnosis crucial for effective management and treatment. Understanding its presentation, potential causes, and appropriate diagnostic approaches is essential in providing optimal care for patients affected by this complex inflammatory condition.
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Affiliation(s)
- Haider Ghumman
- Ophthalmology, University of South Florida (USF) Health Morsani College of Medicine, Tampa, USA
| | | | - Anosh Dadabhoy
- Ophthalmology, University of South Florida (USF) Health Morsani College of Medicine, Tampa, USA
| | - Ussama Ghumman
- Transplant Hepatology, University of Texas Health Science Center at San Antonio, San Antonio, USA
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11
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Anns KM, Salar M, Salar H, Khan F, Memon WA, Aman M, Zafar U, Minhas K, Zafar H, Shahid J. Duodenal stricture secondary to IgG4-related chronic sclerosing duodenitis-A case report with review of the literature. Clin Case Rep 2024; 12:e8980. [PMID: 38808195 PMCID: PMC11130227 DOI: 10.1002/ccr3.8980] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/23/2024] [Revised: 04/01/2024] [Accepted: 04/28/2024] [Indexed: 05/30/2024] Open
Abstract
Key Clinical Message This case highlights the importance of a definite diagnosis of an IgG4-related chronic sclerosing duodenitis based on histological and radiological findings to rule out any malignancy in the mass. While dealing with patients having concentric duodenal thickening resulting in stricture formation, one should think of inflammatory etiology as well. IgG4-related disease is one of these inflammatory disorders where we see soft tissue thickening without a large mass or any associated lymphadenopathy as in our case. Abstract Immunoglobulin G4-related disease (IgG4-RD) is distinguished as an infiltration of IgG-4-positive plasmacytes involving inflammatory lesions across multiple organs which is accompanied by raised IgG4 levels in the serum. Several inflammatory disorders are recognized as part of the IgG4-RD family based on shared histopathological features, which include Mikulicz's disease, chronic sclerosing sialadenitis, or Riedel's thyroiditis. Our case highlights a distinctive presentation of IgG4-related diseases; a 58-year-old man presenting with duodenal stricture highly suspicious of a duodenal mass/ampullary mass later found to be due to IgG4-related sclerosing duodenitis with negative malignancy on biopsy. We present the diagnostic challenges faced and relevant findings noted.
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Affiliation(s)
| | - Musa Salar
- Medical College, The Aga Khan UniversityKarachiPakistan
| | - Hashim Salar
- Medical College, The Aga Khan UniversityKarachiPakistan
| | | | - Wasim Ahmed Memon
- Department of RadiologyThe Aga Khan University HospitalKarachiPakistan
| | - Muhammad Aman
- Department of RadiologyThe Aga Khan University HospitalKarachiPakistan
| | - Uffan Zafar
- Department of RadiologyThe Aga Khan University HospitalKarachiPakistan
| | - Khurram Minhas
- Department of HistopathologyThe Aga Khan University HospitalKarachiPakistan
| | - Hasnain Zafar
- Department of General SurgeryThe Aga Khan University HospitalKarachiPakistan
| | - Jehanzeb Shahid
- Department of RadiologyThe Aga Khan University HospitalKarachiPakistan
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12
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Nomura S, Kaji M, Shiina N, Chiba R, Cho Y, Shiiya H, Kato T. Multiple anaplastic lymphoma kinase-positive primary inflammatory myofibroblastic tumors with spontaneously expanding and shrinking nodules in both lungs: a case report. GENERAL THORACIC AND CARDIOVASCULAR SURGERY CASES 2024; 3:29. [PMID: 39516994 PMCID: PMC11533626 DOI: 10.1186/s44215-024-00153-7] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Subscribe] [Scholar Register] [Received: 12/05/2023] [Accepted: 04/26/2024] [Indexed: 11/16/2024]
Abstract
BACKGROUND Inflammatory myofibroblastic tumors (IMTs) are uncommon neoplasms most prevalent in individuals under 40 years old and predominantly in the lungs. Despite their rarity, multiple anaplastic lymphoma kinase (ALK)-positive IMTs, especially those of various sizes, have not been widely reported. This report describes a case of multiple ALK-positive IMTs in the lungs, aiming to further our understanding of their behavior and management. CASE PRESENTATION Herein, we present the case of a 64-year-old woman who presented with an abnormal shadow on chest examination. Chest computed tomography revealed a main tumor in the right middle lobe and multiple irregularly shaped small nodules in both lungs. Thus, thoracoscopic wedge resection of the left lower lobe was performed for diagnosis. Pathological findings indicated smooth muscle proliferation without malignancy. IMT was diagnosed following thoracoscopic right middle lobectomy. Twenty months postoperatively, one residual nodule shrank, but another grew. CONCLUSIONS This is the first report of multiple ALK-positive IMTs in both lungs, highlighting the need for definitive diagnosis and treatment of IMTs based on surgical resection. Although caution is required in patients with lymph node metastases or distant metastases, careful follow-up is acceptable unless there is a tendency for nodules to increase in size on imaging.
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Affiliation(s)
- Shunsuke Nomura
- Department of Thoracic Surgery, Sapporo Minami-Sanjo Hospital, 4-2 S3W6, Chuou-Ku, Sapporo, Hokkaido, 060-0063, Japan
| | - Mitsuhito Kaji
- Department of Thoracic Surgery, Sapporo Minami-Sanjo Hospital, 4-2 S3W6, Chuou-Ku, Sapporo, Hokkaido, 060-0063, Japan
| | - Nobuyuki Shiina
- Department of Thoracic Surgery, Sapporo Minami-Sanjo Hospital, 4-2 S3W6, Chuou-Ku, Sapporo, Hokkaido, 060-0063, Japan
| | - Ryohei Chiba
- Department of Thoracic Surgery, Sapporo Minami-Sanjo Hospital, 4-2 S3W6, Chuou-Ku, Sapporo, Hokkaido, 060-0063, Japan
| | - Yasushi Cho
- Department of Thoracic Surgery, Sapporo-Kosei General Hospital, N3E8, Chuou-Ku, Sapporo, Hokkaido, 060-0033, Japan
| | - Haruhiko Shiiya
- Department of Thoracic Surgery, Hokkaido University Hospital, N14W5, Kita-Ku, Sapporo, Hokkaido, 060-8648, Japan
| | - Tatsuya Kato
- Department of Thoracic Surgery, Hokkaido University Hospital, N14W5, Kita-Ku, Sapporo, Hokkaido, 060-8648, Japan.
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13
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Huang M, Huang M, Gao X. Atypical inflammatory pseudotumor of the liver. Asian J Surg 2024; 47:1115-1116. [PMID: 37996373 DOI: 10.1016/j.asjsur.2023.10.133] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/28/2023] [Accepted: 10/26/2023] [Indexed: 11/25/2023] Open
Affiliation(s)
- Mengna Huang
- Department of MRI, The First Affiliated Hospital of Zhengzhou University, Zhengzhou, 450052, China.
| | - Mengyue Huang
- Department of MRI, The First Affiliated Hospital of Zhengzhou University, Zhengzhou, 450052, China
| | - Xuemei Gao
- Department of MRI, The First Affiliated Hospital of Zhengzhou University, Zhengzhou, 450052, China
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14
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Khan F, Shahid J, Saleem A, Khawaja UA, Memon WA, Zafar U, Hameed TA, Abbasher Hussien Mohamed Ahmed K. IgG4-related inflammatory pancreatic head pseudotumor mirrors pancreatic head tumor: A novel case series with a review of the literature. Clin Case Rep 2024; 12:e8467. [PMID: 38317667 PMCID: PMC10839121 DOI: 10.1002/ccr3.8467] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/04/2023] [Revised: 01/12/2024] [Accepted: 01/19/2024] [Indexed: 02/07/2024] Open
Abstract
Key Clinical Message In this noteworthy case series regarding pancreatic pseudo tumors, we intend to spread knowledge among physicians for the diagnostic and therapeutic approach and eventual disease prognosis. Abstract Inflammatory pseudotumor of pancreatic head greatly mimics pancreatic head tumor. One of them is IgG4-related pancreatic disease, which is commonly mistaken as neoplastic disease on imaging. In our novel case series, we report three cases of IgG4-related pancreatic head pseudotumor with patients ranging from 35 to 72 years of age. Patients presented with jaundice and abdominal pain. Alongside initial laboratory workup, abdominal CTs and serum IgG4 levels were also obtained. Imaging features in conjunction with IgG4 levels confirmed the diagnosis of IgG4-related autoimmune pancreatitis. Pancreatic pseudotumors are notorious for being often reported as real tumors. Through our noteworthy case series, we intend to highlight the imaging features and laboratory markers that are crucial in such cases to avoid invasive procedures.
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Affiliation(s)
- Faheemullah Khan
- Department of RadiologyAga Khan University HospitalKarachiPakistan
| | - Jehanzeb Shahid
- Department of RadiologyAga Khan University HospitalKarachiPakistan
| | - Amna Saleem
- Jinnah Medical and Dental CollegeKarachiPakistan
| | | | | | - Uffan Zafar
- Department of RadiologyAga Khan University HospitalKarachiPakistan
| | - Tariq Abdul Hameed
- Department of RadiologyIndiana University School of MedicineIndianapolisIndianaUSA
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15
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Wu KY, Kulbay M, Daigle P, Nguyen BH, Tran SD. Nonspecific Orbital Inflammation (NSOI): Unraveling the Molecular Pathogenesis, Diagnostic Modalities, and Therapeutic Interventions. Int J Mol Sci 2024; 25:1553. [PMID: 38338832 PMCID: PMC10855920 DOI: 10.3390/ijms25031553] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/31/2023] [Revised: 01/21/2024] [Accepted: 01/23/2024] [Indexed: 02/12/2024] Open
Abstract
Nonspecific orbital inflammation (NSOI), colloquially known as orbital pseudotumor, sometimes presents a diagnostic and therapeutic challenge in ophthalmology. This review aims to dissect NSOI through a molecular lens, offering a comprehensive overview of its pathogenesis, clinical presentation, diagnostic methods, and management strategies. The article delves into the underpinnings of NSOI, examining immunological and environmental factors alongside intricate molecular mechanisms involving signaling pathways, cytokines, and mediators. Special emphasis is placed on emerging molecular discoveries and approaches, highlighting the significance of understanding molecular mechanisms in NSOI for the development of novel diagnostic and therapeutic tools. Various diagnostic modalities are scrutinized for their utility and limitations. Therapeutic interventions encompass medical treatments with corticosteroids and immunomodulatory agents, all discussed in light of current molecular understanding. More importantly, this review offers a novel molecular perspective on NSOI, dissecting its pathogenesis and management with an emphasis on the latest molecular discoveries. It introduces an integrated approach combining advanced molecular diagnostics with current clinical assessments and explores emerging targeted therapies. By synthesizing these facets, the review aims to inform clinicians and researchers alike, paving the way for molecularly informed, precision-based strategies for managing NSOI.
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Affiliation(s)
- Kevin Y. Wu
- Department of Surgery, Division of Ophthalmology, University of Sherbrooke, Sherbrooke, QC J1G 2E8, Canada; (K.Y.W.)
| | - Merve Kulbay
- Department of Ophthalmology & Visual Sciences, McGill University, Montreal, QC H4A 0A4, Canada
| | - Patrick Daigle
- Department of Surgery, Division of Ophthalmology, University of Sherbrooke, Sherbrooke, QC J1G 2E8, Canada; (K.Y.W.)
| | - Bich H. Nguyen
- CHU Sainte Justine Hospital, Montreal, QC H3T 1C5, Canada
| | - Simon D. Tran
- Faculty of Dental Medicine and Oral Health Sciences, McGill University, Montreal, QC H3A 1G1, Canada
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16
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Zhu X, Liu Y, Guo W, Liang Q, Pan C, Tan B, Yu Y. Epidemiology, characteristics, and prognostic factors of lymphoplasmacyte-rich meningioma: a systematic literature review. BMC Cancer 2024; 24:110. [PMID: 38254159 PMCID: PMC10801983 DOI: 10.1186/s12885-023-11811-4] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/26/2023] [Accepted: 12/28/2023] [Indexed: 01/24/2024] Open
Abstract
BACKGROUNDS Lymphoplasmacyte-rich meningioma(LPM) is a rare subtype of meningioma with a low degree of malignancy and an overall preferable prognosis. The purpose of this article is to increase the understanding of the disease, reduce misdiagnosis, and improve prognosis. METHODS A search was conducted in the PubMed database for English articles published from 1993 to 2023. The keywords were "lymphoplasmacyte-rich (all fields) and meningioma (all fields) and English (lang)" and "lymphoplasmacyte-rich meningioma (title/abstract) and English (lang)".We further analyzed the clinical manifestations, imaging manifestations, pathological features, treatment strategies, and prognosis of LPM.The possible prognostic indicators were analyzed by the log-rank test and Pearson's chi-squared test. RESULTS Fourteen reports with 95 LPM patients were included in this report, including 47 males and 48 females who were diagnosed between the ages of 9 and 79, with an average age of 45 years. The most common clinical manifestations are headache and limb movement disorders. In most cases, the tumor occurred on the convex portion of the brain. All tumors showed significant enhancement, with homogeneous enhancement being more common, and most patients showed peritumoral edema. Postoperative pathological EMA, LCA, and vimentin positivity were helpful for the final diagnosis of the patient. Log-rank tests showed a correlation between complete resection and better prognosis and recurrence. CONCLUSION There is a lack of significant differences in the clinical symptoms and imaging manifestations of LPM compared to other diseases that need to be differentiated, and a clear diagnosis requires pathological examination. After standardized surgical treatment, the recurrence rate and mortality rate of LPM are both low. Complete surgical resection of tumors is associated with a better prognosis and lower recurrence rate.
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Affiliation(s)
- Xiaoxi Zhu
- Department of Neurosurgery, First Hospital of Jilin University, 71 Xinmin Street, Changchun, 130021, Jilin, China
| | - Yanhua Liu
- Department of Neurosurgery, First Hospital of Jilin University, 71 Xinmin Street, Changchun, 130021, Jilin, China
| | - Weiwei Guo
- The Hospital of Northeast Normal University, Changchun, China
| | - Qi Liang
- Department of Neurosurgery, First Hospital of Jilin University, 71 Xinmin Street, Changchun, 130021, Jilin, China
| | - Chengliang Pan
- Department of Neurosurgery, First Hospital of Jilin University, 71 Xinmin Street, Changchun, 130021, Jilin, China
| | - Bin Tan
- Department of Neurosurgery, First Hospital of Jilin University, 71 Xinmin Street, Changchun, 130021, Jilin, China
| | - Ying Yu
- Department of Neurosurgery, First Hospital of Jilin University, 71 Xinmin Street, Changchun, 130021, Jilin, China.
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17
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Yamashita T, Matsubayashi Y, Mochizuki T. Traumatic tumor hemorrhage of inflammatory myofibroblastic tumor of the lung. Respir Med Case Rep 2024; 47:101981. [PMID: 38288137 PMCID: PMC10823134 DOI: 10.1016/j.rmcr.2024.101981] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/30/2023] [Revised: 12/31/2023] [Accepted: 01/08/2024] [Indexed: 01/31/2024] Open
Abstract
A 23-year-old female with a history of idiopathic epilepsy was found to have a right chest cavity shadow in a school health checkup 5 years before. CT revealed a thin-walled cavity lesion in the right middle lobe containing a ball-like mass, showing air crescent sign. After falling due to a seizure, she was transported by ambulance and admitted. CT revealed diffuse ground-glass opacities throughout the right lung field. Bronchoscopy revealed bloody bronchial alveolar lavage fluid. Due to the tumor hemorrhage, an elective simple right middle lobe resection was performed without complications. The initial immunohistochemical staining was negative for ALK using ALK1 clone; however, subsequent staining of ALK by D5F3 and 5A4 clone was positive. Immunostaining findings led to a diagnosis of inflammatory myofibroblastic tumor. The patient remains under regular observation and has experienced no recurrence over the 6-year postoperative period. This case contains two different points: the first is that a cavity lesion of inflammatory myofibroblastic tumor may cause traumatic bleeding and should be treated with caution; the second is that attention should be paid to differences in stainability among clones when diagnosing inflammatory myofibroblastic tumor.
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Affiliation(s)
- Takashi Yamashita
- Department of Thoracic Surgery, Iwata City Hospital, 512-3, Ohkubo, Iwata, Shizuoka, 438-8550, Japan
| | - Yuta Matsubayashi
- Department of Thoracic Surgery, Iwata City Hospital, 512-3, Ohkubo, Iwata, Shizuoka, 438-8550, Japan
| | - Takahiro Mochizuki
- Department of Thoracic Surgery, Iwata City Hospital, 512-3, Ohkubo, Iwata, Shizuoka, 438-8550, Japan
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18
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Almajali F, Almajali M, Schwetye K, Alexopoulos G, Khan M, Coppens J, Mercier P. IgG4-negative pituitary inflammatory pseudotumor with sphenoidal involvement resembling a macroadenoma. Br J Neurosurg 2023; 37:1886-1892. [PMID: 33063545 DOI: 10.1080/02688697.2020.1834509] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/12/2020] [Accepted: 10/06/2020] [Indexed: 10/23/2022]
Abstract
BACKGROUND Inflammatory pseudotumors (IPTs) are rare benign conditions of unknown etiology that can affect any part of the body. IPTs are most commonly associated with Immunoglobulin G4 (IgG4)-related disease. Central nervous system IPTs, especially with pituitary involvement, are even rarer entities. The presence of an IgG4-negative pituitary IPT with simultaneous extracranial involvement has not been reported. CASE REPORT We present the case of a 41-year-old female with past medical history of rheumatoid arthritis and a diagnosis of pituitary IPT with coexisting sphenoidal (extracranial) involvement mimicking a pituitary macroadenoma at presentation. The patient underwent multiple consecutive biopsies, and an extensive workup prior to establishing the diagnosis. Laboratory work-up showed normal serum IgG4 and unremarkable liver function tests. CONCLUSION Pituitary lesions with simultaneous sphenoidal involvement in patients with IgG4-negative systemic inflammatory disease should raise the clinical suspicion for intracranial IPTs, as these tumors can mimic aggressive counterparts causing adjacent bony erosion, and local invasion.
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Affiliation(s)
- Fawwaz Almajali
- Department of Neurology, Saint Louis University, St. Louis, MO, USA
| | | | - Kate Schwetye
- Department of Pathology, Saint Louis University, St. Louis, MO, USA
| | | | - Maheen Khan
- Department of Neurosurgery, Saint Louis University, St. Louis, MO, USA
| | - Jeroen Coppens
- Department of Neurosurgery, Saint Louis University, St. Louis, MO, USA
| | - Philippe Mercier
- Department of Neurosurgery, Saint Louis University, St. Louis, MO, USA
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19
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Wu H, Ding YW, Yan ZC, Wei M, Wang XD, Zhang HZ. Multiple inflammatory pseudotumor formation after craniopharyngioma resection via an extended nasal endoscopic approach: A case report. World J Clin Cases 2023; 11:7724-7731. [PMID: 38078120 PMCID: PMC10698460 DOI: 10.12998/wjcc.v11.i31.7724] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/12/2023] [Revised: 09/27/2023] [Accepted: 10/17/2023] [Indexed: 11/06/2023] Open
Abstract
BACKGROUND This report describes a case of intracranial multiple inflammatory pseudotumors (IP) after endoscopic resection of a craniopharyngioma, which is relatively rarely reported in the literature, and neurosurgeons should be aware of its existence. CASE SUMMARY Herein, we report the case of a 56-year-old man who developed decreased visual acuity and blurred vision without obvious cause or inducement on April 27, 2020. To seek further treatment, he went to the Department of Neurosurgery, Clinical Medical College, Yangzhou University. After falling ill, there was no nausea, vomiting, limb convulsions, obvious disturbance of consciousness, speech disorders, cough, or persistent fever. The neurological examination findings were normal, and pituitary magnetic resonance imaging (MRI) revealed multiple nodules with abnormal signals in the sellar region. The diagnosis was craniopharyngioma. We performed total resection of the tumor via transnasal endoscopy, and the postoperative pathology suggested that the type of tumor was craniopharyngioma. Six months after the operation, the patient experienced sudden hearing loss in the right ear, tinnitus in both ears, and numbness on the right side of the face and head. Meanwhile, cranial MRI showed multiple IP. After steroid hormone and anti-inflammatory therapy, the above symptoms did not significantly improve. Finally, the patient's symptoms were well improved by surgery, and the postoperative pathological diagnosis was multiple IP. CONCLUSION Intracranial inflammatory pseudotumor is a benign disease with slow progression, but the clinical symptoms and imaging findings are not typical, there are no pathological findings, and the diagnosis is relatively difficult. Most of the cases are treated by surgical resection, and the prognosis is good after surgery.
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Affiliation(s)
- Hao Wu
- Department of Neurosurgery, The Second Affiliated Hospital of Xi’an Medical University, Xi'an 710038, Shaanxi Province, China
- Department of Neurosurgery, Clinical Medical College, Yangzhou University, Yangzhou 225001, Jiangsu Province, China
| | - Yu-Wei Ding
- Department of Neurosurgery, The Yangzhou School of Clinical Medicine of Dalian Medical University, Yangzhou 225001, Jiangsu Province, China
| | - Zheng-Cun Yan
- Department of Neurosurgery, Clinical Medical College, Yangzhou University, Yangzhou 225001, Jiangsu Province, China
| | - Min Wei
- Department of Neurosurgery, Clinical Medical College, Yangzhou University, Yangzhou 225001, Jiangsu Province, China
| | - Xiao-Dong Wang
- Department of Neurosurgery, Clinical Medical College, Yangzhou University, Yangzhou 225001, Jiangsu Province, China
| | - Heng-Zhu Zhang
- Department of Neurosurgery, Clinical Medical College, Yangzhou University, Yangzhou 225001, Jiangsu Province, China
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20
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Nguyen H, Pham DH, Luong TH. Successful surgical management of an aggressive IgG4-related sclerosing esophageal inflammatory pseudotumor: a case report and review of literature. J Cardiothorac Surg 2023; 18:215. [PMID: 37403075 DOI: 10.1186/s13019-023-02317-y] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/11/2022] [Accepted: 06/28/2023] [Indexed: 07/06/2023] Open
Abstract
BACKGROUND Inflammatory pseudotumor (IPT) of the esophagus is a very rare benign lesions which clinical presentation is not clear and difficult to make a definitive diagnosis preoperatively. CASE PRESENTATION In this report, we presented a case of a 24-year-old female with signs of severe malnutrition state due to dysphagia increasing gradually and losing 10 kg in weight for 2 months. Comprehensive preoperative radiologic investigations were proceeded with a circumferential severe stricture caused smooth submucosal swelling in the esophagus under 23 cm from the upper dental arch and two times of negative biopsy. Due to the aggressive clinical symptoms and gross lesion characteristics, the patient underwent laparoscopic-thoracoscopic esophagectomy and reconstruction with a gastric tube. Histopathological examination showed that the squamous epithelium of the esophagus had a small, benign nucleus, the submucosal layer and the smooth muscle layer increased fibrous, with infiltrating many lymphocytes, plasma cells, and macrophages. Immunohistochemical staining was negative for CD68, CD34, Desmin and ALK markers, and there was an increase in the number of IgG4-positive plasma cells. The final diagnosis was an aggressive IgG4-related sclerosing esophageal inflammatory pseudotumor. CONCLUSIONS Inflammatory pseudotumor of the esophagus is an extremely rare benign lesion but could led to aggressive clinical presentation. The gold standard of diagnosis is histopathological examination of surgically removed specimens. Radical resection is still the most efficient treatment method.
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Affiliation(s)
- Hoang Nguyen
- Department of Gastrointestinal and Hepatobiliary surgery, Hanoi Medical University Hospital, Hanoi, Vietnam
- Department of Surgery, Hanoi Medical University, 1st Ton That Tung Street, Dong Da, Ha Noi, Hanoi, 11521, Vietnam
| | - Duc Huan Pham
- Center for Gastroenterology - Hepatobiliary - Urology - Vinmec Times City International Hospital, Hanoi, Vietnam
| | - Tuan Hiep Luong
- Department of Surgery, Hanoi Medical University, 1st Ton That Tung Street, Dong Da, Ha Noi, Hanoi, 11521, Vietnam.
- Department of Gastrointestinal and Hepato-pancreato-biliary surgery, Bach Mai Hospital, Hanoi, Vietnam.
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Miguez González J, Calaf Forn F, Pelegrí Martínez L, Lozano Arranz P, Oliveira Caiafa R, Català Forteza J, Palacio Arteaga LM, Losa Gaspà F, Ramos Bernadó I, Barrios Sánchez P, Ayuso Colella JR. Primary and secondary tumors of the peritoneum: key imaging features and differential diagnosis with surgical and pathological correlation. Insights Imaging 2023; 14:115. [PMID: 37395913 DOI: 10.1186/s13244-023-01417-6] [Citation(s) in RCA: 12] [Impact Index Per Article: 6.0] [Reference Citation Analysis] [Abstract] [Key Words] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/26/2022] [Accepted: 03/30/2023] [Indexed: 07/04/2023] Open
Abstract
Peritoneal malignancies represent a diagnostic challenge for abdominal radiologists, oncologists, surgeons and pathologists in multidisciplinary teams, who must address their differential diagnosis, staging and treatment. In this article, we explain the pathophysiology of these processes and lay out the role of different imaging techniques in their evaluation. Then, we review the clinical and epidemiological aspects, the main radiological features and the therapeutic approaches for each primary and secondary peritoneal neoplasm, with surgical and pathological correlation. We further describe other rare peritoneal tumors of uncertain origin and a variety of entities that may mimic peritoneal malignancy. Finally, we summarize the key imaging findings of each peritoneal neoplasm to facilitate an accurate differential diagnosis that may impact patient management.Clinical relevance statementImaging plays an essential role in the evaluation of peritoneal malignancies, assessing their extension, detecting unfavorable sites of involvement and facilitating an accurate differential diagnosis, helping to choose the best therapeutic approach.
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Affiliation(s)
- Javier Miguez González
- Department of Radiology, Complex Hospitalari Universitari Moisès Broggi, Consorci Sanitari Integral, Sant Joan Despí, Barcelona, Spain.
| | - Francesc Calaf Forn
- Department of Radiology, Complex Hospitalari Universitari Moisès Broggi, Consorci Sanitari Integral, Sant Joan Despí, Barcelona, Spain
| | - Laura Pelegrí Martínez
- Department of Radiology, Complex Hospitalari Universitari Moisès Broggi, Consorci Sanitari Integral, Sant Joan Despí, Barcelona, Spain
| | - Pilar Lozano Arranz
- Department of Radiology, Complex Hospitalari Universitari Moisès Broggi, Consorci Sanitari Integral, Sant Joan Despí, Barcelona, Spain
| | - Rafael Oliveira Caiafa
- Department of Radiology, Complex Hospitalari Universitari Moisès Broggi, Consorci Sanitari Integral, Sant Joan Despí, Barcelona, Spain
| | - Jordi Català Forteza
- Department of Radiology, Complex Hospitalari Universitari Moisès Broggi, Consorci Sanitari Integral, Sant Joan Despí, Barcelona, Spain
| | - Lina Maria Palacio Arteaga
- Department of Pathology, Complex Hospitalari Universitari Moisès Broggi, Consorci Sanitari Integral, Sant Joan Despí, Barcelona, Spain
| | - Ferrán Losa Gaspà
- Department of Medical Oncology, Institut Català d'Oncologia Hospitalet, Complex Hospitalari Universitari Moisès Broggi, Consorci Sanitari Integral, Barcelona, Spain
| | - Isabel Ramos Bernadó
- Peritoneal Surface Malignancies Unit, Department of Surgery, Complex Hospitalari Universitari Moisès Broggi, Consorci Sanitari Integral, Sant Joan Despí, Barcelona, Spain
| | - Pedro Barrios Sánchez
- Former Director of the Peritoneal Carcinomatosis Program of Catalonia, Former Head of the Peritoneal Surface Malignancies Unit, Department of Surgery, Complex Hospitalari Universitari Moisès Broggi, Consorci Sanitari Integral, Sant Joan Despí, Barcelona, Spain
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Iwai H, Yanagawa N, Deguchi H, Tomoyasu M, Shigeeda W, Kaneko Y, Yoshimura R, Kanno H, Sugai M, Shikanai S, Sugai T, Saito H. Surgical treatment for lung metastasis of inflammatory myofibroblastic tumor of the lung: A case report. Thorac Cancer 2023. [PMID: 37146628 DOI: 10.1111/1759-7714.14914] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/15/2023] [Accepted: 04/13/2023] [Indexed: 05/07/2023] Open
Abstract
Inflammatory myofibroblastic tumor (IMT) is a rare disease that is considered an intermediate neoplasm, with the risk of recurrence and metastasis. Surgical treatment is the standard therapy for IMT, although there are only a few reports of surgery for lung metastasis of pulmonary IMT. We opine that surgical treatment might be effective not only for localized tumors, but also for cases of lung metastasis of IMT.
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Affiliation(s)
- Hidenobu Iwai
- Department of Thoracic Surgery, Iwate Medical University, Iwate, Japan
| | - Naoki Yanagawa
- Department of Molecular Diagnostic Pathology, Iwate Medical University, Iwate, Japan
| | - Hiroyuki Deguchi
- Department of Thoracic Surgery, Iwate Medical University, Iwate, Japan
| | - Makoto Tomoyasu
- Department of Thoracic Surgery, Iwate Medical University, Iwate, Japan
| | - Wataru Shigeeda
- Department of Thoracic Surgery, Iwate Medical University, Iwate, Japan
| | - Yuka Kaneko
- Department of Thoracic Surgery, Iwate Medical University, Iwate, Japan
| | - Ryuichi Yoshimura
- Department of Thoracic Surgery, Iwate Medical University, Iwate, Japan
| | - Hironaga Kanno
- Department of Thoracic Surgery, Iwate Medical University, Iwate, Japan
| | - Mayu Sugai
- Department of Molecular Diagnostic Pathology, Iwate Medical University, Iwate, Japan
| | - Shunsuke Shikanai
- Department of Molecular Diagnostic Pathology, Iwate Medical University, Iwate, Japan
| | - Tamotsu Sugai
- Department of Molecular Diagnostic Pathology, Iwate Medical University, Iwate, Japan
| | - Hajime Saito
- Department of Thoracic Surgery, Iwate Medical University, Iwate, Japan
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Ramos JG, Ochoa A, Cicutti S, Del Rio R, Lubienieki F. Inflammatory myofibroblastic tumor of the lung involving the central nervous system in pediatric patients. Childs Nerv Syst 2023; 39:1137-1145. [PMID: 37017800 DOI: 10.1007/s00381-023-05925-0] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/11/2023] [Accepted: 03/14/2023] [Indexed: 04/06/2023]
Abstract
INTRODUCTION The inflammatory myofibroblastic tumor (IMT) is a very rare lesion with an incidence of less than 0.1% of total neoplasms and with main affection in the lungs. Involvement in the central nervous system is extremely rare, but with a much more aggressive course than IMT diagnosed in the rest of the body. We report the 2 cases presented in our neurosurgery department to date; both were treated satisfactorily without intercurrences in 10 years of follow-up. HISTORICAL BACKGROUND The World Health Organization described the IMT as a distinctive lesion composed of myofibroblastic spindle cells accompanied by an inflammatory infiltrate of plasma cells, lymphocytes, and eosinophils. CLINICAL PRESENTATION Clinical manifestations of patients with CNS IMT vary and may consist of headache, vomiting, seizures, and blindness. Seizures are the most common symptom in patients with focal lesions. DIAGNOSIS The true origin of this entity remains to be elucidated, but to date, etiologies ranging from chromosomal alterations to autoimmune or postinfectious mechanisms have been described. Due to its rarity and non-specificity in imaging, the final diagnosis of IMT in the brain parenchyma relies on pathological examination. MANAGEMENT Treatment options are controversial and include total or subtotal removal, high-dose steroids, and radiation therapy. In the last decade, the development of ALK Tyrosine Kinase Inhibitors allows the possibility of chemotherapy in those patients harboring ALK mutations. CONCLUSION IMT is a rare tumor that can exceptionally be found in the CNS. The cause is still unknown although the different studies focus on a neoplastic origin. The diagnosis is based in the use of different modalities of imaging and with histological confirmation. Optimal management is gross total resection whenever possible, is the only established curative treatment. Further research with longer follow-up is needed to clarify the natural history of this rare tumor.
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Affiliation(s)
- Javier Gonzalez Ramos
- Division of Pediatric Neurosurgery, Hospital de Pediatría Juan P. Garrahan, Combate de los Pozos 1881, ZC: 1245, Buenos Aires, Argentina.
| | - Adalberto Ochoa
- Division of Pediatric Neurosurgery, Hospital de Pediatría Juan P. Garrahan, Combate de los Pozos 1881, ZC: 1245, Buenos Aires, Argentina
| | - Santiago Cicutti
- Division of Pediatric Neurosurgery, Hospital de Pediatría Juan P. Garrahan, Combate de los Pozos 1881, ZC: 1245, Buenos Aires, Argentina
| | - Ramiro Del Rio
- Division of Pediatric Neurosurgery, Hospital de Pediatría Juan P. Garrahan, Combate de los Pozos 1881, ZC: 1245, Buenos Aires, Argentina
| | - Fabiana Lubienieki
- Division of Pathology, Hospital de Pediatría Juan P. Garrahan, Buenos Aires, Argentina
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Oh JS, Kim HW, Park SB, Kang DH, Choi CW, Kim SJ, Nam HS, Ryu DG. A large and pedunculated inflammatory pseudotumor with pseudosarcomatous change of the cecum mimicking a malignant polyp: a case report and literature review. Clin Endosc 2023; 56:119-124. [PMID: 34275257 PMCID: PMC9902694 DOI: 10.5946/ce.2021.081] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/24/2021] [Accepted: 04/05/2021] [Indexed: 02/04/2023] Open
Abstract
Inflammatory pseudotumor (IPT) is a rare benign tumor of unknown etiology that can occur in almost any organ system. It has neoplastic features such as local recurrence, invasive growth, and vascular invasion, leading to the possibility of malignant sarcomatous changes. The clinical presentations of colonic IPT may include abdominal pain, anemia, a palpable mass, and intestinal obstruction. A few cases of colonic IPT have been reported, but colonic IPT with pedunculated morphology is very rare. Furthermore, since it can mimic malignant polyps, understanding the endoscopic findings of colonic IPT is important for proper treatment. Herein, we present a case of colonic IPT with pseudosarcomatous changes, presenting as a large polyp, mimicking a malignant polyp in the cecum, along with a literature review.
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Affiliation(s)
- Jong Suk Oh
- Department of Internal Medicine, Pusan National University School of Medicine and Research Institute for Convergence of Biomedical Science and Technology, Pusan National University Yangsan Hospital, Yangsan, Korea
| | - Hyung Wook Kim
- Department of Internal Medicine, Pusan National University School of Medicine and Research Institute for Convergence of Biomedical Science and Technology, Pusan National University Yangsan Hospital, Yangsan, Korea,Correspondence: Hyung Wook Kim Department of Internal Medicine, Pusan National University Yangsan Hospital, Pusan National University School of Medicine and Research Institute for Convergence of Biomedical Science and Technology, 20 Geumo-ro, Mulgeum-eup, Yangsan 50612, Korea E-mail:
| | - Su Bum Park
- Department of Internal Medicine, Pusan National University School of Medicine and Research Institute for Convergence of Biomedical Science and Technology, Pusan National University Yangsan Hospital, Yangsan, Korea
| | - Dae Hwan Kang
- Department of Internal Medicine, Pusan National University School of Medicine and Research Institute for Convergence of Biomedical Science and Technology, Pusan National University Yangsan Hospital, Yangsan, Korea
| | - Cheol Woong Choi
- Department of Internal Medicine, Pusan National University School of Medicine and Research Institute for Convergence of Biomedical Science and Technology, Pusan National University Yangsan Hospital, Yangsan, Korea
| | - Su Jin Kim
- Department of Internal Medicine, Pusan National University School of Medicine and Research Institute for Convergence of Biomedical Science and Technology, Pusan National University Yangsan Hospital, Yangsan, Korea
| | - Hyeong Seok Nam
- Department of Internal Medicine, Pusan National University School of Medicine and Research Institute for Convergence of Biomedical Science and Technology, Pusan National University Yangsan Hospital, Yangsan, Korea
| | - Dae Gon Ryu
- Department of Internal Medicine, Pusan National University School of Medicine and Research Institute for Convergence of Biomedical Science and Technology, Pusan National University Yangsan Hospital, Yangsan, Korea
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Differential Diagnosis of Thoracoacromial Artery Pseudoaneurysm from Shoulder Inflammatory Pseudotumor: A Case Report. Diagnostics (Basel) 2022; 13:diagnostics13010082. [PMID: 36611374 PMCID: PMC9818345 DOI: 10.3390/diagnostics13010082] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/10/2022] [Revised: 11/23/2022] [Accepted: 12/21/2022] [Indexed: 12/29/2022] Open
Abstract
Diagnosing shoulder tumors is a challenge because the joint is very complex, and a static examination can misdiagnose some tumors. However, we found that a pseudoaneurysm provides a differential diagnosis of a tumor, and to that end, we present a case that mimics shoulder infection. The patient was an 80-year-old female who had a history of coronary artery disease and end-stage renal disease under regular hemodialysis and complained of right shoulder swelling and progression. A magnetic resonance imaging (MRI) scan revealed an abscess, inflammatory pseudotumor (IPT), and osteomyelitis of the humerus. Computed tomography (CT)-guided pigtail drainage was performed twice without significant improvement. An angiogram revealed a right shoulder pseudoaneurysm fed by the acromial branch of the thoracoacromial artery. After endovascular coiling, the patient was discharged and outpatient follow-up was arranged. If aspiration of the abscess leads to only mild improvement in shoulder swelling, further evaluation should be arranged. An angiogram examination is a good method for diagnosing and designing operations, and endovascular treatment is good for preventing injury to the muscle, tendon, nerve, or blood vessels.
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Li YY, Zang JF, Zhang C. Laparoscopic treatment of inflammatory myofibroblastic tumor in liver: A case report. World J Clin Cases 2022; 10:11853-11860. [PMID: 36405255 PMCID: PMC9669864 DOI: 10.12998/wjcc.v10.i32.11853] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/11/2022] [Revised: 07/12/2022] [Accepted: 08/30/2022] [Indexed: 11/07/2022] Open
Abstract
BACKGROUND Inflammatory myofibroblastic tumor in the liver (IMTL) is a rare borderline mesenchymal tumor. Neither clinical symptoms nor laboratory tests have absolute specificity for the diagnosis of IMTL, and imaging also lacks obvious specificity. Although there are sporadic reports of recurrence after surgical treatment, surgical resection is the mainstay of treatment.
CASE SUMMARY A 29-year-old man complained of general weakness, slight discomfort in the upper abdomen, with a history of upper respiratory tract infection for 1 wk before admission. Plain and enhanced upper abdominal magnetic resonance imaging showed a mass in liver segments II and III (48 mm × 53 mm). He was treated by laparoscopic left lateral segmentectomy. Postoperative pathological examination with hematoxylin and eosin staining suggested that the mass in liver segments II and III was IMTL. During 21 mo postoperative follow-up, no obvious residual or recurrent lesions were observed.
CONCLUSION There is a risk of malignant degeneration in IMTL. The principal choice of treatment is laparoscopic left lateral segmentectomy.
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Affiliation(s)
- Yang-Yang Li
- Department of Hepatobiliary Surgery, Taizhou People’s Hospital, The Fifth Affiliated Hospital of Medical School of Nantong University, Taizhou 225300, Jiangsu Province, China
| | - Jin-Feng Zang
- Department of Hepatobiliary Surgery, Taizhou People’s Hospital, The Fifth Affiliated Hospital of Medical School of Nantong University, Taizhou 225300, Jiangsu Province, China
| | - Chi Zhang
- Department of Hepatobiliary Surgery, Taizhou People’s Hospital, The Fifth Affiliated Hospital of Medical School of Nantong University, Taizhou 225300, Jiangsu Province, China
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Gao SQ, Bao YJ, Luo JS. Inflammatory Myofibroblastic Tumor of the Hilar Bile Duct: A Case Report and Literature Review. Front Surg 2022; 9:928669. [PMID: 36211291 PMCID: PMC9537756 DOI: 10.3389/fsurg.2022.928669] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/26/2022] [Accepted: 06/03/2022] [Indexed: 11/13/2022] Open
Abstract
Background Inflammatory myofibroblastic tumor (IMT) is a very rare tumor and occurs seldom in the biliary tract. IMT can occur in any part of the body and in people of any age; however, it most commonly occurs in children or adolescents. Its etiology and pathogenesis are currently unknown. The clinical manifestations of a hilar inflammatory myofibroblastic tumor are atypical, and the imaging examination is nonspecific. The diagnosis is mainly based on histopathology and immunohistochemistry findings, and surgical resection is the preferred treatment method. Case Description Herein, we report a rare case of hilar bile duct IMT and review the related literature. Our patient was a 54-year-old woman presenting with a 1-day history of upper abdominal pain as the main clinical symptom. She was misdiagnosed as having cholangiocarcinoma before the surgery. She underwent surgery and was ultimately diagnosed with IMT based on histopathology and immunohistochemistry findings. On 1-year follow-up, no tumor recurrence or related complications were noted. Conclusions We hope this case report helps clinicians gain a deeper understanding of biliary IMT of the hilum.
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Liver Abscess Complicated with an Inflammatory Myofibroblastic Tumor. J Belg Soc Radiol 2022; 106:78. [PMID: 36213372 PMCID: PMC9503893 DOI: 10.5334/jbsr.2756] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/08/2022] [Accepted: 08/31/2022] [Indexed: 11/30/2022] Open
Abstract
Inflammatory myofibroblastic tumors have a wide spectrum of biological behaviour and are composed of inflammatory cells and myofibroblastic spindle cells. Tumors may infrequently involve the liver. Imaging findings are non-specific. Teaching Point: Radiologists should be familiar with inflammatory myofibroblastic tumors as a diagnostic consideration to avoid unnecessary surgery.
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Ahmed M, Buti YJ, Bawahab A, Ghaly H, Elsayed R. Small Bowel Obstruction as a Result of Inflammatory Pseudotumor. Cureus 2022; 14:e28707. [PMID: 36204020 PMCID: PMC9527131 DOI: 10.7759/cureus.28707] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 09/02/2022] [Indexed: 12/02/2022] Open
Abstract
Inflammatory pseudotumor is a rare benign neoplasm that has been described in nearly the entire body and is often mistaken for malignancy. The exact etiology remains unknown. We present a case of small bowel obstruction secondary to an inflammatory pseudotumor. The patient’s symptoms and radiological findings were very concerning for underlying malignancy. En-block resection was recommended to prevent a recurrence. We aim to shed light on this rare cause of small bowel obstruction.
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30
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Immunoglobulin G4-related inflammatory pseudotumor of the right ventricle with right coronary artery occlusion. J Cardiol Cases 2022; 26:293-296. [DOI: 10.1016/j.jccase.2022.05.019] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/28/2022] [Revised: 05/27/2022] [Accepted: 05/31/2022] [Indexed: 11/23/2022] Open
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Lee CS, Kim JS, Rodriguez R, Krell RW. Anaplastic Lymphoma Kinase Positive Mesenteric Inflammatory Myofibroblastic Tumor in Adult Woman. Cureus 2022; 14:e24422. [PMID: 35637807 PMCID: PMC9126854 DOI: 10.7759/cureus.24422] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 04/23/2022] [Indexed: 11/25/2022] Open
Abstract
Inflammatory myofibroblastic tumors (IMTs) are rare mesenchymal neoplasms containing spindle cells and inflammatory components that can be locally aggressive. They have unclear biological behavior and may recur after resection. A 31-year-old woman presented with three months of cough, fatigue, weight loss, abdominal pain, anemia, and elevated inflammatory markers. CT showed a large well-circumscribed enhancing mass in the right colic mesentery. The patient underwent a laparoscopic right colectomy. Pathologic review showed fascicular spindle cells with admixed chronic inflammatory cells. Cells stained diffusely positive for SMA and anaplastic lymphoma kinase (ALK), diagnostic of an IMT. Post-operatively, the patient reported symptom resolution and had normalization of lab values. She remains disease-free at 20 months. IMT is rare in adults, accounting for 0.7%-1.0% of lung tumors. Up to 30% of patients present with elevated inflammatory markers. On imaging, IMTs are soft tissue masses with variable enhancement and fibrosis, often suspected to be malignant neoplasms. Up to 80% of IMTs are driven by altered tyrosine kinase signaling and half of IMTs express ALK, which may be treated in unresectable/recurrent cases using ALK-inhibitors. IMT may recur in 10%-15% of patients. The roles of adjuvant treatments are unclear given the rarity and unpredictable biological behavior. Long-term follow-up with regular radiologic and laboratory surveillance is recommended given possible local recurrence. IMTs are best managed in a multidisciplinary setting given their unpredictable nature. Surgery is the mainstay of IMT treatment with long-term control expected in >80% of adult patients.
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32
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Bandara GBKD, Jayarajah U, Rodrigo VSD. Renal inflammatory myofibroblastic tumour presenting with a large retroperitoneal abscess. SAGE Open Med Case Rep 2022; 10:2050313X221089484. [PMID: 35401980 PMCID: PMC8984842 DOI: 10.1177/2050313x221089484] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/02/2021] [Accepted: 03/07/2022] [Indexed: 11/21/2022] Open
Abstract
Inflammatory myofibroblastic tumour arising from the kidney is a rare occurrence. In this
case report, we present a rare case of inflammatory myofibroblastic tumour arising from
the kidney diagnosed after the presentation with a large retroperitoneal abscess for the
first time in literature. A 55-year-old woman with diabetes mellitus presented to us with
painful lump in the left lumbar region of the back for 1-week duration. On examination,
there was a firm, diffuse lump in the left lumbar region of the back. Her inflammatory
markers were high, but the serum creatinine and blood urea were within the normal range.
Abdominal ultrasonography showed a distorted left kidney with a heterogeneous mass
consisting cystic and solid components measuring approximately 7 × 9 × 8 cm in size. A
contrast-enhanced computed tomography scan showed an 11 × 9 × 9 cm-sized low-density mass
posterior to the left kidney with multiple contrast-enhancing septations which appeared
posterior to but separate from the left kidney within the left renal fascia. There was
breeching of the Gerota’s fascia with extension posteriorly up to subcutaneous tissue.
Since the radiological opinion was in favour of a complex perinephric abscess, open
drainage of the abscess was performed after failed attempts of ultrasound-guided drainage.
The biopsy of the abscess wall was suggestive of a renal cell carcinoma and radical
nephrectomy was planned. Due to tumour invasion, the radical nephrectomy was combined with
a splenectomy and the specimen sent for histology. It showed an inflammatory
myofibroblastic tumour or pseudotumour with the periphery showing ulceration and abscess
formation. The patient had an uneventful recovery following surgery. Thus, we report the
first case of renal inflammatory myofibroblastic tumour presenting with a large
retroperitoneal abscess extending to the subcutaneous tissue plane. Final diagnosis was
made only after radical surgery which was curative.
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Affiliation(s)
| | - Umesh Jayarajah
- Department of Surgery, District General Hospital, Chilaw, Sri Lanka
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Common Presentation of Uncommon Disease: Inflammatory myofibroblastic tumor of the lung, case report. Respir Med Case Rep 2022; 37:101656. [PMID: 35516792 PMCID: PMC9065430 DOI: 10.1016/j.rmcr.2022.101656] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/25/2022] [Accepted: 04/20/2022] [Indexed: 11/23/2022] Open
Abstract
Background Inflammatory myofibroblastic tumor (IMT) of the lung is a rare lung tumor, accounting for 0.7% of all lung tumors. They are usually benign, but can invade surrounding structures, undergo malignant transformation, recur, or even metastasize. Case report We report a 44-year-old adult diabetic male from Saudi Arabia who had been suffering from cough with severe sputum and left shoulder pain for 2 weeks. Chest radiography (X-ray and computed tomography (CT)) revealed the presence of a mass lesion in the left lower upper lobe with central cavitation. The diagnosis of inflammatory myofibroblast lung tumor was confirmed by histological and immunohistochemical examination of the CT guided lung biopsy. The patient was successfully treated with surgical resection of the tumor by left limited thoracotomy with safety margin, and IMT was also documented. Conclusion A high degree of suspicion of a solitary pulmonary mass is required for diagnosis and management of an inflammatory myofibroblastic lung tumor. The clinical and radiologic presentation of an inflammatory myofibroblastic tumor is nonspecific and the diagnosis is rarely made before surgical biopsy. Histologic and immunohistochemical examination is usually required to confirm the diagnosis and prevent recurrence.
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Goyal L, Rao S, Reddy GS, Agarwal P. Inflammatory myofibroblastic tumor of anterior maxillary gingiva: An unusual clinical presentation. J Oral Maxillofac Pathol 2022; 26:S73-S76. [PMID: 35450239 PMCID: PMC9017838 DOI: 10.4103/jomfp.jomfp_338_20] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/08/2020] [Revised: 05/28/2021] [Accepted: 05/31/2021] [Indexed: 11/04/2022] Open
Abstract
Inflammatory myofibroblastic tumor (IMT) is a rare benign neoplasm with variable clinical presentation. We hereby present a case of a 27-year-old female who presented to us with an unsuspecting gingival overgrowth in her anterior gingiva. This article aims to describe IMT as a rare intraoral entity which may involve the anterior maxillary gingiva, involving or sparing the underlying bone, as only a handful of such cases have been reported so far. Surgical excision is the mainstay of treatment. Biopsy and histopathological examination of even the smallest lesions is a must. Timely diagnosis and intervention can prevent severe morbidity that can arise if these lesions are left undiagnosed as aggressive management of lesions that become extensive is mandatory.
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Affiliation(s)
- Lata Goyal
- Department of Dentistry, Division of Periodontics, All India Institute of Medical Sciences, Bathinda, Punjab, India
| | - Shalinee Rao
- Department of Pathology, All India Institute of Medical Sciences, Rishikesh, Uttarakhand, India
| | - Gosla Srinivas Reddy
- Department of Dentistry (Craniomaxillofacial Surgery), All India Institute of Medical Sciences, Rishikesh, Uttarakhand, India
| | - Padmanidhi Agarwal
- Department of Dentistry, Dr. Ram Manohar Lohia Institute of Medical Sciences, Lucknow, Uttar Pradesh, India
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Patil PH, Pereira T, Winnier JJ, Shetty JS. Inflammatory myofibroblastic tumor in the retromolar region of an 8-year-old child: A rare finding. J Oral Maxillofac Pathol 2022; 26:S80-S83. [PMID: 35450238 PMCID: PMC9017824 DOI: 10.4103/jomfp.jomfp_363_20] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/05/2020] [Revised: 05/29/2021] [Accepted: 06/07/2021] [Indexed: 11/04/2022] Open
Abstract
Inflammatory myofibroblastic tumor (IMT) of the oral cavity is an extremely rare finding. The etiology and pathogenesis of IMT is controversial and unclear. The tumor requires complete surgical excision and continuous monitoring of clinical consequences. The present article describes the clinical, histological, operative and immunohistochemical features of a case of IMT in the mandibular retromolar region of an 8-year-old male. Histologically, the lesion shows myofibroblastic spindle cell proliferations with infiltrative margins in an inflammatory background. Immunohistochemically, the myofibroblastic spindle cells in the present case were positive for α-smooth muscle actin and CD68 due to which the diagnosis of IMT was confirmed.
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Affiliation(s)
- P Harshali Patil
- Department of Pedodontics and Preventive Dentistry, D.Y. Patil University School of Dentistry, Navi Mumbai, Maharashtra, India
| | - Treville Pereira
- Department of Oral pathology and Microbiology, D.Y. Patil University School of Dentistry, Navi Mumbai, Maharashtra, India
| | - J Jasmin Winnier
- Department of Pedodontics and Preventive Dentistry, D.Y. Patil University School of Dentistry, Navi Mumbai, Maharashtra, India
| | - J Subraj Shetty
- Department of Oral pathology and Microbiology, D.Y. Patil University School of Dentistry, Navi Mumbai, Maharashtra, India
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Kovač JD, Janković A, Đikić-Rom A, Grubor N, Antić A, Dugalić V. Imaging Spectrum of Intrahepatic Mass-Forming Cholangiocarcinoma and Its Mimickers: How to Differentiate Them Using MRI. Curr Oncol 2022; 29:698-723. [PMID: 35200560 PMCID: PMC8870737 DOI: 10.3390/curroncol29020061] [Citation(s) in RCA: 22] [Impact Index Per Article: 7.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/29/2021] [Revised: 01/23/2022] [Accepted: 01/29/2022] [Indexed: 12/14/2022] Open
Abstract
Intrahepatic cholangiocarcinoma (ICC) is the second most common primary hepatic malignancy, with mass-forming growth pattern being the most common. The typical imaging appearance of mass-forming ICC (mICC) consists of irregular ring enhancement in the arterial phase followed by the progressive central enhancement on portal venous and delayed phases. However, atypical imaging presentation in the form of hypervascular mICC might also be seen, which can be attributed to distinct pathological characteristics. Ancillary imaging features such as lobular shape, capsular retraction, segmental biliary dilatation, and vascular encasement favor the diagnosis of mICC. Nevertheless, these radiological findings may also be present in certain benign conditions such as focal confluent fibrosis, sclerosing hemangioma, organizing hepatic abscess, or the pseudosolid form of hydatid disease. In addition, a few malignant lesions including primary liver lymphoma, hemangioendothelioma, solitary hypovascular liver metastases, and atypical forms of hepatocellular carcinoma (HCC), such as scirrhous HCC, infiltrative HCC, and poorly differentiated HCC, may also pose a diagnostic dilemma by simulating mICC in imaging studies. Diffusion-weighted imaging and the use of hepatobiliary contrast agents might be helpful for differential diagnosis in certain cases. The aim of this manuscript is to provide a comprehensive overview of mICC imaging features and to describe useful tips for differential diagnosis with its potential mimickers.
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Affiliation(s)
- Jelena Djokic Kovač
- Center for Radiology and Magnetic Resonance Imaging, University Clinical Centre of Serbia, Pasterova No. 2, 11000 Belgrade, Serbia;
- Faculty of Medicine, University of Belgrade, Dr Subotica No. 8, 11000 Belgrade, Serbia; (N.G.); (A.A.); (V.D.)
- Correspondence: ; Tel.: +381-65-8270-290
| | - Aleksandra Janković
- Center for Radiology and Magnetic Resonance Imaging, University Clinical Centre of Serbia, Pasterova No. 2, 11000 Belgrade, Serbia;
| | - Aleksandra Đikić-Rom
- Department of Pathology, University Clinical Centre of Serbia, Pasterova No.2, 11000 Belgrade, Serbia;
| | - Nikica Grubor
- Faculty of Medicine, University of Belgrade, Dr Subotica No. 8, 11000 Belgrade, Serbia; (N.G.); (A.A.); (V.D.)
- Clinic for Digestive Surgery, University Clinical Centre of Serbia, Koste Todorovica Street, No. 6, 11000 Belgrade, Serbia
| | - Andrija Antić
- Faculty of Medicine, University of Belgrade, Dr Subotica No. 8, 11000 Belgrade, Serbia; (N.G.); (A.A.); (V.D.)
- Clinic for Digestive Surgery, University Clinical Centre of Serbia, Koste Todorovica Street, No. 6, 11000 Belgrade, Serbia
| | - Vladimir Dugalić
- Faculty of Medicine, University of Belgrade, Dr Subotica No. 8, 11000 Belgrade, Serbia; (N.G.); (A.A.); (V.D.)
- Clinic for Digestive Surgery, University Clinical Centre of Serbia, Koste Todorovica Street, No. 6, 11000 Belgrade, Serbia
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Geiselmann MT, Acampa DJ, Melamed J, Arif F, Takabe K, Seitelman E, Datta R, Gunasekaran G, Takahashi H. Immunoglobulin G4-Negative Inflammatory Pseudotumors of the Pancreas. World J Oncol 2022; 12:240-245. [PMID: 35059084 PMCID: PMC8734500 DOI: 10.14740/wjon1432] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/01/2021] [Accepted: 11/25/2021] [Indexed: 11/11/2022] Open
Abstract
Inflammatory pseudotumor (IPT) can occur in any organ, but rarely shows pancreatic involvement. While surgical excision has been recommended as the primary treatment for IPT of the pancreas in the past, some authors suggest observation while medical management often results in regression. Corticosteroids, nonsteroidal anti-inflammatory drugs and immunosuppressive therapy have been used to treat IPTs. Spontaneous regression has also been reported in IPT managed without surgical intervention. A 62-year-old female was evaluated for worsening abdominal pain and a mass in the neck of the pancreas that was identified on ultrasound. Further imaging with magnetic resonance imaging revealed a pancreatic mass with dilated pancreatic duct and an atrophic parenchyma of the pancreatic neck. Her serum tumor markers were not elevated. As this lesion appeared to be resectable pancreatic cancer based on cross-sectional imaging, no biopsy was performed prior to surgical resection. Distal pancreatectomy and splenectomy was recommended and the patient desired to proceed. Her recovery was uneventful with no postoperative complications, including pancreatic fistula. Final pathology revealed a lesion consistent with the diagnosis of immunoglobulin G4 (IgG4)-negative IPT without neoplasm. IPT of the pancreas is a difficult entity to diagnose and treat due to clinical and imaging characteristics closely resembling pancreatic adenocarcinoma. Biopsy with immunohistochemical analysis can be useful in diagnosing IPT; however, symptomatic lesions and concerning findings on cross-sectional imaging may warrant more definitive surgical intervention.
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Affiliation(s)
- Matthew T Geiselmann
- New York Institute of Technology, College of Osteopathic Medicine, Old Westbury, NY, USA.,Both authors contributed equally
| | - Daniel J Acampa
- New York Institute of Technology, College of Osteopathic Medicine, Old Westbury, NY, USA.,Both authors contributed equally
| | - Joshua Melamed
- Department of Surgery, Mount Sinai South Nassau, Oceanside, NY, USA
| | - Farzana Arif
- Department of Pathology, Mount Sinai South Nassau, Oceanside, NY, USA
| | - Kazuaki Takabe
- Department of Surgical Oncology, Roswell Park Comprehensive Cancer Center, Buffalo, NY, USA.,Department of Surgery, University at Buffalo Jacobs School of Medicine and Biomedical Sciences, the State University of New York, Buffalo, NY, USA.,Department of Breast Surgery and Oncology, Tokyo Medical University, Tokyo, Japan.,Department of Surgery, Yokohama City University, Yokohama, Japan.,Department of Surgery, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan
| | - Eric Seitelman
- Department of Surgery, Mount Sinai South Nassau, Oceanside, NY, USA
| | - Rajiv Datta
- Department of Surgery, Mount Sinai South Nassau, Oceanside, NY, USA
| | - Ganesh Gunasekaran
- Department of Surgery, Mount Sinai South Nassau, Oceanside, NY, USA.,Department of Surgery, Division of HPB Surgery, Mount Sinai Hospital, New York, NY, USA
| | - Hideo Takahashi
- Department of Surgery, Mount Sinai South Nassau, Oceanside, NY, USA
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38
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Zulu S, Kruger C, Morare N, Montwedi D, Ngwenya S. Inflammatory pseudo-tumour of the colon mimicking acute appendicitis: A case report. Int J Surg Case Rep 2022; 90:106695. [PMID: 34999473 PMCID: PMC8749171 DOI: 10.1016/j.ijscr.2021.106695] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/01/2021] [Revised: 12/10/2021] [Accepted: 12/14/2021] [Indexed: 11/30/2022] Open
Abstract
Introduction Presentation of case Discussion Conclusion
Inflammatory pseudotumours are often mistaken for malignant lesions This error can result in unnecessary resection being performed We investigated whether radiography can diagnose inflammatory pseudotumours Our findings reveal that radiography may not be effective in this context Further studies may be required to prevent unnecessarily extensive resection
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Affiliation(s)
- S Zulu
- Department of Surgery, University of Pretoria, South Africa.
| | - C Kruger
- Department of Surgery, University of Pretoria, South Africa
| | - N Morare
- Department of Surgery, University of Pretoria, South Africa
| | - D Montwedi
- Department of Surgery, University of Pretoria, South Africa
| | - S Ngwenya
- Department of Surgery, University of Pretoria, South Africa
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39
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Zarrouki S, Marouf R. Mini-invasive endoscopic approach to tracheal inflammatory myofibroblastic tumor in a young woman: A case report. Ann Med Surg (Lond) 2022; 73:103208. [PMID: 35070283 PMCID: PMC8767228 DOI: 10.1016/j.amsu.2021.103208] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/09/2021] [Revised: 12/19/2021] [Accepted: 12/19/2021] [Indexed: 11/22/2022] Open
Abstract
INTRODUCTION Inflammatory myofibroblastic tumor (IMT) of the trachea is rare tumor mostly found in children and young adults. CASE REPORT We report a case of a 28 year-old woman who presented chronic isolated coughing. Chest CT scan showed a tracheal tumor. Rigid bronchoscopy allowed the complete removal of the tumor, and histology confirmed the diagnosis of IMT. 12 months follow-up found no recurrence. DISCUSSION IMT is a rare tumor exhibiting both benign and aggressive behaviour. The endoscopic approach of tracheal should be considered when there is a minimal tracheal wall invasion. CONCLUSION Through this case, we want to emphasise the role of rigid bronchoscopy in the complete removal of endotracheal IMT.
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Affiliation(s)
- Sara Zarrouki
- Thoracic Surgery Departement, Mohammed VI University Hospital Center, Oujda, Morocco
- Mohammed First University, Faculty of Medicine and Pharmacy, Oujda, Morocco
| | - Rachid Marouf
- Thoracic Surgery Departement, Mohammed VI University Hospital Center, Oujda, Morocco
- Mohammed First University, Faculty of Medicine and Pharmacy, Oujda, Morocco
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40
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Boriçi S, Tanka M, Serbo L. Inflammatory myofibroblastic tumour of the lung: Case report and review of the literature. Respirol Case Rep 2022; 10:e0885. [PMID: 34934506 PMCID: PMC8652042 DOI: 10.1002/rcr2.885] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/05/2021] [Revised: 11/16/2021] [Accepted: 11/18/2021] [Indexed: 11/10/2022] Open
Abstract
Inflammatory myofibroblastic tumour (IMT) of the lung is a rare tumour encountered in children. Although it is seen mostly in paediatric ages, a small number of cases exists in the literature. It may appear as an inflammatory mass or may have the characteristics of a tumour with the ability for recidivism and metastasis. Careful follow-up of the cases is required to differentiate between the two. We present the case of a 4-year-old girl who presented with cough, and the chest x-ray and computed tomography scan revealed a tumour mass in the right lung. After lobectomy, histological examination combined with immunohistochemical study discovered an IMT of the lung.
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Affiliation(s)
- Sonila Boriçi
- Service of Pulmonology and AllergyUniversity Hospital Center “Mother Tereza”TiranaAlbania
| | - Marjeta Tanka
- Service of Pulmonology and AllergyUniversity Hospital Center “Mother Tereza”TiranaAlbania
| | - Luljeta Serbo
- Service of Pulmonology and AllergyUniversity Hospital Center “Mother Tereza”TiranaAlbania
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41
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Craig E, Wiltsie LM, Beaupin LK, Baig A, Kozielski R, Rothstein DH, Li V, Twist CJ, Barth M. Anaplastic lymphoma kinase inhibitor therapy in the treatment of inflammatory myofibroblastic tumors in pediatric patients: Case reports and literature review. J Pediatr Surg 2021; 56:2364-2371. [PMID: 33676744 DOI: 10.1016/j.jpedsurg.2021.02.004] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/07/2020] [Revised: 01/21/2021] [Accepted: 02/02/2021] [Indexed: 10/22/2022]
Abstract
BACKGROUND Inflammatory myofibroblastic tumors (IMTs) are a rare subtype of inflammatory pseudotumor frequently associated with rearrangement of the anaplastic lymphoma kinase (ALK) gene. Their treatment has historically relied on at-times challenging and morbid surgical excision. Recent studies have shown that neo/adjuvant therapy with ALK inhibitors can significantly enhance outcomes in select patients. METHODS A systematic literature review was performed to characterize comprehensive treatment of ALK-positive IMTs in the pediatric population. This report also includes two patients from our home institutions not previously reported in the literature. RESULTS We identified a total of 27 patients in 12 studies in addition to 2 patients from the senior authors' institution for a total of 29 patients (median age, 7 years; 52% male). The IMTs comprised a wide range of anatomic locations. Almost half (12, 41.3%) were treated with ALK-inhibitors alone and felt to be in remission. The remainder was treated with ALK-inhibitors either before or after surgery and had a curative response. CONCLUSIONS ALK-positive IMTs can be successfully treated with ALK-inhibition alone or in combination with surgical resection. Further genetic characterization may be helpful in determining more precise treatment and defining needed durations thereof.
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Affiliation(s)
- Ethan Craig
- Department of Surgery, University at Buffalo Jacobs School of Medicine and Biomedical Sciences, Buffalo, NY, United States
| | - Laura M Wiltsie
- Division of Pediatric Hematology/Oncology, University at Buffalo Jacobs School of Medicine and Biomedical Sciences, Buffalo, NY, United States
| | - Lynda K Beaupin
- Division of Pediatric Hematology/Oncology, University at Buffalo Jacobs School of Medicine and Biomedical Sciences and Department of Pediatrics, Roswell Park Comprehensive Cancer Center, Buffalo, NY, United States
| | - Ayesha Baig
- Division of Pediatric Gastroenterology, University at Buffalo Jacobs School of Medicine and Biomedical Sciences, Buffalo, NY, United States
| | - Rafal Kozielski
- Department of Pathology, John R. Oishei Children's Hospital, Buffalo, NY, United States
| | - David H Rothstein
- Department of Pediatric Surgery John R. Oishei Children's Hospital and Department of Surgery, University at Buffalo Jacobs School of Medicine and Biomedical Sciences, Buffalo, NY, United States.
| | - Veetai Li
- Department of Pediatric Neurosurgery, John R. Oishei Children's Hospital and Department of Neurosurgery, University at Buffalo Jacobs School of Medicine and Biomedical Sciences, Buffalo, NY, United States
| | - Clare J Twist
- Division of Pediatric Hematology/Oncology, University at Buffalo Jacobs School of Medicine and Biomedical Sciences and Department of Pediatrics, Roswell Park Comprehensive Cancer Center, Buffalo, NY, United States
| | - Matthew Barth
- Division of Pediatric Hematology/Oncology, University at Buffalo Jacobs School of Medicine and Biomedical Sciences and Department of Pediatrics, Roswell Park Comprehensive Cancer Center, Buffalo, NY, United States
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42
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Kourtidis S, Saravakos P, Fiehn C, Preyer S. Inflammatory Pseudotumor in the Parapharyngeal Space: Is It Possible to Diagnose by Exclusion? Cureus 2021; 13:e18907. [PMID: 34804740 PMCID: PMC8599394 DOI: 10.7759/cureus.18907] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 10/19/2021] [Indexed: 11/17/2022] Open
Abstract
Inflammatory pseudotumor (IP) is a rare pathologic condition that easily can be confounded with malignancy. The clinical presentation depends on the site of occurrence and the radiological or laboratory findings are not specific. Diagnosis can be established only with histology. We report a case of a 64-year-old woman with IP in an uncommon localization, the parapharyngeal space extending to skull base. Although the diagnosis was not certain after histopathological examination, broad diagnostic workup helped to exclude malignancy or bacterial infection and led to diagnosis of an IP by exclusion. We observed a good clinical and radiological regression of symptoms after administration of oral immunosuppressants, confirming the immunological mechanism of the disease.
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Affiliation(s)
- Savvas Kourtidis
- Otorhinolaryngology and Head and Neck Surgery, Charité Universitätsmedizin, Berlin, DEU
| | | | - Christoph Fiehn
- Rheumatology and Clinical Immunology, Medical Centre Baden-Baden, Baden-Baden, DEU
| | - Serena Preyer
- Otorhinolaryngology and Head and Neck Surgery, ViDia Kliniken, Karlsruhe, DEU
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43
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Rodriguez JD, Selleck AM, Abdel Razek AAK, Huang BY. Update on MR Imaging of Soft Tissue Tumors of Head and Neck. Magn Reson Imaging Clin N Am 2021; 30:151-198. [PMID: 34802577 DOI: 10.1016/j.mric.2021.06.019] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/16/2022]
Abstract
This article reviews soft tissue tumors of the head and neck following the 2020 revision of WHO Classification of Soft Tissue and Bone Tumours. Common soft tissue tumors in the head and neck and tumors are discussed, along with newly added entities to the classification system. Salient clinical and imaging features that may allow for improved diagnostic accuracy or to narrow the imaging differential diagnosis are covered. Advanced imaging techniques are discussed, with a focus on diffusion-weighted and dynamic contrast imaging and their potential to help characterize soft tissue tumors and aid in distinguishing malignant from benign tumors.
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Affiliation(s)
- Justin D Rodriguez
- Department of Radiology, Duke University, 2301 Erwin Rd, Durham, NC 27705, USA
| | - A Morgan Selleck
- Department of Otolaryngology/Head and Neck Surgery, University of North Carolina Hospitals, 170 Manning Drive, CB 7070, Physicians Office Building, Rm G190A, Chapel Hill, NC 27599, USA
| | | | - Benjamin Y Huang
- Department of Radiology, UNC School of Medicine, 101 Manning Drive, CB#7510, Chapel Hill, NC 27599, USA.
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44
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A rare cause of chronic dysphagia: pulmonary inflammatory myofibroblastic tumor with distal esophagus invasion. J Cardiothorac Surg 2021; 16:286. [PMID: 34627314 PMCID: PMC8501738 DOI: 10.1186/s13019-021-01662-0] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/21/2020] [Accepted: 09/19/2021] [Indexed: 02/08/2023] Open
Abstract
Background Inflammatory myofibroblastic tumor (IMT) is rare intermediate tumor, which happens mostly in children and young adults. Case presentation Reported is the successful treatment of a 29-year-old man presented with progressively dysphagia and weight loss. No other abnormal symptoms were observed. The contrast enhanced computed tomography (CT) revealed a dumbbell-shaped lesion between lung and esophagus. Finally, it was pathologically diagnosed as pulmonary IMT invading to the distal esophagus after operation. The patient underwent partial esophagectomy and left lower lobectomy, and was discharged on 10th postoperative day. Conclusions IMT is a rare lesion that usually occurs in the lung, but pulmonary IMT with distal esophagus invasion has not been described previously. Discriminating untypical symptom, completed resection, pathological expertise and closed follow-up will reach the successful diagnosis and treatment.
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45
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Perkins SJ, Gao R, Glazer TA, Zhao CX, Basura G, McKean EL. Treatment and Prognosis of Inflammatory Pseudotumor of the Skull Base. J Neurol Surg B Skull Base 2021; 83:e555-e563. [DOI: 10.1055/s-0041-1735558] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/12/2021] [Accepted: 07/25/2021] [Indexed: 10/20/2022] Open
Abstract
Abstract
Background Inflammatory pseudotumor (IPT) of the skull base is a rare, locally destructive lesion managed with a variety of treatments. We explore the impact of treatment on outcome and assess the prognosis of IPT.
Methods This is a retrospective review of IPT of the skull base at a tertiary academic medical center. The primary outcome was radiographic progression after treatment. Outcome versus tumor location was also examined and a prognostic model was developed using a logistic regression.
Results The demographics of 21 patients with IPT are reported. Treatment consisted of corticosteroids (in 80.1% of patients), disease modifying antirheumatic drugs (DMARDs; 33.3%), surgical resection (28.6%), radiation (23.8%), antibiotics (14.3%), chemotherapy (rituximab; 9.5%), and antivirals (4.8%). At 50.7 months, 50.8% had radiographic progression. Local therapy trended toward having a better response than systemic therapy (p = 0.60). IPT of the orbit required 2.4 treatment modalities, compared with 2.0 for pharyngeal IPT, and 1.3 for posterior skull base masses (p = 0.14). A total of 75% orbital IPT underwent radiographic progression, compared with 71% of pharyngeal IPT and 50% of posterior skull base masses (p = 0.62). Sixteen patients were used to create the logistic model of radiographic progression. The Cox–Snell R
2 was 0.71 (p = 0.03). No individual variables were statistically significant.
Conclusion To our knowledge, this is among the largest sample of cases describing the presentation, treatment, and prognosis of IPT of the skull base. Our data suggest that there may be an improved response with local therapy over systemic therapy and better prognosis among posterolateral skull base masses.
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Affiliation(s)
- Sidney J. Perkins
- Medical School, University of Michigan Medical School, Ann Arbor, Michigan, United States
| | - Rebecca Gao
- Department of Otolaryngology—Head and Neck Surgery, University of Michigan, Ann Arbor, Michigan, United States
| | - Tiffany A. Glazer
- Department of Surgery, Division of Otolaryngology-Head and Neck Surgery, University of Wisconsin, Madison, Wisconsin, United States
| | - Cher X. Zhao
- Department of Otolaryngology, Harvard Medical School, Pediatric Otolaryngology, Massachusetts Eye and Ear, Boston, MA 02114, United States
| | - Gregory Basura
- Department of Otolaryngology—Head and Neck Surgery, University of Michigan, Ann Arbor, Michigan, United States
| | - Erin L. McKean
- Department of Otolaryngology—Head and Neck Surgery, University of Michigan, Ann Arbor, Michigan, United States
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Chen HC, Fu Q, Song Y, Wang DL. Clinical and pathological characteristics of patients with pulmonary inflammatory pseudotumors: An 18-year retrospective study of 31 cases. Medicine (Baltimore) 2021; 100:e27040. [PMID: 34477134 PMCID: PMC8415998 DOI: 10.1097/md.0000000000027040] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/05/2020] [Accepted: 08/02/2021] [Indexed: 01/05/2023] Open
Abstract
To investigate the clinical and pathological characteristics in patients with pulmonary inflammatory pseudotumors (PIP).This retrospective study included 31 patients with PIP from 2001 to 2019. Preoperative computed tomography scan was performed in all patients. Clinical and pathological characteristics were collected and analyzed.Thirty-one patients (16 female and 15 male) were recruited, with a median age of 57 years (range, 11-72 years). Eight (25.8%) patients were asymptomatic, and the others had symptoms characterized by cough with sputum, chest and back pain, dry cough, fever and blood in sputum, or hemoptysis. All cases were single lesions, including 23 cases in the right lung, and 8 cases in the left lung. Computed tomography scan demonstrated irregular lobulated nodules or masses in 14 patients, and regular round or oval nodules or masses in 11 cases. The blurred edge of tumors and spiculation was found in 12 cases. Microscopic results were characterized by the collection of inflammatory mesenchymal cells. Immunohistochemical examination showed vimentin, smooth muscle actin, and anaplastic lymphoma kinase positive. Complete tumor resection was obtained in all cases. No recurrence or metastasis was observed during the follow-up period.PIP has a variety of manifestations. Preoperative diagnosis is difficult to reach. The final diagnosis still depends on the pathological and immunohistochemical examination. Complete surgical resection is the main treatment at present, and the overall prognosis is good.
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Affiliation(s)
- Heng-Chi Chen
- Department of Thoracic Surgery, National Cancer Center/National Clinical Research Center for Cancer/Cancer Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
| | - Qiang Fu
- Department of Thoracic Surgery, National Cancer Center/National Clinical Research Center for Cancer/Cancer Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
| | - Yan Song
- Department of Pathology, National Cancer Center/National Clinical Research Center for Cancer/Cancer Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
| | - Da-Li Wang
- Department of Thoracic Surgery, National Cancer Center/National Clinical Research Center for Cancer/Cancer Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
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Primitivo A, Oliveira MH, Gonçalves A. IgG4-related hepatic inflammatory pseudotumour: could MRI suggest the correct diagnosis? BMJ Case Rep 2021; 14:e244572. [PMID: 34426430 PMCID: PMC8383849 DOI: 10.1136/bcr-2021-244572] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 08/08/2021] [Indexed: 11/04/2022] Open
Abstract
We report a case of a 62-year-old woman, HIV positive, with a 3-week history of jaundice and elevated cholestatic enzymes. Imaging studies displayed intrahepatic biliary dilatation and a central liver lesion. Pathology described lesions of active cholangitis, lymphoplasmacytic infiltration and fibrosis, suggesting a hepatic inflammatory pseudotumour (IPT) IgG4 related. IgG4-related lymphoplasmacytic form of IPT belongs to IgG4-related diseases. We discuss the importance to include IgG4-related hepatic IPT as part of the differential diagnosis of any liver lesion, highlighting potential imaging clues that may help in establishing the correct diagnosis.
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Affiliation(s)
- Ana Primitivo
- Radiology, Hospital Beatriz Ângelo, Loures, Portugal
| | | | - Afonso Gonçalves
- Radiology, Hospital Beatriz Ângelo, Loures, Portugal
- Radiology, Hospital Garcia de Orta EPE, Almada, Portugal
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48
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Imamura K, Hosoya M, Kasuya K, Shimanuki MN, Shinden S, Ogawa K, Oishi N. Labyrinthine destruction caused by inflammatory pseudotumor of the temporal bone: A report of three cases and review of the literature. Laryngoscope Investig Otolaryngol 2021; 6:857-865. [PMID: 34401513 PMCID: PMC8356880 DOI: 10.1002/lio2.609] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/11/2021] [Revised: 05/12/2021] [Accepted: 06/16/2021] [Indexed: 11/26/2022] Open
Abstract
BACKGROUND Inflammatory pseudotumors (IPTs) are rare, idiopathic, and inflammatory lesions that are histopathologically benign. Here, we present three cases of labyrinthine destruction caused by an IPT. METHODS The first patient was a 74-year-old male with a mass lesion extending from the inner ear to the external ear canal. The second patient was a 62-year-old female with a foliated polycystic lesion in the petrous bone on the dorsal side of the left internal auditory canal. The third patient was a 68-year-old female with a mass extending from the inner ear to the middle ear, destroying the semicircular canal and cochlea. RESULTS In two cases, we performed surgical resection successfully with no recurrence. In the other case, the lesion showed shrinkage after chemotherapy for colorectal cancer incidentally found during the examination process. CONCLUSION Surgical technique and indication for IPT should be based on the location and function of the lesion. In addition, there is room to consider pharmacotherapy as a treatment option for IPT of the temporal bone. LEVEL OF EVIDENCE 4.
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Affiliation(s)
- Kanako Imamura
- Department of Otorhinolaryngology‐Head and Neck SurgeryKeio University School of MedicineTokyoJapan
| | - Makoto Hosoya
- Department of Otorhinolaryngology‐Head and Neck SurgeryKeio University School of MedicineTokyoJapan
| | - Kento Kasuya
- Department of Otorhinolaryngology‐Head and Neck SurgeryKeio University School of MedicineTokyoJapan
| | - Marie N. Shimanuki
- Department of OtolaryngologySaiseikai Utsunomiya HospitalUtsunomiya‐shiJapan
| | - Seiichi Shinden
- Department of OtolaryngologySaiseikai Utsunomiya HospitalUtsunomiya‐shiJapan
| | - Kaoru Ogawa
- Department of Otorhinolaryngology‐Head and Neck SurgeryKeio University School of MedicineTokyoJapan
| | - Naoki Oishi
- Department of Otorhinolaryngology‐Head and Neck SurgeryKeio University School of MedicineTokyoJapan
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Strainiene S, Sedleckaite K, Jarasunas J, Savlan I, Stanaitis J, Stundiene I, Strainys T, Liakina V, Valantinas J. Complicated course of biliary inflammatory myofibroblastic tumor mimicking hilar cholangiocarcinoma: A case report and literature review. World J Clin Cases 2021; 9:6155-6169. [PMID: 34368338 PMCID: PMC8316968 DOI: 10.12998/wjcc.v9.i21.6155] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/29/2021] [Revised: 04/30/2021] [Accepted: 05/20/2021] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND The inflammatory myofibroblastic tumor (IMT) is a rare, idiopathic, usually benign, mass-forming disease with myofibroblastic proliferation and a varying amount of inflammatory cells. Although it can affect various organs, the biliary tract is a rare localization of primary IMT, clinically, endoscopically and radiologically imitating cholangiocarcinoma. The treatment options are based only on clinical practice experience. CASE SUMMARY A 70-year-old woman was referred to our center due to progressive fatigue, weight loss, abdominal pain, night sweats, and elevated liver enzymes. Magnetic resonance cholangiopancreatography and endoscopic retrograde cholangiopancreatography (ERCP) revealed proximal common hepatic duct and hilar biliary strictures extending bilaterally to lobular bile ducts. Although initial clinical, endoscopic and radiological signs were typical for hilar cholangiocarcinoma, histological examination showed no signs of malignancy. In total, 8 biopsies using different approaches were performed (several biopsies from dominant stricture during ERCP and direct cholangioscopy; ultrasound-guided liver biopsy; diagnostic laparoscopy with liver and lymph node biopsies). Histological examination revealed signs of IMT, and the final diagnosis of biliary IMT was stated. Although IMT is usually a benign disease, in our case, it was complicated. All pharmacological treatment measures were ineffective. The patient still needs permanent stenting, suffers from recurrent infections and mechanical jaundice. Despite that, the patient already survived 24 mo. CONCLUSION IMT presenting with hilar biliary strictures is a unique diagnostic and clinical challenge as it is indistinguishable from cholangiocarcinoma, and there are no evidence-based treatment options. Our goal is to increase the understanding of this rare disease and its possible course.
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Affiliation(s)
- Sandra Strainiene
- Clinic of Gastroenterology, Nephrourology and Surgery, Institute of Clinical Medicine, Vilnius University, Vilnius 01513, Lithuania
| | | | - Juozas Jarasunas
- Department of Radiology, Nuclear Medicine and Medical Physics, Institute of Clinical Medicine, Vilnius University, Vilnius 01513, Lithuania
| | - Ilona Savlan
- Clinic of Gastroenterology, Nephrourology and Surgery, Institute of Clinical Medicine, Vilnius University, Vilnius 01513, Lithuania
| | - Juozas Stanaitis
- Clinic of Gastroenterology, Nephrourology and Surgery, Institute of Clinical Medicine, Vilnius University, Vilnius 01513, Lithuania
| | - Ieva Stundiene
- Clinic of Gastroenterology, Nephrourology and Surgery, Institute of Clinical Medicine, Vilnius University, Vilnius 01513, Lithuania
| | - Tomas Strainys
- Clinic of Anesthesiology and Intensive Care Institute of Clinical Medicine, Vilnius University, Vilnius 01513, Lithuania
| | - Valentina Liakina
- Clinic of Gastroenterology, Nephrourology and Surgery, Institute of Clinical Medicine, Vilnius University, Vilnius 01513, Lithuania
- Department of Chemistry and Bioengineering, Faculty of Fundamental Science, Vilnius Gediminas Technical University, Vilnius 10223, Lithuania
| | - Jonas Valantinas
- Clinic of Gastroenterology, Nephrourology and Surgery, Institute of Clinical Medicine, Vilnius University, Vilnius 01513, Lithuania
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Suzuki S, Ohtani M, Matsuo Y, Yoshida M, Goto A, Fukuda M, Mimasaka S. A forensic autopsy case: Sudden unexpected death due to cardiac inflammatory myofibroblastic tumor. Leg Med (Tokyo) 2021; 53:101931. [PMID: 34147823 DOI: 10.1016/j.legalmed.2021.101931] [Citation(s) in RCA: 4] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/06/2021] [Revised: 04/16/2021] [Accepted: 05/09/2021] [Indexed: 11/19/2022]
Abstract
We report an autopsy case of a 25-year-old man with no medical history who died suddenly in an Internet cafe. He was found in cardiorespiratory arrest and did not respond to cardiopulmonary resuscitation. Traumatic lesions were not observed on his body. An autopsy was performed to investigate the cause of death. Upon examination, we discovered a heart tumor that infiltrated from the outside wall to the outflow tract of the left ventricle. Left ventricular outflow tract obstruction due to a cardiac tumor was considered the mechanism of death. Histological examination identified an inflammatory myofibroblastic tumor (IMT). The final diagnosis was death secondary to circulatory failure due to a cardiac IMT. Additionally, a cardiac tumor was diagnosed using post-mortem computed tomography. Only few cases of sudden unexpected death due to cardiac IMT have been reported; we report this case along with a review of the literature.
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Affiliation(s)
- Shoken Suzuki
- Department of Forensic Sciences, Akita University Graduate School of Medicine, Akita, Japan; Department of Dentistry and Oral Surgery, Akita University Graduate School of Medicine, Akita, Japan.
| | - Maki Ohtani
- Department of Forensic Sciences, Akita University Graduate School of Medicine, Akita, Japan.
| | - Yuhei Matsuo
- Department of Forensic Sciences, Akita University Graduate School of Medicine, Akita, Japan.
| | - Makoto Yoshida
- Department of Cellular and Organ Pathology, Akita University Graduate School of Medicine, Akita, Japan.
| | - Akiteru Goto
- Department of Cellular and Organ Pathology, Akita University Graduate School of Medicine, Akita, Japan.
| | - Masayuki Fukuda
- Department of Dentistry and Oral Surgery, Akita University Graduate School of Medicine, Akita, Japan.
| | - Sohtaro Mimasaka
- Department of Forensic Sciences, Akita University Graduate School of Medicine, Akita, Japan.
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