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Park HJ, Geum SY, Yoo JH, Park JW, Shin SH, Ye MK. Fibrous dysplasia of the maxillary bone secondary to sinus surgery in an elderly patient: A case report. EAR, NOSE & THROAT JOURNAL 2025; 104:275S-278S. [PMID: 36357084 DOI: 10.1177/01455613221139403] [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] [Indexed: 12/22/2023] Open
Abstract
Fibrous dysplasia (FD) is a rare benign disease that replaces a normal bone with abnormal fibrous and weak osseous tissue. It is usually detected in childhood and rarely occurs in old age. Although the disease is known to be caused by a genetic mutation, only a single case of FD secondary to surgery is reported in the literature. We report a case of monostotic FD of the maxillary sinus in a 70-year-old Asian woman who presented with incidental calcific lesion in the maxillary sinus on a brain computed tomography scan. At 32 months prior to presentation, the patient had undergone an endoscopic sinus surgery for a fungal ball of the same sinus. The lesion was removed by endoscopic surgery, and the histopathological evidence was consistent with FD. To the best of our knowledge, this is the second case of a postsurgical craniofacial FD, and a rare case that occurred in old age.
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Affiliation(s)
- Hee Jun Park
- Department of Otorhinolaryngology-Head and Neck Surgery, School of Medicine, Catholic University of Daegu, Daegu, Korea
| | - Sang Yen Geum
- Department of Otorhinolaryngology-Head and Neck Surgery, School of Medicine, Catholic University of Daegu, Daegu, Korea
| | - Jae Ho Yoo
- Department of Otorhinolaryngology-Head and Neck Surgery, School of Medicine, Catholic University of Daegu, Daegu, Korea
| | - Jin Woo Park
- Department of Otorhinolaryngology-Head and Neck Surgery, School of Medicine, Catholic University of Daegu, Daegu, Korea
| | - Seung Heon Shin
- Department of Otorhinolaryngology-Head and Neck Surgery, School of Medicine, Catholic University of Daegu, Daegu, Korea
| | - Mi Kyung Ye
- Department of Otorhinolaryngology-Head and Neck Surgery, School of Medicine, Catholic University of Daegu, Daegu, Korea
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Eduardo da Silva C, Clara Thibes A, Vidaletti T. Primary jugular foramen fibrous dysplasia: surgically nuanced video of extradural infratemporal transjugular approach. Illustrative case. JOURNAL OF NEUROSURGERY. CASE LESSONS 2024; 8:CASE24396. [PMID: 39401464 PMCID: PMC11488373 DOI: 10.3171/case24396] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Subscribe] [Scholar Register] [Received: 06/20/2024] [Accepted: 07/25/2024] [Indexed: 10/20/2024]
Abstract
BACKGROUND Fibrous dysplasia of the bone is a disease caused by a somatic GNAS mutation that affects craniofacial bones and can have a mass effect on different neurovascular structures. The authors present the first case of primary jugular foramen fibrous dysplasia with occlusion of the transverse and sigmoid sinuses. OBJECTIVE A 33-year-old man presented with a history of dizziness and occasional dysphagia over the past year. Magnetic resonance imaging showed a uniform enhanced mass in the left jugular foramen, with complete blockage of the adjacent transverse and sigmoid sinuses. The computed tomography scan revealed a cystic bone lesion of the jugular foramen. The patient underwent a gross-total removal of the tumor through an infratemporal transjugular approach with complete preservation of the lower cranial nerves. LESSONS This first reported case of primary jugular foramen fibrous dysplasia highlights the importance of considering this diagnosis when evaluating jugular fossa lesions. Understanding the anatomy of the infratemporal and jugular fossae, along with proficiency in microsurgical techniques, is essential for removing such tumors while preserving cranial nerve functions and the patient's quality of life. https://thejns.org/doi/10.3171/CASE24396.
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Affiliation(s)
- Carlos Eduardo da Silva
- Centro de Neurologia e Neurocirurgia – CNNc, Hospital Ernesto Dornelles, Porto Alegre, Rio Grande do Sul, Brazil
- Anatomy, DCBS, Universidade Federal de Ciências da Saúde de Porto Alegre – UFCSPA, Porto Alegre, Rio Grande do Sul, Brazil
| | - Ana Clara Thibes
- Centro de Neurologia e Neurocirurgia – CNNc, Hospital Ernesto Dornelles, Porto Alegre, Rio Grande do Sul, Brazil
| | - Tamara Vidaletti
- Centro de Neurologia e Neurocirurgia – CNNc, Hospital Ernesto Dornelles, Porto Alegre, Rio Grande do Sul, Brazil
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Kaur H, Mohanty S, Kochhar GK, Iqbal S, Verma A, Bhasin R, Kochhar AS. Comprehensive management of malocclusion in maxillary fibrous dysplasia: A case report. World J Clin Cases 2021; 9:10671-10680. [PMID: 35005000 PMCID: PMC8686154 DOI: 10.12998/wjcc.v9.i34.10671] [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: 04/06/2021] [Revised: 07/14/2021] [Accepted: 10/20/2021] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Fibrous dysplasia (FD) is a developmental hamartomatous bone disease characterized by a blend of fibrous and osseous entities. Though rarely malignant, the tumor can vary from being small and asymptomatic, to a fairly large sized lesion, progressing gradually, compromising occlusion and facial esthetics. Treatment approach depends on the stage of skeletal maturity. It primarily involves surgical management for stabilizing the disease process. Post-surgical comprehensive dental treatment is necessary for restoring form and function of the jaws and teeth. This article describes comprehensive orthodontic management of severe malocclusion in a surgically operated case of FD maxilla.
CASE SUMMARY A 19-year female presented with a chief complaint of excessive gingival display when smiling. Dental history included swelling of gums around the upper right front teeth, diagnosed at the age of 15 as FD of the right anterior maxillary segment and treated with surgical recontouring of the dysplastic bone. The clinical and radiological examinations showed adequate post-surgical healing. The surgically treated dysplastic area presented with right canting of the maxillary anterior occlusal plane. The maxillary teeth were torqued palatally, with the root of the right maxillary canine exposed clinically. We discuss sequential management of the associated malocclusion with comprehensive fixed orthodontics, along with special precautions taken to prevent reactivation of the quiescent and healed lesion.
CONCLUSION The adequate healing of fibro-dysplastic bone post-surgery must be allowed before initiating orthodontic tooth movement in the dysplastic bone. Periodic follow-ups are needed to monitor stability of occlusion and any relapse of the lesion.
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Affiliation(s)
- Harneet Kaur
- Department of Orthodontics and Dentofacial Orthopaedics, Faculty of Dentistry, Jamia Millia Islamia, Delhi 110025, Delhi, India
| | - Sujata Mohanty
- Department of Oral and Maxillofacial Surgery, Maulana Azad Institute of Dental Sciences, Delhi 110002, Delhi, India
| | - Gulsheen Kaur Kochhar
- Department of Pediatric and Preventive Dentistry, National Dental College and Hospital, Derabassi 140507, Punjab, India
| | - Shahid Iqbal
- Faculty of Dentistry, Jamia Millia Islamia, Delhi 110025, Delhi, India
| | - Anjali Verma
- Department of Oral and Maxillofacial Surgery, Maulana Azad Institute of Dental Sciences, Delhi 110002, Delhi, India
| | - Ritasha Bhasin
- Faculty of Dentistry, University of Toronto, Toronto M5G1G6, Ontario, Canada
| | - Anuraj Singh Kochhar
- Department of Orthodontics and Dentofacial Orthopaedics, Faculty of Dentistry, University of Toronto, Toronto M5G0C1, Ontario, Canada
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Jaulent P, Vignot E, Chapurlat R. Fibrous dysplasia of occipital bone revealed by acute intracranial hypertension. Osteoporos Int 2019; 30:691-693. [PMID: 30327834 DOI: 10.1007/s00198-018-4737-9] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/04/2018] [Accepted: 10/09/2018] [Indexed: 02/07/2023]
Abstract
Fibrous dysplasia of bone is a stem cell bone disease due to a somatic GNAS mutation which can affect craniofacial bones. Although craniofacial fibrous dysplasia is a benign and progressive disorder, it can cause mass effect on the cranial structures. We describe an 18-year-old man, without past medical history, came at the emergency department with progressively worsening headache, associated with vomiting. Cranial bone CT and then brain MRI revealed fibrous dysplasia of occipital bone with intraosseous cyst, compression of right sigmoid vein. An angiography was performed to stent the right sigmoid vein and symptoms had completely resolved only a few hours after the procedure. Then, a treatment by bisphosphonates was introduced. We believe this is the first description of sigmoid vein compression by a bone cyst, requiring stenting. MRI should be performed urgently in case of unusual severe headache or rapidly evolving neurologic impairment in patients with craniofacial fibrous dysplasia. Treatment of fibrous dysplasia is a controversial subject. In cases with neurologic complications, surgery or endovascular treatment might be performed.
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Affiliation(s)
- P Jaulent
- Hospices Civils de Lyon, Hôpital Edouard Herriot, Pavillon F Rhumatologie, Lyon, France
- Université de Lyon, Université Claude Bernard Lyon 1, Lyon, France
| | - E Vignot
- Hospices Civils de Lyon, Hôpital Edouard Herriot, Pavillon F Rhumatologie, Lyon, France
- Université de Lyon, Université Claude Bernard Lyon 1, Lyon, France
- INSERM UMR 1033, Lyon, France
| | - R Chapurlat
- Hospices Civils de Lyon, Hôpital Edouard Herriot, Pavillon F Rhumatologie, Lyon, France.
- Université de Lyon, Université Claude Bernard Lyon 1, Lyon, France.
- INSERM UMR 1033, Lyon, France.
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Umerani MS, Bakhshi SK, Abbas A, Sharif S, Arshad S. Post-traumatic fibrous dysplasia of the parietal bone: A rare entity. Asian J Neurosurg 2017; 12:547-550. [PMID: 28761541 PMCID: PMC5532948 DOI: 10.4103/1793-5482.148800] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/13/2022] Open
Abstract
Fibrous dysplasia (FD) is a rare fibro-osseous lesion in which normal bone is replaced by abnormal fibrous tissue. Although a congenital disorder, a single case report of traumatic etiology had been described in the literature. We report a case of monostotic FD of the parietal calvarium in a 21-year-old female patient who presented to us with a single swelling in the parietal region been noticed after head injury sustained at the age of 7 years. After imaging investigations, the lesion was excised via craniotomy followed by cranioplasty in the same sitting. The histopathological evidence was suggestive of FD. To the best of our knowledge, this is the second case of a posttraumatic cranial FD and the first case describing the growing mass in the parietal bone secondary to head injury.
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Affiliation(s)
| | | | - Asad Abbas
- Department of Surgery, Ziauddin University Hospital, Karachi, Pakistan
| | - Salman Sharif
- Department of Neurosurgery, Liaquat National Hospital, Karachi, Pakistan
| | - Sidra Arshad
- Department of Histopathology, Aga Khan University Hospital, Karachi, Pakistan
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Basaran R, Kaksi M, Gur E, Efendioglu M, Balkuv E, Sav A. Monostotic fibrous dysplasia involving occipital bone: a case report and review of literature. Pan Afr Med J 2014; 19:124. [PMID: 25745531 PMCID: PMC4341266 DOI: 10.11604/pamj.2014.19.124.5203] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/05/2014] [Accepted: 09/27/2014] [Indexed: 12/20/2022] Open
Abstract
Fibrous dysplasia (FD) is a progressive systemic bone tumour of young and it can be seen on cranial bones. FD is divided into three types according to radiological features. The second most common subtype is polyostotic subtype. With this article, we aimed to review and present clinical features, radiological examination, differential diagnosis and treatment management of a case of solitary monostotic fibrous dysplasia of occipital bone. 15 years old female patient admitted to our hospital for a bump and in the back of his head that she noticed 1 month ago. Her physical and neurological examination was normal. On cranial CT examination we detected a bony defect. Her gadolinium enhanced cranial MRI revealed bony defect along with massive gadolinium enhancement in adjacent tissue. On histopathologic examination; PANCK, CD68, CD1a were found negative and CD45, S-100, Vimentine were found positive. Ki-67 was 4,8%. In conclusion, fibrous dysplasia is a progressive bone disease of the young patients. Despite its resemblance to a benign lesion by not being symptomatic it can progress and cause severe bony defects and skin lesions. Total surgical resection is necessary and sufficient for total treatment.
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Affiliation(s)
- Recep Basaran
- Dr. Lutfi Kirdar Kartal Education and Research Hospital, Department of Neurosurgery, Istanbul, Turkey
| | - Mustafa Kaksi
- Eyup State Hospital, Department of Neurosurgery, Istanbul, Turkey
| | - Erdal Gur
- Dr. Lutfi Kirdar Kartal Education and Research Hospital, Department of Neurosurgery, Istanbul, Turkey
| | - Mustafa Efendioglu
- Dr. Lutfi Kirdar Kartal Education and Research Hospital, Department of Neurosurgery, Istanbul, Turkey
| | - Ece Balkuv
- Istanbul Medeniyet University Goztepe Education and Research Hospital, Department of Neurology, Istanbul, Turkey
| | - Aydin Sav
- Acibadem University School of Medicine, Department of Pathology, Istanbul, Turkey
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