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Kobayashi M, Maniwa T, Chikaraishi H, Samejima H, Horiguchi J, Kanzaki R, Kamiura S, Okami J. Pulmonary metastases of a uterine smooth muscle tumor of uncertain malignant potential presenting as growing bullae: a case report. J Surg Case Rep 2025; 2025:rjaf106. [PMID: 40040775 PMCID: PMC11879138 DOI: 10.1093/jscr/rjaf106] [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: 12/28/2024] [Accepted: 02/11/2025] [Indexed: 03/06/2025] Open
Abstract
Postoperative recurrences of uncertain malignant potential [(smooth muscle tumors of uncertain malignant potential (STUMPs)] have been reported, with lung as the most common site. Herein, we describe pulmonary metastases of a uterine STUMP presenting as bullae in a female patient. Computed tomography revealed two pulmonary metastases in the left lung with several bullae in both lungs, and the patient was referred to our department for pulmonary metastasectomy. Despite mild right pneumothorax on admission, left pulmonary metastasectomy and bullectomy were safely performed, with an uneventful postoperative course. Right pneumothorax recurred four days after discharge; hence, surgery was indicated. Although the air leak point was not a bulla but rather a pleural dissemination nodule, metastasectomy with bullectomy was performed. Pathological examination suggested that the bullae developed owing to retraction and collapse of the alveoli surrounding the pulmonary metastases. Bullae formed during the clinical course of malignancy have the potential of pulmonary metastases.
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Affiliation(s)
- Masao Kobayashi
- Department of General Thoracic Surgery, Osaka International Cancer Institute, 3-1-69, Otemae, Chuo-Ku, Osaka 540-0008, Japan
| | - Tomohiro Maniwa
- Department of General Thoracic Surgery, Osaka International Cancer Institute, 3-1-69, Otemae, Chuo-Ku, Osaka 540-0008, Japan
| | - Hisaya Chikaraishi
- Department of General Thoracic Surgery, Osaka International Cancer Institute, 3-1-69, Otemae, Chuo-Ku, Osaka 540-0008, Japan
| | - Hironobu Samejima
- Department of General Thoracic Surgery, Osaka International Cancer Institute, 3-1-69, Otemae, Chuo-Ku, Osaka 540-0008, Japan
| | - Julian Horiguchi
- Department of General Thoracic Surgery, Osaka International Cancer Institute, 3-1-69, Otemae, Chuo-Ku, Osaka 540-0008, Japan
| | - Ryu Kanzaki
- Department of General Thoracic Surgery, Osaka International Cancer Institute, 3-1-69, Otemae, Chuo-Ku, Osaka 540-0008, Japan
| | - Shoji Kamiura
- Department of Gynecology, Osaka International Cancer Institutex, 3-1-69, Otemae, Chuo-Ku, Osaka 540-0008, Japan
| | - Jiro Okami
- Department of General Thoracic Surgery, Osaka International Cancer Institute, 3-1-69, Otemae, Chuo-Ku, Osaka 540-0008, Japan
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Yang Q, Madueke-Laveaux OS, Cun H, Wlodarczyk M, Garcia N, Carvalho KC, Al-Hendy A. Comprehensive Review of Uterine Leiomyosarcoma: Pathogenesis, Diagnosis, Prognosis, and Targeted Therapy. Cells 2024; 13:1106. [PMID: 38994959 PMCID: PMC11240800 DOI: 10.3390/cells13131106] [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: 05/19/2024] [Revised: 06/14/2024] [Accepted: 06/21/2024] [Indexed: 07/13/2024] Open
Abstract
Uterine leiomyosarcoma (uLMS) is the most common subtype of uterine sarcomas. They have a poor prognosis with high rates of recurrence and metastasis. The five-year survival for uLMS patients is between 25 and 76%, with survival rates approaching 10-15% for patients with metastatic disease at the initial diagnosis. Accumulating evidence suggests that several biological pathways are involved in uLMS pathogenesis. Notably, drugs that block abnormal functions of these pathways remarkably improve survival in uLMS patients. However, due to chemotherapy resistance, there remains a need for novel drugs that can target these pathways effectively. In this review article, we provide an overview of the recent progress in ascertaining the biological functions and regulatory mechanisms in uLMS from the perspective of aberrant biological pathways, including DNA repair, immune checkpoint blockade, protein kinase and intracellular signaling pathways, and the hedgehog pathway. We review the emerging role of epigenetics and epitranscriptome in the pathogenesis of uLMS. In addition, we discuss serum markers, artificial intelligence (AI) combined with machine learning, shear wave elastography, current management and medical treatment options, and ongoing clinical trials for patients with uLMS. Comprehensive, integrated, and deeper insights into the pathobiology and underlying molecular mechanisms of uLMS will help develop novel strategies to treat patients with this aggressive tumor.
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Affiliation(s)
- Qiwei Yang
- Department of Obstetrics and Gynecology, University of Chicago, Chicago, IL 60637, USA; (O.S.M.-L.); (H.C.); (A.A.-H.)
| | | | - Han Cun
- Department of Obstetrics and Gynecology, University of Chicago, Chicago, IL 60637, USA; (O.S.M.-L.); (H.C.); (A.A.-H.)
| | - Marta Wlodarczyk
- Department of Biochemistry and Pharmacogenomics, Faculty of Pharmacy, Medical University of Warsaw, Banacha 1B, 02-097 Warsaw, Poland;
| | - Natalia Garcia
- Greehey Children’s Cancer Research Institute, University of Texas Health Science Center, San Antonio, TX 78229, USA;
- Department of Cell Systems and Anatomy, University of Texas Health Science Center, San Antonio, TX 78229, USA
| | - Katia Candido Carvalho
- Laboratório de Ginecologia Estrutural e Molecular (LIM 58), Disciplina de Ginecologia, Departamento deObstetricia e Ginecologia, Hospital das Clinicas da Faculdade de Medicina da Universidade de Sao Paulo (HCFMUSP), São Paulo 05403-010, Brazil;
| | - Ayman Al-Hendy
- Department of Obstetrics and Gynecology, University of Chicago, Chicago, IL 60637, USA; (O.S.M.-L.); (H.C.); (A.A.-H.)
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Incognito GG, D'Urso G, Incognito D, Lello C, Miceli A, Palumbo M. Management of a giant uterine smooth muscle tumor of uncertain malignant potential in a 32-year-old woman: case report and review of the literature. Minerva Obstet Gynecol 2022; 74:466-470. [PMID: 35912464 DOI: 10.23736/s2724-606x.22.05126-0] [Citation(s) in RCA: 3] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 06/15/2023]
Abstract
Uterine smooth muscle tumors of uncertain malignant potential (STUMP) represent a group of rare uterine smooth muscle tumors not diagnosed unequivocally as benign or malignant. To data, diagnostic criteria, malignant potential, surgical management, and follow-up of these neoplasms remain controversial. Considering that STUMP and leiomyoma are not significantly different in terms of clinical presentation and preoperative sonographic characteristics, it might be difficult to distinguish between the two affections prior to pathological confirmation at surgery. All cases should be managed by multidisciplinary tumor teams and patients' follow-up should comprise consultation with a gynecologic oncologist and a close surveillance because of the possibility of recurrence or metastasis. We present the case of a 32-year-old nulliparous woman admitted to our gynecology clinic. She was asymptomatic and only complained an increase in abdominal volume started during the past 6 months. A transabdominal and transvaginal pelvic ultrasound revealed a large heterogeneous tumor mass measuring 190×163 mm, color score 2, expanded in the left iliac fossa, suspected for benign uterine myoma. Subsequent magnetic resonance imaging confirmed a large pelvic-abdominal tumor located near the left posterior-lateral uterine wall with areas of necrosis, suggestive of subserosal leiomyoma with cystic degeneration. The patient underwent a median longitudinal laparotomy for excision of the pelvic mass. The patient was normally discharged five days after surgery in good health conditions. The final histological examination was compatible with STUMP. At present, the patient has had no relapses or metastases and she is undergoing follow-up.
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Affiliation(s)
- Giosuè G Incognito
- Department of General Surgery and Medical Surgical Specialties, University of Catania, Catania, Italy -
| | - Gisella D'Urso
- Department of General Surgery and Medical Surgical Specialties, University of Catania, Catania, Italy
| | - Dalila Incognito
- Anesthesia and Intensive Care Unit, Department of Medical and Surgical Sciences, Magna Graecia University, Catanzaro, Italy
| | - Chiara Lello
- Department of Drug and Health Sciences, University of Catania, Catania, Italy
| | - Alessia Miceli
- Department of Human Pathology in Adulthood and Childhood "G. Barresi", University of Messina, Messina, Italy
| | - Marco Palumbo
- Department of General Surgery and Medical Surgical Specialties, University of Catania, Catania, Italy
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Di Giuseppe J, Grelloni C, Giuliani L, Delli Carpini G, Giannella L, Ciavattini A. Recurrence of Uterine Smooth Muscle Tumor of Uncertain Malignant Potential: A Systematic Review of the Literature. Cancers (Basel) 2022; 14:cancers14092323. [PMID: 35565452 PMCID: PMC9104240 DOI: 10.3390/cancers14092323] [Citation(s) in RCA: 18] [Impact Index Per Article: 6.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/09/2022] [Revised: 04/30/2022] [Accepted: 05/05/2022] [Indexed: 02/05/2023] Open
Abstract
Background: This study aimed to systematically review the existing literature on uterine smooth muscle tumor of uncertain malignant potential (STUMP) to provide information about characteristics and outcomes of patients and the risk factors for recurrence over a period of 60 years (1960−2021). Methods: According to PRISMA guidelines, we searched for "uterine smooth muscle tumor of uncertain malignant potential" in PubMed (all fields) and Scopus (Title/Abstract/Keywords) databases (accessed on 1 January 2022). Relevant articles were obtained in full-text format and screened for additional references. The only filter used was the English language. Studies including full case description of patients with histopathological diagnosis of STUMP in accordance with Stanford criteria were included. Results: Thirty-four studies, including 189 cases, were included. The median age was 43 years, and in 21.5% of cases there was a recurrence of the disease. Bivariate analysis showed a significant association between use of morcellation without bag and risk of recurrence (p = 0.001). Unprotected morcellation during demolitive or conservative surgery was independently associated with a higher risk of disease recurrence with a relative risk of 2.94 (p < 0.001). A significant progressive decrease in the recurrence rate was observed over time (r = −0.671, p = 0.008). The percentage of patients who underwent surgery followed by in-bag protected morcellation significantly increased after the publication of the U.S. Food and Drug Administration alert about the risk linked to this procedure (p = 0.01). Conclusions: Unprotected morcellation of the lesion is associated with the relapse of the disease. However, this clinical condition showed a drastic decrease over time. This could likely be due to the increased awareness by surgeons of the importance of customizing surgical treatment.
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A large and late mediastinal metastasis from a uterine smooth muscle tumour of uncertain malignant potential: A case report. Int J Surg Case Rep 2021; 90:106734. [PMID: 34972012 PMCID: PMC8717254 DOI: 10.1016/j.ijscr.2021.106734] [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: 11/27/2021] [Revised: 12/22/2021] [Accepted: 12/22/2021] [Indexed: 11/23/2022] Open
Abstract
INTRODUCTION Smooth muscle tumours of uncertain malignant potential (STUMP) are mesenchymal uterine tumours with a malignant potential found somewhere between that of benign leiomyomas and leiomyosarcomas. Only three cases of STUMP pulmonary metastasis exist in the literature. CASE PRESENTATION We report the case of a 63 year-old female patient presenting with dysphonia secondary to recurrent laryngeal nerve paralysis due to an enormous mediastinal STUMP metastasis, 14 years after having undergone a total hysterectomy with bilateral salpingo-oophorectomy. A successful left pneumonectomy was performed and the mass weighing 1570 g was histologically confirmed as a STUMP metastasis. DISCUSSION Only three cases of pulmonary metastasis from STUMP have been reported in the recent literature and no robust information exists about the metastatic nature of STUMP. No cases exist in the literature of a successful pneumonectomy performed for a STUMP metastasis of such large dimensions presenting after many years from the diagnosis of the primary uterine lesion. Physicians should keep in mind that the finding of a smooth muscle tumour in the lung of a woman should promptly raise the suspicion of a metastatic uterine malignancy, even several years after diagnosis of the primary lesion. CONCLUSION Despite the size, invasiveness and late presentation of the STUMP metastasis, the surgery was successful and the patient no longer requires oxygen therapy.
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Ultrasound and clinical characteristics of uterine smooth muscle tumors of uncertain malignant potential (STUMPs). Eur J Obstet Gynecol Reprod Biol 2020; 251:167-172. [PMID: 32505056 DOI: 10.1016/j.ejogrb.2020.05.040] [Citation(s) in RCA: 16] [Impact Index Per Article: 3.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/06/2020] [Revised: 05/20/2020] [Accepted: 05/21/2020] [Indexed: 02/07/2023]
Abstract
OBJECTIVE(S) Smooth muscle tumors of uncertain malignant potential are rare uterine neoplasms. Their identification through imaging is still limited due to the few available descriptions in the scientific literature. The objective of this paper is to provide clinical and ultrasound features that could support an early identification of these neoplasms. STUDY DESIGN We retrospectively evaluated preoperative sonographic data of patients receiving a histopathological diagnosis of smooth muscle tumors of uncertain malignant potential between 2014 and 2019 at the S. Anna Hospital (Turin, Italy), a tertiary gynecological center. Tumors were characterized on the basis of ultrasound images using terms and definitions according to the morphological uterus sonographic assessment group. RESULTS A total of fourteen patients with smooth muscle tumors of uncertain malignant potential (20 lesions, including 18 pure and 2 with associated leiomyosarcoma) were identified. The median age was 47 years (range 28-77) and nine (64%) patients were of reproductive age. Six patients (43%) were asymptomatic, two (14%) presented with abdominal pain, two (14 %) with menorrhagia and four (29%) with both symptoms. Two (14%) patients developed local recurrences as uterine smooth muscle tumor of uncertain malignant potential and leiomyosarcoma, respectively. At ultrasound imaging, nine (69%) smooth muscle tumors of uncertain malignant potential were poorly or moderately vascularized and nine (82%) showed both circumferential and intra-lesional flows. Only three (15%) showed shadowing. The outlines were well-defined in seventeen cases (85%) and most (90%) showed isoechoic or mixed echogenicity with microcystic anechoic areas in fourteen (70%) of cases. CONCLUSION(S) Sonographic features of smooth muscle tumors of uncertain malignant potential may vary and a pathognomonic description has not been recognized. However, the identification of single or multiple lesions with specific ultrasound features should raise the suspicion of tumors of uncertain malignant potential. These features include isoechogenicity or mixed echogenicity, regular borders, presence of internal microcystic and anechoic areas, circumferential and intralesional vascularization ranging from minimal to high and absence of shadowing.
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Gadducci A, Zannoni GF. Uterine smooth muscle tumors of unknown malignant potential: A challenging question. Gynecol Oncol 2019; 154:631-637. [PMID: 31326137 DOI: 10.1016/j.ygyno.2019.07.002] [Citation(s) in RCA: 49] [Impact Index Per Article: 8.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/30/2019] [Revised: 06/26/2019] [Accepted: 07/01/2019] [Indexed: 12/11/2022]
Abstract
Uterine smooth muscle tumors of unknown malignant potential [STUMP]s are neoplasms with pathological features that preclude an equivocal diagnosis of leiomyosarcoma, but that do not fulfill the criteria for leiomyoma or its variants, and raise concerns that the tumors may behave in a malign fashion. Total hysterectomy with or without bilateral salpingo-oophorectomy is the standard treatment if fertility is completed, whereas myomectomy alone can be taken into consideration in young patients who desire to preserve childbearing potential. A careful surveillance every 6 months for 5 years and then yearly is strongly warranted. Patients with STUMP can relapse as either STUMP or leiomyosarcoma in approximately 11-13% of the cases, and their 5-year overall survival ranges from 92 to 100%. The present paper reviews the clinicopathological features of uterine STUMPs with a particular focus on most commonly accepted histopathological criteria for the diagnosis and on biological behaviour of these controversial neoplasms.
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Affiliation(s)
- Angiolo Gadducci
- Department of Clinical and Experimental Medicine, Division of Gynecology and Obstetrics, University of Pisa, Italy.
| | - Gian Franco Zannoni
- Division of Anatomic Pathology and Histology - Fondazione Policlinico Universitario A. Gemelli IRCCS, Università Cattolica del Sacro Cuore School of Medicine, Rome, Italy
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Saoud MK, Benchiba I, Mamouni N, Errarhay S, Bouchikhi C, Banani A. [Uterine fibroid associated with smooth muscle tumor of uncertain malignant potential (STUMP): about a case]. Pan Afr Med J 2019; 31:151. [PMID: 31086612 PMCID: PMC6488250 DOI: 10.11604/pamj.2018.31.151.14135] [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: 10/15/2017] [Accepted: 01/31/2018] [Indexed: 11/11/2022] Open
Abstract
STUMP (smooth muscle tumor of uncertain malignant potential) are smooth muscle tumors that cannot be diagnosed as either benign or malignant due to their morphological characteristics. We here report the case of a 44 year old patient with no notable medical history presenting with an increase in the abdominal volume. Pelvic ultrasound and CT TAP scan were performed showing voluminous mass with tissue and fluid-filled component in the uterus. The patient underwent exploratory laparotomy during which two masses were detected, one in the uterus and the other in the retroperitoneal area. Anatomopathologic examination showed uterine myoma associated with a STUMP.
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Affiliation(s)
| | - Imane Benchiba
- Service Gynécologie-Obstétrique I, CHU Hassan II, Fès, Maroc
| | - Nisrine Mamouni
- Service Gynécologie-Obstétrique I, CHU Hassan II, Fès, Maroc
| | - Sanaa Errarhay
- Service Gynécologie-Obstétrique I, CHU Hassan II, Fès, Maroc
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Fukui K, Takase N, Miyake T, Hisano K, Maeda E, Nishimura T, Abe K, Kozuki A, Tanaka T, Harada N, Takamatsu M, Kaneda K. Review of the literature laparoscopic surgery for metastatic hepatic leiomyosarcoma associated with smooth muscle tumor of uncertain malignant potential: Case report. World J Hepatol 2018; 10:402-408. [PMID: 29713398 PMCID: PMC5924371 DOI: 10.4254/wjh.v10.i4.402] [Citation(s) in RCA: 2] [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: 01/19/2018] [Revised: 03/10/2018] [Accepted: 04/03/2018] [Indexed: 02/06/2023] Open
Abstract
Metastatic hepatic leiomyosarcoma is a rare malignant smooth muscle tumor. We report a case of metastatic hepatic leiomyosarcoma associated with smooth muscle tumor of uncertain malignant potential (STUMP). A 68-year-old female presented with a liver mass (60 mm × 40 mm, Segment 4). She underwent left salpingo-oophorectomy for an ovary tumor with STUMP in a broad ligament 6 years ago. Though FDG-PET showed obvious metabolically active foci, abnormal metabolically active foci other than the lesion were not detected. A malignant liver tumor was strongly suspected and laparoscopic partial liver resection was performed with vessel-sealing devices using the crush clamping method and Pringle maneuver. Immunohistochemical findings revealed metastatic liver leiomyosarcoma associated with STUMP in a broad ligament. This case is an extremely rare case of malignant transformation from primary STUMP to metastatic hepatic leiomyosarcoma. It provides important evidence regarding the treatment for metastatic hepatic leiomyosarcoma associated with STUMP.
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Affiliation(s)
- Keisuke Fukui
- Department of Surgery, Kakogawa Central City Hospital, Kakogawa 675-8611, Japan
| | - Nobuhisa Takase
- Department of Surgery, Kakogawa Central City Hospital, Kakogawa 675-8611, Japan
| | - Taiichiro Miyake
- Department of Surgery, Kakogawa Central City Hospital, Kakogawa 675-8611, Japan
| | - Koji Hisano
- Department of Surgery, Kakogawa Central City Hospital, Kakogawa 675-8611, Japan
| | - Eri Maeda
- Department of Surgery, Kakogawa Central City Hospital, Kakogawa 675-8611, Japan
| | - Tohru Nishimura
- Department of Surgery, Kakogawa Central City Hospital, Kakogawa 675-8611, Japan
| | - Koichiro Abe
- Department of Surgery, Kakogawa Central City Hospital, Kakogawa 675-8611, Japan
| | - Akihito Kozuki
- Department of Surgery, Kakogawa Central City Hospital, Kakogawa 675-8611, Japan
| | - Tomohiro Tanaka
- Department of Surgery, Kakogawa Central City Hospital, Kakogawa 675-8611, Japan
| | - Naoki Harada
- Department of Surgery, Kakogawa Central City Hospital, Kakogawa 675-8611, Japan
| | - Manabu Takamatsu
- Department of Surgery, Kakogawa Central City Hospital, Kakogawa 675-8611, Japan
| | - Kunihiko Kaneda
- Department of Surgery, Kakogawa Central City Hospital, Kakogawa 675-8611, Japan
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Conconi D, Chiappa V, Perego P, Redaelli S, Bovo G, Lavitrano M, Milani R, Dalprà L, Lissoni AA. Potential role of BCL2 in the recurrence of uterine smooth muscle tumors of uncertain malignant potential. Oncol Rep 2016; 37:41-47. [PMID: 28004108 PMCID: PMC5355714 DOI: 10.3892/or.2016.5274] [Citation(s) in RCA: 10] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/23/2016] [Accepted: 03/30/2016] [Indexed: 12/15/2022] Open
Abstract
Uterine smooth muscle tumors are the most common female genital tract neoplasms. While leiomyosarcoma has been studied at length, smooth muscle tumors of uncertain malignant potential (STUMPs) still have ambiguous and unresolved issues, with a risk of relapse and evolution largely undefined. We performed an array comparative genomic hybridization analysis on a primitive STUMP and its local recurrence, histologically diagnosed as undifferentiated sarcoma. To the best of our knowledge, our report is the first genomic study on primitive STUMPs and the different relapsed tumors. The results showed few copy number alterations shared between both samples and the high heterogeneity in the STUMP was apparently lost in the sarcoma. Surprisingly the STUMP presented an amplification of the BCL2 gene, not observed in the relapsed tumor. Additionally, fluorescence in situ hybridization and immunohistochemical staining were performed to confirm BCL2 amplification and expression in these samples and in two other cases of primitive STUMPs and their corresponding relapsed tumors. The presence of BCL2 in multiple copies and expression in the two primitive STUMPs and two relapsed tumors was confirmed. The marked amplification of the BCL2 gene present in the primitive STUMP and the multiple copies also observed in other cases, suggest its potential role as a marker of STUMP malignant potential and recurrence.
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Affiliation(s)
- Donatella Conconi
- School of Medicine and Surgery, University of Milano-Bicocca, I-20900 Monza, Italy
| | - Valentina Chiappa
- Department of Obstetrics and Gynecology, San Gerardo Hospital, I-20900 Monza, Italy
| | - Patrizia Perego
- Unit of Pathology, San Gerardo Hospital, I-20900 Monza, Italy
| | - Serena Redaelli
- School of Medicine and Surgery, University of Milano-Bicocca, I-20900 Monza, Italy
| | - Giorgio Bovo
- Unit of Pathology, San Gerardo Hospital, I-20900 Monza, Italy
| | - Marialuisa Lavitrano
- School of Medicine and Surgery, University of Milano-Bicocca, I-20900 Monza, Italy
| | - Rodolfo Milani
- School of Medicine and Surgery, University of Milano-Bicocca, I-20900 Monza, Italy
| | - Leda Dalprà
- School of Medicine and Surgery, University of Milano-Bicocca, I-20900 Monza, Italy
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Lacrimal Sac Smooth Muscle Tumor of Uncertain Malignant Potential. Ophthalmic Plast Reconstr Surg 2016; 33:S29-S31. [PMID: 27306954 DOI: 10.1097/iop.0000000000000726] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/26/2022]
Abstract
A 13-year-old male presented with recurrent left nasolacrimal duct obstruction following endoscopic dacryocystorhinostomy 4 years prior at an outside institution. The past medical history was significant for stage IV neuroblastoma, diagnosed at age 2, requiring surgical resection, induction chemotherapy, autologous bone marrow transplantation and radiation, currently in remission. Preoperative CT scan demonstrated a 2 cm ovoid mass centered in the left lacrimal fossa, consistent with dacryocystocele; however, a solid tumor could not be ruled out. Subsequent surgical exploration of the lacrimal sac revealed a friable, solid mass filling the lacrimal sac, and extending into the duct. The mass was grossly resected with preservation of the lacrimal drainage system and placement of indwelling silicone stents. Histopathology confirmed the diagnosis of smooth muscle tumor of uncertain malignant potential. The patient remained free of epiphora and showed no clinical or radiographic evidence of recurrence at 6 months of follow up.
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