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Cholangiocarcinoma accompanied by desmoid-type fibromatosis

Hepatobiliary & Pancreatic Diseases International 2011年第2期10卷 214-217页

作者： Nan Xu,Zhe-Yu Chen,Lu-Nan Yan,Jia-Ying Yang,Wen-Tao Wang and Shu-Guang JingDepartment of Liver and Vascular Surgeryand Department of Hepato-biliary-pancreatic Surgery,West China Hospital,Sichuan University, Chengdu 610041,China [a] Department of Liver and Vascular Surgery West China Hospital Sichuan University Chengdu 610041 China [b] Department of Hepato-biliary-pancreatic Surgery West China Hospital Sichuan University Chengdu 610041 China

BACKGROUND:Cholangiocarcinoma complicated by intraabdominal desmoid-type fibromatosis(DTF)is uncommon. There are no reports on patients with this type of fibromatosis, in which the pre-operative treatment(including diagnosis), surgical approach,post-operative pathologic reports,and prognosis are discussed. METHOD:The clinicopathological features of a 49-year-old man were retrospectively analyzed. RESULTS:Cholangiocarcinoma located in the inferior segment of the bile duct was considered pre-operatively on the basis of clinical *** the time of pancreaticoduodenectomy,the mesojejunum was stiff without nodules or a mass at a distance of approximately 80 cm from the ligament of *** excision of the entire lesion of the intestinal mesenteric contracture and its subsidiary was ***-operative pathologic findings confirmed an adenocarcinoma located at the extremity of the common bile duct and infiltrating the full thickness of the common bile duct as well as the deep muscular layer of the *** contracted jejunal mesentery was shown to have *** patient was alive with no evidence of recurrence after a follow-up of 6 months. CONCLUSIONS:The patient had a rare hereditary disease with intra-abdominal DTF,which manifests the characteristics of an aggressive growth pattern and a high rate of local recurrence; conservative therapy is *** excision of the fibromatous lesion during pancreaticoduodenectomy may maximally decrease the risk of local recurrence.

关键词： cholangiocarcinoma desmoid-type fibromatosis diagnosis pancreaticoduodenectomy pathology

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Abnormality of chromosome 8 in desmoid-type fibromatosis

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Chinese Medical Journal 2007年第9期120卷 838-840页

作者： YANG Ji-long WANG Jian ZHOU Xiao-yan LI Xiao-qiu ZHU Xiong-zeng Department of Pathology Cancer Hospital Fudan University Shanghai 200032 China

desmoid-type fibromatoses are clonal fibroblastic proliferations that arise in deep soft tissues and are characterized by infiltrative growth and a tendency toward local recurrence. The molecular genetic studies show that there are chromosomal abnormalities relating to the pathogenesis of this lesion, such as trisomy 8. However, there are different results concerning this type of abnormality. Fluorescence in situ hybridization （FISH） is a sensitive and reproducible technique that combines molecular cytogenetics with morphologic information and brings them together in a single frame for evaluation. To explore the controversy of the aneuploid abnormality of chromosome 8, we collected specimens of desmoid-type fibromatosis to determine whether the existence of trisomy 8 chromosome abnormalities correlates with clinicopathologic features, as well as to investigate the feasibility of detecting trisomy 8 in FFPE tissue by FISH method.

关键词： desmoid-type fibromatosis fluorescence in situ hybridization trisomy 8

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A Case of Shoulder desmoid-type fibromatosis Missed on an Initial Work Up: Case Report and Literature Review

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Journal of Biomedical Science and Engineering 2020年第10期13卷 223-227页

作者： Michael Byrd William Barfield Emily Darr College of Medicine Medical University of South Carolina Charleston SC USA Department of Orthopaedics & Physical Medicine Medical University of South Carolina Charleston SC USA

desmoid-type fibromatosis (DF) is a rare, locally invasive, non-metastasizing soft tissue proliferation derived from mesenchymal progenitor cells. The incidence of DF is 2 to 4 per million per year in the general population and typically affects adults between the ages of 35 - 40. desmoid-type fibromatosis can either be sporadic or associated with mutation in the adenomatous polyposis coli gene. Trauma, surgery, pregnancy, and oral contraceptives have been identified as risk factors for the development of desmoid-type fibromatosis. MRI is the standard for image characterization, and CT image-guided core needle biopsy for diagnosis. “Wait and see” is the current management recommendation, and studies of y-secretase inhibitors and tyrosine kinase inhibitors have shown promise in the treatment of desmoid-type fibromatosis. This report presents a case of rare right shoulder desmoid type fibromatosis in a 48-year-old male that was missed on an initial workup including EMG/NCS and shoulder MRI, and demonstrates the importance of revisiting the diagnostic process if a former workup has yielded an unclear clinical picture.

关键词： desmoid-type fibromatosis All Rehabilitation All Pain

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Coexistence of duodenum derived aggressive fibromatosis and paraduodenal hydatid cyst: A case report and review of literature

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World Journal of Gastrointestinal Surgery 2018年第8期10卷 90-94页

作者： Sami Akbulut Mehmet Yilmaz Saadet Alan Mehmet Kolu Nese Karadag Department of Surgery and Liver Transplant Institute Inonu University Faculty of Medicine Department of Surgery Inonu University Faculty of Medicine Department of Pathology Inonu University Faculty of Medicine Department of Radiology Inonu University Faculty of Medicine

Intra-abdominal aggressive fibromatosis is a locally aggressive tumor mostly originating from the mesentery or retroperitoneal space, infiltrating adjacent tissues, and very rarely metastasizing to distant organs. There are only two case reports in the English language literature where intra-abdominal aggressive fibromatosis originated from the intestinal wall. In this study, we aimed to report a case of aggressive fibromatosis originating from the muscularis propria layer of the duodenum and invading pancreas. Another interesting aspect of this case is that a primary paraduodenal hydatid cyst was incidentally detected in the surgical specimen. A 46-year-old female patient presented to our clinic with postprandial nausea and vomiting. A contrast-enhanced abdominal computerized tomography revealed a mass lesion with a size of 100 mm × 80 mm which originated from the distal pancreas and compressed the gastric pilor externally. Upon exploration the distal part of duodenum, proximaljejunum, and pancreatic mass were noted to form a conglomerated structure. Therefore, the fourth part of the duodenum, a 25 cm part of the proximal jejunum, distal pancreas, and the spleen were excised enbloc. The pathology report of the specimen indicated fibromatosis with a diameter of 55 mm that originated from the muscularis propria of the duodenum and extended into the pancreatic parenchyma. There was also an incidentally detected 10 mm paraduodenal hydatid cyst. No tumor recurrence was detected at a follow-up period of 24 mo. In conclusion, the most ideal treatment of desmoid-type fibromatosis is surgical resection of the mass lesion with clean surgical borders. Although rare, this tumor may originate from the intestinal wall. Histopathological verification is of great significance for a proper diagnosis.

关键词： Duodenal wall Hydatid cyst Aggressive fibromatosis Intra-abdominal fibromatosis desmoid tumor desmoid-type fibromatosis

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Adult Neuromuscular Choristoma in the Intraconal Region of the Orbital Muscle: A Case Report

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Journal of Biosciences and Medicines 2024年第11期12卷 206-212页

作者： Xuecheng Ge Liangping Luo Department of Radiology First Affiliated Hospital of Jinan University Guangzhou China The Guangzhou Key Laboratory of Molecular and Functional Imaging for Clinical Translation Department of Radiology The First Affiliated Hospital of Jinan University Guangzhou China Department of Radiology and Nuclear Medicine The Fifth Affiliated Hospital of Jinan University (Shenhe Peoples Hospital) Heyuan China

Background: Neuromuscular choristoma is a rare benign peripheral neuropathy composed of ectopic mature muscle fibers and nerve bundles, usually involving large nerve roots or trunks, such as brachial plexus and sciatic nerve. NMC usually occurs in childhood, and some cases are congenital. Here, we report a case of adult orbital intraconal NMC. The resected specimens were fish-like and tough. Histological pathology suggested that the specimen was composed of mature skeletal muscle tissue interspersed with peripheral nerve bundles. Histopathological examination revealed that the left orbital mass was composed of mature skeletal muscle tissue interspersed with surrounding nerve fascicles. Immunohistochemistry: S-100 protein was positive. In general, postoperative histopathological examination ultimately determined the diagnosis of NMC in the intraconal region of the orbital muscle. Case Presentation: A 51-year-old female patient was admitted to the hospital due to periocular pain for 2 weeks. Orbital CT scan showed an irregular soft tissue density in the left orbital muscle cone area, and the boundary between the local and the left lateral rectus muscle was unclear. Magnetic resonance imaging showed that there was an oval abnormal signal in the posterior lateral space of the left eyeball, with a clear edge and a size of about 22 mm × 8 mm. The boundary between the local area and the left lateral rectus muscle was unclear, and the optic nerve was compressed to the right side. The T1 WI showed low signal, T2-FS showed high and low mixed signal, and the enhanced scan showed continuous obvious enhancement. Eventually, the patient underwent surgical resection of the lesion. Conclusions: NMC is a rare benign peripheral neuropathy, especially NMC in the orbital muscle cone. There is no specificity in clinical and imaging examinations. Accurate diagnosis before surgical resection is very challenging for clinicians and radiologists. Importantly, we can differentiate orbital NMC

关键词： Neuromuscular Choristoma desmoid-type fibromatosis Orbit Case Report

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