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Intraosseous synovial sarcoma of the distal ulna: a case report and review of the literature

BACKGROUND: Synovial sarcoma is a relatively rare type of soft tissue sarcoma. The commonly observed symptom is a deep-seated palpable mass accompanied by pain or tenderness. Thus, it is considered a soft tissue sarcoma and rarely occurs primarily in bone. However, only few studies have been reporte...

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Autores principales: Fujibuchi, Taketsugu, Miyawaki, Joji, Kidani, Teruki, Imai, Hiroshi, Kiyomatsu, Hiroshi, Kitazawa, Riko, Miura, Hiromasa
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6359868/
https://www.ncbi.nlm.nih.gov/pubmed/30709383
http://dx.doi.org/10.1186/s12885-019-5325-x
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author Fujibuchi, Taketsugu
Miyawaki, Joji
Kidani, Teruki
Imai, Hiroshi
Kiyomatsu, Hiroshi
Kitazawa, Riko
Miura, Hiromasa
author_facet Fujibuchi, Taketsugu
Miyawaki, Joji
Kidani, Teruki
Imai, Hiroshi
Kiyomatsu, Hiroshi
Kitazawa, Riko
Miura, Hiromasa
author_sort Fujibuchi, Taketsugu
collection PubMed
description BACKGROUND: Synovial sarcoma is a relatively rare type of soft tissue sarcoma. The commonly observed symptom is a deep-seated palpable mass accompanied by pain or tenderness. Thus, it is considered a soft tissue sarcoma and rarely occurs primarily in bone. However, only few studies have been reported on intraosseous synovial sarcoma, and reports on cases with cytogenetic or molecular confirmation are even rarer. We report a case of intraosseous synovial sarcoma of the distal ulna that has been confirmed using histopathological examination and molecular analysis. CASE PRESENTATION: A 77-year-old female was referred to our hospital with a 1-month history of right wrist pain after housework. Clinical and imaging findings suggested a benign bone tumor that was enhanced by Gd-DTPA. It was thought that the tumor was possibly an enchondroma. Initially, we planned to evaluate the benignancy of the tumor with intraoperative frozen section, followed by curettage and bone graft at one stage However, when considering carefully, characteristics of the tumor did not perfectly match those of any diagnostic categories including enchondroma. Therefore, an incisional biopsy was performed and revealed that the tumor was synovial sarcoma. Following an elaborate plan, the patient underwent a wide resection of the tumor at the distal part of the right ulna. Reverse transcription-polymerase chain reaction (RT-PCR) from the resected specimen and sequencing of RT-PCR products demonstrated a chimeric SYT-SSX1 transcript, confirming the diagnosis of synovial sarcoma. CONCLUSIONS: Synovial sarcoma is seldom considered in differential diagnosis of bone tumors because it is difficult to line up such an unusual diagnosis as a differential diagnosis. When the lesion does not perfectly fit into any diagnostic category, when the initial image diagnosis appears unconvincing, biopsy and pathology are indicated, recalling Jaffe’s triangle. According to these diagnostic processes, the patient successfully completed the treatment for this rare intraosseous synovial sarcoma, following a careful plan based on the preoperative diagnosis.
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spelling pubmed-63598682019-02-07 Intraosseous synovial sarcoma of the distal ulna: a case report and review of the literature Fujibuchi, Taketsugu Miyawaki, Joji Kidani, Teruki Imai, Hiroshi Kiyomatsu, Hiroshi Kitazawa, Riko Miura, Hiromasa BMC Cancer Case Report BACKGROUND: Synovial sarcoma is a relatively rare type of soft tissue sarcoma. The commonly observed symptom is a deep-seated palpable mass accompanied by pain or tenderness. Thus, it is considered a soft tissue sarcoma and rarely occurs primarily in bone. However, only few studies have been reported on intraosseous synovial sarcoma, and reports on cases with cytogenetic or molecular confirmation are even rarer. We report a case of intraosseous synovial sarcoma of the distal ulna that has been confirmed using histopathological examination and molecular analysis. CASE PRESENTATION: A 77-year-old female was referred to our hospital with a 1-month history of right wrist pain after housework. Clinical and imaging findings suggested a benign bone tumor that was enhanced by Gd-DTPA. It was thought that the tumor was possibly an enchondroma. Initially, we planned to evaluate the benignancy of the tumor with intraoperative frozen section, followed by curettage and bone graft at one stage However, when considering carefully, characteristics of the tumor did not perfectly match those of any diagnostic categories including enchondroma. Therefore, an incisional biopsy was performed and revealed that the tumor was synovial sarcoma. Following an elaborate plan, the patient underwent a wide resection of the tumor at the distal part of the right ulna. Reverse transcription-polymerase chain reaction (RT-PCR) from the resected specimen and sequencing of RT-PCR products demonstrated a chimeric SYT-SSX1 transcript, confirming the diagnosis of synovial sarcoma. CONCLUSIONS: Synovial sarcoma is seldom considered in differential diagnosis of bone tumors because it is difficult to line up such an unusual diagnosis as a differential diagnosis. When the lesion does not perfectly fit into any diagnostic category, when the initial image diagnosis appears unconvincing, biopsy and pathology are indicated, recalling Jaffe’s triangle. According to these diagnostic processes, the patient successfully completed the treatment for this rare intraosseous synovial sarcoma, following a careful plan based on the preoperative diagnosis. BioMed Central 2019-02-01 /pmc/articles/PMC6359868/ /pubmed/30709383 http://dx.doi.org/10.1186/s12885-019-5325-x Text en © The Author(s). 2019 Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
spellingShingle Case Report
Fujibuchi, Taketsugu
Miyawaki, Joji
Kidani, Teruki
Imai, Hiroshi
Kiyomatsu, Hiroshi
Kitazawa, Riko
Miura, Hiromasa
Intraosseous synovial sarcoma of the distal ulna: a case report and review of the literature
title Intraosseous synovial sarcoma of the distal ulna: a case report and review of the literature
title_full Intraosseous synovial sarcoma of the distal ulna: a case report and review of the literature
title_fullStr Intraosseous synovial sarcoma of the distal ulna: a case report and review of the literature
title_full_unstemmed Intraosseous synovial sarcoma of the distal ulna: a case report and review of the literature
title_short Intraosseous synovial sarcoma of the distal ulna: a case report and review of the literature
title_sort intraosseous synovial sarcoma of the distal ulna: a case report and review of the literature
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6359868/
https://www.ncbi.nlm.nih.gov/pubmed/30709383
http://dx.doi.org/10.1186/s12885-019-5325-x
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