Benign multicystic peritoneal mesothelioma occurring in bilateral inguinal canals metachronously: a case report

Abstract Background Benign multicystic peritoneal mesothelioma (BMPM) is a benign tumor that usually occurs in middle-aged females. Although several published studies have reported the occurrence of this tumor in the abdominal cavity, few have documented its development in the inguinal region. Case...

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Main Authors: Hiroyuki Oshikiri, Yohei Ozawa, On Suzuki, Masahiro Usuda, Go Miyata
Format: Article
Language:English
Published: Japan Surgical Society 2022-03-01
Series:Surgical Case Reports
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Online Access:https://doi.org/10.1186/s40792-022-01399-5
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author Hiroyuki Oshikiri
Yohei Ozawa
On Suzuki
Masahiro Usuda
Go Miyata
author_facet Hiroyuki Oshikiri
Yohei Ozawa
On Suzuki
Masahiro Usuda
Go Miyata
author_sort Hiroyuki Oshikiri
collection DOAJ
description Abstract Background Benign multicystic peritoneal mesothelioma (BMPM) is a benign tumor that usually occurs in middle-aged females. Although several published studies have reported the occurrence of this tumor in the abdominal cavity, few have documented its development in the inguinal region. Case presentation We present a case of a 48-year-old female presenting with a bulge in her left inguinal region. Physical examination revealed a golf ball-sized nodule in the left inguinal region that could not be pushed back into the abdominal cavity. Contrast-enhanced computed tomography showed a multicystic tumor; therefore, the patient was diagnosed with inguinal hernia or hydrocele of the Nuck’s canal. We performed surgical resection and hernia repair using the mesh plug method. The resected specimen was 80 mm in length and contained a multicystic tumor. Pathological examination showed that the cyst wall was lined by a single layer of cuboidal to single layer squamous epithelium. Immunohistochemistry revealed positivity for calretinin in the epithelial cells, for which a diagnosis of BMPM was established. The patient returned to our hospital after 5 years with symptoms similar to the previous episode, but this time in the right inguinal region. Imaging studies showed a tumor in the right inguinal region with the same characteristics as the previous one. The patient underwent tumor resection and hernia repair using the same technique. The resected tumor was 45 mm in length and had characteristics similar to the previously resected tumor. The presence of calretinin and D2-40 on immunohistochemistry led to the diagnosis of BMPM. There was no recurrence of BMPM for 33 months after the secondary surgery. Conclusions Here we present the first report of metachronous BMPM occurring in bilateral inguinal canals. Although the pathogenesis of BMPM remains unclear, reactive changes have been suggested to cause tumors originating from the groin. The treatment of choice for BMPM is surgical resection. For diagnosis, pathological examination with immunostaining can be useful. The most appropriate postoperative follow-up for inguinal BMPM is controversial, and the accumulation of more inguinal BMPM cases is needed.
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spelling doaj-art-f570b6d4d96b45d09f04bb8fc158e0872025-08-02T23:34:45ZengJapan Surgical SocietySurgical Case Reports2198-77932022-03-01811610.1186/s40792-022-01399-5Benign multicystic peritoneal mesothelioma occurring in bilateral inguinal canals metachronously: a case reportHiroyuki Oshikiri0Yohei Ozawa1On Suzuki2Masahiro Usuda3Go Miyata4Department of Digestive Surgery, Iwate Prefectural Central HospitalDepartment of Surgery, Tohoku University Graduate School of MedicineDepartment of Digestive Surgery, Iwate Prefectural Central HospitalDepartment of Digestive Surgery, Iwate Prefectural Central HospitalDepartment of Digestive Surgery, Iwate Prefectural Central HospitalAbstract Background Benign multicystic peritoneal mesothelioma (BMPM) is a benign tumor that usually occurs in middle-aged females. Although several published studies have reported the occurrence of this tumor in the abdominal cavity, few have documented its development in the inguinal region. Case presentation We present a case of a 48-year-old female presenting with a bulge in her left inguinal region. Physical examination revealed a golf ball-sized nodule in the left inguinal region that could not be pushed back into the abdominal cavity. Contrast-enhanced computed tomography showed a multicystic tumor; therefore, the patient was diagnosed with inguinal hernia or hydrocele of the Nuck’s canal. We performed surgical resection and hernia repair using the mesh plug method. The resected specimen was 80 mm in length and contained a multicystic tumor. Pathological examination showed that the cyst wall was lined by a single layer of cuboidal to single layer squamous epithelium. Immunohistochemistry revealed positivity for calretinin in the epithelial cells, for which a diagnosis of BMPM was established. The patient returned to our hospital after 5 years with symptoms similar to the previous episode, but this time in the right inguinal region. Imaging studies showed a tumor in the right inguinal region with the same characteristics as the previous one. The patient underwent tumor resection and hernia repair using the same technique. The resected tumor was 45 mm in length and had characteristics similar to the previously resected tumor. The presence of calretinin and D2-40 on immunohistochemistry led to the diagnosis of BMPM. There was no recurrence of BMPM for 33 months after the secondary surgery. Conclusions Here we present the first report of metachronous BMPM occurring in bilateral inguinal canals. Although the pathogenesis of BMPM remains unclear, reactive changes have been suggested to cause tumors originating from the groin. The treatment of choice for BMPM is surgical resection. For diagnosis, pathological examination with immunostaining can be useful. The most appropriate postoperative follow-up for inguinal BMPM is controversial, and the accumulation of more inguinal BMPM cases is needed.https://doi.org/10.1186/s40792-022-01399-5Benign multicystic peritoneal mesotheliomaMetachronous bilateral inguinal tumorInguinal noduleImmunohistochemistryCalretininD2-40
spellingShingle Hiroyuki Oshikiri
Yohei Ozawa
On Suzuki
Masahiro Usuda
Go Miyata
Benign multicystic peritoneal mesothelioma occurring in bilateral inguinal canals metachronously: a case report
Surgical Case Reports
Benign multicystic peritoneal mesothelioma
Metachronous bilateral inguinal tumor
Inguinal nodule
Immunohistochemistry
Calretinin
D2-40
title Benign multicystic peritoneal mesothelioma occurring in bilateral inguinal canals metachronously: a case report
title_full Benign multicystic peritoneal mesothelioma occurring in bilateral inguinal canals metachronously: a case report
title_fullStr Benign multicystic peritoneal mesothelioma occurring in bilateral inguinal canals metachronously: a case report
title_full_unstemmed Benign multicystic peritoneal mesothelioma occurring in bilateral inguinal canals metachronously: a case report
title_short Benign multicystic peritoneal mesothelioma occurring in bilateral inguinal canals metachronously: a case report
title_sort benign multicystic peritoneal mesothelioma occurring in bilateral inguinal canals metachronously a case report
topic Benign multicystic peritoneal mesothelioma
Metachronous bilateral inguinal tumor
Inguinal nodule
Immunohistochemistry
Calretinin
D2-40
url https://doi.org/10.1186/s40792-022-01399-5
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