Remote Recurrence of Benign Multicystic Peritoneal Mesothelioma

Our plain-English summary of Remote Recurrence of Benign Multicystic Peritoneal Mesothelioma, by C.E. Lee, A. Agrawal (Journal of Obstetrics and Gynaecology Canada, 2017).

The study

• Authors: C.E. Lee, A. Agrawal
• Journal: Journal of Obstetrics and Gynaecology Canada
• Published: 2017;39(11):1042-1045
• Type of article: Case report
• Cited by: 8 articles, as counted by NIH iCite
• Read it: PubMed · Publisher

Abstract

Background: Benign peritoneal cystic mesothelioma (BPCM) is a rare disease entity that arises from mesothelioma cells. We describe a rare case of BPCM recurrence 36 years after its initial presentation.

Case: A 62-year-old woman was referred to an outpatient gynaecologic oncology clinic with an incidental finding of multiple pelvic cysts. She had a preceding history of known BPCM treated with extensive debulking surgery. She presented after 36 years of clinical remission. A repeat laparotomy for a debulking surgical procedure confirmed a recurrence of BPCM.

Conclusion: Our current case represents a woman with a remote recurrence of BPCM after initial optimal debulking surgery. Her clinical presentation of recurrence after 36 years illustrates the need for long-term follow-up and clinical suspicion in symptomatic patients with previously diagnosed BPCM.

Crown Copyright © 2017. Published by Elsevier Inc. All rights reserved.

In plain English

This case report described one woman whose benign peritoneal cystic mesothelioma (BPCM) came back after a long remission. BPCM is a rare, non-cancerous tumour of the lining of the abdomen. The article did not mention asbestos or any occupational or environmental exposure. After an operation for a benign ovarian cyst at 17, she had surgery at 26 for pelvic lesions confirmed as BPCM. Nine months later a large pelvic cyst led to a second BPCM operation, including removal of her uterus, tubes and ovaries, aimed at optimal debulking. She then took hormone replacement therapy and stayed in clinical remission for 36 years. Shortly after she stopped it, at age 62, scans done for another problem showed multiple pelvic cysts. The cysts stayed stable for a year. Surgery then removed all visible disease, and pathology confirmed benign mesothelial cysts. The authors concluded that the case shows the need for long-term follow-up even after optimal surgery. They noted that the timing of the recurrence was not definite, and suggested the case may argue somewhat against the theory that BPCM depends on hormones.

We wrote this plain-English summary from the full article with the help of AI, and a second AI model checked each sentence against the article. It is not the authors' wording; the abstract and the article are their own account.

What the study did and found

• Design: case report
• Who was studied: One woman, aged 62 when referred to an outpatient gynaecologic oncology clinic, with a history of BPCM first treated surgically at age 26 (authors at the University of Saskatchewan, Saskatoon)
• Exposure: No asbestos or other occupational or environmental exposure is reported. The article describes surgery and hormone replacement therapy in this patient, and the timing of recurrence relative to stopping that therapy.
• Main results:
  • After a laparotomy for a benign ovarian cyst at age 17, the patient had a first laparotomy for BPCM at age 26 for multiple pelvic lesions confirmed by pathology.
  • Nine months later a large pelvic peritoneal cyst led to a repeat laparotomy with total abdominal hysterectomy and removal of both ovaries and tubes, with the goal of optimal debulking. Pathology again confirmed BPCM.
  • After surgery she was prescribed hormone replacement therapy and remained in clinical remission for 36 years.
  • Shortly after she stopped hormone replacement therapy, multiple pelvic cysts were found incidentally during investigations for cholestatic symptoms. They were seen on CT and MRI in several pelvic locations, including along blood vessels, the rectum and the urinary bladder.
  • The lesions stayed stable in size over a year. All visible disease was removed at laparotomy, and pathology confirmed benign mesothelial cysts with benign nuclei and no papillary architecture.
• The authors' conclusion: The authors concluded that the case highlights the importance of long-term follow-up to monitor for remote recurrence even after optimal debulking surgery for BPCM. They also said the case may lend some opposition to the theory that BPCM depends on hormones.
• Limitations the authors state:
  • The authors stated that the time of recurrence is not definite. The cysts may have been present long before she stopped the hormones, since they remained stable in size and number for almost a year.

Topics

Medical terms in this study, explained in our Lexicon: Peritoneal Mesothelioma, Mesothelioma, Cytoreductive Surgery, Laparotomy.

Related studies

• A recurrence of benign multicystic peritoneal mesothelioma treated through laparoscopic excision: a case report and review of the literature — T.D. Witek, J.W. Marchese, et al. (2014)
• Benign multicystic peritoneal mesothelioma: a rare cause of intra-abdominal cystic disease — B. Campbell, D. Mehanna, et al. (2017)
• Multicystic peritoneal mesothelioma: A short review — C.H. Zhang, J.W. Yu, et al. (2017)
• Benign multicystic mesothelioma and peritoneal inclusion cysts: are they the same clinical and histopathological entities? A systematic review to find an evidence-based management — A.M.C. Rapisarda, A. Cianci, et al. (2018)
• Benign Multicystic Peritoneal Mesothelioma: AIRP Best Cases in Radiologic-Pathologic Correlation — R. Lee, A. Tong, et al. (2016)
• Pediatric Benign Cystic Peritoneal Mesothelioma — J. Durell, H. Dagash, et al. (2016)

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