Benign multicystic mesothelioma and peritoneal inclusion cysts: are they the same clinical and histopathological entities? A systematic review to find an evidence-based management

Our plain-English summary of Benign multicystic mesothelioma and peritoneal inclusion cysts: are they the same clinical and histopathological entities? A systematic review to find an evidence-based management, by A.M.C. Rapisarda, A. Cianci, S. Caruso, et al (Archives of Gynecology and Obstetrics, 2018).

The study

• Authors: A.M.C. Rapisarda, A. Cianci, S. Caruso, S.G. Vitale, G. Valenti, E. Piombino, S. Cianci
• Journal: Archives of Gynecology and Obstetrics
• Published: 2018;297(6):1353-1375
• Type of article: Systematic review
• Cited by: 54 articles, as counted by NIH iCite
• Read it: PubMed · Publisher

Abstract

Purpose: Peritoneal mesothelial cysts (PMC) are a clinical dilemma because of their true pathogenic nature. Many definitions have been associated with PMC, including "benign multicystic mesothelioma", "cystic mesothelioma", "multilocular peritoneal inclusion cysts", ''inflammatory cysts of the peritoneum" or "postoperative peritoneal cyst".

Methods: We herein performed a systematic review of the literature focusing on clinical and histopathological aspects of PMC, diagnosis, and therapies. Moreover, we described our experience with a case of PMC in a young female.

Results: Since there is often a history of prior surgery or inflammatory disease, most authors consider PMC of reactive origin. However, in some cases they occur without any documentable signs of disease or injury. A variety of clinical findings can complicate the preoperative assessment and a multitude of histological pictures may potentially lead to a misdiagnosis. The absence of a uniform treatment strategy and lack of long-term follow-up often hinder the accurate definition leading to unnecessary or unnecessarily aggressive therapy.

Conclusions: PMC are more common than had previously been thought. Most authors consider them non-neoplastic; thus the designation of "peritoneal inclusion cyst" is preferable. The term "mesothelioma" should be used only in cases of histological evidences of atypia. The high rates of recurrence suggest that the goal of treatment should not be necessarily complete eradication, but symptomatic relief through individualized treatment. This is a topic of particular importance, especially in young female where recurrence rates could be lower than those reported in adults and where an improperly aggressive treatment could have repercussions on fertility.

In plain English

This article was a systematic review of reports on peritoneal mesothelial cysts (PMC), cysts of the abdominal and pelvic lining also called "benign multicystic mesothelioma" or "peritoneal inclusion cysts", plus one case report. The authors searched PubMed, found 338 articles from 1960 to 2017, and selected 157. They analysed 21 published cases in young females selected for ages 10 to 20 (actual range 11 to 20, mean 16.7 years at diagnosis). Pelvic pain was the most common symptom (ten cases), and ten patients had had earlier surgery. Most were treated by surgical removal. Recurrence occurred in three cases over a mean follow-up of 33.9 months; one patient was lost to follow-up and eight cases had no recurrence data. In the authors' own case, a 19-year-old woman with chronic pelvic pain, the cyst was removed by laparoscopy and no mass was seen 3 months later. The authors concluded that "peritoneal inclusion cyst" is the preferable name and treatment should aim at symptom relief. They noted reports were mostly small with short follow-up. The article did not study asbestos exposure.

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: Systematic review of published case reports and case series, combined with a single case report from the authors' own department
• Who was studied: 157 selected articles from a PubMed search that found 338 articles (published 1960 to 2017). A sub-analysis covered 21 published cases in young females, selected for ages 10–20 years. The cases' actual ages ranged from 11 to 20 years, with a mean age at diagnosis of 16.7 years. Separately, one 19-year-old woman was treated by the authors.
• Exposure: Not studied. The article examines peritoneal mesothelial cysts and their diagnosis and treatment. Asbestos is mentioned only in the discussion, in describing other studies of malignant mesothelioma.
• Main results:
  • The search found 338 articles published from 1960 to 2017. 181 were excluded and 157 were selected.
  • Among the 21 young female cases, the mean age at diagnosis was 16.7 years (range 11–20 years).
  • Among the 21 young female cases, pelvic pain was the most common presenting symptom (ten cases).
  • Among the 21 young female cases, ten patients had undergone previous surgery.
  • In most of the young female cases, treatment was surgical removal by laparoscopy or laparotomy. Three cases were treated by cyst drainage.
  • In the young female cases, mean follow-up was 33.9 months (range 2–253 months). One patient was lost to follow-up and recurrence was not reported in eight cases.
  • Recurrence occurred in three of the young female cases. In two of these, the previous treatment was cyst drainage.
  • In the authors' own case, a 19-year-old woman with chronic pelvic pain, the cyst was removed by laparoscopy and the diagnosis of PMC was confirmed by histology. No pelvic mass and no raised cancer markers were found 3 months after the operation.
• The authors' conclusion: The authors concluded that most authors regard these lesions as non-neoplastic, so "peritoneal inclusion cyst" is the preferable name. In their view, treatment should aim at symptom relief rather than complete eradication. They also suggested that recurrences may follow a less aggressive course in young females.
• Limitations the authors state:
  • The published literature consists mostly of case reports and small case series.
  • Short follow-up times in published reports made firm conclusions difficult.
  • Some features of the young female cases were only partially reported by the original authors.
  • Most young female case reports did not record tumor markers, so this aspect could not be analysed.
  • Some findings in the young female group may reflect the small sample size.

Topics

The National Library of Medicine indexes this study under subjects our site covers: Mesothelioma, Peritoneal mesothelioma.

Medical terms in this study, explained in our Lexicon: Multicystic Mesothelioma (MCM), Systematic Review, Mesothelioma, Peritoneum, Misdiagnosis.

Related studies

• Multilocular peritoneal inclusion cyst (multicystic mesothelioma) with hyaline globules — J. Lamovec, J. Sinkovec (1996)
• Collagenous Spherulosis in Well-Differentiated Papillary Mesothelioma in a Female With Multilocular Peritoneal Inclusion Cysts — B. AbdullGaffar, L.G. Raman (2019)
• Loose mesothelial cysts in the peritoneal cavity — Y. Ramirez, J. Rosai, et al. (2010)
• A rare case of benign multicystic peritoneal mesothelioma misdiagnosed as hydatid cyst found in the liver parenchyma and abdomen cavity of a male with asbestos exposure — A.B. Kelarji, M.S. Alshutaihi, et al. (2021)
• Selected topics in peritoneal pathology — P.M. Baker, P.B. Clement, et al. (2014)
• Peritoneal, benign, cystic mesothelioma with free-floating cysts, re-examined by new methods. A case report — O.H. Iversen, T. Hovig, et al. (1988)

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