Benign cystic mesothelioma associated with ipsilateral renal agenesis: a case report and review of literature

Our plain-English summary of Benign cystic mesothelioma associated with ipsilateral renal agenesis: a case report and review of literature, by D. Murro, A. Harbhajanka, B. Mahon, et al (Pediatric and Developmental Pathology, 2014).

The study

• Authors: D. Murro, A. Harbhajanka, B. Mahon, D. Deziel
• Journal: Pediatric and Developmental Pathology
• Published: 2014;17(6):487-90
• Type of article: Case report, Review
• Cited by: 7 articles, as counted by NIH iCite
• Read it: PubMed · Publisher

Abstract

Benign Cystic Mesothelioma (BCM) is an uncommon peritoneal lesion that usually occurs in reproductive age females with a history of abdominal surgery. Occasional expression of estrogen and progesterone receptor in these cells may explain female predilection. Reports of BCM in males are rare. We describe a case of BCM associated with ipsilateral renal agenesis in a young male without any surgical history. The cyst lining stained positive for cytokeratin, Wilms Tumor-1, epithelial membrane antigen, CD10, estrogen receptor, and progesterone receptor, and negative for PAX-8. Only three cases of BCM associated with congenital renal anomalies have been reported. To the best of our knowledge, this is the first case of BCM associated with ipsilateral renal agenesis in an adult male and the first male case of BCM displaying estrogen and progesterone receptor positivity. Such a case reveals the presence of congenital anomalies should be considered in patients with BCM.

In plain English

This article was a case report with a short review of earlier published cases. It described one 26-year-old man with a history of hypertension and no prior surgery. He came to an emergency department with a month of worsening abdominal and right flank pain. Scans showed many fluid-filled cysts in the abdomen and pelvis and no normal right kidney. During surgery, no right kidney or right ureter was found, and tests done during the operation showed the cysts were negative for malignancy. Laboratory staining of the cyst lining was consistent with benign cystic mesothelioma (BCM), a non-cancerous peritoneal condition also called peritoneal inclusion cysts. Some lining cells were estrogen receptor positive, and progesterone receptor showed focal positivity. Five weeks later he was doing well but was told his cysts were likely to recur. The authors stated that, to their knowledge, this was the first reported case of BCM with a missing kidney on the same side (ipsilateral renal agenesis) in an adult male, and the first male case of BCM positive for these hormone receptors. They concluded that congenital anomalies should be considered in patients with BCM. The article did not discuss asbestos.

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 of one patient, with a brief review of published cases of peritoneal cysts and benign cystic mesothelioma associated with renal anomalies
• Who was studied: One 26-year-old man with a history of hypertension who presented to an emergency department with abdominal and right flank pain
• Exposure: No asbestos or other exposure was studied or mentioned. The authors note the patient had no prior surgery.
• Main results:
  • CT scans showed a complex cystic mass with multiple septations and no normal right kidney.
  • At exploratory laparotomy, no right kidney or right ureter was present.
  • Removed specimens were right pelvic cysts (4.6 X 4.2 X 3.5 cm), peritoneal cysts (7 X 6.5 X 1 cm) and liver cysts (9.5 X 5 X 2.5 cm). Individual cyst diameters ranged from 2.5 to 5.5 cm.
  • The cyst lining cells were positive for markers including CK5, AE1/AE3, CD10, WT-1, EMA and calretinin, and negative for Pax8, CD31, CD34, mucin and PSA. Some epithelial cells were estrogen receptor positive, and progesterone receptor showed focal positivity.
  • The findings were consistent with a diagnosis of benign cystic mesothelioma.
  • Five weeks after surgery the patient was doing well. He was counseled to continue follow-up because his cysts were likely to recur.
• The authors' conclusion: The authors concluded that, to their knowledge, this was the first case of BCM associated with ipsilateral renal agenesis in an adult male and the first male BCM case with estrogen and progesterone receptor positivity. They stated that congenital anomalies should be considered in patients with BCM, and that their patient could support a developmental origin of BCM.
• Limitations the authors state:
  • The authors note that most published BCM cases did not report WT-1 staining, which limits comparison with their case.

Topics

Medical terms in this study, explained in our Lexicon: Mesothelioma, Epithelial Membrane Antigen (EMA).

Related studies

• Peritoneal benign cystic mesothelioma: a case report and review of the literature — S. van Ruth, M.W. Bronkhorst, et al. (2002)
• van Ruth S, Bronkhorst MWGA, van Coeverden F, Zoetmulder FAN. Peritoneal benign cystic mesothelioma: a case report and review of literature. Eur J Surg Oncol 2002; 28(2): 192-5 — K. Sethna, F. Mohamed, et al. (2002)
• Benign cystic mesothelioma of the peritoneum in a child-case report and review of the literature — V.C. Shakya, C.S. Agrawal, et al. (2011)
• Giant benign cystic peritoneal mesothelioma: US, CT, and MRI findings — A. Ozgen, D. Akata, et al. (1998)
• Imaging appearance of benign multicystic peritoneal mesothelioma: a case report and review of the literature — V. Mehta, V. Chowdhary, et al. (2017)
• Pediatric Benign Cystic Peritoneal Mesothelioma — J. Durell, H. Dagash, et al. (2016)

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