Case report: malignant peritoneal mesothelioma in two siblings

Our plain-English summary of Case report: malignant peritoneal mesothelioma in two siblings, by A.H. Picklesimer, V. Zanagnolo, T.H. Niemann, et al (Gynecologic Oncology, 2005).

The study

• Authors: A.H. Picklesimer, V. Zanagnolo, T.H. Niemann, L.A. Eaton, L.J. Copeland
• Journal: Gynecologic Oncology
• Published: 2005;99(2):512-6
• Type of article: Case report
• Cited by: 6 articles, as counted by NIH iCite
• Read it: PubMed · Publisher

Abstract

Background: Mesothelioma is a rare tumor, linked with occupational asbestos exposure. This association has been used to explain clustering of cases within families. Newer evidence, however, supports a possible genetic predisposition for this tumor.

Case: Our patient's brother was diagnosed with advanced stage malignant peritoneal mesothelioma in August 1995 at age of 42, he underwent tumor-reductive surgery followed by chemotherapy. He underwent a repeat cytoreductive surgery in September 1996 and further chemotherapy. He died of disease in December 1996. Our patient underwent cytoreductive surgery in May 1999 at age of 49 for advanced stage malignant peritoneal mesothelioma with suboptimal debulking. She received multiple chemotherapy regimens, including most recently experimental targeted agents, for slow progressing disease. She is presently alive with clinical disease 6 years from diagnosis.

Conclusion: This is the first report of two siblings of different gender with malignant peritoneal mesothelioma and only average environmental asbestos exposure. It is highly likely that the family described in this case report has some form of inherited susceptibility to malignancy cancer gene, HLA type, or tumor suppressor gene mutation.

In plain English

This case report described a brother and sister, two of twelve children, who both developed malignant peritoneal mesothelioma (a cancer of the abdominal lining), confirmed by tissue examination. The brother, born in 1953, was a salesman with a possible history of asbestos exposure at his elementary school. He first sought care in August 1995, had two tumor-reducing operations with chemotherapy, and died in December 1996. The sister, born in 1950, taught for 25 years at the elementary school both had attended and recalled the summer when asbestos-containing insulation was removed from it. She had surgery in May 1999, received many chemotherapy regimens including experimental drugs, and was still living when the report was written. Other relatives had many cancers, including a maternal aunt with pleural mesothelioma. The authors described the siblings as having no occupational or other significant asbestos exposure. They wrote that it was highly likely the family had some form of inherited susceptibility, and said future cases should be evaluated for genetic abnormalities.

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 two siblings (familial mesothelioma), with a description of the family's cancer history
• Who was studied: A brother (born 1953) and a sister (born 1950), two of twelve children in one family, both with histologically verified malignant peritoneal mesothelioma. The brother was treated starting in 1995 and 1996, and the sister starting in 1999. The report gives a US university institution but no family location.
• Exposure: No fiber type is named. The brother had a possible history of asbestos exposure at his elementary school. The sister taught for 25 years at the same school and recalled the summer when asbestos-containing insulation was removed from the building. The authors describe the siblings as having no occupational or other significant asbestos exposure. Elsewhere they call it 'only average environmental asbestos exposure' and 'no definitive asbestos exposure'.
• Main results:
  • Brother: worked as a salesman, with a possible history of asbestos exposure at his elementary school.
  • Brother: peritoneal mesothelioma was confirmed by tissue examination after surgery. He had a steady decline and died in December 1996. No autopsy was performed.
  • Sister: an elementary school teacher for 25 years at the school both siblings attended. She recalled the removal of asbestos-containing insulation from the building.
  • Sister: peritoneal mesothelioma was confirmed by tissue examination after surgery in May 1999. She received multiple chemotherapy regimens and was still living at the time of the report.
  • Family: many cancers in other relatives, including one of three maternal aunts who died with cancer, whose cancer was a pleural mesothelioma.
• The authors' conclusion: The authors concluded that this was the first report of two siblings of different gender with malignant peritoneal mesothelioma without definitive asbestos exposure. In their abstract they call it highly likely that the family has some form of inherited susceptibility. In their discussion they say the case suggests the possibility of an inherited predisposition, and that future cases should be evaluated for genetic abnormalities.

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: Peritoneal Mesothelioma, Mesothelioma, Asbestos Exposure, Chemotherapy, Cytoreductive Surgery, Tumor Suppressor Gene.

Related studies

• Detecting germline BAP1 mutations in patients with peritoneal mesothelioma: benefits to patient and family members — M. Kittaneh, C. Berkelhammer (2018)
• Well-differentiated papillary mesothelioma: clustering in a Portuguese family with a germline BAP1 mutation — C. Ribeiro, S. Campelos, et al. (2013)
• Hemizygosity of Nf2 is associated with increased susceptibility to asbestos-induced peritoneal tumours — J. Fleury-Feith, C. Lecomte, et al. (2003)
• BAP1 mutation is a frequent somatic event in peritoneal malignant mesothelioma — H. Alakus, S.E. Yost, et al. (2015)
• Differential Susceptibility of Human Pleural and Peritoneal Mesothelial Cells to Asbestos Exposure — J. Dragon, J. Thompson, et al. (2015)
• Genomic copy number alterations in 33 malignant peritoneal mesothelioma analyzed by comparative genomic hybridization array — P. Chirac, D. Maillet, et al. (2016)

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