Two familial mesothelioma cases with high concentrations of soluble cytokeratin 19 fragment in pleural fluid

Our plain-English summary of Two familial mesothelioma cases with high concentrations of soluble cytokeratin 19 fragment in pleural fluid, by J. Hiyama, M. Marukawa, Y. Shiota, et al (Internal Medicine, 1998).

The study

• Authors: J. Hiyama, M. Marukawa, Y. Shiota, T. Ono, S. Imai, K. Motohiro, J. Aoki, N. Sasaki, K. Taniyama, H. Mashiba
• Journal: Internal Medicine
• Published: 1998;37(4):407-10
• Type of article: Case report
• Cited by: 8 articles, as counted by NIH iCite
• Read it: PubMed · Publisher

Abstract

We report two cases of diffuse malignant pleural mesothelioma occurring almost simultaneously in one family. Patient 1 was a 42-year-old Japanese man who had worked as an electrical engineer for 25 years. Patient 2, his mother, was 69 years old. She lived for 10 years with patient 1 after he started his work, and also worked at a shipyard herself for 6 years. The concentrations of cytokeratin subunit 19 fragment (CYFRA 21-1) in pleural fluid of the two patients were 1,500 ng/ml, and 1,200 ng/ml, respectively. Measurement of CYFRA 21-1 concentration in the pleural fluid may be a useful tool for a diagnosis of malignant mesothelioma.

In plain English

This case report described a son and mother treated at Kure Kyosai Hospital in Kure, Japan. Both had biphasic diffuse malignant pleural mesothelioma confirmed by open biopsy, and they were admitted in June 1995, two weeks apart. Patient 1, aged 42, had worked about 25 years as an electrical engineer, with occupational asbestos exposure from handling insulators and wiring. Patient 2, his mother, aged 69, had worked 4 years at a shipyard and 2 at a steel plant, and lived with him about 10 years after he began work. Lung tissue held 86 asbestos bodies per 5 g of wet tissue in the son, and eight in the mother. Fiber types were not reported. Pleural fluid CYFRA 21-1, a tumor marker, was 1,500 ng/ml in the son and 1,200 ng/ml in the mother. These levels were significantly higher than in 32 benign pleural effusion cases (median 23.5 ng/ml; p less than 0.02). The authors said asbestos could not be excluded as a cause, but a genetic factor may have been responsible, given the low asbestos load. They concluded that measuring CYFRA 21-1 in pleural fluid may help diagnosis.

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 related patients, with a comparison of pleural fluid CYFRA 21-1 against 32 benign pleural effusion cases
• Who was studied: A 42-year-old Japanese man and his 69-year-old mother, both admitted to Kure Kyosai Hospital, Kure, Japan, in June 1995 (the mother two weeks after the son)
• Exposure: Son: occupational asbestos exposure as an electrical engineer for about 25 years, handling insulators and wiring; smoker for 20 years (Brinkman index 500). Mother: work at a shipyard for 4 years and a steel plant for 2 years, plus indirect asbestos exposure at home from her son; never smoked. Fiber types were not identified.
• Main results:
  • Son (patient 1): pleural fluid CYFRA 21-1 concentration was 1,500 ng/ml
  • Son: biopsy of parietal pleura nodules showed biphasic mesothelioma
  • Son: 86 asbestos bodies per 5 g of wet lung tissue (37 ferrous bodies and 49 naked bodies)
  • Mother (patient 2): pleural fluid CYFRA 21-1 concentration was 1,200 ng/ml
  • Mother: open biopsy nodules showed biphasic mesothelioma
  • Mother: only eight asbestos bodies per 5 g of wet lung tissue, all naked bodies
  • Comparison group: 32 benign pleural effusion cases (including tuberculosis pleurisy, heart failure, and 3 benign asbestos pleural effusions)
  • CYFRA 21-1 in the two mesothelioma cases was significantly higher than in the benign cases. The benign cases had a median of 23.5 ng/ml (interquartile range 8.1 to 36.0 ng/ml). The Mann-Whitney U test comparing the two groups gave p less than 0.02.
  • The authors stated that asbestos exposure could not be excluded as a cause of these familial cases, because mesothelioma may be associated with minimal exposure
  • The authors suggested a genetic factor may have been responsible, because of the low asbestos load in lung tissue
• The authors' conclusion: The authors concluded that measuring CYFRA 21-1 in pleural fluid may be useful in diagnosing malignant pleural mesothelioma, and that further studies are needed.

Topics

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

Medical terms in this study, explained in our Lexicon: Mesothelioma, Pleural Mesothelioma, Malignant Mesothelioma.

Related studies

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• Mesothelin-related predictive and prognostic factors in malignant mesothelioma: a nested case-control study — O.D. Roe, J. Creaney, et al. (2008)
• Evaluation of pleural CYFRA 21-1 and carcinoembryonic antigen in the diagnosis of malignant pleural effusions — G. Salama, M. Miédougé, et al. (1998)
• Value of the mesothelium-associated antibodies thrombomodulin, cytokeratin 5/6, calretinin, and CD44H in distinguishing epithelioid pleural mesothelioma from adenocarcinoma metastatic to the pleura — P.M. Cury, D.N. Butcher, et al. (2000)

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