Heterologous sarcomatoid pleural mesothelioma with osteosarcomatous differentiation: a report of autopsy case that accomplished trimodality therapy and review of the literature

Our plain-English summary of Heterologous sarcomatoid pleural mesothelioma with osteosarcomatous differentiation: a report of autopsy case that accomplished trimodality therapy and review of the literature, by H. Itano, T. Takeda, T. Yamada, et al (General Thoracic and Cardiovascular Surgery, 2020).

The study

• Authors: H. Itano, T. Takeda, T. Yamada, M. Koide, T. Kobayashi
• Journal: General Thoracic and Cardiovascular Surgery
• Published: 2020;68(8):871-879
• Type of article: Case report, Review
• Cited by: 3 articles, as counted by NIH iCite
• Read it: PubMed · Publisher

Abstract

Heterologous mesothelioma is a very rare subtype of sarcomatoid mesothelioma characterized by the presence of malignant heterologous elements. A 69-year-old man with a strong history of asbestos exposure presented with a 5-cm mass in his chest wall, destroying the right 5th rib and spreading along the parietal pleura, on a CT. Biopsy revealed heterologous mesothelioma with osteosarcomatous elements, following which left extrapleural pneumonectomy was performed with combined resection of pericardium, hemidiaphragm, and 4th, 5th, and 6th costal segments. A small cytokeratin-positive epithelioid component in the resected tumor definitively confirmed the diagnosis. Post-operative chemotherapy and intensity-modulated radiotherapy were undertaken. After 12-month disease-free period post treatment, rapid intraperitoneal recurrence resulted in death. Autopsy revealed no tumors in the left thorax. We present here a case of heterologous osteosarcomatous pleural mesothelioma that followed a unique clinical course after trimodality therapy. In addition, literature of 54 cases of the similar heterologous mesothelioma was reviewed.

In plain English

This was a case report with a review of published cases. It described a 69-year-old man in Japan, a demolition worker for 40 years, with heavy asbestos exposure. Biopsied lung held 35,523 asbestos bodies per gram of dry lung. Tests showed heterologous sarcomatoid pleural mesothelioma with bone-forming (osteosarcomatous) features, a rare subtype. In 2010 he had surgery removing the left lung and pleura, the pericardium, half the diaphragm and three rib segments. He then had chemotherapy and radiotherapy. The radiotherapy was stopped early after breathing problems from radiation pneumonitis and possible cytomegalovirus pneumonia. After 12 disease-free months, the cancer returned rapidly in the abdomen and he died within a month. Autopsy found no tumor in the left chest but found tumors in the right lung, the right pleura and the abdomen. Reviewing 54 published similar cases, the authors found asbestos exposure confirmed in 34 (63%) and a median survival of 6.0 months among 36 cases with data. The authors concluded that the treatment achieved local control but could not prevent abdominal recurrence.

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 (with autopsy) plus a review of 54 published cases of the same tumor type
• Who was studied: one 69-year-old man treated at a hospital in Uji, Japan, first seen in April 2010 with left-sided chest pain; plus 54 previously published cases of pleural heterologous mesothelioma with osteo- or chondrosarcomatous elements
• Exposure: occupational asbestos exposure as a demolition worker for 40 years; asbestos body count of 35,523 per gram of dry lung in biopsied lung. No fiber type is given.
• Main results:
  • Diagnosis in the patient: heterologous sarcomatoid pleural mesothelioma with osteosarcomatous differentiation. It was confirmed on the surgical specimen by finding a small epithelioid mesothelioma component that was positive for mesothelial and epithelial markers.
  • The patient underwent left extrapleural pneumonectomy on July 13, 2010, with removal of the pericardium and hemidiaphragm and the 4th, 5th and 6th rib segments.
  • The patient received 3 courses of cisplatin and pemetrexed. Radiotherapy (IMRT) was stopped at a total dose of 43.2 Gy because of respiratory insufficiency from radiation pneumonitis and possible cytomegalovirus pneumonia.
  • After a 12-month disease-free period following trimodality therapy, the patient had rapid intraperitoneal recurrence and died within a month.
  • The patient's serum alkaline phosphatase was 402 IU/mL before surgery (normal range 115–359 IU/mL). It normalized after surgery, then rose rapidly with the recurrence, peaking at 4209 IU/mL at death.
  • At the patient's autopsy, the left thorax was tumor-free. Autopsy found pulmonary metastases in the right middle and lower lobes, small right pleural dissemination, calcified mesenteric tumors up to 11 cm, numerous peritoneal nodules and 3500 cc of malignant ascites.
  • In the 54 reviewed published cases, median age at diagnosis was 68.0 years (range 27–85). There were 45 men (83%) and 9 women (17%). Asbestos exposure was confirmed in 34 cases (63%) and parietal pleural plaques were identified in 20 (37%).
  • In the reviewed cases, median survival among 36 cases with available data was 6.0 months after diagnosis. In the 7 cases with diffuse spread and diffuse calcification, median survival was 5.0 months.
  • Among the 54 reviewed cases, only 2 underwent extrapleural pneumonectomy, none had trimodality therapy, and only 7 (13%) had an autopsy. The authors described the present case as the first reported such case to undergo trimodality therapy with outcome assessed by autopsy.
• The authors' conclusion: The authors concluded that trimodality therapy achieved local control in this patient but could not prevent abdominal recurrence. They also concluded that immunohistochemistry panels were effective in diagnosis.

Topics

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

Medical terms in this study, explained in our Lexicon: Sarcomatoid, Pleural Mesothelioma, Autopsy, Trimodality Therapy, Mesothelioma, Heterologous Elements, Asbestos Exposure, Parietal Pleura.

Related studies

• Sarcomatoid pleural mesothelioma with osteosarcomatous, chondrosarcomatous and rhabdomyoblastic elements: an extremely rare autopsy case — Y. Tsukamoto, H. Hao, et al. (2015)
• Patterns of failure after trimodality therapy for malignant pleural mesothelioma — E.H. Baldini, A. Recht, et al. (1997)
• Post-irradiation pericardial malignant mesothelioma: an autopsy case and review of the literature — M. Bendek, M. Ferenc, et al. (2010)
• Sarcomatoid mesothelioma with osteoid differentiation — M. Evison, P.A. Crosbie, et al. (2013)
• Malignant mesothelioma with heterologous elements: clinicopathological correlation of 27 cases and literature review — S. Klebe, A. Mahar, et al. (2008)
• Primary malignant pericardial sarcomatoid mesothelioma: An autopsy report — H. Muta, Y. Sugita, et al. (2017)

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