Pleural Rosai-Dorfman disease complicated with renal clear cell carcinoma: a case report and literature review

Our plain-English summary of Pleural Rosai-Dorfman disease complicated with renal clear cell carcinoma: a case report and literature review, by S. Tang, X. Yang, S. Wang, et al (Frontiers in Oncology, 2024).

The study

• Authors: S. Tang, X. Yang, S. Wang, Q. Xiao
• Journal: Frontiers in Oncology
• Published: 2024;14:1476243
• Type of article: Case report
• Read it: PubMed · Publisher · Free full text

Abstract

Background: Rosai-Dorfman disease (RDD), also known as sinus histiocytosis with massive lymphadenopathy, is a rare non-malignant disorder characterized by excessive proliferation of histiocytes, the cause of which remains unknown. Although the lymph nodes are the most commonly affected site, some patients may present with extranodal involvement, particularly in the skin, nasal cavity, eyes, and bones. In this report, we aim to present a unique case of RDD with pleural involvement in a 61-year-old patient. To our knowledge, only a few cases of RDD primarily manifesting as pleural disease have been reported thus far.

Case Summary: A 61-year-old male with left brachial plexus neuropathy was found to have a space-occupying lesion in the left posterior pleura. Initial chest CT showed a 5.7cm × 1.3cm soft tissue density shadow, which grew to 6.8cm × 1.7cm two years later. Given a history of clear cell renal cell carcinoma, pleural metastasis was suspected. The patient declined a needle biopsy but underwent surgical excision. Postoperative pathology and immunohistochemistry confirmed Rosai-Dorfman disease (RDD). After surgery, the patient received anti-infection, phlegm reduction, rehydration, and analgesia treatments and recovered well.

Conclusion: RDD originating in pleura is rare, its clinical and imaging manifestations lack specificity, and it is easy to be misdiagnosed. It is crucial to consider the possibility of RDD when encountering nodular pleural masses or lamellar soft tissue lesions. Differential diagnosis should include pleural mesothelioma, pleural metastases, solitary fibrous neoplasms, and schwannomas of the pleura. Ultimately, a definitive diagnosis can be achieved through pathology and immunohistochemistry analysis.

Copyright © 2025 Tang, Yang, Wang and Xiao.

In plain English

This article reported a single patient and briefly reviewed earlier published cases. It does not mention asbestos. The patient, a 61-year-old man in Chongqing, China, had had surgery for left renal clear cell carcinoma, a kidney cancer, one year earlier. A chest CT scan more than two years before had shown a soft tissue shadow on the left pleura (the lining around the lung) of about 5.7cm by 1.3cm. A later scan showed it at about 6.8 by 1.7cm. Spread of the kidney cancer was suspected. The patient declined a needle biopsy and had the lesion removed by thoracoscopic surgery. Tissue tests confirmed Rosai-Dorfman disease, a rare overgrowth of immune cells called histiocytes, arising in the pleura. CT scans up to 2.5 years after surgery showed mild left pleural thickening. The authors concluded that pleural Rosai-Dorfman disease is easily misdiagnosed and must be told apart from pleural mesothelioma, pleural metastases, solitary fibrous tumors and schwannomas. They added that tissue examination is needed to confirm the 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 one patient, with a literature review of previously reported pleural Rosai-Dorfman disease cases
• Who was studied: one 61-year-old man with a left posterior pleural lesion present for over two years, seen at the Army Specialty Medical Center (authors' affiliations in Chongqing, China)
• Main results:
  • The patient had a left renal clear cell carcinoma removed one year before presentation
  • On the initial chest CT, the larger pleural lesion measured about 5.7cm by 1.3cm, with no abnormality in the adjacent ribs
  • Two years later, a follow-up enhanced CT showed the lesion had grown to about 6.8 by 1.7cm
  • Because of the kidney cancer history, spread of cancer to the pleura was suspected
  • The patient refused a needle biopsy and had the lesion removed by thoracoscopic surgery
  • During surgery, a small amount of carbon dust deposition was seen in the lung
  • Pathology and immunohistochemistry led to a definitive diagnosis of primary Rosai-Dorfman disease of the pleura. The tissue was S100 positive, and no BRAF V600E mutation was detected
  • Immunohistochemistry showed scattered IgG4 expression, which the authors said indicated a potential association with IgG4-related diseases
  • Follow-up CT scans at 4 months, 1.5 years and 2.5 years after surgery showed mild left pleural thickening and minimal chronic inflammation in nearby lung tissue
• The authors' conclusion: The authors concluded that pleural Rosai-Dorfman disease is rare and easily misdiagnosed. They said it must be distinguished from pleural metastatic tumors, pleural mesothelioma, solitary fibrous tumors of the pleura and schwannoma, and that histopathological examination is needed to confirm the diagnosis.

Topics

Medical terms in this study, explained in our Lexicon: Carcinoma, Lymphadenopathy, Pleura, Metastasis, Needle Biopsy, Pathology, Immunohistochemistry, Differential Diagnosis.

Related studies

• Rosai-Dorfman disease presenting as a solitary mediastinal mass — A.I. Hida, S. Yagi, et al. (2009)
• A Case of Rosai-Dorfman Disease Presenting as an Isolated Pleural Plaque — A. Rathinam, S. Gupta, et al. (2023)
• Carbonic anhydrase IX is expressed in mesothelioma and metastatic clear cell renal cell carcinoma of the lung — M.L. Ramsey, B.J. Yuh, et al. (2012)
• Genomic profiling of the genes on chromosome 3p in sporadic clear cell renal cell carcinoma — Y. Togo, Y. Yoshikawa, et al. (2016)
• Acne inversa complicated by squamous cell carcinoma in association with diffuse malignant peritoneal mesothelioma arising in the absence of predisposing factors: a case report — V. Barresi, E. Vitarelli, et al. (2008)
• Primary pleural epithelioid mesothelioma of clear cell type: a case report and review of current literature — C. Gkogkou, K. Samitas, et al. (2011)

Browse more research in Frontiers in Oncology, or return to medical research.