Case report reveals an unprecedented instance of xanthomatous pleuritis in a child with recurrent pneumonia, suggesting a critical role for histopathology in diagnosis.
Background Xanthomatous pleuritis is an exceptionally rare benign inflammatory condition characterized by aggregates of lipid-laden macrophages within the pleura. To the best of our knowledge, only three adult cases have been reported worldwide, associated with chronic pleural inflammation or infection. Pediatric pleural xanthomas are exceedingly uncommon, and xanthomatous pleuritis has not been previously reported in a child. Case Presentation We report a 3.5-year-old girl with recurrent pneumonia, chronic respiratory symptoms, and Failure to Thrive (FTT). Chest X ray (CXR) revealed cardiomegaly and subpleural nodules, followed by high resolution Computed Tomography scan (CT scan) showing patchy right-sided consolidation, mild pleural thickening, subpleural nodules and loculated effusion. Diagnostic thoracentesis was performed; however, only a minimal amount of loculated fluid was obtained. CT-guided biopsy of a pleural-based nodule demonstrated numerous lipid-laden (foamy) macrophages with scattered multinucleated histiocytes, without granuloma or malignancy, supporting the diagnosis of xanthomatous pleuritis. Further evaluations for tuberculosis, cystic fibrosis, and primary immunodeficiency were negative. Echocardiography identified a patent ductus arteriosus (PDA). The child improved clinically with intravenous (IV) broad-spectrum antibiotics and then after Amoxicillin-Clavulanate therapy. She later underwent successful PDA closure. Conclusion To the best of our knowledge, this case represents the first pediatric case with xanthomatous pleuritis described in the literature and emphasizes the importance of histopathologic evaluation in children with unexplained recurrent pulmonary disease.
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