Esophageal Xanthogranulomatous Inflammation with Peripheral Eosinophilia in an Immunosuppressed Patient with Rheumatoid Arthritis: A Rare Case Report and Focused Literature Review

Background: Xanthogranulomatous inflammation of the esophagus is an exceedingly rare lesion characterized by extensive xanthogranulomatous tissue infiltration, often mimicking neoplasms. The association of esophageal xanthogranuloma with peripheral eosinophilia in immunosuppressed patients remains poorly understood, posing diagnostic challenges.Objectives: This study aimed to present a rare case of esophageal xanthogranuloma with tissue eosinophilia in a patient with rheumatoid arthritis on multiple immunosuppressive agents. In addition, this study examined clinicopathological features, assessed potential pathogenic linkages, and revealed diagnostic considerations.Methods: A ۵۲-year-old man with a ۱۷-year history of rheumatoid arthritis on immunosuppressants presented with progressive dysphagia and gastrointestinal bleeding. The diagnostic workup included contrast-enhanced CT, upper endoscopy, endoscopic ultrasound (EUS), and additional biopsies, all of which were inconclusive. Due to persistent symptoms and nondiagnostic minimally invasive procedures, a transhiatal esophagectomy was performed. Histopathological examination of the resected specimen was conducted, including routine hematoxylin and eosin (H&E) staining. Immunohistochemistry (IHC) was not performed owing to institutional limitations. Laboratory data included peripheral eosinophil counts, with emphasis on absolute eosinophil count. The literature review also included published cases of esophageal xanthogranulomas and eosinophil-associated esophageal lesions.Results: Histology revealed a well-defined intramural esophageal mass demonstrating extensive xanthogranulomatous inflammation, rich in lipid-laden macrophages, multinucleated giant cells, and eosinophils, confirmed qualitatively. No evidence of malignancy or infectious granulomatous disease was identified. The patient recovered uneventfully post-surgery, with symptomatic improvement. Moreover, literature review demonstrated that esophageal xanthogranulomas are rare, with few cases reported, and their pathogenesis remains speculative, particularly regarding eosinophil involvement and immunosuppression.Conclusion: This case revealed the importance of considering xanthogranulomatous inflammation in the differential diagnosis of esophageal intramural lesions in immunosuppressed patients with peripheral eosinophilia. Definitive diagnosis relies on histopathology, with surgical excision often necessary when less invasive methods are inconclusive. Further studies are warranted to elucidate the potential pathogenic links and the role of eosinophils in such lesions.

کلیدواژه ها:

Esophageal xanthogranuloma ، Eosinophilia ، Immunosuppression ، Granulomatous inflammation ، Benign esophageal lesion