Razavi Journal of Medicine

Razavi Journal of Medicine

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

Articles in Press

Document Type : Case Report/Series

Authors
Sepehr Sadrizadeh 1
Ali Sadrizadeh 2
Mohammad Hassan Jokar 3
Alireza Bary 4
Reza Sahabi 5
Alireza Sharifian-Attar 6
1 Department of Medicine, Faculty of Medicine, Mashhad University of Medical Sciences, Mashhad, Iran.
2 Cardiothoracic Surgery and Transplant Research Center, Imam Reza Hospital, Faculty of Medicine, Mashhad University of Medical Sciences, Mashhad, Iran.
3 Rheumatic Diseases Research Center, School of Medicine, Mashhad University of Medical Sciences, Mashhad, Iran.
4 Department of Hematology and Oncology, Ghaem Hospital, Mashhad University of Medical Sciences, Mashhad, Iran.
5 Department of Pathology, Razavi Hospital, Mashhad, Iran
6 Department of Anesthesia, Mashhad University of Medical Sciences, Mashhad, Iran
10.30483/rjm.2026.254775.1524
Abstract
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 52-year-old man with a 17-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.
Keywords
Esophageal xanthogranuloma
Eosinophilia
Immunosuppression
Granulomatous inflammation
Benign esophageal lesion

Acknowledgements: Not applicable.

Consent for Publication: Written informed consent was obtained from the patient for the publication of this case report and any accompanying images. 

Availability of Data and Materials: The data supporting the findings of this study are available from the corresponding author upon reasonable request. The minimal dataset necessary to interpret, replicate, and build upon the findings is accessible, subject to privacy considerations.

Competing Interests: The authors declare no conflicts of interest, financial or non-financial.

Funding: This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Ethics Approval and Consent to Participate: The study was conducted in accordance with the ethical principles of the Declaration of Helsinki and institutional policies governing publication of clinical case reports. Written informed consent was also obtained from the patient for the publication of the clinical information and accompanying images. Besides, all identifying information was removed to preserve patient confidentiality. 

Authors' Contributions: Sepehr Sadrizadeh conceptualized; Ali Sadrizadeh supervised and validated; Mohammad Hassan Jokar and Alireza Bary conducted the investigation and methodology; Reza Sahabi and Ali Reza Sharifian Attar drafted and edited the manuscript. All authors reviewed and approved the final version.

Open Access Policy: This is an open access article under the terms of the Creative Commons Attribution License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited. To view a copy of this licence, visit https://creativecommons.org/licenses/by/4.0/

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Razavi Journal of Medicine

Articles in Press, Accepted Manuscript
Available Online from 01 August 2026