Cecal appendicitis as a rare manifestation of paracoccidioidomycosis: A case report and systematic review of the literature

盲肠阑尾炎是副球孢子菌病的一种罕见表现:病例报告及文献系统综述

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Abstract

BACKGROUND: Paracoccidioidomycosis (PCM) is a systemic mycosis endemic to Latin America, with the acute/subacute form predominantly affecting children and young adults. Cases of cecal appendicitis caused by Paracoccidioides spp. have rarely been reported. This study aimed to describe the clinical manifestations and evolution of a case of cecal appendicitis due to PCM and to conduct a systematic literature review. CASE PRESENTATION: We present the case of a 20-year-old male with generalized lymphadenopathy who was diagnosed with PCM and treated with oral trimethoprim-sulfamethoxazole. After the initial improvement, the patient returned with clinical deterioration. The treatment was changed to liposomal amphotericin B. Six days later; the patient developed an acute abdomen and underwent exploratory laparotomy with appendectomy. Histopathological examination confirmed acute granulomatous appendicitis due to PCM, and the patient showed postoperative clinical improvement. A systematic review were conducted using Embase, Web of Science, Lilacs, Medline, LIEPCS, PubMed, SciELO, and Gray Literature databases. Of the ten identified articles included in the systematic review, most case reports with a low risk of bias were found in South American countries. Seven patients were confirmed appendicitis due to PCM through biopsy, whereas one had confirmed PCM at another site. Two patients were initially misdiagnosed with Crohn's. Most studies have reported favorable outcomes. CONCLUSION: Appendicitis caused by PCM is rare, even in endemic countries. It has a benign course when properly treated with both clinical and surgical management. This should be considered in the differential diagnosis of acute abdomen with lymphadenopathy in endemic regions.

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