Clinical Case Summary

A 46-year-old male with stenosing colonic Crohn’s disease (CD) and perianal fistulas, treated with Infliximab and Azathioprine, achieved clinical and endoscopic remission (CDAI 35, fecal calprotectin 73 µg/g, and SES-CD 1) after 36 months of therapy. Following three years of immunosuppression, he developed dysphagia, weight loss, fever and night sweats – no respiratory symptoms were present. The patient was hospitalized for further evaluation and nutritional support. Infectious screening ruled out bacterial infections, CMV, EBV, HIV, HBV, HCV, tuberculosis, toxoplasmosis, and syphilis. Upper gastrointestinal endoscopy revealed active, ill-defined ulcers in the larynx and pharynx [1] ; two well-defined, clean-based ulcers with non-erythematous edges in the distal esophagus; and elevated, reddened, non-friable post-bulbar lesions. Pharyngeal biopsies collected by the otolaryngology team demonstrated a marked inflammatory response with loose granulomas. Fungal testing revealed numerous fungi consistent with Histoplasma capsulatum (Figure 3B). CT scans indicated adrenal thickening and enlarged para-aortic lymph nodes, supporting the diagnosis of disseminated histoplasmosis. The patient was treated with liposomal amphotericin B, followed by oral itraconazole, resulting in clinical improvement. Given the remission of CD and the confirmed disseminated granulomatous infection, immunosuppressive therapy was discontinued. This case highlights the risk of opportunistic infections in patients with inflammatory bowel disease (IBD) undergoing long-term immunosuppression, particularly when in anti-TNF combination therapy. Gastrointestinal histoplasmosis, especially in the absence of pulmonary symptoms, is a rare infection that is often misdiagnosed due to symptom overlap with IBD. Clinicians must maintain a high index of suspicion to ensure early diagnosis and appropriate management, as delayed treatment is potentially life-threatening.

References

Castro HM, Fullana MP, Canale HL. Disseminated histoplasmosis in immunosuppressed patient. Med Clin (Barc). 2021 Jan 8;156(1):48. English, Spanish. doi: 10.1016/j.medcli.2019.10.002. Epub 2019 Nov 26. PMID: 31784112.