Gastrointestinal histoplasmosis complicating pediatric Crohn disease: A case report and review of literature

Abstract

BACKGROUND

Infection with Histoplasma capsulatum (H. capsulatum) can lead to disseminated disease involving the gastrointestinal tract presenting as diffuse abdominal pain and diarrhea which may mimic inflammatory bowel disease (IBD).

CASE SUMMARY

We report a case of 12-year-old boy with presumptive diagnosis of Crohn disease (CD) that presented with several months of abdominal pain, weight loss and bloody diarrhea. Colonoscopy showed patchy moderate inflammation characterized by erythema and numerous pseudopolyps involving the terminal ileum, cecum, and ascending colon. Histologic sections from the colon biopsy revealed diffuse cellular infiltrate within the lamina propria with scattered histiocytic aggregates, and occasional non-necrotizing granulomas. Grocott-Gomori’s Methenamine Silver staining confirmed the presence of numerous yeast forms suggestive of Histoplasma spp., further confirmed with positive urine Histoplasma antigen (6.58 ng/mL, range 0.2-20 ng/mL) and serum immunoglobulin G antibodies to Histoplasma (35.9 EU, range 10.0-80.0 EU). Intravenous amphotericin was administered then transitioned to oral itraconazole. Follow-up computed tomography imaging showed a left lower lung nodule and mesenteric lymphadenopathy consistent with disseminated histoplasmosis infection.

CONCLUSION

Gastrointestinal involvement with H. capsulatum with no accompanying respiratory symptoms is exceedingly rare and recognition is often delayed due to the overlapping clinical manifestations of IBD. This case illustrates the importance of excluding infectious etiologies in patients with “biopsy-proven” CD prior to initiating immunosuppressive therapies. Communication between clinicians and pathologists is crucial as blood cultures and antigen testing are key studies that should be performed in all suspected cases of histoplasmosis to avoid misdiagnosis and inappropriate treatment.

Keywords: Crohn disease; Disseminated histoplasmosis; Endoscopy; Colon; Inflammatory bowel disease; Immunosuppression; Case report

Core Tip: Impaired cell-mediated immunity is known to increase the risk for disseminated histoplasmosis and has been described in the setting of Crohn disease (CD) treated with immunosuppressant agents. Endoscopically, the appearance of histoplasmosis varies and includes features of inflammatory mucosal changes. Increasing awareness of this condition is critical to avoid misdiagnosis and inappropriate treatment, particularly in the setting of underlying CD. While no specific recommendations are available, immunosuppressive therapy may be safely initiated in some cases when there appears to be effective response to antifungal therapy and the patient can be monitored closely.