Fatal gastrointestinal histoplasmosis 15 years after orthotopic liver transplantation

Abstract

We report a case of ileo-colonic Histoplasmosis without apparent respiratory involvement in a patient who had previously undergone an orthotopic liver transplant (OLT) for primary biliary cholangitis 15 years earlier. The recipient lived in the United Kingdom, a non-endemic region for Histoplasmosis. However, she had previously lived in rural southern Africa prior to her OLT. The patient presented with iron deficiency anaemia, diarrhoea, abdominal pain and progressive weight loss. She reported no previous foreign travel, however, it later became known that following her OLT she had been on holiday to rural southern Africa. On investigation, a mild granulomatous colitis primarily affecting the right colon was identified, that initially improved with mesalazine. Her symptoms worsened after 18 mo with progressive ulceration of her distal small bowel and right colon. Mycobacterial, Yersinia, cytomegalovirus and human immunodeficiency virus infections were excluded and the patient was treated with prednisolone for a working diagnosis of Crohn’s disease. Despite some early symptom improvement following steroids, there was subsequent deterioration with the patient developing gram-negative sepsis and multi-organ failure, leading to her death. Post-mortem examination revealed that her ileo-colonic inflammation was caused by Histoplasmosis.

Keywords: Histoplasmosis, Orthotopic liver transplant, Primary biliary cholangitis, Primary biliary cholangitis

Core tip: Histoplasmosis is an endemic fungal infection in many parts of the world; the majority of hosts remain asymptomatic. Clinical manifestations are most commonly pulmonary. We present an unusual case of Histoplasmosis occurring in a patient who was living in a non-endemic region, developed the disease after 15 years of immunosuppression following an orthotopic liver transplant, she presented with no pulmonary symptoms but rather luminal GI and systemic symptoms. This highlights the importance of considering Histoplasmosis within the differential of immunosuppressed patients with a past relevant travel history who present with diarrhea, weight loss, abdominal pain and granulomatous colitis.