Case Report
Copyright ©The Author(s) 2017. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Gastroenterol. Nov 21, 2017; 23(43): 7807-7812
Published online Nov 21, 2017. doi: 10.3748/wjg.v23.i43.7807
Fatal gastrointestinal histoplasmosis 15 years after orthotopic liver transplantation
Nikita Agrawal, David EJ Jones, Jessica K Dyson, Tim Hoare, Sharon A Melmore, Stephanie Needham, Nick P Thompson
Nikita Agrawal, Nick P Thompson, Department of Gastroenterology, Newcastle Hospitals NHS Foundation Trust, Newcastle Upon Tyne NE7 7DN, United Kingdom
David EJ Jones, Jessica K Dyson, Faculty of Medical Sciences, Newcastle University, Newcastle Upon Tyne NE7 7DN, United Kingdom
Tim Hoare, Department of Radiology, Newcastle Hospitals NHS Foundation Trust, Newcastle Upon Tyne NE7 7DN, United Kingdom
Sharon A Melmore, Stephanie Needham, Department of Histopathology, Newcastle Hospitals NHS Foundation Trust, Newcastle Upon Tyne NE7 7DN, United Kingdom
Author contributions: All authors contributed to writing this case report and all authors apart from Agrawal N were involved in the care of this patient.
Informed consent statement: Consent for publication granted by next of kin.
Conflict-of-interest statement: No conflict of interests are declared by any authors.
Open-Access: This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See:
Correspondence to: Nick P Thompson, MD, Department of Gastroenterology, Newcastle Hospitals NHS Foundation Trust, High Heaton, Newcastle Upon Tyne NE7 7DN, United Kingdom.
Telephone: +44-191-2448584 Fax: +44-191-2231249
Received: July 19, 2017
Peer-review started: July 20, 2017
First decision: August 10, 2017
Revised: September 28, 2017
Accepted: October 17, 2017
Article in press: October 17, 2017
Published online: November 21, 2017

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.