Hemophagocytic Lymphohistiocytosis Associated With Bartonella Peliosis Hepatis in an HIV-positive Kidney Transplant Patient

• Type of Material: Article
• Authors: Danielle Steed, Emory University; Jeffrey Collins, Emory University; Alton B Farris III, Emory University; Jeannette Guarner, Emory University; Dilek Yarar, Kidney Specialists of Kentucky; Rachel Jacqueline Friedman-Moraco, Emory University; Tristan Doane, Emory University; Stephanie Marie Pouch, Emory University; G Marshall Lyon III, Emory University; Michael Woodworth, Emory University
• Language: English
• Date: 2022-04-29
• Publisher: Elsevier
• Publication Version: Author Accepted Manuscript (After Peer Review)
• Copyright Statement: © 2022 Elsevier Ltd. All rights reserved.
• License: Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International
• Final Published Version (URL): https://doi.org/10.1016%2FS1473-3099(22)00276-6
• Title of Journal or Parent Work: The Lancet Infectious Diseases
• Volume: 22
• Issue: 10
• Start Page: e303
• End Page: e309
• Grant/Funding Information: We appreciate thoughtful support from Jane E. Koehler MA, MD in the Division of Infectious Diseases at the University of California, San Francisco during the clinical care of this patient. This work was supported in part by the National Institute of Allergy and Infectious Disease under Award Number K23AI144036 to MHW. The content is solely the responsibility of the authors and does not necessarily represent the official views of the National Institutes of Health. The authors would like to thank the participants, physicians, investigators, and staff of the HOPE in Action Prospective Observational Study of HIV+ Deceased Donor Transplant for HIV+ Recipients [NCT02602262]. This publication was supported in part by the National Institute of Allergy and Infectious Diseases under Award Number K23AI144036 to MHW, which had no role in writing of this manuscript or decision to submit for publication.
• Keywords: bacillary peliosis hepatis; hemophagocytic lymphohistiocytosis; Kidney transplantation; bartonella; HIV-positive
• Research Categories: Biology, Microbiology; Health Sciences, Epidemiology; Health Sciences, Medicine and Surgery

Abstract

Bacillary peliosis hepatis is a well-recognized manifestation of disseminated Bartonella infection that can occur in immunocompromised individuals. Hemophagocytic lymphohistiocytosis (HLH) is an immune-mediated condition with features that can overlap with a severe primary infection such as disseminated Bartonella infection. We report a case of bacillary peliosis hepatis and secondary HLH due to disseminated Bartonella infection in a kidney transplant recipient with well-controlled human immunodeficiency virus (HIV) infection. The patient had two weeks of fever and abdominal pain and was found to have hepatomegaly. He recalled exposure to a sick dog but had no recalled cat exposures. Laboratory evaluation was notable for pancytopenia and cholestatic injury. This patient met greater than five of eight clinical criteria for HLH. Pathology review of a bone marrow core biopsy identified hemophagocytosis. A transjugular liver biopsy was performed, and histopathology review identified peliosis hepatis. Warthin-Starry staining of the bone marrow showed pleiomorphic coccobacillary organisms. The Bartonella IgG titer was 1:512 and Bartonella-specific DNA targets were detected by peripheral blood PCR. Treatment with doxycycline, increased prednisone, and holding the mycophenolate component of his transplant immunosuppression regimen resulted in an excellent clinical response. Secondary HLH can be difficult to distinguish from severe systemic infection. A high index of suspicion can support the diagnosis of systemic Bartonella infection in those who present with HLH, especially in patients with hepatomegaly, immunosuppression, and germane animal exposures.