A New Complication of Stem Cell Transplantation: Measles Inclusion Body Encephalitis

Author:

Freeman Alexandra F.1,Jacobsohn David A.2,Shulman Stanford T.1,Bellini William J.3,Jaggi Preeti1,Leon Guillermo de4,Keating Gesina F.5,Kim Francine6,Pachman Lauren M.1,Kletzel Morris2,Duerst Reggie E.2

Affiliation:

1. Division of Infectious Disease, Department of Pediatrics

2. Division of Hematology/Oncology/Transplant, Department of Pediatrics

3. Centers for Disease Control and Prevention, Atlanta, Georgia

4. Department of Pathology

5. Division of Neurology

6. Division of Diagnostic Imaging

Abstract

Measles inclusion body encephalitis (MIBE) is a disease of the immunocompromised host and typically occurs within 1 year of acute measles infection or vaccination. We report a 13-year-old boy who had chronic granulomatous disease and presented 38 days after stem cell transplantation with afebrile focal seizures that progressed despite multiple anticonvulsants. After an extensive diagnostic evaluation, brain biopsy was performed, revealing numerous intranuclear inclusion bodies consistent with paramyxovirus nucleocapsids. Measles studies including reverse transcriptase-polymerase chain reaction and viral growth confirmed measles virus, genotype D3. Immunohistochemistry was positive for measles nucleoprotein. Despite intravenous ribavirin therapy, the patient died. MIBE has not been described in stem cell recipients but is a disease of immunocompromised hosts and typically occurs within 1 year of measles infection, exposure, or vaccination. Our case is unusual as neither the patient nor the stem cell donor had apparent recent measles exposure or vaccination, and neither had recent travel to measles-endemic regions. The patient had an erythematous rash several weeks before the neurologic symptoms; however, skin biopsy was consistent with graft-versus-host disease, and immunohistochemistry studies for measles nucleoprotein were negative. As measles genotype D3 has not been seen in areas where the child lived since his early childhood, the possibility of an unusually long latency period between initial measles infection and MIBE is raised. In addition, this case demonstrates the utility of brain biopsy in the diagnosis of encephalitis of unknown cause in the immunocompromised host.

Publisher

American Academy of Pediatrics (AAP)

Subject

Pediatrics, Perinatology, and Child Health

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1. Rubella virus chronic inflammatory disease and other unusual viral phenotypes in inborn errors of immunity;Immunological Reviews;2023-11-27

2. Measles Virus and the Central Nervous System: An Update;Seminars in Pediatric Neurology;2023-10

3. Sarampión;EMC - Pediatría;2023-09

4. Spotting a potential threat: Measles among pediatric solid organ transplantation recipients;Pediatric Transplantation;2023-03-15

5. Rubeola Virus;Principles and Practice of Pediatric Infectious Diseases;2023

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