A new complication of stem cell transplantation

Measles inclusion body encephalitis

Alexandra F. Freeman, David A. Jacobsohn, Stanford T. Shulman, William J. Bellini, Preeti Jaggi, Guillermo De Leon, Gesina F. Keating, Francine Kim, Lauren M. Pachman, Morris Kletzel, Reggie E. Duerst

Research output: Contribution to journalArticle

34 Citations (Scopus)

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.

Original languageEnglish (US)
JournalPediatrics
Volume114
Issue number5
DOIs
StatePublished - Nov 2004
Externally publishedYes

Fingerprint

Subacute Sclerosing Panencephalitis
Measles
Stem Cell Transplantation
Immunocompromised Host
Vaccination
Nucleoproteins
Biopsy
Stem Cells
Infection
Immunohistochemistry
Genotype
Chronic Granulomatous Disease
Intranuclear Inclusion Bodies
Nucleocapsid
Measles virus
Ribavirin
Brain
Graft vs Host Disease
Encephalitis
Neurologic Manifestations

Keywords

  • Encephalitis
  • Immunocompromised host
  • Measles

ASJC Scopus subject areas

  • Pediatrics, Perinatology, and Child Health

Cite this

Freeman, A. F., Jacobsohn, D. A., Shulman, S. T., Bellini, W. J., Jaggi, P., De Leon, G., ... Duerst, R. E. (2004). A new complication of stem cell transplantation: Measles inclusion body encephalitis. Pediatrics, 114(5). https://doi.org/10.1542/peds.2004-0949

A new complication of stem cell transplantation : Measles inclusion body encephalitis. / Freeman, Alexandra F.; Jacobsohn, David A.; Shulman, Stanford T.; Bellini, William J.; Jaggi, Preeti; De Leon, Guillermo; Keating, Gesina F.; Kim, Francine; Pachman, Lauren M.; Kletzel, Morris; Duerst, Reggie E.

In: Pediatrics, Vol. 114, No. 5, 11.2004.

Research output: Contribution to journalArticle

Freeman, AF, Jacobsohn, DA, Shulman, ST, Bellini, WJ, Jaggi, P, De Leon, G, Keating, GF, Kim, F, Pachman, LM, Kletzel, M & Duerst, RE 2004, 'A new complication of stem cell transplantation: Measles inclusion body encephalitis', Pediatrics, vol. 114, no. 5. https://doi.org/10.1542/peds.2004-0949
Freeman AF, Jacobsohn DA, Shulman ST, Bellini WJ, Jaggi P, De Leon G et al. A new complication of stem cell transplantation: Measles inclusion body encephalitis. Pediatrics. 2004 Nov;114(5). https://doi.org/10.1542/peds.2004-0949
Freeman, Alexandra F. ; Jacobsohn, David A. ; Shulman, Stanford T. ; Bellini, William J. ; Jaggi, Preeti ; De Leon, Guillermo ; Keating, Gesina F. ; Kim, Francine ; Pachman, Lauren M. ; Kletzel, Morris ; Duerst, Reggie E. / A new complication of stem cell transplantation : Measles inclusion body encephalitis. In: Pediatrics. 2004 ; Vol. 114, No. 5.
@article{600663a498d24ba0b735ddfe98df6e7e,
title = "A new complication of stem cell transplantation: Measles inclusion body encephalitis",
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.",
keywords = "Encephalitis, Immunocompromised host, Measles",
author = "Freeman, {Alexandra F.} and Jacobsohn, {David A.} and Shulman, {Stanford T.} and Bellini, {William J.} and Preeti Jaggi and {De Leon}, Guillermo and Keating, {Gesina F.} and Francine Kim and Pachman, {Lauren M.} and Morris Kletzel and Duerst, {Reggie E.}",
year = "2004",
month = "11",
doi = "10.1542/peds.2004-0949",
language = "English (US)",
volume = "114",
journal = "Pediatrics",
issn = "0031-4005",
publisher = "American Academy of Pediatrics",
number = "5",

}

TY - JOUR

T1 - A new complication of stem cell transplantation

T2 - Measles inclusion body encephalitis

AU - Freeman, Alexandra F.

AU - Jacobsohn, David A.

AU - Shulman, Stanford T.

AU - Bellini, William J.

AU - Jaggi, Preeti

AU - De Leon, Guillermo

AU - Keating, Gesina F.

AU - Kim, Francine

AU - Pachman, Lauren M.

AU - Kletzel, Morris

AU - Duerst, Reggie E.

PY - 2004/11

Y1 - 2004/11

N2 - 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.

AB - 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.

KW - Encephalitis

KW - Immunocompromised host

KW - Measles

UR - http://www.scopus.com/inward/record.url?scp=16644396341&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=16644396341&partnerID=8YFLogxK

U2 - 10.1542/peds.2004-0949

DO - 10.1542/peds.2004-0949

M3 - Article

VL - 114

JO - Pediatrics

JF - Pediatrics

SN - 0031-4005

IS - 5

ER -