Possible Interferon Response in a Child with Measles Encephalitis during Immunosuppression

Simpson, R; Eden, O. B.

doi:10.3109/00365548409070407

Cited by 23 publications

(6 citation statements)

References 15 publications

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“…Interferon-␣ has been used in 3 patients; 2 had no improvement in neurologic symptoms, and 1 improved temporarily but died soon after from underlying acute lymphoblastic leukemia. 20,21 One should maintain a high index of suspicion for MIBE in afebrile immunocompromised patients who present with focal status epilepticus. Typically, these patients have had recent measles infection or exposure; however, in our case, no history of either could be ascertained.…”

Section: Discussionmentioning

confidence: 99%

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

Freeman¹,

Jacobsohn²,

Shulman³

et al. 2004

Pediatrics

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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 transcriptasepolymerase 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. I n addition to acute encephalitis and subacute sclerosing panencephalitis in immunocompetent hosts, measles virus can cause measles inclusion body encephalitis (MIBE) in immunocompromised hosts. 1,2 MIBE presents typically with focal and intractable seizures within 1 year of initial measles infection. The mortality rate is high, and no effective treatment exists. 3 We report a patient who had chronic granulomatous disease (CGD) and developed fatal MIBE after nonmyeloablative cytoreduction and allogeneic stem cell transplantation (SCT). There was no history of recent or remote measles infection or exposure. CASE REPORTSThe patient was a 13-year-old Mexican-American boy who received a diagnosis of X-linked CGD at 2 years of age after serratia lymphadenitis. The patient was born in the United States and lived in Chicago but traveled frequently to urban Mexico, most recently 4 years before this illness. He received 1 documented measles-mumps-rubella vaccine at one year of age, and had no history of a measles-like illness or measles exposure at any time. A second measles-mumps-rubella vaccine was not documented. Despite prophylactic therapy for CGD with trimethoprim-sulfamethoxazole and thrice-weekly ␥-interferon, he had multiple staphylococcal liver abscesses that required extensive surgical drainage. Poor compliance with interferon ...

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Section: Discussionmentioning

confidence: 99%

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

Freeman¹,

Jacobsohn²,

Shulman³

et al. 2004

Pediatrics

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“…"-'3 In contrast, two similar studies using dual photon absorptiometry showed only small differences in the bone mineral content of cases and controls.5 14 One interpretation of these findings is that the Singh index reflects some aspect of bone morphology that is more closely related to bone strength than is the bone mineral content as measured by photon absorptiometry.…”

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confidence: 98%

Measles in children who have malignant disease.

Kernahan¹,

McQuillin²,

Craft³

1987

BMJ

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“…Simpson and Eden [55] reported the case of a 4-year-old boy with ALL in remission, who developed an encephalopathy following an attenuated measles vaccine. The boy's deterioration was halted when he was treated with interferon.…”

Section: Discussion Of Pathological Findingsmentioning

confidence: 99%