Fulminant central demyelinating lesions associated with infliximab treatment in a child with Crohn's disease: A case report

Infliximab is a tumor necrosis factor alpha (TNFα) inhibitor used for treating rheumatic disorders, inflammatory bowel disease, and other conditions. Despite the recent increase in reports of central demyelinating lesions associated with the use of TNFα inhibitors, most cases have been observed in adults with gradual onset and only mild neurological sequelae. We report a pediatric case of fulminant demyelinating lesions associated with infliximab administration. Our patient was an 8-year-old male diagnosed with ileocolonic Crohn's disease. Around 6 h after his second infliximab administration, he developed headache, eye pain, nausea, and loss of consciousness. He was subsequently intubated due to respiratory depression. Brain magnetic resonance imaging revealed a prominent mass effect in the pons with diffusion restriction extending from the brain stem to the cervical spinal cord, as well as scattered lesions in the deep gray matter and cerebral hemispheres with hemorrhage. The lesion distribution was similar to that of acute disseminated encephalomyelitis (ADEM). Furthermore, the rapid progression of symptoms and intracerebral bleeding were consistent with those observed in acute hemorrhagic leukoencephalitis, a severe variant of ADEM. Despite performing plasma exchange and steroid pulse therapy, the patient remained unconscious with quadriplegia and severe brainstem dysfunction and was maintained on tracheostomy-based ventilation. Although extremely rare, central demyelinating lesions associated with TNFα inhibitors can develop among pediatric cases. Clinicians should therefore be aware of the possibility of a fulminant course in children.