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Adalimumab/certolizumab-pegol/infliximab Neuromyelitis optica spectrum disorder with cheiro-oral syndrome and transverse myelitis: 3 case reports

In case series, three women aged 43–65 years were described, who developed demyelination characterised as neuromyelitis optica spectrum disorder with cheiro-oral syndrome or transverse myelitis during treatment with adalimumab, certolizumab pegol or infliximab [routes, dosages and durations of treatments to reactions not stated; not all indications stated]. Case 1: The 65-year-old woman developed paraethesia of tongues, lips, feet and hands, immediately after receiving first dose of certolizumab pegol [certolizumab]. Examination showed reduced sensation of the tongue and perioral region. An MRI of the brain showed signal abnormality centred in the mid brain at the level of interpeduncular notch. The demyelination characterised as cheiro-oral syndrome suggestive of neuromyelitis optica spectrum disorder. Treatment with certolizumab pegol was discontinued and two months later MRI showed resolution of the lesion along with improvement in her symptoms. Antibody testing was nonsignificant for Aquaporin-4 antibodies. Case 2: The 63-year-old woman developed lower limb sensory disturbance, after receiving first dose of infliximab for rheumatoid arthritis. Examination showed urinary retention, bilateral leg weakness and a sensory level to the upper abdomen. Her MRI showed an isolated T8/9 lesion and CSF was negative for oligoclonal bands. The demyelination characterised as transverse myelitis. Treatment with infliximab was discontinued and no further episodes of demyelination were observed. Case 3: The 43-year-old woman presented with subtle left leg weakness. She had been receiving adalimumab for 2 years. Her examination showed an extensor plantar response and hyperreflexia. Her MRI was significant for lesions at C2 and T9 and CSF was negative for oligoclonal bands. The demyelination characterised as transverse myelitis. Treatment with adalimumab was discontinued and no further episodes of demyelination were observed. Triplett JD, et al. TNF-alpha inhibitor therapy associated demyelination. Journal of Neurology, Neurosurgery and Psychiatry 88: e20-e21 (plus poster) abstr. 54, No. 5, May 803449342 2017. Available from: URL: http://doi.org/10.1136/jnnp-2017-316074.54 [abstract]

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Reactions 25 Jan 2020 No. 1788