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Background. In the past the occurrence of neuromyelitis optica in Malaysia was thought to be uncommon and the frequency of anti‐aquaporin‐4 Ig G antibody was unknown. Objective. To evaluate the frequency of anti‐aquaporin‐4 Ig G antibody (Anti‐AQP4 antibody) amongst patients with neuromyelitis optica (NMO) and its spectrum disorders (NMOSD) and the differences between the seropositive and seronegative groups. Methods. Retrospectively, 96 patients with NMO/high risk syndromes for NMOSD (HRS‐NMOSD) were identified out of 266 patients with idiopathic inflammatory demyelinating disease from a single center hospital based registry. Anti‐AQP4 seropositivity was found in 38/48 (79.2%) with NMO, 12/21 (57.1%) with brain involvement at high risk for NMOSD, 12/15 (80%) with transverse myelitis (i.e., 11/15 with relapsing transverse myelitis and one with monophasic transverse myelitis), and 3/7 (42 …