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Dear Editor, Neuromyelitis optica spectrum disorder (NMOSD) presents with characteristic patterns of hemispheric white-matter lesions in magnetic resonance imaging (MRI), including tumefactive lesions, long spindle-like or radial-shape lesions following white-matter tracts, and asymmetric extensive and confluent hemispheric lesions.1 However, since bilateral symmetrical extensive white-matter lesions mimicking leukodystrophy have been previously reported in only three cases,2-4 its presence is not widely recognized. Here we report on a patient with anti-aquaporin-4 (anti-AQP4)-positive NMOSD who exhibited leukodystrophylike white-matter involvement. A 67-year-old female was admitted with a 4-day history of visual loss and ocular pain in the left eye. Nine years previously she had been admitted for right optic neuritis, which was her first clinical event. At that time her visual acuity on the right side had deteriorated to hand movements, whereas it was 20/20 on the left. Brain MRI revealed only subtle hyperintensities in the left temporal deep white matter, pons, and middle cerebellar peduncles (Fig. 1A– D). The presence of NMO-IgG and anti-AQP4 antibodies was not checked at that time. Her right-side visual function remained unchanged thereafter. Following the first attack, she remained stable and free of additional attacks related to demyelinating diseases of the central nervous system until the present attack, and had not been taking any medication to prevent such attacks. At the present admission she had visual acuity of counting fingers close to the face in the left eye, while that in the right eye was similar to the previous visual acuity. There was no limb weakness, sensory disturbance, or ataxia. Her score on the Mini Mental State Examination was 28. Brain MRI performed at this time revealed bilateral symmetrical extensive hyperintensities in the subcortical and periventricular white matter, external and internal capsules, temporal pole white matter, pons, middle cerebellar peduncles, and deep cerebellar white matter (Fig. 1E–H). She was a nonsmoker and nondrinker, had no history of migraine, and had no known diabetes or other medical conditions other than hypertension. There was no family history of inherited disease. A thorough clinical history-taking and medication review revealed no clear evidence of exposure to any substances including antineoplastic agents, immunosuppressive drugs, antimicrobial agents, abused drugs, or environmental toxins, with the exception of antihypertensive medication (telmisartan/amlodipine 40/5 mg once daily). Laboratory tests searching for inherited or immune-mediated conditions such as leukodystrophies (arylsulfatase A and very-long-chain fatty acids), mitochondrial disorders (lactate and pyruvate), and Hashimoto’s encephalopathy (thyroid hormone and antithyroid antibodies) all produced unremarkable findings. A cerebrospinal fluid examination showed normal cell count and protein level. Anti-AQP4 antibody was strongly positive (intensity, Jun Seok Lee Moon Kyung Choi Jae-Joon Lee Hung Youl Seok

A Little-Known Brain Imaging Feature in Neuromyelitis Optica Spectrum Disorder: A Leukodystrophy-Like Pattern