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Case Study of a 16-Year-Old with Hypomyelination and Basal Ganglia Atrophy (H-ABC)

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A 16-year-old male with

progressive walking difficulty and dystonia

• Teaching NeuroImages

Neurology

• Resident & Fellow Section

© 2019 American Academy of Neurology Vinayagamani S, et al.

(2)

Vignette

© 2019 American Academy of Neurology

• A16-year-old-male patient, born of non-consanguineous parentage, had an uneventful birth history and mild global developmental delay.

• He had progressive gait disturbances since 2 years of age.

• Examination revealed generalized dystonia, scanning speech, pendular nystagmus, cerebellar ataxia, and spasticity.

Vinayagamani S, et al.

(3)

Imaging

© 2019 American Academy of Neurology Vinayagamani S, et al.

(4)

DIAGNOSIS

© 2019 American Academy of Neurology

• MRI brain showed features of hypomyelination with atrophy of the basal ganglia and cerebellum (H-ABC).

• Next generation exome sequencing showed a heterozygous mutation in exon 4 of TUBB4A (c.731G>A) confirming the diagnosis of H-ABC

• Conclusions

• The presence of generalised dystonia and atrophic/absent putamen are the two features that distinguish H-ABC from the other hypomyelinating

leukodystrophies like Pelizaeus-Merzbacher disease, Salla disease and Tay syndrome.1,2

Vinayagamani S, et al.

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