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A KMT2B Frameshift Variant Causing Focal Dystonia Restricted to the Oromandibular Region After Long-Term Follow-up

Authors :
Alfand Marl F. Dy Closas
Katja Lohmann
Ai Huey Tan
Norlinah Mohamed Ibrahim
Jia Lun Lim
Yi Wen Tay
Kalai Arasu Muthusamy
Azlina Binti Ahmad-Annuar
Christine Klein
Shen-Yang Lim
Source :
Journal of Movement Disorders. 16:91-94
Publication Year :
2023
Publisher :
The Korean Movement Disorder Society, 2023.

Abstract

KMT2B-linked dystonia (DYT-KMT2B) is a childhood-onset dystonia syndrome typically beginning in the lower limbs and progressing caudocranially to affect the upper limbs with eventual prominent craniocervical involvement. Despite its recent recognition, it now appears to be one of the more common monogenic causes of dystonia syndromes. Here, we present an atypical case of DYT-KMT2B with oromandibular dystonia as the presenting feature, which remained restricted to this region three decades after symptom onset. This appears to be the first reported case of DYT-KMT2B from Southeast Asia and provides further supporting evidence for the pathogenic impact of the KMT2B c.6210_6213delTGAG variant.

Subjects

Subjects :
Neurology
Neurology (clinical)

Details

ISSN :
20934939 and 2005940X
Volume :
16
Database :
OpenAIRE
Journal :
Journal of Movement Disorders
Accession number :
edsair.doi...........b17d0501300a982ab0380799e7fb45c8