| Literature DB >> 33141444 |
Paulo Victor Sgobbi Souza1, Bruno Mattos Lombardi Badia1, Igor Braga Farias1, Wladimir Bocca Vieira de Rezende Pinto1, Acary Souza Bulle Oliveira1, Hasan Orhan Akman2, Salvatore DiMauro2.
Abstract
Adult polyglucosan body disease (APBD) represents a complex autosomal recessive inherited neurometabolic disorder due to homozygous or compound heterozygous pathogenic variants in GBE1 gene, resulting in deficiency of glycogen-branching enzyme and secondary storage of glycogen in the form of polyglucosan bodies, involving the skeletal muscle, diaphragm, peripheral nerve (including autonomic fibers), brain white matter, spinal cord, nerve roots, cerebellum, brainstem and to a lesser extent heart, lung, kidney, and liver cells. The diversity of new clinical presentations regarding neuromuscular involvement is astonishing and transformed APBD in a key differential diagnosis of completely different clinical conditions, including axonal and demyelinating sensorimotor polyneuropathy, progressive spastic paraparesis, motor neuronopathy presentations, autonomic disturbances, leukodystrophies or even pure myopathic involvement with limb-girdle pattern of weakness. This review article aims to summarize the main clinical, biochemical, genetic, and diagnostic aspects regarding APBD with special focus on neuromuscular presentations.Entities:
Keywords: GBE1 gene; glycogen; glycogen storage disease; motor neuron disease; neurogenetics; polyglucosan body disease
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Year: 2020 PMID: 33141444 DOI: 10.1002/jimd.12325
Source DB: PubMed Journal: J Inherit Metab Dis ISSN: 0141-8955 Impact factor: 4.982