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@ARTICLE{Hengel:140192,
author = {Hengel, H. and Keimer, R. and Deigendesch, W. and Rieß, A.
and Marzouqa, H. and Zaidan, J. and Bauer, P. and Schöls,
L.},
title = {{GPT}2 mutations cause developmental encephalopathy with
microcephaly and features of complicated hereditary spastic
paraplegia.},
journal = {Clinical genetics},
volume = {94},
number = {3-4},
issn = {0009-9163},
address = {Malden, Mass.},
publisher = {Wiley},
reportid = {DZNE-2020-06514},
pages = {356-361},
year = {2018},
abstract = {Various genetic defects can cause intellectual and
developmental disabilities (IDDs). Often IDD is a symptom of
a more complex neurodevelopmental or neurodegenerative
syndrome. Identifying syndromic patterns is substantive for
diagnostics and for understanding the pathomechanism of a
disease. Recessive glutamate pyruvate transaminase (GPT2)
mutations have recently been associated with IDD in 4
families. Here, we report a novel recessive GPT2 stop
mutation p.Gln24* causing a complex IDD phenotype in a
homozygous state in 5 patients from 2 consanguineous Arab
families. By compiling clinical information of these
individuals and previously described GPT2 patients a
recognizable neurodevelopmental and potentially
neurodegenerative phenotype can be assigned consisting of
intellectual disability, pyramidal tract affection with
spastic paraplegia, microcephaly and frequently epilepsy.
Because of the consistent presence of pyramidal tract
affection in GPT2 patients, we further suggest that GPT2
mutations should be considered in cases with complex
hereditary spastic paraplegia.},
keywords = {Adolescent / Adult / Brain Diseases: genetics / Child /
Consanguinity / Female / Humans / Male / Mutation / Pedigree
/ Spastic Paraplegia, Hereditary: genetics / Transaminases:
genetics / GPT2 protein, human (NLM Chemicals) /
Transaminases (NLM Chemicals)},
cin = {AG Gasser / AG Schöls},
ddc = {610},
cid = {I:(DE-2719)1210000 / I:(DE-2719)5000005},
pnm = {345 - Population Studies and Genetics (POF3-345)},
pid = {G:(DE-HGF)POF3-345},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:29882329},
doi = {10.1111/cge.13390},
url = {https://pub.dzne.de/record/140192},
}