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@ARTICLE{Hengel:153290,
author = {Hengel, Holger and Buchert, Rebecca and Sturm, Marc and
Haack, Tobias B and Schelling, Yvonne and Mahajnah, Muhammad
and Sharkia, Rajech and Azem, Abdussalam and Balousha,
Ghassan and Ghanem, Zaid and Falana, Mohammed and Balousha,
Osama and Ayesh, Suhail and Keimer, Reinhard and
Deigendesch, Werner and Zaidan, Jimmy and Marzouqa, Hiyam
and Bauer, Peter and Schöls, Ludger},
title = {{F}irst-line exome sequencing in {P}alestinian and
{I}sraeli {A}rabs with neurological disorders is efficient
and facilitates disease gene discovery.},
journal = {European journal of human genetics},
volume = {28},
number = {8},
issn = {1476-5438},
address = {Basingstoke},
publisher = {Stockton Press},
reportid = {DZNE-2020-01287},
pages = {1034 - 1043},
year = {2020},
abstract = {A high rate of consanguinity leads to a high prevalence of
autosomal recessive disorders in inbred populations. One
example of inbred populations is the Arab communities in
Israel and the Palestinian Authority. In the Palestinian
Authority in particular, due to limited access to
specialized medical care, most patients do not receive a
genetic diagnosis and can therefore neither receive genetic
counseling nor possibly specific treatment. We used
whole-exome sequencing as a first-line diagnostic tool in 83
Palestinian and Israeli Arab families with suspected
neurogenetic disorders and were able to establish a probable
genetic diagnosis in $51\%$ of the families (42 families).
Pathogenic, likely pathogenic or highly suggestive candidate
variants were found in the following genes extending and
refining the mutational and phenotypic spectrum of these
rare disorders: ACO2, ADAT3, ALS2, AMPD2, APTX, B4GALNT1,
CAPN1, CLCN1, CNTNAP1, DNAJC6, GAMT, GPT2, KCNQ2, KIF11,
LCA5, MCOLN1, MECP2, MFN2, MTMR2, NT5C2, NTRK1, PEX1,
POLR3A, PRICKLE1, PRKN, PRX, SCAPER, SEPSECS, SGCG,
SLC25A15, SPG11, SYNJ1, TMCO1, and TSEN54. Further, this
cohort has proven to be ideal for prioritization of new
disease genes. Two separately published candidate genes
(WWOX and PAX7) were identified in this study. Analyzing the
runs of homozygosity (ROHs) derived from the Exome
sequencing data as a marker for the rate of inbreeding,
revealed significantly longer ROHs in the included families
compared with a German control cohort. The total length of
ROHs correlated with the detection rate of recessive
disease-causing variants. Identification of the
disease-causing gene led to new therapeutic options in four
families.},
keywords = {Arabs: genetics / Female / Gene Frequency / Genetic Loci /
Genetic Predisposition to Disease / Humans / Male / Nervous
System Diseases: genetics / Pedigree / Exome Sequencing:
standards / Exome Sequencing: statistics $\&$ numerical
data},
cin = {AG Gasser 1},
ddc = {610},
cid = {I:(DE-2719)1210000},
pnm = {345 - Population Studies and Genetics (POF3-345)},
pid = {G:(DE-HGF)POF3-345},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:32214227},
pmc = {pmc:PMC7382450},
doi = {10.1038/s41431-020-0609-9},
url = {https://pub.dzne.de/record/153290},
}