Publication:
Clinical exome sequencing in neuromuscular diseases: an experience from Turkey

dc.contributor.departmentKUTTAM (Koç University Research Center for Translational Medicine)
dc.contributor.departmentKUH (Koç University Hospital)
dc.contributor.departmentSchool of Medicine
dc.contributor.kuauthorAvcı, Şahin
dc.contributor.kuauthorBörklü Yücel, Esra
dc.contributor.kuauthorEraslan, Serpil
dc.contributor.kuauthorKayserili, Hülya
dc.contributor.kuauthorOflazer, Piraye
dc.contributor.kuauthorVanlı-Yavuz, Ebru Nur
dc.contributor.kuauthorDemiriz, Çiğdem
dc.contributor.schoolcollegeinstituteKUH (KOÇ UNIVERSITY HOSPITAL)
dc.contributor.schoolcollegeinstituteResearch Center
dc.contributor.schoolcollegeinstituteSCHOOL OF MEDICINE
dc.date.accessioned2024-11-09T23:10:45Z
dc.date.issued2020
dc.description.abstractNeuromuscular diseases (NMDs) encompass a variety of ailments from muscular dystrophies to ataxias, in the course of which the functioning of the muscles is eventually either directly or indirectly impaired. The clinical diagnosis of a particular NMD is not always straightforward due to the clinical and genetic heterogeneity of the disorders under investigation. Traditional diagnostic tools such as electrophysiological tests and muscle biopsies are both invasive and painful methods, causing the patients to be reluctant. Next-generation sequencing, on the other hand, emerged as an alternative method for the diagnosis of NMDs, both with its minimally invasive nature and fast processing period. In this study, clinical exome sequencing (CES) was applied to a cohort of 70 probands in Turkey, 44 of whom received a final diagnosis, representing a diagnostic rate of 62.9%. Out of the 50 mutations identified to be causal, 26 were novel in the known 27 NMD genes. Two probands had complex/blended phenotypes. Molecular confirmation of clinical diagnosis of NMDs has a major prognostic impact and is crucial for the management and the possibility of alternative reproductive options. CES, which has been increasingly adopted to diagnose single-gene disorders, is also a powerful tool for revealing the etiopathogenesis in complex/blended phenotypes, as observed in two probands of the cohort.
dc.description.indexedbyWOS
dc.description.indexedbyScopus
dc.description.indexedbyPubMed
dc.description.issue8
dc.description.openaccessNO
dc.description.publisherscopeInternational
dc.description.sponsoredbyTubitakEuN/A
dc.description.volume41
dc.identifier.doi10.1007/s10072-020-04304-w
dc.identifier.eissn1590-3478
dc.identifier.issn1590-1874
dc.identifier.scopus2-s2.0-85081027177
dc.identifier.urihttps://doi.org/10.1007/s10072-020-04304-w
dc.identifier.urihttps://hdl.handle.net/20.500.14288/9532
dc.identifier.wos551151100027
dc.keywordsNext-generation sequencing (NGS)
dc.keywordsClinical exome sequencing (CES)
dc.keywordsNeuromuscular disease (NMD)
dc.keywordsDual diagnosis
dc.keywordsDysferlinopathy
dc.keywordsSarcoglycanopathy
dc.language.isoeng
dc.publisherSpringer-Verlag Italia Srl
dc.relation.ispartofNeurological Sciences
dc.subjectClinical neurology
dc.subjectNeurosciences
dc.titleClinical exome sequencing in neuromuscular diseases: an experience from Turkey
dc.typeJournal Article
dspace.entity.typePublication
local.contributor.kuauthorBörklü Yücel, Esra
local.contributor.kuauthorDemiriz, Çiğdem
local.contributor.kuauthorAvcı, Şahin
local.contributor.kuauthorVanlı-Yavuz, Ebru Nur
local.contributor.kuauthorEraslan, Serpil
local.contributor.kuauthorOflazer, Piraye
local.contributor.kuauthorKayserili, Hülya
local.publication.orgunit1SCHOOL OF MEDICINE
local.publication.orgunit1Research Center
local.publication.orgunit1KUH (KOÇ UNIVERSITY HOSPITAL)
local.publication.orgunit2KUTTAM (Koç University Research Center for Translational Medicine)
local.publication.orgunit2KUH (Koç University Hospital)
local.publication.orgunit2School of Medicine
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