TY - JOUR
T1 - Congenital myasthenic syndrome in Israel
T2 - Genetic and clinical characterization
AU - Aharoni, Sharon
AU - Sadeh, Menachem
AU - Shapira, Yehuda
AU - Edvardson, Simon
AU - Daana, Muhannad
AU - Dor-Wollman, Talia
AU - Mimouni-Bloch, Aviva
AU - Halevy, Ayelet
AU - Cohen, Rony
AU - Sagie, Liora
AU - Argov, Zohar
AU - Rabie, Malcolm
AU - Spiegel, Ronen
AU - Chervinsky, Ilana
AU - Orenstein, Naama
AU - Engel, Andrew G.
AU - Nevo, Yoram
N1 - Publisher Copyright:
© 2016 Elsevier B.V.
PY - 2017/2/1
Y1 - 2017/2/1
N2 - The objective of the study was to evaluate the epidemiology of patients with congenital myasthenic syndrome (CMS) in Israel. Targeted mutation analysis was performed based on the clinical symptoms and electrophysiological findings for known CMS. Additional specific tests were performed in patients of Iranian and/or Iraqi Jewish origin. All medical records were reviewed and clinical data, genetic mutations and outcomes were recorded. Forty-five patients with genetic mutations in known CMS genes from 35 families were identified. Mutations in RAPSN were identified in 13 kinships in Israel. The most common mutation was c.-38A>G detected in 8 patients of Iranian and/or Iraqi Jewish origin. Four different recessive mutations in COLQ were identified in 11 kinships, 10 of which were of Muslim-Arab descent. Mutations in CHRNE were identified in 7 kinships. Less commonly detected mutations were in CHRND, CHAT, GFPT1 and DOK7. In conclusion, mutations in RAPSN and COLQ are the most common causes of CMS in our cohort. Specific mutations in COLQ, RAPSN, and CHRNE occur in specific ethnic populations and should be taken into account when the diagnosis of a CMS is suspected.
AB - The objective of the study was to evaluate the epidemiology of patients with congenital myasthenic syndrome (CMS) in Israel. Targeted mutation analysis was performed based on the clinical symptoms and electrophysiological findings for known CMS. Additional specific tests were performed in patients of Iranian and/or Iraqi Jewish origin. All medical records were reviewed and clinical data, genetic mutations and outcomes were recorded. Forty-five patients with genetic mutations in known CMS genes from 35 families were identified. Mutations in RAPSN were identified in 13 kinships in Israel. The most common mutation was c.-38A>G detected in 8 patients of Iranian and/or Iraqi Jewish origin. Four different recessive mutations in COLQ were identified in 11 kinships, 10 of which were of Muslim-Arab descent. Mutations in CHRNE were identified in 7 kinships. Less commonly detected mutations were in CHRND, CHAT, GFPT1 and DOK7. In conclusion, mutations in RAPSN and COLQ are the most common causes of CMS in our cohort. Specific mutations in COLQ, RAPSN, and CHRNE occur in specific ethnic populations and should be taken into account when the diagnosis of a CMS is suspected.
KW - CHRNE
KW - COLQ
KW - Congenital myasthenic syndrome (CMS)
KW - Genetic mutations
KW - RAPSN
UR - http://www.scopus.com/inward/record.url?scp=85009382832&partnerID=8YFLogxK
U2 - 10.1016/j.nmd.2016.11.014
DO - 10.1016/j.nmd.2016.11.014
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C2 - 28024842
AN - SCOPUS:85009382832
VL - 27
SP - 136
EP - 140
JO - Neuromuscular Disorders
JF - Neuromuscular Disorders
SN - 0960-8966
IS - 2
ER -