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Biallelic variants in ZNF142 lead to a syndromic neurodevelopmental disorder

Authors :
Christensen, Maria B.
Levy, Amanda M.
Mohammadi, Nazanin A.
Niceta, Marcello
Kaiyrzhanov, Rauan
Dentici, Maria Lisa
Al Alam, Chadi
Alesi, Viola
Benoit, Valérie
Bhatia, Kailash P.
Bierhals, Tatjana
Boßelmann, Christian M.
Buratti, Julien
Callewaert, Bert
Ceulemans, Berten
Charles, Perrine
De Wachter, Matthias
Dehghani, Mohammadreza
D'haenens, Erika
Doco-Fenzy, Martine
Geßner, Michaela
Gobert, Cyrielle
Guliyeva, Ulviyya
Haack, Tobias B.
Hammer, Trine B.
Heinrich, Tilman
Hempel, Maja
Herget, Theresia
Hoffmann, Ute
Horvath, Judit
Houlden, Henry
Keren, Boris
Kresge, Christina
Kumps, Candy
Lederer, Damien
Lermine, Alban
Magrinelli, Francesca
Maroofian, Reza
Vahidi Mehrjardi, Mohammad Yahya
Moudi, Mahdiyeh
Müller, Amelie J.
Oostra, Anna J.
Pletcher, Beth A.
Ros-Pardo, David
Samarasekera, Shanika
Tartaglia, Marco
Van Schil, Kristof
Vogt, Julie
Wassmer, Evangeline
Winkelmann, Juliane
Zaki, Maha S.
Zech, Michael
Lerche, Holger
Radio, Francesca Clementina
Gomez-Puertas, Paulino
Møller, Rikke S.
Tümer, Zeynep
Christensen, Maria B.
Levy, Amanda M.
Mohammadi, Nazanin A.
Niceta, Marcello
Kaiyrzhanov, Rauan
Dentici, Maria Lisa
Al Alam, Chadi
Alesi, Viola
Benoit, Valérie
Bhatia, Kailash P.
Bierhals, Tatjana
Boßelmann, Christian M.
Buratti, Julien
Callewaert, Bert
Ceulemans, Berten
Charles, Perrine
De Wachter, Matthias
Dehghani, Mohammadreza
D'haenens, Erika
Doco-Fenzy, Martine
Geßner, Michaela
Gobert, Cyrielle
Guliyeva, Ulviyya
Haack, Tobias B.
Hammer, Trine B.
Heinrich, Tilman
Hempel, Maja
Herget, Theresia
Hoffmann, Ute
Horvath, Judit
Houlden, Henry
Keren, Boris
Kresge, Christina
Kumps, Candy
Lederer, Damien
Lermine, Alban
Magrinelli, Francesca
Maroofian, Reza
Vahidi Mehrjardi, Mohammad Yahya
Moudi, Mahdiyeh
Müller, Amelie J.
Oostra, Anna J.
Pletcher, Beth A.
Ros-Pardo, David
Samarasekera, Shanika
Tartaglia, Marco
Van Schil, Kristof
Vogt, Julie
Wassmer, Evangeline
Winkelmann, Juliane
Zaki, Maha S.
Zech, Michael
Lerche, Holger
Radio, Francesca Clementina
Gomez-Puertas, Paulino
Møller, Rikke S.
Tümer, Zeynep
Source :
Christensen , M B , Levy , A M , Mohammadi , N A , Niceta , M , Kaiyrzhanov , R , Dentici , M L , Al Alam , C , Alesi , V , Benoit , V , Bhatia , K P , Bierhals , T , Boßelmann , C M , Buratti , J , Callewaert , B , Ceulemans , B , Charles , P , De Wachter , M , Dehghani , M , D'haenens , E , Doco-Fenzy , M , Geßner , M , Gobert , C , Guliyeva , U , Haack , T B , Hammer , T B , Heinrich , T , Hempel , M , Herget , T , Hoffmann , U , Horvath , J , Houlden , H , Keren , B , Kresge , C , Kumps , C , Lederer , D , Lermine , A , Magrinelli , F , Maroofian , R , Vahidi Mehrjardi , M Y , Moudi , M , Müller , A J , Oostra , A J , Pletcher , B A , Ros-Pardo , D , Samarasekera , S , Tartaglia , M , Van Schil , K , Vogt , J , Wassmer , E , Winkelmann , J , Zaki , M S , Zech , M , Lerche , H , Radio , F C , Gomez-Puertas , P , Møller , R S & Tümer , Z 2022 , ' Biallelic variants in ZNF142 lead to a syndromic neurodevelopmental disorder ' , Clinical Genetics , vol. 102 , no. 2 , pp. 98-109 .
Publication Year :
2022

Abstract

Biallelic variants of the gene encoding for the zinc-finger protein 142 (ZNF142) have recently been associated with intellectual disability (ID), speech impairment, seizures, and movement disorders in nine individuals from five families. In this study, we obtained phenotype and genotype information of 26 further individuals from 16 families. Among the 27 different ZNF142 variants identified in the total of 35 individuals only four were missense. Missense variants may give a milder phenotype by changing the local structure of ZF motifs as suggested by protein modeling; but this correlation should be validated in larger cohorts and pathogenicity of the missense variants should be investigated with functional studies. Clinical features of the 35 individuals suggest that biallelic ZNF142 variants lead to a syndromic neurodevelopmental disorder with mild to moderate ID, varying degrees of delay in language and gross motor development, early onset seizures, hypotonia, behavioral features, movement disorders, and facial dysmorphism. The differences in symptom frequencies observed in the unpublished individuals compared to those of published, and recognition of previously underemphasized facial features are likely to be due to the small sizes of the previous cohorts, which underlines the importance of larger cohorts for the phenotype descriptions of rare genetic disorders.

Details

Database :
OAIster
Journal :
Christensen , M B , Levy , A M , Mohammadi , N A , Niceta , M , Kaiyrzhanov , R , Dentici , M L , Al Alam , C , Alesi , V , Benoit , V , Bhatia , K P , Bierhals , T , Boßelmann , C M , Buratti , J , Callewaert , B , Ceulemans , B , Charles , P , De Wachter , M , Dehghani , M , D'haenens , E , Doco-Fenzy , M , Geßner , M , Gobert , C , Guliyeva , U , Haack , T B , Hammer , T B , Heinrich , T , Hempel , M , Herget , T , Hoffmann , U , Horvath , J , Houlden , H , Keren , B , Kresge , C , Kumps , C , Lederer , D , Lermine , A , Magrinelli , F , Maroofian , R , Vahidi Mehrjardi , M Y , Moudi , M , Müller , A J , Oostra , A J , Pletcher , B A , Ros-Pardo , D , Samarasekera , S , Tartaglia , M , Van Schil , K , Vogt , J , Wassmer , E , Winkelmann , J , Zaki , M S , Zech , M , Lerche , H , Radio , F C , Gomez-Puertas , P , Møller , R S & Tümer , Z 2022 , ' Biallelic variants in ZNF142 lead to a syndromic neurodevelopmental disorder ' , Clinical Genetics , vol. 102 , no. 2 , pp. 98-109 .
Notes :
application/pdf, English
Publication Type :
Electronic Resource
Accession number :
edsoai.on1349067773
Document Type :
Electronic Resource