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DOI 10.3390/genes14101843

Медицинская библиотека · Genes · 2023

From Feeding Challenges to Oral-Motor Dyspraxia: A Comprehensive Description of 10 New Cases with CTNNB1 Syndrome.

Onesimo Roberta, Sforza Elisabetta, Trevisan Valentina, Leoni Chiara, Giorgio Valentina, Rigante Donato, Kuczynska Eliza Maria, Proli Francesco, Agazzi Cristiana, Limongelli Domenico, Digilio Maria Cistina, Dentici Maria Lisa, Macchiaiolo Maria, Novelli Antonio, Bartuli Andrea, Sinibaldi Lorenzo, Tartaglia Marco, Zampino Giuseppe

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Аннотация

CTNNB1 syndrome is an autosomal-dominant neurodevelopmental disorder featuring developmental delay; intellectual disability; behavioral disturbances; movement disorders; visual defects; and subtle facial features caused by de novo loss-of-function variants in the CTNNB1 gene. Due to paucity of data, this study intends to describe feeding issues and oral-motor dyspraxia in an unselected cohort of 10 patients with a confirmed molecular diagnosis. Pathogenic variants along with key information regarding oral-motor features were collected. Sialorrhea was quantified using the Drooling Quotient 5. Feeding abilities were screened using the Italian version of the Montreal Children's Hospital Feeding Scale (I-MCH-FS). Mild-to-severe coordination difficulties in single or in a sequence of movements involving the endo-oral and peri-oral muscles were noticed across the entire cohort. Mild-to-profuse drooling was a commonly complained-about issue by 30% of parents. The mean total I-MCH-FS t-score equivalent was 43.1 ± 7.5. These findings contribute to the understanding of the CTNNB1 syndrome highlighting the oral motor phenotype, and correlating specific gene variants with clinical characteristics.

Для цитирования: Onesimo Roberta, Sforza Elisabetta, Trevisan Valentina, Leoni Chiara, Giorgio Valentina, Rigante Donato, Kuczynska Eliza Maria, Proli Francesco, Agazzi Cristiana, Limongelli Domenico, Digilio Maria Cistina, Dentici Maria Lisa, Macchiaiolo Maria, Novelli Antonio, Bartuli Andrea, Sinibaldi Lorenzo, Tartaglia Marco, Zampino Giuseppe. From Feeding Challenges to Oral-Motor Dyspraxia: A Comprehensive Description of 10 New Cases with CTNNB1 Syndrome.. Genes. 2023. DOI: 10.3390/genes14101843.

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