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DOI 10.1007/s10048-025-00822-x

Медицинская библиотека · Neurogenetics · 2025

A novel FBXW11 variant in a patient with neurodevelopmental, jaw, eye, and digital syndrome.

Maznina Anna, Molodtsova-Zolotukhina Daria, Andreeva Nina, Esibov Anton, Bostanova Fatima M, Sharkov Artem, Doroshchuk Natalya A, Sagaydak Olesya V, Groznova Olga S, Woroncow Mary, Bogdanov Viktor P, Volchkov Pavel Y

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Аннотация

Neurodevelopmental, jaw, eye, and digital syndrome (NEDJED) is a rare autosomal dominant condition that has demonstrated diverse phenotypes. This is the second case report published on this condition, covering the disease history of an 8 year old patient with a severe manifestation of the disease. The patient was born with hydrocephalus, and demonstrated major developmental delay as he aged. Whole-genome sequencing of the patient and his parents was conducted, detecting a de novo variant, NM_001378974.1:c.1220 A > T [p.Lys407Ile], located in the conserved WD4 region of the WD40 domain of FBXW11, which is consistent with all previously reported patients. The phenotype of the patient is presented with a focus on MRI and EEG features, including images and detailed description for both. While the patient's phenotype is overall consistent with previous findings, there are a number of major factors we believe are caused by the FBXW11 variant that have not been previously described, such as the patient's complete inability to walk.

Для цитирования: Maznina Anna, Molodtsova-Zolotukhina Daria, Andreeva Nina, Esibov Anton, Bostanova Fatima M, Sharkov Artem, Doroshchuk Natalya A, Sagaydak Olesya V, Groznova Olga S, Woroncow Mary, Bogdanov Viktor P, Volchkov Pavel Y. A novel FBXW11 variant in a patient with neurodevelopmental, jaw, eye, and digital syndrome.. Neurogenetics. 2025. DOI: 10.1007/s10048-025-00822-x.

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