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KBG syndrome is a rare autosomal dominant disorder caused by ANKRD11 variants, characterized by distinctive craniofacial features, short stature, skeletal anomalies, and neurodevelopmental impairment. Regional studies, particularly from underrepresented populations, are essential to broaden the clinical and molecular spectrum of the syndrome. We retrospectively analyzed the clinical, molecular, and anthropometric data of 23 Turkish individuals with molecularly confirmed KBG syndrome. Molecular analyses included whole exome sequencing, clinical exome sequencing, and targeted gene panels. Variants were classified according to ACMG guidelines, and phenotypic features were systematically assessed using standardized criteria. Eighteen distinct ANKRD11 pathogenic or likely pathogenic variants were identified, including eight novel variants, predominantly located in exon 9. Short stature (height SDS < -2) was documented in 30.4% of patients. Intellectual disability was observed in 82.6%, and behavioral anomalies in 52.2% of the cohort. Craniofacial dysmorphism, including long philtrum, triangular facial shape, and macrodontia, was prominent. No genotype-phenotype correlation could be established. This study expands the mutational landscape of ANKRD11 and underscores the clinical variability of KBG syndrome in a Turkish cohort. Our findings emphasize the importance of integrating molecular and detailed clinical evaluations for early diagnosis, especially in diverse populations with limited prior representation.
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http://dx.doi.org/10.1002/ajmg.a.64128 | DOI Listing |
Genes (Basel)
July 2025
Department of Human Pathology of Adulthood and Childhood, University of Messina, Via Consolare Valeria1, 98125 Messina, Italy.
Background: SET domain-containing 5 (SETD5) is a member of the protein lysine-methyltransferase family. gene mutations cause disorders of the epigenetic machinery which determinate phenotypic overlap characterized by several abnormalities. gene variants have been described in patients with KBG and Cornelia de Lange (CdL) syndromes.
View Article and Find Full Text PDFMedicine (Baltimore)
August 2025
Kirk Kerkorian School of Medicine, University of Nevada, Las Vegas, Las Vegas, NV.
Rationale: KBG syndrome is a rare, autosomal dominant neurodevelopmental disorder characterized by developmental delay, macrodontia, distinctive facial features, and a range of systemic manifestations.
Patient Concerns: We report a pediatric patient with a history of global developmental delay, autism spectrum disorder, sensorineural hearing loss, and spastic diplegia who developed episodic, unilateral dystonic spells beginning at age 7, leading to impaired mobility.
Diagnoses: Initial genetic testing revealed a maternally inherited 3p26 duplication, which did not fully account for the patient's clinical presentation.
J AAPOS
July 2025
Department of Ophthalmology, Seoul National University College of Medicine, Seoul National University Bundang Hospital, Seongnam, Korea. Electronic address:
KBG syndrome is characterized by intellectual disability, and craniofacial and skeletal abnormalities. We report the case of a 5-year-old boy with KBG syndrome with 16q24.3 microdeletion who showed complex strabismus.
View Article and Find Full Text PDFAm J Med Genet A
July 2025
Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.
KBG syndrome is one of the most frequent neurodevelopmental disorders and is caused by ANKRD11 variants. Postnatal short stature is observed in ~50% of patients. Recombinant human growth hormone (rhGH) has become a valuable treatment for patients with growth hormone deficiency (GHD) along with Prader-Willi and Turner syndrome.
View Article and Find Full Text PDFFront Pediatr
June 2025
Department of Pediatrics, West China Second University Hospital, Sichuan University, Chengdu, Sichuan, China.
The KBG syndrome (KBGS) affects several systems caused by the mutation of the gene. The main manifestations of KGBS included hearing loss, feeding difficulties, craniofacial abnormalities, tooth deformity, and developmental delay (delayed overall development, convulsions, and intellectual abnormalities). Only 10%-26% of patients with KBG syndrome have congenital heart disease, including atrial and ventricular septal defects.
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