Chen Ting-Yi, Chen Yi, Tang Lan-Fang
Department of Pulmonology, Children's Hospital of Zhejiang University School of Medicine & National Clinical Research Center for Child Health, Hangzhou, China.
Am J Med Genet A. 2025 Jun;197(6):e63996. doi: 10.1002/ajmg.a.63996. Epub 2025 Jan 30.
Congenital heart defects and skeletal malformations syndrome (CHDSKM) is a rare autosomal dominant genetic disorder characterized by specific clinical features, including dysmorphic facial traits, congenital heart defects, skeletal abnormalities, joint issues, and failure to thrive. The novelty of this case lies in the identification of a novel mutation in the ABL1 gene, expanding the genetic spectrum associated with this syndrome. A 5.9-year-old boy was referred to the clinic due to growth retardation and intellectual disability. Clinical evaluation revealed several hallmark features of CHDSKM, including distinct facial dysmorphisms such as a broad forehead, frontal bossing, micrognathia, low-set ears, and short palpebral fissures. The patient was diagnosed with congenital heart defects, including a ventricular septal defect, atrial septal defect, and patent ductus arteriosus. Skeletal malformations included scoliosis and finger contractures. Additionally, he exhibited developmental delay, gastrointestinal issues such as umbilical hernia and intestinal malrotation, intellectual disability, and dysgenesis of the corpus callosum, which are atypical for this syndrome. Molecular genetic analysis identified a de novo mutation (c.898C>G) in exon 5 of the ABL1 gene, resulting in a novel missense mutation (p.Gln300Glu). This case emphasizes the importance of considering CHDSKM in the differential diagnosis of children with growth and developmental concerns. The identification of a novel mutation in the ABL1 gene highlights the critical role of early molecular genetic testing, which can facilitate improved management and support for affected individuals and their families.
先天性心脏缺陷与骨骼畸形综合征(CHDSKM)是一种罕见的常染色体显性遗传病,具有特定的临床特征,包括面部畸形特征、先天性心脏缺陷、骨骼异常、关节问题和生长发育迟缓。该病例的新颖之处在于在ABL1基因中鉴定出一种新的突变,扩展了与该综合征相关的基因谱。一名5.9岁男孩因生长发育迟缓及智力障碍被转诊至诊所。临床评估发现了CHDSKM的几个标志性特征,包括明显的面部畸形,如额头宽阔、额部隆起、小颌畸形、低位耳和睑裂短小。该患者被诊断患有先天性心脏缺陷,包括室间隔缺损、房间隔缺损和动脉导管未闭。骨骼畸形包括脊柱侧弯和手指挛缩。此外,他还表现出发育迟缓、胃肠道问题,如脐疝和肠旋转不良、智力障碍以及胼胝体发育不全,这些在该综合征中并不典型。分子遗传学分析在ABL1基因第5外显子中鉴定出一个新发突变(c.898C>G),导致一个新的错义突变(p.Gln300Glu)。该病例强调了在对生长发育有问题的儿童进行鉴别诊断时考虑CHDSKM的重要性。ABL1基因中一个新突变的鉴定突出了早期分子遗传学检测的关键作用,这有助于改善对受影响个体及其家庭的管理和支持。