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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.
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http://dx.doi.org/10.1002/ajmg.a.63996 | DOI Listing |
Br J Haematol
September 2025
Department of Pediatrics, Stanford University, Stanford, California, USA.
Chronic myeloid leukaemia (CML) accounts for 2% of leukaemias in children and 9% in adolescents. While the BCR::ABL1 fusion gene remains a hallmark across all age groups, emerging evidence suggests that paediatric CML exhibits unique biological and clinical characteristics compared to its adult counterpart. Children often present with more aggressive clinical features and show distinct treatment response patterns.
View Article and Find Full Text PDFLeuk Res Rep
August 2025
Department of Hematology, The Second Hospital & Clinical Medical School, Lanzhou University, No. 82, Cuyingmen, Lanzhou, Gansu Province 730030, China.
The use of TKIs has significantly improved the prognosis of CML. However, a small subset of patients still experience poor outcomes. We present a rare case of Ph-AML following a diagnosis of CML.
View Article and Find Full Text PDFFuture Oncol
September 2025
Division of Leukemia, Department of Medical Oncology, Dana-Farber Cancer Institute, Boston, MA, USA.
Philadelphia chromosome-positive acute lymphoblastic leukemia (Ph+ ALL) is characterized by the fusion gene which produces a constitutively active tyrosine kinase which drives disease pathogenesis and is associated with resistance to conventional chemotherapy. Intensive cytotoxic chemotherapy followed by allogeneic hematopoietic stem cell transplantation (HSCT), the historical treatment paradigm for Ph+ ALL, was associated with poor outcomes. The introduction of inhibitors of ABL1 revolutionized the treatment of Ph+ ALL.
View Article and Find Full Text PDFInt J Lab Hematol
September 2025
Department of Medical Oncology, Dr BRA IRCH, All India Institute of Medical Sciences (AIIMS), New Delhi, India.
Introduction: B-cell acute lymphoblastic leukemia (B-ALL) is genetically heterogeneous. We assessed the utility of FusionPlex ALL targeted RNA sequencing panel in detecting gene fusions and other genomic lesions in B-ALL.
Methods: The high-risk B-ALL, negative for common recurrent gene fusions (RGF), that is, BCR::ABL1, ETV6::RUNX1, TCF3::PBX1 and KMT2A::AFF1, were analysed with RNA-based targeted sequencing 81-gene-panel FusionPlex ALL (IDT, USA).
Blood
September 2025
Centre for Cancer Biology, SA Pathology, Adelaide, Australia.
Genomic profiling in chronic-phase chronic myeloid leukemia (CP-CML) patients demonstrated somatic variants in blood cancer-related genes (CGVs) and rearrangements associated with the formation of the Philadelphia-chromosome (Ph-associated rearrangements) at diagnosis, collectively termed additional genetic abnormalities (AGA). AGAs had a negative impact on failure-free survival and molecular response in imatinib-treated patients. We investigated whether treatment with more potent therapies could overcome the negative impact of AGAs at diagnosis.
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