| Literature DB >> 28095295 |
Eleonora Palagano1, Mary A Slatter2, Paolo Uva3, Ciro Menale4, Anna Villa4, Mario Abinun5, Cristina Sobacchi4.
Abstract
Osteopetrosis (OPT) is a rare skeletal disorder with phenotypic and genotypic heterogeneity: a variety of clinical features besides the bony defect may be present, and at least ten different genes are known to be involved in the disease pathogenesis. In the framework of this heterogeneity, we report the clinical description of a neonate, first child of consanguineous parents, who had osteoclast-rich osteopetrosis and bone marrow failure in early life, but no other usual classical features of infantile malignant OPT, such as visual or hearing impairments. Because of the severe presentation at birth, the patient received Hematopoietic Stem Cell Transplantation (HSCT) at 2months of age with successful outcome. Post-HSCT genetic investigation by means of exome sequencing identified a novel homozygous mutation in the Fermitin Family Member 3 (FERMT3) gene, which was predicted to disrupt the functionality of its protein product kindlin 3. Our report provides information relevant to physicians for recognizing patients with one of the rarest forms of infantile malignant OPT, and clearly demonstrates that HSCT cures kindlin 3 deficiency with severe phenotype.Entities:
Keywords: Bleeding; Integrin; Osteoclast
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Year: 2017 PMID: 28095295 DOI: 10.1016/j.bone.2017.01.012
Source DB: PubMed Journal: Bone ISSN: 1873-2763 Impact factor: 4.398