Literature DB >> 28095295

Hematopoietic stem cell transplantation corrects osteopetrosis in a child carrying a novel homozygous mutation in the FERMT3 gene.

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.
Copyright © 2017 Elsevier Inc. All rights reserved.

Entities:  

Keywords:  Bleeding; Integrin; Osteoclast

Mesh:

Substances:

Year:  2017        PMID: 28095295     DOI: 10.1016/j.bone.2017.01.012

Source DB:  PubMed          Journal:  Bone        ISSN: 1873-2763            Impact factor:   4.398


  5 in total

1.  Bone marrow transplantation as a therapy for autosomal dominant osteopetrosis type 2 in mice.

Authors:  Imranul Alam; Rita L Gerard-O'Riley; Dena Acton; Sara L Hardman; Madeline Murphy; Marta B Alvarez; Rachel J Blosser; Anthony Sinn; Edward F Srour; Melissa A Kacena; Michael J Econs
Journal:  FASEB J       Date:  2022-09       Impact factor: 5.834

Review 2.  Genetics of Osteopetrosis.

Authors:  Eleonora Palagano; Ciro Menale; Cristina Sobacchi; Anna Villa
Journal:  Curr Osteoporos Rep       Date:  2018-02       Impact factor: 5.096

3.  RNA-seq analysis of chondrocyte transcriptome reveals genetic heterogeneity in LG/J and SM/J murine strains.

Authors:  X Duan; L Cai; E J Schmidt; J Shen; E D Tycksen; R J O'Keefe; J M Cheverud; M F Rai
Journal:  Osteoarthritis Cartilage       Date:  2020-01-14       Impact factor: 6.576

Review 4.  One Disease, Many Genes: Implications for the Treatment of Osteopetroses.

Authors:  Sara Penna; Valentina Capo; Eleonora Palagano; Cristina Sobacchi; Anna Villa
Journal:  Front Endocrinol (Lausanne)       Date:  2019-02-19       Impact factor: 5.555

Review 5.  Clinical Application of Genome and Exome Sequencing as a Diagnostic Tool for Pediatric Patients: a Scoping Review of the Literature.

Authors:  Hadley Stevens Smith; J Michael Swint; Seema R Lalani; Jose-Miguel Yamal; Marcia C de Oliveira Otto; Stephan Castellanos; Amy Taylor; Brendan H Lee; Heidi V Russell
Journal:  Genet Med       Date:  2018-05-14       Impact factor: 8.822

  5 in total

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