Skeletal Changes After Hematopoietic Stem Cell Transplantation in Osteopetrosis.


Journal

Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research
ISSN: 1523-4681
Titre abrégé: J Bone Miner Res
Pays: United States
ID NLM: 8610640

Informations de publication

Date de publication:
09 2020
Historique:
received: 15 10 2019
revised: 30 03 2020
accepted: 16 04 2020
pubmed: 25 4 2020
medline: 7 7 2021
entrez: 25 4 2020
Statut: ppublish

Résumé

Osteopetrosis is a rare skeletal dysplasia resulting from an osteoclast defect leading to increased bone mass and density. Hematopoietic stem cell transplantation can rescue the disease phenotype and prevent complications. However, little is known about the skeletal changes hematopoietic stem cell transplantation induces in patients with this disease. The purpose of this study was to describe the skeletal changes after hematopoietic stem cell transplantation in a retrospective cohort of patients diagnosed with osteopetrosis in one medical center over 13 years. For this purpose, all available epidemiological, hematological, biochemical, and radiographic data were collected and quantitatively analyzed. We found a significant early change in bone metabolism markers coinciding with hematopoietic recovery after stem cell transplantation. Hematopoietic stem cell transplantation induced a later significant improvement in both skeletal mineral distribution and morphology but did not lead to complete radiological normalization. Presumably, changes in bone metabolism, skeletal mineral distribution, and morphology were the result of renewed osteoclast function enabling bone remodeling. We propose that biochemical bone metabolism markers and radiological indices be routinely used to evaluate response to hematopoietic stem cell transplantation in patients with osteopetrosis. © 2020 American Society for Bone and Mineral Research.

Identifiants

pubmed: 32329913
doi: 10.1002/jbmr.4037
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

1645-1651

Informations de copyright

© 2020 American Society for Bone and Mineral Research.

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Auteurs

Galina Shapiro (G)

Medical Corps, Israel Defense Forces, Ramat Gan, Israel.

Jorge Fishleder (J)

Pediatric Orthopedics Unit, Department of Orthopedic Surgery, Hadassah-Hebrew University Medical Center, Jerusalem, Israel.

Polina Stepensky (P)

Department of Bone Marrow Transplantation and Cancer Immunotherapy, Hadassah-Hebrew University Medical Center, Jerusalem, Israel.

Naum Simanovsky (N)

Pediatric Orthopedics Unit, Department of Orthopedic Surgery, Hadassah-Hebrew University Medical Center, Jerusalem, Israel.

Vladimir Goldman (V)

Pediatric Orthopedics Unit, Department of Orthopedic Surgery, Hadassah-Hebrew University Medical Center, Jerusalem, Israel.

Ron Lamdan (R)

Pediatric Orthopedics Unit, Assuta Ashdod Medical Center, Ashdod, Israel.

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