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Spontaneous reshaping of vertebral fractures in an adolescent with osteogenesis imperfecta

BACKGROUND: Vertebral compression fractures (VFs) are a common and severe finding in patients with osteoporosis. In children, VFs have the unique potential to reshape and regain their original configuration. Spontaneous vertebral body reshaping (i.e., medication-unassisted) has been reported in seco...

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Autores principales: Montero-Lopez, Rodrigo, Laurer, Elisabeth, Tischlinger, Katharina, Nagy, Dóra, Scala, Mario, Kranewitter, Wolfgang, Webersinke, Gerald, Hörtenhuber, Thomas, Högler, Wolfgang
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Elsevier 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9178468/
https://www.ncbi.nlm.nih.gov/pubmed/35693066
http://dx.doi.org/10.1016/j.bonr.2022.101595
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author Montero-Lopez, Rodrigo
Laurer, Elisabeth
Tischlinger, Katharina
Nagy, Dóra
Scala, Mario
Kranewitter, Wolfgang
Webersinke, Gerald
Hörtenhuber, Thomas
Högler, Wolfgang
author_facet Montero-Lopez, Rodrigo
Laurer, Elisabeth
Tischlinger, Katharina
Nagy, Dóra
Scala, Mario
Kranewitter, Wolfgang
Webersinke, Gerald
Hörtenhuber, Thomas
Högler, Wolfgang
author_sort Montero-Lopez, Rodrigo
collection PubMed
description BACKGROUND: Vertebral compression fractures (VFs) are a common and severe finding in patients with osteoporosis. In children, VFs have the unique potential to reshape and regain their original configuration. Spontaneous vertebral body reshaping (i.e., medication-unassisted) has been reported in secondary osteoporosis. Here we describe a previously unreported spontaneous vertebral reshaping in an adolescent with osteogenesis imperfecta (OI) with multiple vertebral fractures. CASE REPORT: A 17-year-old female was diagnosed with OI type I at 5 years of age caused by a novel frameshift variant in COL1A1 (NM_000088.4: c.540delC; p.Met181TrpfsTer84). Due to parental reservations about medication, she had never received bisphosphonate or any other bone active therapy. A lateral spine X-ray demonstrated transparent bones and no VF. However, previous spine X-rays taken at age of 6 years at an external institution showed VFs in T5–7 (Genant semiquantitative method grade I-II). The two lateral spine x-rays, taken 11 years apart, demonstrate that substantial spontaneous vertebral reshaping occurred without bone active therapy during puberty. DISCUSSION: Vertebral reshaping is explained by the stabilization of bone mineral density (BMD) and the remaining growth capacity the children. We hypothesize that spontaneous reshaping may occur in milder forms of OI, and that puberty may be a key mediator of the phenomenon. In all children with OI and vertebral fractures, we nevertheless recommend bisphosphonate therapy since it improves bone mass, BMD, vertebral shape, physical activity and reduces fracture rates.
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spelling pubmed-91784682022-06-10 Spontaneous reshaping of vertebral fractures in an adolescent with osteogenesis imperfecta Montero-Lopez, Rodrigo Laurer, Elisabeth Tischlinger, Katharina Nagy, Dóra Scala, Mario Kranewitter, Wolfgang Webersinke, Gerald Hörtenhuber, Thomas Högler, Wolfgang Bone Rep Case Report BACKGROUND: Vertebral compression fractures (VFs) are a common and severe finding in patients with osteoporosis. In children, VFs have the unique potential to reshape and regain their original configuration. Spontaneous vertebral body reshaping (i.e., medication-unassisted) has been reported in secondary osteoporosis. Here we describe a previously unreported spontaneous vertebral reshaping in an adolescent with osteogenesis imperfecta (OI) with multiple vertebral fractures. CASE REPORT: A 17-year-old female was diagnosed with OI type I at 5 years of age caused by a novel frameshift variant in COL1A1 (NM_000088.4: c.540delC; p.Met181TrpfsTer84). Due to parental reservations about medication, she had never received bisphosphonate or any other bone active therapy. A lateral spine X-ray demonstrated transparent bones and no VF. However, previous spine X-rays taken at age of 6 years at an external institution showed VFs in T5–7 (Genant semiquantitative method grade I-II). The two lateral spine x-rays, taken 11 years apart, demonstrate that substantial spontaneous vertebral reshaping occurred without bone active therapy during puberty. DISCUSSION: Vertebral reshaping is explained by the stabilization of bone mineral density (BMD) and the remaining growth capacity the children. We hypothesize that spontaneous reshaping may occur in milder forms of OI, and that puberty may be a key mediator of the phenomenon. In all children with OI and vertebral fractures, we nevertheless recommend bisphosphonate therapy since it improves bone mass, BMD, vertebral shape, physical activity and reduces fracture rates. Elsevier 2022-06-03 /pmc/articles/PMC9178468/ /pubmed/35693066 http://dx.doi.org/10.1016/j.bonr.2022.101595 Text en © 2022 Published by Elsevier Inc. https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
spellingShingle Case Report
Montero-Lopez, Rodrigo
Laurer, Elisabeth
Tischlinger, Katharina
Nagy, Dóra
Scala, Mario
Kranewitter, Wolfgang
Webersinke, Gerald
Hörtenhuber, Thomas
Högler, Wolfgang
Spontaneous reshaping of vertebral fractures in an adolescent with osteogenesis imperfecta
title Spontaneous reshaping of vertebral fractures in an adolescent with osteogenesis imperfecta
title_full Spontaneous reshaping of vertebral fractures in an adolescent with osteogenesis imperfecta
title_fullStr Spontaneous reshaping of vertebral fractures in an adolescent with osteogenesis imperfecta
title_full_unstemmed Spontaneous reshaping of vertebral fractures in an adolescent with osteogenesis imperfecta
title_short Spontaneous reshaping of vertebral fractures in an adolescent with osteogenesis imperfecta
title_sort spontaneous reshaping of vertebral fractures in an adolescent with osteogenesis imperfecta
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9178468/
https://www.ncbi.nlm.nih.gov/pubmed/35693066
http://dx.doi.org/10.1016/j.bonr.2022.101595
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