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Osteogenesis Imperfecta: characterization of fractures during pregnancy and post-partum

BACKGROUND: Pregnancy and breastfeeding are associated with bone density loss. Fracture occurrence during pregnancy and post-partum, and its determinants, remain poorly known in Osteogenesis Imperfecta (OI). The aim of this study was to characterize fractures that occurred during pregnancy and post-...

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Autores principales: Koumakis, Eugénie, Cormier-Daire, Valérie, Dellal, Azeddine, Debernardi, Marc, Cortet, Bernard, Debiais, Françoise, Javier, Rose-Marie, Thomas, Thierry, Mehsen-Cetre, Nadia, Cohen-Solal, Martine, Fontanges, Elisabeth, Laroche, Michel, Porquet-Bordes, Valérie, Marcelli, Christian, Benachi, Alexandra, Briot, Karine, Roux, Christian, Cormier, Catherine
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8796450/
https://www.ncbi.nlm.nih.gov/pubmed/35090500
http://dx.doi.org/10.1186/s13023-021-02148-x
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author Koumakis, Eugénie
Cormier-Daire, Valérie
Dellal, Azeddine
Debernardi, Marc
Cortet, Bernard
Debiais, Françoise
Javier, Rose-Marie
Thomas, Thierry
Mehsen-Cetre, Nadia
Cohen-Solal, Martine
Fontanges, Elisabeth
Laroche, Michel
Porquet-Bordes, Valérie
Marcelli, Christian
Benachi, Alexandra
Briot, Karine
Roux, Christian
Cormier, Catherine
author_facet Koumakis, Eugénie
Cormier-Daire, Valérie
Dellal, Azeddine
Debernardi, Marc
Cortet, Bernard
Debiais, Françoise
Javier, Rose-Marie
Thomas, Thierry
Mehsen-Cetre, Nadia
Cohen-Solal, Martine
Fontanges, Elisabeth
Laroche, Michel
Porquet-Bordes, Valérie
Marcelli, Christian
Benachi, Alexandra
Briot, Karine
Roux, Christian
Cormier, Catherine
author_sort Koumakis, Eugénie
collection PubMed
description BACKGROUND: Pregnancy and breastfeeding are associated with bone density loss. Fracture occurrence during pregnancy and post-partum, and its determinants, remain poorly known in Osteogenesis Imperfecta (OI). The aim of this study was to characterize fractures that occurred during pregnancy and post-partum in OI patients. RESULTS: We conducted a retrospective multicentric study including a total of 50 previously pregnant OI women from 10 Bone Centers in France. Among these patients, 12 (24%) patients experienced fractures during pregnancy or in the 6 months following delivery, and 38 (76%) did not experience any fracture. The most frequent localizations were: proximal femur (25%), spine (25%), distal femur (12.5%), and pelvis (12.5%). Fractures during pregnancy occurred during the third trimester and post-partum fractures occurred with a mean delay of 2 months following delivery. No fractures occurred during childbirth. We next compared the 12 patients with pregnancy or post-partum fractures with the 38 patients without fractures. Mean age at pregnancy was 32.7 ± 3.1 years-old in the fractured group, vs 29.3 ± 5.0 years-old in the non-fractured group (p = 0.002). Breastfeeding was reported in 85.7% of patients in the fractured group, vs 47.1% in the non-fractured group (p = 0.03). All patients with post-partum fractures were breastfeeding. Bone mineral density was significantly lower in patients with pregnancy-related fractures compared with other patients: spine Z-score − 2.9 ± 1.6DS vs − 1.5 ± 1.7DS (p = 0.03), and total hip Z-score − 2.0 ± 0.7DS vs − 0.5 ± 1.4DS (p = 0.04). At least one osteoporosis-inducing risk factor or disease other than OI was identified in 81.8% vs 58.6% of fractured vs non-fractured patients (not significant). Fracture during pregnancy or post-partum was not associated with the severity of OI. Bisphosphonates before pregnancy were reported in 16.7% and 21.1% of patients with pregnancy-related fractures and non-fractured patients, respectively (not significant). CONCLUSIONS: OI management during pregnancy and post-partum should aim for optimal control of modifiable osteoporosis risk factors, particularly in patients with low BMD. Breastfeeding should be avoided.
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spelling pubmed-87964502022-02-03 Osteogenesis Imperfecta: characterization of fractures during pregnancy and post-partum Koumakis, Eugénie Cormier-Daire, Valérie Dellal, Azeddine Debernardi, Marc Cortet, Bernard Debiais, Françoise Javier, Rose-Marie Thomas, Thierry Mehsen-Cetre, Nadia Cohen-Solal, Martine Fontanges, Elisabeth Laroche, Michel Porquet-Bordes, Valérie Marcelli, Christian Benachi, Alexandra Briot, Karine Roux, Christian Cormier, Catherine Orphanet J Rare Dis Research BACKGROUND: Pregnancy and breastfeeding are associated with bone density loss. Fracture occurrence during pregnancy and post-partum, and its determinants, remain poorly known in Osteogenesis Imperfecta (OI). The aim of this study was to characterize fractures that occurred during pregnancy and post-partum in OI patients. RESULTS: We conducted a retrospective multicentric study including a total of 50 previously pregnant OI women from 10 Bone Centers in France. Among these patients, 12 (24%) patients experienced fractures during pregnancy or in the 6 months following delivery, and 38 (76%) did not experience any fracture. The most frequent localizations were: proximal femur (25%), spine (25%), distal femur (12.5%), and pelvis (12.5%). Fractures during pregnancy occurred during the third trimester and post-partum fractures occurred with a mean delay of 2 months following delivery. No fractures occurred during childbirth. We next compared the 12 patients with pregnancy or post-partum fractures with the 38 patients without fractures. Mean age at pregnancy was 32.7 ± 3.1 years-old in the fractured group, vs 29.3 ± 5.0 years-old in the non-fractured group (p = 0.002). Breastfeeding was reported in 85.7% of patients in the fractured group, vs 47.1% in the non-fractured group (p = 0.03). All patients with post-partum fractures were breastfeeding. Bone mineral density was significantly lower in patients with pregnancy-related fractures compared with other patients: spine Z-score − 2.9 ± 1.6DS vs − 1.5 ± 1.7DS (p = 0.03), and total hip Z-score − 2.0 ± 0.7DS vs − 0.5 ± 1.4DS (p = 0.04). At least one osteoporosis-inducing risk factor or disease other than OI was identified in 81.8% vs 58.6% of fractured vs non-fractured patients (not significant). Fracture during pregnancy or post-partum was not associated with the severity of OI. Bisphosphonates before pregnancy were reported in 16.7% and 21.1% of patients with pregnancy-related fractures and non-fractured patients, respectively (not significant). CONCLUSIONS: OI management during pregnancy and post-partum should aim for optimal control of modifiable osteoporosis risk factors, particularly in patients with low BMD. Breastfeeding should be avoided. BioMed Central 2022-01-28 /pmc/articles/PMC8796450/ /pubmed/35090500 http://dx.doi.org/10.1186/s13023-021-02148-x Text en © The Author(s) 2022 https://creativecommons.org/licenses/by/4.0/Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/ (https://creativecommons.org/publicdomain/zero/1.0/) ) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Research
Koumakis, Eugénie
Cormier-Daire, Valérie
Dellal, Azeddine
Debernardi, Marc
Cortet, Bernard
Debiais, Françoise
Javier, Rose-Marie
Thomas, Thierry
Mehsen-Cetre, Nadia
Cohen-Solal, Martine
Fontanges, Elisabeth
Laroche, Michel
Porquet-Bordes, Valérie
Marcelli, Christian
Benachi, Alexandra
Briot, Karine
Roux, Christian
Cormier, Catherine
Osteogenesis Imperfecta: characterization of fractures during pregnancy and post-partum
title Osteogenesis Imperfecta: characterization of fractures during pregnancy and post-partum
title_full Osteogenesis Imperfecta: characterization of fractures during pregnancy and post-partum
title_fullStr Osteogenesis Imperfecta: characterization of fractures during pregnancy and post-partum
title_full_unstemmed Osteogenesis Imperfecta: characterization of fractures during pregnancy and post-partum
title_short Osteogenesis Imperfecta: characterization of fractures during pregnancy and post-partum
title_sort osteogenesis imperfecta: characterization of fractures during pregnancy and post-partum
topic Research
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8796450/
https://www.ncbi.nlm.nih.gov/pubmed/35090500
http://dx.doi.org/10.1186/s13023-021-02148-x
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