Atypical femoral fractures in a Mexican cohort of children and adolescents with osteogenesis imperfecta. Analysis of trajectories.
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| Title: | Atypical femoral fractures in a Mexican cohort of children and adolescents with osteogenesis imperfecta. Analysis of trajectories. |
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| Alternate Title: | Fracturas femorales atípicas en una cohorte mexicana de niños y adolescentes con osteogénesis imperfecta. Análisis de trayectorias. |
| Authors: | Bremer, A.1,2, Clark, P.1,3 osteoclark@gmail.com, Méndez-Sánchez, L.1,4, García-de la Torre, G.1,5 |
| Source: | Acta Ortopédica Mexicana. Nov/Dec2025, Vol. 39 Issue 6, p363-368. 6p. |
| Subjects: | OSTEOGENESIS imperfecta, FEMORAL fractures, FRACTURE strength, ODDS ratio, ZOLEDRONIC acid, CHILD patients, OI SA, INTERDISCIPLINARY research |
| Abstract (English): | Introduction: osteogenesis imperfecta (OI) is a rare inherited bone disorder resulting from defects in type I collagen synthesis or structure. It is characterized by increased bone fragility, recurrent fractures, and early- onset hearing loss. Management requires a multidisciplinary approach aimed at reducing fracture incidence, enhancing mobility, and promoting functional independence. Since 1998, bisphosphonates (BPs) have been used as compassionate therapy in OI. Objective: to characterize children with osteogenesis imperfecta (OI) and report the incidence of atypical femoral fractures (AFFs) in relation to the duration of zoledronic acid treatment. Material and methods: a single group ambispective cohort study of pediatric patients diagnosed with osteogenesis imperfecta according to the Sillence criteria and treated with zoledronic acid (0.1 mg/kg/year); the retrospective period spanned from January 2008 to July 2017, while the prospective period extended from October 2017 to March 2024. Results: a total of 68 patients were included, with 24 fractures identified in 21 patients (31%). Fractures were more frequent in males (67%), adolescents (76%) and those receiving continuous treatment (76%). The duration of treatment before developing an AFF was also shorter in males (69 months) compared to females (77 months). Cox regression analysis showed that male patients with OI had a hazard ratio (HR) 3.13 (95% CI 1.18-8.26, p = 0.021) indicating a significantly higher risk of developing an AFF compared to female patients. Conclusion: there may be an association between prolonged use of zoledronic acid and the occurrence of AFFs. Male and adolescents exhibited a higher risk of AFFs (HR 3.13, 95% CI 1.18-8.26) These fractures may not be limited to the femur, as other long bones could also be affected. Larger cohorts and longer follow-up periods, including control groups not exposed to zoledronic acid, are needed to establish causation. [ABSTRACT FROM AUTHOR] |
| Abstract (Spanish): | Introducción: la osteogénesis imperfecta (OI) es un trastorno óseo hereditario poco frecuente causado por defectos en la síntesis o estructura del colágeno tipo I. Se caracteriza por una mayor fragilidad ósea, fracturas recurrentes y pérdida auditiva de inicio temprano. Su tratamiento requiere un enfoque multidisciplinario dirigido a reducir la incidencia de fracturas, mejorar la movilidad y promover la independencia funcional. Desde 1998, los bifosfonatos (BF) se utilizan como tratamiento compasivo en la OI. Objetivo: caracterizar a niños y adolescentes con osteogénesis imperfecta (OI) y reportar la incidencia de fracturas femorales atípicas (FFA) en relación con la duración del tratamiento con ácido zoledrónico. Material y métodos: estudio de cohorte ambispectivo de un solo grupo, conformado por pacientes pediátricos diagnosticados con OI según los criterios de Sillence y tratados con ácido zoledrónico (0.1 mg/kg/año). El período retrospectivo abarcó de enero de 2008 a julio de 2017, y el periodo prospectivo de octubre de 2017 a marzo de 2024. Resultados: se incluyeron 68 pacientes, con un total de 24 fracturas identificadas en 21 pacientes (31%). Las fracturas fueron más frecuentes en niños (67%), adolescentes (76%) y en aquellos que recibieron tratamiento continuo (76%). La duración del tratamiento antes de presentar una FFA fue menor en hombres (69 meses) en comparación con mujeres (77 meses). El análisis de regresión de Cox mostró que los pacientes con OI presentaron una razón de riesgo (HR) de 3.13 (intervalo de confianza [IC] del 95%: 1.18-8.26; p = 0.021), indicando un riesgo significativamente mayor de desarrollar una FFA en comparación con las pacientes femeninas. Conclusión: podría existir una asociación entre el uso prolongado de ácido zoledrónico y la aparición de FFA. Los varones y los adolescentes presentaron un mayor riesgo de fractura (HR 3.13; IC95%: 1.18-8.26). Estas fracturas podrían no limitarse únicamente al fémur, ya que otros huesos largos también podrían verse afectados. Se requieren cohortes de mayor tamaño y periodos de seguimiento más prolongados, incluyendo grupos control no expuestos a ácido zoledrónico, para establecer una relación causal. [ABSTRACT FROM AUTHOR] |
| Copyright of Acta Ortopédica Mexicana is the property of Sociedad Mexicana de Ortopedia, AC and its content may not be copied or emailed to multiple sites without the copyright holder's express written permission. Additionally, content may not be used with any artificial intelligence tools or machine learning technologies. However, users may print, download, or email articles for individual use. This abstract may be abridged. No warranty is given about the accuracy of the copy. Users should refer to the original published version of the material for the full abstract. (Copyright applies to all Abstracts.) | |
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| Items | – Name: Title Label: Title Group: Ti Data: Atypical femoral fractures in a Mexican cohort of children and adolescents with osteogenesis imperfecta. Analysis of trajectories. – Name: TitleAlt Label: Alternate Title Group: TiAlt Data: Fracturas femorales atípicas en una cohorte mexicana de niños y adolescentes con osteogénesis imperfecta. Análisis de trayectorias. – Name: Author Label: Authors Group: Au Data: <searchLink fieldCode="AR" term="%22Bremer%2C+A%2E%22">Bremer, A.</searchLink><relatesTo>1,2</relatesTo><br /><searchLink fieldCode="AR" term="%22Clark%2C+P%2E%22">Clark, P.</searchLink><relatesTo>1,3</relatesTo><i> osteoclark@gmail.com</i><br /><searchLink fieldCode="AR" term="%22Méndez-Sánchez%2C+L%2E%22">Méndez-Sánchez, L.</searchLink><relatesTo>1,4</relatesTo><br /><searchLink fieldCode="AR" term="%22García-de+la+Torre%2C+G%2E%22">García-de la Torre, G.</searchLink><relatesTo>1,5</relatesTo> – Name: TitleSource Label: Source Group: Src Data: <searchLink fieldCode="JN" term="%22Acta+Ortopédica+Mexicana%22">Acta Ortopédica Mexicana</searchLink>. Nov/Dec2025, Vol. 39 Issue 6, p363-368. 6p. – Name: Subject Label: Subjects Group: Su Data: <searchLink fieldCode="DE" term="%22OSTEOGENESIS+imperfecta%22">OSTEOGENESIS imperfecta</searchLink><br /><searchLink fieldCode="DE" term="%22FEMORAL+fractures%22">FEMORAL fractures</searchLink><br /><searchLink fieldCode="DE" term="%22FRACTURE+strength%22">FRACTURE strength</searchLink><br /><searchLink fieldCode="DE" term="%22ODDS+ratio%22">ODDS ratio</searchLink><br /><searchLink fieldCode="DE" term="%22ZOLEDRONIC+acid%22">ZOLEDRONIC acid</searchLink><br /><searchLink fieldCode="DE" term="%22CHILD+patients%22">CHILD patients</searchLink><br /><searchLink fieldCode="DE" term="%22OI+SA%22">OI SA</searchLink><br /><searchLink fieldCode="DE" term="%22INTERDISCIPLINARY+research%22">INTERDISCIPLINARY research</searchLink> – Name: Abstract Label: Abstract (English) Group: Ab Data: Introduction: osteogenesis imperfecta (OI) is a rare inherited bone disorder resulting from defects in type I collagen synthesis or structure. It is characterized by increased bone fragility, recurrent fractures, and early- onset hearing loss. Management requires a multidisciplinary approach aimed at reducing fracture incidence, enhancing mobility, and promoting functional independence. Since 1998, bisphosphonates (BPs) have been used as compassionate therapy in OI. Objective: to characterize children with osteogenesis imperfecta (OI) and report the incidence of atypical femoral fractures (AFFs) in relation to the duration of zoledronic acid treatment. Material and methods: a single group ambispective cohort study of pediatric patients diagnosed with osteogenesis imperfecta according to the Sillence criteria and treated with zoledronic acid (0.1 mg/kg/year); the retrospective period spanned from January 2008 to July 2017, while the prospective period extended from October 2017 to March 2024. Results: a total of 68 patients were included, with 24 fractures identified in 21 patients (31%). Fractures were more frequent in males (67%), adolescents (76%) and those receiving continuous treatment (76%). The duration of treatment before developing an AFF was also shorter in males (69 months) compared to females (77 months). Cox regression analysis showed that male patients with OI had a hazard ratio (HR) 3.13 (95% CI 1.18-8.26, p = 0.021) indicating a significantly higher risk of developing an AFF compared to female patients. Conclusion: there may be an association between prolonged use of zoledronic acid and the occurrence of AFFs. Male and adolescents exhibited a higher risk of AFFs (HR 3.13, 95% CI 1.18-8.26) These fractures may not be limited to the femur, as other long bones could also be affected. Larger cohorts and longer follow-up periods, including control groups not exposed to zoledronic acid, are needed to establish causation. [ABSTRACT FROM AUTHOR] – Name: Abstract Label: Abstract (Spanish) Group: Ab Data: Introducción: la osteogénesis imperfecta (OI) es un trastorno óseo hereditario poco frecuente causado por defectos en la síntesis o estructura del colágeno tipo I. Se caracteriza por una mayor fragilidad ósea, fracturas recurrentes y pérdida auditiva de inicio temprano. Su tratamiento requiere un enfoque multidisciplinario dirigido a reducir la incidencia de fracturas, mejorar la movilidad y promover la independencia funcional. Desde 1998, los bifosfonatos (BF) se utilizan como tratamiento compasivo en la OI. Objetivo: caracterizar a niños y adolescentes con osteogénesis imperfecta (OI) y reportar la incidencia de fracturas femorales atípicas (FFA) en relación con la duración del tratamiento con ácido zoledrónico. Material y métodos: estudio de cohorte ambispectivo de un solo grupo, conformado por pacientes pediátricos diagnosticados con OI según los criterios de Sillence y tratados con ácido zoledrónico (0.1 mg/kg/año). El período retrospectivo abarcó de enero de 2008 a julio de 2017, y el periodo prospectivo de octubre de 2017 a marzo de 2024. Resultados: se incluyeron 68 pacientes, con un total de 24 fracturas identificadas en 21 pacientes (31%). Las fracturas fueron más frecuentes en niños (67%), adolescentes (76%) y en aquellos que recibieron tratamiento continuo (76%). La duración del tratamiento antes de presentar una FFA fue menor en hombres (69 meses) en comparación con mujeres (77 meses). El análisis de regresión de Cox mostró que los pacientes con OI presentaron una razón de riesgo (HR) de 3.13 (intervalo de confianza [IC] del 95%: 1.18-8.26; p = 0.021), indicando un riesgo significativamente mayor de desarrollar una FFA en comparación con las pacientes femeninas. Conclusión: podría existir una asociación entre el uso prolongado de ácido zoledrónico y la aparición de FFA. Los varones y los adolescentes presentaron un mayor riesgo de fractura (HR 3.13; IC95%: 1.18-8.26). Estas fracturas podrían no limitarse únicamente al fémur, ya que otros huesos largos también podrían verse afectados. Se requieren cohortes de mayor tamaño y periodos de seguimiento más prolongados, incluyendo grupos control no expuestos a ácido zoledrónico, para establecer una relación causal. [ABSTRACT FROM AUTHOR] – Name: AbstractSuppliedCopyright Label: Group: Ab Data: <i>Copyright of Acta Ortopédica Mexicana is the property of Sociedad Mexicana de Ortopedia, AC and its content may not be copied or emailed to multiple sites without the copyright holder's express written permission. Additionally, content may not be used with any artificial intelligence tools or machine learning technologies. However, users may print, download, or email articles for individual use. This abstract may be abridged. No warranty is given about the accuracy of the copy. Users should refer to the original published version of the material for the full abstract.</i> (Copyright applies to all Abstracts.) |
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| RecordInfo | BibRecord: BibEntity: Identifiers: – Type: doi Value: 10.35366/121814 Languages: – Code: eng Text: English PhysicalDescription: Pagination: PageCount: 6 StartPage: 363 Subjects: – SubjectFull: OSTEOGENESIS imperfecta Type: general – SubjectFull: FEMORAL fractures Type: general – SubjectFull: FRACTURE strength Type: general – SubjectFull: ODDS ratio Type: general – SubjectFull: ZOLEDRONIC acid Type: general – SubjectFull: CHILD patients Type: general – SubjectFull: OI SA Type: general – SubjectFull: INTERDISCIPLINARY research Type: general Titles: – TitleFull: Atypical femoral fractures in a Mexican cohort of children and adolescents with osteogenesis imperfecta. Analysis of trajectories. Type: main BibRelationships: HasContributorRelationships: – PersonEntity: Name: NameFull: Bremer, A. – PersonEntity: Name: NameFull: Clark, P. – PersonEntity: Name: NameFull: Méndez-Sánchez, L. – PersonEntity: Name: NameFull: García-de la Torre, G. IsPartOfRelationships: – BibEntity: Dates: – D: 01 M: 11 Text: Nov/Dec2025 Type: published Y: 2025 Identifiers: – Type: issn-print Value: 23064102 Numbering: – Type: volume Value: 39 – Type: issue Value: 6 Titles: – TitleFull: Acta Ortopédica Mexicana Type: main |
| ResultId | 1 |
