Combined gait analysis and radiologic examination in children with X-linked hypophosphatemia.


Journal

Clinical biomechanics (Bristol, Avon)
ISSN: 1879-1271
Titre abrégé: Clin Biomech (Bristol, Avon)
Pays: England
ID NLM: 8611877

Informations de publication

Date de publication:
05 2023
Historique:
received: 28 11 2022
revised: 19 04 2023
accepted: 23 04 2023
medline: 22 5 2023
pubmed: 7 5 2023
entrez: 6 5 2023
Statut: ppublish

Résumé

X-linked hypophosphataemia causes bone deformities and gait abnormalities that tend to worsen with age in the absence of appropriate treatment. However, doctors do not currently use quantitative tools to characterize these symptoms and their possible interactions. Radiographs and 3D gait data from 43 non-surgical growing children with X-linked hypophosphataemia were acquired prospectively. Data from age-matched typically developing children were used to form the reference group. Subgroups based on radiological parameters were compared with each other and with the reference population. Linear correlations between radiographic parameters and gait variables were examined. X-linked hypophosphatemic patients differed from the control group in pelvic tilt, ankle plantarflexion, knee flexion moment and power. High correlations with tibiofemoral angle were found for trunk lean, knee and hip adduction, and knee abduction moment. The Gait Deviation Index was below 80 for 88% of the patients with a high tibiofemoral angle (varus). Compared to other subgroups, varus patients had augmented trunk lean (+3°) and knee adduction (+10°) and decreased hip adduction (-5°) and ankle plantarflexion (-6°). Femoral torsion was associated with alterations in rotation at the knee, and hip. Gait abnormalities induced in X-linked hypophosphataemia have been described in a large cohort of children. Links between gait alterations and lower limb deformities were found, with varus deformities standing out. Since bony deformities appear when X-linked hypophosphatemic children start walking and have been found to alter gait patterns, we suggest that combining radiology with gait analysis may improve the clinical management of X-linked hypophosphataemia.

Sections du résumé

BACKGROUND
X-linked hypophosphataemia causes bone deformities and gait abnormalities that tend to worsen with age in the absence of appropriate treatment. However, doctors do not currently use quantitative tools to characterize these symptoms and their possible interactions.
METHODS
Radiographs and 3D gait data from 43 non-surgical growing children with X-linked hypophosphataemia were acquired prospectively. Data from age-matched typically developing children were used to form the reference group. Subgroups based on radiological parameters were compared with each other and with the reference population. Linear correlations between radiographic parameters and gait variables were examined.
FINDING
X-linked hypophosphatemic patients differed from the control group in pelvic tilt, ankle plantarflexion, knee flexion moment and power. High correlations with tibiofemoral angle were found for trunk lean, knee and hip adduction, and knee abduction moment. The Gait Deviation Index was below 80 for 88% of the patients with a high tibiofemoral angle (varus). Compared to other subgroups, varus patients had augmented trunk lean (+3°) and knee adduction (+10°) and decreased hip adduction (-5°) and ankle plantarflexion (-6°). Femoral torsion was associated with alterations in rotation at the knee, and hip.
INTERPRETATION
Gait abnormalities induced in X-linked hypophosphataemia have been described in a large cohort of children. Links between gait alterations and lower limb deformities were found, with varus deformities standing out. Since bony deformities appear when X-linked hypophosphatemic children start walking and have been found to alter gait patterns, we suggest that combining radiology with gait analysis may improve the clinical management of X-linked hypophosphataemia.

Identifiants

pubmed: 37148614
pii: S0268-0033(23)00105-5
doi: 10.1016/j.clinbiomech.2023.105974
pii:
doi:

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

105974

Informations de copyright

Copyright © 2023 Elsevier Ltd. All rights reserved.

Déclaration de conflit d'intérêts

Declaration of Competing Interest Professor Linglart received grants and fee reimbursement from Kyowa Kirin. Professor Skalli is co-inventor of the biplanar imaging system (co-author in patents), without personal financial benefit: royalties rewarded to non-profit research and education structures. The other authors declare that they have no competing interest.

Auteurs

Aurore Bonnet-Lebrun (A)

Institut de Biomecanique Humaine Georges Charpak, Arts et Metiers Sciences et Technologies, 151 Boulevard de l'Hôpital, 75013 Paris, France. Electronic address: aurore.bonnet-lebrun@ensam.eu.

Agnès Linglart (A)

APHP, Service d'endocrinologie pédiatrique, Hôpital Bicêtre Paris Sud, 94270 Le Kremlin-Bicêtre, France; Centre de référence Maladies Rares du Métabolisme du Calcium et du Phosphore, 94270 Le Kremlin Bicetre, France.

Marine De Tienda (M)

Institut de Biomecanique Humaine Georges Charpak, Arts et Metiers Sciences et Technologies, 151 Boulevard de l'Hôpital, 75013 Paris, France; APHP, Service d'orthopédie infantile, Hôpital Necker Enfants Malades, 75015 Paris, France.

Virginie Nguyen Khac (V)

Institut de Biomecanique Humaine Georges Charpak, Arts et Metiers Sciences et Technologies, 151 Boulevard de l'Hôpital, 75013 Paris, France; APHP, Service d'orthopédie infantile, Hôpital Necker Enfants Malades, 75015 Paris, France.

Younes Ouchrif (Y)

Institut de Biomecanique Humaine Georges Charpak, Arts et Metiers Sciences et Technologies, 151 Boulevard de l'Hôpital, 75013 Paris, France; APHP, Service d'orthopédie infantile, Hôpital Necker Enfants Malades, 75015 Paris, France.

Jugurtha Berkenou (J)

Centre de référence Maladies Rares du Métabolisme du Calcium et du Phosphore, 94270 Le Kremlin Bicetre, France.

Hélène Pillet (H)

Institut de Biomecanique Humaine Georges Charpak, Arts et Metiers Sciences et Technologies, 151 Boulevard de l'Hôpital, 75013 Paris, France.

Ayman Assi (A)

Laboratory of Biomechanics and Medical Imaging, Faculty of Medicine, Saint-Joseph University, Beirut, Lebanon.

Philippe Wicart (P)

Institut de Biomecanique Humaine Georges Charpak, Arts et Metiers Sciences et Technologies, 151 Boulevard de l'Hôpital, 75013 Paris, France; APHP, Service d'orthopédie infantile, Hôpital Necker Enfants Malades, 75015 Paris, France.

Wafa Skalli (W)

Institut de Biomecanique Humaine Georges Charpak, Arts et Metiers Sciences et Technologies, 151 Boulevard de l'Hôpital, 75013 Paris, France.

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Classifications MeSH