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. 2025 Mar;67(3):307-321.
doi: 10.1111/dmcn.16124. Epub 2024 Oct 21.

Incidence of hip problems in developmental central hypotonia: A scoping review

Roslyn W Livingstone  1 , Ginny S Paleg  2 , M Wade Shrader  3 , Freeman Miller  3 , Elisabet Rodby-Bousquet  4   5
Affiliations

Incidence of hip problems in developmental central hypotonia: A scoping review

Roslyn W Livingstone et al. Dev Med Child Neurol. 2025 Mar.

Abstract

Aim: To describe what is known about hip problems in individuals with developmental central hypotonia.

Method: Searches were conducted in five databases to October 2023. Down syndrome was excluded from this analysis of less well-known genetic diagnoses. At least two reviewers independently screened titles, abstracts, read full-text articles, and extracted data.

Results: Of 89 full-text articles, 79 met inclusion criteria. Studies included 544 individuals aged 1 month to 63 years with Kabuki, 49, XXXXY, Prader-Willi, PURA, Koolen de Vries, Emanuel, TRPM3, Wolf-Hirschhorn, and other rare syndromes. Most diagnoses may be associated with a combination of differences in hip structure or stability that are evident at birth, or develop in early infancy, with increasing hip dysplasia and subluxation over time. Joint or ligamentous laxity was most reported along with hypotonia and hypermobility as risk factors. Limited data were identified about conservative or surgical intervention and outcomes in these populations.

Interpretation: Children with significant hypotonia, with or without a confirmed genetic diagnosis, are at increased risk of hip problems that may be missed with standard neonatal screening. Ultrasound is recommended between 6 weeks and 6 months, and annual orthopaedic review with regular radiographs for older children and adults with significant and persistent hypotonia.

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Conflict of interest statement

Ginny S. Paleg has worked as an educational consultant for Prime Engineering, a manufacturer of supported standing and stepping devices. She is not an employee, and owns no stock. Prime Engineering provided no funding for this study, and had no influence on study conduct or reporting. Roslyn W. Livingstone, M. Wade Shrader, Freeman Miller and Elisabet Rodby‐Bousquet have no interests which might be perceived as posing a conflict or bias.

Figures

FIGURE 1
FIGURE 1
Map illustrating origins of studies included in this review.
FIGURE 2
FIGURE 2
Tree chart illustrating relative proportions of cases reported for each diagnosis in included studies.
FIGURE 3
FIGURE 3
(a) Terms used to indicate timing of hip problems in included studies. (b) Age at onset reported in individual cases. (c) Hip problems reported across all included studies. (d) Risk factors for hip problems reported.
FIGURE 4
FIGURE 4
(a) Hip problems reported from different geographical regions. Bar length indicates percentage of reports for that region while numbers indicate actual number of reports. (b) Hip problems reported over time. Bar height indicates percentage of reports for that period while numbers indicate actual numbers of reports. (c) Interventions reported for individual cases reported in studies. (c) Comparison of successful versus unsuccessful (required further surgical intervention) femoral/acetabular osteotomies, or more conservative treatment (includes splinting, casting, open/closed reduction, traction, and/or soft‐tissue releases).
FIGURE 5
FIGURE 5
(a) Onset reported across diagnoses, by percentage for each diagnosis and by numbers of cases. (b) Hip problems reported across diagnoses, by percentage of reports for each diagnosis and actual numbers of reports.
See this image and copyright information in PMC

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