Is Motor Performance in 5.5-Year-Old Children Associated with the Presence of Generalized Joint Hypermobility? Rosa M. de Boer, PPT, MSc 1 , Leo A. van Vlimmeren, PPT, PhD 2,3 , Mark C. Scheper, PT, MSc 4,5 , Maria W. G. Nijhuis-van der Sanden, PPT, PhD 2,3 , and Raoul H. H. Engelbert, PPT, PhD 4,5 Objective To determine the prevalence of generalized joint hypermobility (GJH) in Dutch children aged 5.5 years, and to examine the association between GJH and motor performance and development over time. Study design A prospective cohort of 249 children was recruited. GJH was assessed with the Beighton test at age 5.5 years. Motor performance was evaluated at age 2.0 years using the Bayley Scales of Infant Development, Second Edition and at age 5.5 years using the Movement Assessment Battery for Children–Second Edition (subscore categories: manual dexterity, aiming and catching, and static and dynamic balance). Results In 249 children, the prevalence of GJH, defined by the Beighton test score, was 34.1% for a score $4, 22.5% for a score $5, and 16.5% for a score $6. No significant association was found between GJH and total motor performance. Manual dexterity in girls (Beighton score $4) was positively associated with higher level of motor performance (b [SE] = 0.38 [0.17]; P = .028), ranging from +0.04 SD to +0.72 SD, even after correction for covariates. A significant interaction between GJH and body mass index (BMI) growth was found, indicating that the effect of GJH on the rate of development of motor performance declines with increasing BMI growth (b = 0.05 [0.02]; P = .031). Conclusion In this healthy pediatric cohort, GJH was present in one-third of the sample, and no significant association was found between GJH and total motor performance. The effect of GJH on the rate of development of motor performance appears to decline with increasing BMI growth. Longitudinal prospective studies are recommended to detect influences of GJH on motor performance over time, as well as the influence of body composition and Beighton cutoff points. (J Pediatr 2015;-:---). G eneralized joint hypermobility (GJH) is common in children. When arthralgia in more than 4 joints is present for longer than 3 months without any signs of rheumatic, neurologic, skeletal, or metabolic disease, hypermobility syndrome (HMS) can be diagnosed. 1 The presence of GJH in children is commonly detected using the Beighton test. This measure is considered the gold standard from infancy to old age. 2-5 Use of the Beighton test is controversial, however, owing to a lack of standardization in children, as well as a lack of age-, sex-, and ethnicity-specific cutoff values. 2,4,6 The prevalence of GJH is unclear, with extensive variation reported in the relevant literature. This could be explained by the absence of an international consensus and the lack of operationalization standards, which vary according to differences in study populations, cutoff levels, and actual administration of the Beighton test. 2,6-8 Murray et al 9 reported GJH prevalence ranging between 2% and 55% in various pediatric populations. Recent studies have examined the heterogeneity of GJH in the Caucasian population by comparing the prevalence of GJH at varying cutoff levels and taking age and sex into consideration. 2,4,6,10,11 Differences in cutoff levels also might be explained by the more precise and detailed descriptions of standard operating procedures used for measuring local joint hypermobility and GJH, as first described by Juul-Kristensen et al. 3 The concurrent validity of the Beighton test in relation to goniometry has been found to be high. 4 Regarding predictive value, one study found that 10-year-old children diagnosed with GJH and musculoskeletal pain using the Beighton test had an increased risk of persistent pain at age 14 years. 12 Despite the prevalence of GJH in normal populations, 13,14 much remains unknown about its consequences. GJH has been associated with a wide variety of musculoskeletal complaints, including joint pain, dysfunction of various organ systems (eg, blood vessels and skin), and psychosocial problems. 2,6,15-18 GJH is associated with an increased incidence of motor delay in infancy, with From the 1 Primary Practice for Pediatric Physiotherapy, Heerhugowaard; 2 Department of Rehabilitation, Pediatric Physical Therapy, Radboud University Medical Center; 3 Scientific Institute for Quality of Healthcare, Radboud University Medical Center, Nijmegen; 4 Education of Physiotherapy, University of Applied Sciences Amsterdam; and 5 Department of Rehabilitation, University Hospital Amsterdam (AMC), Amsterdam, The Netherlands Supported by Scientific Committee of the Royal Dutch Society of Physiotherapy (BU002/10). The authors declare no conflicts of interest. 0022-3476/$ - see front matter. Copyright ª 2015 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jpeds.2015.06.034 AICC Akaike information criterion BMI Body mass index BSID-II Bayley Scales of Infant Development, Second Edition GJH Generalized joint hypermobility HMS Hypermobility syndrome MD Mean difference Movement ABC-2 Movement Assessment Battery for Children–Second Edition 1