More than a foot : gait, gross motor skills, neurodevelopmental difficulties and health-related quality of life in children with idiopathic clubfoot

Introduction: Idiopathic clubfoot (IC) is one of the most common congenital musculoskeletal deformities and can affect one or both feet of the child. However, knowledge beyond the structural deformity is still limited. This thesis aimed to evaluate unexplored fields by including measures that might affect the children’s overall health. This included evaluation of gait, gross motor skills, neurodevelopmental difficulties (NDD), and health-related quality of life (HRQoL). Methods and participants: An explorative cross-sectional research approach of four studies divided into two parts was used. In part one, gait and gross motor skills were evaluated in 59 and 47 children with IC, respectively, born in 2005–2008 and treated in Stockholm County (mean age 5.4 ± 0.5 years). Gait was evaluated using three-dimensional gait analysis, and gross motor skills were evaluated using the Clubfoot Assessment Protocol. For comparison, 28 typically developing (TD) children were recruited (mean age 5.5 ± 0.6 years). In part two, NDD and HRQoL were surveyed in children with IC born in 2004–2007 in Stockholm and Skåne counties. Parents to 106 children with IC (mean age 9.4 ± 0.6 years) answered the Five to Fifteen questionnaire and Roye ́s Disease-specific instrument to operationalise NDD and satisfaction of the clubfoot treatment outcome, respectively. The children answered the generic HRQoL questionnaire EQ-5D-Y. A general population sample of 109 community schoolchildren (mean age 9.5 ± 0.6 years) was used for comparison. Results: Children with IC were found to have significant gait deviations, gross motor deficits and asymmetries at five years of age. No differences were found in bilateral and unilateral IC with the main concerns regarding decreased dorsi-plantar flexion range and ankle power generation in gait in both groups, compared with the TD children. In terms of gross motor skills, one-leg hop and stand deviated in 85 and 87% of the children, respectively. Associations between gross motor skills and passive range of motion of the foot, gait, and initial clubfoot severity were generally poor. The contralateral leg in children with unilateral IC showed no discrete gait or gross motor deficits; however, global gait measures revealed gait modifications. In nine-year-old children with IC, significantly more NDD were found in comparison with the general population, and group differences were evident in the domains of motor skills, perception, and language and in the subdomains of gross and fine motor skills, relation in space, comprehensive, and expressive language skills. Thirty-one per cent of the children in the IC sample were defined as experiencing NDD of clinical relevance. In this subgroup, parents reported significantly lower satisfaction of the clubfoot treatment outcome. In the overall IC sample of nine-year-old children, the children reported a satisfying overall health status despite more HRQoL problems, mainly regarding pain and discomfort. Neither being born with one or two affected feet nor sex affected NDD or HRQoL, however NDD negatively affected HRQoL. Conclusions: Children with bilateral and unilateral IC have similar marked gait and gross motor deficits on the affected side, thus indicating similar clubfoot severity and development. Nonetheless, awareness of foot involvement is important because of pronounced asymmetries in children with unilateral IC. Gross motor skills are not related to several foot measures and should be considered a different outcome entity in the evaluation of IC. Therefore, gross motor assessment as well as pain management should be emphasised in the follow-up of children with IC. Finally, health care providers should be attentive to NDD in children with IC appearing more often than would be expected by chance, especially since NDD negatively affect clubfoot treatment outcome and HRQoL. LIST OF SCIENTIFIC PAPERS I. Lööf E, Andriesse H, André M, Böhm S, Broström EW. Gait in 5-yearold children with idiopathic clubfoot: A cohort study of 59 children, focusing on foot involvement and the contralateral foot. Acta Orthopaedica. 2016, 87: 52228 II. Lööf E, Andriesse H, André M, Böhm S, Iversen MD, Broström EW. Gross motor skills in children with idiopathic clubfoot and the association between gross motor skills, foot involvement, gait and foot motion. Journal of Pediatric Orthopaedics. 2017, Feb 24. doi: 10.1097/BPO.0000000000000964 III. Lööf E, Andriesse H, Broström EW, André M, Bölte S. Neurodevelopmental difficulties in children with idiopathic clubfoot. Developmental Medicine & Child Neurology. 2019, 61: 98-104 IV. Lööf E, Andriesse H, Broström EW, André M, Böhm S, Bölte S. Neurodevelopmental difficulties negatively affect health-related quality of life in children with idiopathic clubfoot. Acta Paediatrica. 2018, Dec 27. doi: 10.1111/apa.14709