Exploring pain interference and self-perceived health status in children with osteogenesis imperfecta - a cross-sectional study

Abstract

Background Chronic pain may affect and interfere in children’s everyday life and can be present in children with Osteogenesis Imperfecta (OI). However, the knowledge is still sparse to what extent pain is present, how pain interfere in children’s everyday life and affect their self-perceived health status. The purpose of the study was therefore to explore presence of chronic pain, pain interference in daily life, and self-perceived health status in children with OI. Methods Children with OI, aged 6–18 years, were recruited consecutively to this cross-sectional study. Participants answered a standardised interview including five pre-structured questions, and the Numeric Pain Rating Scale (NPRS), the Pain Interference Index, and a questionnaire concerning self-perceived health status the Patient Reported Outcomes Measurement Information System Pediatric-25 Profile v1.1 (PROMIS-25). Results Twenty-eight children (median: 11 years, IQR 6) with OI type I, III, or IV participated. Pain was present in 27 of 28 children and interfered in their everyday life regardless of OI-type, sex, and age. The median NPRS for average pain intensity was 4 (IQR 2), the median for pain frequency was 2–3 times/week, and the median frequency of school absence due to pain was 2–3 times per month. The most common pain locations were back and feet. Pain in the feet was more frequently reported in children with type I (p = 0.032), and pain in the hip was more often reported in children ≥13 years (p = 0.011). The children were asked what they thought to be the cause of pain and the most frequent response was “walking long distances”. Self-perceived health status for mobility was lower than the general population, and lowest for children with type III (p = 0.016). Pain interference was associated with children’s self-perceived health status (rs = 0.84, p < 0.001). Conclusion Almost all children experienced pain, which interfered in children’s everyday lives, affected participation in various activities and was associated with reduced self-perceived health status. If children avoid physical activities because of pain, it might cause a vicious circle of inactivity, which further decreases bone density and increase the risk of fractures. The results emphasize the importance to offer adequate pain reducing interventions.

The IMPACT survey: a mixed methods study to understand the experience of children, adolescents and adults with osteogenesis imperfecta and their caregivers

“…This study found that, irrespective of age, individuals with OI experience numerous signs, symptoms and events that affect their HRQoL. Pain was highly prevalent and impactful on the HRQoL of individuals with OI, as reported in past research [ 20 , 31 – 33 ]. Notably, fatigue, which has rarely been explored in children, was prevalent in children in this sample, but much less so than adults or adolescents.…”

The impact of foot orthoses on gait in children with Osteogenesis Imperfecta type I, III and IV – a cross-sectional study

“…As outlined throughout this study, foot deformity is not the sole negative influence on gait in children with OI, as the condition also involves muscle weakness, joint hypermobility, and malalignment of the of the skeleton [ 1 , 40 , 42 , 43 ]. In a recent cross-sectional study exploring pain and pain interference in children with OI, with a partly overlapping cohort as the present study ( n = 5), the most common cause of non-fracture pain explained by children was “walking for longer distances” [ 20 ]. In a retrospective cohort study, Adib et al defined clinical characteristics of 124 children (median age of 12 years) with joint hypermobility-related presentations [ 45 ].…”

“…In children with juvenile idiopathic arthritis, foot orthoses, as compared to shoes only, have been reported to reduce pain and improve function [ 18 ], and redrestribute high pressure areas of the hind-foot and forefoot [ 19 ]. A recent review, evaluating foot orthoses for treating pediatric flat feet, concluded that attention should be directed towards pediatric foot conditions causing pain, limited function, or reduced quality of life [ 18 ], all of which are common difficulties for children with OI [ 7 , 8 , 20 ].…”

Formulation and Evaluation of Diclofenac Potassium Gel in Sports Injuries with and without Phonophoresis

“…NPRS [ 52 ] is often made up of a sequence of numbers and oral anchors that indicate the full range of pain intensity ( Figure 10 ). Patients usually rate their pain on a scale of 0 to 10, 0 to 20, or 0 to 100.…”