International audienceIdiopathic scoliosis is a spine disorder of unknown origin with 1.5-3% prevalence in the general population. Besides the large multifactorial form sample of idiopathic scoliosis, there is a good evidence for the existence of a monogenic subgroup in which the disease is inherited in a dominant manner. However, results from literature suggest a strong heterogeneity in the locations of the mutated genes. Using a high resolution genome-wide scan, we performed linkage analyses in three large multigenerational idiopathic scoliosis families compatible with dominant inheritance including 9 to 12 affected members or obligate carriers. In two of these families, our results suggested intra-familial genetic heterogeneity whereas, in the other, we observed a perfect marker disease co-segregation in two regions at 3q12.1 and 5q13.3. We can state that one of these two locations is a novel idiopathic scoliosis disease gene locus, since the probability of having this perfect co-segregation twice by chance in the genome is very low (p=0.001). Lastly, in all three families studied, linkage to the previously mapped dominant idiopathic scoliosis loci on chromosomes 19p13.3, 17p11.2, 9q34, 17q25 and 18q is unlikely, confirming that there is a high genetic heterogeneity within the subgroup of dominant forms of idiopathic scoliosis
Aim
To describe coping strategies in children and adolescents with cerebral palsy (CP), relative to age.
Method
Patients were prospectively recruited from two paediatric rehabilitation centres in France. The Pediatric Pain Coping Inventory – French and Structured Pain Questionnaire were completed by an experienced professional for each child.
Results
One hundred and forty‐two children with CP were included (80 males, 62 females; median age 12y; IQR=8–15y). They generally used fewer coping strategies than typically developing children (‘Seeks social support and action’: 12.47 vs 12.85, p=0.477; ‘Cognitive self‐instruction’: 9.28 vs 10.90, p<0.001; ‘Distraction’: 4.89 vs 7.00, p<0.001; ‘Problem solving’: 4.43 vs 5.19, p<0.001). In the CP group, ‘Seeks social support and action’ decreased with age (p=0.021) and ‘Cognitive self‐instruction’ increased with age (p<0.001). ‘Problem solving’ and ‘Distraction’ did not change with age. Coping strategies were influenced by Gross Motor Function Classification System level (p=0.022) and history of surgery (p=0.002).
Interpretation
Children with CP generally used fewer coping strategies than typically developing children and tended to rely on social support. Use of active strategies increased with age; however, they appeared later than in typically developing children and were used to a lesser extent.
What this paper adds
Children with cerebral palsy (CP) use fewer pain‐coping strategies than typically developing children.
Children with CP tend to use social support to cope with pain.
Children with CP learn more appropriate strategies from previous painful experiences.
Active coping strategies appear later but remain underused in children with CP.
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