1. Stress in Parents of Children With Genetically Determined Leukoencephalopathies: A Pilot Study
- Author
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Aaron Spahr, E. Malvey-Dorn, S. Chandratre, F Pelletier, B. Osterman, M. Desmeules, G. Roedde, Marie Emmanuelle Dilenge, S. Chenier, Albert Larbrisseau, P. Marois, A Mirchi, Luan Tran, Elsa Rossignol, K. Y. Lim, Nancy Braverman, E. Dermer, Michael Shevell, J. Reggin, Mark A. Tarnopolsky, Guillaume Sébire, J. Laflamme, Kether Guerrero, Catalina Maftei, Geneviève Legault, Soad M. Ahmed, Sunita Venkateswaran, Daniela Pohl, Daniela Buhas, Philippe Major, I. Paradis, John J. Mitchell, Geneviève Bernard, M. Sullivan, Bernard Brais, Nicolas Chrestian, Myriam Srour, Michel Sylvain, EM Riou, and A. Nadeau
- Subjects
Adult ,Male ,Parents ,0301 basic medicine ,Pediatrics ,medicine.medical_specialty ,Pilot Projects ,Early death ,030105 genetics & heredity ,White matter ,03 medical and health sciences ,0302 clinical medicine ,Quality of life ,Leukoencephalopathies ,Surveys and Questionnaires ,Stress (linguistics) ,Humans ,Medicine ,Child ,business.industry ,Leukodystrophy ,Infant ,medicine.disease ,Cross-Sectional Studies ,medicine.anatomical_structure ,Child, Preschool ,Pediatrics, Perinatology and Child Health ,Female ,Neurology (clinical) ,Parental stress ,business ,Stress, Psychological ,030217 neurology & neurosurgery - Abstract
Genetically determined leukoencephalopathies comprise a group of rare inherited white matter disorders. The majority are progressive diseases resulting in early death. We performed a cross-sectional pilot study including 55 parents from 36 families to assess the level of stress experienced by parents of patients with genetically determined leukoencephalopathies, aged 1 month to 12 years. Thirty-four mothers and 21 fathers completed the Parenting Stress Index–4th Edition. One demographic questionnaire was completed per family. Detailed clinical data was gathered on all patients. Statistical analysis was performed with total stress percentile score as the primary outcome. Mothers and fathers had significantly higher stress levels compared with the normative sample; 20% of parents had high levels of stress whereas 11% had clinically significant levels of stress. Mothers and fathers had comparable total stress percentile scores. We identified pediatric behavioral difficulties and gross motor function to be factors influencing stress in mothers. Our study is the first to examine parental stress in this population and highlights the need for parental support early in the disease course. In this pilot study, we demonstrated that using the Parenting Stress Index–4th Edition to assess stress levels in parents of patients with genetically determined leukoencephalopathies is feasible, leads to valuable and actionable results, and should be used in larger, prospective studies.
- Published
- 2020