Stress and coping in parents of children with Prader‐Willi syndrome: Assessment of the impact of a structured plan of care

Abstract

Hyperphagia, developmental delays, and maladaptive behaviors are common in Prader-Willi syndrome (PWS) likely resulting in heightened parental stress. Objectives were to evaluate stress, describe usefulness of coping behaviors, and assess the impact of a structured Plan of Care (PC) on parents with children with PWS. Parents answered Perceived Stress Scale (PSS-14), Coping Health Inventory for Parents (CHIP), and narrative/demographic surveys. The PC was introduced to a cohort of parents after completion of the PSS-14 and CHIP and re-administered 4-6 month after the introduction of the PC. Higher parental stress (n = 57) was observed compared to the general population, and associated with parent's age, number of children living at home, and child's age and residential setting. "Maintaining family integration, cooperation, and an optimistic definition of the situation" was the most useful coping pattern. Thirty-eight parents answered the PSS-14 and CHIP after the PC. Parental stress decreased after the PC (P = 0.035). Coping behaviors related to "maintaining family integration" increased after the PC (P = 0.042). Women and men preferred different coping patterns before and after the PC. In conclusion, parental stress is increased in PWS, and a PC decreased stress and increased coping behaviors related to family stability for parents with children with PWS.

Impacts of caring for a child with the CDKL5 disorder on parental wellbeing and family quality of life

“…Although there is an increasing number of studies examining maternal health [9, 10], research specifically targeting families caring for a child with a genetic disorder remains relatively scarce. While impaired mental health has been reported for those with a child with Down syndrome [11] and RTT [12, 13], and increased stress for those with a child with Prader-Willi syndrome [14], there are no data on the health of parents caring for a child with the CDKL5 disorder.…”

Stress and Coping in Caregivers of Children with RASopathies: Assessment of the Impact of Caregiver Conferences

“…Baseline stress scores in our cohort were higher than those reported for caregivers of individuals with Alzheimer's disease, 22 but lower than those reported for caregivers of children with cerebral palsy 23 and Prader-Willi syndrome. 8 There are likely multiple factors that contribute to the variability of stress levels among caregivers of different disorders or even the same disorder. Hassall et al studied 46 mothers of children with intellectual impairment and their results supported their hypotheses that parenting stress was higher when behavioral difficulties of the child were more severe and when the mothers displayed a more external locus of control for parenting.…”

“…Numerous studies have utilized the CHIP instrument to evaluate coping behaviors of caregivers of children with chronic disease. 8,11,26,27 A higher percentage of the maximum score for each subscale indicates a higher use of that coping mechanism. In previous studies of caregivers of individuals with other chronic illnesses including epilepsy, juvenile idiopathic arthritis, Prader-Willi syndrome, and various physical disabilities maximum percentage scores have ranged from 66 to 77% for the CHIP I, 45 to 74% for the CHIP II, and 61 to 69% for the CHIP III.…”

“…[3][4][5][6] There have also been several studies evaluating interventions aimed to improve stress levels and coping behaviors among caregivers of children with many different neurodevelopmental disorders. 7,8 However, there is limited information in caregivers of individuals with RASopathies.…”

Brazilian study of adaptation and psychometric properties of the Coping Health Inventory for Parents

“…The CHIP has been administered in various international studies in order to measure coping patterns of parents of individuals with different diagnoses and health conditions, such as cancer, Prader-Willi syndrome, autism spectrum disorders, and asthma (Garro 2011;Lakkis et al 2015;Tvrdik et al 2015). Considering the paucity of instruments for investigating parental coping specifically in the area of health care in Brazil and the importance of understanding the means by which such strategies aid the process of the parents' adaptation to the child's chronic or acute illness, the present study sought to translate and culturally adapt the CHIP for a Brazilian sample, as well as to investigate the preliminary psychometric properties of the scale.…”