PDF herunterladen
J Pediatr Genet 2020; 09(04): 235-242
DOI: 10.1055/s-0040-1712178
Original Article
Georg Thieme Verlag KG Stuttgart · New York

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

Athena Ganetsos
1   Division of Medical Genetics, Department of Pediatrics, Stanford University, Palo Alto, California, United States
,
Ellyn Farrelly
1   Division of Medical Genetics, Department of Pediatrics, Stanford University, Palo Alto, California, United States
,
Pilar Magoulas
2   Department of Molecular and Human Genetics, Baylor College of Medicine, Texas Children's Hospital, Houston, Texas, United States
,
David A. Stevenson 
1   Division of Medical Genetics, Department of Pediatrics, Stanford University, Palo Alto, California, United States
› Institutsangaben
Weitere Informationen

Publikationsverlauf

14. Februar 2020

20. April 2020

Publikationsdatum:
22. Mai 2020 (online)

   Lizenzen und Reprints

Abstract

The study aimed to assess baseline stress and coping mechanisms among caregivers of children with RASopathies (i.e., cardiofaciocutaneous and Costello's syndrome) and the impact of attending biennial caregiver conferences. Caregivers completed the Perceived Stress Scale, Coping Health Inventory for Parents, and demographic surveys prior to family conferences, and 1- and 6-month postconferences. Baseline stress was increased and associated with child age, parental age, and number of conferences attended. After 1 month, caregiver stress was lowered among men and caregivers attending ≥2 support conferences.

Keywords

Costello syndrome - cardiofaciocutaneous syndrome - RASopathies
 

  • References

  • 1 Tajan M, Paccoud R, Branka S, Edouard T, Yart A. The RASopathy family: consequences of germline activation of the RAS/MAPK pathway. Endocr Rev 2018; 39 (05) 676-700
    CrossrefPubMedGoogle Scholar
  • 2 Alfieri P, Piccini G, Caciolo C. , et al. Behavioral profile in RASopathies. Am J Med Genet A 2014; 164A (04) 934-942
    PubMedGoogle Scholar
  • 3 Baker BL, Blacher J, Olsson MB. Preschool children with and without developmental delay: behaviour problems, parents' optimism and well-being. J Intellect Disabil Res 2005; 49 (Pt 8): 575-590
    CrossrefPubMedGoogle Scholar
  • 4 Singer GH. Meta-analysis of comparative studies of depression in mothers of children with and without developmental disabilities. Am J Ment Retard 2006; 111 (03) 155-169
    CrossrefPubMedGoogle Scholar
  • 5 Estes A, Munson J, Dawson G, Koehler E, Zhou XH, Abbott R. Parenting stress and psychological functioning among mothers of preschool children with autism and developmental delay. Autism 2009; 13 (04) 375-387
    CrossrefPubMedGoogle Scholar
  • 6 Pierpont EI, Wolford M. Behavioral functioning in cardiofaciocutaneous syndrome: Risk factors and impact on parenting experience. Am J Med Genet A 2016; 170 (08) 1974-1988
    CrossrefPubMedGoogle Scholar
  • 7 Neece CL. Mindfulness-based stress reduction for parents of young children with developmental delays: implications for parental mental health and child behavior problems. J Appl Res Intellect Disabil 2014; 27 (02) 174-186
    CrossrefPubMedGoogle Scholar
  • 8 Tvrdik T, Mason D, Dent KM. , et al. Stress and coping in parents of children with Prader-Willi syndrome: Assessment of the impact of a structured plan of care. Am J Med Genet A 2015; 167A (05) 974-982
    PubMedGoogle Scholar
  • 9 Cohen S, Kamarck T, Mermelstein R. A global measure of perceived stress. J Health Soc Behav 1983; 24 (04) 385-396
    CrossrefPubMedGoogle Scholar
  • 10 Cohen S, Williamson G. Perceived stress in a probability sample of the United States. In: Spacapan S, Oskamp S. , eds. The Social Psychology of Health. Newbury Park, CA: Sage; 1988
    Google Scholar
  • 11 McCubin I, McCubin M, Patterson J, Cauble A, Wilson L, Warwick W. CHIP-Coping Health Inventory for Parents: an assessment of parental coping patterns in the care of the chronically ill child. J Marriage Fam 1983; 45: 359-370
    CrossrefPubMedGoogle Scholar
  • 12 Adviento B, Corbin IL, Widjaja F. , et al. Autism traits in the RASopathies. J Med Genet 2014; 51 (01) 10-20
    CrossrefPubMedGoogle Scholar
  • 13 Garg S, Brooks A, Burns A. , et al. Autism spectrum disorder and other neurobehavioural comorbidities in rare disorders of the Ras/MAPK pathway. Dev Med Child Neurol 2017; 59 (05) 544-549
    CrossrefPubMedGoogle Scholar
  • 14 Bizaoui V, Gage J, Brar R, Rauen KA, Weiss LA. RASopathies are associated with a distinct personality profile. Am J Med Genet B Neuropsychiatr Genet 2018; 177 (04) 434-446
    CrossrefPubMedGoogle Scholar
  • 15 Galéra C, Delrue MA, Goizet C. , et al. Behavioral and temperamental features of children with Costello syndrome. Am J Med Genet A 2006; 140 (09) 968-974
    PubMedGoogle Scholar
  • 16 Axelrad ME, Schwartz DD, Katzenstein JM, Hopkins E, Gripp KW. Neurocognitive, adaptive, and behavioral functioning of individuals with Costello syndrome: a review. Am J Med Genet C Semin Med Genet 2011; 157C (02) 115-122
    PubMedGoogle Scholar
  • 17 Bouma R, Schweitzer R. The impact of chronic childhood illness on family stress: a comparison between autism and cystic fibrosis. J Clin Psychol 1990; 46 (06) 722-730
    CrossrefPubMedGoogle Scholar
  • 18 Randall P, Parker J. Supporting the Families of Children with Autism. Chichester: Wiley; 1999
    Google Scholar
  • 19 McKinney B, Peterson RA. Predictors of stress in parents of developmentally disabled children. J Pediatr Psychol 1987; 12 (01) 133-150
    CrossrefPubMedGoogle Scholar
  • 20 Weiss MJ. Hardiness and social support as predictors of stress in mothers of typical children, children with autism, and children with mental retardation. Autism 2002; 6 (01) 115-130
    CrossrefPubMedGoogle Scholar
  • 21 Sakkalou E, Sakki H, O'reilly MA, Salt AT, Dale NJ. Parenting stress, anxiety, and depression in mothers with visually impaired infants: a cross-sectional and longitudinal cohort analysis. Dev Med Child Neurol 2018; 60 (03) 290-298
    CrossrefPubMedGoogle Scholar
  • 22 Cristancho-Lacroix V, Wrobel J, Cantegreil-Kallen I, Dub T, Rouquette A, Rigaud AS. A web-based psychoeducational program for informal caregivers of patients with Alzheimer's disease: a pilot randomized controlled trial. J Med Internet Res 2015; 17 (05) e117
    CrossrefPubMedGoogle Scholar
  • 23 Thanou E, Tsiou C, Kattami C, Chrousos GP, Darviri C. A stress management and health promotion intervention for parents of children with cerebral palsy and motor disability at the Infant Department of the Greek Center for the Protection and Rehabilitation of the Disabled (ELEPAP). Quasi-experimental study. Psychology (Irvine) 2016; 7: 557-564
    PubMedGoogle Scholar
  • 24 Hassall R, Rose J, McDonald J. Parenting stress in mothers of children with an intellectual disability: the effects of parental cognitions in relation to child characteristics and family support. J Intellect Disabil Res 2005; 49 (Pt 6): 405-418
    CrossrefPubMedGoogle Scholar
  • 25 Dellve L, Samuelsson L, Tallborn A, Fasth A, Hallberg LR. Stress and well-being among parents of children with rare diseases: a prospective intervention study. J Adv Nurs 2006; 53 (04) 392-402
    CrossrefPubMedGoogle Scholar
  • 26 Hobdell EF, Grant ML, Valencia I. , et al. Chronic sorrow and coping in families of children with epilepsy. J Neurosci Nurs 2007; 39 (02) 76-82
    CrossrefPubMedGoogle Scholar
  • 27 Cavallo S, Feldman DE, Swaine B, Meshefedjian G, Malleson PN, Duffy CM. Is parental coping associated with quality of life in juvenile idiopathic arthritis?. Pediatr Rheumatol Online J 2009; 7: 7
    CrossrefPubMedGoogle Scholar
  • 28 Gripp KW, Morse LA, Axelrad M. , et al. Costello syndrome: clinical phenotype, genotype, and management guidelines. Am J Med Genet A 2019; 179 (09) 1725-1744
    CrossrefPubMedGoogle Scholar