Pediatric neurofibromatosis 1 and parental stress: a multicenter study.
BACKGROUND
Neurofibromatosis 1 (NF1) is a complex and multifaceted neurocutaneous syndrome with many and varied comorbidities. The literature about the prevalence and degree of maternal stress and the impact of NF1 in the parent-child interaction is still scant. The aim of this study was to evaluate the prevalence of maternal stress in a large pediatric sample of individuals affected by NF1.


METHODS
Thirty-seven children (19 boys, 18 girls) of mean age 7.86±2.94 (range 5-11) years affected by typical NF1 and a control group comprising 405 typically developing children (207 boys, 198 girls; mean age 8.54±2.47 years) were included in this study. To assess parental stress, the mothers of all individuals (NF1 and comparisons) filled out the Parenting Stress Index-Short Form test.


RESULTS
The two study groups were comparable for age (P=0.116), gender (P=0.886), and body mass index adjusted for age (P=0.305). Mothers of children affected by NF1 reported higher mean Parenting Stress Index-Short Form scores on the Parental Distress domain (P<0.001), Difficult Child domain (P<0.001), and Total Stress domain than the mothers of typically developing children (controls) (P<0.001). No significant differences between the two groups were found for the Parent-Child Dysfunctional Interaction domain (P=0.566) or Defensive Responding domain scores (P=0.160).


CONCLUSIONS
NF1 is considered a multisystemic and complex disease, with many still unrecognized features in pediatric patients and in their families. In this light, our findings about the higher levels of maternal stress highlight the importance of considering the environmental aspects of NF1 management in developmental age.
Clinic of Child and Adolescent Neuropsychiatry, Department of Mental Health, Physical and Preventive Medicine, Second University of Naples, Naples, Italy.
Maria
M
Esposito
Department of Psychiatry, "Magna Graecia" University of Catanzaro, Catanzaro, Italy.
Rosa
R
Marotta
Child Neuropsychiatry, Department of Psychology, University of Palermo, Palermo, Italy.
Michele
M
Roccella
Unit of Child and Adolescent Neuropsychiatry, University of Perugia, Perugia, Italy.
Beatrice
B
Gallai
Child Neuropsychiatry, Department of Psychology, University of Palermo, Palermo, Italy.
Lucia
L
Parisi
Department of Psychiatry, "Magna Graecia" University of Catanzaro, Catanzaro, Italy.
Serena Marianna
SM
Lavano
Clinic of Child and Adolescent Neuropsychiatry, Department of Mental Health, Physical and Preventive Medicine, Second University of Naples, Naples, Italy.
Marco
M
Carotenuto
Parental Stress Index; children; maternal stress; von Recklingausen disease