Skip to main content
SpringerLink
Log in

Psychological, cognitive and maternal stress assessment in children with primary ciliary dyskinesia

  • Original article
  • Published:
World Journal of Pediatrics Aims and scope Submit manuscript

Abstract

Background

Primary ciliary dyskinesia (PCD) is a rare disorder due to structure and functional abnormalities of respiratory cilia. There are no reports on the behavioral and psychological aspects of children and adolescents with PCD. This study was undertaken to assess the cognitive and behavioural characteristics, and the parental stress of a population of school-aged children with PCD.

Methods

Ten PCD and 34 healthy school-aged children underwent Wechsler Intelligence Scale for Children-III edition, Child Behavior Check-List questionnaire (CBCL), Parenting Stress Index-Short Form tests in order to perform a behavioural and psychological evaluation.

Results

PCD children showed significant behavioral and social competent problems in CBCL scale than control children, in particular with regard to internalizing problems score (P<0.001). Parental distress, parent-child interaction and total stress in the mothers of PCD patients were higher than those in the controls’ parents (P<0.001).

Conclusion

Our findings pinpoint the importance of specific psychological support in the clinical management of children with PCD.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Similar content being viewed by others

References

  1. Barbato A, Frischer T, Kuehni CE, Snijders D, Azevedo I, Baktai G, et al. Primary ciliary dyskinesia: a consensus statement on diagnostic and treatment approaches in children. Eur Respir J 2009;34:1264–1276.

    Article  PubMed  CAS  Google Scholar 

  2. Afzelius BA. A human syndrome caused by immotile cilia. Science 1976;193:317–319.

    Article  PubMed  CAS  Google Scholar 

  3. El Zein L, Omran H, Bouvagnet P. Lateralization defects and ciliary dyskinesia: lessons from algae. Trends Genet 2003;19:162–167.

    Article  PubMed  Google Scholar 

  4. McManus IC, Mitchison HM, Chung EM, Stubbings GF, Martin N. Primary ciliary dyskinesia (Siewert’s/Kartagener’s syndrome): respiratory symptoms and psycho-social impact. BMC Pulm Med 2003;3:4.

    Article  PubMed  Google Scholar 

  5. Pifferi M, Bush A, Di Cicco M, Pradal U, Ragazzo V, Macchia P, et al. Health-related quality of life and unmet needs in patients with primary ciliary dyskinesia. Eur Respir J 2010;35:787–794.

    Article  PubMed  CAS  Google Scholar 

  6. Veale BM. Meeting the challenge of chronic illness in general practice. Med J Aust 2003;179:247–249.

    PubMed  Google Scholar 

  7. Glasscoe CA, Quittner AL. Psychological interventions for people with cystic fibrosis and their families. Cochrane Database Syst Rev 2008:16:CD003148.

    Google Scholar 

  8. Esposito M, Gallai B, Parisi L, Roccella M, Marotta R, Lavano SM, et al. Maternal stress and childhood migraine: a new perspective on management. Neuropsychiatr Dis Treat 2013;9:351–355.

    Article  PubMed  Google Scholar 

  9. Pfeffer PE, Pfeffer JM, Hodson ME. The psychosocial and psychiatric side of cystic fibrosis in adolescents and adults. J Cyst Fibros 2003;2:61–68.

    Article  PubMed  CAS  Google Scholar 

  10. Eddy ME, Carter BD, Kronenberger WG, Conradsen S, Eid NS, Bourland SL, et al. Parent relationships and compliance in cystic fibrosis. J Pediatr Health Care 1998;12:196–202.

    Article  PubMed  CAS  Google Scholar 

  11. Spieth LE, Stark LJ, Mitchell MJ, Schiller M, Cohen LL, Mulvihill M, et al. Observational assessment of family functioning at mealtime in preschool children with cystic fibrosis. J Pediatr Psychol 2001;26:215–224.

    Article  PubMed  CAS  Google Scholar 

  12. Koscik RL, Farrell PM, Kosorok MR, Zaremba KM, Laxova A, Lai HC, et al. Cognitive function of children with cystic fibrosis: deleterious effect of early malnutrition. Pediatrics 2004;113:1549–1558.

    Article  PubMed  Google Scholar 

  13. Koinis-Mitchell D, McQuaid EL, Seifer R, Kopel SJ, Nassau JH, Klein RB, et al. Symptom perception in children with asthma: cognitive and psychological factors. Health Psychol 2009;28:226–237.

    Article  PubMed  Google Scholar 

  14. McCrimmon RJ, Ryan CM, Frier BM. Diabetes and cognitive dysfunction. Lancet 2012;16:379:2291–2299.

    Article  Google Scholar 

  15. Esposito M, Pascotto A, Gallai B, Parisi L, Roccella M, Marotta R, et al. Can headache impair intellectual abilities in children? An observational study. Neuropsychiatr Dis Treat 2012;8:509–513.

    Article  PubMed  Google Scholar 

  16. Montella S, Alving K, Maniscalco M, Sofia M, De Stefano S, Raia V, et al. Measurement of nasal nitric oxide by hand-held and stationary devices. Eur J Clin Invest 2011;41:1063–1070.

    Article  PubMed  CAS  Google Scholar 

  17. Santamaria F, De Stefano S, Montella S, Barbarano F, Iacotucci P, Ciccarelli R, et al. Nasal nitric oxide assessment in primary ciliary dyskinesia using aspiration, exhalation, and humming. Med Sci Monit 2008;14:CR80–85.

    PubMed  CAS  Google Scholar 

  18. WMA declaration of Helsinki-Ethical principles for medical research involving human subjects, 2008. http://www.wma.net/en/30publications/10policies/b3/ (accessed November 25, 2012).

  19. Orsini A, Picone L. WISC III-Contributo alla taratura italiana. Firenze: Giunti O.S. Organizzazioni Speciali, 2006.

    Google Scholar 

  20. Wechsler D. Wechsler Intelligence Scale for Children, 3rd ed. San Antonio: the Psychological Corporation, 1991.

    Google Scholar 

  21. Achenbach TM. Manual for the Child Behavior Checklist/4-18 and 1991 Profile. Burlington: University of Vermont Department of Psychiatry, 1991.

    Google Scholar 

  22. Abidin RR. Parenting stress index, srd ed: professional manual. Lutz: Psychological Assessment Resources Inc, 1995:53–71.

    Google Scholar 

  23. Abidin RR. Parenting stress index: a measure of the parent-child system. In: Zalaquett CP, Wood R, eds. Evaluating stress-a book of resources. Lanham: Scarecrow Press, 1997:277–291.

    Google Scholar 

  24. Lessenberry BM, Rehfeldt RA. Evaluating stress levels of parents of children with disabilities. Except Child 2004;70:231–244.

    Google Scholar 

  25. Abidin RR. Parenting Stress index/short form manual. Los Angeles, Calif, USA: Western Psychological Services, 1990.

    Google Scholar 

  26. Carson DK, Schauer RW. Mothers of children with asthma: perceptions of parenting stress and the mother-child relationship. Psychol Rep 1992;71:1139–1148.

    PubMed  CAS  Google Scholar 

  27. Wysocki T, Huxtable K, Linscheid TR, Wayne W. Adjustment to diabetes mellitus in preschoolers and their mothers. Diabetes Care 1989;12:524–529.

    Article  PubMed  CAS  Google Scholar 

  28. Ievers CE, Drotar D. Family and parental functioning in cystic fibrosis. J Dev Behav Pediatr 1996;17:48–55.

    Article  PubMed  CAS  Google Scholar 

  29. Walker LS, Ford MB, Donald WD. Cystic fibrosis and family stress: effects of age and severity of illness. Pediatrics 1987;79:239–246.

    PubMed  CAS  Google Scholar 

  30. Foland-Ross LC, Gotlib IH. Cognitive and neural aspects of information processing in major depressive disorder: an integrative perspective. Front Psychol 2012;3:489.

    Article  PubMed  Google Scholar 

  31. Racer KH, Dishion TJ. Disordered attention: implications for understanding and treating internalizing and externalizing disorders in childhood. Cogn Behav Pract 2012;19:31–40.

    Article  PubMed  Google Scholar 

  32. Vuontela V, Carlson S, Troberg AM, Fontell T, Simola P, Saarinen S, et al. Working memory, attention, inhibition, and their relation to adaptive functioning and behavioral/emotional symptoms in school-aged children. Child Psychiatry Hum Dev 2013;44:105–122.

    Article  PubMed  Google Scholar 

  33. Carotenuto M, Esposito M, Parisi L, Gallai B, Marotta R, Pascotto A, et al. Depressive symptoms and childhood sleep apnea syndrome. Neuropsychiatr Dis Treat 2012;8:369–373.

    Article  PubMed  Google Scholar 

  34. Hung JW, Wu YH, Yeh CH. Comparing stress levels of parents of children with cancer and parents of children with physical disabilities. Psychooncology 2004;13:898–903.

    Article  PubMed  Google Scholar 

  35. Taanila A, Kokkonen J, Järvelin MR. The long-term effects of children’s early-onset disability on marital relationships. Dev Med Child Neurol 1996;38:567–577.

    Article  PubMed  CAS  Google Scholar 

  36. Quittner AL, Opipari LC, Espelage DL, Carter B, Eid N, Eigen H. Role strain in couples with and without a chronic illness: associations with marital satisfaction, intimacy and daily mood. Health Psychol 1998;17:112–124.

    Article  PubMed  CAS  Google Scholar 

  37. Hodgkinson R, Lester H. Stresses and coping strategies of mothers living with a child with cystic fibrosis: implications for nursing professionals. J Adv Nurs 2002;39:377–383.

    Article  PubMed  Google Scholar 

  38. Hermanns J, Florin I, Dietrich M, Rieger C, Hahlweg K. Maternal criticism, mother-child interaction, and bronchial asthma. J Psychosom Res 1989;33:469–476.

    Article  PubMed  CAS  Google Scholar 

  39. Weinstein AG, Faust DS, McKee L, Padman R. Outcome of short-term hospitalization for children with severe asthma. J Allergy Clin Immunol 1992;90:66–75.

    Article  PubMed  CAS  Google Scholar 

  40. Hullmann SE, Wolfe-Christensen C, Ryan JL, Fedele DA, Rambo PL, Chaney JM, et al. Parental overprotection, perceived child vulnerability, and parenting stress: a cross-illness comparison. J Clin Psychol Med Settings 2010;17:357–365.

    Article  PubMed  Google Scholar 

Download references

Author information

Authors and Affiliations

  1. Clinic, Department of Mental and Physical Health, and Preventive Medicine, Second University of Naples, Naples, Italy

    Marco Carotenuto, Maria Esposito, Francesca Di Pasquale & Francesca Santamaria

  2. Department of Pediatrics, Federico II University, Naples, Italy

    Sara De Stefano & Francesca Santamaria

  3. Clinic of Child and Adolescent Neuropsychiatry, Second University of Naples, Via Sergio Pansini 5 PAD, 11, 80131, Naples, Italy

    Maria Esposito

Authors
  1. Marco Carotenuto
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Maria Esposito
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Francesca Di Pasquale
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Sara De Stefano
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Francesca Santamaria
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Maria Esposito.

Rights and permissions

Reprints and permissions

About this article

Cite this article

Carotenuto, M., Esposito, M., Di Pasquale, F. et al. Psychological, cognitive and maternal stress assessment in children with primary ciliary dyskinesia. World J Pediatr 9, 312–317 (2013). https://doi.org/10.1007/s12519-013-0441-1

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s12519-013-0441-1

Key words

Search

Navigation