Quality of Life and Anxiety in Parents of Children with an Anorectal Malformation or Hirschsprung Disease: The First Year after Diagnosis

Abstract

Introduction In 2012, we started the KLANKbord-study. A quality of life (QoL) study that follows patients with an anorectal malformation (ARM) or Hirschsprung disease (HD) and their parents from diagnosis till the age of 18 years. We hypothesized that the diagnosis of ARM or HD initially has a negative influence on QoL and anxiety levels of parents, but that this influence will diminish over time. The aim of this study is to see whether QoL and anxiety levels of parents change within the first year after the diagnosis.

Methods Parents of all children born with ARM or HD, were eligible for this study. Within 3 months after the diagnosis ARM or HD, parents received a set of validated QoL questionnaires (measurement 1). Measurement 2 was 12 months after the first questionnaire.

Main Results During measurement 1 mothers (n = 20) scored significantly higher on the social (p value, 0.01; 95% confidence interval [CI], 0.3946–3.1528) and environmental domain (p value, 0.01; 95% CI, 0.4449–2.2851) of the World Health Organization Quality of Life-BREF (WHOQOL-BREF) compared with the known reference values. Fathers (n = 19) scored significantly higher on the physical (p value, 0.01; 95% CI, 0.2964–1.8072), psychological (p value, 0.001; 95% CI, 0.7697–2.4757), and environmental domain (p value, 0.003; 95% CI, 0.5586–2.4214) than the reference values. Comparison of all domains of the WHOQOL-BREF for mothers and fathers between measurements did not show a significant difference. Anxiety levels of mothers were lower during measurement 2 compared with measurement 1. Anxiety levels of fathers were higher during measurement 2 compared with measurement 1. These differences are not significant. Anxiety levels of mothers were significantly higher than anxiety levels of fathers during measurement 1 (p value, 0.002; 95% CI, 0.808–2,956). During measurement 2 this difference in anxiety of mothers versus fathers did not exist (p value, 0.373; 95% CI, −1.157 to 2.922).

Conclusion A negative influence on the QoL of parents having a child with ARM or HD, compared with the reference population was not seen in this population. QoL did not change significantly during the first year for both fathers and mothers. Anxiety levels of mothers did decline during this first year. The number of parents included in this study is still small, which might influence our results. Therefore, we will continue the KLANKbord-study indefinitely.

Keywords

anorectal malformation - Hirschsprung disease - quality of life - anxiety

• References

• 1 Holschneider AM, Hudson JM eds. Anorectal Malformations in Children. Embryology, Diagnosis, Surgical Treatment, Follow-up. Berlin, Heidelberg: Springer; 2006
• 2 Chumpitazi BP, Nurko S. Defecation disorders in children after surgery for Hirschsprung disease. J Pediatr Gastroenterol Nutr 2011; 53 (1) 75-79
• 3 Streisand R, Braniecki S, Tercyak KP, Kazak AE. Childhood illness-related parenting stress: the pediatric inventory for parents. J Pediatr Psychol 2001; 26 (3) 155-162
• 4 Hassink EA, Brugman-Boezeman AT, Robbroeckx LM , et al. Parenting children with anorectal malformations: implications and experiences. Pediatr Surg Int 1998; 13 (5–6) 377-383
• 5 Funakosi S, Hayashi J, Kamiyama T , et al. Psychosocial liaison-consultation for the children who have undergone repair of imperforate anus and Hirschsprung disease. J Pediatr Surg 2005; 40 (7) 1156-1162
• 6 The WHOQOL group. Development of the WHOQOL: Rationale and current status. Int J Ment Health 1994; 23 (3) 24-56
• 7 Rojas-Carrasco KE. The State-Trait Anxiety Inventory for parents who have hospitalized children in an intensive care unit [in Spanish]. Rev Med Inst Mex Seguro Soc 2010; 48 (5) 491-496
• 8 Gau BS, Chen YC, Lo LH , et al. Clinical applicability of the World Health Organization Quality of Life Scale Brief Version (WHOQOL-BREF) to mothers of children with asthma in Taiwan. J Clin Nurs 2010; 19 (5–6) 811-819
• 9 The WHOQOL Group. Development of the World Health Organization WHOQOL-BREF quality of life assessment. Psychol Med 1998; 28 (3) 551-558
• 10 Marteau TM, Bekker H. The development of a six-item short-form of the state scale of the Spielberger State-Trait Anxiety Inventory (STAI). Br J Clin Psychol 1992; 31 (Pt 3) 301-306
• 11 Spielberger CD, Gorsuch RL, Lushene RE. The State-Trait Anxiety Inventory manual. Palo Alto, CA: Consulting Psychologists Press; 1970
• 12 van der Bij AK, de Weerd S, Cikot RJ, Steegers EA, Braspenning JC. Validation of the dutch short form of the state scale of the Spielberger State-Trait Anxiety Inventory: considerations for usage in screening outcomes. Community Genet 2003; 6 (2) 84-87
• 13 Van der Ploeg HM, Defares PB, Spielberger CD. A Dutch-Language Adaptation of the Spielberger State-Trait Anxiety Inventory. Lisse, The Netherlands: Swets & Zeitlinger; 1980
• 14 Witvliet M, Sleeboom C, de Jong J, van Dijk A, Zwaveling S, van der Steeg A. Anxiety and quality of life of parents with children diagnosed with an anorectal malformation or Hirschsprung disease. Eur J Pediatr Surg 2014; 24 (1) 70-74
• 15 Carvalho AE, Linhares MB, Padovani FH, Martinez FE. Anxiety and depression in mothers of preterm infants and psychological intervention during hospitalization in neonatal ICU. Span J Psychol 2009; 12 (1) 161-170
• 16 Streisand R, Mackey ER, Elliot BM , et al. Parental anxiety and depression associated with caring for a child newly diagnosed with type 1 diabetes: opportunities for education and counseling. Patient Educ Couns 2008; 73 (2) 333-338
• 17 Allen R, Newman SP, Souhami RL. Anxiety and depression in adolescent cancer: findings in patients and parents at the time of diagnosis. Eur J Cancer 1997; 33 (8) 1250-1255
• 18 Carvalho AE, Martinez FE, Linhares MB. Maternal anxiety and depression and development of prematurely born infants in the first year of life. Span J Psychol 2008; 11 (2) 600-608
• 19 Bottos AM, Zanon E, Sartori MT, Girolami A. Psychological aspects and coping styles of parents with Haemophilic child undergoing a programme of counselling and psychological support. Haemophilia 2007; 13 (3) 305-310