Abstract
Purpose
To determine whether caregivers of children with single ventricle heart defects identified as having feeding disorder will report more frequent feeding dysfunction, or maladaptive mealtime behavior and/or interactions, when compared to reference populations.
Methods
As part of routine evaluation, parents of children evaluated at the Feeding, Swallowing, and Nutrition Center at the Children's Hospital of Wisconsin completed previously validated questionnaires to assess feeding dysfunction and parental stress. Parental responses for single ventricle patients were compared to all other children evaluated with a feeding disorder.
Results
Questionnaires were completed in 8 patients with single ventricle heart defects. The mean age was 36 ± 23 months, with 5 females (63%). Mean weight-for-age Z score was -1.4 ± 0.9. Compared to non-cardiac feeding clinic children, there was more reported child resistance to eating (83 ± 15% versus 44 ± 2%; p = 0.05). Single ventricle parents were more likely to report distress (50 ± 18% versus 21 ± 2%; p = 0.04) and a difficult child (63 ± 17% versus 31 ± 2%; p = 0.05). There was also more defensive responding among parents of single ventricle children (63 ± 17% versus 29 ± 2%; p = 0.04).
Conclusion
Single ventricle patients evaluated for disordered feeding more frequently showed resistance to eating and parental distress than non-cardiac feeding clinic patients. Parents of these children underestimated the degree of feeding difficulty by defensive responding and had more parental distress. These findings suggest that feeding dysfunction can contribute to longer term feeding and growth problems in single ventricle patients with feeding disorder.
Keywords: Single ventricle, feeding, nutrition, growth
Introduction
Children with congenital heart disease face significant risk of growth failure and feeding difficulties (1,2). Furthermore, those with single ventricle physiology are at particular risk of associated feeding complications such as an increased risk of necrotizing enterocolitis, poor growth and feeding disorders(2,3). These patients require staged palliation, often with an initial surgery in the first few days of life with prolonged disruption of oral feeding due to preoperative concern for NEC and prolonged critical illness during the post-operative recovery. Frequently patients with hypoplastic left heart syndrome (HLHS) require placement of a gastrostomy (G) tube in the post-operative period for feeding disorder(3,4). Many single ventricle patients show catch up growth after bidirectional cavopulmonary anastomosis(5). However, inadequate intake requiring nasogastric (NG) or G tube supplementation persists in 18% of patients in the period between bidirectional cavopulmonary anastomosis and Fontan and 5% of patients after Fontan(6). This persistence of feeding difficulties is not well understood. Feeding difficulties are associated with interstage mortality and earlier bidirectional cavopulmonary anastomosis(4), and low weight for age z-score at the time of cavopulmonary anastomosis surgery is also associated with longer hospital stay post-operatively(7). These factors are likely a cause of significant parental anxiety in regards to feeding and nutritional status. Over time, parental stress can lead to dysfunctional feeding strategies, which could explain the persistence of feeding problems in some cases. To date, no study has quantified the psychosocial stress and evaluated feeding dysfunction, or maladaptive mealtime behaviors or interactions, in single ventricle families. We sought to assess the psychosocial stress in both the parents and patients using previously validated questionnaires. The Parenting Stress Index (PSI) is a questionnaire that has been used to assess the stress present in a parent-child interaction(8). The About Your Child's Eating (AYCE) questionnaire and the Mealtime Behavior Questionnaire (MBQ) have been validated to measure the child-parent interaction as it specifically relates to feeding(9,10). We assessed the hypothesis that in patients with single ventricle physiology and feeding disorder, feeding dysfunction is more common than a reference population also with feeding disorder.
Methods
The study protocol was reviewed and approved by the institutional review board of the Children's Hospital of Wisconsin and informed consent was obtained. All patients presenting to the Feeding, Swallowing, and Nutrition Center at the Children's Hospital of Wisconsin were administered the Mealtime Behavior Questionnaire, About Your Child's Eating, and/or the Parenting Stress Index. Those with single ventricle physiology were isolated from the database and analyzed. These patients were then compared to previously published normal controls and to non-cardiac patients presenting to the same clinic. Data are expressed as means ± standard deviation and comparisons between groups were performed using chi-squared testing.
Results
From 2003-2010, 8 patients with single ventricle physiology were evaluated in the Feeding, Swallowing, and Nutrition Center at the Children's Hospital of Wisconsin and completed appropriate questionnaires. Cardiac anatomy consisted of: 4 patients with HLHS, 2 patients with pulmonary atresia with an intact ventricular septum, 1 patient with unbalanced atrioventricular septal defect and 1 patient with tricuspid atresia with interrupted aortic arch. At the time of questionnaire administration the mean age was 36 ± 23 months with 5 females (63%). Six patients were status post Fontan while 2 were status post cavopulmonary anastomosis without Fontan completion. The mean weight-for-age Z score was -1.4 ± 0.9. G tubes were present in 7 patients (89%) for supplementation of oral feeds. All single ventricle patients had choking, gagging or vomiting. These patients were compared to patients presenting to the feeding clinic feeding disorder related to a variety of underlying problems such as extreme prematurity, cerebral palsy and solid organ transplantation. The mean age was 40 ± 31 months. Results of comparison with normal controls can be seen in Table 1 and with other feeding clinic patients in Table 2. Single ventricle patients had significantly more resistance to eating than the normal controls (83 vs. 1.3%, p value < 0.0001) and the non-cardiac feeding disorder group (44%, p value = 0.05). Parents of single ventricle patients more frequently had distress and rated their child as difficult more often than non-cardiac feeding disorder patients. There was also significantly more defensive responding in the single ventricle group than the feeding disorder control group (63 vs. 29%; p value = 0.04).
Table 1.
Comparison of single ventricle patients with normal controls.
| Single ventricle patients | Normal controls | ||
|---|---|---|---|
|
| |||
| Feature | Percent Positive | Percent positive | p value |
| MBQ | N=7 | N=355 | |
| Distraction/Avoidance | 29 | 6.1 | 0.02 |
| Food manipulation | 43 | 5.0 | <0.0001 |
| Aggression | 14 | 4.1 | 0.2 |
| Choking, gagging, vomiting | 100 | 7.6 | <0.0001 |
| AYCE | N=6 | N=384 | |
| Child resistance to eating | 83 | 1.3 | <0.0001 |
| Positive mealtime environment | 33 | 4.4 | 0.0005 |
| Parent aversion to mealtime | 67 | 1.0 | <0.0001 |
MBQ – Mealtime Behavior Questionnaire, AYCE – About your Child's Eating, PSI – Parental Stress Index
Table 2.
Comparison of single ventricle patients with non-cardiac patients presenting to the Feeding, Swallowing and Nutrition clinic.
| Single ventricle patients | Other feeding disorder patients | ||
|---|---|---|---|
|
| |||
| Feature | Percent Positive | Percent positive | p value |
| MBQ | N=7 | N=665 | |
| Distraction/Avoidance | 29 | 23 | 0.73 |
| Food manipulation | 43 | 51 | 0.68 |
| Aggression | 14 | 30 | 0.36 |
| Choking, gagging, vomiting | 100 | 71 | 0.09 |
| AYCE | N=6 | N=745 | |
| Child resistance to eating | 83 | 44 | 0.05 |
| Positive mealtime environment | 33 | 50 | 0.42 |
| Parent aversion to mealtime | 67 | 37 | 0.13 |
| PSI | N=8 | N=669 | |
| Total stress | 38 | 29 | 0.60 |
| Parental distress | 50 | 21 | 0.04 |
| Dysfunctional interaction | 50 | 25 | 0.10 |
| Difficult child | 63 | 31 | 0.05 |
| Defensive responding | 63 | 29 | 0.04 |
MBQ – Mealtime Behavior Questionnaire, AYCE – About your Child's Eating, PSI – Parental Stress Index
Discussion
Of all cardiac patients, those with single ventricle physiology have the most feeding difficulties and are slowest to reach enteral feeding goals(11,12). Investigations exclusively of the single ventricle population have shown that factors such as timing of enteral feeds, duration of ventilation, total hospital course and readmission rates were not associated with poor growth (13). Many strategies are used to overcome the poor feeding and growth encountered in single ventricle patients, including supplementation of volume with tube feedings, whether by G tube or NG tube, and increases in caloric density. NG or G tube supplementation of feedings is reported in 25-75% of patients prior to bidirectional Glenn(6,13). In spite of these strategies, there continues to be growth problems. At the time of cavopulmonary anastamosis 50% of patients were severely underweight (14) and 89% failed to meet CDC standards for growth(7).
Home monitoring strategies are used at our institution and many others, which includes measurement of daily weight, with guidelines to report a loss of >30g in 24 hours, failure to gain at least 20g in 3 days or daily intake of <100ml/kg/day. This has improved interstage survival, with those in breach of guidelines undergoing earlier cavopulmonary anastamosis(4). The majority of violations of these guidelines were isolated to feeding or growth problems. The use of this aggressive nutritional plan has resulted in normal growth for interstage patients with improved mortality (15). However, this may place a greater stress on families at mealtime. Parents realize that poor growth and intake may equate to worse outcomes or at the very least, further medical evaluation including possible inpatient monitoring.
After cavopulmonary anastomosis surgery, there is significant catch up growth in most single ventricle patients but inadequate intake requiring NG or G tube supplementation persists in 18% of patients in the pre-Fontan period and 5% of patients after Fontan(6). This raises concern that environmental, and more specifically psychosocial, factors could play a significant role in these patients. We proposed that over time feeding problems could be replaced by dysfunctional feeding strategies that create a cycle of poor feeding and further stress and feeding dysfunction. These data indicate that single ventricle patients presenting to the feeding clinic are more resistant to feeding and therefore, there is more parental distress than in non-cardiac patients with similar feeding disorder. Single ventricle parents also significantly underestimate the problems with defensive responding, or trying to normalize their child's behavior.
There are limitations to this data, in particular the small sample size and patient selection, in that all the patients were presenting to the Feeding, Swallowing, and Nutrition Center. Future study may include administration of the questionnaires to a large cohort of single ventricle patients to determine the true incidence of feeding dysfunction in this group and risk factors for development of feeding dysfunction.
Conclusion
Single ventricle patients with feeding disorder show more resistance to feeding than non-cardiac patients with feeding disorder. Single ventricle caregivers normalize their child's behavior with defensive responding. Early assessment for feeding dysfunction is warranted in single ventricle patients with feeding disorder.
Footnotes
None of the authors have any potential conflicts of interest, including financial interests or relationships to industry, relevant to the subject matter or materials discussed in the manuscript.
Author Contributions: Garick Hill – data collection, data analysis including statistics, article drafting and revision, approval of final article.
Alan Silverman – concept/design, data collection, data interpretation, critical revisions, approval of final article.
Richard Noel – concept/design, critical revisions, approval of final article.
Peter Bartz – concept/design, critical revisions, approval of final article.
References
- 1.Varan B, Tokel K, Yilmaz G. Malnutrition and growth failure in cyanotic and acyanotic congenital heart disease with and without pulmonary hypertension. Arch Dis Child. 1999 Jul;81(1):49–52. doi: 10.1136/adc.81.1.49. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 2.Maurer I, Latal B, Geissmann H, Knirsch W, Bauersfeld U, Balmer C. Prevalence and predictors of later feeding disorders in children who underwent neonatal cardiac surgery for congenital heart disease. Cardiol Young. 2011 Jun;21(3):303–309. doi: 10.1017/S1047951110001976. [DOI] [PubMed] [Google Scholar]
- 3.Jeffries HE, Wells WJ, Starnes VA, Wetzel RC, Moromisato DY. Gastrointestinal morbidity after Norwood palliation for hypoplastic left heart syndrome. Ann Thorac Surg. 2006 Mar;81(3):982–987. doi: 10.1016/j.athoracsur.2005.09.001. [DOI] [PubMed] [Google Scholar]
- 4.Ghanayem NS, Hoffman GM, Mussatto KA, Cava JR, Frommelt PC, Rudd NA, et al. Home surveillance program prevents interstage mortality after the Norwood procedure. J Thorac Cardiovasc Surg. 2003 Nov;126(5):1367–1377. doi: 10.1016/s0022-5223(03)00071-0. [DOI] [PubMed] [Google Scholar]
- 5.Srinivasan C, Jaquiss RD, Morrow WR, Frazier EA, Martin D, Imamura M, et al. Impact of staged palliation on somatic growth in patients with hypoplastic left heart syndrome. Congenit Heart Dis. 2010 Nov-Dec;5(6):546–551. doi: 10.1111/j.1747-0803.2010.00457.x. [DOI] [PubMed] [Google Scholar]
- 6.Vogt KN, Manlhiot C, Van Arsdell G, Russell JL, Mital S, McCrindle BW. Somatic growth in children with single ventricle physiology impact of physiologic state. J Am Coll Cardiol. 2007 Nov 6;50(19):1876–1883. doi: 10.1016/j.jacc.2007.07.050. [DOI] [PubMed] [Google Scholar]
- 7.Anderson JB, Beekman RH, 3rd, Border WL, Kalkwarf HJ, Khoury PR, Uzark K, et al. Lower weight-for-age z score adversely affects hospital length of stay after the bidirectional Glenn procedure in 100 infants with a single ventricle. J Thorac Cardiovasc Surg. 2009 Aug;138(2):397–404.e1. doi: 10.1016/j.jtcvs.2009.02.033. [DOI] [PubMed] [Google Scholar]
- 8.Pedersen SD, Parsons HG, Dewey D. Stress levels experienced by the parents of enterally fed children. Child Care Health Dev. 2004 Sep;30(5):507–513. doi: 10.1111/j.1365-2214.2004.00437.x. [DOI] [PubMed] [Google Scholar]
- 9.Davies WH, Ackerman LK, Davies CM, Vannatta K, Noll RB. About Your Child's Eating: factor structure and psychometric properties of a feeding relationship measure. Eat Behav. 2007 Dec;8(4):457–463. doi: 10.1016/j.eatbeh.2007.01.001. [DOI] [PubMed] [Google Scholar]
- 10.Berlin KS, Davies WH, Silverman AH, Woods DW, Fischer EA, Rudolph CD. Assessing Children's Mealtime Problems With the Mealtime Behavior Questionnaire. Children's Health Care. 2010;39(2):142–156. 04/23; 2012/02. [Google Scholar]
- 11.Natarajan G, Reddy Anne S, Aggarwal S. Enteral feeding of neonates with congenital heart disease. Neonatology. 2010;98(4):330–336. doi: 10.1159/000285706. [DOI] [PubMed] [Google Scholar]
- 12.Davis D, Davis S, Cotman K, Worley S, Londrico D, Kenny D, et al. Feeding difficulties and growth delay in children with hypoplastic left heart syndrome versus d-transposition of the great arteries. Pediatr Cardiol. 2008 Mar;29(2):328–333. doi: 10.1007/s00246-007-9027-9. [DOI] [PubMed] [Google Scholar]
- 13.Anderson JB, Beekman RH, 3rd, Eghtesady P, Kalkwarf HJ, Uzark K, Kehl JE, et al. Predictors of poor weight gain in infants with a single ventricle. J Pediatr. 2010 Sep;157(3):407–13. 413.e1. doi: 10.1016/j.jpeds.2010.04.012. [DOI] [PubMed] [Google Scholar]
- 14.Kelleher DK, Laussen P, Teixeira-Pinto A, Duggan C. Growth and correlates of nutritional status among infants with hypoplastic left heart syndrome (HLHS) after stage 1 Norwood procedure. Nutrition. 2006 Mar;22(3):237–244. doi: 10.1016/j.nut.2005.06.008. [DOI] [PubMed] [Google Scholar]
- 15.Hehir DA, Cooper DS, Walters EM, Ghanayem NS. Feeding, growth, nutrition, and optimal interstage surveillance for infants with hypoplastic left heart syndrome. Cardiol Young. 2011 Dec;21(Suppl 2):59–64. doi: 10.1017/S1047951111001600. [DOI] [PubMed] [Google Scholar]