Gastrointestinal Motility and Sensory Abnormalities May Contribute to Food Refusal in Medically Fragile Toddlers
Zangen, Tsili; Ciarla, Carla; Zangen, Samuel; Di Lorenzo, Carlo; Flores, Alex F.; Cocjin, Jose; Reddy, Sarabudla Narasimha; Rowhani, Anita; Schwankovsky, Lenore; Hyman, Paul E.
Journal of Pediatric Gastroenterology and Nutrition . 37(3):287-293, September 2003.
Department of Pediatrics, Kansas University Medical Center; *Pediatric Gastroenterology and Nutrition, Schneider Children’s Medical Center, Israel; †Pediatric unit, Santa Maria delle Grazie Hospital, Naples, Italy; ‡GI Department, Children’s Hospital of Pittsburgh, PA; §Pediatric Gastroenterology, Newton-Wellesley Hospital, Newton, MA
Received October 11, 2001; accepted March 15, 2003.
Address correspondence and reprint requests to Dr. Tsili Zangen, Department of Pediatric Gastroenterology and Nutrition, Schneider Children’s Medical Center of Israel, 14 Kaplan street, Petah-Tiqva, Israel, (e-mail: firstname.lastname@example.org).
Background: In chronically ill children who refuse to eat, surgery to correct anatomic problems and behavioral treatments to overcome oral aversion often succeed. A few patients fail with standard treatments. The aims of the study were to: 1) investigate motility and gastric sensory abnormalities and 2) describe treatment that was individualized based on pathophysiology in children who failed surgery and behavioral treatments.
Methods: We studied 14 patients (age 1.5-6; mean 2.5; M/F: 7/7). All had a lifelong history of food aversion and retching or vomiting persisting after feeding therapy and fundoplication. All were fed through gastrostomy or gastro-jejunostomy tubes. We studied esophageal and antroduodenal manometry, and gastric volume threshold for retching. We identified when gastric antral contractions were associated with retching and pain. A multidisciplinary treatment program included a variable combination of continuous post-pyloric feedings, drugs to decrease visceral pain, drugs for motility disorders, and behavioral, cognitive, and family therapy. We interviewed parents 2-6 months following testing to evaluate symptoms, mode of feeding and emotional health.
Results: We found a motility disorder alone in 2, decreased threshold for retching alone in 5 and both motility and sensory abnormalities in 7. After treatment, 6 of 14 (43%) began eating orally and 80% had improved emotional health. Retching decreased from 15 episodes per day to an average of 1.4 per day (p <0.01).
Conclusions: Upper gastrointestinal motor and/or sensory disorders contributed to reduced quality of life for a majority of children and families with persistent feeding problems. A multidisciplinary approach improved symptoms and problems in these children
Children with developmental disorders and those with chronic disease appear to be at risk for chronic feeding problems and food refusal(1,2). In a recent study, children referred for feeding problems were categorized using a multidimensional classification system including anatomic abnormalities, neurologic conditions, behavioral issues, cardiac and respiratory problems and metabolic dysfunction (3). Developmental delay was a problem for 74% of all subjects, often in association with anatomic and behavioral problems. Children with psychomotor retardation display a high rate of oral dysfunction (4) with associated aversive experiences (aspiration and choking), food refusal after prolonged airway intubation or tube feeding, and persistent vomiting and retching despite aggressive medical and surgical treatment .
A frequent approach to the evaluation of infants and toddlers who refuse to eat has been to distinguish between medical and behavioral conditions.
This dichotomization sometimes fails when there is a complex interaction among anatomic, physiologic, behavioral and environmental factors. For example a combination of intensive behavioral therapy and nutritional interventions eliminated tube feeding requirements in 47% of children with lifelong food aversion.
We hypothesize that motor and sensory disorders may have contributed to persistent food refusal in some of the children who failed the intensive behavioral therapy. Infants may refuse to eat because it is painful or unpleasant to eat, or they fear eating will be a dysphoric experience. Abnormal antroduodenal manometry (8), gastric antral electrical dysrhythmias (9) and delayed gastric emptying (10), were associated with persistent symptoms of retching, vomiting and food aversion after fundoplication. It seems probable that the abnormalities in motility and visceral sensation existed before fundoplication, and contributed to the pathogenesis of chronic symptoms prompting surgery. The purposes of the current study were 1) to investigate the possible contribution of abnormalities in upper gastrointestinal motility and gastric sensation in medically fragile children referred for refusal to eat; 2) to pilot multidisciplinary treatment plans individualized to each patient's pathophysiology.
We studied 14 children (age range 1.5-6 years; 7 male): 11 with cerebral palsy and moderate to severe developmental delay, 1 with congenital chronic renal failure, and 2 with feeding problems that started after fundoplication but no other known disease. Twelve had received intensive neonatal care, 9 because of prematurity with either respiratory or neurologic complications and 3 because of perinatal hypoxic-ischemic encephalopathy. All presented with a life long history of food aversion, vomiting and/or retching persisting after multiple medical and surgical interventions, including one or two fundoplications (Table 1). The patient with renal failure was considered by his nephrologist as having satisfactory metabolic control. All patients had trials of drugs to treat gastroesophageal reflux disease, including cisapride, metoclopramide, H2 histamine receptor antagonists and/or proton pump inhibitors. All had received months of feeding therapy from occupational or speech therapists. We discontinued medications affecting motility at least 2 days before testing