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A B S T R AC T
This article describes a 6-year-old boy with Down Syndrome who was referred with
a refractory feeding disorder and his successful treatment using a multidisciplinary
treatment team approach. The case highlights the complex nature of feeding
disorders in this population, as well as the need to consider multiple issues during
their assessment and treatment.
K E Y WO R D S
attention deficit/hyperactivity disorder, Down syndrome, feeding disorder
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successful treatment (Calvert et al., 1976). In this article we describe a DS patient with
feeding difficulties that demonstrates the multifactorial nature of these complex and
difficult-to-treat disorders. We also describe a multidisciplinary treatment approach that
led to successful resolution of his feeding disorder.
Case report
Martin is a 6-year-old boy diagnosed with DS. He was referred for evaluation of his
aversion to solid food and failure-to-thrive. At the time of admission, Martin was fed
exclusively using a bottle, and would drink only Pediasure®. Martin would occasionally
eat ice cream and pickles, or lick potato chips, but otherwise ate no solid food. His mother
also reported that he would vomit several times/day, particularly when nervous or agitated.
Although Martin’s weight did not place him at immediate medical risk, his refusal to eat
or drink in public meant that he was not able to attend school.
G R E T C H E N D. F L A N AG A N , R D
is a Registered Dietician specializing in pediatrics who
works in the Department of Clinical Nutrition at Lucile Packard and as part of the multi-
disciplinary team for treating pediatric feeding disorders.
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Feeding history
Martin had trouble feeding during the first two weeks of life, and a nasogastric tube was
placed. He was fed with expressed breast milk given in a bottle for the first two months of
life. He was then hospitalized for two weeks for a respiratory infection and resumed
normal bottle-feeding after discharge. Baby foods were introduced without difficulty
around five months of age. At six months of age, Martin was rehospitalized for a lung infec-
tion complicated by severe vomiting and diarrhea and he regressed to bottle-feeding. He
refused to resume solid feeding and was fed exclusively with a bottle from this date
onwards. Martin was seen for a feeding evaluation at age two years. His mother reported
that the clinician attempted to force Martin to take food by mouth, resulting in him
choking and becoming cyanotic. A second attempt at treatment resulted in dehydration,
followed by a three-week hospitalization. During this period, Martin again acquired a
secondary lung infection, and was subjected to multiple blood tests and medical
procedures, which his mother believed were quite traumatic. After this date, Martin’s
mother gave up on all further attempts to wean him from the bottle.
Social history
Martin had three elder siblings, all healthy and without history of feeding difficulties. He
lived with his parents, and 18-year-old brother. His mother was hearing impaired. He had
attended school but had been placed on home school because of the school’s concerns
about dehydration. He was doing poorly academically, and had been asked to repeat
kindergarten. His mother reported that he got on well with other children, but functioned
socially at the level of a 2–3-year-old.
Formulation
Martin was assessed as having an aversion to solid food with no underlying medical basis.
He was able to chew and swallow solid food, but was heavily dependent on bottle-feeding
as his primary source of nutrition. It was hypothesized that traumatic experiences associ-
ated with attempts to introduce solid food had contributed to his feeding aversion. Over
the years, Martin appeared to have developed negative behavioral responses to oral
feeding efforts that had left his family reliant on Pediasure® to meet his nutritional needs.
His mother’s anxiety about his weight and her perception of his medical fragility resulted
in her being nervous about trying alternate foods or feeding strategies. Other relevant
issues included oral-motor difficulties, reduced appetite and interest in food, and
communication difficulties. Finally, his need for the presence of his mother for bottle-
feeding suggested the possibility of separation–anxiety issues.
Treatment course
Martin was admitted to an inpatient medical–psychiatry unit that is based in a general
pediatric hospital and which has been described in more detail by Lock and Steiner (1999).
The unit specializes in the management of children and adolescents with mixed medical
and psychiatric disorders, including patients with feeding disorders. The unit differs from
other general pediatric units in that the nursing staff have additional training in pediatric
psychiatry issues and there is close daily consultation with other disciplines including occu-
pational therapy and nutrition. Martin’s treatment involved the collaboration of several
disciplines, as outlined below, and comprised three weeks of inpatient treatment followed
by three weeks of day treatment.
Gastroenterology
Gastroenterology work-up including esophagogastroduodenoscopy, esophageal transit
study and gastric emptying study confirmed that there were no inflammatory, gross
anatomical or dysmotility disorders to explain Martin’s feeding difficulties. He was started
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on Periactin 2 mg p.o. b.i.d. in an attempt to stimulate his appetite but there were no
additional medical interventions.
Nutrition
Martin presented in a well-nourished state. His weight of 19.2 kg placed his weight above
the 75th percentile for age on a DS growth chart. Martin’s calorie needs were estimated
to be 70–90 calories/kg body weight. His nutritional needs were being met with 7 cans of
Pediasure® consumed by bottle throughout the day and night. The goal of treatment was
to introduce a variety of solid foods. Initially, Martin was allowed to continue his bottle-
feedings of Pediasure® during the daytime, however, in his night-time feedings, water was
progressively substituted for the Pediasure® in an attempt to stimulate his daytime
appetite. Gradually, over a three-week period, the daytime bottle-feedings were also
omitted – although not until he was comfortably drinking from a cup with a straw – first
by reducing the volume of Pediasure® in each bottle, and then the frequency. Close moni-
toring of his vital signs prevented Martin from becoming dehydrated, and was one of the
justifications for initiating his treatment as an inpatient. Martin’s caloric intake was moni-
tored daily. Initially, his caloric intake dropped to an average of 40 calories/kg of body
weight. However, by the end of the hospitalization his average caloric intake was 77
calories/kg of body weight meeting his estimated nutritional needs.
Psychiatry
Psychiatric assessment suggested that Martin met criteria for attention deficit/hyperactiv-
ity disorder (ADHD), based on history, direct observation and Conner Scale ratings
(Conners & Kronsberg, 1985). Martin’s level of hyperactivity was such that it directly inter-
fered with his ability to sustain adequate attention during meals. His ADHD symptoms
diminished following treatment with dextroamphetamine (5 mg p.o. Qam; 2.5 mg p.o.
Q2:00 pm). Although psychostimulants are commonly associated with appetite suppres-
sion, the benefits of his improved attention span in facilitating his treatment participation
outweighed any possible negative gastrointestinal side effects. The recognition and treat-
ment of his ADHD was an important breakthrough in allowing Martin to take better
advantage of other treatment interventions.
Several additional relevant behavioral and psychological factors were identified. First
was that Martin’s previous history of being ‘medically fragile’ created a fear in his mother
about the potential consequences of weight loss. This fear allowed Martin to manipulate
and intimidate his mother who also lacked strategies or the confidence to make changes
to the problematic feeding routine. She was also skeptical about the treatment plan. For
example, she questioned his diagnosis of ADHD, and believed that he was just ‘energetic’.
In individual meetings with the therapist, she expressed difficulties believing that Martin
would ever attain sufficient independence to leave home. It was hypothesized that his
mother’s fearfulness about the potential risks of dehydration resulted in her being unwill-
ing to try new initiatives that entailed reducing his bottle-feeding.
Key to making progress with Martin was intensive family therapy with his mother. The
initial focus of the work was primarily educational and supportive, for example, helping
her accept the potential value of a trial of stimulants for his attention deficits as well as
helping her set limits around his requests for bottle-feeding during the phase of eliminat-
ing his night-time bottle. Education focused on efforts to decrease his mother’s fear around
the consequences of temporary weight loss which is always anticipated when embarking
on treatment of this nature. It was explained that efforts to change the feeding routine
would inevitably lead to protest and food refusal on the part of Martin, but that there was
a safe margin in which to allow weight loss during his inpatient admission. Weekly
meetings were critical to helping her develop trust in the treatment team.
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Outcome
By the time of discharge, Martin was eating large bowls of thick soup-based mixtures,
with fork-mashed vegetables, meats, noodles, and yogurt with fruit pieces. His lingual
range of movements had improved greatly although he was still limited to munching and
mashing soft foods. He accepted a wider variety of tastes and textures including dry
textured foods. He was able to complete his meals within a 30-minute interval, without
the use of videotapes or other distractions. Martin’s admission weight was 19.2 kg, and
his discharge weight 18.5 kg which had been anticipated given the discontinuation of his
high calorie supplement.
Martin returned home with his mother to a rural area without access to services from OT
or S&L. Telephone contact was maintained to monitor his progress. At six months follow-
up, his weight had increased in line with expectations based on his growth charts indicating
that he was able to maintain a safe body weight while making the transition from
Pediasure® to a solid food diet. He continued to eat a wide variety of foods, although still
required food to be fork mashed to facilitate eating. He was able to feed himself indepen-
dently at school, although at home, his mother still tended to play an active role in feeding.
Discussion
Feeding problems may be classified based on their etiology as being related to medical
conditions, oral-motor dysfunction or delay, or behavioral mismanagement (Linscheid
et al., 1995). Pipes and Holm (1980) report that retention of infantile feeding habits and
inappropriate feeding practices on the part of the parents are two of the important
behavioral issues in children with DS. Our case report illustrates the multifactorial
nature of feeding disorders, particularly in a child with DS, speech and language delays
and comorbid ADHD. One major principle behind our approach is our belief that treat-
ment needs to be multidisciplinary in nature and closely coordinated in order to address
each respective issue.
Table 1 outlines the diagnostic and treatment approach used by our treatment team.
Core to our approach is the multidisciplinary team and close collaboration of the disci-
plines of GI, psychiatry, OT, S&L, nursing and nutrition. Also important in the assess-
ment of children who have failed outpatient treatment is the option of inpatient
admission (Linscheid et al., 1995). This allows the team to take temporary control of the
feeding situation and test new feeding approaches while the child is under close medical
observation. This can be particularly helpful when parents have excessive concerns about
the consequences of temporary weight loss.
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Table 1. Feeding disorders in children with Down syndrome: Diagnostic and treatment issues
Diagnostic issues
• Medical issues
• Oral-motor dysfunction
• Sensory defensiveness
• Parental anxiety
• Reinforcement of negative feeding patterns
• Communication difficulties
• Comorbid psychiatric illness
Behavioral treatments
• Contingent social attention
• Behavioral modification interventions
• Appetite manipulation
• Family therapy
Occupational therapy
• Structure and time limits on meals
• Use of incentives, distraction and behavior modification techniques during meals
• Exercises to improve oral-motor skills
• Gradual introduction of foods with increased textures
• Education of parent about feeding techniques
Speech and language
• Exercises to improve oral-motor skills and speech intelligibility
• Teaching of gestures and verbalizations to improve expressive language
• Cueing methods to encourage verbal expression
Assessment
Assessment of our patients includes a detailed clinical interview with the parent to cover
the feeding history, nature of the feeding difficulties, current mealtime practices and
routine, and previous efforts used to encourage eating. This interview is supplemented
with direct observation of a typical meal, either a videotape of a home meal, or in the
hospital. During the direct observation, data is gathered on the child’s behavioral
reaction to feeding, as well as the nature of the interaction between the parent and child.
Finally, we review the history and current nutritional aspects of the child’s diet and its
relationship with the child’s growth and weight. Careful nutritional assessment of the
caloric needs of the child is essential before starting treatment (Cloud, 1993).
Medical issues It is important to first clarify with parents that linear growth is reduced
in children with DS so that they have appropriate expectations. There are revised growth
charts available specifically for use in children with DS (Cronk et al., 1988). Dental
problems, including malocclusions and malalignment of teeth may also interfere with
nutrition (Patterson & Ekvall, 1993). It is also common for patients to have complicat-
ing medical issues, either gastrointestinal related, or those affecting cardiovascular and
pulmonary systems. Table 2 shows some of the common medical conditions that may
interfere with feeding.
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Conditioned anxiety at the sight of food may provoke a number of avoidance behaviors
that include crying, gagging, or choking at the site of food, refusal to chew or swallow,
or lack of awareness of hunger. These conditioned responses may persist long after the
initial traumatic event has ended, and perpetuate a pattern of selective food refusal or
feeding difficulties, particularly if the avoidance behaviors result in the withdrawal of
food or demand to eat (Linscheid et al., 1995). These issues played a central role in the
origin of our patient’s feeding aversion.
Oral-motor dysfunction Our patient also demonstrated significant difficulties with oral-
motor function that interfered with the transition to solid food. These difficulties
included problems with chewing and manipulating food placed in the mouth, and conse-
quent difficulties swallowing partially chewed food despite a normal swallowing study.
Chewing difficulties are commonly reported in children with DS. This may be a result of
reductions in the size of the oral cavity relative to the tongue due to underdevelopment
of the maxilla and nasal bones, reduced tone of the facial musculature, and inefficiency
of the tongue in lateralizing food for chewing and swallowing (Gisel, Lange, & Niman,
1984; Shapiro, Gorlin, Redman, & Bruhl, 1967; Spizer, Rabinowitch, & Wyhor, 1961).
In addition, children with DS often have prolonged chewing times and take longer to
complete meals (Calvert et al., 1976). Delays in fine motor skills may also result in
difficulties with self-feeding skills (Cullen et al., 1981).
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Reinforcement of negative feeding patterns Chatoor, Schaefer, Dickson, and Egan (1985)
outlined a developmental approach to conceptualize feeding disorders in younger
children and describe the way struggles around food may represent efforts at separ-
ation–individuation. It is expected that oppositional behaviors, both during feeding and
in other situations, are not only expected, but also provide an important opportunity for
children to mature socially (Linscheid et al., 1995). At around one year of age, the
feeding situation changes from one in which the parent has primary control to one of
shared control between parent and child, and the child’s food refusal may represent their
effort to express their autonomy. This may become an issue in children with medical
problems in which the maintenance of an adequate diet is in conflict with the child’s
developmental desire for greater autonomy. Caregivers may unwittingly reinforce these
struggles by their increased attention to their child during mealtimes, particularly when
there is a component of increased parental anxiety (Linscheid et al., 1995).
Communication difficulties Delays in both expressive and receptive language are common
in DS and also contribute to behavioral difficulties that may be acted out at meal times.
Our patient had significant delays in his S&L and he was not able to communicate his
desires clearly, leading to frustration on the part of both child and parent. Communi-
cation difficulties also interfered with efforts to implement treatment interventions, for
example, in trying to ascertain whether the patient understood the proposed behavior-
modification program.
Comorbid psychiatric disorders Although there have been conflicting findings on the
presence of psychiatric disorders in children with DS, several studies have reported a
higher incidence of behavioral problems, specifically oppositional and externalizing
behaviors, and hyperactivity (Green, Dennis, & Bennets, 1989; Pueschel, Bernier, &
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Pezzulo, 1991). Martin’s treatment illustrates well the importance of a careful psychiatric
assessment, as the presence of a comorbid psychiatric disorder, such as ADHD, may
interfere with the feeding process, as well as limiting the ability to take advantage of
treatment interventions. The treatment of Martin’s ADHD resulted in his increased
ability to focus and participate in and benefit from his OT and S&L sessions.
Behavioral interventions
Linscheid et al. (1995) provided a useful overview of some of the behavioral inter-
ventions used in the treatment of feeding disorders.
Contingent social attention One of the core approaches to encourage feeding is the use
of social attention, in the form of praise and verbal encouragement that is made contin-
gent upon desired feeding behaviors. This is a particularly useful modality in children
with DS, who are often described as social and gregarious, and motivated by the desire
for social approval. Contingent social attention is paired with what has been described
as ‘differential social attention’, or the planned ignoring of the child for food refusal or
tantrums. This technique has been applied in a number of studies of children with feeding
disorders, including children with developmental and cognitive delays (Butterfield &
Parson, 1973).
Behavioral approaches A number of programs describe the use of rewards for desired
feeding behaviors, including those of television time, play with toys, or awarding tokens
or stickers that may be redeemed for prizes (Linscheid et al., 1995). Although children
with DS commonly have reduced intelligence (Patterson & Ekwall, 1993), we have found
that even children with quite delayed cognition are able to grasp the concept of rewards
for appropriate behaviors. Martin responded well to the use of videotapes as a reward
for completing meals. Food refusal, throwing of food, and retention of food in the mouth,
all common in DS (Pipes & Holm, 1980), as well as in our patient, were reduced using
behavioral modification measures. We advocate the use of television time after meals as
a reward, rather than during meals as a distraction. Many parents of children with short
attention spans are fed in front of a television which may actually interfere with the
child’s ability to concentrate on the feeding process (Cloud, 1993).
Appetite manipulation It is common for children with feeding disorders to have lower
appetites than do normal children. The use of appetite stimulants, including Periactin,
was found useful in our case to increase appetite. Changing the feeding schedule in
children who have previously had free access to food by only allowing food at mealtimes
can help establish a normal pattern of appetite and satiation (Cloud, 1993). We reduced
the number of calories given in liquid form by either eliminating snacks and substituting
bottle-feeds with water in order that Martin would arrive at meals feeling hungry.
Family therapy Psychiatric issues played a major role in Martin’s feeding disorder. Aside
from the assessment for psychiatric disorders, such as ADHD mentioned above, we have
noted that parents in particular are anxious and often hesitant to try out new approaches.
It is common for parents to equate good parenting with successful feeding with the result
that the pressure to get their child to eat may interfere with their ability to set appro-
priate limits on access to food outside meals (Linsheid et al., 1995). Exploration of the
parents’ resistance to new treatment interventions is critical, as well as providing support
and reinforcement when parents are able to make changes in their parenting and feeding
techniques. Directly addressing the parental fear of the medical consequences of
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temporary weight loss was important with our patient to facilitate his progress (Cloud,
1993). Psychiatric consultation also played an important role in supporting the treatment
team and helping interpret the behavior of Martin and his mother.
Occupational therapy
It is important to keep in mind that Martin had an absence of age-expected hunger and
interest in food. With this in mind, the motivation for change needed to shift from food
to another incentive. Initially, this was accomplished using immediate reinforcement, for
example, being given a turn to place a puzzle piece after taking a bite and later by
allowing an activity such as watching a video after the meal. Distraction such as watching
a video or playing with toys early in the process served to mask negative sensory experi-
ences such as the introduction of texture. It also proved to be the major motivation for
sitting longer for meals in the absence of normal hunger. Eventually, Martin’s feeding
became more spontaneous and it is generally possible to discontinue the program of
incentives.
Parents need constant support and reassurance throughout the process, especially as
setbacks are common. Helping the parents to set goals that are attainable is important.
A goal may be as small as increasing volume input by 5 ml. Positive reinforcement for
small gains is critical not only for the child but also for the parent. In treatments of limited
duration, the transition to chewing often does not take place and longer term outpatient
follow-up should be expected. With support from a therapist in the patient’s community
and access to consultation with the feeding team, progress can continue after discharge.
Conclusion
Feeding disorders in patients with DS are often the final outcome following the coales-
cence of a number of contributing issues. These may include mechanical and sensory
issues, effects of prior medical trauma, and comorbid psychiatric illness. Behaviors that
might not ordinarily lead to major feeding difficulties may often be reinforced and
supported by responses of caregivers permeating the entire parent–child relationship.
Our case illustrates the complexity and multifactorial nature of feeding disorders in
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