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Clinical Research

Nutrition in Clinical Practice


Volume 00 Number 0
Feeding Disorders in Children With Tracheostomy Tubes xxx 2020 1–7
© 2020 American Society for
Parenteral and Enteral Nutrition
DOI: 10.1002/ncp.10551
wileyonlinelibrary.com
Jennifer Henningfeld, MD1 ; Cecilia Lang, APNP2 ; Gina Erato, BA3 ;
Alan H. Silverman, PhD3 ; and Praveen S. Goday, MBBS, CNSC, FAAP3

Abstract
Background: We aimed to describe feeding dysfunction in a group of children with tracheostomy. Methods: Single-center,
retrospective chart review of all children with a tracheostomy who were evaluated by our interdisciplinary feeding program.
Demographic and diagnostic data, nutrition variables, acceptance of food consistencies, as well as 2 validated psychometric
instruments for assessment of feeding dysfunction were analyzed. Results: Thirteen tracheostomy-dependent children (5/13; 38%
ventilator dependent) were evaluated at a median age of 51 months (interquartile range [IQR], 26–69). The majority of children
(8/13; 62%) underwent evaluation after decannulation. Four children (30%) had a history of a cuffed tracheostomy tube. Eleven
children (85%) used a speaking valve prior to decannulation, only 2 of whom started before initial discharge with a tracheostomy.
Children with a tracheostomy had low-median weight- and height-for-age z-scores (−1.27 and −1.73, respectively), with normal-
median body mass index (BMI)–for-age z-score (0.175). Children received 75% of feedings via tube feeding (IQR, 13%–97%).
Compared with other children with feeding disorders, children with tracheostomy had delays in initial acceptance of most food
textures and general diet, and the Mealtime Behavior Questionnaire showed significantly worse overall scores (P = .01), and the
About Your Child’s Eating survey showed significantly higher parental perception of resistance to eating (P = .0001). Conclusion:
Requirement of enteral nutrition, poor oral-feeding skills, chronic malnutrition, and worse mealtime behaviors are associated
with tracheostomy. A history of ventilator dependence, cuffed tracheostomy, and inpatient speaking valve–use were infrequently
associated with interdisciplinary feeding-program evaluation. (Nutr Clin Pract. 2020;00:1–7)

Keywords
child; deglutition disorders; enteral nutrition; feeding and eating disorders of childhood; feeding behavior; growth and development;
pediatrics; swallowing; tracheostomy

Introduction ideally without laryngeal penetration or aspiration.7 About


one-third of all children with feeding difficulties have
Feeding in typical children progresses seamlessly from a aspiration.8 Medical literature has debated the effect of a
completely liquid diet through a variety of textures until the tracheostomy on swallowing, with some literature suggest-
child is consuming the complex diet that adults eat. Pediatric ing increased risk of dysphagia and aspiration. Studies have
feeding disorder has recently been defined as impaired oral
intake that is not age-appropriate and associated with
From the 1 Medical College of Wisconsin, Division of Pediatric
a medical, nutrition, feeding skill, and/or psychosocial Pulmonology and Sleep Medicine, Milwaukee, Wisconsin, USA;
dysfunction.1 In infants and children with tracheostomy, 2 Children’s Hospital of Wisconsin, Department of Respiratory Care

prolonged hospitalization, apart from being associated Services, Milwaukee, Wisconsin, USA; and the 3 Medical College of
with chronic malnutrition, impedes the usual progression Wisconsin, Division of Pediatric Gastroenterology and Nutrition,
of feeding, and these infants miss critical windows for Milwaukee, Wisconsin, USA.
feeding that affect feeding progression.2 This altered feeding Financial disclosure: None declared.
progression in children with tracheostomy has not been Conflicts of interest: Dr Goday serves as consultant to Nutricia and
subjected to detailed examination from an interdisciplinary serves on a Data Safety and Monitoring Board for Shire
perspective.3,4 Finally, feeding in an otherwise medically Pharmaceuticals. The remaining authors have no conflicts of interest
relevant to this article to disclose.
complex child is likely to cause social stressors on both the
child and family alike, which may result in dysfunctional Received for publication November 12, 2019; accepted for publication
May 31, 2020.
feeding strategies.5,6 Research is lacking on psychosocial
stressors because of dysfunctional feeding, mealtime This article originally appeared online on xxxx 0, 2020.
behaviors, and interactions in children with tracheostomies. Corresponding Author:
Swallowing is a complex neuromuscular process leading Praveen S. Goday, MBBS, CNSC, FAAP, 8701 W Watertown Plank
Road, Milwaukee, WI 53226, USA.
to delivery of a food or liquid bolus to the stomach, Email: pgoday@mcw.edu
2 Nutrition in Clinical Practice 00(0)

shown ≤87% prevalence of aspiration in tracheostomy- questionnaires, and anthropometric data at the time of the
dependent adult patients.9,10 Multiple mechanisms for initial evaluation by the FSNC. Institutional review board
dysphagia and aspiration have been theorized in patients approval was obtained. Questionnaires were administered
with tracheostomy, including hindrance of laryngeal as routine screening during FSNC evaluation for clinical
elevation,9-11 esophageal obstruction or compression purposes, and results were obtained via chart review.
with an inflated cuff,12 diminution of the laryngeal
protective reflex,13,14 and atrophy of laryngeal muscles
and uncoordinated closure due to chronically bypassing
Anthropometric and Nutrition Measures
the upper airways.15,16 The indication for tracheostomy We obtained anthropometric data and nutrition intake
has evolved over time, including bypassing upper airway data, including enteral nutrition support and historical oral
obstruction and acting as a conduit for long-term invasive intake data. Oral and enteral intake was evaluated using
ventilation, with the latter becoming a more frequent a 3-day diet record. It is standard practice in our clinic to
indication. Although certain diseases require lifelong assess overall calorie and protein intake and micronutrient
tracheostomy, decannulation is possible in some children. nutriture based on the diet record. For the purpose of
As children advance through the liberation process, they this study, we only recorded overall calorie intake and
progress in their developmental milestones, including oral- percentage of calorie intake, orally and enterally.
feeding skills. A recent study evaluated all children who
were successfully weaned from mechanical ventilation and
decannulated.17 At the time of initial discharge with a
Feeding-Skill Measures
tracheostomy, most children were tube fed; however, at the We assigned the ability to accept specific food textures to
time of decannulation most children were tolerating some the following ages: smooth purees – 6 months, mashed
oral feedings. It also showed that 60% of swallow studies foods and textured purees – 8 months, easily dissolvable
performed prior to decannulation-approved oral feedings.17 solids – 9 months, combination foods – 12 months, diced
Our aim was to retrospectively evaluate the characteris- solids – 14 months, toddler foods – 16 months, and general
tics of children with tracheostomy who required evaluation diet – 18 months. Corrections were made for prematurity
in the Feeding, Swallowing and Nutrition Center (FSNC) at when relevant. The delay in acceptance of a category was
Children’s Hospital of Wisconsin in Milwaukee, Wisconsin. calculated by subtracting the age at which the child accepted
In addition to nutrition status, we also hoped to evaluate the particular category vs the assigned age at which the child
the psychosocial stress of both caretakers and children with should have been able to accept that specific texture of food.
either a history of or a current tracheostomy tube. We In an effort to categorize delay in acceptance of specific
hypothesized that feeding dysfunction and oropharyngeal texture categories, when a child was not yet accepting a
developmental delays will be more common in children specific category at the time of the FSNC visit, the age at
with a tracheostomy or a history of a tracheostomy when the time of the visit was used as the age of acceptance of
compared with age-matched controls with known feeding that food.
dysfunction. We also hypothesized that there will be an
increase in stressful mealtime behaviors.
Developmental and Behavioral Measures
We also obtained psychological and behavioral measures
Materials and Methods of child and caregiver functioning within a feeding
This is a single-center, retrospective chart-review study of context. Specifically, measures assessed problem-feeding
all children aged <21 years who were discharged home from behaviors, the caregiver and child feeding dynamic,
the Children’s Hospital of Wisconsin with a tracheostomy, caregiver/parenting stress, and global child development.
with or without home mechanical ventilation, between The Mealtime Behavior Questionnaire (MBQ) is a 33-
2000 and 2015 and who also required evaluation at FSNC. question Likert scale instrument that measures caregiver
We excluded patients with a congenital gastroesophageal perceptions of child feeding dysfunction.18 Specific
malformation that required surgical repair. Along with subscales include child distraction/avoidance, food
demographic data, we collected pertinent pulmonary manipulation problems, mealtime aggression, and choking,
and nutrition data. From a pulmonary perspective, gagging, and vomiting, which yield a composite total score.
we collected age of tracheostomy placement, need for The About Your Child’s Eating (AYCE) questionnaire
mechanical ventilation, use of a cuffed tracheostomy is a 25-question, caregiver-completed Likert scale instru-
tube, use of a one-way speaking valve and tracheostomy ment that measures elements of the caregiver/child rela-
cap, and decannulation (if performed). From a nutrition tionship within a feeding context.19 Subscales include child
standpoint, we collected data regarding acceptance of resistance to eating, positive mealtime environment, and
various food consistencies, developmental and behavioral parental aversion to mealtime.19
Henningfeld et al 3

The Parenting Stress Index–Short Form (PSI-SF) is a Table 1. Patient Characteristics.


caregiver self-report instrument that was used to assess
Characteristic N (%)
the overall level of stress that parents/caregivers experience
when conducting parenting tasks.20 The PSI-SF is a 36- Gender, male 8 (62%)
item instrument that yields a total stress score derived from Race
3 subscales: parental distress, parent-child dysfunctional White 8 (62%)
interaction, and difficult child.20 African American 2 (15%)
The Child Development Inventory (CDI) is a 300-item, Other 3 (23%)
caregiver observational scale to describe areas of develop- Tracheostomy characteristics
Tracheostomy only 8 (62%)
ment in children aged 15 months to 6 years. This inven- Tracheostomy with home mechanical ventilation 5 (38%)
tory specifically assesses social, self-help, motor, language Diagnosis leading to tracheostomy
development, and preschool achievement skills. The CDI Upper airway 3 (23%)
derives a total development score and developmental age Lower airway 7 (54%)
estimates.21 Both upper and lower airway 3 (23%)
All the above behavioral and developmental measures
have been shown to have good internal consistency and Control characteristics
construct validity.18-21
Characteristic N (%)

Controls Gender, male 8 (62%)


Race
For the purpose of comparing children with feeding disor- White 8 (62%)
ders who do not have tracheostomy with the children with African American 2 (15%)
feeding disorders that do have tracheostomies, a sample Other 3 (23%)
was drawn from an existing clinical database of children Age Median 60 months (IQR, 28-78)
previously diagnosed with a feeding disorder. The children a IQR, interquartile range.
with tracheostomy were matched according to their age, sex,
race, and general development with other children receiving
treatment at the FSNC. behavioral differences between children with tracheostomy
and children with known feeding disorders, matched sample
Data Analysis t-tests were conducted on total score and subscales of the
MBQ and subscales of the AYCE. Independent sample
Participants. Paired samples t-tests were conducted on t-tests were also conducted to detect differences between
demographic and developmental variables of sex, race, age, groups on a measure of caregiver stress (based on the PSI),
and general development (as measured by the total CDI including a total score and subscales of parent distress,
score) to ensure appropriate matching and to determine parent-child difficult interactions, and difficult child.
whether factors unrelated to tracheostomy status could
confound the study results. Additionally, paired samples Results
t-tests were conducted on measures of caregiver stress
(based on the PSI). Thirteen tracheostomy-dependent children underwent eval-
uation at FSNC during the study period (Table 1).
Anthropometric and Nutrition. To obtain comparative data
regarding anthropometric, nutrition, gastrointestinal, and Tracheostomy Characteristics
oral feeding, χ 2 analyses were conducted on weight-for- Of the 13 children who underwent evaluation at the FSNC,
age z-scores, height-for-age z-scores, BMI-for-age z-scores, all children had their tracheostomy placed within 7 months
and oral vs enteral feeding. Percentages of children who of birth. Only 5 children (38%) required home mechanical
are physically able to accept thin liquids across groups and ventilation. Tracheostomy cuff was used inpatient for
children who can accept thickened liquids across groups 4 children (31%) with only 3 of those children (23%)
were also evaluated by χ 2 analyses. needing a cuff as an outpatient. All children in the study
were weaned off mechanical ventilation and ultimately
Behavioral. To detect behavioral differences between chil- decannulated. Eleven children (84%) attempted a one-
dren with tracheostomy and a community sample, a one- way speaking valve prior to decannulation, with 1 child
sample t-test was conducted comparing the tracheostomy attempting prior to initial discharge with tracheostomy.
group with the mean and SD values of total score and Ten children (77%) attempted tracheostomy capping prior
subscales of the MBQ and subscales of the AYCE. To detect to decannulation. Three children were unable to cap their
4 Nutrition in Clinical Practice 00(0)

Table 2. Comparison of Growth and Feeding in Children With Tracheostomy and Controls.

Tracheostomy (mean ± SD) Controls (mean ± SD) P-value

Anthropometric measurements
Weight-for-age z-score −1.32 ± 0.94 −1.02 ± 2.05 .029
Height-for-age z-score −1.77 ± 0.97 −1.02 ± 1.49 .091
Body mass index (BMI)–for-age z-score 0.15 ± 1.01 −0.27 ± 1.67 .047
Oral vs enteral feeding
% of calories by enteral tube 55 ± 45.7 29.9 ± 43.4 .37
% of calories taken orally 45 ± 45.7 70.2 ± 43.4 .37

n (%) n (%) P-value

Acceptance of thin and thick liquids


% able to accept thin liquids 12 (92.3) 12 (92.3) 1.0
% able to accept thickened liquids 11 (84.6) 12( 92.3) .56

Table 3. Comparison of Delay in Acceptance of Various Food Consistencies in Children With A Tracheostomy and Controls.

Tracheostomy (mean ± SD) Controls (mean ± SD)


Delay in months P-value

Smooth purees 13.6 ± 19.5 2.5 ± 3.8 .059


Mashed foods 18.4 ± 20.8 4.6 ± 10.0 .06
Textured foods 23.8 ± 22.9 5.8 ± 10.3 .008*
Easily dissolvable solids 24.2 ± 21.9 8.1 ± 12.4 .052
Combination foods 28.4 ± 23.9 7.8 ± 12.3 .003
Diced solids 27.9 ± 23.3 7.5 ± 12.4 .01*
Toddler foods 26.1 ± 23.1 7.5 ± 12.5 .01*
General diet 24.8 ± 22.3 13.3 ± 19.2 .3

*p < 0.05.

tracheostomy due to upper airway obstruction, requiring with tracheostomy were receiving a larger percentage of
laryngotracheal construction prior to decannulation. their energy needs via enteral nutrition (55 ± 45.7) vs
controls (29.9 ± 43.4), but the results were not statistically
significant (P = .37) (Table 2). All children were receiving the
Anthropometric and Nutrition Data rest of their energy intake orally (ie, 45% [± 54.7] in children
Median age at evaluation in FSNC was 51 months (in- with tracheostomy vs 70.1% [± 56.6] in the controls).
terquartile range, 26–69). The majority of children (8/13; At the initial presentation to the feeding clinic, there was
62%) underwent evaluation after decannulation. Of the no difference in the ability of patients with tracheostomy
5 children who were ventilator dependent, none required and controls to accept thin liquids (12; 92.3% vs 12; 92.3%;
mechanical ventilation at the time of evaluation in FSNC. P = 1.0) or thickened liquids (11; 84.6% vs 12; 92.3%; P =
Tracheostomy-dependent children had lower weight-for-age 0.56) (Table 2). None of the children had any restrictions in
and height-for-age z-scores when compared with controls regard to solid-food consumption.
with feeding disorders. Their BMI-for-age z-scores were There was a delay in acceptance of most food textures by
normal (Table 2). Six of the 13 children with tracheostomy children with a tracheostomy when compared with controls
(46.1%) and 3 of 13 controls (23.1%) had height z-scores < (Table 3).
−2, but none in either group had height z-scores < −3. There was no statistically significant difference between
Children with a tracheostomy were statically more likely children with tracheostomy and controls in regard to CDI,
to have an enteral feeding tube than controls (10 vs 5, ages, aspiration on last swallow study, gagging, abdominal
respectively; P = .047). All children who required an enteral pain, constipation, vomiting, diarrhea, acid-suppression
feeding tube had a gastrostomy tube, except for 1 child with medication, food allergies, and environmental allergies (data
a tracheostomy who had a gastrojejunal tube. The children not shown).
Henningfeld et al 5

Figure 1. Frequency of feeding-disorder subtypes in children with tracheostomy and controls, based on the Mealtime Behavior
Questionnaire (MBQ) and About Your Child’s Eating (AYCE) questionnaires. Measure subscales and ranges: Mealtime Behavior
Questionnaire total score (33–165): distraction/avoidance (10–50), food manipulation (6–30), aggression (6–30), and choking,
gagging, vomiting (3–15); About Your Child’s Eating total score (1–100): child resistance to eating (11–55), positive mealtime
environment (4–20), and parent aversion to mealtime (5–25).

Mealtime Behaviors Children with tracheostomy appear to have worse feed-


ing disorders than other children with feeding disorders.
When compared with matched controls on a measure of They are much more likely to have a gastrostomy tube
mealtime behavior problems, the tracheostomy-dependent than matched controls. Our small study suggests that
children had significantly lower distraction/avoidance, more tracheostomy-dependent children have a trend toward re-
food manipulation problems, less mealtime aggression, ceiving more calories via the enteric tube. Their ability
higher choking/gagging/vomiting, and a higher total scale to accept liquid safely at the time of evaluation was no
score. On a measure of caregiver/child feeding interac- different than from controls; however, there were delays
tions, the tracheostomy-dependent children had signifi- in dietary progression through various dietary textures.
cantly higher resistance to eating when compared with Interestingly, our study did not demonstrate any increase
controls; however, tracheostomy-dependent children and in feeding disorders in children who required mechanical
controls had no difference between mealtime environment ventilation, cuffed tracheostomy, or the use of a speaking
and parent aversion to eating (Figure 1). valve or tracheostomy capping prior to decannulation. The
discovery of feeding disorders and lack of identifiable risk
factors within the tracheostomy-dependent population in
Discussion this study may reflect the timing of the tracheostomy.
Our preliminary study is one of the first detailed explo- Since tracheostomies are often placed following prolonged
rations of feeding disorders in a small group of children intubation at young ages, oral aversion is common. Addi-
with tracheostomy. All of these tracheostomy-dependent tionally, the tracheostomy is often placed or remains in place
children met the criteria for pediatric feeding disorder during critical windows of feeding-skills development, and
by exhibiting significant feeding issues, including over- therefore, feeding skills are likely to be impacted.
all feeding-skill issues, poor overall growth, and psy- Adult literature supports the use of a speaking valve to
chological issues associated with feeding and mealtime help decrease aspiration and gain oral skills;22,23 however,
behaviors. this has not been supported by pediatric data.24 We also did
6 Nutrition in Clinical Practice 00(0)

not find a significant correlation between speaking valve and and safety should be emphasized, and caregivers should
feeding disorders in our pediatric population, but to draw be provided with ample support to prevent the onset of
conclusions from our study would be premature because behavioral etiologies of further feeding problems.
of the small sample size. The discordance between adult This study has limitations. First, this study represents a
and pediatric data may be explained by the fact adults very small cohort of a much larger tracheostomy-dependent
often had normal swallowing mechanics prior to an insult population because of our selection criteria. Further study
requiring placement of a tracheostomy, whereas children of all children with tracheostomy would help capture those
with tracheostomy often never acquired normal swallowing tracheostomy-dependent children who will never eat orally
skills prior to tracheostomy. That said, there are significant because of a variety of factors, children who always ate by
theoretical benefits to the speaking valve, and early use of mouth without feeding issues, and children with feeding dys-
the speaking valve should be encouraged until more data function who have the potential to eat by mouth but are not
are available. being actively advanced toward oral feedings by their care-
Overall, the tracheostomy-dependent children had very givers or therapists. This small cohort also limits our ability
low weights and heights. Since the heights were much more to show statistical significance and may underestimate the
affected than the weights, they had normal BMIs. Given differences between the aged-matched controls. Second, it
that most of these children had gastrostomy tubes, it is is a retrospective study and does not show the progression
likely that their weights were being managed through use of skills over time. Prospective multi-institutional studies
of these tubes, leading to normal BMIs. Six of the children across the continuum of pediatric tracheostomy are needed
had height z-scores < −2, but none had height z-scores to further characterize the feeding disorders in this cohort.
< −3; such scores would have qualified these children for One of the goals of pediatric tracheostomy is to allow
chronic malnutrition, based on World Health Organization children to achieve their full developmental potential. This
(WHO) criteria25 but not on the American Society for study emphasizes the need to support children outside
Parenteral and Enteral Nutrition/Academy of Nutrition of their airway and mechanical ventilation. Pediatric tra-
and Dietetics (AND) criteria.26 Stunting, based on the cheostomy programs should increase collaboration with a
WHO criterion (height z-score < −2), is a well-established variety of other specialists, such as registered dietitians,
risk marker of poor child development. Stunting before the speech therapists, occupational therapists, gastroenterolo-
age of 2 years predicts poorer cognitive and educational gists, and psychologists, to help children maximize their
outcomes in later childhood and adolescence.27,28 Stunting full global developmental potential while they are acquiring
is also associated with a reduction in schooling, lower test oral-feeding skills.
performances, lower household per capita expenditure, and
a greater likelihood of living in poverty in the developing
Statement of Authorship
world.29 Our findings are similar to the stunted growth
seen in children with extremely low birth weight30 and J. Henningfeld contributed to the design of the work, collected
hypoplastic left heart syndrome,31 likely reflecting that these data, wrote the first draft, and approved the final manuscript;
C. Lang contributed to the design of the work, helped with
children had significant medical problems commencing at
manuscript redrafting, and approved the final manuscript; G.
birth.
Erato collected data, helped with manuscript redrafting, and
As children progress toward liberation from respiratory approved the final manuscript; A. H. Silverman contributed
technology, they also make gains in lungs maturation and to the design of the work, helped with manuscript redrafting,
developmental gains outside of their airway , such as and approved the final manuscript; P. S. Goday contributed to
requiring less speech, physical, and occupational therapy the design of the work, interpretation of the data, helped with
and a decreased incidence of gastrostomy tube requirement manuscript redrafting, and approved the final manuscript.
at the time of decannulation when compared with tra-
cheostomy placement.30 This study shows the progression References
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