Impoacto de Los Problemas Respiatorios en Pc
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Transcript of Impoacto de Los Problemas Respiatorios en Pc
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Impact of feedingproblems onnutritional intake andgrowth: Oxford
Feeding Study II
P B Sullivan* MA MD FRCP FRCPCH, University Lecturer in
Paediatrics, Department of Paediatrics;
E Juszczak, Statistician, Centre for Statistics in Medicine;B R Lambert SRD MSc, Paediatric Dietician;
M Rose MRCP MRCPCH, University of Oxford;
M E Ford-Adams MRCP MRCPCH, Paediatric Registrar,
Department of Paediatrics, John Radcliffe Hospital;
A Johnson MA FRCP FRCPCH, National Perinatal
Epidemiology Unit, University of Oxford, Oxford, UK.
*Correspondence to first author atUniversity of Oxford,
Department of Paediatrics, John Radcliffe Hospital, Oxford,
OX3 9DU, UK.
E-mail: [email protected]
Poor nutritional status and growth failure are common in
children with cerebral palsy (CP). The aim of this study was
to assess, within a subgroup of a large and clearly defined
population of children with disabilities, the impact of feeding
difficulties on (1) the quality (micronutrient intake) andquantity (macronutrient intake) of their diet and (2) their
growth. One hundred children with disabilities (40 females,
60 males; mean age 9 years, SD 2 years 5 months; range 4
years 6 months to 13 years 7 months) underwent a detailed
dietetic analysis and a comprehensive anthropometric
assessment. Diagnostic categories of disability were: CP
(n=90); global developmental delay (n=3); Marfan syndrome
(n=1); intractable epilepsy (n=2); agenesis of the corpus
callosum (n=2); methyl malonic aciduria (n=1); and
congenital rubella (n=1). Neurological impairment was
classified according to difficulty with mobility which was
graded as mild (little or no difficulty walking), moderate
(difficulty walking but does not need aids or a helper), andsevere (needs aids and/or a helper or cannot walk). Results
confirmed the significant impact of neurological impairment
in children on body growth and nutritional status becoming
worse in those with a greater degree of motor impairment.
The major nutritional deficit was in energy intake, with only
one fifth reportedly regularly achieving over 100% estimated
average requirement (EAR), whilst micronutrient intake was
less markedly impaired and protein intake was normal in this
group (96% above EAR). Many children with neurological
impairment would benefit from individual nutritional
assessment and management as part of their overall care.
Poor nutritional status and growth failure are common in
children with cerebral palsy (Thommessen et al. 1991,
Stallings et al. 1995). It has been reported in several studies
that such growth failure can be related to an inadequate food
intake, resulting from self-feeding impairment and oral-motor
dysfunction (Krick and Van Duyn 1984, Gisel and Patrick
1988, Rempel et al. 1988, Stallings et al. 1993, Reilly et al. 1996,
Stallings et al. 1996). However, many of these studies contain
only a small number of participants. In an attempt to overcome
this limitation, we have recently reported the first large-scaleepidemiological study of feeding and nutritional problems
in disabled children with neurological impairment, called
the Oxford Feeding Study (Sullivan et al. 2000). The results
of this study highlighted that feeding problems in children
with neurological impairment are indeed common and
severe and are a cause of much concern to parents.
Patrick and Gisel (1990), on the basis of their experience
with feeding neurologically impaired children, suggest that
nutritional problems are caused primarily by energy deficits
rather than by deficits of protein, vitamins or minerals. They
maintain that it is because the diet given to people with dis-
abilities is qualitatively good but deficient in volume and that
the malnutrition found in individuals with disabilities israrely associated with either skin changes or hair or mucosal
changes. This is, of course, quite different from the clinical
picture of childhood malnutrition seen in the developing
world where micronutrient deficiencies (especially of vita-
min A and zinc) together with protein deficiency are much
more prominent. The qualitative adequacy but quantitative
deficiency of the diets provided for children with disabilities
might also be one of the reasons that such individuals can
survive in a moderately malnourished state for many years.
The aims of the present study were: (1) to test the hypoth-
esis that the diet in children with disabilities is lacking in
quantity rather than quality and (2) to demonstrate that the
dietary deficit and associated growth impairment are worse
in those with more severe levels of motor deficit.
Method
A description of the study population and the selection
procedure for the children studied is provided in detail else-
where (Sullivan et al. 2000). Briefly, a target population of neu-
rologically impaired children with oral-motor dysfunction
was identified using the Oxford Register of Early Childhood
Impairments (ORECI). All had been born to mothers resi-
dent in the four counties of Berkshire, Buckinghamshire,
Northamptonshire and Oxfordshire in England at the time of
delivery. In this cross-sectional study, 377 children (aged 4 to 14
years), born between 1984 and 1995, with motor or feeding
problems recorded on the ORECI database were selected fordetailed analysis. A questionnaire was sent to 377 parents of
these children and replies were received from 271 (72%). The
questionnaire consisted of 26 questions relating to the gener-
al health of the child, the childs nutritional status, feeding
abilities, the influence of feeding impairment on the family,
and contact with health care professionals. The question-
naire was constructed in such a way that it was possible to
investigate the relationship between the degree of motor dis-
ability (e.g. mobility) and oral-motor impairment (e.g. drool-
ing and speech impairment) and difficulties encountered with
feeding. In the original study questionnaire (Sullivan et al.
2000), neurological impairment in participants was classified
Developmental Medicine & Child Neurology2002, 44: 461467 461
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according to type and mobility. Difficulty was graded as mild
(little or no difficulty walking), moderate (difficulty walking
but does not need aids or a helper), and severe (needs aids
and/or a helper or cannot walk).
In this follow-up study, one hundred of the 271 families
who returned the questionnaire were then randomly select-
ed to represent a subgroup of the study population. The ran-
dom selection was stratified so that equal numbers were
selected from each of the four counties. After giving their
consent, families of the subgroup (n=100) were visited athome by a research dietitian trained in qualitative research
techniques and anthropometry. The aims of the home visits
were (1) to confirm by interview answers given in the origi-
nal study questionnaire, (2) to undertake a detailed dietetic
analysis, and (3) to perform a comprehensive anthropomet-
ric assessment of each affected child.
Approval for this project was obtained from each of the
seven Clinical Research Ethics Committees covering the geo-
graphical area under study. Permission to use the ORECI
database was obtained by the ORECI Management Group.
After consent from the participants family doctors and in-
formed consent from parents/caregivers had been obtained,
the study questionnaire (which also asked whether or notthey would give consent to be visited at home) was sent to
parents/caregivers of children in order to characterize the
nature and extent of the feeding disability.
DIETETIC ASSESSMENT
Dietary information was obtained from parents in two ways.
Firstly, a verbal dietary history was taken to establish the childs
normal nutritional intake. This included details of foods and
drinks typically eaten, quantities consumed, feeding habits
and methods, information about items of food and drink
taken on a more irregular basis, and a 24-hour dietary recall by
a dietitian during a home visit. Secondly, all parents were
asked to keep a 3-day dietary diary (Gersovitz et al.1978,
Acheson et al. 1980). This diary was specifically designed forthe study and had simple written instructions to aid comple-
tion. Parents also received detailed verbal instructions on how
to fill in the diary and they were encouraged to write down as
much information as possible about the foods and drinks pre-
sented to and consumed by the child. Detailed information
was requested on food brand names used, methods of cook-
ing, and preparation and food consistency. Estimates of the
amounts of food or drink (those actually ingested by the child
as well as those offered in an attempt to get some idea of food
loss through spillage) were recorded using handy household
measures and weights (e.g. cups, fluid ounces, tablespoons).
Other data collected included the time in minutes taken for
the child to consume each meal, snack, or drink and the extentto which the child was able to self-feed. Two weekdays and one
weekend day were used in the recording period to allow for
any potential differences in food consumption patterns on
different days of the week. The verbal information obtained
during the visit and in particular the 24-hour dietary recall
was used to help validate the information in the 3-day diet
diary. For those children whose parents failed to return a
completed food diary, the 24-hour recall data were analyzed
and used to provide information on their nutritional intakes.
Dietary intake data were analyzed using proprietary soft-
ware (Dietplan 5, Forestfield Software Limited). Nutrient
intake was expressed as a percentage of the Department of
Health (1991) Dietary Reference Values for Food Energy and
Nutrients for the United Kingdom.
ANTHROPOMETRY
Detailed anthropometry was performed and included
measurement of body weight (Marsden Wheelchair Weigher,
Marsden Weighing Machine Group Ltd., London), upper arm
and lower leg length (Harpenden Anthropometer, Child
Growth Foundation, London), mid-upper arm circumference
(MUAC; Harpenden Anthropometric tape), triceps and sub-scapular skinfold thickness (skinfold callipers, Holtain Ltd.,
Crymmych, UK), and occipito-frontal circumference (Lasso
Headtape Measure, Child Growth Foundation, UK). To reduce
the potential for measurement error, all measurements were
performed by the same trained operator and each measure-
ment was repeated three times consecutively and the mean
value calculated. Accurate length and height measurements
are difficult to obtain in children with disabilities because of
limb contractures and scoliosis. For this reason, we chose to
record upper arm length and lower leg length (Spender et al.
1989) as measures of linear growth. In order to identify possi-
ble differences in limb length and body symmetry in those
children with a right or left-sided hemiplegia, measurementswere taken from both the right and left sides of the body.
However, to make the Table readable, we only present results
for left-sided measurements (correlation between left and
right-sided measurements was very high: the lowest Pearsons
correlation coefficient was 0.92 for left and right upper arm). In
order to facilitate internal and external comparisons adjusting
for the known confounding effects of age and sex, measure-
ments were expressed as standardizedzscores of available age-
and sex-specific reference population standards (a negativez
score indicates below average). For weight and occipito-frontal
circumference, measurements were standardized to the 1990
British Growth reference centiles (Freeman et al. 1995), upper
arm and lower leg length measurements were standardized
to American reference standards for cerebral palsy as none areavailable for British children (Spender et al. 1989), and mid
upper arm circumference and skinfold measurements were
standardized to American data (Frisancho 1988).
STATISTICAL ANALYSIS
Descriptive and statistical analyses were performed using
SPSS (version 10.0) and STATA (version 6.0). The continuous
variables of age, weight, and standardized z scores were
investigated for departure from normality both informally,
by assessing the shape when looking at a histogram, and for-
mally, using the Shapiro-Wilk test for normality. The majority
of children were in the lowest centile for their measurements
and, therefore, the data were, by nature, skewed.Having examined the histograms and tests of normality, we
decided to present descriptive statistics appropriate for skewed
data, and perform non-parametric tests throughout to investi-
gate differences between the disability groups with respect to
demographic, ponderal (weight, MUAC, triceps and subscapu-
lar skinfold thickness), and skeletal measurements.
For the continuous variables of age and weight, summary
statistics including the median and the range are given (Table
II). In addition, for the standardizedzscores, we present the
proportion of children below a defined range expressed as a
percentage (Table II). The lower end of the defined range
was azscore of 1.96 (i.e. 2.5% of values were expected to lie
462 Developmental Medicine & Child Neurology 2002, 44: 461467
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below this level) and the denominator for this percentage
was as the number of valid measurements. We acknowledge
that 1.96 is an arbitrary cut-off (representing approximately
2SDs below mean) and that some children did indeed regis-
ter measurements above a zscore of +1.96; however, the
focus of this study is the lower end of the weight spectrum
and hence a one-sided aspect is presented here.
Comparison of age and standardized z scores across all
three disability groups was performed using the Kruskal-Wallis
(equality of populations rank) test. Post-hoc comparisons con-trasting mild and moderate disability combined versus severe
disability were also performed using the Kruskal-Wallis test,
equivalent to performing a Mann-WhitneyUtest.
For the binary variable, sex, the distribution of males and
females across the three disability groups was investigated
using the 2 test.
Results
The hundred children with disabilities visited in their homes
were evenly spread across the four counties of Berkshire,
Buckinghamshire, Northamptonshire, and Oxfordshire. In
the vast majority of cases, the caregiver was the childs own
mother (in four cases it was a foster mother). The mean ageof the children was 9 years (SD 2 years 5 months), ranging from
4 years 6 months to 13 years 7 months (40 females 60 males).
There was no statistically significant difference between the
disability groups with respect to the distributions of age and
sex, that is, the groups were well balanced for these charac-
teristics. Forty-eight children were unable to walk and 21
needed both aids and a helper to be mobile; these 69 children
were classified for the purposes of analysis as the severely dis-
abled group. Nineteen children had moderate difficulty with
walking, but no aids nor helpers were required for them to
be mobile (moderately disabled group). The remaining 12
children had either little or no difficulty with walking (mild-
ly disabled group).
Cerebral palsy (CP) was the reason for disability in 90children, of whom 64 (71%) had spastic quadriplegia. The
remainder of the diagnostic categories were as follows: glob-
al developmental delay (n=3), Marfan syndrome (n=1),
intractable epilepsy (n=2), agenesis of the corpus callosum
(n=2), methyl malonic aciduria (n=1), and congenital
rubella (n=1). In terms of responses to the study question-
naire, when compared with children in the total study popu-
lation (n=271), those children in the home visit study
(n=100) were not substantially different in terms of age,
sex, or geographical locality within the region. Nor were
they different in the important areas of motor impairment,
oral-motor impairment, feeding problems, and nutrition;
therefore, we consider them a representative sample of the
total population of children with CP with or without motor
dysfunction.
Table I shows the relationship between ability to self-feed
and disability and indicates that the majority of children with
a severe degree of motor impairment were unable to self-
feed and required help. All of the children with a mild to
moderate degree of motor impairment were able to be fed by
mouth and none always needed help with feeding.
DIETETIC ASSESSMENT
Fifty-three caregivers returned dietary diaries. The verbal
diet history information was used to provide information on
nutrient intakes in 41 participants. Six children, all with
severe disabilities, were given proprietary enteral feeds via
their gastrostomy tubes and in these cases nutritional intake
was calculated from information provided by the product
manufacturers.
Figure 1 shows nutrient intakes with respect to the per-
centage of children achieving over 100% of estimated aver-
age requirement (EAR). Nutrient intake analysis revealed
that energy intakes were generally low with only 20% of chil-
dren having energy intakes above the EAR for their age and59% of the group with severe disabilities consuming below
80% EAR compared with 16% of the group with moderate
disabilities, though this difference was not statistically signif-
icant (Kruskal-Wallis test comparing mild plus moderate vs
severe disability, p=0.09). Ninety-six out of 100 children,
however, achieved a protein intake above the EAR and this
was consistent across the three groups.
Mean intake of the micronutrients: sodium, potassium,
magnesium, phosphorus, copper, iodine, B1, B2, B6, and
B12, met reference nutrient intake (RNI) for all children and
for each disability group, but the ranges of intakes were
wide. It is noteworthy that iron, for instance, fell below the
normal range for nearly half the participants. Interestingly, in
22 out of 33 of the children with severe disabilities, milk ormilk-based drinks provided the highest proportion of energy
in their diets out of all the items they consumed. Milk is a
poor source of iron and may explain this finding together
with the observation that the majority of these children met
their RNIs for calcium, phosphorus, thiamin, riboflavin, B6,
and B12 more than adequately.
Half of the children with severe disabilities failed to meet at
least 81% of the RNI for potassium, iron, copper, magnesium,
and zinc. Selenium, vitamin A, niacin, and folate deficiencies
were also seen in moderate and mild disability groups.
Retinol (vitamin A) intakes were relatively low across all
the groups with only 26% of children meeting the RNI.
Nutritional Intake and Growth in DisabilityP B Sullivan et al. 463
Table I: Feeding ability with respect to degree of motor disability,n (%)
Severe disability Mild or moderate disability
(n=69) (n=31)
Always needs help 33 (48) 0 (0)
Some difficulty, needs help 15 (22) 23 (74)
Can feed but slow and messy, help given 12 (17) 2 (6.5)
Tube fed/not fed by mouth 6 (9) 0 (0)
Some difficulty, no help 2 (3) 4 (13)
Self feeds 1 (1) 2 (6.5)
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464 Developmental Medicine & Child Neurology 2002, 44: 461467
Table II: Summary statistics for continuous variables (zscores are standardized for age and sex): median, proportion (%) of
children more than 2SDs below mean (forzscores only) and range
Index All children Disability group
n=100 Mild Moderate Severe
n=12 n=19 n=69
Age (y) 9.04 9.63 9.33 8.92
(4.5 to 13.6) (6.2 to 12.8) (6.4 to 13.3) (4.5 to 13.6)
Weight (kg) 22.60 29.45 25.90 20.40
(11.1 to 66.4) (12.7 to 43.5) (17.8 to 66.4) (11.1 to 51.2)
Weight,zscorea 1.43d (38%) 0.31 (8%) 0.62 (16%) 1.87f(49%)
(10.2 to 2) (4.8 to 1.6) (2.1 to 2) (10.2 to 1.7)
Occipito-frontal measurement,zscorea 1.92 (48%) 0.45 (25%) 1.76 (33%) 2.26h (56%)
(7.3 to 1.9) (3.3 to 0.8) (3 to 0.8) (7.3 to 1.9)
Upper arm length,zscoreb 0.000d (6%) 0.55 (0%) 0.90 (0%) 0.35f(9%)
(3.5 to 4.7) (1.5 to 2.2) (0.8 to 2.9) (3.5 to 4.7)
Lower leg length,zscoreb 1.27d (36%) 0.000 (0%) 0.27 (5%) 2.08f(52%)
(5.9 to 3.6) (1.4 to 1.8) (2.1 to 3.6) (5.9 to 2.8)
Mid upper arm circumference,zscorec 0.69 (9%) 0.39 (0%) 0.70 (0%) 0.74 (13%)
(3.5 to 2.4) (1.4 to 1.9) (1.5 to 2.1) (3 to 2.4)
Triceps skinfold,zscorec 0.63e (1%) 0.25 (0%) 0.25 (0%) 0.89g (2%)
(2.1 to 2.9) (1.5 to 2) (1.2 to 2.3) (2.1 to 2.9)
Subscapular skinfold,zscorec 0.29 (0%) 0.18 (0%) 0.063 (0%) 0.40h (0%)
(1.2 to 3.9) (0.8 to 1.9) (0.8 to 1.7) (1.2 to 3.9)
aReference population,1990 British Growth reference centiles (Freeman et al. 1995); bReference population, Spender et al. 1989;cReference population, Frisancho 1988; dp0.001; ep0.05; results of Kruskal-Wallis test across all 3 disability groups; fp0.001; gp0.01;hp
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ANTHROPOMETRY
Table II shows summary statistics for the measurements re-
corded for the whole group of participants and with respect to
level of motor disability. Figures 2 to 5 examine the relation-
ship between level of motor dysfunction and standardized
body weight, lower leg length, upper arm length, and occipi-
to-frontal measurement (head circumference) respectively,
using dotplots. Dotplots present all the observations, in this
casezscores by disability group, with the added features of a
horizontal line representing the mean value for each refer-ence population (i.e. zscore=0), and short horizontal line
indicating the median value for each disability group.
Over three-quarters of children in this study group were
below mean weight for their age and sex when compared
with the standards for children without disabilities and this
deficit was most marked in those with severe motor disability
where their median value approximates to 2SDs below the
reference mean value (Fig. 2).
Linear growth, as measured by limb length, was similarly
impaired and it is noteworthy that the compromise in linear
growth was more marked in the lower limbs and almost exclu-
sively in the severe disability group. For lower leg length, the
mild and moderate disability groups had typically normal
measurements, while in the severe disability group, half weremore than 2SDs below the reference mean value (Fig. 3).
In the upper limb, around three-quarters of the mild and
moderate disability groups were above the reference popula-
tion mean (Fig. 4), whereas in the severe disability group, the
Nutritional Intake and Growth in DisabilityP B Sullivan et al. 465
Figure 2:Dotplot of bodyweight z score by disability group.
Line indicates median value for each disability group.
Figure 3:Dotplot of lower leg length z score by disability
group. Line indicates median value for each disability group.
Figure 4:Dotplot of upper arm length z score by disability
group. Line indicates median value for each disability group.
Figure 5:Dotplot of head circumference z score by disability
group. Line indicates median value for each disability group.
Mild Moderate Severe
Disability Group
4
3
2
1
0
1
2
3
4
5
6
Lowerle
glengthz
score
Mild Moderate Severe
Disability Group
3
2
1
0
1
2
34
5
6
7
Headcircu
mferencez
score
Mild Moderate Severe
Disability Group
3
2
1
0
12
3
4
5
6
7
8
9
10
Weightz
scor
e
Mild Moderate Severe
Disability Group
5
4
3
2
1
0
1
2
3
4
Upperarmlengthz
score
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medianzscore was 0.35 when compared with the Spender
(1989) reference population. It should be remembered here
that this reference population for linear growth is a CP popula-
tion (Spender 1989) and thus this growth distinction between
mobile and immobile children with CP is all the more marked.
Head growth (Fig. 5) was also reduced in children with
neurological impairment and worsened with severity of
motor disability such that the medianzscores for those with
mild, moderate, or severe motor disability were 0.5, 1.8,
and 2.3, respectively.Another way of looking at the measurement of head cir-
cumference is that, overall, nearly half of all children (n=48)
were below our defined threshold of 2SDs below the mean,
with a trend according to severity of disability: 1 in 4 for the
mild disability group, 1 in 3 for the moderate disability group,
and 1 in 2 for those with severe motor impairment.
Ponderal measurements, that is, weight, MUAC, triceps
and subscapular skinfold thickness, were most markedly atyp-
ical in proportion to the degree of motor disability; weight for
age, for instance, was below the reference range in 8%, 16%,
and 49% of participants for the mild, moderate, and severe dis-
ability groups, respectively (Table II). That is, for the most
severely disabled group, we would expect 2.5% of observationsto lie below our defined range, however, 49% of observations
were below this level. Moreover, when post-hoc comparisons
were made forzscores across disability groups for the ponder-
al measurements, the difference between mild and moderate
combined versus severe disability groups was statistically sig-
nificant for weight (p
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Effect of severity of motor impairment on growth and
nutritional intake
Although we did not use a standard scale of mobility but
rather based our classification on the answers given in the
questionnaire in our original study (Sullivan 2000), we were
nevertheless able to identify clearly those children with the
most severe degree of motor disability. In a recent prospec-
tive follow-up of 321 children born with bilateral CP born in
London between 1989 and 1992 (Lin et al. 1999), only 38%
attained unsupported walking. Using the classification in ourstudy, the figure was very similar with only 31% being able to
walk unsupported.
Conclusion
This study has confirmed the significant impact of neurologi-
cal impairment in children on body growth and nutritional
status. Moreover, it has demonstrated that these deficits are
worse in those with a greater degree of motor impairment.
The major nutritional deficit was in energy intake, whilst
micronutrient intakes were less markedly impaired and pro-
tein intake was normal in this group of children. Many chil-
dren with neurological impairment, regardless of the degree
of disability, would benefit from individual nutritional assess-ment and management as part of their overall care. Dietitians
have a very important role in ensuring that this is achieved and
should be actively involved in the process of nutritional assess-
ment. Further research is required to determine the appropri-
ate energy intake required to maintain energy balance in
children with neurological impairment.
Accepted for publication 10th January 2002.
AcknowledgementsThe authors are grateful to the managing committee of the OxfordRegister of Early Childhood Impairment (ORECI) for permission touse the database. This study was made possible by a grant from the
Anglia and Oxford Research and Development Committee.
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