Feeding interventions for growth and development in infants

with cleft lip, cleft palate or cleft lip and palate (Protocol)

Glenny AM, Hooper L, Shaw WC, Reilly S, Reid J

This is a reprint of a Cochrane protocol, prepared and maintained by The Cochrane Collaboration and published in The Cochrane
Library 2001, Issue 3
http://www.thecochranelibrary.com

Feeding interventions for growth and development in infants with cleft lip, cleft palate or cleft lip and palate (Protocol)
Copyright © 2004 The Cochrane Collaboration. Published by John Wiley & Sons, Ltd.
 TABLE OF CONTENTS
BACKGROUND . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 1
OBJECTIVES . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 2
CRITERIA FOR CONSIDERING STUDIES FOR THIS REVIEW . . . . . . . . . . . . . . . . . . 2
SEARCH STRATEGY FOR IDENTIFICATION OF STUDIES . . . . . . . . . . . . . . . . . . . . 2
METHODS OF THE REVIEW . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 3
POTENTIAL CONFLICT OF INTEREST . . . . . . . . . . . . . . . . . . . . . . . . . . . 4
SOURCES OF SUPPORT . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 4
REFERENCES . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 4
COVER SHEET . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 5

Feeding interventions for growth and development in infants with cleft lip, cleft palate or cleft lip and palate (Protocol) i
Copyright © 2004 The Cochrane Collaboration. Published by John Wiley & Sons, Ltd
Feeding interventions for growth and development in infants
with cleft lip, cleft palate or cleft lip and palate (Protocol)
Glenny AM, Hooper L, Shaw WC, Reilly S, Reid J
This record should be cited as:
Glenny AM, Hooper L, Shaw WC, Reilly S, Reid J. Feeding interventions for growth and development in infants with cleft lip,
cleft palate or cleft lip and palate. (Protocol) The Cochrane Database of Systematic Reviews , Issue . Art. No.: CD003315. DOI:
10.1002/14651858.CD003315.
This version first published online: 23 July 2001 in Issue , .
Date of most recent substantive amendment: 01 April 2001
BACKGROUND
Cleft lip and cleft palate are common birth defects, between them
affecting about one baby of every 700 born. The Cleft Lip and
Palate Association in the UK (CLAPA 2001) define cleft lip as ’an
opening in the upper lip between the mouth and the nose... it can
range from a slight notch in the coloured portion of the lip to
complete separation in one or both sides of the lip extending up
and into the nose’. They explain cleft palate as occurring when ’the
roof of the mouth is not joined completely....[this can] range from
just an opening at the back of the soft palate to a nearly complete
separation of the roof of the mouth (soft and hard palate)’. These
two conditions may arise early in pregnancy as an unborn baby
is developing, and may occur independently or together (in cleft
lip and palate). The cleft may be on one side (unilateral) or both
sides (bilateral).
Because the lip and/or palate are affected, feeding of the baby is an
immediate concern and there is evidence of delay in growth of cleft
children as compared to those without clefting. A cleft lip may
be problematic in creating a seal around the nipple, but closure
can still generally be obtained. A cleft palate, however, prevents
the baby from creating negative pressure necessary in feeding and
may also lead to breathing problems during feeding. At the very
least this lengthens feeding times considerably. Clefting of the hard
palate also limits the normal use of the tongue to compress the
nipple (Shprintzen 1995).
Avedian 1980 found that though cleft babies were born on the
30th weight centile on average, they had fallen to the 20th centile
at one and two months of age, achieving the 30th centile again
only at six months. Jensen 1983 studied Danish boys aged six to
20 years with clefts and compared them with a control group,
finding delayed skeletal maturity in boys with clefts over the whole
period. Jones 1988 found that weight gain per week was lower in
Feeding interventions for growth and development in infants with cleft lip, cleft palate or cleft lip and palate (Protocol)
Copyright © 2004 The Cochrane Collaboration. Published by John Wiley & Sons, Ltd
neonates with clefts than without. More recently Lee 1997 found
that clefting was associated with significant growth faltering in
early infancy, but that children had attained expected weight and
height by about two years. Clefting has also been associated with
increased risks of failure to thrive (Marcovitch 1994) and severe
dehydration (Livingstone 2000).
There appear to be additional developmental delays in some chil-
dren with cleft lip and/or palate, which may also be potentially
linked to nutritional status early in life. Jocelyn 1996 found that
cleft children had significantly lower scores on tests of cognition,
comprehension and expressive language abilities than matched
control children at 12 and 24 months. Neiman 1997 found that at
36 months cleft toddlers showed significantly lower developmen-
tal performance in fine motor, gross motor and expressive language
skills compared with non-cleft toddlers. Maternal attachment may
also be affected (Speltz 1990).
In an effort to combat reduced weight for height, a variety of advice
and devices are recommended to aid feeding in babies with clefts.
These include a variety of modified bottles and teats (Clarren 1987;
CLAPA 2001), measures supplemental to breastfeeding (Clarren
1987; Wide Smiles 2001), obturating plates (Chen 1990; Balluff
1986; Nagda 1996; Kogo 1997) and advice or training to parents
(Richard 1991; Danner 1992). (An obturating plate is an acrylic
appliance placed over the gums of the babies upper jaw so that
the cleft between the mouth and nose is covered. They may be
passive, or occasionally are used to adjust the relationship between
the jaw segments). The methods suggested vary with cleft type
and severity (Clarren 1987), from centre to centre (Shaw 1999),
as well as pre- and post-operatively.
This review aims to assess the effects of these feeding interventions
in babies with cleft lip and /or palate on growth, development and
parental satisfaction.
1
OBJECTIVES
This review aims to compare the effects of differing interventions
such as maternal advice and support, modified bottles and/or teats,
obturating plates and supplemental breastfeeding in:
• babies with cleft lip prior to closure
• babies with cleft palate prior to closure
• babies with cleft lip and palate prior to closure
• babies following cleft lip closure
• babies following cleft palate closure.
Interventions in the first six months from term and used with
breast milk or formula feeding only will be considered, but out-
comes (measures of growth, development and parental satisfac-
tion) may be measured at any time including adulthood.
CRITERIA FOR CONSIDERING
STUDIES FOR THIS REVIEW
SEARCH STRATEGY FOR
IDENTIFICATION OF STUDIES
See: search strategy
The search will attempt to identify all relevant studies irrespective
of language. Papers not in English will be considered if they can
be translated.
Electronic searching - The databases to be searched will include:
Cochrane Central Register of Controlled Trials (CENTRAL)
Cochrane Oral Health Group Specialised Register
MEDLINE (from 1966)
EMBASE (from 1980)
CINAHL (from 1982)
Psych Info (from 1967)
AMED (from 1985)
Sensitive search strategies will be developed for each database
(available from the authors on request) using a combination of
controlled vocabulary and free-text around the following search
terms: cleft lip, cleft palate, harelip, mouth abnormalities, bottle
feeding, breast feeding, breastfeeding, infant nutrition.

The search strategy for MEDLINE (Ovid) is given as an example

Types of studies below:

Controlled clinical trials (randomised and non-randomised) will 1 (cleft$ adj5 (lip$ or palat$ or oral or orofacial)).mp. [mp=title,
be included. abstract,
registry number word, mesh subject heading]
Types of participants 2 harelip$.mp. [mp=title, abstract, registry number word,
Babies born with cleft lip, cleft palate or cleft lip and palate up to mesh subject heading]
the age of six months (from term). The babies may be waiting for 3 hare-lip$.mp. [mp=title, abstract, registry number word,
an operation to close the cleft, recovering from one, or between mesh subject heading]
operations. Babies with Pierre Robin sequence or other syndromes 4 exp Cleft lip/
will be not be included in the review. 5 exp Cleft palate/
6 *Mouth abnormalities/
Types of intervention 7 1 or 2 or 3 or 4 or 5 or 6
Interventions may include modified bottles, cups, spoons, pumps, 8 breastfe?d$.mp. [mp=title, abstract, registry number word,
positions, techniques and/or teats (supplemental to breastfeeding mesh subject heading]
or instead of breastfeeding), obturating plates or maternal advice 9 bottlefe?d$.mp. [mp=title, abstract, registry number word,
and support (during breast or bottle feeding). Any intervention mesh subject heading]
can be compared to any other or to un-modified equipment or 10 breast-fe?d$.mp. [mp=title, abstract, registry number word,
advice. mesh subject heading]
11 bottle-fe?d$.mp. [mp=title, abstract, registry number word,
Types of outcome measures mesh subject heading]
Outcomes measured may include 12 (fe?d$ adj5 (bottle$ or breast)).mp. [mp=title, abstract,
registry number word, mesh subject heading]
• measures of growth (weight until age 16, growth centiles to age 13 exp Infant nutrition/
16, head circumference upto 12 months), 14 teat$.mp. [mp=title, abstract, registry number word, mesh
• development (standard childhood development indices, em- subject heading]
ployment or marital status in adulthood), 15 (plate$ adj5 obturat$).mp. [mp=title, abstract, registry
number word, mesh subject heading]
• parental satisfaction (assessment of confidence with feeding, or 16 “feeding plate$”.mp. [mp=title, abstract, registry number
that the child is getting enough feed, time taken feeding). word, mesh subject heading]
Feeding interventions for growth and development in infants with cleft lip, cleft palate or cleft lip and palate (Protocol) 2
Copyright © 2004 The Cochrane Collaboration. Published by John Wiley & Sons, Ltd
17 *Feeding behavior/
18 *Feeding methods/
19 8 or 9 or 10 or 11 or 12 or 13 or 14 or 15 or 16 or 17 or 18
20 7 and 19
In addition to the handsearching already being carried out by the
Cochrane Collaboration (see master list www.cochrane.org) the
following journals will be handsearched;
• Cleft Palate Journal (1964 (vol 1) to 1990); name changed to
Cleft Palate - Craniofacial Journal (1991 onwards)
• Journal of Maxillofacial Surgery (1973 (vol 1) to 1986); name
changed to Journal of Cranio-Maxillo-Facial Surgery (1987
onwards)
• British Journal of Plastic Surgery (1966 (vol 19) onwards)
• Plastic and Reconstructive Surgery (1966 (vol 38) onwards)
Experts in the field will be approached to identify both
unpublished and ongoing studies. The National Research
Register, National Institutes of Health, Clinical Trial Service
Unit, Medical Research Council and Medical Editors Trial
Amnesty registers will all be searched through the Current
Controlled Trials webpage (http://www.controlled-trials.com).
Searches of SIGLE (System for Information on Grey Literature in
Europe), the International Register of Perinatal Trials, and CRISP
(Computer Retrieval of Information on Scientific Projects) will
also be undertaken.
Reference lists of all relevant review articles and trials identified
for inclusion in the systematic review will be screened to identify
any additional studies.
METHODS OF THE REVIEW
Study selection
The titles and abstracts (when available) of all reports identified
through the electronic searches will be scanned independently by
two reviewers. For studies appearing to meet the inclusion criteria,
or for which there is insufficient data in the title and abstract
to make a clear decision, the full report will be obtained. The
full reports obtained from all the electronic and other methods
of searching will be assessed independently by two reviewers to
establish whether the studies meet the inclusion criteria or not.
Disagreements will be resolved by discussion. Where resolution is
not possible, a third reviewer will be consulted. All studies meeting
the inclusion criteria will then undergo validity assessment and
data extraction. Studies rejected at this or subsequent stages will be
recorded in the table of excluded studies, and reasons for exclusion
recorded.
Quality assessment
Feeding interventions for growth and development in infants with cleft lip, cleft palate or cleft lip and palate (Protocol)
Copyright © 2004 The Cochrane Collaboration. Published by John Wiley & Sons, Ltd
The quality assessment of the included trials will be undertaken
independently and in duplicate by the two reviewers as part of the
data abstraction process.
Four main quality criteria will be examined;
1) Randomisation procedure, recorded as
Truly (computer generated random numbers, coin toss, etc...)
Quasi (allocation according to birth-date, patient number, etc...)
Unclear/not stated
Not done
2) Allocation concealment, recorded as;
(A) Adequate
(B) Unclear
(C) Inadequate
(D) Not used
as described in the Cochrane Reviewer’s Handbook.
3) Blind outcome assessment
4) Completeness of follow-up and intention to treat analysis
Further quality assessment will be carried out to assess definition of
exclusion/inclusion criteria, adequate definition of success criteria
and comparability of control and treatment groups at entry. The
quality assessment criteria will be pilot tested using several articles.
Data Extraction
Data will be extracted by two reviewers independently using
specially designed data extraction forms. The data extraction
forms will be piloted on several papers and modified as required
before use. Any disagreement will be discussed and a third
reviewer consulted where necessary. Authors will be contacted for
clarification or missing information whenever possible. Data will
be excluded until further clarification is available if agreement
cannot be reached.
For each trial the following data will be recorded;
• Date of the study, year of publication, and country of origin.
• Details of the participants including demographic
characteristics, source of recruitment and criteria for inclusion.
• Details on the type and duration of intervention
• Details of the outcomes reported, including method of
assessment (where measurement scales are used it will be
recorded whether or not they have been validated).
Data Synthesis
For dichotomous outcomes, the estimate of effect of an
intervention will be expressed as relative risks together with
95% confidence intervals. For continuous outcomes, means and
standard deviations will be used to summarise the data for each
group.
Clinical heterogeneity will be assessed by examining the types
of participants, interventions and outcomes in each study. Only
if there are studies of similar comparisons reporting the same
3
outcome measures will meta-analysis be attempted. Relative risks
will be combined for dichotomous data, and standardised mean
differences for continuous data, using a fixed effects model.
The significance of any discrepancies in the estimates of the
treatment effects from the different trials will be assessed by means
of Cochran’s test for heterogeneity. If any significant statistical
heterogeneity (P<0.1) is detected, it is planned to re-assess the
significance of the treatment effects by using a random effects
model.
Sensitivity analyses will be undertaken to examine the effect
of randomisation, allocation concealment and blind outcome
assessment on the overall estimates of effect. In addition, the effect
of including unpublished literature on the review’s findings will
also be examined.
Where possible, subgroup analyses will be undertaken to compare:
results for cleft lip only (pre- and post-closure), cleft palate only
(pre- and post-closure) and cleft lip and palate.

POTENTIAL CONFLICT OF
INTEREST

None known

SOURCES OF SUPPORT

External sources of support

• No sources of support supplied

Internal sources of support
• University Dental Hospital of Manchester UK

REFERENCES

Additional references Clarren 1987

Avedian 1980 Clarren SK, Anderson B, Wolf LS. Feeding infants with cleft lip,
Avedian LV, Ruberg RL. Impaired weight gain in cleft palate infants. cleft palate or cleft lip and palate. Cleft Palate Journal 1987;24(3):
Cleft Palate Journal 1980;17(1):24-26. 244-249.
Danner 1992
Balluff 1986 Danner SC. Breastfeeding the infant with a cleft defect. NAACOGS
Balluff MA, Udin RD. Using a feeding appliance to aid the infant Clinical Issues in Perinatal & Womens Health Nursing 1992;3(4):634-
with a cleft palate. Ear, Nose and Throat Journal 1986;65(7):316-320. 639.
Chen 1990 Jensen 1983
Chen HJ, Wang CH, Wang CC, Shieh TY. [A modified technique of Jensen BL, Dahl E, Kreiborg S. Longitudinal study of body height,
obturator fabrication for cleft palate infants] [Chinese]. Kao-Hsiung radius length and skeletal maturity in Danish boys with cleft lip and
i Hsueh Ko Hsueh Tsa Chih [Kaohsiung Journal of Medical Sciences] palate. Scandinavian Journal of Dental Research 1983;91(6):473-481.
1990;6(10):546-550.
Jocelyn 1996
CLAPA 2001 Jocelyn LJ, Penko MA, Rode HL. Cognition, communication and
Cleft Lip, Palate Association. Questions and answers. CLAPA website hearing in young children with cleft lip and palate and in control
2001. children: a longitudinal study. Pediatrics 1996;97(4):529-534.
Feeding interventions for growth and development in infants with cleft lip, cleft palate or cleft lip and palate (Protocol) 4
Copyright © 2004 The Cochrane Collaboration. Published by John Wiley & Sons, Ltd
Jones 1988
Jones WB. Weight gain and feeding in the neonate with cleft: a three-
center study. Cleft Palate Journal 1988;25(4):379-384.
Kogo 1997
Kogo M, Okada G, Ishii S, Iida S, Matsuya T. Breast feeding for
cleft lip and palate patients, using the Hotz-type plate. Cleft Palate-
Craniofacial Journal 1997;34(4):351-353.
Lee 1997
Lee J, Nunn J, Wright C. Height and weight achievement in cleft lip
and palate. Archives of Disease in Childhood 1997;76(1):70-72.
Livingstone 2000
Livingstone VH, Willis CE, Abdel-Wareth LO, Thiessen P, Lock-
itch G. Neonatal hypernatremic dehydration associated with breast-
feeding malnutrition: a retrospective survey. Canadian Medical Asso-
ciation Journal 2000;162(5):647-652.
Marcovitch 1994
Marcovitch H. Fortnightly review: failure to thrive. British Medical
Journal 1994;308(6920):35-38.
Nagda 1996
Nagda S, Deshpande DS, Mhatre SW. Infant palatal obturator. Jour-
nal of the Indian Society of Pedodontics & Preventive Dentistry 1996;
14(1):24-25.
Neiman 1997
Neiman GS, Savage HE. Development of infants and toddlers with
clefts from birth to three years of age. Cleft Palate-Craniofacial Journal
1997;34(3):218-225.
Richard 1991
Richard ME. Feeding the newborn with cleft lip and/or palate: the
enlargement, stimulate, swallow, rest (ESSR) method. Journal of Pe-
diatric Nursing: Nursing Care of Children and Families 1991;6(5):
317-321.
Shaw 1999
Shaw WC, Bannister RP, Roberts CT. Assisted feeding is more reliable
for infants with clefts–a randomized trial. Cleft Palate-Craniofacial
Journal 1999;36(3):262-268.
Shprintzen 1995
Shprintzen RJ, Bardach J. Cleft Palate Speech Management: a multi-
disciplinary approach. First Edition. St. Louis: Mosby, 1995.
Speltz 1990
Speltz ML, Armsden GC, Clarren SS. Effects of craniofacial birth
defects on maternal functioning post-infancy. J Pediatr Psychol 1990;
15(2):177-195.
Wide Smiles 2001
Wide Smiles. Breastfeeding the cleft-affected newborn and Special
considerations of the bottle-fed baby with a cleft. Wide Smiles web
site (www.widesmiles.org) 2001.

COVER SHEET
Title Feeding interventions for growth and development in infants with cleft lip, cleft palate or
cleft lip and palate
Authors Glenny AM, Hooper L, Shaw WC, Reilly S, Reid J
Contribution of author(s) Information not supplied by author
Issue protocol first published 2001/4
Date of most recent amendment 25 February 2004
Date of most recent 01 April 2001
SUBSTANTIVE amendment
Review expected to be published in Issue 3, 2004
What’s New Information not supplied by author
DOI 10.1002/14651858.CD003315
Cochrane Library number CD003315
Editorial group Cochrane Oral Health Group
Editorial group code HM-ORAL

Feeding interventions for growth and development in infants with cleft lip, cleft palate or cleft lip and palate (Protocol) 5
Copyright © 2004 The Cochrane Collaboration. Published by John Wiley & Sons, Ltd