The Burden of Care for Children With Unilateral Cleft

Lip: A Systematic
Review of Revision Surgery

Dipresentasikan : Dionesia Kidi Making S. Ked

Pembimbing : dr. Alders A. K. Nitbani, Sp.B

A Systematic Review and Meta-Analysis

Thomas J. Sitzman, M.D., Sarah M. Coyne, B.S., Maria T. Britto, M.D., M.P.H.
ABSTRACT

To identify the
average rate of Two investigators
revision surgery independently screened all
following cleft lip abstracts and determined
repair. eligibility from review of full
manuscripts using
prespecified inclusion and
exclusion criteria.
ABSTRACT
Results: The search identified 3034 articles. Of those, 45
met the inclusion criteria. Studies were primarily case
series and retrospective cohort studies, with only one
randomized controlled trial.

One-third of studies (n=15) did not describe how the

study sample was selected. Follow-up duration was not
reported in one-fourth of studies (n=11).

Nasolabial aesthetics were reported in 44% of studies (n=20).

The incidence of revision surgery ranged from 0% to 100%.
Meta-analysis was precluded because of study heterogeneity
(I2 = 97%).
ABSTRACT
METHODS
 Using the Institute of
Medicine (IOM) Standards for
Systematic Reviews (2011),
methods of study identification,
inclusion criteria, and analysis
were specified in advance and
documented in a protocol
(protocol available from the
authors on request).
 Search Strategy
An electronic search of PubMed (1946 to 2013),
CINAHL (1960 to 2013), and SCOPUS (1823 to
2013) was performed from database inception
through March 2013.
Search terms were cleft lip and surgery. The
search was restricted to studies of infants,
children, or adolescents.
Reference lists from included studies were
visually searched for additional studies of interest
by one reviewer.
 Eligibility Criteria
Inclusion criteria were published studies reporting
incidence of cleft lip revision surgery after primary
unilateral cleft lip repair in patients younger than 18
years.
Exclusion criteria were nonEnglish-language articles,
review articles, case series with fewer than 20 patients,
duplicate publications of the same patient cohort, and
manuscripts not available after extensive search by a
medical librarian.
Case series with fewer than 20 patients were excluded
because the estimate of revision surgery in such small
series has limited precision.
 Study Selection
The screening process is summarized in Figure 1.
Citations were screened on title and abstract
against the inclusion and exclusion criteria.
Studies reporting any outcome measure were
included at the initial screen to prevent excluding
studies that reported incidence of revision
surgery as a secondary outcome.
Final assessment for eligibility was determined by
review of full articles. Reports were not blinded
for authorship or affiliation.
Flow diagram of study selection. When a study included distinct patient cohorts,
incidence of revision surgery in each cohort was collected
 Data Extraction
Data were extracted from included studies
using a piloted form. A second investigator
independently reviewed the data extraction
for accuracy and completeness.
Authors of primary studies were not
contacted to provide missing or additional
data.
 Assessment of Study Strengths and
Limitations
Included studies were evaluated on timing of
outcome assessment, method of sample
selection, and follow-up duration.
All studies were graded using the Oxford Center
of Evidence-Based Medicine classification
(OCEBM Levels of Evidence Working Group,
2011).
Potential for selection bias was evaluated by
contrasting studies with relatively high and low
rates of revision surgery.
 RESULTS
Study Selection
The database search strategy identified 3034
articles. Forty-five articles met the
prespecified inclusion criteria. From these 45
studies, data were extracted for 62 unique
patient cohorts. Included studies are
summarized in Table 1.
 Incidence of Cleft Lip Revision
The incidence of cleft lip revision ranged from 0%
to 100% (Table 1).
There was substantial variation in incidence of
revision among the included studies (e.g.,
heterogeneity), which was confirmed by the I
test (I = 97%).
This heterogeneity persisted even within the
subgroups of cleft lip only (I =92%) and cleft lip
with cleft palate (97%).
This heterogeneity precluded metaanalysis.
Instead, we focused on qualitative synthesis.
 DISCUSSION
The burden of care for a patient with cleft lip includes
both primary repair and potentially multiple revision
procedures.
The results of this systematic review show the burden
of revision procedures for cleft lip varied broadly within
the literature, ranging from 0% to 100%.
Incidence of revision in the three largest patient
cohorts with followup beyond 1 year was 28%
(Abyholm et al., 1981), 30% (Henkel et al., 1998), and
36% (Mackay et al., 1999). Incidence of revision in the
sole randomized controlled trial was 24% (Bongaarts et
al., 2006).
 DISCUSSION
This review raises the question of why the incidence of
revision surgery varies so profoundly between centers.
Results of this study suggest that differences in follow-
up durationmay explain part of the variation; all of the
studies that reported a revision surgery incidence
greater than 75% evaluated patients at skeletal
maturity, while among the studies that reported a
revision surgery incidence less than 10%, follow-up
duration was less than 1 year or not reported in 27%.
 DISCUSSION
Health literacy, socioeconomic status, cultural
preferences, insurance status, and access to
health care likely also affect the use of revision
surgery, affecting whether surgeons offer
revision surgery, whether patients request
revision surgery, and whether patients and
families agree to undergo revision surgery
 DISCUSSION
Several authors have suggested that a reduced
incidence of revision surgery can be achieved
through improved technique, experience,
and/or dentofacial orthopedics
 Implications for Future Research
The findings of this review have important implications for future
research. The limitations of the existing literature demonstrate that
the burden of surgical care for patients with cleft lip remains poorly
understood.
Alternative approaches are necessary to determine the true
burden of care. The ideal approach would be a prospective
longitudinal observational cohort study of children undergoing cleft
lip repair.
Alternatively, a longitudinal cohort could be assembled
retrospectively from a population-based birth defects registry, a
cleft or craniofacial patient registry, or a national database in which
the unit of analysis is the individual.
The results of such a longitudinal cohort study, whether
prospectively and retrospectively assembled, would improve our
understanding of the patients burden of care.
 Implications for Surgeons
The surgeon directly affects the incidence of
revision surgery through the quality of their
initial repair. For surgeons looking to reduce
the need for revision, an important finding of
this review is that vermillion asymmetries are
the most common indication for revision of
the cleft lip. There is an anatomic basis for
these vermillion asymmetries; vermillion
height asymmetries 1 mm between the
medial and lateral lip height
 Implications for Surgeons
occur in 89% of patients with unilateral cleft lip
(Sitzman and Fisher, 2013). Techniques for correcting
the natural asymmetry in vermillion height have been
discussed (Millard, 1960; Noordhoff, 1984; Stal and
Hollier, 2002; Stal et al., 2009), yet a minority of
surgeons routinely employ them (Sitzman et al., 2008).
The nuance to choosing the point of closure on the
lateral lip element has also been highlighted
(Noordhoff, 1984; Losee et al., 2003). The high
prevalence of vermillion asymmetries at revision
surgery suggests that changing the surgical approach to
closing the vermillion may improve results of primary
surgery and thus reduce the need for lip revision.
 CONCLUSIONS
This systematic review of cleft lip surgery demonstrates
substantial variation in the incidence of revision
surgery.
For patients, this review suggests it is not possible to
accurately estimate the burden of care for their cleft
lip.
A logical next step would be a national assessment of
variation in cleft outcomes, including the burden of
care, using a populationbased birth defects registry or
a longitudinal national database in which the unit of
analyses is the individual
 CONCLUSIONS
Participation of cleft surgeons would be
essential to these efforts, as surgeons play a
central role in the quality of the initial repair
and the subsequent evaluation for revision.
Obtaining a more thorough understanding of
the sources for variation in revision surgery
may illuminate opportunities to reduce the
burden of surgical care and improve patient
outcomes.