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Cleft Palate Craniofac J. Author manuscript; available in PMC 2021 July 01.
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Published in final edited form as:

Cleft Palate Craniofac J. 2020 July ; 57(7): 860–871. doi:10.1177/1055665620902883.

The Effectiveness of Palate Re-Repair for Treating

Velopharyngeal Insufficiency: A Systematic Review and Meta-
Analysis
Nicole M. Kurnik, MD1, Erica M. Weidler, MEd2, Kari M. Lien, MA3, Kelly N. Cordero, PhD4,
Jessica L. Williams, MS5, M’hamed Temkit, PhD6, Stephen P. Beals, MD7, Davinder J.
Singh, MD8, Thomas J. Sitzman, MD, MPH9
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1.NicoleM. Kurnik, MD. Resident, Division of Plastic Surgery, Mayo Clinic Arizona; Scottsdale,
Arizona.
2.EricaM. Weidler, MEd. Research Associate, Department of Clinical Research, Phoenix
Children’s Hospital; Phoenix, Arizona.
3.KariM. Lien, MA. Graduate Student, Department of Speech and Hearing Science, Arizona State
University; and Speech Language Pathologist, Barrow Cleft and Craniofacial Center; Phoenix,
Arizona.
4.KellyN. Cordero, PhD. Adjunct Faculty, Department of Speech and Hearing Science, Arizona
State University; and Speech Language Pathologist, Barrow Cleft and Craniofacial Center;
Phoenix, Arizona.
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5.JessicaL. Williams, MS. Adjunct Faculty, Department of Speech and Hearing Science, Arizona
State University; and Speech Language Pathologist, Barrow Cleft and Craniofacial Center;
Phoenix, Arizona.
6.M’hamed Temkit, PhD. Statistician, Department of Clinical Research, Phoenix Children’s
Hospital; Phoenix, Arizona.
7.Stephen P. Beals, MD. Professor, Division of Plastic Surgery, Mayo Clinic Arizona; and Barrow
Cleft and Craniofacial Center; Phoenix, Arizona.
8.Davinder J. Singh, MD. Associate Professor, Division of Plastic Surgery, Phoenix Children’s
Hospital; Division of Plastic Surgery, Mayo Clinic Arizona; and Barrow Cleft and Craniofacial
Center; Phoenix, Arizona.
9.Thomas J. Sitzman, MD, MPH. Assistant Professor, Division of Plastic Surgery, Phoenix
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Children’s Hospital; Division of Plastic Surgery, Mayo Clinic Arizona; and Barrow Cleft and
Craniofacial Center; Phoenix, Arizona.

Abstract

Corresponding Author: Thomas J. Sitzman, MD, MPH, Division of Plastic Surgery, Phoenix Children’s Hospital, 1920 E Cambridge
Rd, Suite 201, Phoenix, AZ 85006 USA, tsitzman@phoenixchildrens.com, Office: 602.933.2670, Fax: 602.933.4320.
Authors’ roles: The study was conceived by T.J.S. All authors participated in study design, analysis, and interpretation. N.M.K. and
T.J.S. wrote the first draft of the article. All authors revised the article critically for important intellectual content. All authors approved
the final version submitted for publication. All authors agree to be accountable for all aspects of the work.
PROSPERO registration: CRD42018096063
 Kurnik et al. Page 2

Background: Palate re-repair has been proposed as an effective treatment for velopharyngeal
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insufficiency (VPI) with a low risk of obstructive sleep apnea (OSA). The authors conducted a
systematic review and meta-analysis to determine the proportion of patients achieving normal
speech resonance following palate re-repair for VPI, the proportion developing OSA, and the
criteria for patient selection that are associated with increased effectiveness.

Methods: PubMed, Embase, and Scopus were searched from inception through April 2018 for
English language articles evaluating palate re-repair for the treatment of VPI in patients with a
repaired cleft palate. Inclusion criteria included reporting of hypernasality, nasal air emission,
nasometry, additional VPI surgery, and/or OSA outcomes. Meta-analysis was conducted using
random effects models. Risk of bias was assessed regarding criteria for patient selection, blinding
of outcome assessors, and validity of speech assessment scale.

Results: Eighteen studies met inclusion criteria. The incidence of achieving no consistent
hypernasality follow palate re-repair was 61% (95% CI, 44–75%). The incidence of additional
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surgery for persistent VPI symptoms was 21% (95% CI, 12–33%). The incidence of OSA was
28% (95% CI, 13–49%). Criteria for selecting patients to undergo re-repair varied, with anterior/
sagittal position of palatal muscles (33%) and small velopharyngeal gap (22%) being the most
common. No specific patient selection criteria led to superior speech outcomes (p=0.6572).

Conclusions: Palate re-repair achieves normal speech resonance in many but not all patients
with VPI. Further research is needed to identify the specific exam and imaging findings that
predict successful correction of VPI with re-repair.

Keywords
cleft palate; velopharyngeal insufficiency; re-repair; palatoplasty; speech; systematic review; meta-
analysis
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INTRODUCTION
Between 20% and 50% of children with cleft palate will develop velopharyngeal
insufficiency (VPI) following cleft palate repair (Kummer, 2009; Chim et al., 2015; Naran et
al., 2017). Traditional methods for treating VPI include pharyngeal flap and sphincter
pharyngoplasty. Both operations are highly effective; a recent meta-analysis determined
overall effectiveness for achieving normal resonance was 76% for pharyngeal flap and 61%
for sphincter pharyngoplasty (de Blacam et al., 2018). However, both operations are
associated with a substantial risk of obstructive sleep apnea (OSA), estimated at 19%−93%
(Orr et al., 1987; Wells et al., 1999; Liao et al., 2004; Abyholm et al., 2005; Yamaguchi et
al., 2016). Thus, while pharyngeal flap and sphincter pharyngoplasty are highly effective in
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treating VPI, they can also produce substantial morbidity through OSA.

Reducing the risk of OSA is important given its negative health effects. OSA can lead to
metabolic and cardiovascular derangements including increased fasting glucose levels,
elevated triglyceride levels, hypertension, chronic inflammation and elevated pulmonary
artery pressure and endothelial dysfunction (Blechner and Williamson, 2016; Hilmisson et
al., 2019). OSA has also been associated with cognitive and behavioral deficits in children
(Gottlieb et al., 2003; Kaemingk et al., 2003; Mulvaney et al., 2006; Hvolby, 2015). Given

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the serious morbidities associated with OSA, the reported incidence of OSA as high as 93%
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after pharyngeal flap and sphincter pharyngoplasty is concerning.

Palate re-repair is an attractive alternative for treating VPI given its lower risk of OSA
(Mehendale et al., 2013). Re-repair involves repositioning and reconstructing the velar
muscles. Re-repair can be performed using a straight-line incision in the oral mucosa of the
palate (i.e. Sommerlad re-repair) as initially described by Dellon and Edgerton (Dellon and
Edgerton, 1969; Edgerton and Dellon, 1971) and further described and evaluated by
Sommerlad et al (1994). Re-repair can also be performed using a double opposing Z-plasty
(i.e. Furlow palatoplasty) (Randall et al., 1986; Chen et al., 1994). While pharyngeal flaps
and sphincter pharyngoplasties create a permanent obstruction or narrowing of the
oropharynx that can lead to OSA, palate re-repair has been proposed to have a lower risk of
OSA because it produces no change in the size of the oropharyngeal passage or obstruction
to air flow (Liao et al., 2004; Mehendale et al., 2013). Re-repair has also been shown to be
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up to 82% effective in treating VPI (Sommerlad et al., 2002). This has led some authors to
propose palate re-repair as the primary treatment modality for patients with VPI (Liao et al.,
2004).

Despite the apparent benefits of palate re-repair, this technique has not been widely adopted.
In a recent systematic review of VPI treatments only 7.9% of patients received palatoplasty
while 88% of patients received pharyngeal flap or sphincter pharyngoplasty (de Blacam et
al., 2018). The lower utilization of palate re-repair may be due to concerns about its
effectiveness or uncertainty about which patients are most likely to improve with re-repair.

This systematic review and meta-analysis was conducted to determine the proportion of
patients achieving normal resonance following cleft palate re-repair for VPI from all
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published clinical studies and case series. Our primary measure of normal resonance was
defined as no consistent hypernasality on perceptual evaluation. We also evaluated complete
resolution of hypernasality, resolution of nasal air emission, re-operation for persistent VPI,
and the incidence of OSA. Further, the indications for palate re-repair in each study and the
outcomes among children with and without syndromic diagnoses were evaluated. The study
provides a detailed picture of how palate re-repair is being utilized by cleft providers and
how patients considering palate re-repair should be counseled about the indications, risks,
and benefits of the procedure.

METHODS
Protocol Development
Methods of study identification, inclusion criteria, and analysis were specified in advance
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and documented in a protocol according to the Institute of Medicine Standards for

Systematic Reviews (2011) and the Preferred Reporting Items for Systematic reviews and
Meta-Analyses for Protocols 2015 (PRISMA-P 2015) guidelines (Moher et al., 2015). The
protocol was registered on the PROSPERO register of systematic reviews prior to study
identification. The protocol was amended twice during the study: (1) the criterion for OSA
was changed from “clinical diagnosis of OSA” to “apnea-hypopnea index (AHI) >1” based
on guidelines from the American Academic of Sleep Medicine (Dehlink and Tan, 2016;

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Berry et al., 2018), and (2) the definition of palate re-repair was changed from “velar muscle
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reconstruction or velar lengthening” to “velar muscle reconstruction with or without velar

lengthening.” The PRISMA reporting guidelines were followed (Moher et al., 2009).

Search Strategy
An electronic search of PubMed, Embase, Scopus, and Web of Science was performed from
database inception through April 20, 2018. Search terms included cleft palate, re-repair,
palatoplasty, furlow, double opposing z-plasty, velopharyngeal insufficiency, hypernasality,
and nasal air emission. The search was restricted to English language articles. A detailed
search strategy is included in Appendix 1 (Supplemental Digital Content). Reference lists
from included studies were visually searched for additional studies of interest by one
investigator (T.J.S.).
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Eligibility Criteria
Inclusion criteria were: (1) English language articles, (2) published in the medical literature,
(3) reporting both the number of patients undergoing palate re-repair for VPI and the number
of those patients with any one of the following outcomes: hypernasality, nasal air emission,
normal resonance, additional VPI surgery, and/or OSA. Patients of any age were considered,
as long as they had a history of cleft palate (including submucous cleft palate), cleft palate
repair, and a diagnosis of VPI. Studies were excluded if they did not specify that patients
had previously undergone cleft palate repair, if patients had undergone a velopharyngeal
surgery other than palate repair prior to re-repair, or if they underwent re-repair at the same
time as another velopharyngeal surgery. Review articles and studies with fewer than ten
patients were excluded.
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Study Selection
The study selection process is summarized in Figure 1. Citations were screened on title and
abstract against the inclusion and exclusion criteria. Screening was performed by two
investigators working independently, with disagreements resolved by a third investigator.
Final assessment for eligibility was determined by review of full articles. Final assessment
was performed by two investigators working independently, with disagreements resolved by
consensus of four investigators (N.M.K, K.M.L., K.N.C., and T.J.S.).

Data Collection
Data was extracted by a single investigator (N.M.K.) using a pilot tested form. Two speech
language pathologists (K.N.C. and K.M.L.) with experience in VPI evaluation and
management then independently reviewed the data extraction for accuracy and
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completeness. Because there was substantial heterogeneity in reporting of methods and

outcomes among the included studies, extracted data was then independently re-reviewed for
accuracy and completeness by a surgeon with experience in VPI management (T.J.S.).
Disagreements were resolved by discussion among four investigators (N.M.K, K.M.L.,
K.N.C., and T.J.S.). Authors of primary studies were not contacted to provide missing or
additional data.

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Data extracted from each study included: (1) study design (clinical trial vs cohort study vs
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case series, prospective vs retrospective data collection for outcomes relative to initiation of
the study, reporting on consecutively treated subjects); (2) characteristics of study population
(age, cleft type, presence of syndromic diagnoses, number of surgeons and centers providing
treatment); (3) indications for selecting re-repair as the VPI treatment operation; (4)
technique of palate re-repair; (5) proportion of study population achieving each of the
following outcomes of interest:

• No hypernasality

• No consistent hypernasality (i.e. hypernasality described as none or inconsistent)

• Less than mild hypernasality (i.e. hypernasality described as none, inconsistent,

borderline, or minimal)

• No consistent nasal air emission

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• Nasometric scores within normal range

• Additional velopharyngeal surgery for persistent VPI

• Diagnosis of OSA (apnea-hypopnea index [AHI] >1)

and; (6) method of outcome assessment (blinding of assessors to treatment history, time
from re-repair to outcome assessment, scale or instrument used for assessing each outcome).

Technique of palate re-repair was collected verbatim from each included study. A panel of
three cleft surgeons (T.J.S., D.J.S., S.P.B.) then reviewed these technique descriptions,
created three distinct categories which captured all descriptions (Furlow double-opposing Z-
plasty [Furlow DOZ], radical intra-velar veloplasty [radical IVVP], and radical IVVP with
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palatal lengthening), and assigned each reported technique to one of these categories.

Among studies that reported incidence of OSA for subjects undergoing re-repair, data was
also collected on incidence of OSA in subjects undergoing sphincter pharyngoplasty and/or
pharyngeal flap when available.

Assessment of Risk of Bias

Risk of bias was assessed at both the study and outcome levels in an unblinded manner.
Study-level bias was assessed by describing the method for assigning subjects to re-repair
and whether outcomes were reported on a consecutively-treated patient cohort. Outcome-
level bias was assessed by determining if outcome assessors were blinded to treatment
history, by evaluating the validity of the hypernasality assessment scale or instrument for
outcome assessment, by evaluating whether outcomes were evaluated prospectively or
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retrospectively (e.g. through review of medical records), and by describing the follow-up
duration (time from re-repair to outcome assessment).

Data Synthesis and Statistical Analysis

For each study, incidence was reported for each of the following outcomes: no hypernasality,
no consistent hypernasality, less than mild hypernasality, no consistent nasal air emission,
nasometric scores within normal range, additional velopharyngeal surgery for persistent

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VPI, and diagnosis of OSA. Incidence of no consistent hypernasality was pre-specified as

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the primary outcome of interest. Forest plots were constructed using 95% Wald confidence
intervals. The I2 test was performed to evaluate heterogeneity across studies. Meta-analysis
was performed with a random effects model to address variability across studies. Subgroup
analyses were pre-specified and included comparison by indication for selecting re-repair,
technique of re-repair, and inclusion or exclusion of subjects with syndromic diagnoses.
Comparison among subgroups was performed with ANOVA using a random effects model.
Statistical analyses were performed in R Studio Version 1.0.153 (RStudio, Inc., Boston,
MA).

RESULTS
Study Selection
The search strategy identified 780 unique citations. Screening of titles and abstracts
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identified 103 potentially relevant articles. Full text review of these articles identified
eighteen studies meeting the inclusion criteria. The most frequent reason for exclusion was
subjects not undergoing re-repair for VPI (e.g. including patients with unrepaired
submucous cleft palate). The study selection process is shown in Figure 1.

Study Characteristics
The eighteen studies meeting inclusion criteria are summarized in Table 1. All studies
included patients with cleft palate with or without cleft lip (CP±CL). All studies described
patients treated at a single-center, with up to four surgeons operating on the patient cohort.

Risk of Bias
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The risk of study-level bias and outcome-level bias was assessed for each study and is
reported in Table 2.

There was evidence of study-level bias among some of the included studies. The indications
for performing re-repair varied among the studies, and five studies (28%) failed to report
how the decision to perform re-repair was made. While ten studies (56%) reported outcomes
on a consecutive sample of patients, one study (6%) did not include a consecutive sample
and seven studies (39%) did not indicate information about sample consecutiveness.

Evidence of outcome-level bias was also present among many of the included studies. Of the
thirteen studies reporting perceptual speech outcomes, only two (15%) blinded the raters to
treatment history and only three used a validated assessment scale (23%). Five studies
prospectively collected patient outcomes. Time from surgery to outcome assessment varied
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between six and twenty-five months among the included studies (Table 1).

Speech Outcomes
Individual results for each study and each speech outcome are reported in Table 1, Figures
2–3, and Supplemental Digital Figures 1-6. The overall incidence of achieving no consistent
hypernasality following palate re-repair was 61% (95% CI, 44–75%) (Figure 2). The
incidence of achieving no hypernasality, a more stringent outcome, was 53% (95% CI, 40–

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65%) (Supplemental Digital Figure 1). The incidence of less than mild hypernasality, a less
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stringent outcome, was 65% (95% CI, 54–75%) (Supplementary Digital Figure 2). The
incidence of no consistent nasal air emission was 78% (95% CI, 60–89%) (Supplementary
Digital Figure 3). The incidence of additional velopharyngeal surgery for persistent VPI
symptoms was 21% (95% CI, 12–33%) (Supplementary Digital Figure 4). There was
substantial heterogeneity across studies for all outcomes (I2 range 67–82%).

Predictors of re-repair success

Three general techniques for re-repair were identified: Furlow DOZ, radical IVVP, and
radical IVVP with mucosal lengthening. Radical IVVP had a higher incidence of achieving
no consistent nasal air emission compared to Furlow DOZ (p=0.0081) (Supplemental Digital
Figure 3). For the remainder of the speech outcomes there was no significant difference
among techniques (p>0.10)
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The indication for performing re-repair was not associated with the incidence of achieving
no consistent hypernasality (p=0.6572) (Figure 3). The inclusion or exclusion of subjects
with syndromic diagnoses was not associated with incidence of achieving no consistent
hypernasality (p=0.7657) (Supplemental Digital Figure 6).

Obstructive Sleep Apnea

Incidence of OSA for each study is reported in Table 1. Figure 4 includes both the incidence
of OSA among included studies and, when reported in the included studies, the incidence of
OSA in subjects undergoing sphincter pharyngoplasty and/or pharyngeal flap. The overall
incidence of OSA following re-repair was 28% (95% CI, 13–49%). The incidence of OSA
following re-repair was substantially lower than the incidence of OSA following pharyngeal
flap (86%; 95% CI, 63–96%; p=0.0007).
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Only one study reported incidence of OSA both before and after re-repair. In that study of
thirteen patients, OSA grade improved in four patients (31%), remained the same in four
patients (31%), and became worse in five patients (38%).(Mehendale et al., 2013) Of the five
patients who showed deterioration, four developed mild OSA and one developed mild-to-
moderate OSA.

DISCUSSION
Velopharyngeal insufficiency (VPI) can have many different causes and presentations. An
unrepaired submucous cleft palate, a short and/or immobile soft palate, and anteriorly
oriented or discontinuous palatal muscles all represent distinct causes of VPI (Sie et al.,
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2001; Sie and Chen, 2007; Gart and Gosain, 2014; Perry and Kuehn, 2015; Naran et al.,
2017; de Blacam et al., 2018). Ideally, providers identify the causative factor(s) for VPI in
each patient through their history, exam and imaging studies, and then select a treatment
approach best suited to each patient’s pathophysiology (Marsh, 2004; Shprintzen and
Marrinan, 2009; Hopper et al., 2014; Kummer et al., 2015; Naran et al., 2017). To achieve
this ideal, the effectiveness of VPI surgical approaches must be considered for specific
patient presentations.

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The most common presentation of VPI is hypernasality and/or audible nasal air emission
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after primary cleft palate repair. Pharyngeal flap and sphincter pharyngoplasty have been
evaluated in this population through a randomized controlled trial and no significant
difference in efficacy or side effects was found between these two approaches (Abyholm et
al., 2005). Effectiveness of palate re-repair in this population is less well understood. The
present study addressed this knowledge gap by conducting a systematic review and meta-
analysis of palate re-repair specifically in children with VPI after primary cleft palate repair.
This is a substantially different approach compared to prior studies that evaluated palate re-
repair when used for all causes of VPI (Sie et al., 2001; Perkins et al., 2005; Dailey et al.,
2006; Milczuk et al., 2007; Mann et al., 2011; de Blacam et al., 2018).

A key finding of this study was that three types of surgical techniques are being employed
under the label of palate re-repair: Furlow DOZ (Chen et al., 1994), radical IVVP
(Sommerlad et al., 1994; Sommerlad et al., 2002), and radical IVVP with palatal
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lengthening via either buccal myomucosal flaps (Robertson et al., 2008; Logjes et al., 2017)
or mucosal Z-plasty (Woo et al., 2014; Logjes et al., 2017). Among these three surgical
techniques, the only significant difference in outcomes between them was that radical IVVP
had a higher incidence of achieving no consistent nasal air emission compared to Furlow
DOZ (92% vs 63%, p=0.0081). There was no statistically significant difference among the
three techniques in incidence of hypernasality or receipt of additional VPI surgery (p>0.10).
This finding may indicate that the three distinct re-repair techniques have similar efficacy, or
it may be due to underlying differences in patient populations or methods of outcome
measure among the included studies.

Another key finding of this study was that indications for selecting re-repair to treat VPI
vary among surgeons. The most commonly cited indications for selecting re-repair were:
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anterior, sagittal, or non-transverse position of the palatal muscles on clinical exam (n=6,
33%) (Chen et al., 1994; Sommerlad et al., 1994; Wojcicki and Wojcicka, 2008; Mehendale
et al., 2013; Elsherbiny et al., 2018), and small VP gap on nasopharyngoscopy (n=4, 22%)
(Chen et al., 1994; Madrid et al., 2011; Barbosa et al., 2013; Yamaguchi et al., 2016). In
contrast to this targeted application of re-repair, two studies used re-repair as their treatment
for all children with VPI after CP repair (Deren et al., 2005; Woo et al., 2014). This variation
in surgical indications for re-repair suggests that surgeons remain uncertain which patients
are most likely to benefit from re-repair.

While no significant difference in effectiveness of re-repair was identified among the

different indications, two trends deserve discussion. First, three studies used palatal muscle
position and VP gap as indications for re-repair and also reported hypernasality outcomes;
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one of these studies reported an 84% success rate using a radical IVVP (Sommerlad et al.,
2002) and the other an 89% success rate with Furlow DOZ (Chen et al., 1994). This suggests
that velar muscle repositioning via either radical IVVP or Furlow DOZ is highly effective in
correcting VPI in the setting of anterior, sagittal, or non-transverse positioned palatal
muscles. The second notable trend is that among the two studies using palate re-repair for all
patients with VPI after primary palate repair and reporting hypernasality outcomes
(Nakamura et al., 2003; Deren et al., 2005), only 43% achieved resolution of hypernasality.
These findings suggest that patient selection based on physical exam and imaging findings

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may play an important role in determining the effectiveness of palate re-repair (Perkins et
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al., 2005). Research comparing these patient selection criteria should be a priority for the
field.

Limitations
Results of the present study must be interpreted in the context of the study design. As a
systematic review, the strength and quality of the study’s findings are dependent upon the
sample size and risk of bias in the included studies. The study’s overall estimate of re-
repair’s effectiveness for achieving the primary outcome of no consistent hypernasality is
quite strong, since it is based on 283 subjects from nine studies. However, the strength of the
study’s other comparisons, particularly comparisons among the different surgical techniques
and surgical indications, are much weaker since they are based on substantially smaller
samples. These limited samples highlight the need for future studies.
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Quality of the present study’s findings is a concern due to potential reporting bias in the
included studies. Eight of the eighteen studies (44%) failed to report outcomes for a
consecutive patient cohort and five studies (28%) failed to state how they selected patients to
receive re-repair. If substantial reporting bias is present in these studies, then estimates of re-
repair effectiveness may be overly optimistic.

Another concern is the risk of bias in assessment of speech outcomes. Among the thirteen
studies reporting speech outcomes, only two (15%) blinded outcome assessors to treatment
history and only three (23%) used a validated scale for assessing speech outcomes.
Variability in assessment scales raises substantial concerns that speech outcomes were not
assessed similarly among the included studies – a finding demonstrated by both the
substantial heterogeneity in speech outcomes among the included studies and by prior
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studies showing moderate reliability across uncalibrated speech outcome raters (Chapman et
al., 2016). The present study attempted to address this bias by using random-effects models
that assume there are underlying differences between all studies; however, this approach
incompletely addresses the issue. Further, even studies using validated rating scales for
perceptual assessment are subject to imperfect rater reliability. These challenges in
consistency and reliability for speech outcome reporting remain a recognized limitation in
both cleft palate and VPI research (Whitehill, 2002; Sell, 2005; Chapman et al., 2016;
Kummer et al., 2016; de Blacam et al., 2018), and for this reason caution should be
exercised in all comparisons across studies.

Finally, it should be noted that variations in surgical expertise and the details of surgical
technique may contribute to differences in effectiveness of palate re-repair, and to
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differences in effectiveness between re-repair techniques or surgical indications. The present

study attempted to address this variation by using random-effects models that assume there
are underlying differences between all studies, but the overall estimates of effectiveness
generated by these models may under- or over-estimate the effectiveness of the technique in
any one surgeon’s hands. Further study is needed to understand how surgeon expertise and
details of surgical technique influence effectiveness of palate re-repair.

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Conclusions
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Palate re-repair is a promising surgical treatment of VPI in patients with a repaired cleft
palate. Among studies reporting use of re-repair in this population, re-repair corrected
hypernasality in 61% of patients (95% CI, 44–75%) and corrected nasal air emission in 78%
of patients (95% CI, 60–89%). Re-repair also carries a substantially lower risk of OSA than
pharyngeal flap (28% vs 86%, p=0.0007). Further research is needed to directly compare re-
repair to pharyngeal flap and sphincter pharyngoplasty and to identify which pre-operative
history, examination, and imaging findings are most predictive of VPI symptom resolution
following re-repair.

Included Citations(Chen et al., 1994; Sommerlad et al., 1994; Sommerlad et al., 2002;
Nakamura et al., 2003; Liao et al., 2004; Deren et al., 2005; Noorchashm et al., 2006;
Robertson et al., 2008; Wojcicki and Wojcicka, 2008; Madrid et al., 2011; Barbosa et al.,
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2013; Koh et al., 2013; Mehendale et al., 2013; Woo et al., 2014; Yamaguchi et al., 2016;
Logjes et al., 2017; Abdel-Aziz et al., 2018; Elsherbiny et al., 2018)

Supplementary Material
Refer to Web version on PubMed Central for supplementary material.

Acknowledgments
Financial Disclosure Statement: Dr. Sitzman received support from the National Institute of Dental and
Craniofacial Research (K23 DE025023). No other external funding was provided for this manuscript. The authors
have no other financial relationships relevant to this article to disclose.

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Figure 1. Flow diagram of study selection.

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VPI, velopharyngeal insufficiency

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Figure 2. Incidence of no consistent hypernasality.

This outcome includes subjects with hypernasality described as none or inconsistent
following palate re-repair. No significant difference among re-repair techniques (p=0.7337).
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 Kurnik et al. Page 16
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Figure 3. Influence of re-repair indications.

This figure compares the incidence of no consistent hypernasality across studies based on
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the indications by which subjects were selected to undergo re-repair. No significant

difference among re-repair indications (p=0.6572).

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Figure 4. Incidence of OSA.

This outcome includes subjects with an apnea-hypopnea (AHI) index > 1 on overnight
polysomnography following palate re-repair. The incidence of OSA following re-repair is
substantially lower than the incidence of OSA following pharyngeal flap (p=0.0007).
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Table 1.

Included Studies.

Subjects achieving specified outcomes (%)

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Age at Months No
Surgical Surgery Follow-up No consistent Less than mild consistent Normal Additional
1 2 3 No 4 5 nasal air nasometry 6 7
Study Technique n (range) (range) hypernasality hypernasality hypernasality emission scores VPI surgery OSA

Abdel-Aziz Furlow DOZ 12 7 (–) 6 (–) – – – – – – 3 (25)

2018
Chen 1994 Furlow DOZ 18 – (3–23) 12 (12–48) – 16 (89) – – – – –
Deren 2005 Furlow DOZ 27 7 (4–13) 12 (–) 10 (37) 10 (37) 10 (37) 16 (59) – – –
Koh 2013 Furlow DOZ 13 5 (3–6) 8 (–) 9 (69) 9 (69) 9 (69) 8 (62) – – –
Liao 2004 Furlow DOZ 20 6 (–) 6 (–) – – – – – – 4 (20)
Madrid 2011 Furlow DOZ 10 15 (–) 12 (–) – – – – – – 1 (10)
Noorchasm Furlow DOZ 19 6 (4–15) 9 (3–25) 13 (68) 13 (68) 13 (68) 13 (68) – – –
2006
Wojcicki 2008 Furlow DOZ 44 12 (6–25) 7 (3–12) 10 (23) 10 (23) 28 (64) – 14 (32) – –
Woo 2014 Furlow DOZ 30 7 (3–30) – (–) – – – – – 11 (37) –
Yamaguchi Furlow DOZ 46 7 (–) 38 (–) – – – – – 9 (20) –
2016
Barbosa 2013 Radical IVVP 38 21 (6–52) 14 (–) – – – – 13 (34) – –
Elsherbiny Radical IVVP 111 6 (3–15) 25 (3–96) – – – – – 33 (30) –
2018
Mehendale Radical IVVP 13 10 (1–16) 7 (–) – – – – – – 7 (54)
2013
Nakamura Radical IVVP 13 11 (5–19) 14 (6–36) 8 (53) 8 (53) 8 (53) – – – –
2003

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Sommerlad Radical IVVP 32 14 (4–37) 6 (–) – – 25 (81) 26 (84) – 2 (6) –
1994
Sommerlad Radical IVVP 85 11 (3–48) 6 (–) 53 (62) 71 (84) 71 (84) 82 (96) – 10 (12) –
2002
Logjes 2017 Radical IVVP 42 5 (2–17) 14 (6–25) 8 21 (55) 21 (55) – – 7 (17) –
with palatal 21 (55)
lengthening
Robertson Radical IVVP 22 9 (1–23) – (–) 9 11 (65) 11 (65) 13 (76) – 11 (50) –
2008 with palatal 11 (65)
lengthening
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Subjects achieving specified outcomes (%)

Age at Months No
Surgical Surgery Follow-up No consistent Less than mild consistent Normal Additional
1 2 3 No 4 5 nasal air nasometry 6 7
Study Technique n (range) (range) hypernasality hypernasality hypernasality emission scores VPI surgery OSA
Kurnik et al.

Woo 2014 Radical IVVP 22 8 (–) – (–) – – – – – 0 (0) –

with palatal
lengthening

1
Technique categorized as Furlow double-opposing Z-plasty (Furlow DOZ), radical intra-velar veloplasty (radical IVVP), and radical IVVP with palatal lengthening
2
Mean subject age if reported, otherwise median age
3
Mean follow-up duration if available, otherwise minimum duration of follow-up
4
Hypernasality described as none or inconsistent
5
Hypernasality described as none, inconsistent, borderline, or minimal
6
Additional velopharyngeal surgery for persistent VPI
7
OSA, obstructive sleep apnea
8
Logjes et al. collected post-operative speech samples on only 38 subjects, percentages presented for speech outcomes are among the 38 evaluated subjects
9
Robertson et al. collected post-operative speech samples on only 17 subjects, percentages presented for speech outcomes are among the 17 evaluated subjects

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Table 2.

Re-Repair Indication, Study Design and Assessment of Risk of Bias.

Prospective vs
Retrospective Perceptual speech
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Measurement of Included assessment scale/

1 Study 2 Consecutively Patients w/ No. of 3 Blinded speech
Study Re-Repair Indication Design Outcomes Treated Patients Syndromes Surgeons instrument assessment

Abdel-Aziz 2018 4 Cohort – – No 1 – –

–
Barbosa 2013 Small gap Cohort – – – 4 – –
Chen 1994 5 Cohort Retrospective Yes – – Undefined scale No
Small gap or sagittal
6
muscle
Deren 2005 All patients Cohort Prospective Yes No 3 Trier, 1985 No
Elsherbiny 2018 Sagittal muscle Cohort Retrospective Yes Yes 1 Pittsburgh Weighted No
7
Speech Score
Koh 2013 – Cohort Retrospective – No 1 In-house scale No
Liao 2004 – Cohort Prospective Yes No 1 – –
Logjes 2017 – Cohort Retrospective Yes Yes 1 In-house scale No
Madrid 2011 Small gap Cohort – – No 1 – –
Mehendale 2013 Sagittal muscle Cohort Retrospective – Yes 1 – –
Nakamura 2003 All patients Cohort Retrospective Yes – 3 In-house scale No
Noorchasm 2006 – Cohort Retrospective – – 1 Pittsburgh Weighted No
4
Speech Score
Robertson 2008 Short or scarred palate Cohort Retrospective No Yes 1 Bzock Screening Test, No
1989
Sommerlad 1994 Sagittal muscle Cohort Prospective Yes No 1 In-house scale Yes

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Sommerlad 2002 Sagittal muscle Cohort Prospective Yes No 1 Cleft Audit Protocol for Yes
4
Speech
Wojcicki 2008 Sagittal muscle Cohort Prospective – – 1 In-house scale No
Woo 2014 All patients Cohort Retrospective Yes No – Undefined scale No
Yamaguchi 2016 Small gap Cohort Retrospective Yes No 1 In-house scale No

1
Patient selection criteria for deciding who received palate re-repair
2
Prospective versus retrospective measurement of outcome data relative to initiation of the protocol for study data collection (e.g. a study was considered prospective if there was a specific protocol in place
for collection of consistent, reliable data on the study outcomes prior to collection of any study-specific data).
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3
Empty cells indicate study did not report perceptual speech outcomes (e.g. OSA reporting only)
4
–, not reported or not applicable
5
Small velopharyngeal gap on nasopharyngoscopy
6
Sagittal, anterior, or non-transverse orientation of the velar muscles on intraoral examination
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7
Validated assessment tool

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