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Systematic Review of Postoperative Velopharyngeal Insufficiency: Incidence and Association With Palatoplasty Timing and Technique

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Cleft palate is among the most common congenital disorders worldwide and is correctable through surgical intervention. Sub-optimal surgical results may cause velopharyngeal insufficiency (VPI). When symptomatic, VPI can cause hypernasal or unintelligible speech. The postoperative risk of VPI varies significantly in the literature but may be attributed to differences in study size, cleft type, surgical technique, and operative age. To identify the potential impact of these factors, a systematic review was conducted to examine the risk of VPI after primary palatoplasty, accounting for operative age and surgical technique. A search of PubMed, Embase, and Web of Science was completed for original studies that examined speech outcomes after primary palatoplasty. The search identified 4740 original articles and included 35 studies that reported mean age at palatoplasty and VPI-related outcomes. The studies included 10,795 patients with a weighted mean operative age of 15.7 months (range: 3.1-182.9 mo), and 20% (n=2186) had signs of postoperative VPI. Because of the heterogeneity in reporting of surgical technique across studies, small sample sizes, and a lack of statistical power, an analysis of the VPI risk per procedure type and timing was not possible. A lack of data and variable consensus limits our understanding of optimal timing and techniques to reduce VPI occurrence. This paper presents a call-to-action to generate: (1) high-quality research from thoughtfully designed studies; (2) greater global representation; and (3) global consensus informed by high-quality data, to make recommendations on optimal technique and timing for primary palatoplasty to reduce VPI.
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Systematic Review of Postoperative
Velopharyngeal Insufficiency: Incidence
and Association With Palatoplasty Timing
and Technique
Meredith D. Xepoleas, BA,*Priyanka Naidu, MBChB, MSc,*
Eric Nagengast, MD, MPH,*
Zach Collier, MD,*
Delaney Islip, BS,
Jagteshwar Khatra, BS,
Allyn Auslander, PhD,*
§
Caroline A. Yao, MD, MS,*
David Chong, MBBS,
and William P. Magee, III, MD, DDS
§
Abstract: Cleft palate is among the most common congenital
disorders worldwide and is correctable through surgical inter-
vention. Sub-optimal surgical results may cause velopharyngeal
insufciency (VPI). When symptomatic, VPI can cause hyper-
nasal or unintelligible speech. The postoperative risk of VPI
varies signicantly in the literature but may be attributed to
differences in study size, cleft type, surgical technique, and op-
erative age. To identify the potential impact of these factors, a
systematic review was conducted to examine the risk of VPI
after primary palatoplasty, accounting for operative age and
surgical technique. A search of PubMed, Embase, and Web of
Science was completed for original studies that examined speech
outcomes after primary palatoplasty. The search identied 4740
original articles and included 35 studies that reported mean age
at palatoplasty and VPI-related outcomes. The studies included
10,795 patients with a weighted mean operative age of
15.7 months (range: 3.1182.9 mo), and 20% (n =2186) had
signs of postoperative VPI. Because of the heterogeneity in re-
porting of surgical technique across studies, small sample sizes,
and a lack of statistical power, an analysis of the VPI risk per
procedure type and timing was not possible. A lack of data and
variable consensus limits our understanding of optimal timing
and techniques to reduce VPI occurrence. This paper presents a
call-to-action to generate: (1) high-quality research from
thoughtfully designed studies; (2) greater global representation;
and (3) global consensus informed by high-quality data, to
make recommendations on optimal technique and timing for
primary palatoplasty to reduce VPI.
Key Words: Velopharyngeal insufciency, cleft palate, palato-
plasty, timing, technique
(J Craniofac Surg 2023;34: 16441649)
Orofacial clefts are among the most common congenital
anomalies worldwide. The global prevalence of cleft
palate globally ranges from 1.4 per 10,000 births in Cuba to
25.3 per 10,000 births in Canada.
1
This congenital anomaly is
correctable through cost-effective surgical interventions.
24
One study estimated that the repair of cleft lip or palate has
the same cost effectiveness of vaccines in low-income and-
middle-income countries.
5
Cleft-related speech impairment
can be profound for the patient, impacting ability to seek
gainful employment and ultimately resulting in signicant
economic impact for households and communities.
2,4
The primary goals of the cleft palate repair are to restore
functional anatomy, to close the defect, and restore normal
speech. Unfortunately, maxillary growth deciency, palatal
stulae, and inadequate speech are still common complications.
Delayed surgery, improper surgical technique, and scarring can
produce sub-optimal surgical outcomes, which may result in
velopharyngeal insufciency (VPI).
6,7
Velopharyngeal in-
sufciency occurs as a result of a short, immobile soft palate
that does not close adequately against the back of the throat,
resulting in hypernasal or unintelligible speech.
8
The rate of VPI
after primary palatoplasty varies between 5% and 30%
916
in
the literature, which may be attributed to differences in study
size, cleft severity, surgical technique, and operative age of the
study populations.
17
There is still no international consensus on the appropriate
timing of cleft palate repair and technique preferences are still
debated.
18,19
Most literature suggests primary palatoplasty
performed after 18 months leads to worse speech and a higher
risk of developing VPI.
9,2024
However, some centers still ad-
vocate for delayed palatoplasty to limit midface hypoplasia.
25
Cleft palate can present in varying degrees of severity with or
without cleft lip involvement, which makes it difcult for one
From the *Operation Smile Inc, Virginia Beach, VA; Division of
Plastic and Reconstructive Surgery, Keck School of Medicine of
USC; University of California, Los Angeles, School of Dentistry;
§Division of Plastic and Maxillofacial Surgery, Childrens Hospital
Los Angeles; Department of Plastic Surgery, Shriners Hospital for
Children, Los Angeles, CA; and ¶Royal Childrens Hospital, Mel-
bourne, VIC, Australia.
Received June 8, 2021.
Accepted for publication May 19, 2023.
The authors report no conicts of interest.
Address correspondence and reprint requests to William P Magee III,
MD, DDS, Childrens Hospital Los Angeles, 4650 Sunset Blvd. MS
#96. Los Angeles, CA 90027; E-mail: wmagee@chla.usc.edu
Supplemental Digital Content is available for this article. Direct URL
citations are provided in the HTML and PDF versions of this article
on the journals website, www.jcraniofacialsurgery.com.
This is an open access article distributed under the terms of the Creative
Commons Attribution-Non Commercial-No Derivatives License 4.0
(CCBY-NC-ND), where it is permissible to download and share the
work provided it is properly cited. The work cannot be changed in
any way or used commercially without permission from the journal.
Copyright © 2023 The Author(s). Published by Wolters Kluwer Health,
Inc. on behalf of Mutaz B. Habal, MD.
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000009555
ORIGINAL ARTICLE
1644 The Journal of Craniofacial Surgery Volume 34, Number 6, September 2023
singular surgical technique to repair all presentations.
26
Sur-
geons are often taught one cleft palate repair technique through
mentorship and utilize or modify that one technique for the
entirety of their careers. The reluctance to change from a
technique one gains prociency in and undertake a new learning
curve discourages change in a surgeons approach. Fur-
thermore, technique variations and modications make com-
paring surgical outcomes challenging. Surgeon skill itself is
another major factor pivotal to postoperative outcomes.
27
The timing of cleft palate repair is also difcult to control as
it depends on timely presentation of the patient to care, which is
often delayed in resource-limited settings.
28
Furthermore, the
assessment of VPI is not standardized, is subjective, and can
vary across languages and settings, confounding the comparison
of post-palatoplasty outcomes.
29
Understanding appropriate timing and technique in cleft
palate repair is imperative in minimizing the negative compli-
cations, yet consensus on both is still variable. We hypothesize
that there is a lack of high-quality data to standardize recom-
mendations. This systematic review aimed to (1) objectively
summarize existing data on potential impact of primary pala-
toplasty operative age and surgical technique on the risk of
developing postoperative VPI, and (2) comment on whether this
data can be used to inform best practice and provide recom-
mendations for next steps.
MATERIALS AND METHODS
A systematic review of the literature was conducted after the
Preferred Reporting Items for Systematic Reviews and Meta
Analyses (PRISMA) guidelines.
30
A detailed protocol was
registered to PROSPERO (CRD42020193982).
31
Information Sources and Search Strategy
A search of PubMed, Embase, and Web of Science was
conducted on April 6, 2020 and included twelve search terms
(Supplemental Appendix 1, Supplemental Digital Content 1,
http://links.lww.com/SCS/F263). The search terms focused on
the speech outcomes and the associated surgical interventions of
the palate. There was no restriction on publication date. Gray
literature was not searched and there was no contact with study
authors.
Eligibility Criteria and Study Selection
Eligibility criteria included full-text articles published in
English that recorded VPI speech measures after primary pal-
atoplasty. All cleft palate phenotypes were included. The studies
were required to include mean age at primary palatoplasty and
a measure of either VPI, hypernasality, or a rate of secondary
surgical intervention for speech outcomes. Studies were required
to have complete speech outcomes correlated with mean age at
surgery for a given study population. This review only examined
non-syndromic patients to limit confounding factors in the
speech outcomes. All studies with an incorrect study focus or
missing inclusion criteria were excluded. Case reports, edito-
rials, reviews, and articles without original data were not in-
cluded in the analysis.
In the rst stage, 2 unblinded reviewers (M.X. and D.I.)
screened all titles independently to assess relevance to this re-
views objective. In the second stage, once the discrepancies
between reviewerslists were resolved, the 2 reviewers in-
dependently screened the remaining study abstracts for in-
clusion criteria. Discrepancies between reviewerslists were
solved through discussion. In the nal stage, 3 reviewers in-
dependently assessed full-text articles (M.X., D.I., J.K.).
Disagreements were resolved by discussion and articles that did
not meet inclusion criteria were excluded.
Data Extraction
Quantitative data were extracted from the full-text studies
including patient demographics, surgical factors, and surgical
outcomes in relation to speech. Data extracted from full-text
articles by independent reviewers (M.X., D.I., J.K.) included
author, year of publication, study design, population size, se-
verity of cleft, palatoplasty operative age (age at soft palate
repair if 2-stage), surgical technique (if available), outcomes of
the population in relation to speech (VPI, hypernasality, or
incidences of secondary surgery for speech), and age at speech
assessment. This data were aggregated into a chart created on
Microsoft Excel (Microsoft Corporation).
Data Analysis and Synthesis of Results
The data extracted from included articles were too hetero-
geneous for a meta-analysis. To determine timing as a risk for
VPI symptoms after primary palatoplasty, the study population
was divided into 4 quartiles based on mean operative age: 0 to
6 months (Q1), >6 to 12 months (Q2), >12 to 24 months (Q3),
and over 24 months (Q4). To determine the risk of VPI symp-
toms after primary palatoplasty by surgical technique, a sub-
group analysis was attempted using data from studies that
reported common palatoplasty techniques (Supplemental
Appendix 1, Supplemental Digital Content 1, http://links.lww.
com/SCS/F263). Patients qualied as having VPI symptoms for
our analysis through 3 different criteria: those with post-
operative VPI, hypernasality, or who underwent a second
speech surgery. Patients with normal speech outcomes were
considered not to have any evidence of VPI. Data were ana-
lyzed using descriptive statistics, χ
2
tests, and Kruskal-Wallis
tests. Signicance was set at P<0.05.
Quality Assessment
Three authors (M.X., D.I., J.K.) assessed 4 factors of study
methodology to determine the risk of bias present in the in-
cluded studies. Judgements were made by one of the authors
and checked by one other. The factors included:whether the
speech assessor was unblinded to the patients surgical or
medical history (detection bias), whether there were speech as-
sessment outcomes not reported for the entire study population
(attrition bias), whether there were any speech assessments de-
scribed in the methods that were not reported in the results
(reporting bias), and whether a formal speech protocol was used
for the speech assessments (other bias). Each bias was rated as
either high risk, low risk or unable to be assessed. Selection and
performance bias were not assessed as this review covered sur-
gical interventions. The authors believed it to be likely that
random sequence generation and allocation concealment would
not be present in the included studies, as this would mean
withholding the appropriate surgical treatment from some pa-
tients. In addition, for performance bias, the authors believed it
to be unlikely that the patients and study personnel would be
blinded to type of surgical interventions. Therefore, the authors
assumed selection and performance bias to be present in the
included studies. Publication bias was assumed to be present as
the authors only assessed published peer-reviewed studies.
RESULTS
Study Selection
The search identied 4740 original articles (Fig. 1). A total of
4636 were excluded and 104 full-text articles were separately
The Journal of Craniofacial Surgery Volume 34, Number 6, September 2023 Systematic Review of Postoperative VPI
Copyright © 2023 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of Mutaz B. Habal, MD. 1645
reviewed by 3 authors (M.X., D.I., and J.K.). Sixty-nine of the
104 were excluded for failure to report age at primary
palatoplasty (n =16), failure to report complete speech
outcomes after primary palatoplasty (n =36), or reporting the
presence of a syndrome in their populations (n =17). The 35
included studies reported mean age at palatoplasty and VPI-
related outcomes.
Quality Assessment
Quality assessment of the 35 included studies is shown in
Fig. 2. Most studies (n =28, 80%) failed to specify whether
speech assessment was blinded. All studies were low risk for
reporting bias. Six (17%) did not include speech assessment
results for the entire study population. Formal speech
assessment protocols were reported in 26 studies (74%).
Study Characteristics
Twenty-one included studies (60%) were retrospective and 14
(40%) were prospective. Most were published between 2010 and
2020 (n =27, 77%) (Supplemental Appendix 1, Supplemental
Digital Content 1, http://links.lww.com/SCS/F263). The authors
of these 35 studies were associated with institutions in 16
countries, with the majority (n =11, 31%) having authors af-
liated with institutions in the United States of America (Sup-
plemental Appendix 1, Supplemental Digital Content 1, http://
links.lww.com/SCS/F263). The speech outcome used to measure
VPI presence was reported as hypernasality in 10 studies (29%),
incidence of secondary surgery in 18 studies (51%), and VPI in 7
studies (20%). The majority of studies with speech assessments
(n =33, 94%) examined speech outcomes at age 4 years or older
(n =29, 88%). This age group is unlikely to develop subsequent
VPI and earlier assessment is often unreliable due to age.
32
Operative Age
The 35 included studies had 10,795 patients with a weighted
mean operative age of 15.7 months (range: 3.1182.9 mo)
(Supplemental Appendix 1, Supplemental Digital Content 1,
http://links.lww.com/SCS/F263), and 20% (n =2186) had signs
of postoperative VPI. The majority (n =8489, 78.6%) had pal-
atoplasty between age 6 and 12 months (Q2) (Supplemental
Table 1, Supplemental Digital Content 2, http://links.lww.com/
SCS/F264), and this group had the lowest occurrence of VPI
(18.1%, P<0.010). There was a signicant difference between
Q3 and Q4 (P=0.007), but not between Q4 and Q1 (P=0.409,
Supplemental Table 2, Supplemental Digital Content 2, http://
links.lww.com/SCS/F264).
Surgical Techniques
Becaues of the heterogeneity in reporting surgical technique
across studies an analysis of the VPI risk per procedure was not
possible. Nine studies (26%) mentioned only the name of the
surgical procedure used, 15 studies (43%) described the surgical
procedure in writing or diagrams, and 7 (20%) studies men-
tioned only the name of the surgical technique and cited a cleft
palate repair technique paper. An assessment of surgical tech-
niques was not applicable for 4 retrospective papers that did not
collect technique information. Eleven studies (31%) included
modications to techniques.
DISCUSSION
This is the largest review to date examining the risk of devel-
oping VPI after primary palatoplasty with respect to operative
timing and technique; illustrating that up to 20% of primary
cleft palate patients are at risk of postoperative VPI. Previous
literature reviews report a 20% to 30% incidence of VPI after
primary palatoplasties in non-syndromic patients.
29,33
Risk of
VPI may also depend on surgical technique and expertise, cleft
phenotype, and cleft severity.
27
In previous studies, higher rates
of VPI have been suggested with more severe clefts (Veau III
and IV),
32,34
male sex, shorter palate length, large cleft width,
and patients with cleft lip and palate compared with isolated
cleft palate.
35
This analysis found that primary palatoplasty performed
between ages 6 and 12 months was associated with a sig-
nicantly lower risk of VPI; corroborating the long-standing
idea that operative age plays a signicant role in postoperative
speech outcomes.
36
However, the authors cannot conclusively
advocate for 6 to 12 months as the optimal timeframe owing to
FIGURE 1. The PRISMA flowchart of results. PRISMA indicates Preferred
Reporting Items for Systematic Reviews and Meta Analyses.
FIGURE 2. Results of the risk of bias assessment.
Xepoleas et al The Journal of Craniofacial Surgery Volume 34, Number 6, September 2023
1646 Copyright © 2023 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of Mutaz B. Habal, MD.
a number of reasons: (1) studies included in this analysis lacked
statistical power owing to small sample sizes; (2) timing and
technique were analyzed in isolation without consideration of
other important contributing factors, such as cleft severity,
phenotype, and surgeon expertise; (3) measurement of speech
outcomes lacked standardized assessments; and (4) nearly 80%t
of patients underwent palatoplasty between 6 and 12 months,
which could indicate the inuence of certain protocols. For
example, the Rule of 10s has inuenced many surgeons to
perform cleft lip repair at 10 weeks of age; to ensure adequate
postoperative recovery, the cleft palate in these children was
often repaired after 6 months of age.
37,38
Our results do not indicate an advantage to early (Q1) or late
(Q4) palatoplasty. There is variable consensus on timing of
palatoplasty to optimize speech outcomes. Kaplan suggested
palatoplasty before 6 months to ensure proper anatomy at the
time of speech development at 9 to 12 months.
21
Whereas
Murthy et al. and Zhao et al. reported slightly superior speech
outcomes after late palatoplasty,
9,39
other studies report poorer
speech outcomes.
9,2124
Many surgeons would recommend
performing primary palatoplasty before 12 months of age to
correct cleft palate anatomy before speech patterns have de-
veloped. It is theorized that younger children have more time to
learn and practice speaking with a repaired mechanism, pro-
viding an opportunity to ameliorate erroneous speech
patterns.
36
Intuitively, primary palatoplasty between 0 and
6 months would result in lower rates of VPI, particularly when
compared with primary palatoplasty after 2 years of age, con-
tradicting the ndings of this analysis.
However, the benet of palatoplasties earlier than 6 months
need to be measured against the risk of midface hypoplasia,
which has signicant consequences. Furthermore, early repair
before 6 months may be difcult due to small anatomy and
airways that increase risk for anesthesia complications, airway
obstruction, and lack of respiratory reserve.
40
Variability in reporting standards among the studies limited
our analysis of the timing of cleft palate repair. Among the 35
included studies there was no consistency in (1) the scale/
measurement used to evaluate VPI; (2) reporting of VPI out-
comes; (3) reporting of operative age; and (4) reporting surgical
technique with descriptions or citations. In addition, this review
excluded 52 articles for failure to report basic demographics
within their studies such as mean operative age and complete
postoperative speech outcomes.
Timing of cleft palate repair is variable between centers
globally. This variability also exists in the United States, for
example, patients in the Midwest tend to undergo palatoplasty
later than the West (14.3 versus 13.2 mo) which is even later
than in the Northeast (12.9 mo). Eighty-ve percent of centers
in the United States perform cleft palate repair after 6 months of
age. Even in our institution at the Childrens Hospital Los
Angeles/ University of Southern California there is variation
among surgeons. This variability could be attributed to what
surgeons learned during their training or the practical reasons
that occur from patient presentation to anesthesia preparation.
There are many reasons that contribute to the lack of universal
protocols for cleft palate repair. The search for optimal timing is
a meticulous investigation for optimal speech results against
known iatrogenic effects of palatal surgery demonstrated to
have variable degrees of growth disturbances.
An analysis of surgical technique was not possible owing to a
lack of standardized technique nomenclature. Techniques are
named but, are often modied (described and undescribed).
Less than half the articles included descriptions and/or diagrams
of the techniques used. This was further compounded by in-
consistent reporting on the descriptions of surgical techniques
among the studies and inadequate information to appropriately
classify and compare techniques.
Limitations
Of note, VPI is not denitely a complication of palatoplasty
owing to the natural incidence of VPI due to factors beyond the
surgeons control, such as soft tissue availability, cleft width,
and palatal length. These factors are highly variable and the
most extreme severity may be impossible to overcome with a
singular surgery. More data are needed to better understand the
factors that contribute to the occurrence of VPI. Despite these
inherent factors, this systematic review demonstrates that opti-
mization of technique and timing may reduce VPI. Palatoplasty
has been shown to improve articulation patterns, regardless of
the presence of VPI. However, it is important to note that re-
ducing VPI may not correct all aspects of speech development.
VPI is one component of speech development and palatoplasty
alone has been demonstrated to improve articulation patterns
regardless of VPI.
The generalizability of our report relies on the quality and
consistency of the underlying study data. Biases may result from
retrospective studies and those that did not include details about
blinding reviewers, speech assessment protocols, or surgical
details. Furthermore, confounding factors such as surgeon ex-
pertise, and cleft severity and phenotype, were not analyzed due
to inadequate data. Longitudinal data collection of patients
who undergo palatoplasty is challenging owing to poor long-
term follow-up and lack of standardized outcomes assessments
and metrics. In 2001, the Eurocleft study found that 201 Eu-
ropean cleft centers had 194 different protocols for the man-
agement of unilateral cleft palate alone.
41
Speech outcomes
were dened by rate of secondary surgery, VPI, or hypernasality
instead of a standardized single measurement. As some sur-
geons or centers are more active in treating mild or moderate
VPI, the rate of secondary surgery may not consistently repre-
sent speech outcomes. There was minimal individual patient-
level data reported in the studies, so mean ages for entire
cohorts were utilized and may be subject to outlier inuences
compared with other methods of central tendency. The high
variability in operative age within the Q4 group may also
contribute to the statistical differences seen between this group
and Q2 and Q3. Furthermore, statistical power of included
studies was highly variable owing to small sample sizes. More
accurate data collected through further prospective studies is
crucial to inform timing and technique of primary palatoplasty.
The need for larger longitudinal studies was addressed by the
creation of the Universal Parameters for Reporting Speech
Outcomes, which was cited as a protocol in only in 5
studies.
9,40,4245
Although outside the scope of this paper, we cannot deny the
role that hearing has on speech development. There is variable
consensus in the literature on the extent to which this can im-
pact speech or be a confounding factor for VPI. If we had a
better understanding of the implications of palatoplasty on
hearing and effusions, it may help to construct more sophisti-
cated treatment algorithms. As future studies are planned, it
would be ideal to include these factors to gain a more complete
understanding of the implications of an important and common
surgery.
Recommendations
The authors would recommend future studies on cleft palate
repair include the following: (1) a thorough description of the
surgical technique either through diagrams or narratives; (2) a
The Journal of Craniofacial Surgery Volume 34, Number 6, September 2023 Systematic Review of Postoperative VPI
Copyright © 2023 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of Mutaz B. Habal, MD. 1647
standardized assessment of resonance and articulation after the
Universal Parameters for Reporting Speech Outcomes com-
pleted by blinded speech language pathologists; and (3) a mean
age of the study population stratied by surgical technique and
cleft phenotype. Few included studies incorporated all these
elements.
9,43,46
Further recommendations include improved
data collection methods facilitated by (1) standardized metrics
of evaluation; (2) national audits completed by blinded speech
language pathologists to report on speech outcome of each cleft
center for further comparison between international centers;
and (3) central and shared databases. In addition, the tendency
for surgeons to only learn one palatoplasty technique is a lim-
iting factor for comparison of surgical techniques as surgeon
skill is a confounding factor for outcomes. In one study of this
review, surgeons were taught a second technique to serve as a
control against themselves.
47
Despite the intention, there will
still be a bias towards the technique the surgeon has more initial
experience with, making this confounding factor difcult to
account for. As technology evolves, opportunities to utilize ar-
ticial intelligence to help us more readily obtain results and
impact treatment algorithms are an option but need to be well
organized. Organizations such as Operation Smile have started
to explore ways to evaluate speech through articial intelligence
to increase access to care low-resource settings. Further research
on this topic is required.
Velopharyngeal insufciency is a complication that causes
substantial morbidity and developmental delay as the childs
ability to communicate in both social and educational settings is
impaired. This systematic review highlights the lack of high-
powered, well-designed studies in primary cleft palate repair
which limits our ability to understand optimal timing and
techniques that would limit VPI occurrence. Furthermore, low-
income and middle-income countries (86% of the worlds pop-
ulation) are highly under-represented in the literature. These
gaps in the literature should be seen as an opportunity to answer
an important question that could substantially reduce morbidity
and improve quality of life. This paper should be viewed as a
call-to-action to generate: (1) high-quality research from
thoughtfully designed studies; (2) greater global representation;
and (3) global consensus once the high-quality evidence has
been reviewed. This consensus can only be encouraged through
collaboration and working towards a common goal of ensuring
best outcomes for patients requiring primary palatoplasty.
CONCLUSION
Palatoplasty is life-changing surgery; however, our study
demonstrates that 20% of patients who undergo primary
palatoplasty are at risk of developing VPI. Despite inherent
factors that may be difcult to overcome in patients with cleft,
such as cleft width, soft tissue availability, and palatal length,
it is still important that we strive for optimal results for both
technique and timing. Although earlier cleft palate repairs
suggest diminished rates of VPI, optimal timing for primary
palatoplasty cannot be concluded from this study. Similarly,
our study did not uncover a statistically superior surgical
technique to limit postoperative VPI, and could not account
for important confounders, such as surgeon skill, cleft se-
verity, and phenotype, owing to inadequate published data.
This is further complicated by inconsistent nomenclature of
techniques and the lack of widely accepted standardized pro-
tocols and outcomes metrics for cleft surgery. This study
demonstrates the need to standardize technique classication
and speech evaluation to improve data collection and analysis.
Future studies based on more reliable data can inform
palatoplasty timing and technique to minimize VPI, while
accounting for the risk of midface hypoplasia.
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The Journal of Craniofacial Surgery Volume 34, Number 6, September 2023 Systematic Review of Postoperative VPI
Copyright © 2023 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of Mutaz B. Habal, MD. 1649
Article
Background Very little is known about how the velopharynx and levator veli palatini muscle develop in utero. The purpose of this study was to describe craniofacial, velopharyngeal, and levator veli palatini dimensions in a group of infants born prematurely and imaged before 40 weeks gestation. Methods A retrospective, descriptive study design was utilized to examine the MRI scans of 6 infants less than 40 weeks’ gestation. Imaging was initially completed for medically necessity and pulled from patients’ charts retrospectively for the purpose of this study. Craniofacial, velopharyngeal, and levator veli palatini dimensions were analyzed. Results All linear measures were consistently shorter across all variable categories. While effective VP ratio was less favorable for speech in infants under 40 weeks’ gestation, angle measures such as LVP angle of origin, NSB angle, SNA angle, and SNB angle were relatively unchanged. Conclusions Linear craniofacial, VP, and LVP variables tend to be smaller in infants under 40 weeks’ gestation than those reported within the first 6 months of life while angulation is relatively similar. Future research in this area may be relevant to better diagnosis of craniofacial conditions in utero.
Article
Speech production in general and in patients with cleft palate in particular is multifactorial. In addition to the complex velopharyngeal mechanism, all structures of the oral cavity have some contribution for correct speech production. Therefore, in addition to the velopharynx the surgeons and other cleft/craniofacial team members need to perform a thorough and complete evaluation of all structures to fully appreciate the causative factor(s) responsible for inadequate speech production after palatoplasty and to prescribe the most appropriate, personalized management plan. The purpose of this communication is to stress the importance of areas other than the velopharyngx that could have a negative impact on speech. More specifically, the issues of palatoplasty failures and palatal revisions will be presented. This is an area of significant importance and could represent the first line of defense before considering procedures altering the anatomy of the velopharynx, such as pharyngeal flaps, pharyngoplasties, and others. Issues covering the effects of skeletal and dental problems, such as malocclusion, partial or complete edentulism can also affect speech but are outside the scope of this communication.
Article
Definitive oronasal separation through closure of the velopharyngeal (VP) sphincter is necessary for the development of normal speech and feeding. Individuals with velopharyngeal incompetence or insufficiency often exhibit hypernasal speech, poor speech intelligibility, and nasal regurgitation. Assessment of VP sphincter function using nasopharyngoscopy is a key element in identifying VP dysfunction. A foundational understanding of normal anatomy and physiology of the velopharyngeal mechanism is paramount to successful diagnosis. This includes recognition of 4 distinct VP sphincter closure patterns: coronal, sagittal, circular, and circular with Passavant’s ridge. In this study, the authors showcase 2 patients with velopharyngeal competence who presented to an ear, nose, and throat clinic for nasopharyngoscopic evaluation. This study sought to demonstrate the use of nasopharyngoscopy to recognize velopharyngeal closure patterns and discuss how they may influence the surgical management of VP dysfunction.
Article
Full-text available
A fenda palatina é um defeito de causa multifatorial que acontece ainda na vida intrauterina, pois não há completa fusão dos processos maxilares. Dessa forma, a palatoplastia visa o reparo da fissura e dispõe de técnicas que atuam para a melhoria da qualidade de vida do paciente. Objetivo: Avaliar as técnicas utilizadas na cirurgia de palatoplastia, destacando a que apresenta melhores resultados quanto ao manejo e pós-operatório. Metodologia: Estudo de revisão integrativa da literatura, realizando busca em bases de dados Scielo e Pubmed no período de janeiro a outubro de 2023. Os artigos selecionados tiveram corte temporal de 2004 a 2023, e dos 545 artigos encontrados, 25 foram utilizados. Resultados: Os autores relataram a prevalência das fissuras e sua classificação. Além disso, quanto as técnicas, abordaram resultados semelhantes nas taxas de sucesso pós-operatório e manejo, ademais, chegaram à conclusão de que existem técnicas que aumentam a incidência de fistula e da nasalância. Discussão: Houve debate dos autores quanto a predileção das fissuras palatinas. Houve discordância quanto a preferência da cirurgia em um estágio e a de dois estágios. A técnica de Furlow e Bardach apresentaram resultados semelhantes e demostraram melhores taxas de sucesso. Conclusão: Portanto, conclui-se que a escolha da técnica cirúrgica quanto ao manejo, fica a escolha do profissional. Já na efetividade do tratamento, as técnicas de Furlow e Bardach demostraram serem mais eficazes.
Article
Full-text available
Background: This systematic review aims to inform the development of a screening tool which pre-operatively predicts which children are likely to develop velopharyngeal insufficiency, one of the causes of poor speech outcomes, following cleft palate repair. This would be highly beneficial as it would inform pre-operative counselling of parents, allow targeted speech and language therapy, and enable meaningful comparison of outcomes between surgeons, techniques, and institutions. Currently, it is unclear which factors influence speech outcomes. A systematic review investigating the non-interventional factors which potentially influence speech outcomes following cleft palate repair is warranted. This may be illuminating in itself or provide foundations for future studies. Methods: A systematic review will be carried out according to Cochrane methodology and reported according to PRISMA guidelines (PLoS Med 6: e1000097, 2009). Systematic review software will be used to facilitate three-stage screening by two independent reviewers experienced in cleft lip and palate. Thereafter, data extraction and GRADE assessment will be performed in duplicate by five independent reviewers experienced in cleft lip and palate. Studies reporting the proportion of patients who were recommended or underwent secondary speech surgery for velopharyngeal insufficiency following primary surgery for cleft palate will be included. The study findings will be tabulated and summarised. The primary outcome measure will be further speech surgery (either recommended or performed). The secondary outcome measure will be perceptual speech assessment for the presence of velopharyngeal insufficiency. A meta-analysis is planned. However, if this is not possible, due to the anticipated marked heterogeneity of study characteristics, pre-operative assessment, and the recorded outcome measures, a narrative synthesis will be undertaken. Discussion: This systematic review may provide sufficient data to inform the development of a screening tool to predict the risk of velopharyngeal insufficiency prior to cleft palate repair. However, it is anticipated that these findings will provide the foundation for future studies in this area. Systematic review registration: Registered on 19 December 2016 with PROSPERO CRD42017051624.
Article
Full-text available
Studies on the impact of cleft palate surgery on speech with stringent methodology are called for, since we still do not know the best timing or the best method for surgery. The purpose was to report on speech outcome for all Swedish-speaking 5-year-olds born with a non-syndromic unilateral cleft lip and palate (UCLP), in 2008–2010, treated at Sweden’s six cleft palate centres, and to compare speech outcomes between centres. Speech was assessed in 57 children with percent consonants correct adjusted for age (PCC-A), based on phonetic transcriptions from audio recordings by five independent judges. Also, hypernasality and perceived velopharyngeal function were assessed. The median PCC-A for all children was 93.9, and medians in the different groups varied from 89.9 to 96.8. In the total group, 9 children (16%) had more than mild hypernasality. Twenty-two children (38.5%) were perceived as having competent/sufficient velopharyngeal function, 25 (44%) as having marginally incompetent/insufficient velopharyngeal function, and 10 children (17.5%) as having incompetent/insufficient velopharyngeal function. Ten children were treated with secondary speech improving surgery and/or fistula surgery. No significant differences among the six groups, with eight to ten children in each group, were found. The results were similar to those in other studies on speech of children with UCLP, but poorer than results in normative data of Swedish-speaking 5-year-olds without UCLP. Indications of differences in frequency of surgical treatment and speech treatment between centres were observed.
Article
Full-text available
The aim of the present study is to test the feasibility of modified Z-plasty palatoplasty for cleft palate repair in surgeries and provide a new surgical method. Forty cleft palate patients were selected as participants and divided into 2 groups in random. Twenty patients in the experiment group were treated by modified Z-plasty palatoplasty while the other 20 patients in the control group by double opposing Z-plasty and Sommerlad palatoplasty. By evaluating and observing postoperative velopharyngeal movement, speech intelligibility, nasal leaking, analysis of CSL (Computer Structure Language) and X-ray velopharyngeal lateral radiographs, Modified Z-plasty palatoplasty achieved better results than traditional operation. Satisfactory linguistic effects on incomplete cleft palate can be observed after modified Z-plasty palatoplasty treatment. So this method may be used as a clinical choice.
Article
Full-text available
Background:. To determine best practices, surgeons who perform cleft palate surgery or surgery for velopharyngeal insufficiency need to be able to compare their outcomes in normalizing the velopharyngeal valve. Methods:. We conducted a comprehensive review of articles that reported speech/resonance outcomes following palatoplasty or surgery for velopharyngeal insufficiency. We analyzed protocols that were used and how the results were reported. We found 170 articles, published between 1990 and 2014, that met our inclusion criteria. Results:. Most studies (66%) had a sample size of
Article
Full-text available
Background There is a lack of high-level evidence on the surgical management of cleft palate. An appreciation of the differences in the complication rates between different surgical techniques and timing of repair is essential in optimizing cleft palate management. MethodA comprehensive electronic database search will be conducted on the complication rates associated with cleft palate repair using MEDLINE, EMBASE, and the Cochrane Central Register of Controlled Trials. Two independent reviewers with expertise in cleft pathology will screen all appropriate titles, abstracts, and full-text publications prior to deciding whether each meet the predetermined inclusion criteria. The study findings will be tabulated and summarized. The primary outcomes will be the rate of palatal fistula, the incidence and severity of velopharyngeal insufficiency, and the rate of maxillary hypoplasia with different techniques and also the timing of the repair. A meta-analysis will be conducted using a random effects model. DiscussionThe evidence behind the optimal surgical approach to cleft palate repair is minimal, with no gold standard technique identified to date for a certain type of cleft palate. It is essential to appreciate how the complication rates differ between each surgical technique and each time point of repair, in order to optimize the management of these patients. A more critical evaluation of the outcomes of different cleft palate repair methods may also provide insight into more effective surgical approaches for different types of cleft palates.
Article
Background: The treatment plan for cleft lip and palate varies among centers and requires long-term evaluation of its final outcome. Methods: A consecutive series of patients born from 1994 to 1996 were reviewed. Inclusion criteria were complete unilateral cleft lip and palate, undergoing all treatment procedures performed by the team, and continuous follow-ups until 20 years of age. Exclusion criteria were incomplete data, having microform cleft lip on the contralateral side, presence of the Simonart band, and other abnormalities. Results: A total of 72 patients were included. Average age at final evaluation was 21.3 years; 83.3 percent of patients underwent one-stage rotation-advancement lip repair and 16.7 percent underwent two-stage repair with an initial adhesion cheiloplasty. All patients underwent palate repair using the two-flap method at an average age of 12.3 months. Velopharyngeal insufficiency occurred and required surgical interventions in 19.4 percent during the preschool age and in 16.7 percent at the time of alveolar bone grafting; 56.9 percent of patients underwent secondary lip/nose revision during the growing age. Regular orthodontic treatment was administered to 34.7 percent of patients between 12 and 16 years of age. Orthodontic treatment and orthognathic surgery were applied in 37.5 percent of the patients after maturity. The average number of surgical procedures to complete the treatment was 4.8 per patient. Conclusions: This treatment protocol provided generally acceptable final outcome after the 20-year follow-up. Some results were less ideal and have resulted in modifications of the planning and methods in the protocol. Clinical question/level of evidence: Therapeutic, IV.
Article
Objective Early palate repair is recommended to minimize the development of disordered speech. We studied the speech outcome of late palate repair in 131 patients. The success of late palate repair is questioned because of the persistence of learned, compensatory misarticulations that are difficult to correct in spite of the establishment of correct palatal anatomy and a competent velopharyngeal mechanism. The objective of this study is to highlight the speech results following late primary repair of the palate. Settings and Design Retrospective analysis of speech outcomes in 131 patients with cleft lip and palate who underwent primary palate repair after the age of 10 years between November 2000 and December 2004. None of the patients had received supervised institution-based speech therapy. However, all patients were counseled and oriented and demonstrated the correct place and manner of articulation for the phonemes misarticulated by them. Preoperative and 6- to 12-month postoperative speech samples were assessed within the parameters of articulation, hypernasality, nasal air emission, and speech intelligibility. Outcome The analysis indicated improvement in all speech parameters leading to an overall improvement in postoperative intelligibility for most patients. Conclusion Although definite improvement occurs in all parameters of speech following late primary palate repair, residual speech problems persist in most patients, requiring further evaluation and appropriate treatment.
Article
Objective To investigate whether delayed hard palate repair resulted in better midfacial growth in the long term than previously achieved with “conventional” surgical methods of palatal closure. Design and Setting Long-term cephalometric data from patients with unilateral cleft lip and palate were available from two Scandinavian cleft centers. The patients had been treated by different regimens, particularly regarding the method and timing of palatal surgery. Patients were analyzed retrospectively, and one investigator digitized all radiographs. Patients Thirty consecutively treated subjects from each center, with cephalograms taken at three comparable stages between 10 and 16 years of age. Results and Conclusions Patients whose hard palates were repaired late (early soft palate closure followed by delayed hard palate repair at the stage of mixed dentition) had significantly better midfacial development than patients in whom the hard palate was operated on early with a vomer flap, and then during the second year of life, the soft palate was repaired with a push-back procedure. As the growth advantage in the delayed hard palate repair group was accomplished without impeding long-term speech development, the delayed repair regimen proved to be a good alternative in surgical treatment of patients with unilateral cleft lip and palate.
Article
Background: A lack of high-level evidence exists on the outcomes of different cleft palate repair techniques. A critical appreciation for the complication rates of common repair techniques is paramount to optimize cleft palate care. Methods: A literature search was conducted for articles on the measurement of fistula and velopharyngeal insufficiency (VPI) rates following cleft palate repair. Study quality was determined using validated scales. The heterogeneity between studies was evaluated using the I2 statistic. Random-effect model analysis and forest plots were used to report pooled relative risks (RRs) with 95% confidence intervals for treatment effect. P-values of 0.05 were considered statistically significant. Results: Of 2386 studies retrieved, 852 underwent screening and 227 met inclusion criteria (130 studies (57%) on fistulas and 122 studies (54%) on VPI). Meta-analyses were performed using 32 studies. The Furlow technique was associated with less postoperative fistulae than the von Langenbeck and Veau/Wardill/Kilner techniques (RR = 0.56 [0.39-0.79], p < 0.01 and RR = 0.25 [0.12-0.52], p < 0.01, respectively). One-stage repair was associated with less fistulae compared to two-stage repair (RR = 0.42 [0.19-0.96], p = 0.04). The Furlow repair was also associated with a less VPI than the Bardach palatoplasty (RR = 0.41 [0.23, 0.71], p < 0.01), and the one-stage repair was associated with a reduction in VPI rates compared to two-stage repair (RR = 0.55 [0.32, 0.95], p = 0.03). Conclusion: The Furlow repair is associated with less risk of fistula formation than the von Langenbeck and Veau/Wardill/Kilner techniques and less VPI compared to the Bardach repair. One-stage repair is associated with less risk of fistula formation and VPI than two-stage repair.