ORIGINAL ARTICLE
Incidence of Fistula Formation and Velopharyngeal
Insufficiency in Early Versus Standard Cleft Palate Repair
Michael J. Eliason, MD, Stephen Hadford, MD,y Lauren Green, MPH,z
and Travis Reeves, MD§
Abstract: The goals of cleft palate repair are well-established;
however, there does exist difference in practice patterns regarding
the most appropriate patient age for palatoplasty. The optimal timing
is debated and influenced by cleft type, surgical technique, and
the surgeon’s training. The objective of this study was to compare
the rates of post-operative fistula formation and velopharyngeal
insufficiency (VPI) in ‘‘early’’ versus ‘‘standard’’ cleft palate repair
Downloaded from http://journals.lww.com/jcraniofacialsurgery by BhDMf5ePHKbH4TTImqenVPVWEjt0ik0+2SWL5rxP3YjdkSkST9W0NTw4iOz49ejv1qXnvfidNCk= on 07/05/2020
in a cohort of patients treated at a single craniofacial center.
A retrospective chart review identified 525 patients treated for
cleft palate from 2000 to 2017 with 216 meeting inclusion criteria.
‘‘Early repair’’ is defined as palatoplasty before 6-months of age
(108 patients). ‘‘Standard repair’’ is palatoplasty at or beyond
6-months old (108 patients). Rates of fistula formation were found
to be significantly higher in early repairs (Chi-square statistic 9.0536,
P value ¼ 0.0026). Development of VPI was not significantly
different between the 2 groups (Chi-square statistic 1.2068,
P value ¼ 0.27196). As expected, the incidence of post-palatoplasty
VPI was significantly higher in patients who had a post-operative
fistula when compared to those who healed without fistula formation
(Chi-square statistic 4.3627, P value ¼ 0.0367).
There is significant debate regarding the optimal timing of cleft
repair to maximize speech outcomes and minimize risks. The authors’
data show that post-operative fistula formation occurs at a higher rate
when performed prior to 6 months old. Furthermore, while the rate of
VPI was not significantly affected by age at time of surgery, it was
significantly higher in those who experienced a post-operative fistula.
From the Naval Medical Center Portsmouth, Department of Otolaryngol-
ogy, Portsmouth, VA; yCleveland Clinic, Head and Neck Institute,
Cleveland, OH; zHealthcare Analytics and Delivery Science Institute;
and §Children’s Hospital of the King’s Daughters, Department of
Otolaryngology, Norfolk, VA.
Received August 4, 2019.
Accepted for publication December 16, 2019.
Address correspondence and reprint requests to Michael J. Eliason, MD,
Department of Otolaryngology, 620 John Paul Jones Circle, Ports-
mouth, VA 23708; E-mail: mitcheliason@hotmail.com,
Michael.J.Eliason4.mil@mail.mil).
The views expressed in this article are those of the author and do not
necessarily reflect the official policy or position of the Department of the
Navy, Department of Defense, or the U.S. Government.
One of the authors is a military service member. This work was prepared as
part of his official duties. The authors have no conflicts of interest to
disclose.
Supplemental digital contents are available for this article. Direct URL
citations appear in the printed text and are provided in the HTML and
PDF versions of this article on the journal’s Web site (www.jcraniofa-
cialsurgery.com).
Copyright # 2020 by Mutaz B. Habal, MD
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000006307
980 The Journal of Craniofacial Surgery  Volume 31, Number 4, June 2020
Copyright © 2020 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
Key Words: Cleft palate, craniofacial, palatoplasty, surgical
timing
(J Craniofac Surg 2020;31: 980–982)
O ver 2600 children are born annually in the United States with a
cleft palate (CP), and isolated orofacial clefts represent one of
the most common birth defects.1– 2 The goals of CP repair include
separating the nasal and oral cavities to establish normal velophar-
yngeal function and to preserve maxillofacial growth.3 This is
typically accomplished through a meticulous multilayer closure
to prevent fistula formation. Though the goals of cleft palate repair
are mostly agreed upon, some aspects of care are debated and vary
amongst surgeons and craniofacial centers. One specific contro-
versy is the optimal timing for palate repair; this decision is
influenced by cleft type/width, surgical protocol, and training
backgrounds, but all seek to optimize outcomes and minimize
complications. Furthermore, the impact of the timing specifically
on the rate of post-operative fistula formation and speech outcomes
is unclear, and there is limited data with conflicting conclusions in
the literature. Most centers agree that performing surgery prior to
18-months of age is ideal, and the majority perform repair around
9 to 10 months to allow closure of the oro-nasal fistula prior to
speech development. The plastic surgery literature specifically
highlights earlier repair, and some limited studies suggest a better
speech outcome.3– 5 Our institution is unique in that we utilize both
otolaryngology-trained and pediatric plastic surgery- trained sur-
geons for cleft repair. The two otolaryngologists perform repair at
the standard time (8–10 months), and the plastic surgeons perform
an earlier repair (<6 months) in select patients. Therefore, the
objective for this study was to assess the effect of surgical timing on
the rate of post-operative palatal fistula formation and velophar-
yngeal insufficiency (VPI) at a single, tertiary care craniofacial
center.
MATERIALS AND METHODS
After obtaining approval from the local Institutional Review Board
(IRB), a retrospective chart review of outpatient records from a
single craniofacial center associated with The Children’s Hospital
of King’s Daughters (CHKD), a tertiary care medical center, was
performed over a seventeen-year period from January 2000 to July
2017. Patients were included if they were born after January 1,
2000, underwent primary cleft palate repair at CHKD, and had
sufficient medical record data documented by an otolaryngologist
or plastic surgeon from CHKD to permit assessment for post-
operative complications. Patients were excluded if their surgery
was done at an outside institution, if there was no documented
follow-up, or if their Veau classification of cleft palate could not be
determined from record review. Five hundred twenty-five patients
were initially identified as being followed for cleft palate during this
timeframe and 216 met all inclusion criteria. Primary outcome data
included the patient age at time of cleft palate repair, incidence of
post-operative palatal fistula and/or velopharyngeal insufficiency.
The Journal of Craniofacial Surgery  Volume 31, Number 4, June 2020
Additional data included gender, associated syndrome, cleft type,
fistula requiring surgery, and VPI treatment (speech therapy versus
surgery). Both paper and electronic medical records were searched
when looking for patient information. ‘‘Early repair’’ was defined as
palatoplasty before 6-months of age. ‘‘Standard repair’’ was defined
as palatoplasty at or beyond 6-months of age.
All statistical analyses were performed in collaboration with the
Eastern Virginia Medical School (EVMS) Healthcare Analytics and
Delivery Science Institute (HADSI) using SAS version 9.4 (SAS
Institute, Cary, NC). To test for violations of normalcy, univariate
analysis was performed to examine the distribution of the only
continuous variable, patient age. Patient descriptive statistics of
categorical variables were summarized and presented as count
(percent). Association of fistula and VPI outcomes were tested
between early palate repair (<6 months) and late palate repair (6
months), patients with an associated syndrome, gender, and cleft
palate type using Pearson Chi-square test. The incidence of fistula
development and VPI were calculated and compared between early
and late palate repair groups. Multiple logistic regression models
were fit using maximum likelihood estimates to determine inde-
pendent predictors of fistula formation and VPI outcomes including
those predictors that were statistically significant in univariate
analyses. All hypothesis testing was carried out at the 95% signifi-
cance level, unless otherwise specified, with a P value of <0.05
accepted as statistically significant.
RESULTS
Supplemental Digital Content, Table 1, http://links.lww.com/SCS/
B228, details the demographics of the 216 patients included in the
study that underwent cleft palate repair surgery at CHKD between
January 1, 2000 and July 31, 2017 (Supplemental Digital Content,
Table 1, http://links.lww.com/SCS/B228). Eight surgeons with
either plastic surgery or otolaryngology training performed the
216 surgeries. The age at initial cleft palate repair ranged from
0.20 to 25.5 months (mean, 6.6 months 5.5). Patient age values
were skewed towards the natural limit of 0 and were not normally
distributed (t ¼ 17.61, P < 0.0001). Details regarding the incidence
of cleft palate based on their Veau classification, associated
syndrome, and speech evaluations are shown in Supplemental
Digital Content, Table 1, http://links.lww.com/SCS/B228. The
Veau classification was used as a simple and standardized means
of communicating the extent of a palatal cleft.
The incidence of fistula development in the early palate repair
group was 39.8% and 21.3% in the late repair group, with a
significantly increased odds ratio of 2.45 (P ¼ 0.0030, 95% CI
1.357, 4.429). The incidence of fistula development for all types
of reparative surgery was 31.0%; however, this shows a dramatic
decrease over time (Fig. 1), and current rates are consistent with
national averages. Supplemental Digital Content, Table 2, http://
links.lww.com/SCS/B228, shows the association between develop-
ing a fistula and the independent patient predictors. Unilateral clefts
and clefts involving the hard and soft palate had the highest
incidence of fistula, 37.5% and 30.0% respectively. However, there
was not a significant association between cleft type and fistula
formation (x2 U 4.0045, P ¼ 0.2610). There was also no significant
difference in fistula formation between males and females
(x2 U .8702, P ¼ 0.3509). Median age was significantly lower in
patients who formed a fistula (4.0 months) compared to patients
who did not form a fistula (6.0 months) (Z ¼ 2.883, P ¼ 0.0039).
Presence of an associated genetic syndrome did not differ signifi-
cantly in fistula formation.
Only 15.1% of patients who underwent cleft palate surgery
developed clinically relevant velopharyngeal insufficiency. Early
palate repair patients had a slightly higher rate of VPI compared
to late palate repair patients, 19.7% and 13.8% respectively
# 2020 Mutaz B. Habal, MD
Copyright © 2020 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
Fistuale and VPI After Palatoplasty
FIGURE 1. The annual rate of post-operative fistula formation and VPI
downtrended at this institution during the timeframe studied. This is
consistent with the decreasing rate of ‘‘early’’ repairs being performed at this
institution.
(Supplemental Digital Content, Table 3, http://links.lww.com/
SCS/B228). However, the association between early palate repair
and late palate repair and VPI was not statistically significant
(x2 ¼ 1.2483, P ¼ 0.2639). Similar to fistula formation, the median
age for patients with VPI after cleft surgery was lower than patients
who did not have VPI, but the difference was not statistically
significant. Patients with fistula development post-surgery had
statistically significantly higher rates of VPI (23.8%) compared
to patients who did not develop a fistula (12.3%) (x2 ¼ 9.0536,
P ¼ 0.0026). The odds of a patient having VPI after formation of a
fistula were 2.18 times higher than those patients who did not have a
fistula formation after surgery (P ¼ 0.0475, 95% CI 1.00, 4.73).
Unlike fistula formation, there was a significant relationship
between having an associated syndrome and developing VPI
post-surgery (x2 ¼ 5.6266, P ¼ 0.0177). Fistula formation and
having an associated syndrome were both identified as significant
predictors of VPI in the logistic regression model.
Importantly, both the upper and lower extremes of age appear to
have much a higher incidence of fistula, with 0 to 3.9 months and
>12 months showing rates of 50% and 29.2%, respectively. A
similar pattern is seen when looking at VPI, with the percentage of
patients developing VPI at 0 to 3.9 months and >12 months being
19.4% and 25.0%, respectively. The age group with both the lowest
incidence of fistula and VPI is within the 6 to 7.9-month group.
DISCUSSION
While the goal and techniques for surgical correction of cleft palate
are well established, the opinion on timing for repair remains
heterogeneous due to the perceived impact on maxillofacial growth,
speech development, and avoidance of velopharyngeal insufficiency
and palatal fistula.6-10 Advocates of early repair consider that nor-
malization of the palate optimizes a child’s speech development.
Specifically, it is thought that coordination of the oropharyngeal
musculature starts in early infancy and that closure of the oro-nasal
fistula permits this prelinguistic development that ultimately leads to
better speech outcomes.11–14 Proponents of the early timing postulate
that early surgery permits healing and resolution of post-surgical
edema prior to onset of typical palatal coordination efforts.11,15 In
addition to these factors, early CP repair may permit breastfeeding at
an earlier age and improve infant-mother bonding.16 In contrast,
advocates of a more delayed repair cite disruption of palatal growth
centers as a cause for impaired maxillofacial growth and resultant
midface hypoplasia.17 The data to support this impairment of midface
growth has been debated and uncertainty persists regarding this
impact of surgical timing and techniques.3,18–20
The optimal timing of CP surgery tends to differ based on the
specialty training of the cleft surgeon. While most cleft surgeries are
done by those trained in plastic surgery, there is a subset of
approximately 20% of the cleft surgeons who are otolaryngologists.
A recent study utilizing the National Surgical Quality Improvement
Program pediatric database found that plastic surgeons and otolar-
yngologists have no difference in timing of cleft lip repair, rhino-
plasty, nor surgical complications. However, plastic surgeons tend
to perform CP repair surgery significantly earlier than otolaryngol-
ogists.3–4 Of note, both plastic surgeons and otolaryngologists
981
Eliason et al The Journal of Craniofacial Surgery
performed the CP repairs in the cohort studied for this retrospective
review with plastic surgeons performing all of the repairs done prior
to 6 months of age.
Historically, some patients at our craniofacial center underwent
extremely early cleft palate repair (<3 months). As far as we know,
this practice has not been studied in the literature and is not standard
nationally. This is no longer practiced at our institution as the
perceived benefits to this type of repair do not outweigh the risks,
particularly with regard to fistula formation. In fact, after removing
the 40 patients who underwent palatoplasty at younger than 3-
months old, the overall incidence of post-operative fistula reduced
from 31.0% to 26.7%. However, the incidence of those who
underwent surgery at 3 to 6 months old was still significantly
higher at 33.8% compared to the 21.3% in those children older than
6-months.
The data in this study does provide insight on the complications of
post-operative VPI, palatal fistula formation, and resultant impact on
the primary outcome of speech development. Specifically, early CP
repair, as defined by performing surgery prior to 6 months of age
showed a statistically significant increase in the rate of palatal fistula
formation. Specifically, the rate of fistula in the early cohort was
twice the rate of those performed after 6 months (40.9% versus
20.0%). Furthermore, covariables such as patient gender, associated
syndrome, and cleft type were assessed to ensure appropriate
data distribution, and none were significantly associated.
The overall rate of VPI was less than the rate palatal fistula
formation. While the rate of VPI was higher in the early group, the
results did not achieve significance. However, VPI was signifi-
cantly higher in those with associated syndromes and as expected in
those who experienced a concomitant palatal fistula. Only prospec-
tive study with careful evaluation of other speech outcomes would
truly uncover the full advantages and disadvantages of early repair
on articulation and compensatory errors.
The main strength of this study is the divergent practice patterns
within a single craniofacial center that allow for comparison of palate
repair in two age groups. This uncommon situation allowed for a
relatively large scale, retrospective analysis that has not been previ-
ously performed. However, there are limitations. Specifically, during
the period reviewed the institution’s means of maintaining medical
records transitioned from paper to electronic, and it is likely that the
216 who met inclusion criteria did not encompass all consecutive cleft
palate surgeries during this time. We estimate that the number missed
during this transition is minimal. Furthermore, given the inherent
faults of a retrospective study, there was no means to ensure exact
parity across all demographics, cleft types, and surgeries performed.
Although we sought to control for various confounding factors,
we cannot ignore that various surgeons with a variety of training
backgrounds performed the operations using different techniques.
While there will likely continue to be debate regarding the
optimal timing of cleft repair to maximize speech outcomes and
minimize surgical complications, the data show that post-operative
fistula formation occurs at a significantly greater incidence when
performed on patients prior to 6 months old. Furthermore, while the
rate of VPI was not significantly affected by age at time of surgery,
it was significantly higher in those who experienced a post-opera-
tive fistula and in those with an associated syndrome. These data are
the result of a retrospective review, but are compelling due to the
sample size, length of follow-up, and divergent practice patterns
that allowed for comparison within a single institution.
982 # 2020 Mutaz B. Habal, MD
Copyright © 2020 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
 Volume 31, Number 4, June 2020
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