KEY WORDS
Cleft palate - palatal fistula - palatoplasty - tongue flap
INTRODUCTION
This study was conducted to evaluate the incidence and management of palatal fistulas
in patients with cleft palate. Cleft lip and palate is one of the most common congenital
anomalies worldwide affecting babies 1:700 live birth.[[1]] Cleft lip and palate together occur in every 1:1289 live births, cleft lip alone
occurs 1:1000 live births, more commonly among the boys and three times more frequent
than the cleft palate alone. Cleft palate alone occurs 1: 2500 live births with a
higher incidence among the female babies.[[2]] Cleft lip and palate is the most frequent diagnosis accounting for 46% of all the
cleft populations.
Cleft palate repair aims to attain the development of normal speech without significantly
impairing maxillary outgrowth, as well as minimising hearing loss and middle ear complications.[[3]] In managing patients with cleft palate, the most controversial issues include the
timing of surgery, speech development and facial growth.[[4]] The ideal age for cleft palate surgery is usually 9–18 months. Speech and hearing
are improved by cleft palate repair before 24 months of age. Delayed closure (after
5 years) is associated with retarded growth of the maxillofacial region.
The incidence of fistulas after palatoplasty ranges from 3% to 38%.[[5]] Larger studies report rates in the range of 10%–20%.[[6]] Palatal fistulas may present as asymptomatic holes or may cause such symptoms as
speech problems, nasal regurgitation of fluids or difficulty in maintaining oral hygiene.
The most common locations for fistulas are at the region around the incisive foramen,
at the posterior nasal spine and the uvula.
All post-operative fistulas are found to be contributed to either failure of healing
or breakdown of the original cleft palate repair. The incidence is highly variable
although the primary cause remains the same in most, which is due to closure under
tension and infection.[[7]] Failure of healing of the palatal wound post-repair may lead to scarring and fistula.[[8]] Anatomically, the cleft size as well as the technique of repair are factors which
influence fistula occurrence.[[9]] Cleft size affects the difficulty of surgical repair and thus, indirectly affects
post-operative maxillary growth.[[10]] Facial and palatal growth retardation following cleft repair is said to be due
to the destruction of blood supply and scar formation.
SUBJECTS AND METHODS
The study determines the incidence and management protocol of cleft palatal fistulas
in a series of primary cleft palate repair surgeries. It is a retrospective analysis
of total 185 palatal fistulas out of which 132 cases had been operated at our institute
for primary palatoplasty, and the rest 53 were the outside-operated cases [[Table 1]]. The technique for primary palatoplasty at our institute was von Langenbeck palatoplasty
and two-flap palatoplasty with radical muscle dissection and posterior sling formation.
The incidence of palatal fistulas and their management by various methods has been
discussed. The gender and the age predilection has also been discussed, laying stress
on the location and size of palatal fistulas along with the rate of complication.
Table 1
Rate of fistula formation
|
Diagnosis
|
Cases operated at our institute
|
Fistula formation in our cases
|
Percentage rate of fistula formation in our cases (%)
|
|
CLP: Cleft lip and palate
|
|
Unilateral CLP
|
1342
|
56
|
04.17
|
|
Bilateral CLP
|
402
|
85
|
21.14
|
|
Incomplete CLP
|
316
|
12
|
03.79
|
|
Total
|
2060
|
153
|
|
The clinical records of the patients with palatal fistulas who underwent fistula repair
between 2004 and 2016 were retrospectively reviewed. This included 132 cases operated
at our institute for primary palatoplasty, and the rest 53 were the outside-operated
cases. Patients included are those who had a complete operative and follow-up medical
records with a minimum follow-up period of at least 2 months. Other variables of interest
are noted for each patient, which include gender, age, type of cleft, location and
size of fistula, method of repair and complication if any reported. The patients with
bilateral as well as unilateral cleft lip and palate were included. Isolated cleft
palate patients were also included in the study. Fistulas could be single or multiple
in number. Anterior palatal fistulas were considered to be in the anterior part of
the hard palate, middle palatal fistulas at the junction of hard and soft palate,
and posterior palatal fistulas were confined to the soft palate. Palatal fistulas
were subdivided into three types depending on their size-small (<2 mm), medium (2–5
mm) and large (>5 mm).
RESULTS
Between 2004 and 2016, a total of 2060 patients were operated for primary cleft palate
repair at our institute, out of which out of which total 153 patients were diagnosed
for palatal fistula. Of these 153 patients, 21 patients were not operated as 10 of
them had pinpoint fistulas, which resolved with time and 11 of them did not turn up
for follow-up and rest of 132 patients were operated for palatal fistula repair. Hence,
the true incidence rate of palatal fistula in our series was 7.427% (153 out of 2060
patients). Out of a total number of 185 palatal fistulas operated at our institute,
122 (65.94%) were anterior palatal fistulas, 45 (24.32%) were middle palatal fistulas,
17 (9.18%) were posterior palatal fistulas and one very large fistula almost whole
of the palate (0.54%). Outside operated cases had more number of anterior palatal
fistulas, and the size of the fistulas was similar to our study (mostly ranging from
2 mm to 5 mm).
In terms of age, the most common age group at which palatal fistula cases turned up
for fistula closure was 6–10 years (23.78%), followed by <5 years (17.29%), 21–25
years (17.29%), 26–30 years (14.05%), 16–20 years (12.43%), 11–15 years (9.73%), 31–35
years (3.78%) and 36–40 years (1.62%).
The reason for less number of patients operated under the age of 5 years is because
this series includes a large number of patients who reported late for palatal repair
or palatal fistula repair (when palate has been operated outside). Most of our own
cases were taken up for palatal fistula repair after 6 months of palatoplasty.
In terms of gender, male predominance was reported as out of total 185 cases of palatal
fistulas operated, 120 were male (64.86%) and 65 were female (35.13%).
Incidence wise, cleft palatal fistulas were most commonly seen in patients having
bilateral cleft lip and palate (21.14%), followed by unilateral cleft lip and palate
(4.17%) and isolated cleft palate (3.79%) [[Table 1]].
Fistula rate was found to be maximum in case of bilateral cleft lip and palate patients
(21.14%) whereas it was comparatively less in case of unilateral cleft lip and palate
patients (4.17%) and incomplete cleft palate patients (3.79%) as shown in [Table 1]. Number of cases operated for anterior palatal fistula were 122, middle palate fistula
was 45, posterior palatal fistula was 17 and one case of large palatal fistula involving
almost whole of palate operated by radial artery forearm flap (RAFF) as shown in [Table 2].
Table 2
Plan of fistula repair
|
Palatal fistula
|
|
Operated at our institute primarily
|
132
|
|
Primarily outside operated
|
53
|
|
Total
|
185
|
|
Anterior palatal fistula
|
|
|
Tongue flap
|
80
|
|
Local flap
|
42
|
|
Total
|
122
|
|
Middle palatal fistula
|
|
|
Langenbeck palatoplasty
|
45
|
|
Posterior palatal fistula
|
|
|
Langenbeck palatoplasty
|
17
|
|
Large palatal fistula almost involving whole palate
|
|
|
Radial artery forearm flap
|
1
|
|
Total
|
185
|
In terms of management, all palatal fistulas were segregated into groups based on
location and size. Location wise, it has been divided into anterior, middle and posterior
palatal fistulas, while according to size, it has been classified into <2 mm (small),
2–5 mm (medium) and >5 mm (large).
Out of 122 anterior palatal fistulas, 26 cases (21.31%) were of the size of <2 mm,
76 cases (62.29%) were of the size of 2–5 mm and 20 cases (16.39%) was of the size
>5 mm.
Out of 45 middle palatal fistulas, 19 cases were of the size (<2 mm), 25 cases (56.53%)
of the size of 2–5 mm and 1 case (2.17%) was of the size of >5 mm.
Out of 17 cases of posterior palatal fistulas, 7 cases (41.18%) were of the size <2
mm, 9 cases (52.94%) were of the size of 2–5 mm and 1 case (5.88%) was of the size
of >5 mm.
Most of the anterior palatal fistulas (80 cases) were closed using tongue flap [[Table 2] and [Figures 1]
[2]
[3]] followed by local flaps (21 cases) which included local mucoperiosteal flaps or
extended alveolar mucoperiosteal flap and mucosal flaps [[Figures 4]
[5]]. Twenty cases of anterior palatal fistulas were closed using redopalatoplasty by
two flap technique [[Figures 6]
[7]
[8]]. One case of anterior palatal fistula of size 2–5 mm was operated using buccal
mucosal flap [[Figures 9]
[10]
[11]].
Figure 1: Anterior palatal fistula >5 mm
Figure 2: Tongue flap attachment
Figure 3: Tongue flap for anterior palatal fistula closure after division and insetting
Figure 4: Anterior palatal fistula 2 mm to be corrected by using local flap (extended alveolar
mucoperiosteal flap)
Figure 5: Fistula closure by local flap - extended alveolar mucoperiosteal flap
Figure 6: Anterior palatal fistula to be corrected by two flap palatoplasty
Figure 7: Two flap palatoplasty for fistula closure
Figure 8: Postoperative follow-up (2 months)
Figure 9: Defect of anterior palatal fistula to be corrected by buccal mucosal flap
Figure 10: Harvesting of buccal mucosal flap
Figure 11: Result after insetting of flap and closure of fistula palatoplasty
Of 80 cases of anterior palatal fistulas closed by tongue flap [[Figures 1]
[2]
[3]], we encountered dehiscence in 3 patients (3.75%). All the three patients were of
the age <5 years and were further managed by flap reattachment. On follow-up, these
cases healed well. In this way, tongue flap was considered as a successful treatment
of choice for managing challenging anterior palatal fistulas hard to repair.[[11]]
Middle and posterior palatal fistulas were treated by redo-palatoplasty using von
Langenbeck technique [[Figures 12]
[13]].
Figure 12: Soft palatal defect to be corrected by langenbeck palatoplasty
Figure 13: Langenbeck palatoplasty for correction of defect
One case of very large palatal fistula involving almost the whole of the palate was
treated by RAFF [[Figures 14]
[15]
[16]].
Figure 14: Radial forearm free flap: Large palatal fistula involving almost whole of palate
Figure 15: Radial forearm free flap: Fistula closure by radial forearm flap
Figure 16: Radial forearm free flap: Post-operative result
The complication rate for tongue flap was reported to be 3.75% (3 cases), due to flap
dehiscence in three patients, which required reattachment. The recurrence rate was
noted in these patients after 6 months of follow-up. In case of local flaps, the complication
rate was 11.9% (5 cases) out of which two cases were of two flap palatoplasty. These
were further managed by redo-palatoplasty and tongue flap in case of comparatively
large palatal fistulas. In case of middle palatal fistulas complication rate was 8.69%
(4 patients), and in case of posterior palatal fistulas, it was 5.88% (1 patient).
DISCUSSION
Despite improved technique of repair of cleft palate, fistula occurrence is still
a possibility either due to an error in surgical technique or due to poor tissue quality
of the patient. The incidence of palatal fistulas is more common in bilateral cases
than unilateral ones, as reported by Musgrave and Bremner.[[8]] Our study also reports a higher incidence of fistula in patients having bilateral
cleft lip and palate (21.14%) as compared to unilateral cleft lip and palate (4.17%)
and isolated cleft palate (3.79%).
About 3.75% of cases operated by tongue flap had residual fistulas due to flap dehiscence.
These were further managed by tongue reattachment. The post-operative period of follow-up
was 6 months.
Michael H. Carstens[[11]] conducted a study encountering anterior palatal fistula in 77.5% of cases. In most
of the cases, this results from failure to achieve control of the anterior nasal floor.
We also report a high incidence of anterior palatal fistulas (65.94%) as compared
to middle palatal fistulas (24.32%) and posterior palatal fistulas (9.18%). One very
large palatal fistula, almost involving the whole of the palate was also reported.
In case of anterior palatal fistulas, the most common technique used for closure included
local flaps (extended alveolar mucoperiosteal flaps and buccal mucosal flaps), two
flap palatoplasty and tongue flap. The condition of native tissue was a good factor
which helped the surgeon to decide the technique to close similar types of fistulas.
Local flaps were used only in those cases where the native tissue (with rugae) was
available. Otherwise, we prefer bringing virgin tissue from other areas. Local flaps
were mostly done in cases <2 mm of size and where the condition of native tissue was
favourable for the closure of fistula. In case of scarred tissue, compromised vascularity
and fibrosis, the local flap cannot be easily mobilised, and chances of flap necrosis
are high.
Although commonly the anterior palatal fistula closure is established by the use of
local flap at times, the size and site of fistula make use of local flap for its repair
is a remote possibility. Hence, for anterior palatal fistula >2 mm size, tongue flap
was used as a workhorse flap in our series of patients. The use of tongue flap gave
promising results in case of difficult anterior palatal fistulas with a shortage of
tissue. The advantage includes the central position of tongue in the floor of mouth
facilitating mobility and positioning of the flap. Success depends on proper flap
elevation, tension-free nasal layer closure, edge-to-edge approximation of flap with
palatal tissues and good tension-free primary closure of donor area near base of the
flap.[[12]] Middle and posterior palatal fistulas were mostly treated using von Langenbeck
palatoplasty. Radial artery forearm-free flap was used in case of very large palatal
fistula. Although Cohen et al. reported recurrence rate of 37%,[[13]] our study shows a very low rate of 7.427% which is in concordance with the study
of Musgrave and Bremner[[8]] with a recurrence rate of 7%. Many authors have published very different data about
the incidence of oronasal fistulas. The lowest incidence (0%) was published by Stewart
et al.,[[14]] followed by 0.7% by Brusati and Mannucci,[[15]] 0.76% by Losee et al.,[[16]] 3.4% by Wilhelm et al.,[[17]] 3.6% by Khosla et al.,[[18]] 4.7% by Inman et al.,[[19]] 8.7% by Muzaffar et al.,[[20]] 12.8% by Phua and Chalain,[[21]] 15% Sommerlad.[[22]] The overall recurrence rate of fistula was nil in case of tongue flap whereas it
was 11.9% in case of local flaps, 8.69% in case of middle palatal fistulas and 5.88%
in case of posterior palatal fistulas. In our series of tongue flap patients, only
three cases of flap dehiscence were noted, which were managed further by reattachment
of flap. None had a residual fistula in 6 months of follow_up. Hence, tongue flap
was successfully used in managing challenging anterior palatal fistulas.[[12]
[23]
[24]]
CONCLUSION
Tongue flap remains the flap of choice for managing very difficult and challenging
anterior palatal fistulas, compared to local flaps. For middle and posterior palatal
fistulas, von Langenbeck palatoplasty gave good results. For very large palatal fistulas,
free flap (radial forearm flap) makes a good choice.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms.
In the form the patient(s) has/have given his/her/their consent for his/her/their
images and other clinical information to be reported in the journal. The patients
understand that their names and initials will not be published and due efforts will
be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
