Etiology
Most often the palatal fistula is located at the junction of the hard and soft palate closure or between the premaxilla and secondary palate. The symptoms depend on size, position, and general velopharyngeal competence. A fistula may also be caused by trauma, tumor, irradiation, or a rare infectious disease, such as midline granuloma, syphilitic gumma, leprosy, noma, or leishmaniasis [
2]. The most common complaint is uncontrolled regurgitation of fluid into the nose. A large fistula also causes obvious speech defects, whereas a small fistula may result in some speech impairment.
Breakdown of primary palatal repair is one of the major causes of palatal fistula, which is related to tension at the site of closure, necrosis, whether the greater palatine vessel was injured during elevation of the flaps or injection, hematoma, or mechanical trauma before flaps heal. Attempts at closure using only local transposition flaps may be successful, although frequently, closure is not achieved and a smaller oronasal fistula will recur.
Additional attempts to gain closure with local tissue alone often result in repeated failure as the thick and immobile, scarred palatal mucoperiosteum leads to closure under tension with subsequent flap necrosis and wound dehiscence.
Tissue from distant sites has been used including tubed pedicle and flaps from the abdomen, arm, neck, or cervicothoracic region. Others have used cheek and nasolabial flaps to close these palatal defects. Although staged distal tissue transfers may be effective, they require multiple operations, are always cumbersome, leave multiple scars, prevent chewing until final flap division, are bulky when inset, and finally transfer skin rather than mucosa to the roof of the mouth.
Methods
Source of Data
We performed a retrospective study of 40 patients with palatal fistulas who were treated with anteriorly based tongue flap in the Department of Oral and Maxillofacial Surgery, S.D.M. College of Dental Sciences and Hospital (Sattur, Dharwad, India) from January 2000 to January 2007.
Patient Selection Criteria
Selection criteria consisted of the following: (1) fistulas present in anterior and midpalate were considered; (2) the size of the palatal fistula not amenable for local flap closure; (3) history of repeated attempts to achieve the closure of the palatal defect; (4) scarred palate and adjacent tissue.
Method of Study
Patients’ preoperative photographs, clinical records, and preoperative speech analysis were recorded. Sizes of the fistulas were measured preoperatively.
Surgical Technique
The operation was performed with the patient receiving general anesthesia. The unaffected nasal side was used for nasotracheal intubation. After routine intra- and extraoral betadine preparation, sterile fistulas were injected with 2% lidocaine with 1:200,000 adrenaline for homeostasis and ballooning of the tissues for ease of dissection.
Incision was performed around the fistulous tract (
Figure 1 and
Figure 2); mucosalized edges were excised (
Figure 3). The nasal layer was identified and carefully dissected to mobilize the nasal layer, and the nasal floor was reconstructed using 4–0 Vicryl (
Figure 4).
The length of the flap was designed such that 1 to 2 cm of additional tissue would span the posterior edge of the palatal defect; the approximate size of the tongue flap was designed using the coverage of the suture material as a template (
Figure 5 and
Figure 6). Next, with the tongue in an unstrained position, a dorsal flap with an anteriorly based pedicle was designed using the suture material as a template (
Figure 7). The width was dictated by the width of the defect plus 20%. The anteriorly based tongue flap was raised, including 2 to 3 mm of muscle thickness to allow for adequate vascularization (
Figure 8). After mobilization of the lingual flap, the donor site was sutured with 4–0 resorbable interrupted sutures (
Figure 9).
The tongue flap was then rotated forward and sutured to the raw edges of the palatal defect anteriorly and laterally using 4–0 Vicryl (
Figure 10 and
Figure 11). No nasogastric tube was placed to assist in feeding.
Following completion of fistula closure, the patient was assessed under the following criteria at 2 weeks, 1 month, 3 months, 6 months, and thereafter at 1-year intervals: (1) flap viability; (2) fistula closure; (3) residual tongue function and aesthetics; (4) assessment of speech impediment.
Length of follow-up period ranged from 2 weeks to 18 months, with an average length of 15 months.
Speech Assessment
A customized Performa for patients with palatal fistulas was used to document the findings of the speech pathologist. All 40 patients with palatal fistula were evaluated by a speech pathologist preoperatively and at 1, 3, and 6 months and 1 year postoperatively, using the following parameters: (1) articulation and speech intelligibility; (2) nasal emission; (3) hypernasality.
All patients were advised to perform palatal muscular strengthening exercises for 8 weeks starting at 5 weeks postoperatively, such as blowing (balloon, candle, etc.), sucking (using pipes and straws of varying length), and direct stimulation using finger or cotton ear buds. Patients were also given specific speech exercises as instructed by the speech pathologist to improve their articulation.
The degree of nasal emission, hypernasality, and speech intelligibility was recorded on a scale ranging from 0 to 3 (0, normal; 1, mild; 2, moderate; 3, severe). All assessments were done by single speech pathologist. The comparisons of the pre- and postoperative nasal emission, hypernasality, and speech intelligibility were done using the Wilcoxon matched pairs test.
Analysis of the Data
All the results of the study were subjected to statistical analysis.
Discussion
Primary treatment of cleft palate should result in an intact palate with separation of the oral and nasal cavities. However, the published reports of large series indicate fistula can recur in the secondary palate of a small but significant group of patients; the incidence varies from 8.9 to 34% [
2].
Even in the best of hands, an oronasal fistula of the secondary palate may occur postoperatively. A fistula may also be caused by trauma, tumor, irradiation, or a rare infectious disease, such as midline granuloma, syphilitic gumma, leprosy, noma, or leishmaniasis. Breakdown of the primary palatal repair is usually related to tension at the site of closure (often at the junction of the hard and soft palate); necrosis can occur if the greater palatine vessel is injured during elevation of the anterior tip of the push back flap [
2].
The severity of the original defect may also influence the incidence of fistula. Musgrave and Bremner reported a 4.6% incidence in the case of incomplete cleft palate, 7.7% in complete unilateral clefts, and 12.5% in complete bilateral clefts [
6]. In our study, we treated 40 patients with palatal fistula secondary to cleft palate surgery. We encountered 21 bilateral complete cleft lip, alveolus, and palate patients. This accounts for 52% of our study, and there were 11 unilateral complete cleft lip, alveolus, and palate patients (28%) and eight complete palate patients (20%). This is represented in the
Table 4 and
Figure 28.
Fistulas may occur in the labial vestibule, the alveolus, the hard palate, and at the junction between the hard and soft palate. Symptoms of these fistulas may be hypernasality in speech, regurgitation of fluids into the nose, and food lodging in the defect. The symptoms depend to some extent on the site of the fistula [
7]. In accordance with literature, most of the fistulas were located in anterior palate [
8]. In our study, The Pittsburgh Fistula Classification System was used to describe fistula [
5]. Most of the fistulas were seen at the junction of the primary and secondary palate (type V). Of 40 patients, 31 (77%) had fistula at the junction of primary and secondary palate, 3 (8%) had fistula in hard palate (type IV), and 6 (15%) had fistula at the junction of the soft and hard palate (type III), as shown in
Table 1 and
Figure 25.
Attempts at closure using only local transposition flaps may be successful, although frequently this is not achieved and a smaller oronasal fistula will recur. Additional attempts to gain closure with local tissue alone often result in repeated failure as thick and immobile scarred palatal mucoperiosteum leads to closure under tension with subsequent flap necrosis and wound dehiscence [
2].
A variety of both surgical and prosthetic solutions to the problem of inadequate local tissue have been sought. Tissue from distant sites has been used including tubed pedicle flaps from the abdomen, arm, neck, or cervicothoracic region. Others have used cheek and nasolabial flaps to close these palatal defects. Although these staged techniques of distal tissue transfer may be effective, they require multiple operations, are always cumbersome, leave multiple scars, prevent chewing until final flap division, are bulky when inset, and finally transfer skin rather than mucosa to the roof of the mouth [
2].
Free nonvascularized grafts, such as dermis or conchal cartilage, may prove useful, though such grafts are limited to defects less than 5 mm in diameter [
9].
Jackson published his work on 68 patients for closure of secondary palatal fistulas with intraoral tissue and bone grafting. For narrow defects, a vomer flap was raised and closed after arch expansion was done and bone grafting followed the procedure. For wider fistulas, he used tongue flaps. In his study, he used the Veau flap and the buccal flap but found that the tongue flap was excellent for wider defects [
10].
Gordon and Brown provided a brief review of flap techniques for closure of defects of the palate including the Fickling–Inkwell technique, double-layer island flap, double-layer hinged flap, and tongue flap; the authors advise that local flaps be chosen for smaller defects (enough to be covered with a rotated flap) when adjacent healthy tissue is available. However, a larger defect may require reinforcement with tongue flaps [
11].
The FAMM (facial artery musculomucosal) flap as introduced by Pribaz et al. is a valuable option to reconstruct moderate-size defects of the anterior palate [
12]. The FAMM flap has few minor drawbacks. First, the pedicle may be injured during mastication to the point that the pedicle gets severed. The patient must be aware that he or she will have to wear a bite block during the healing period. The incorporation of the flap for 3 weeks is a bothersome stage, and the mucosal paddle is somewhat bulky and requires resurfacing at a later dates. The whole facial artery can also be missing, as reported in well-documented studies, and its presence should be ascertained by a laser Doppler examination. The procedure requires careful surgical planning for optimal results [
13]. Furthermore, speech therapists discourage the use of a flap that, with the inclusion of facial muscles, will likely interfere with further speech development [
9].
Given the limitations of local options for coverage, free tissue transfer may offer an attractive solution. Although the radial forearm flap has been described as the free tissue workhorse of the orofacial reconstructive surgeon, along with the dorsalis pedis flap, free tissue transfer methods are associated with significant donor site morbidity and oftentimes poor cosmesis [
9].
More recently, Cole et al. used decellularized human dermal matrix for the repair of recurrent oronasal fistula. This study retrospectively analyzed five consecutive patients treated with interpositional AlloDerm (AlloDerm, Life-Cell Corporation, Branchburg, NJ) placement between nasal and oral mucosa for the repair of recurrent oronasal fistula; results were assessed, and the authors concluded that the adjunctive placement of intramucosal decellularized dermal graft is effective and reliable for use in the closure of recalcitrant oronasal fistulas [
9]. The main drawback of this study was small sample size and the average length of follow-up of only 7 months. The main disadvantage of AlloDerm is the associated cost [
14]. In this regard, further study is required to assess the efficacy of decellularized dermal matrix over the long term.
Tongue flaps have been used for the reconstruction of various sites including the lower lip, floor of mouth, buccal mucosa, and palate. However, in cleft palate surgery their excellent vascularity and the large amount of tissue they provide render tongue flaps particularly appropriate for the repair of large fistulas in palates scarred by previous surgery [
15,
16,
17].
Tongue flaps have been used to close intraoral defects following tumor surgery, severe infection [
18], trauma, and cleft palate fistulas. According to DeSanto, tongue flaps are also useful after radiation therapy [
19]. Posteriorly based flaps are indicated when treating defects of soft palate, retromolar region, floor of the mouth, and posterior buccal mucosa [
16,
20]. Anteriorly based flaps are useful in the treatment of defects of the hard palate, anterior buccal mucosa, lips, and anterior floor of the mouth [
8,
21].
Pigott et al. [
22] presented their work on 20 patients with palatal fistulas, which were successfully closed with tongue flaps in 17 patients (85%); six required an additional minor procedure, several patients no longer had to wear obturator, speech was improved in nine patients due to reduction in hypernasality, and in eight patients, articulation was noticeably improved. No patient underwent intermaxillary fixation after the procedure. In addition, the authors also give indications for surgical repair of palatal fistula and for use of tongue flap. According to Pigott et al., in attempted palatal fistula with scarred regions, tongue flaps can be successful. The anteriorly based tongue flap has proved to be a reliable way of closing the difficult fistula where symptoms are sufficient to justify the attempt.
Contreras et al. used tongue flap and forehead flap for closure of residual oronasal fistula, and they concluded that when all other surgical possibilities have failed, tongue, vestibular mucosa, pharynx, or forehead flap can be used to restore mucosal continuity [
23].
Gamoletti et al. studied the histological pattern of the reinnervation process of heterotopically transposed lingual flaps in the oral cavity. Two cases were reported: in the first, the tongue flap was used to repair the vermilion of the lower lip, and in the second, for the closure of a posttraumatic defect of the hard palate. The histological findings are similar in the two cases: myelinated and unmyelinated fibers, free nerve endings, and encapsulated receptors are present. The authors concluded that pedicled tongue flaps have proved to be an effective method of repairing defects due to tissue loss in the oral cavity [
24].
Kim et al. used tongue flaps in 16 cases; 13 patients were operated twice for closure of the fistulas [
25]. In our study, 30 patients (75%) were operated for cleft palate previously, seven patients (7%) were operated twice to close the fistulas primarily, and three patients (3%) were operated more than twice in an attempting to close the fistula as shown in
Table 2 and
Figure 26. In our study, fistula was present in anterior and midpalate; the size of the palatal fistulas was not amenable for local flap closure, and a history of repeated attempts to achieve closure of the palatal defect was considered.
Design of Tongue Flaps
The lingual artery is the main artery supplying the tongue. The dorsal lingual artery, a branch of the lingual artery, supplies the dorsum of the tongue, vallecula, epiglottis, tonsils, and adjacent soft palate. The ranine branch unites both dorsal lingual arteries at the tip and provides a rich plexus. Once the lingual artery reaches the anterior edge of the hyoglossus muscle, it divides into its terminal branch: the sublingual and the deep lingual arteries. The sublingual artery travels along the genioglossus and the sublingual gland and has an extensive anastomotic network with the contralateral sublingual artery. The deep lingual artery courses anteriorly, deep to the ventral mucosa. It gives off multiple branches that ascend toward the dorsum of the tongue [
26]. Cadenat et al. have described the rich submucous vascular plexus found in the tongue [
27]. This plexus allows for safe and predictable elevation of thin flaps.
Carlesso et al. described a design for a tongue flap that utilizes the full thickness of the hemi-mobile tongue, which provides mucosal lining, muscle bulk, and a long, supple, nonrestricting pedicle. The flap uses the entire length of the hemi-mobile tongue, based on the midline of the anterior tongue and including the mucosa of the dorsal and ventral surfaces and the bulk of lingual muscle. The mobility of the flap is such that it can be moved in a wide arc in and around the oral cavity, suggesting the applications can be increased to include tissue losses as the result of trauma or the treatment of neoplastic diseases [
28].
Busić et al. used anteriorly based dorsal tongue flaps in 19 cleft patients for closing large palatal defects. The procedure was successful in 17 patients. One patient had partial marginal necrosis after division of the pedicle, another had complete necrosis after division of the pedicle, and another had complete necrosis of the distal part of the flap; the authors concluded that the anteriorly based dorsal tongue flap is a safe and effective method for closure of relatively large palatal defects. The parameters for success include sufficient length of the flap (5 to 6 cm), a flap width some-what larger than the defect, and a flap thickness of ~0.5 cm [
29].
Assunçao presented his experience with thin (3-mm) tongue flaps used to close large anterior palatal fistulas. This technique was used successfully in 12 patients with fistula following surgery for cleft palate. One forked flap and one mushroom-shaped flap that were used to close irregularly shaped fistulas were described. All flaps survived, and there was a partial recurrence of one fistula in only one patient. The results of this series confirm that the thin tongue flap is a safe and reliable technique for the closure of large palatal fistula even when tailored to fit irregularly shaped defects [
30].
Defect size is an important factor. The quality of the local tissue may be unsuitable. Repeated attempts to achieve closure of the palatal defect by transposition of local flaps result in tissue scarring, ischemia, and mucosal irregularity. This triad predisposes to chronic inflammatory changes in the palatal tissues, often compounded by the traumatic and unhygienic insults of orthodontic and obturating appliances [
31].
Kim et al. encountered a maximum fistula size of 5 × 4 cm17; in our study, the maximum fistula size was 8 × 6 cm and minimum was 10 × 8 mm.
In our study, 38 patients (95%) had scar over the palate adjacent to fistula due to a previous surgery, and in two patients (5%) not much scarring was present, which is one of the indications for the use of tongue flaps [
31].
In our study, a dorsal flap with an anteriorly based pedicle was designed. The length of the flap was designed such that 1 to 2 cm of additional tissue would span the posterior edge of the palatal defect; the approximate size of the tongue flap was designed using cover of the suture material as template. The width was dictated by the width of the defect plus 20%. The flap should include 2 mm of muscle thickness to allow for adequate vascularization.
Fistula Closure
For all 40 patients, we used anteriorly based tongue flaps. None of the flaps failed over the long-term follow-up, which indicates its versatility. In the initial stage, complete closure of the fistulas was achieved in all 40 patients; however, three patients (8%) experienced recurrences of the fistula. Fistulas in hard palate (type IV) recurred. These patients were 23, 27, and 28 years of age, and these fistulas were located in hard palate. Secondary fistula closure was achieved by local advancement of the donated tongue tissue. All the patients in whom defect closure was successful experienced complete resolution of nasal regurgitation and reduction in social embarrassment with improvement in their psychological attitude, which is in accordance with previous studies by Coghlan et al. [
31].
Flap Viability
All flaps in 40 patients proved to be viable in the long term, although two flaps (5%) required resuturing, which showed satisfactory results over long-term follow-up.
Residual Tongue Function and Esthetics
In all cases postoperative aesthetics of the donor tongue site were found to be satisfactory. There was no interference with speech with the use of the tongue as a donor site. Oral hygiene and mastication were unimpaired. No patient described sensory or gustatory disability following this procedure.
Speech Assessment
Bradley and Stell treated eight patients who had carcinoma of the oral cavity with tongue flaps. Pre- and postoperative speech analysis (from 5 to 24 months after surgery) was done. None of the patients noticed speech problem postoperatively [
32].
For all 40 patients, preoperative speech analysis was done by speech pathologist, and degree of speech impediment was assessed, including intelligibility, hypernasality, and nasal emission. These results were compared with 1-month, 3-month, 6-month, and 1-year postoperative speech analysis results. All three parameters showed significant improvement over a period of 6 months and 1 year.
Speech Intelligibility
In our study, degree of severity was graded as normal (0), mild (1), moderate (2), or severe (3). Of 40 patients, 10 (25%) were grade 3, 24 (60%) were grade 2, and 6 (15%) were grade 1 (
Table 5).
Following surgery at 1 month, there was no significant improvement in speech intelligibility but at 3 months, there was significant improvement; grade 3 severity disappeared, and 34 patients (85%)showed moderate severity (grade 2) with Z value of 2.254 (
Table 6), which is statistically significant, and six patients (15%) had mild severity. At 6 months, severity showed further marked improvement. In five patients (12.5%), speech intelligibility reached normal level, and 34 patients (85%) had mild and only one patient (2.5%) had moderate score with the Z values of 7.217, which is statistically significant. At 1-year follow-up, there was drastic improvement in speech intelligibility: 16 patients (40%) reached normal level, and 24 patients (60%) had mild severity with Z values of 2.541, which is statistically significant. Improvement between pre- and postoperative regular follow-up interval is represented in
Table 5 and
Table 6 and
Figure 29.
Hypernasality
Degree of severity was graded as normal (0), mild (1), moderate (2), and severe (3). Of 40 patients, 9 (22.5%) had mild, 29 (72.5%) had moderate, and 2 (5.0%) had severe hypernasality preoperatively (
Table 7). By 1 month following surgery, there was no significant change in the degree of severity. At 3 months, 9 patients (22.5%) had normal, 29 (72.5%) had mild, and 2 (5.5%) patients had moderate hypernasality. At 6 months, 36 (90%) had reached normal level and only 4 (10%) patients had mild hypernasality. At 1 year, 37 patients (92.57%) had normal hypernasality and 3 (7.5%) had mild hypernasality. Improvement between pre- and postoperative levels is represented in
Table 7 and
Table 8 and
Figure 30.
Nasal Emission
Nasal emission was assessed preoperatively and graded as normal (0), mild (1), moderate (2), or severe (3). Of 40 patients, 25 (62.5%) had moderate nasal emission, 12 (30.2%) had mild nasal emission, and 3 (7.5%) had severe nasal emission. At 1 month following surgery, there was significant improvement: 3 (7.5%) had normal nasal emission, 31 (77.5%) had mild nasal emission, 5 (12.5%) had moderate nasal emission, and 1 (2.5%) had severe nasal emission. At 3 months, there was further improvement: 14 (35%) had normal nasal emission, 23 (57.5%) had mild nasal emission, and 3 (7.5%) had moderate nasal emission. At 6 months and 1 year, 34 (85%) had normal nasal emission and 6 (15.0%) had mild nasal emission, respectively, showing significant improvement (
Table 9 and
Table 10 and
Figure 31).