Descriptive study of management of palatal fistula in one hundred and ninety-four cleft individuals

Descriptive study of management of palatal fistula in one hundred and ninety-four cleft individuals

Jyotsna Murthy
Department of Plastic Surgery, Sri Ramachandra Medical College and Research Institute, Porur, Chennai, India

Correspondence Address:
Jyotsna Murthy
Department of Plastic Surgery, Sri Ramachandra Medical College and RI, Porur, Chennai-600 116, Tamil Nadu
India

DOI: 10.4103/0970-0358.81447

Objective: Palatal fistula is a significant complication following cleft palate repair. The guidelines of management of the palatal fistula is dependent on the type of cleft, site of fistula, condition of surrounding tissue and associated problem. We studied the management and outcome of 194 cleft palate fistula in our institute. Design: We present the descriptive hospital-based study of management of palatal fistula in 194 cleft patients. We have excluded all the syndromic children and children whose anterior palate was not operated as per protocol. Settings : Of 194 cleft palate fistula, 37 had palate repair in our hospital and 157 were refereed with fistula following palate repair.The patients were evaluated by interdisciplinary team and plan of management was decided. Result : Various parameters like types of cleft, site of fistula and management of fistula were studied in all the patients. Fifty-two percent were in unilateral CLP and 30% in bilateral CLP because unilateral CLP is the commonest type of cleft. Postalveolar and hard palate region contributing to 67% of all fistulae, followed by junctional in (9%). Seventy-two percent of fistula were amenable for repair by local available tissue, 28% needed tongue flap due to shortage of tissue. Minor numbers have failure of procedure for fistula closure needing further management. Conclusions : This descriptive study present analysis of management of fistula in our institute. It also reinforces that patient with bilateral cleft lip and palate more likely to have shortage of local tissue needing the local flaps like tongue flap compare to other cleft types. The surgical management of fistula can be combined to tackle the associated problems.

Keywords: Palatal fistula; tongue flap; post alveolar fistula

How to cite this article:
Murthy J. Descriptive study of management of palatal fistula in one hundred and ninety-four cleft individuals. Indian J Plast Surg 2011;44:41-6
How to cite this URL:
Murthy J. Descriptive study of management of palatal fistula in one hundred and ninety-four cleft individuals. Indian J Plast Surg [serial online] 2011 [cited 2011 May 23];44:41-6. Available from: http://www.ijps.org/text.asp?2011/44/1/41/81447

One of the expected outcomes of palate repair is to achieve complete partition between nasal and oral cavity in addition to good speech. Any failure of achieving complete structural integrity of palate is labeled as an oronasal (palatal) fistula with persistent passage between oral and nasal cavity. The term, palatal fistula, is normally used for residual non-repaired cleft palate or result of breakdown of repaired palate. [1]

Every visible palatal fistula does not need surgical repair. The indications for a fistula repair depend on the associated symptoms, which are in turn related to the size and location of the fistula. The common symptoms which have been extensively discussed in the literature are: 1. Nasal emission causing speech distortions, 2. Leakage of fluid and food into the nasal cavity leading to poor oral hygiene and foul smell. The timing of the fistula repair depends on symptoms like nasal regurgitation, effect on speech and concerned of the patient. It is generally agreed that fistulae causing disturbances in speech should be repaired as soon as possible. A fistula leading to significant regurgitation of fluid/food into the nose, resulting in inflammation of nasal lining and malodor also needs to be repaired early. However, the repair of a small fistula with no effect on speech and occasional regurgitation of fluid/food into nose, can be delayed to be combined with other procedures. Many attempts have been made to classify palatal fistulae according to the site and is the best presented by Cohen et al. [2] Smith et al, presented modified classification and presented fistula in VII categories according to its site with more details. [3]

The speech evaluation by the speech pathologist is essential to diagnose if the fistula is contributing to the speech problems. The speech pathologist also provides information regarding the associated Velo-pharyngeal incompetence (VPI) which may or may not be due to the fistula after closing the fistula with chewing gum temporarily. [4] Similarly, evaluation and suggestions by an orthodontist are essential for a perialveolar fistula. In certain cases, orthodontic treatment e.g., expansion of arches and removal of deciduous or supernumerary teeth may be necessary. This additional information is vital for combining fistula closure with another procedure like VPI correction, alveolar bone graft or lip revision. The factors which need to be studied and examined before fistula repair are presented in [Table 1]. The concerns and complaints of the patients need to be elicited in detail and to be taken into consideration while planning the management.

We analyzed the management of palatal fistula in 194 fistulae in our institute since 2002. Of total 194 fistulae, 37 patients had palate repair in our institute, 157 patients were referred to our institute for fistula repair. Of referral patients, 80% were either detected during camps in rural area or referred by the health workers from rural areas and 20% approached directly to our center for help. Our data were collected as alveolar (which also includes prealveolar and postalveolar), hard palate, combined postalveolar extending to hard palate, junction, soft palate and combination of hard and soft palate fistula. In view of this being a retrograde study, we presented the data according to these sites mentioned above. A small uvular notching or bifid uvula was not considered as fistula, but uvular breakdown was considered as soft palate fistula because majority of them will need repair of soft palate. Of 2387 palate repairs in our institute, 2067 patients were followed up at least once postoperatively within 1 year and 75 had fistula. Out of 75 patients, we have excluded 38 patients with small, non-symptomatic postalveolar defect which was not repaired as the protocol (no anterior palate repair with lip repair) in our institute between 2001 and 2003. However, the significant postalveolar fistulae in these patients which needed closure were considered as a fistula and included in the study. This protocol was changed thereafter due to higher percentage of postalveolar fistula needing closure. Hence, 37 patients had cleft palate repair in our institute, needing fistula repair were included in this study. We also excluded syndromic cleft lip and palate patients having associated with cardiac anomalies, ectodermal dysplasia and Pierre Robin sequence with compromised airway. Eight patients with total breakdown of palate with severe shortage of tissue, who needed the simultaneous tongue flap and pharyngeal flaps, [5] were excluded from this study.

As discussed above, the decision for surgical repair of palatal fistula was done after proper evaluation [Table 1]. The previous surgical techniques, scarring and shortage of tissue, inflammation of tissue and oral hygiene, availability of local tissue and concomitant planned procedures were considered to decide the timing and the surgical procedure for the fistula repair. For fistula in hard palate or junctional area, if adequate local tissue was available, it was closed by the mucoperiosteal flaps with releasing incisions like in Von Langenbeck palatoplasty. [6] In large fistulae and/or fistula extending in postalveolar and alveolar region were closed by two-flap technique and preferably with alveolar extended palatoplasty. [7],[8] Alveolar extension palatoplasty (AEP) flaps were extremely useful [Figure 1]a for fistula in postalveolar region. The AEP flaps could be raised cautiously even in the presence of the previous scars between mucoperiosteum flaps and its extension into alveolus [Figure 1]b. However, an interval of 6 months or more between the palate repair and the fistula repair by AEP flaps is necessary. Figure 1a: Incision for alveolar extension palatoplasty
Figure 1b: Fistula repair by alveolar extended palatoplasty

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For the large fistulae with a shortage of tissue and/or severe scarring preventing mobilization of the palatine tissue, an additional tissue was imported as local flaps according to the site of fistula. For postalveolar or anterior hard palate fistulae, where additional tissue was needed, an anteriorly based tongue flap were done [9] [Figure 2]. The tongue flaps provided oral lining, while the nasal lining was repaired with turn-over flaps from the fistula edges (hinge flaps) and this step was of paramount importance to prevent recurrent fistulae. The tongue flaps were routinely divided between 10 and 12 days, postoperatively.

Patients needing VPI correction and/or with a scarred/broken soft palate with fistula had nasoendoscopy for the structural evaluation. In severely scarred and broken soft palate with large nasopharyngeal defect on endoscopy, a superiorly based pharyngeal flap was used to add tissue for the nasal lining for the soft palate which also helps to improve velopharyngeal competence.

In a situation with failure of multiple attempts of fistula repair, refusal for surgery by patients and associated demands for a tooth prosthesis, prosthetic cover for a fistula was used.

We have analyzed the type of cleft, location of fistulae and type of surgeries performed in all the patients.

We studied distribution of fistula in various types of cleft lip and/or palate as shown in [Table 2]. In our institute, we had comparatively higher fistula occurrence in submucous cleft (6% of total 37 patients). In retrospection, it was noticed that sutures were put in thinner hypoplastic mucoperiosteum, which should have been excised.

The distribution of site of fistula in all 194 palatal fistulae showed most common site is hard palate and in postalveolar region contributing to 67% of all fistulae. This was followed by exclusive postalveolar region (9%) and junctional area [Table 3]. Table 3: Site of fistulae, procedures for fistula repair and adjuvant procedures

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As the majority of the fistula were repaired by different procedure depending on the size of fistula, the availability of local tissue and necessity of adjuvant procedures. The procedures like local flap, Von Lagenback repair, two flap, AEP, palate repair and tongue flap were done to close the fistula. In 65 patients fistula repair was combined with other procedure like VPI correction, ABG and lip revision [Table 3].

Out of total 194 patients had fistula repair, 48% had followed up and 10 patients had residual fistula. All these patients had very poor local condition and oral hygiene. Two of these were operated by AEP flap closure for postalveolar fistula and eight had tongue flap. Of two with AEP flap, one had successful closure with revision surgery while one had prosthetic rehabilitation. Eight out of 54 patients with tongue flap had postoperative problems. Five patients had flap detachment in postoperatively period. Four out of these five patients were resutured in the first 10 days, and two had complete closure while two had persistent fistula. One patient has revision tongue flap after duration of 8 months successfully. Remaining three patients had residual fistula at the posterior border of tongue flap observed during follow-up.

Although palatal fistula is a common morbidity after cleft palate repair, minimal literature is available regarding the guidelines of management. Palatal fistula is a complication of cleft palate repair observed in different studies ranging from 0 to 34%. [2] We have analysed the descriptive data of type of cleft, site of fistula and management of fistula in our institute. From this data, we learned that smaller fistula are tend to be managed in the same hospital, while the larger fistula are more likely to go to specialized center or tertiary center due to difficulty in management and needing multidisciplinary care. The larger fistulae in difficult site like perialveolar region tend to be neglected and need interdisciplinary care in tertiary centers.

Management of cleft palate fistula is multidisciplinary approach and proper evaluation of speech and dental arch in addition to the local tissues are very essential. The functional aspect of the fistula should be given appropriate attention before a decision is reached on its structural surgical repair. After proper evaluation and decision regarding surgical repair, one needs to pay attention to the local tissue condition. The previous surgical techniques, scarring and shortage of tissue, inflammation of tissue and oral hygiene, availability of local tissue and concomitant planned procedures will decide the type of procedure for the fistula repair [Figure 3]. This approach will help to decide the proper timing and appropriate technique for surgical repair. Tongue flap has been a work horse for difficult palatal fistula with shortage of tissue. Similarly, buccal flaps also known as facial mayo-mucosal flap, is the most appropriate for junction fistulae if there if shortage of tissue. We have used the buccal flap for VPI correction but not for fistula repair. However, if utilized in junctional fistula, this will also help to lengthen the palate for VPI correction at the same time. [10],[11] Other flaps which has been described and utilized very rarely in specific indication are, temporalis muscle flap and free microvascular free flaps. [12],[13] However, the later options are more commonly utilized in closing non-cleft palatal reconstruction.

In certain situation like failure of multiple attempts of fistula repair, refusal for surgery by patients and associated demands for tooth prosthesis, the prosthetic cover for fistula can be applied. However, prosthesis can never provide nature barrier like tissue repair and has implication on oral hygiene and dental health.

 
Veau V. Division Palatine. Paris, Masson et Cie, Paris: 1931.  
Cohen SR, Kalinowski J, LaRossa D, Randall P. Cleft palate fistulas: A multivariate statistical analysis of prevalence, etiology, and surgical management. Plast Reconstr Surg 1991;87:1041-7.  
[PUBMED]    Smith DM, Vecchione L, Jang S, Ford M, Deleyiannis FW, Haralam MA, et al. The Pittsburgh fistula classification system: A standardized scheme for the e description of palatal fistulas. Cleft Palate Craniofac J 2007;44:590-4.  
Nanda V, Sharma R, Mehrotra S, Makkar SS, Munjal S. The "chewing gum test" for cleft palate speech. Plast Reconstr Surg 2005;116:18-22.  
Murthy J. Salvaging procedure for mutilated cleft palate by simultaneous tongue and pharyngeal flap surgery. Plast Reconstr Surg 2008;122:29e-30e.  
[PUBMED]  [FULLTEXT]  Muzaffar AR, Byrd HS, Rohrich RJ, Johns DF, LeBlanc D, Beran SJ, et al. Papaioannoua: incidence of cleft palate fistula: An institutional experience with two-stage palatal repair. Plast Reconstr Surg 2001;10:1515-8.  
Denny AD, Amm CA. Surgical technique for the correction of postpalatoplasty fistulae of the hard palate. Plast Reconstr Surg 2005;115:383-7.  
[PUBMED]  [FULLTEXT]  Carsten MH. Sequential cleft management with the sliding sulcus technique and alveolar extension palatoplasty. J Craniofac Surg 1999;10:503-18.   
Jackson IT. Use of tongue flap to resurface lip defect and close palatal fistulae in children. Plast Reconstr Surg 1972;49:537-41.   
[PUBMED]    Abdel-Aziz M. The use of buccal flap in the closure of posterior post-palatoplasty fistula. Int J Pediatr Otorhinolaryngol 2008;72:1657-61.   
[PUBMED]  [FULLTEXT]  Ashtiani AK, Emami SA, Rasti M. Closure of complicated palatal fistula with facial artery musculomucosal flap. Plast Reconstr Surg 2005;116:381-6; discussion 387-8.  
[PUBMED]  [FULLTEXT]  Krimmel M, Hoffmann J, Reinert S. Cleft palate fistula closure with a mucosal prelaminated lateral upper arm flap. Plast Reconstr Surg 2005;116:1870-2.  
[PUBMED]  [FULLTEXT]  Schwabegger AH, Hubli E, Rieger M, Gassner R, Schmidt A, Ninkovic M. Role of free-tissue transfer in the treatment of recalcitrant palatal fistulae among patients with cleft palates. Plast Reconstr Surg 2004;113:1131-9.  
[PUBMED]  [FULLTEXT]  

[Figure 1], [Figure 2], [Figure 3]

[Table 1], [Table 2], [Table 3]

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Management of palatal fistulae

Management of palatal fistulae

Michael H Carstens
Associate Professor of Plastic Surgery, Saint Louis University, Missouri, USA

Correspondence Address:
Michael H Carstens
Department of Plastic Surgery, Saint Louis University, Missouri
USA

DOI: 10.4103/0970-0358.81445

How to cite this article:
Carstens MH. Management of palatal fistulae. Indian J Plast Surg 2011;44:46-9

This retrospective analysis presented by the Sri Ramachandra craniofacial team raises several important issues for all cleft surgeons. First, palatal fistulae continue to be a common management problem. Second, the classification systems currently applied to cleft lip and palate are empiric, archaic, and misleading. Third, without a classification system based on embryologic mechanism, accurate appreciation of outcomes data generated by cleft palate (CP) surgery (i.e., surgical complications and speech) will remain chaotic. Fourth, the distribution pattern of fistulae is directly related to the technical problems engendered by the primary surgery. Fifth, the low incidence of fistulae reported in the Sri Ramchandra Medical College (SRMC) patients vs. those referred from other location points out the importance of surgical protocol and concentration of expertise. Sixth, the best treatment for palate fistula is prevention using dissection techniques based on developmental anatomy of the cleft and executed with precision. Seventh, established fistulae require the same careful embryologic analysis; closure unusually involves a reapplication of techniques that should have been employed at the primary operation.

Fistula rates reported in the literature range as high as 58% with a recurrence rate of nearly 33%. [1] These by definition result from a prior surgical procedure. Does particular operation predispose to fistula formation? The problem is complicated by a worldwide lack of consensus regarding a treatment protocol. Surgical management has been studied from the standpoint of timing (early vs. late) [2],[3] and staging [4],[5] but not by embryologic mechanism. In short, raw data regarding what factors are associated with a fistula is difficult to interpret.

The problem comes from our inability to stratify clefts by embryologic mechanism. A CP resulting from a small vomer is different from one involving a deficient palatine bone. Soft palate clefts involving the tensor alone can have perfectly good constrictor function. A submucous cleft with global involvement of third arch muscles will have a radically different prognosis. In short, continuing to compare apples with oranges makes outcome analysis unreliable.

CPs come in different flavours. Anatomic defects seen in CP involve distinct developmental fields, each of which contains a variety of tissues: epithelium, dermis and submucosa, cartilage, bone, fascia, and muscle. Regardless of content, the tissues in each developmental field share a common neurovascular supply. Knowledge of the nerve supply to any given field allows one to determine precisely where it originates in the embryo. [6],[7]

Two types of mesenchymal structures contribute to development of the hard and soft palate: neural crest bone fields and paraxial mesoderm muscle fields. These, in turn, can be related to the developmental sector of the CNS that supplies innervation. These sectors are known as neuromeres and their individual nomenclature comes from the names applied to the original three-part embryonic brain.

All components of the hard palate, both hard and soft, are supplied by nerves having their nuclei within the hindbrain or rhombencephalon. The bones and muscles of the palate are developmentally linked to 12 rhombomeres (r0-r11). For example, all bones of the palate are innervated by V2, the nucleus of which resides in the second rhombomere. Thus, neural crest cells destined to become premaxilla, vomer, inferior turbinate, prepalatine, palatine, and maxilla all rise in the neural fold directly above r2. Clefts involving the hard palate result when deficits in one or more of these populations create palatal bone fields that are malformed, small, or downright absent.

In a similar way, all striated craniofacial muscles develop from paraxial mesoderm (PAM) lie down along both sides of the CNS. PAM begins with the hindbrain at the level of r0 (the future isthmus and pituitary) and extends backward all the way to the tail. Just as the neuraxis is segmented into neuromeres, PAM is also segmented into hollow balls called somitomeres. Sm1-Sm7 give rise to the extraocular muscles and the muscles of pharyngeal arches 1-3. Since these somitomere are incompletely separated, a potential confluence of the mesenchyme exists. This explains why the muscles of the second arch migrate widely over (and under) those of the first arch.

Beginning with the eighth somitomere, the mesoderm undergoes a further rearrangement. The result is that Sm8 becomes the first somite. All somites are surrounded by epithelium and contain subunits: bone-forming sclerotomes, dermis-forming dermatomes, and muscle-forming myotomes. The muscle content of all mammalian somitomeres and somites has been mapped out. Thus, Sm4 provides tensor veli palatini. All remaining soft palate muscles (including superior constrictor) arise from Sm7. Clefts of the soft palate result when deficits in one or more of these mesodermal populations create muscle fields that are malformed/mal-innervated, small, absent, or downright absent.

Fistulas are man-made creations. They result from inadequate surgical dissection of soft tissues from the margins of the cleft. Two anatomic sites are typically involved. Trouble is most often seen along the primary palate, with overlap into the adjacent secondary palate. In the SRMC series, anterior hard palate fistulae were encountered in 77.5% cases. In most cases, this results from failure to achieve control of the anterior nasal floor. Primary surgery, be it for the cleft palate or for an associated cleft lip, is the "golden opportunity" to access the anterior palate. After the primary repair (and with further growth) approximation of the palate margins makes dissection of the nasal layer more difficult.

The other common site of fistula formation is the posterior margin of the hard palate. Here, once again, the problem is one of the inadequate mobilization. The horizontal plate of the palate bone, normally having a rectangular shape, is often attenuated into a triangle. The mucoperiosteum of the nasal floor is tractioned forward. Confluence of soft tissues between the vomer and the hard palate presents difficulties as well. The stakes get higher when a Furlow Z-plasty is used for a primary palate repair. Clefts of moderate to severe width do not lend themselves well to this technique. Tissue loss and breakdown at the junction pose challenges for the most experienced operator.

SRMC reports one-year follow-up in 2067 patients with only 37 fistulae (1.9%). This number is no accident. First and foremost, reduction of fistulae by SRMC team is designed with extensive experience and careful dissection. However, it must be stated that any cleft surgeon can achieve similar results, provided that she/he slows down and targets dissection of the anterior and posterior palate margins to achieve a tension-free, watertight closure.[Table 1], [Table 2]

With these results as a benchmark, what steps can be taken to prevent fistula formation? Recalling the sporting dictum "the best defense is a good offense," the goal should be to seal up the nasal lining of the anterior palate. This is done by dissecting out, and uniting, two adjacent neuroangiosomes: medial sphenopalatine and lateral sphenopalatine. This concept lies at the heart of DFR (developmental field reassignment) cleft surgery. [8],[9]

The Medial Sphenopalatine artery (SPA M ) flap consists of mucoperiosteum of the vomer in continuity with that of the posterolateral "shoulder" of the premaxilla. These two components are, in turn, continuous with the soft tissues of the non-philtral prolabium (NPP). Lateral Sphenopalatine artery flap SPA L is the mucoperiosteal lining of the nasal hard palate. After SPA M and SPAM are united, NPP is folded sideways 90 degrees and sutured anteriorly, thus lengthening the nasal floor. The NPP flap contains mesenchyme originally designated for the premaxilla, specifically the lateral incisor zone and frontal process zones.

Elevating the mucoperiosteum from the nasal aspect of the palate is like putting the bell on the tiger; it sounds easy in theory but proves difficult in execution. Intraoral access gets trickier the more anterior one proceeds. Extraoral access proves a useful alternative. An incision in the lateral nasal wall just anterior to the inferior turbinate (and behind the nostril sill) accomplishes two goals: (1) external rotation of the nostril sill and (2) direct access to the piriform rim and the palatal shelf. One first slides an amalgam packer or angled elevator posteriorly just beneath the turbinate anterior and proceeds posteriorly all the way to the soft palate. Next, one takes the dissection medially elevating the nasal mucoperiosteum from lateral to medial. Finally, one incises along the cleft margin to elevate the flap.

Note that the above manoeuvres can be readily accomplished during initial surgery in three scenarios: (1) complete closure of the CP (with cleft lip repair performed secondarily; (2) as part of a lip adhesion procedure for a wide alveolar cleft; and (3) during DFR (developmental field reassignment) cleft lip repair.

Turning our attention to the posterior margin of the hard palate, let us see how the nasal lining can be mobilized. Achieving a complete release may require subperiosteal elevation of lateral nasopharyngeal mucosa. This is accomplished by carefully freeing the nasal lining from the posterior margin of the palatine bone. One then proceeds laterally until encountering the medial pterygoid plate. Here the subperiosteal elevation follows the pterygoid cephalad toward the cranial base. If required, the release can be taken up to the nasal roof. The oral tissues are often tethered with the greater palatine neurovascular pedicle. Under direct vision, the pedicle can be readily isolated. If required, osteotomy of the foramen with a 2-mm osteotome is fast and straightforward. The cuts are U-shaped and directed medially, taking care that the osteotome does not slip downward into the canal.

Regarding secondary fistula closure, we shall proceed from posterior to anterior. A posterior fistula will require release of nasal mucoperiosteum as discussed above. Should it occur in the context of a failed Furlow repair, one may need import of posteriorly based buccinators flaps to fill the deficit. Fistulae in the midportion of the hard palate require isolation of nasal lining, turning it inward carefully. Advancement of mucoperiosteal for oral closure is assisted by an Alveolar Extension Palatoplasty (AEP) -type gingival release. The extra tissue harvested from the lingual margins makes AEP flaps wider that those produced by Von Langenbeck technique. [10] When the fistula is anterior, AEP flaps need to be taken anteriorly, all the way to the cleft margin. Since the AEP flaps can be so readily mobilized, one should "cheat" when incising along the cleft margin. The incision can be designed to favour the oral side of the cleft by several millimetres, thus providing extra tissue to turn into the floor of the nose.

Finally, it is worth noting that the preponderance of patients in this paper presents as outside referrals (cleft camps, etc.). These patients are more likely to have recalcitrant fistulae. In many instances, their previous surgical management is unknown. In like manner, the fistula rate for this subset of patients is unknown. Those patients collected from within the SRMC system are a much smaller fraction of the total clefts performed. Their surgical treatment follows a known protocol. It would be interesting to separate out the distribution of clefts between the two groups to see if the fistulae seen in the SRMC versus those of the referral patients follow different patterns.

In summation, this reviewer would like to congratulate the SRMC team for a careful analysis of palatal fistulae. The two groups represent very different surgical approaches (eclectic vs. standardized). Further study of this experience will undoubtedly prove rewarding. This commentary is designed to highlight common anatomic pitfalls in CP repair and how these can contribute to a fistula. Surgical techniques based upon an understanding of developmental anatomy are valuable to prevent fistula formation and to reconstruct fistulae when these are encountered.

 
Landheer JA, Breugem CC, van der Molen M. Fistula incidence and predictors of fistula occurrence after cleft palate repair: Two-stage closure versus one-stage closure. Cleft Palate Craniofac J 2010;47:623-30.  
Lehner B, Wiltfang J, Strobel-Schwarthoff K, Benz M, Hirschfelder U, Neukam FW. Influence of early hard palate closure in unilateral and bilateral cleft lip and palate on maxillary transverse growth during the first four years of life. Cleft Palate Craniofac J 2003;40:126-30.  
[PUBMED]  [FULLTEXT]  Friede H, Enemark H. Long-term evidence for favorable midfacial growth after delated hard palate repair in UCLP patients. Cleft Palate Craniofac J 2001;38:323-9.  
[PUBMED]  [FULLTEXT]  Nollett PJ, Katsaros C, van't Hof MA, Semb G, Shaw WC, Kuijper-Jagtman AM. Cephalometric evaluation of long-term craniofacial development in unilateral cleft lip and palate patients treated with delayed hard palate closure. Int J Oral Maxillofac Surg 2008;37:123-30.  
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Carstens MH. Neural tube programming and the pathogenesis of craniofacial clefts, part I: The neuromeric organization of the head and neck. Handb Clin Neurol 2007;87:247-76.  
Carstens MH. Neural tube programming and the pathogenesis of craniofacial clefts, part II: Mesenchyme, pharyngeal arches, developmental fields, and the assembly of the human face. Hanb Clin Neurol 2007;87:277-339.  
Neuroembryology and functional anatomy of Cranio-facial Clefts 2009;42; 19-34.   
Carstens MH. Developmental field reassignment in unilateral cleft lip: Reconstruction of the premaxilla. In: Losee JE, Kirschner RE, editors. Comprehensive Cleft Care. New York: McGraw Hill; 2009. p. 241-66.   
Carstens MH. Sequential cleft management with the sliding sulcus technique and alveolar extension palatoplasty. J Craniofac Surg 1999;10:503-18.  
[PUBMED]    

[Table 1], [Table 2]

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Management of urethrocutaneous fistula after hypospadias surgery - An experience of thirty-five cases

Management of urethrocutaneous fistula after hypospadias surgery - An experience of thirty-five cases

Rajat Kumar Srivastava, Mangesh S Tandale, Nikhil Panse, Anubhav Gupta, Pawan Sahane
Consultant Plastic Surgeon, Saraswati Hospital & Research Centre, Lucknow, India

Correspondence Address:
Rajat Kumar Srivastava
C-300, Niralanagar, Lucknow
India

DOI: 10.4103/0970-0358.81456

Introduction: The commonest complication following hypospadias repair is occurrence of urethrocutaneous fistula. The smaller fistulas (<2 mm) are easier to close with a simple closure whereas larger ones (>2 mm) with good vascular surrounding skin require a local skin flap closure for avoiding overlapping suture lines. For the recurrent/larger fistulas with impaired local surrounding skin - incidence of recurrence is significantly reduced by providing a waterproofing interposition layer. Aims : To study the effect of size, location, number of fistulas and surrounding tissues in selecting the procedure and its outcome. To identify various factors involved in the recurrence and to formulate a management in the cases where recurrence has occurred. Patients and Methods: This study of 35 cases of urethrocutaneous fistula repair was done from July 2006 to May 2009 to achieve better results in fistula management following hypospadias surgery. Statistical analysis used: ?2 test and Fisher's exact test. Results : The overall success rate for fistula repair at first attempt was 89% with success rates for simple closure, layered closure and closure with waterproofing layer being 77%,89% and 100%, respectively. The second attempt success rate at fistula repair for simple closure and closure with waterproofing layer were 33% and 100%, respectively. At third attempt the two recurrent fistulas were managed by simple closure with a waterproofing interposition layer with no recurrence. All the waterproofing procedures in this study had a success rate of 100%. Conclusions: The treatment plan for a fistula must be individualized based on variables which has an effect on the outcome of repair and to an extent dictates the type of repair to be performed. The significantly improved success rates with the addition of a waterproofing layer suggests the use of this interposition layer should be done at the earliest available opportunity to prevent a reccurence rather than to reserve it for future options.

Keywords: Hypospadias; tunicavaginalis; urethrocutaneous fistula; waterproofing layer

How to cite this article:
Srivastava RK, Tandale MS, Panse N, Gupta A, Sahane P. Management of urethrocutaneous fistula after hypospadias surgery - An experience of thirty-five cases. Indian J Plast Surg 2011;44:98-103
How to cite this URL:
Srivastava RK, Tandale MS, Panse N, Gupta A, Sahane P. Management of urethrocutaneous fistula after hypospadias surgery - An experience of thirty-five cases. Indian J Plast Surg [serial online] 2011 [cited 2011 May 23];44:98-103. Available from: http://www.ijps.org/text.asp?2011/44/1/98/81456

Urethrocutaneous fistula formation is the commonest complication of hypospadias repair, with a reported incidence of 4-25% [1] . The successful repair of this lesion depends on several basic principles. Various techniques have been described for fistula repair but with disappointing results. Simple closure although, technically easy [2],[3] bears the potential risk of overlying suture lines and recurrence rates. Different procedures [1],[7] have been tried for repair of larger/multiple fistulas provided the local surrounding skin is vascularized and pliable. For larger/recurrent fistulas with impaired local vascularity an interposition waterproofing layer significantly reduces the recurrence rate of the fistulas [8],[10] .

We have operated on a total of 35 patients which underwent 41 procedures for repair of 60 urethrocutaneous fistulas following hypospadias surgery. The age at fistula repair ranged between 3 and18 years (mean age 7 years). Urethral calibration was routinely done intraoperatively with a urethral sound to exclude any distal stenosis, thereafter presence, location, number of fistulas was assessed, probing every pit in the skin with the probe to avoid missing smaller fistulae under loupe magnification. In doubtful cases methylene blue was injected under pressure from the terminal portion of neourethra while a tourniquet was applied at the base of the penis to occlude the proximal urethra. The fistulas were measured with calipers in the antero-posterior length of the penis although they were ovoid in shape [Figure 1]. A catheter of suitable size was inserted into the urethra and the fistulous tract excised by circumferential incision around the fistula. If the fistulas were located adjacent to each other they were joined into a single larger fistula and then repaired. The number, size of fistulas, status of surrounding skin [Figure 2] and suture material used in repair are shown in [Table 1]. The location of various fistulas is mentioned in [Table 2]. Smaller fistulas were repaired using simple closure technique with interrupted inverting suture line with 6-0 chromic catgut or vicryl. The subcutaneous tissue flaps were closed with 5-0 chromic catgut, respectively. For a larger fistula with good surrounding skin following simple closure of fistula site, the skin was closed by a layered closure (pants over vest repair) whereas the larger multiple/recurrent fistulas with scarred surrounding skin - an additional local/distant waterproofing flap procedure was incorporated between the fistula and skin layer. Urinary diversion in the form of perurethral catheter was done in cases considering the merits of the fistula; however, it was not considered mandatory in all cases. Table 1: Showing association of various variables with repeat fistula rates

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In this study the majority of fistulas occurred on the day of cathether removal (83%),followed by fistula appearance within 3 days (11%) and 4-7 days (6%), respectively. The majority of patients had no evident cause of fistulation (74%) probably indicating an error in technique of repair with inadequate inversion of mucosa, inadequate layers of closure, ischaemic tissue or overlapping suture line leading to a suture line leak. The other identifiable causes were meatal stenosis (9%), urethral stricture (10%) and suture line dehiscence (7%). On analyzing the effects of different variables on successful outcome of fistula repair it is clear from [Table 1] that number, size, status of surrounding skin, suture material used have a significant effect on the favourable outcome. On applying Fischers exact test for association of these variables on the outcome -P<.05 which was significant. The overall success rate of fistula repair at first attempt was 89% with success rates for simple closure, layered closure and closure with waterproofing layer being 77, 89 and 100%, respectively - which is comparable with other studies [4],[5],[8] [Table 3]. The second attempt success rate of fistula repair with simple closure was 33% which significantly improved to 100% when it was combined with an additional waterproofing interposition layer [Table 3]. At third attempt the two recurrent fistulas of simple closure were managed by simple closure along with waterproofing with tunica vaginalis interposition layer with no recurrences [Table 3]. Most of the recurrences occurred where vicryl was used (success rate 62%) as compared to chromic catgut (success rate 96%) [Table 1] [11] . The various waterproofing procedures used in this study are listed in [Table 4]. The majority of waterproofing procedures performed were distant flaps [Figure 3]a-d owing to the limited availability of unscarred local tissues. Tunica vaginalis as local flap was used in two cases of penoscrotal fistula while penile dartos and scrotal dartos [Figure 4]a-d were used in distal and proximal level fistulas, respectively. Distant flaps (Tunica vaginalis) were used for all varieties of fistula ranging from coronal to penoscrotal levels [Figure 5]. All these waterproofing procedures had a success rate of 100% in our study which is comparable to other studies [4],[8],[9] . The majority of complications in our study were skin necrosis, repeat fistula and meatal stenosis [Table 5]. Apart from managing repeat fistulas, two of the six patients with skin necrosis had superficial necrosis which healed spontaneously while three cases had an additional waterproofing layer providing a barrier layer thus preventing a repeat fistula and so were managed conservatively while one with an additional waterproofing layer required refreshening and resuturing of tunica vaginalis flap along with redraping with circumferential penile skin. The two patients of meatal stenosis were managed by serial dilatations and were reassessed at frequent intervals to look for development of meatal stenosis. Thus early regular follow-up following the repair should be done to look for impending distal obstruction and timely intervention to prevent recurrence of the fistula. Figure 3a: Showing preoperative micturition
Figure 3b: Showing intraop tunica vaginalis flap elevation
Figure 3c: Showing tunica vaginalis flap suturing
Figure 3d: Showing postop micturition

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Figure 4a: Showing preop proximal penile fistula
Figure 4b: Showing scrotal dartos elevation
Figure 4c: Showing scrotal dartos elevation
Figure 4d: Showing post-op micturition

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The incidence of fistula can be used to judge the success of hypospadias surgery ranging from 0 to 23% [5],[8],[10],[12] which in our study is 21% being comparable to other studies.

The cause of fistula remains less known although it is likely that local infection, local ischaemia, inadequate procedure, poor tissue handling, distal obstruction due to distal stenosis or encrustration with severity of hypospadias has significant impact on the outcome of the primary hypospadias repair. On studying the effects and association of variables like size, location, number of fistulas, amount and status of available local penile skin, suture material used on the outcome of the repair-the P-value was <0.05 which was significant (Applying Fischer`s exact test). However, recurrence did not relate to other variables.

The results underline that both simple closure and layered closure of a fistula at first attempt have a comparatively lower success rates [1],[4],[5],[12] .

Why some fistulas recur is uncertain. Beyond any deficiencies of surgical technique or postoperative management there is no clear answer, other than that impaired local vascularity-scarred surrounding skin [Figure 2] might be the plausible explaination.

The success rates at second and third attempts were appreciably lower in which simple closure [4],[5],[10] was attempted alone ranging from 50% to 80% whereas no recurrence was seen in which simple closure with additional waterproofing layer was incorporated. This further proves our point that repair with the same procedure in a locally scarred fistula leads to increased chances of recurrence and any of the waterproofing procedures should be combined to prevent further recurrence.

Shankar et al. [10] in his study of 10 cases of refistulas at second attempt found 50% success rates at third, fourth and fifth attempts but without any waterproofing layer. He limited the use of Tunica vaginalis as a waterproofing layer to third or subsequent repairs. Thus it is clear that with subsequent attempts at fistula repair, the chances of recurrence increases with decrease in success rates owing to the further scarring of the already deficient compromised surrounding skin. In our experience the use of Tunica vaginalis or scrotal dartos tissue in a scarred area as a waterproofing cover at the earliest opportunity decreases the recurrence rate coupled with the fact that the fistulas are small, easily manageable and more importantly reduce the psychological trauma of undergoing repeated surgeries by the patient.

The use of magnification, absorbable suture material and delicate tissue handling are also a must for a favourable result.

The treatment plan for a fistula must be individualized according to the size, location and number of fistulas with due attention to the local surrounding skin which all have an effect on the outcome of repair and to an extent dictates the type of repair to be performed. The minimum time interval of 6 months between any two procedures should be considered for a favourable outcome. The significantly improved success rates in the repair at the first, second and third attempts with the addition of a waterproofing layer suggests the use of this interposition layer should be done at the earliest available opportunity to prevent a reccurence rather than to reserve it for future occasions.

 
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]

[Table 1], [Table 2], [Table 3], [Table 4], [Table 5]

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