Oronasal Fistula As A Cleft Palate Sequela Closure With Tongue Flap

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Med Buccale Chir Buccale 2017;23:192-195 www.mbcb-journal.org


© The authors, 2017
DOI: 10.1051/mbcb/2017012 Médecine Buccale
Chirurgie Buccale

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Up-to Date Review And Case Report

Oronasal fistula as a cleft palate sequela : closure with


tongue flap. Case report with review of the literature
Thomas Leturc1,*, Anne-Gaelle Chaux-Bodard2, Christian Paulus3
1
Interne DES Chirurgie Orale, service de chirurgie maxillo-faciale et stomatologie, Hôpital Lyon Sud, France
2
Maître de Conférences des Universités Praticien Hospitalier, unité fonctionnelle de chirurgie du service de consultations et
traitements dentaires, Lyon, France
3
Praticien hospitalier, service de chirurgie maxillo-faciale et stomatologie, Hôpital Femme Mère Enfant, Lyon, France

(Received: 27 January 2017, accepted: 22 March 2017)

Keywords: Abstract -- Introduction: Oronasal fistula have several etiologies. Most of the time, the origin is congenital
oral fistula / cleft associated with cleft lip and patate patients, or it can be acquired after trauma, infection, neoplasm, postsurgical
palate / surgical complications or radio and chemonecrosis. Excepted for palatal obturators, their treatment is surgical and may use
flaps different intraoral flaps. Observation: A 9-year-old girl was sent to consult presenting an oronasal fistula as a sequela
of a cleft lip and palate, initially treated in Kosovo (Albania). This was an anterior midpalate fistula, large around 2
cm, functionnaly responsible of a nasal speech and food reflux in the nasal airways. The closure has been done with a
tongue flap. Commentary: Most of the oronasal fistulae secondary to a cleft palate are managed with palatal
rotational flaps. Nevertheless, they cannot be used in some cases, especially when the fistula size is too large, the
localization too anterior and with already scarred palatal tissues around. Then, tongue flap may represent a reliable
alternative. Conclusion: The tongue flap allowed a complete closure of the oronasal fistula, with a very good
tolerance from the child. It shows its interest into the list of the numerous existing oral flaps. However, considering
the discomfort of this technique, it must be done in selected cases, with compliant patients.

Introduction cause speech disorders, regurgitation of food in the nasal


cavity, bad odors, bad tastes, as well as upper respiratory tract
Oronasal fistulae are not a common reason for consultation. infections [1].
When the cause has been discovered, it is often a result of The majority of oronasal fistulae are small and can be easily
trauma, infection, tumors, postoperative iatrogenic side managed by palatal flaps.
effects, or osteonecrosis [1]. The main etiology of oronasal Large fistulae are more complex because there is less
fistulae remains congenital, associated with cleft palate [1]. healthy tissue available at the palatal level. Moreover, in cases
Oronasal fistulae are congenital malformations caused by requiring multiple surgical procedures, the scarring aspect of
the anomalies of fusion of the different prominences involved the palatal mucosa compromises the efficacy of any flaps used.
in the embryogenesis of the face, between the fourth and tenth Other flaps, such as a buccal mucosal flap, a buccal flap, a
weeks of development in utero. They represent the most muscular-mucosal flap on the facial artery, tongue flaps, or
common congenital craniofacial malformation occurring in one even the free micro-braided flaps may be used [1,3]. Their
in every 700 births [2]. respective indications are discussed in terms of the fistula
Their management is performed by a multidisciplinary width and location, and the patient’s surgical history, the
team and extends from birth to adolescence with a succession quality of tissues available, compliance, and dentition [1].
of surgical procedures. Nevertheless, the oronasal fistula
frequently persists and is found in 15–35% patients after Observation
primary closure of the palatovelar slit [3,4]. Such fistulae may
A 9-year-old patient came in for a maxillofacial surgical
consultation for advice on the management of an oronasal fistula.
The patient presented with a congenital bilateral cleft lip, which
was treated through three procedures in Kosovo. The cleft lip was
* Correspondence: [email protected] closed but alveolar continuity was found on the left side only.

This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits
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Med Buccale Chir Buccale 2017;23:192-195 T. Leturc et al.

Fig. 1. Oronasal fistula as a cleft palate sequela: initial state. Fig. 3. Tongue flap lifting.

Fig. 4. Stitched tongue flap to the anterior part of the fistula.


Fig. 2. First nasal layer.

At the palatal level, the patient had an extended oral enough to allow the suture of the flap to the palate without any
communication of the primary palate to the velum, responsible tension (Fig. 3). Lingual hemostasis was carried out using
for hyponasal speech and nasal reflux of liquids and solids. The bipolar electrocautery. The tongue was sutured at separate
first surgical intervention was to close the palate using rotational points with Vicryl 3.0. The flap was sutured on the anterior and
flaps. Subsequently, an oronasal fistula recurred between the lateral parts of the fistula, carrying out a second closure, on
palate and the median retro-incisor, and it was 2 cm in diameter separate points with Vicryl 3.0 (Fig. 4).
(type V on the Pittsburgh classification) (Fig. 1). The surgical procedure was simple, with negligible pain and
A tongue flap was made to fuse this oral fistula to the cleft without any complications.
palate. The procedure was performed under general anesthesia Postoperative pain was managed using a combination of
with nasotracheal intubation. The patient was placed in the paracetamol and nalbuphine while the patient was hospital-
dorsal decubitus position with the neck hyperextended. ized. Oral prednisolone at a dose of 1 mg/kg was administered
Infiltration of the palatal and lingual mucosa was performed for the first 2 days after surgery, and antibiotic therapy with
with 8 ml of xylocaine and adrenaline (1/100 000). Corticoste- amoxicillin (2 g/day) was prescribed for 7 days. The food was
roids (methylprednisolone 1 mg/kg) and antibiotics (amoxicillin administered orally, and it was a mixture of liquid and mixed
2 g/day) were administered before surgery. foods, and the patient was asked to communicate in writing.
A hinged flap was placed at the level of the palatal fistula Local care was performed after each meal by the family, by a
allowing the rehabilitation of the first nasal mucosal plane simple rinsing with saline solution. The patient was discharged
(Fig. 2). The lingual flap was attached to the anterior medial 48 h after the procedure, analgesia with level-1 analgesics was
pedicle and adapted to the width of the fistula. It was lifted tapered off and the patient could eat. The patient has coped
with a cold blade with a 5 mm thickness, which was long with this period between the two operating times very well.

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Med Buccale Chir Buccale 2017;23:192-195 T. Leturc et al.

Fig. 5. Flap healing one month after surgery.


Fig. 6. Tongue healing one month after surgery.
The pedicle section of the flap was completed in the second
phase of treatment. It was performed under general anesthesia
after a period of 2 weeks following the first surgery. The postoperative procedures are relatively simple and
An incision was made at the base of the lingual flap, thus common to other lingual surgeries with the immediate risks
releasing its anterior part. during the postoperative period being pain, edema, bleeding,
Lingual hemostasis was performed using bipolar electro- infection, hematoma, and temporary hypoesthesia [5]. The
cautery. The flap was elongated. Hermetic sutures on the main complications are the premature detachment of the flap,
posterior part of the palatal fistula were made by separate necrosis, speech impairment, and donor-site complications.
points with Vicryl 3.0. Outpatient treatment required no Complications are often associated with improper application
additional procedures. The postoperative follow-up evaluations of the technique (flap too short sutured on, rotation too
were very satisfactory with good healing 1 month after surgery large, poor flap design) [5,6]. The tongue flap does not cause
at the level of the palatal fistula (Fig. 5) as well as at the level speech impediments when tongue integrity is not compro-
of the lingual mucosa (Fig. 6). The last follow-up examination, mised [7].
performed 6 months after the procedure, ruled out fistula Technically, it is accepted that the tongue-flap thickness
recurrence, and the patient no longer had nasal reflux when must be 20% wider than the fistula because of a certain degree
eating. An improvement of the phonation was possible thanks of flap retraction [8] and its design may be adapted specifically
to speech therapy. to the fistula shape [9]. Most surgical teams prefer a two-plane
closure. The reconstruction of the nasal plane, either mucosal
Comment from the periphery of the fistula, or from a vomerine flap [10],
allows better support and better flap stability, thus decreasing
In 1901, Eiselberg was the first to describe a pedicle tongue the risk of failure.
flap for use in the reconstruction of intraoral volume defects. Alsalman et al., however, showed a success rate of 100%
Since then, the tongue flap has been used in several procedures with the closure of a palatal fistula using a tongue flap in a
for the reconstruction of the loss of volume from the intraoral single procedure on a series of five cases, where technically the
defects, including the repair of oronasal and sinus connections total closure of the nasal plane was not feasible [11].
and defects in the base, oral mucosa, lips, tongue, and In 1972, Jackson suggested the first use of the tongue flap
oropharynx [1,5]. for the closure of the palatal fistula in children with cleft
Its main limitation is the patient’s tolerance, which is a deformities and showed that the flap was safe and well
determining factor for the success of the technique. Thus, the tolerated by children [12]. Piggot et al. also demonstrated the
primary contraindications for tongue flaps are psychiatric success of the technique in both adults and children, including
disorders, mental retardation, and patients with heavy 100% success in their group for children aged <2.5 years [13].
comorbidities, which can affect the efficacy of general A literature review reveals that the majority of oranasal
anesthesia [5]. Active smoking, unbalanced diabetes, or fistulae in cases of cleft palates are successfully managed by
severe malnutrition, are secondary contraindications that local palatal flaps, but success is limited in more complex
need to be addressed. situations [1,3,14].
The first step is always performed under general anesthesia, Studies of the tongue flaps have been conducted in a few
with nasotracheal intubation. The resection of the pedicle can special situations:
be performed under local anesthesia but requires a good patient – wide fistulae (>1 cm) [3,6,10,15–17];
compliance to achieve hemostasis in good conditions. In a – anterior fistulae [6,10,15,18,19];
young child, as in the case presented, it is necessary to perform – residual fistulae on scars from multiple surgical interventions
this second procedure under general anesthesia. [5,15].

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Med Buccale Chir Buccale 2017;23:192-195 T. Leturc et al.

These three criteria are different in children and adults, 5. Strauss RA, Kain NJ. Tongue flaps. Oral Maxillofac Surg Clin N Am
studies on the closure of oral fistulae using tongue flaps all 2014;26:313–325.
report a high success rate of 85–100% with reliability and 6. Mahajan RK, Chhajlani R, Ghildiyal HC. Role of tongue flap in
palatal fistula repair: a series of 41 cases. Indian J Plast Surg
reproducibility [6,10,13,15–17,20].
2014;47:210–215.
Studies that have studied the result on phonation report 75%
7. Johnson PA, Banks P, Brown AE. Use of the posteriorly based
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670–673.
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size, they are treated successfully with palatal flaps. However, 11. Alsalman AK, Algadiem EA, Alwabari MS, Almugarrab FJ. Single-
when the fistula is wide or in an anterior position and/or when layer closure with tongue flap for palatal fistula in cleft palate
the tissues are scarred, palatal flaps cannot be used. In the case patients. Plast Reconstr Surg Glob Open 2016;4:852.
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success rates with the tongue flaps, including success rates in tissue and bone grafting. Br J Plast Surg 1972;25:93–105.
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fistulae. Br J Plast Surg 1984;37:285–293.
stability of the closure after expansion.
14. Cervenka B, Setabutr D, Rubinstein BK. Surgical repair of the cleft
Tongue-flap surgery, because of it technique and patient palate. Oper Tech Otolaryngol Head Neck Surg 2015;26:
discomfort between the two surgeries, must be used in ideal 121–126.
conditions and on carefully selected patients by a surgeon who 15. Vasishta SMS, Krishnan G, Rai YS, Desai A. The versatility of the
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Trauma Reconstr 2012;5:145–160.
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