|Year : 2014 | Volume
| Issue : 2 | Page : 108-110
Congenital parotid duct fistula: A case report and review of literature
Ravikumar Gopalakrishnan1, Naveen P Ravikumar2
1 Professor of Plastic Surgery, Thanjavur Medical College, Thanjavur, Tamil Nadu, India
2 Research Fellow, Thanjavur Medical College, Thanjavur, Tamil Nadu, India
|Date of Web Publication||16-Oct-2014|
Shanthi Nagar, Thanjuvar - 613 006, Tamil Nadu
Source of Support: None, Conflict of Interest: None
Parotid duct fistula is uncommon but difficult-to-treat complication that often results from a penetrating trauma. While there is general consensus in the literature as to the management of acute parotid injuries, treatment of chronic fistulas remains controversial. Congenital parotid duct fistula is a rare entity. Herein we present a 20-year-old female who presented with a parotid duct fistula since birth. Clinical evaluation revealed no accessory parotid gland or duct. Patient was found to have a combination of more prominent Plica semilunaris of the left eye, a finding not so far reported in the literature. Following complete examination and investigations, intra oral transposition of the fistulous opening and tract was done. We present this case for its rarity and the gratifying outcome of the surgical procedure.
Keywords: Congenital parotid ductal fistula, intra oral transposition of fistula, plica seminularis of the eye
|How to cite this article:|
Gopalakrishnan R, Ravikumar NP. Congenital parotid duct fistula: A case report and review of literature
. Arch Int Surg 2014;4:108-10
| Introduction|| |
Parotid fistula is a rare, devastating disease. It consists of a communication duct between the salivary gland or duct and the skin, through which saliva is discharged.  The most frequent aetiologies are accidental trauma or a post-operative complication after parotid gland surgery or Rhytidectomy. While there is general consensus in the literature as to the management of acute parotid injuries, treatment of chronic fistulas remains controversial. Congenital salivary fistulas are thought to arise from aberrant development of the salivary gland and are extremely uncommon. , Congenital fistula arising from parotid gland duct is still rarer, with only four reported cases.  The external opening of a parotid or accessory parotid fistula is classically located lateral to the angle of the mouth, which corresponds to the site of fusion of the maxillary and mandibular protuberances. ,, An important and consistent association is the presence of peri aural appendages.  We present this case for its rarity,  for the prominent plica semilunaris of the left eye which was not reported earlier and for the surgical intra-oral transposition of the fistula. ,,,.
| Case Report|| |
A 20-year-old female presented with history of intermittent discharge of watery fluid from an opening in the left cheek since birth. The discharge increased on eating and was not associated with any swelling or pain. There was no previous history of swelling, redness, pain, or injury to the region. Patient had normal general examination with normal vital parameters. Systemic examination was within normal limits. Local examination revealed a small punctum with over hanging skin fold, on the left side of the face, at a distance of about 2.0 cm from the angle of the mouth, and 1 cm inferior to a line joining the left tragus and angle of mouth. Skin puckering was seen extending to the angle of the mouth on the left side [Figure 1]. Expression of clear serous fluid was seen after local massage. This outflow increased when the patient swallowed two vitamin C tablets. The outflow of saliva was also increased when digital pressure was applied on the left parotid gland. No associated palpable mass or tenderness was noted. Oral examination revealed normal Stenson's ducts opening bilaterally. A clinical diagnosis of congenital parotid ductal fistula was made. A more prominent plica semilunaris on the left eye was present. Ultra sonogram and computed tomography (CT) scan showed normal parotid glands on both sides [Figure 2]. No accessory parotid gland was seen. Laboratory analysis of fluid from fistulous opening revealed raised serum amylase level of 3500 IU/ml, which confirmed the diagnosis of salivary fistula.
With the patient under naso-tracheal intubation general anaesthesia the external fistulous opening was cannulated with a 24 gauge intravenous catheterunderloup magnification. Methylene blue was injected through the catheter. The anomalous duct in continuity with a small ellipse of skin with catheter in situ was dissected for about 3 cm [Figure 3]. The fistulous opening was transposed intra-orally through a tunnel and sutured to the buccal mucosa with interrupted absorbable sutures. The skin defect was closed. Postoperative period was uneventful. Patient was followed-up for 2 years and the drainage was normal through intra-orally [Figure 4]. No sign of salivary duct stricture or fistula recurrence was observed. Patient was socially and cosmetically satisfied with outcome of treatment [Figure 5].
| Discussion|| |
The congenital diseases of parotid glands include aberrant or ectopic gland, aplasia, hypoplasia, hyperplasia, duct atresia, duct diverticuli, accessory ducts and congenital salivary fistulas. Congenital salivary fistulas can originate from the parotid gland, submandibular gland, ectopic salivary gland, and, rarely, accessory parotid gland. , The sites of opening of the fistulas have been the retroauricular region, the facial skin of the cheek, the oral mucosa, and skin of the cervical region. , Congenital salivary fistulas from the parotid gland are rare and they usually open at the facial skin near the angle of the mouth. ,, They are often accompanied by preauricular appendages.
In the embryo, the parotid gland arises as a solid outgrowth from the wall of the primitive oral cavity into the mesenchyme of the maxillary process. ,, After fusion of the maxillary and mandibular processes, which are both derived from the first branchial arch, the parotid duct takes its normal position inside the mouth at the level of the superior second molar. It is probable that when the parotid gland bud is abnormally located at the edge of the maxillary process, subsequent fusion of the processes places the duct opening on the skin of the face instead of at its typical position inside the mouth. This theory explains why congenital parotid fistulas had the fistulous orifice near the oral commissure in the line of the fusion of the maxillary and mandibular processes. , Hence, the embryologic origin of this anomaly, although related to the branchial apparatus, differs completely from that of the typical branchial cleft fistulas, as well as from that of the ectopic salivary fistulas found at the base of the neck.  This also explains the puckering of skin extending from the fistulous opening to the angle of mouth and upper lip in our case as there was no history of trauma. The diagnostic investigations are sialography, fistulography, CT and magnetic resonance imaging (MRI). , MRI sialography/fistulography, and computed tomography sialography/fistulography are much more expensive than conventional sialography/fistulography. 
Anti cholinergic agents are used to suppress glandular function during healing or in an attempt to close a fistula. A commonly used agent is propantheline bromide (Pro-Banthine), which inhibits the action of acetylcholine at the postganglionic nerve endings of the parasympathetic nervous system. Radiation therapy induces fibrosis and atrophy of the gland. Approximately 1800 rads for more than 6 weeks is required .Radiation is considered for refractory salivary fistulas.  Botulinum toxin A has been used. It has a latency period and requires repeated injections and the effects may not last long enough to bring about complete remission of disease. Fibrin glue has also been used recently and is rendered inactive by saliva leading to recurrence of fistula.
Surgical management includes diversion of parotid secretion into mouth. The various reconstructive methods are delayed primary repair of the duct, reconstruction of the duct with vein graft, mucosal flaps and suture of proximal duct to buccal mucosa.  Parotidectomy, duct ligation and tympanic neurectomy are the other modalities of treatment.
| Conclusion|| |
A rare case of congenital parotid duct fistula associated with more prominent plica seminularis of the left eye is discussed. Intra-oral transposition of the fistulous opening with the help of 24 gauge catheter and methylene blue is presented. This prevents an inadvertent injury to the duct during its mobilisation. The documentation of each surgical step may help other surgeons treat similar conditions in the future.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]