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123 Journal of Medicine and Health Sciences Faculty of Medicine, Srinakharinwirot University

(Vol.15 No.3 December 2008)

Abstract

A 23-year-old man was presented with recurrent left-sided neck abscess which is characteristicclinical features of fourth branchial cleft fistula. The abscess burst spontaneously, forming a fistula. Diagnosisof fourth branchial fistula in this patient was verified by barium radiograph and injection of methylene blueinto the external opening. An internal opening was found at the apex of the left piriform fossa. The currenttreatment of choice involves complete excision of the sinus tract. Endoscopic cauterization of the internalfistulous tract was developed by some authors. It offers several advantages over open excision such asless injuries to important structures. The cost and duration of treatment are also reduced. Microlaryngoscopicelectric cauterization of the internal opening was used to close the fistula. There has been no recurrence ofthe cervical abscess during 10 months of follow-up. Therefore, microlaryngoscopic electric cauterizationseems to be a safe treatment for patients with fourth branchial cleft fistulas.

Key Words: branchial cleft fistula

A case report: Fourth branchial cleft fistula successfully treated withmicrolaryngoscopic cauterization

Niran HunchaisriDepartment of Ophthalmology & Oto-Rhino-Laryngology, Faculty of Medicine, Srinakharinwirot University

/ Case reportJournal of Medicine and Health Sciences (Vol.15 No.3 December 2008)

Niran HunchaisriDepartment of Ophthalmology & Oto-Rhino-Laryngolgy, Faculty of Medicine,Srinakharinwirot University62 moo 7 Ongkharak, Nakhon Nayok 26120, Thailand

124Journal of Medicine and Health SciencesFaculty of Medicine, Srinakharinwirot University(Vol.15 No.3 December 2008)

Fourth branchial cleft fistula 1 Barium methylene blue pyriform fossa sinus tract microlaryngoscope 10 microlaryngoscope fourth branchial cleft fistula

: branchial cleft fistula

.

Fourth branchial cleft fistula microlaryngoscopic cauterization: 1



Branchial cleft or pouch anomalies(sinuses,cysts and fistulas) arise from abnormaldevelopment of the embryological complex knownas the branchial apparatus. These anomalies mostcommonly present with a cervical mass or cyst,or with drainage from a cervical sinus opening.They account for up to 17% of pediatric neckmasses1. Third and fourth branchial pouchsinuses, however, are quite rare, accounting foronly 3-10% of all branchial anomalies2,3. Theseabnormalities often occur as tracts from thepiriform sinus to the thyroid gland and aresometimes referred to as piriform sinus fistulas.Third and fourth branchial fistulas usually presentas fluctuant neck masses located at the anteriorborder of the sternocleidomastoid muscle. Thesebranchial fistulas may also be associated with neckabscess or acute suppurative thyroiditis4.Treatment has traditionally involved excision ofthe sinus tract with partial thyroidectomy, if thethyroid gland is involved, Cannulation of thepiriform sinus fistula during direct laryngoscopyto aid complete surgical excision has also beendescribed in literature5. Some reports

have described endoscopic cauterization of fourthbranchial cleft sinuses with good results6,7. Thisarticle reports an additional case of fourthbranchial cleft fistula which is effectively treatedwith microlaryngoscopic electric cauterization.

Case ReportA 23 year-old man presented with an

abscess on the left side of neck. He had thehistory of neck abscess since childhood, but therewas no recurrence for more than ten years. Last 2years, he started to have few episodes of neckabscess. He was treated at private hospital withmedication and surgical drainage. After the lastepisode, an opening was seen at the prior abscessarea. There was frank leak from the fistula on oralintake of liquids. He was referred to HRH PrincessMaha Chakri Sirindhorn Medical Center for furthermanagement. On examination, there was apinhead-size opening on the left side of neck, justbelow the cricoid cartilage, with scarring aroundthe opening (Fig 1). Barium swallow showedspillage of the dye in the tract from left piriformsinus area to skin (Fig 2).

Figure 1 Suction tip pointed at the opening of the fistula.


Figure 2 Barium swallow showed fistulous tract from piriform sinus to skin (arrow).

He was scheduled for microlaryngoscopiccauterization under general anesthesia. At surgery,after suspension microlaryngoscope was applied,the internal opening of the fistula was identifiedby injection of methylene blue into the externalopening. The dye was seen coming from the apexof the left piriform sinus, anteriorly. The size of theinternal opening was approximately 2 mm. indiameter. The opening in piriform sinus wascauterized by suction cautery until it becameblanching. He was admitted for 3 days forobservation. Nasogastric tube feeding wasadviced for 2 weeks postoperatively. The externalopening was closed at 1-week follow-up period.Lastly, he had no recurrence of neck abscessduring ten months postoperative period.

DiscussionThe human branchial apparatus comprises 5paired mesodermal arches, separated by 4 pairsof endodermal and ectodermal invaginations thatare called pouches and clefts. A branchialfistula forms when there is remnant of both the

the pouch and the cleft with rupture of theinterposed branchial plate. A sinus is formedwhen the tract opens either to the gut or to theskin but not both, whereas a cyst is formed whenthere is no communication to both of them8.It has been found that 95% of the congenitalanomalies of the branchial apparatus are derivedfrom the second branchial arch, pouch, and cleft.The remaining anomalies stem from the first andthird arch. The remnants of the fourth branchialarch are very rare9-10.The theoretical course of athird branchial fistula would pass deep to thirdarch structures and superficial to fourth archstructures. More specifically, it would arise atthe skin near the mid-to-lower third of theanterior border of the sternocleidomastoidmuscle. A third branchial pouch fistula wouldpierce the platysma, ascend along the carotidsheath, pass over the superior laryngeal nerveand deep to the glossopharyngeal nerve, passbehind the internal carotid, pierce the thyrohy-oid membrane, and enter the upper lateralpiriform sinus wall11.



The theoretical course of a fourth branchialfistula would originate at the anterior border of thelower portion of the sternocleidomastoid muscle.It would also pierce the platysma, ascend alongthe carotid sheath, pass under the superiorlaryngeal nerve, but over the recurrent laryngealand hypoglossal nerves. A fourth branchial pouchsinus would then dip back into the chest to passaround the aortic arch on the left and thesubclavian artery on the right. Finally, it wouldascend to enter the larynx near the cricothyroidjoint or through the lower horn of the thyroidcartilage, pass through the inferior constrictor, andenter the apex of the piriform sinus12-13. It isprobably due to the complex and convolutedcourse that a complete fourth branchial fistula hasyet to be reported. Fourth branchial pouchanomalies typically present as a sinus or fistulafrom the apex of the piriform sinus to the deepside of the thyroid gland12. Approximately, 83-97%of them are on the left side14-15. It has beensuggested that this is due to asymmetricalvascular development16. Because of the similarity in their course, itis difficult to differentiate between the fourth andthe third branchial arch anomalies. Both sinusesare expected to open into the piriform sinus. Todifferentiate between these anomalies, surgicalexploration is required. If the sinus passes belowthe superior laryngeal nerve, a fourth branchialpouch sinus is suggested. On the other hand, ifthe sinus passes above the superior laryngealnerve, a third branchial pouch sinus issuggested17. Another clue to the origin of theanomalies is the location of the sinus opening inthe piriform sinus seen from endoscope. An open-ing in the apex of the piriform sinus suggests fourthbranchial cleft anomalies6.

Regardless of the pouch of origin,histologic examination of excised fistulous tractsoften reveals inflammatory changes tononkeratinized stratified squamous or stratifiedcolumnar epithelium. Transitional epithelium andsubepithelial lymphoid infiltrates have also beenreported18. While controversy remains regardingidentification of third and fourth branchial sinuses,the current treatment of choice involves completeexcision of the sinus tract. Some authors developnew treatment by electric cauterization6,7 andchemocauterization19, 20 of the internal sinus orfistulous tract for closing them. Jordan et al6reported a series of seven patients whounderwent endoscopic cauterization of thesinus opening, four of them had no evidence ofrecurrence during 18 months of follow-up. Verretet al7 reported a retrospective study to evaluatethe effectiveness of endoscopic cauterization asdefinitive treatment for fourth branchial cleftsinuses. Seven of the 10 patients treated withendoscopic cauterization of the fourth branchialcleft sinuses showed no recurrence with anaverage follow-up of 3 years. Three of the patientswere unavailable for follow-up, but medicalrecords of the hospital showed no additionaladmissions for those patients for neck masses.No morbidity of the procedure was identified.

The patient in this article was a case offourth branchial arch fistula, which manifested inchildhood with typical history of left-sided neckabscess. The abscess was recurrent and finallyformed a complete fistula, communicating fromneck to the apex of left piriform fossa. Thediagnosis was confirmed by a contrast study(barium swallow) and injection of methylene blueinto the fistula. Under direct laryngoscope, dyewas seen coming from the internal opening at


the apex of piriform sinus. Endoscopiccauterization offers several advantages overthe open procedure. Because of previousinfection and scar tissue, difficult dissections canbe avoided by endoscopy. As no open excisionis required, the risk of injuries to importantstructures are eliminated, i.e. injuries to thesuperior and recurrent laryngeal nerves,esophagus, trachea, and aorta. Also, theduration of treatment and the cost are reducedin endoscopy. Microlaryngoscopic cauterizationwas used in this patient because of the abovereasons. Despite no recurrence of neck abscessduring ten months postoperative period, longterm follow-up is needed.

ConclusionsThe diagnosis and management of fourth

branchial pouch anomalies are challenging.Diagnosis requires a high index of suspicion,from the clinical presentation of recurrent neckor thyroid abscess. Barium radiography andinjection of methylene blue are useful to confirmthe presence of a fistulous tract opening intopiriform apex. Microlaryngoscopic electriccauterization seems to be an effective primarytreatment for patients with a piriform sinusfistula.

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15. Taylor Jr WE, Myer III CM, Hays LL, CottonRT. Acute suppurative thyroiditis in children.Laryngoscope 1982; 92: 1269-73.

16. Rosenfeld RM, Biller HF. Fourth branchialpouch sinus: diagnosis and treatment.Otolaryngol. Head Neck Surg 1991;105:44-50.

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