DOI No.: 10.21176/ ojolhns.0974-5262.2015.09.

SECOND BRANCHIAL SINUS PRESENTING

WITH ORAL HAIR
*Charu Singh, **Jaya Gupta, ***Samiullah, ****Ranveer Singh, *****Pankaj
Verma

ABSTRACT

INTRODUCTION

Branchial sinus anomalies although not rare can

have variable presentations. We present a case of 24year-old female with a second arch branchial pouch
presenting with a hair in the sinus internal opening.
This case illustrates an unusual presentation of
branchial anomaly and also provides possible
explanation for links between branchial arch anomalies
and dermoids or hairy polyp.

Second branchial cleft anomalies are thought to

originate from the branchial apparatus that did not
completely obliterate during head and neck
embryogenesis. The spectrum of developmental
anomalies includes cysts, sinuses and fistulas and
various combinations of these anomalies. Second
branchial cleft anomalies are the most common of the
branchial anomalies (Schoroeder et al 20071 ;
2

Keywords: Second Branchal Sinus, Branchialarch,

Oral Hair, Anomaly.

Address of Correspondence:
Dr Jaya Gupta
Room No 604 TG Campus

Choudhary et al 2003 ).The development of the second

arch takes place over a more extended time period
hence anomalies in this region are more common3.
95% of branchial cleft anomalies originate from the
second branchial cleft4. We present a case report of a
second branchial cleft sinus with an unusual
presentation and an equally unusual treatment.

Khadra, Sitapur road,

India, Ph.: +91-9415979055

E-mail: jayaguptaent@hotmail.com

CASE REPORT
A 24-year-old woman complained of foreign
body sensation throat for 6 months. She came in the
ENT OPD giving history of hair in her mouth. On
examination there was a hair in her oropharynx coming
out from a sinus on her anterior tonsillar pillar. [Figure
1] At the same location on the opposite side also there
was a pit with no opening. There was no history of any
discharge from the sinus. She did not have any pain or
swelling in neck suggestive of any other branchial arch
anomaly. Barium swallow and pharyngoscopy were
unremarkable.

*Associate professor and Head, **Senior resident, ****Assistant professor, *****Senior resident, ENT Department.Integral Institute of medical
sciences and research.Dasauli, Post Bas-ha, Kursi road. Lucknow, ***Associate professor Era Medical college.Sarfarazganj Lucknow, India

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Vol.-9, Issue-I, Jan-June - 2015

Lucknow 226003

DOI No.: 10.21176/ ojolhns.0974-5262.2015.09.1

Fig 1: Sinus opening on anterior tonsillar pillar with hair protruding.

The hair was pulled along with its root and the
patient

got relieved

of the symptoms

Fig 3: Sinogram outlining the sinus tract from sinus opening in

oropharynx till hyoid.

almost

instantaneously. [Figure 2]

The patient is under follow up for past one year

without any symptoms.
DISCUSSION
Pharyngeal arches are arches of mesenchyme
derived from paraxial and lateral mesoderm and neural
cell, which appear in 4th and 5th week of development.
They are covered externally by ectoderm, which forms
successive clefts and internally lined by endoderm, which
5

forms pouches between arches . Branchial anomalies can

be lined with either respiratory or squamous epithelium.
Cysts are often lined by squamous epithelium, whereas
Fig 2: Sinus opening on left side after removal of hair and pit on left
side anterior tonsillar pillar

sinus and fistulas are more likely to be lined with ciliated

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Vol.-9, Issue-I, Jan-June - 2015

columnar epithelium .
The patient was given intravenous antibiotics for

In the present case the ectoderm seems to be lining

three days to reduce any infection, adhesion or

the sinus tract and giving rise to hair. The histological

granulation around the tract to demonstrate entire course

examination did not show any variation from normal skin

of fistula. The sinus was then cannulated with venflon

hair unlike those reported by Farazaneh Agah-Hosseini ,

cannula no 26 from oropharynx as no external opening

was present. Iohexol was injected into the sinus and
patient was radiographed. The radiological image
depicted the sinus tract going up to the hyoid and ending

Miles , and Baughman . In all these cases the etiology

was not clear; however in our case the hair is an
ectodermal derivate, which seems to be embedded in the
cervical sinus during embryogenesis.

in a pouch. [Figure 3] CT scan of the neck did not

elements in association with the tract. The patient being

Other cases in literature where such hair have

been reported along with branchial arch anomalies are

asymptomatic the surgical intervention is not indicated.

hairy polyp.10,11 In these cases the authors have

demonstrate any other ectodermal or mesodermal

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DOI No.: 10.21176/ ojolhns.0974-5262.2015.09.1

polyps

are

developmental

lies in the simplicity and avoidance of extensive

malformations of branchial anomalies. In a case

dissection. However it is a blind procedure with high

reported by Burns, patient had a hairy polyp attached to

the upper pole of tonsil with second arch branchial

risk of injury to surrounding vital structures, hence it is

sinus opening at the junction of upper 2/3 and lower 1/

complete fistula, which are very rare.

not widely practiced. This technique requires a

3.In another case reported by Vaughman, patient had a

15

hairy polyp attached to the palate with a discharging

Pull through technique described by Talaat

first arch sinus. The presence of these hairy polyps or

dermoids in oropharynx in these patients with branchial

which the pharyngeal portion of the fistula is approached

arch anomalies could also be a coincidential finding.

surrounding structures and delivered through mouth. In

However in our case the ectodermal tissue element was

this technique the patient should at least have an external

found in ororpharynx coming out of branchial pouch

opening from where the dissection can be

sinus. This serves as a connecting link between

commenced.

dermoids or hairy polyps and branchial arch anomalies.

Our case adds further weight to the above theory that
dermoids in oropharynx represents developmental
anomalies of branchial arches.

in

per orally after tonsillectomy. The fistula is freed from the

Fistulectomy technique described by Takehito

16

Oshio using nylon thread as a guide wire and traction

on the gauze ball at one end of the fistula. In this method
also at least one external opening is required.

Another rare finding in our case was distinct

opening of the sinus on the anterior tonsillar pillar. This is
the first case in the world literature in which an isolated
2nd arch branchial pouch sinus is reported. The location
of inner opening of 2nd arch branchial pouch sinus has
only been found to lie in close association with tonsillar
fossa as per the methylene blue dye tests. The tract may
end in the upper half of the posterior tonsillar pillar,
supratonsillar fossa or directly on to the tonsillar

Attempts have been made to treat the fistula by

injecting sclerosing agents (Bailey H)1 7 and
18

trichloroactetic acid (Kim et al) electrocautery (Jordan

et al)19 endoscopic diathermy using uretheral diathermy
wire (PA Rhea)20 All these methods carry a definite
risk of damage to important nearby structures and
pharyngeal perforation. The clinical results achieved
by the above authors are encouraging but there is a

surface . In the presenting case not only is the opening

possibility of recurrence of infection due to distal parts

clearly visible, but also its location is rare. Here we

of the sinus tract.

suggest a classification for the inner opening of second

CONCLUSION

12

arch sinus/fistula A- posterior tonsillar pillar, B tonsillar

fossa or supratonsillar cleft, C Anterior tonsillar pillar.

it would be challenging to remove this sinus completely.

In the literature many techniques have been described for
treatment of such branchial anomalies.

Jan-June

uncommon presentation in ENT OPD. However

Due to this rare presentation and no external opening

13

Though second branchial arch anomalies are a not

complete second arch fistulae are rare and comprise
12

2% of all branchial anomalies. This is the first case of

branchial pouch sinus with a hair that has been reported
in world literature (to our knowledge). Not only is this

14

Taylor and Bicknell and Lee and Krishanan

have described stripping of branchial fistula by passing
a stripper inside the tract. The advantage of this method

2015

hairy

IssueI,

that

an exclusive presentation but also it helps to fill the

missing gaps between the dermoids or hairy polyp and
branchial arch anomalies. It also reminds us that the

49

Vol.-9,

suggested

DOI No.: 10.21176/ ojolhns.0974-5262.2015.09.1

anatomy and embryogenesis of neck is complex and

some of its presentations and treatment still eludes us.
DISCLOSURES

10. Burns BV, Axon PV, Pahade A. Hairy polyp

with an ipsilateral branchial sinus:evidence that
hairy polyp is a second branchial arch
malformation. J laryngol Otol. 2001; 115:145-8.

(a) Competing interests/Interests of Conflict- None

(b) Sponsorships - None
(c) Funding - None
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Chaudhary N, Gupta A, Motwani G et al. Fistula

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Mitroi M, Dumitrescu D, Simionescu C et al.

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