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Congenital Periauricular Fistulas: Possible


Variants of the Preauricular Sinus

Article in International Journal of Pediatric Otorhinolaryngology · November 2014


DOI: 10.1016/j.ijporl.2014.08.005

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International Journal of Pediatric Otorhinolaryngology 78 (2014) 1843–1848

Contents lists available at ScienceDirect

International Journal of Pediatric Otorhinolaryngology


journal homepage: www.elsevier.com/locate/ijporl

Congenital periauricular fistulas: Possible variants of the preauricular


sinus
Jae-Ryong Kim a, Do Hun Kim a, Soo Keun Kong b, Pyung Mo Gu a, Tae Ui Hong a,
Byeong Jin Kim a, Kyung Wook Heo a,*
a
Department of Otorhinolaryngology-Head and Neck Surgery, Inje University College of Medicine, Busan Paik Hospital, Busan, Republic of Korea
b
Department of Otorhinolaryngology-Head and Neck Surgery, Pusan National University School of Medicine, Busan, Republic of Korea

A R T I C L E I N F O A B S T R A C T

Article history: Objective: Although most preauricular sinuses are located near the anterior limb of the ascending helix,
Received 17 April 2014 some are located in unusual areas around the auricle, as shown previously in limited reports. This study
Received in revised form 4 August 2014 analyzed the clinical manifestations of congenital periauricular fistulas with unusual fistula locations
Accepted 7 August 2014
and the possible relationship with the classical preauricular sinus.
Available online 15 August 2014
Methods: We reviewed the medical records of patients who underwent congenital periauricular sinus
excision by three surgeons and followed them for more than 6 months. Clinical manifestations were
Keywords:
compared between classical preauricular sinus (classical group) and congenital periauricular fistula
Congenital preauricular fistulas
Surgery
(variant group) patients.
Results: The classical and variant groups included 192 and 20 ears each, respectively. In the variant
group, the fistula locations were in the ascending helix crus (15 ears, most common), infra-auricular area
(3 ears), supra-auricular area (1 ear), and anterior to tragus (1 ear). In ears with fistulas in the ascending
helix crus, the fistulous tract most often showed a medial direction (9 ears). There were 4 ears in the
variant group with fistulous tracts running toward the postauricular area.
Conclusions: Congenital periauricular fistulas can be located anywhere around the auricle, and also
considered the variant of preauricular sinus because the whole tracts were limited to lateral side of
temporalis muscle and parotid gland as well as not associated with external auditory canal and facial
nerve. Among them, the most common variant was a fistula on the ascending helix crus with short
fistulous tract directed medially.
ß 2014 Elsevier Ireland Ltd. All rights reserved.

1. Introduction discernible and repair does not usually require the addition of skin
or cartilage. Of them, absence of or cleft in helix, lobular anomalies,
During the fourth week of gestation, pinna development begins tragal anomalies, preauricular sinuses, auricular appendices, and
from the first and second branchial arches surrounding the first other grade I dysplasia are frequently encountered [3].
branchial cleft. This mesoderm gives rise to six outgrowths, the Classically, a preauricular sinus manifests as a small opening,
hillocks of His, that condense and fuse to form pinnae during the usually near the anterior limb of the ascending helix. Although
fifth and sixth weeks of gestation [1,2]. By the 20th fetal week, as most preauricular sinuses are located anterior to the external
the mandible grows and develops, the pinna migrates cephalad to auditory canal [4], a small portion have been reported and located
attain the adult configuration and location. During this process, ear in unusual areas like the superoposterior edge of the helix, the
deformities such as preauricular sinus can occur due to a variety of tragus, the lobule, the ascending helix crus, supra-auricular area,
internal and external factors, mostly in the upper part of the ear [2]. and the postauricular area [1,5–8]. However, few reports focus on
Minor anomalies of the external ear have been classified by a patients with congenital periauricular fistulas in unusual locations.
surgical perspective, where the structures of a normal ear are Choi et al. [6] have used the term ‘variant of preauricular sinus’
indicating the congenital fistulas at ascending helix crus with
tracts running posterior or posteroinferior direction.
* Corresponding author. Tel.: +82 51 890 6379; fax: +82 51 892 3831. This study reviewed a series of congenital periauricular fistulas
E-mail addresses: heokw96@daum.net, heokw96@inje.ac.kr (K.W. Heo). that had been addressed surgically and classified them according

http://dx.doi.org/10.1016/j.ijporl.2014.08.005
0165-5876/ß 2014 Elsevier Ireland Ltd. All rights reserved.
1844 J.-R. Kim et al. / International Journal of Pediatric Otorhinolaryngology 78 (2014) 1843–1848

to the fistula site to analyze the clinical manifestations of mass, and discharging sinus after healing were regarded as
congenital periauricular fistulas with unusual fistula locations recurrence. The excision of bilateral lesions was considered two
and compare them with classical preauricular sinus. operations.

2. Materials and methods 2.2. Surgical techniques

2.1. Patients Patients under age 10 years and those afraid of the procedure
received general anesthesia, including orotracheal intubation.
This study included only those patients who underwent a Others received local anesthesia using injection of 2% lidocaine HCl
congenital periauricular sinusectomy between July 2007 and and 1:100,000 epinephrine solution.
January 2011, in two tertiary referral centers by three surgeons and Surgery in the classical group was performed as follows. A
who could be followed for at least 6 months. Indications for surgery preauricular elliptical incision around the fistula was created to
included persistent discharge, recurrent infection or both. Patients minimize any cosmetic alteration of esthetic units of the face. The
with acute inflammatory exacerbation underwent the operation 4 fistulous tract was dissected medially to the temporalis fascia and
weeks after the lesion resolved with conservative treatment that posteriorly to cartilage of the ascending helix. A portion of the
included systemic antibiotics, dressing, and drainage of the abscess cartilage or perichondrium of the helix at the base of the sinus was
if needed. Preoperative pure tone audiometry (PTA) was performed also excised to ensure complete tract excision. In cases with a
in a single institute, if possible. previous infection or ruptured sinus, the dissection was completed
The patients were categorized into classical or variant groups after reaching healthy soft tissues anteriorly, the temporalis fascia
according to the location of the congenital fistula opening. The medially, and the helix cartilage posteriorly. The wound was closed
classical group was defined as patients with a fistula located at the in layered fashion, with or without compression as needed [9].
anterior direction of the ascending helix; all patients outside of the Surgery in the variant group was performed as follows. After
classical group were considered part of the variant group. Informed injection of topical vasoconstrictor and anesthetic agents, the sinus
consent was obtained from all patients. The study was approved by was filled with gentian violet. An elliptical incision around the
the Institutional Review Board. fistula and dissection along the fistulous tract was conducted with
The medical records of the two groups were reviewed, and scissors and knives. If the fistulous tract closely approached the
relevant demographic and clinical data were collected pre- and other side of auricular skin toward the postauricular area, a
post-operatively. Postoperative local inflammation, subcutaneous retroauricular incision was done for en bloc-type removal of the

Fig. 1. (A) Preoperative photograph showing two openings in the left auricle in Case 1. The medial opening (arrow) is located at the skin just below the intertragic notch, while
the lateral opening (arrow head) is at the middle of the lobule’s free lateral border. (B) Anteroposterior and lateral fistulogram shows that the medial (black arrow) and lateral
(white arrow head) openings are connected. Dye entering the medial opening (black arrow) flows out to the lateral opening (white arrow head). (C) The excised fistular tract is
1.8  1.9  1.2 cm in size and horseshoe-shaped. The central portion of the tract is incorporated in the antitragal cartilage. (From Heo KW, Congenital auricular fistula
confined to an ear lobule. J. Clin. Otolaryngol. 2007; 18:125–127, with permission).
J.-R. Kim et al. / International Journal of Pediatric Otorhinolaryngology 78 (2014) 1843–1848 1845

Fig. 2. (A) Preoperative photograph showing a congenital fistula at the superior attachment of the right auricle in Case 2. (B) The fistulous tract was running inferiorly with a
1.6-cm length and was completely excised.

tract. Surgical wound closure and compression were conducted in (Fig. 3). Because the patient’s parents wanted a cosmetic operation,
the same fashion as the classical group. Representative cases the fistula and skin tag removal was performed under general
according to fistula location are described below. anesthesia. During the operation, the fistula revealed a 0.4-cm
straight single tributary extending medially to the subcutaneous
2.3. Case 1 tissue. The postoperative course was uneventful.

A 3-year-old female visited the hospital with complaints of a 2.6. Case 4


recurrent, foul, odorous discharge from two openings in the left
auricle that had existed since birth. Physical examination A 3-year-old male came to the hospital with a congenital fistula
confirmed two openings in the left ear lobule. The medial opening at the ascending helix crus and inferior protruding soft tissue
was located in the skin just below the intertragic notch, while the
lateral opening was at the middle of the free lateral border of the
lobule (Fig. 1A). A fistulogram showed that contrast medium
injected into the medial opening moved slightly up to the
antitragal area, proceeded outward about 2 cm, then down a little,
and finally reached the lateral opening (Fig. 1B).
After general anesthesia, circumferential incisions were per-
formed around the medial and lateral openings. The fistula tract
from both openings intersected at the antitragal cartilage, which
was confirmed by insertion of metal and plastic probes. After the
fistula tract from the medial opening was dissected, the whole tract
and the attached antitragal cartilage were excised en bloc. The
excised tract was horseshoe-shaped and 1.8  1.9  1.2 cm long
(Fig. 1C). (This case was previously published as a case report in J.
Clin. Otolaryngol. 2007; 18:125–127, and is included in this study
with permission.)

2.4. Case 2

A 13-year-old female experienced recurrent discharge from an


opening in the right supra-auricular skin from birth. On physical
examination, a fistula was found at the upper attachment of the
right auricle that was surrounded by relatively thickened soft
tissue (Fig. 2A). Under general anesthesia, the fistulous tract was
excised as in the previous case description. The tract was running
inferiorly, located medial to the auricle and lateral to the
temporalis fascia. The dissected tract was 1.6 cm long (Fig. 2B).

2.5. Case 3

A 2-year-old female visited the hospital with a congenital Fig. 3. In case 3, preoperative photograph showing a congenital fistula (arrow) at the
fistula and multiple skin tags at the anterior direction of tragus preauricular area with several skin tags in a 2-year-old girl.
1846 J.-R. Kim et al. / International Journal of Pediatric Otorhinolaryngology 78 (2014) 1843–1848

(Fig. 4A). He had a history of repeated discharge and swelling from Table 1
Characteristics of patients according to group.
the fistula. His case was treated as a variant preauricular sinus, and
the fistula tract was excised using only an elliptical incision around Classical group Variant group
the fistula, and the soft tissue underneath the ascending helix crus Patient number (male: female) 159 (47:112) 19 (6:13)
resolved (Fig. 4B). The wound was repaired in a layered fashion. Ear number 192 20
Mean age (range) (year) 1–76 (18.8) 2 45 (16.7)

3. Results
Nine ears had received previous surgery in the classical group,
The classical group consisted of 159 subjects (47 males, 112 but there were none in the variant group. Forty-five ears in the
females) ranging in age from 1 to 76 years (mean, 18.8 years; classical group had a history of incision and drainage along with 2
Table 1). Of them, 33 patients received simultaneous excision of ears in the variant group. In the variant group, there were 4 ears
bilateral preauricular sinuses. The variant group consisted of 19 with fistulous tracts running toward the postauricular area, which
subjects (6 males, 13 females) ranging in age from 2 to 45 years required additional retroauricular incision. However, no ears in the
(mean, 16.7 years; Table 1). One patient in the variant group classical group required retroauricular incision. After surgery, each
underwent simultaneous excision of bilateral congenital sinuses. group included one ear with postoperative hematoma, which was
There was only one subject (representing case 3) with controlled using conservative methods. Recurrence was observed
associated auricular anomaly, which was skin tags at preauricular in 3 ears in the classical group and none in the variant group
area. No subject showed other head and neck anomaly, including (Table 3).
branchio-oto-renal syndrome. Familial occurrence of a congenital
periauricular fistula was investigated only in the variant group, and 4. Discussion
two subjects had parents (one father and one mother) with a
classical preauricular sinus. Hearing was evaluated preoperatively Preauricular sinus has an estimated incidence of 0.1–0.9% in the
in 50 subjects in the classical group and eight in the variant group. United States and Europe, 2.5% in Taiwan, 4–10% in some African
The audiometry identified five patients with sensorineural hearing countries, and 2.53% in Korea [4,10–13]. However, the incidence of
loss, which was considered presbycusis because all of them were variant-type preauricular sinus is unknown. There have been only
older than 60 years. No case had a conductive hearing loss. two previous studies reporting more than 10 ears with a unusual
In the variant group, fistula locations were ascending helix crus location of fistula of preauricular sinus [1,6]. In the present study,
(15 ears, including Case 4), infra-auricular area (3 ears, including we collected information on 20 ears with non-classical fistula and
Case 1), supra-auricular area (1 ear, Case 2), and anterior to tragus describe the clinical course and surgical approach. We authors
(1 ear, Case 3). In ears with fistulas in the ascending helix crus, the should believe that the term ‘variant of preauricular sinus’ seems
fistulous tract direction was posteroinferior (5 ears), medial (9 appropriate because the congenital periauricular fistulas and
ears), and inferior (1 ear, Case 4). Also, the mean length of fistulous fistula tracts in all cases of this study were related to the auricle
tract was 1.82 cm for the posteroinferior directed tract, 0.67 cm for anatomically, were consistently lateral to the temporalis fascia and
the medial directed tract, and 0.6 cm for the inferior directed tract parotid gland, and did not violate the facial nerve or external
(Table 2). auditory canal. However, it is possible that others would argue

Fig. 4. (A) Preoperative photograph showing a congenital fistula at the ascending helix crus (arrow) with inferiorly bulging soft tissue (arrow head) suggesting the direction of
fistulous tract in case 4. (B) The fistulous tract is excised only with elliptical incision around the fistula and the preoperative soft tissue underneath the ascending helix crus
was resolved.
J.-R. Kim et al. / International Journal of Pediatric Otorhinolaryngology 78 (2014) 1843–1848 1847

Table 2 Davis assumed that the auricle developed from the six hillocks in a
Subdivision of the variant group according to fistula location.
rather precise manner, with hillock 2 producing the tragus, hillock
Fistula location Ear Tract direction (No. of ears)/ 3 the anterior helix, hillock 4 the superior helix and corresponding
number the mean length (cm) antihelical portion, hillock 5 the posterior helix and corresponding
Above ascending Helix crus 15 Posterior–inferior (5)/1.82 antihelical portion, and hillocks 6 and 1 the lobule [4]. Park
Medial (9)/0.67 deduced that the region supplied by the superficial temporal artery
Inferior (1)/0.6 is compatible with the mandibular hillock region and the region
Infra-auricular 3 Transverse (1)/4.9
supplied by the posterior auricular artery is compatible with the
Superior (2)/1.45
Supra-auricular 1 Inferior (1)/1.6 hyoid hillock region [16]. Park also concluded that hillocks 1 and 6
Anterior to tragus 1 Medial (1)/0.6 produce the earlobe, and hillock 4 or 5 produces the antihelix or
helix, hillock 2 produces the tragus, and hillock 3 produces the
ascending helix [16]. In the present study, the fistulous tract course
in the ears with infra-auricular congenital fistulas, including Case
Table 3 1, was from the intertragic notch to the middle of the free lateral
Clinical parameters of ears according to group.
border of the lobule. This finding is consistent with the proposals of
Classical group Variant group Davis and Park, and could be explained by insufficient fusion of
Previous operation 9 (4.7%) 0 (0%) hillocks 1 and 6. In this manner, the fate of the six hillocks of His
History of incision and drainage 45 (23.4%) 2 (10%) would be precisely predicted by the clinical manifestations of the
Required postauricular incision 0 (0%) 4 (20%) variant preauricular sinus.
Posteriorly directed tract 0 (0%) 4 (20%)
Postoperative hematoma 1 (0.5%) 1 (5%)
Recurrence 3 (1.6%) 0 (0%) 5. Conclusion

This report describes a case series of congenital periauricular


fistulas, which are likely variants of a preauricular sinus because
the whole tracts were limited to lateral side of temporalis muscle
another diagnosis besides preauricular sinus in these cases. In
and parotid gland as well as not associated with external auditory
addition, it would be necessary to perform fistulography preoper-
canal and facial nerve. The fistula locations could be anywhere
atively as in Case 1 or to monitor the facial nerve during the
around the auricle. Among them, the most common variant was a
operation if branchial cleft anomalies were suspected.
fistula on the ascending helix crus with a relatively short fistulous
Choi et al. [6] reported that the variant type of preauricular
tract projecting in a medial direction. During diagnosis of a
sinus comprised 10.9% (11/101) of preauricular sinus, that all
congenital lesion around the auricle, the possibility of variant
fistulas were located on the ascending helix crus, and that the
preauricular sinus should always be considered and fistulogram
fistulous tract was directed posteroinferiorly. In this study, the
would be helpful during preoperative evaluation.
variant group was 9.4% (20/212) of whole subjects, which was
similar to Choi et al. However, in fistula cases at the ascending helix
Acknowledgments
crus, the direction of fistulous tract directed posteroinferiorly in
only one third of ears (5/15), for which the majority (4/5) needed
This work was supported by a grant from Research year of Inje
retroauricular incision. The authors are not certain of the reason for
University (Grant number: 20140032) in 2014.
the difference in direction of fistulous tract with on the ascending
The English in this document has been checked by at least
helix crus between this and the previous study [6].
two professional editors, both native speakers of English. For
During the surgery for preauricular sinus, there are several
a certificate, please see: http://www.textcheck.com/certificate/
factors which are known to reduce recurrence. They include
BYEgCJ.
meticulous dissection of the tract by an experienced surgeon,
avoidance of sinus rupture, a supra-auricular approach with
clearance down to the temporalis fascia, closure of wound dead References
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