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PEDOT-7234; No. of Pages 6 International Journal of Pediatric Otorhinolaryngology xxx (2014) xxx–xxx

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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 b

Department of Otorhinolaryngology-Head and Neck Surgery, Inje University College of Medicine, Busan Paik Hospital, Busan, Republic of Korea 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: Received 17 April 2014 Received in revised form 4 August 2014 Accepted 7 August 2014 Available online xxx

Objective: Although most preauricular sinuses are located near the anterior limb of the ascending helix, some are located in unusual areas around the auricle, as shown previously in limited reports. This study analyzed the clinical manifestations of congenital periauricular fistulas with unusual fistula locations and the possible relationship with the classical preauricular sinus. 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 compared between classical preauricular sinus (classical group) and congenital periauricular fistula (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.

Keywords: Congenital preauricular fistulas Surgery

1. Introduction During the fourth week of gestation, pinna development begins from the first and second branchial arches surrounding the first branchial cleft. This mesoderm gives rise to six outgrowths, the hillocks of His, that condense and fuse to form pinnae during the fifth and sixth weeks of gestation [1,2]. By the 20th fetal week, as the mandible grows and develops, the pinna migrates cephalad to attain the adult configuration and location. During this process, ear deformities such as preauricular sinus can occur due to a variety of internal and external factors, mostly in the upper part of the ear [2]. Minor anomalies of the external ear have been classified by a surgical perspective, where the structures of a normal ear are

* Corresponding author. Tel.: +82 51 890 6379; fax: +82 51 892 3831. E-mail addresses: [email protected], [email protected] (K.W. Heo).

discernible and repair does not usually require the addition of skin or cartilage. Of them, absence of or cleft in helix, lobular anomalies, tragal anomalies, preauricular sinuses, auricular appendices, and other grade I dysplasia are frequently encountered [3]. Classically, a preauricular sinus manifests as a small opening, usually near the anterior limb of the ascending helix. Although most preauricular sinuses are located anterior to the external auditory canal [4], a small portion have been reported and located in unusual areas like the superoposterior edge of the helix, the tragus, the lobule, the ascending helix crus, supra-auricular area, and the postauricular area [1,5–8]. However, few reports focus on patients with congenital periauricular fistulas in unusual locations. 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. This study reviewed a series of congenital periauricular fistulas 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.

Please cite this article in press as: J.-R. Kim, et al., Congenital periauricular fistulas: Possible variants of the preauricular sinus, Int. J. Pediatr. Otorhinolaryngol. (2014), http://dx.doi.org/10.1016/j.ijporl.2014.08.005

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to the fistula site to analyze the clinical manifestations of congenital periauricular fistulas with unusual fistula locations and compare them with classical preauricular sinus.

mass, and discharging sinus after healing were regarded as recurrence. The excision of bilateral lesions was considered two 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. Others received local anesthesia using injection of 2% lidocaine HCl and 1:100,000 epinephrine solution. Surgery in the classical group was performed as follows. A preauricular elliptical incision around the fistula was created to minimize any cosmetic alteration of esthetic units of the face. The fistulous tract was dissected medially to the temporalis fascia and posteriorly to cartilage of the ascending helix. A portion of the cartilage or perichondrium of the helix at the base of the sinus was also excised to ensure complete tract excision. In cases with a previous infection or ruptured sinus, the dissection was completed after reaching healthy soft tissues anteriorly, the temporalis fascia medially, and the helix cartilage posteriorly. The wound was closed in layered fashion, with or without compression as needed [9]. Surgery in the variant group was performed as follows. After injection of topical vasoconstrictor and anesthetic agents, the sinus was filled with gentian violet. An elliptical incision around the fistula and dissection along the fistulous tract was conducted with scissors and knives. If the fistulous tract closely approached the other side of auricular skin toward the postauricular area, a retroauricular incision was done for en bloc-type removal of the

This study included only those patients who underwent a congenital periauricular sinusectomy between July 2007 and January 2011, in two tertiary referral centers by three surgeons and who could be followed for at least 6 months. Indications for surgery included persistent discharge, recurrent infection or both. Patients with acute inflammatory exacerbation underwent the operation 4 weeks after the lesion resolved with conservative treatment that included systemic antibiotics, dressing, and drainage of the abscess if needed. Preoperative pure tone audiometry (PTA) was performed in a single institute, if possible. The patients were categorized into classical or variant groups according to the location of the congenital fistula opening. The classical group was defined as patients with a fistula located at the anterior direction of the ascending helix; all patients outside of the classical group were considered part of the variant group. Informed consent was obtained from all patients. The study was approved by the Institutional Review Board. The medical records of the two groups were reviewed, and relevant demographic and clinical data were collected pre- and post-operatively. Postoperative local inflammation, subcutaneous

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).

Please cite this article in press as: J.-R. Kim, et al., Congenital periauricular fistulas: Possible variants of the preauricular sinus, Int. J. Pediatr. Otorhinolaryngol. (2014), http://dx.doi.org/10.1016/j.ijporl.2014.08.005

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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 the same fashion as the classical group. Representative cases according to fistula location are described below. 2.3. Case 1 A 3-year-old female visited the hospital with complaints of a recurrent, foul, odorous discharge from two openings in the left auricle that had existed since birth. Physical examination confirmed two openings in the left ear lobule. The medial opening 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 performed 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.)

(Fig. 3). Because the patient’s parents wanted a cosmetic operation, the fistula and skin tag removal was performed under general anesthesia. During the operation, the fistula revealed a 0.4-cm straight single tributary extending medially to the subcutaneous tissue. The postoperative course was uneventful. 2.6. Case 4 A 3-year-old male came to the hospital with a congenital fistula at the ascending helix crus and inferior protruding soft tissue

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 fistula and multiple skin tags at the anterior direction of tragus

Fig. 3. In case 3, preoperative photograph showing a congenital fistula (arrow) at the preauricular area with several skin tags in a 2-year-old girl.

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(Fig. 4A). He had a history of repeated discharge and swelling from the fistula. His case was treated as a variant preauricular sinus, and the fistula tract was excised using only an elliptical incision around the fistula, and the soft tissue underneath the ascending helix crus resolved (Fig. 4B). The wound was repaired in a layered fashion.

Table 1 Characteristics of patients according to group.

Patient number (male: female) Ear number Mean age (range) (year)

Classical group

Variant group

159 (47:112) 192 1–76 (18.8)

19 (6:13) 20 2 45 (16.7)

3. Results The classical group consisted of 159 subjects (47 males, 112 females) ranging in age from 1 to 76 years (mean, 18.8 years; Table 1). Of them, 33 patients received simultaneous excision of bilateral preauricular sinuses. The variant group consisted of 19 subjects (6 males, 13 females) ranging in age from 2 to 45 years (mean, 16.7 years; Table 1). One patient in the variant group underwent simultaneous excision of bilateral congenital sinuses. There was only one subject (representing case 3) with associated auricular anomaly, which was skin tags at preauricular area. No subject showed other head and neck anomaly, including branchio-oto-renal syndrome. Familial occurrence of a congenital periauricular fistula was investigated only in the variant group, and two subjects had parents (one father and one mother) with a classical preauricular sinus. Hearing was evaluated preoperatively in 50 subjects in the classical group and eight in the variant group. The audiometry identified five patients with sensorineural hearing loss, which was considered presbycusis because all of them were older than 60 years. No case had a conductive hearing loss. In the variant group, fistula locations were ascending helix crus (15 ears, including Case 4), infra-auricular area (3 ears, including Case 1), supra-auricular area (1 ear, Case 2), and anterior to tragus (1 ear, Case 3). In ears with fistulas in the ascending helix crus, the fistulous tract direction was posteroinferior (5 ears), medial (9 ears), and inferior (1 ear, Case 4). Also, the mean length of fistulous tract was 1.82 cm for the posteroinferior directed tract, 0.67 cm for the medial directed tract, and 0.6 cm for the inferior directed tract (Table 2).

Nine ears had received previous surgery in the classical group, but there were none in the variant group. Forty-five ears in the classical group had a history of incision and drainage along with 2 ears in the variant group. In the variant group, there were 4 ears with fistulous tracts running toward the postauricular area, which required additional retroauricular incision. However, no ears in the classical group required retroauricular incision. After surgery, each group included one ear with postoperative hematoma, which was controlled using conservative methods. Recurrence was observed in 3 ears in the classical group and none in the variant group (Table 3). 4. Discussion Preauricular sinus has an estimated incidence of 0.1–0.9% in the United States and Europe, 2.5% in Taiwan, 4–10% in some African countries, and 2.53% in Korea [4,10–13]. However, the incidence of variant-type preauricular sinus is unknown. There have been only two previous studies reporting more than 10 ears with a unusual location of fistula of preauricular sinus [1,6]. In the present study, we collected information on 20 ears with non-classical fistula and describe the clinical course and surgical approach. We authors should believe that the term ‘variant of preauricular sinus’ seems appropriate because the congenital periauricular fistulas and fistula tracts in all cases of this study were related to the auricle anatomically, were consistently lateral to the temporalis fascia and parotid gland, and did not violate the facial nerve or external 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.

Please cite this article in press as: J.-R. Kim, et al., Congenital periauricular fistulas: Possible variants of the preauricular sinus, Int. J. Pediatr. Otorhinolaryngol. (2014), http://dx.doi.org/10.1016/j.ijporl.2014.08.005

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PEDOT-7234; No. of Pages 6 J.-R. Kim et al. / International Journal of Pediatric Otorhinolaryngology xxx (2014) xxx–xxx Table 2 Subdivision of the variant group according to fistula location. Fistula location

Ear number

Tract direction (No. of ears)/ the mean length (cm)

Above ascending Helix crus

15

Infra-auricular

3

Supra-auricular Anterior to tragus

1 1

Posterior–inferior (5)/1.82 Medial (9)/0.67 Inferior (1)/0.6 Transverse (1)/4.9 Superior (2)/1.45 Inferior (1)/1.6 Medial (1)/0.6

Table 3 Clinical parameters of ears according to group.

Previous operation History of incision and drainage Required postauricular incision Posteriorly directed tract Postoperative hematoma Recurrence

Classical group

Variant group

9 (4.7%) 45 (23.4%) 0 (0%) 0 (0%) 1 (0.5%) 3 (1.6%)

0 2 4 4 1 0

(0%) (10%) (20%) (20%) (5%) (0%)

another diagnosis besides preauricular sinus in these cases. In addition, it would be necessary to perform fistulography preoperatively as in Case 1 or to monitor the facial nerve during the operation if branchial cleft anomalies were suspected. Choi et al. [6] reported that the variant type of preauricular sinus comprised 10.9% (11/101) of preauricular sinus, that all fistulas were located on the ascending helix crus, and that the fistulous tract was directed posteroinferiorly. In this study, the 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 crus, the direction of fistulous tract directed posteroinferiorly in only one third of ears (5/15), for which the majority (4/5) needed retroauricular incision. The authors are not certain of the reason for the difference in direction of fistulous tract with on the ascending helix crus between this and the previous study [6]. During the surgery for preauricular sinus, there are several factors which are known to reduce recurrence. They include 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 space, and removal of the connective tissues around the tract. Especially, the connective tissues around the tract contain some elements which would cause draining from the previous tract [9]. Radical removal of them could minimize the recurrence after primary operation. It might have been helpful if preoperative audiometry and familial occurrence were assessed in all subjects. This is a limitation of this study, as of other retrospective and multicenter studies. Further, a greater number of cases would facilitate determination of the exact manifestations of the variants. In another previous report [1], 14 ears showed the variant type of preauricular sinus in the study group (14/206, 6.8%), the locations of which were superior to the auricle, along the ascending helix crus, at the lobule, and posterior to the auricle without a description of the tract length or direction. The locations of the fistulas were similar to ours, which could assist diagnosis of a variant preauricular sinus. There have been several theories regarding auricle development and the fate of the six hillocks of His [4,14,15]. Among them,

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Davis assumed that the auricle developed from the six hillocks in a rather precise manner, with hillock 2 producing the tragus, hillock 3 the anterior helix, hillock 4 the superior helix and corresponding antihelical portion, hillock 5 the posterior helix and corresponding antihelical portion, and hillocks 6 and 1 the lobule [4]. Park deduced that the region supplied by the superficial temporal artery is compatible with the mandibular hillock region and the region supplied by the posterior auricular artery is compatible with the hyoid hillock region [16]. Park also concluded that hillocks 1 and 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 1, was from the intertragic notch to the middle of the free lateral border of the lobule. This finding is consistent with the proposals of Davis and Park, and could be explained by insufficient fusion of hillocks 1 and 6. In this manner, the fate of the six hillocks of His would be precisely predicted by the clinical manifestations of the variant preauricular sinus. 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 and parotid gland as well as not associated with external auditory canal and facial nerve. The fistula locations could be anywhere around the auricle. Among them, the most common variant was a fistula on the ascending helix crus with a relatively short fistulous tract projecting in a medial direction. During diagnosis of a congenital lesion around the auricle, the possibility of variant preauricular sinus should always be considered and fistulogram would be helpful during preoperative evaluation. Acknowledgments This work was supported by a grant from Research year of Inje University (Grant number: 20140032) in 2014. The English in this document has been checked by at least two professional editors, both native speakers of English. For a certificate, please see: http://www.textcheck.com/certificate/ BYEgCJ. References [1] S.W. Yeo, B.C. Jun, S.N. Park, J.H. Lee, C.E. Song, D.H. Lee, The preauricular sinus: factors contributing to recurrence after surgery, Am. J. Otolaryngol. 27 (2006) 396–400. [2] H.C. Lam, G. Soo, P.J. Wormald, C.A. Van Hasselt, Excision of the preauricular sinus: a comparison of two surgical techniques, Laryngoscope 111 (2001) 317–319. [3] A.G. Hunter, T. Yotsuyanagi, The external ear: more attention to detail may aid syndrome diagnosis and contribute answers to embryological questions, Am. J. Med. Genet. A 135 (3) (2005) 237–250. [4] T. Tan, H. Constantinides, T.E. Mitchell, The preauricular sinus: a review of its aetiology, clinical presentation and management, Int. J. Pediatr. Otorhinolaryngol. 69 (2005) 1469–1474. [5] R.G. Chami, J. Apesos, Treatment of asymptomatic preauricular sinuses: challenging conventional wisdom, Ann. Plast. Surg. 23 (1989) 406–411. [6] S.J. Choi, Y.H. Choung, K. Park, J. Bae, H.Y. Park, The variant type of preauricular sinus: postauricular sinus, Laryngoscope 117 (2007) 1798–1802. [7] S. Minkowitz, F. Minkowitz, Congenital aural sinuses, Surg. Gynecol. Obstet. 118 (1964) 801–806. [8] P.H. Chang, C.M. Wu, An insidious preauricular sinus presenting as an infected postauricular cyst, Int. J. Clin. Pract. 59 (2005) 370–372. [9] K.W. Heo, M.J. Baek, S.K. Park, Pressure dressing after excision of preauricular sinus: suture transfixion of silicone sheets, J. Laryngol. Otol. 123 (2009) 1367– 1370. [10] V. Meggyessy, K. Mehes, Preauricular pits in Hungary: epidemiologic and clinical observations, J. Craniofac. Genet. Dev. Biol. 2 (1982) 215–218.

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Congenital periauricular fistulas: possible variants of the preauricular sinus.

Although most preauricular sinuses are located near the anterior limb of the ascending helix, some are located in unusual areas around the auricle, as...
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