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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 28  |  Issue : 3  |  Page : 258-261

Hair in the wrong place: A rare case of pilonidal sinus in preauricular sinus tract


Department of ENT, Chettinad Hospital and Research Institute, Kelambakkam, Tamil Nadu, India

Date of Submission28-Jun-2022
Date of Decision07-Jul-2022
Date of Acceptance12-Aug-2022
Date of Web Publication21-Nov-2022

Correspondence Address:
Dr. Priya Kanagamuthu
Department of ENT, Chettinad Academy of Research and Education, Chettinad Hospital and Research Institute, Kelambakkam - 603 103, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/indianjotol.indianjotol_106_22

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  Abstract 


Introduction: The preauricular sinus is a congenital abnormality which was initially identified by Van Heusinger in 1864. Preauricular sinus development is strongly related to the development of auricle in the sixth week of pregnancy. Preauricular sinus can be inherited or are sporadic. Most cases are sporadic and over half of all cases are unilateral. Bilateral cases are more frequent in inherited cases. Case Report: A 10 year old boy was brought with complaints of discharge from left pre auricular region on and off since birth. History of frequent episodes of discharge from left pre auricular region about 2-3 episodes per year. Local examination showed preauricular sinus tract at the root of helix on both sides. There was no discharge,no warmth, no tenderness. The child underwent necessary investigations and pre aneasthetic fitness was obtained. Pre auricular sinus tract excision on left side under general anasthesia was done. Multiple sinus tracts were excised and sent for histopathological examination which showed evidence of pilonidal sinus. There was presence of sinus tract line by epidermis, lumen filled with keratin and hair shafts. The child was brought for follow up on postoperative day 10, the sutured site was found to be healthy. Suture removal was done and the wound was healing well. The child is on regular follow up and has shown no signs of any recurrence. Discussion: The only definite treatment of recurrent infective sinuses is complete excision of the sinus tract. In our case, supra aural elliptical incision was placed and all visible sinus tracts were cleared and sent for histopathological analysis. When these sinuses get infected, most frequent cause being with gram-positive bacteria, the exudates should be sent for culture, and the appropriate antibiotics should be administered. The temporalis fascia, the cartilage of the helix, and the auditory canal are identified using the supra-auricular approach. An en bloc excision of the sinus is then carried out. Pilonidal sinus is a difficult condition that causes suffering and embarrassment to its victims. Despite it being rare and atypical, pilonidal sinus could also occur in the auricular region. Although pilonidal sinus has been reported in a number of uncommon areas of the body besides the sacrococcygeal region, this is the second case of pilonidal sinus that we are aware of in the preauricular region that is reported in literature. Conclusion: Thus we present a very rare case of pilonidal sinus in pre auricular region. Only two cases have been reported till date in literature and our case is the second one to be reported in the pre auricular region.

Keywords: Hair follicle, preauricular sinus, tract


How to cite this article:
Anand R, Kanagamuthu P. Hair in the wrong place: A rare case of pilonidal sinus in preauricular sinus tract. Indian J Otol 2022;28:258-61

How to cite this URL:
Anand R, Kanagamuthu P. Hair in the wrong place: A rare case of pilonidal sinus in preauricular sinus tract. Indian J Otol [serial online] 2022 [cited 2022 Dec 6];28:258-61. Available from: https://www.indianjotol.org/text.asp?2022/28/3/258/361634




  Introduction Top


The preauricular sinus is a congenital abnormality which was initially identified by Van Heusinger in 1864. Preauricular sinus development is strongly related to the development of auricle in the 6th week of pregnancy. Six mesenchymal proliferations make up hillocks of His, three of which emerge from the first branchial arch from the caudal border and three from the cephalic border of the second branchial arch. These hillocks fuse together to form the complete auricle.[1]

The preauricular sinus can be inherited or sporadic. Most cases are sporadic, and over half of all cases are unilateral.[2] Bilateral cases are more frequent in inherited cases. Incomplete autosomal dominance with poor penetrance is the pattern of inheritance. In 3%–10% of cases, preauricular sinuses are symptoms of various illnesses or syndromes, most commonly in connection with deafness and the branchio-oto-renal (BOR) syndrome. Recent research has mapped a potential locus for congenital preauricular fistula to chromosome 8q11.1–q13.3.13. Although the true prevalence is not well established, preauricular sinuses are thought to occur most commonly in Afro-American populations. The incidence of preauricular sinuses has been estimated to be 0.1%–0.9% in the United States, 0.9% in England, 1.6%–2.5% in Taiwan, and 4%–10% in some areas of Africa.[3],[4],[5]


  Case Report Top


A 10-year-old boy was brought with complaints of discharge from the left preauricular region on and off since birth. There was a history of frequent episodes of discharge from the left preauricular region about 2–3 episodes per year. Discharge was yellow in color, was serous in nature. Discharge did not contain any blood and was not associated with any foul smell. Discharge from the pre auricular sinus was aggravated with episodes of upper respiratory tract infection. There was a history of frequent episodes of upper respiratory tract infection. There were no other significant complaints. An antenatal, postnatal, and development history was normal.

On examination, the child was conscious oriented and comfortable afebrile. Systemic examination was normal. Local examination showed a preauricular sinus tract at the root of the helix on both sides [Figure 1] and [Figure 2]. There was no discharge, no warmth, and no tenderness.
Figure 1: Preauricular sinus tract on the left root of the helix

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Figure 2: Preauricular sinus tract on the right root of the helix

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The child underwent necessary investigations, and preanesthetic fitness was obtained. Preauricular sinus tract excision on the left side under general anesthesia was done. Multiple sinus tracts were excised and sent for histopathological examination [Figure 3]. The incision site was sutured with nonabsorbable 3-0 Ethilon, and the child was treated with a course of oral and topical antibiotics and analgesics. Sterile dressing was applied.
Figure 3: Intraoperative picture showing preauricular sinus tract

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Histopathology

The histopathology report showed pilonidal sinus. According to the histopathology report, there was presence of a sinus tract that was lined by epidermis. The tract lumen was filled with keratin and hair shafts. There was surrounding chronic inflammatory changes and presence of hyaline cartilage as shown in the histopathological slide pictures [Figure 4] and [Figure 5].
Figure 4: Low-power microscopic picture of the hair shaft in the sinus

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Figure 5: The presence of hair shafts (blue arrows)

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The child was brought for follow-up on postoperative day 10 [Figure 6], and the sutured site was found to be healthy. Suture removal was done, and the wound was healing well. The child is on regular follow-up and has shown no signs of any recurrence.
Figure 6: Postoperative picture showing the healing scar

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  Discussion Top


Embryology

In the 4th week of fetal development, six pairs of branchial arches form from ectoderm, mesoderm, and endoderm [Figure 7]. The inner pharyngeal pouch lined by endothelium and the outer branchial cleft lined by ectoderm are separated from each other by a two-layered membrane. Each arch gives rise to muscle, skin, and cartilage and is innervated by a cranial nerve. The middle ear,  Eustachian tube More Details, and mastoid antrum develop from the first pharyngeal pouch. The tympanic membrane arises from the closing membrane. Many theories have been put forward to explain the abnormal developmental process of pinna resulting in a congenital preauricular sinus. Only the first branchial cleft persists as the external ear canal. All other clefts get resorbed.
Figure 7: The various stages of embryological development of the pinna (a-e)

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Mesoderm and ectoderm from the first and second branchial arches and first branchial cleft merge to develop into the pinna. The six hillocks of His are formed by mesenchymal proliferation during the 6th week of embryonic development. Three hillocks combine on the caudal aspect of the first branchial arch, and three on the cephalic aspect of the second branchial arch. Eventually, the hillocks develop and unite to form the pinna.[6],[7]

Pathogenesis

The preauricular sinus could be caused by a defective closure of the dorsal portion of the first branchial cleft, or an inadequate fusion of the first arch hillocks, or an entrapment of ectodermal folds during auricular development. One of these may result in a congenital preauricular sinus.[8],[9],[10] The cutaneous opening of the preauricular sinus is typically found on or close to the ascending limb of the helical rim and tragus [Figure 8]. It can also open along the superior posterior helical margin. Congdon et al. suggested clinical classification according to the sites of orifice of preauricular sinus by incomplete fusion theory.[11],[12] A few syndromes, namely the BOR syndrome, Beckwith–Wiedemann syndrome,  Treacher Collins syndrome More Details, and hemicranial microsomia syndrome, have also been linked to the development of the preauricular sinus.[13]
Figure 8: The various types of auricular sinuses based on their position

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Treatment

When other congenital anomalies coexist with these sinuses, auditory testing and renal ultrasound should be considered. When these sinuses get infected, the most frequent cause being with Gram-positive bacteria, the exudates should be sent for culture, and the appropriate antibiotics should be administered. The majority of cases are asymptomatic. However, certain cases may present with facial cellulitis or recurring preauricular abscesses, which would necessitate antibiotic therapy against the causative organism and incision and drainage. Surgical excision is indicated when there is no active discharge.[13] The only definite treatment of recurrent infective sinuses is complete excision of the sinus tract.[14] In our case, supra-aural elliptical incision was placed and all visible sinus tracts were cleared and sent for histopathological analysis.

The most common cause of recurrence is simple excision without magnification. The use of probes, however, can cause minor tracts to go unnoticed and occasionally lead to false routes. Methylene blue dye instillation frequently leads to diffuse discoloration of the surgical field and the neglect of smaller areas. In the presence of clogged tract, the dye may not penetrate inside.[15],[16],[17] The temporalis fascia, the cartilage of the helix, and the auditory canal are identified using the supra-auricular approach. An en bloc excision of the sinus is then carried out. Less than 5% of occurrences have been reported. Some studies suggest that including a piece of cartilage like the root of the helix, tragus, or auditory canal in the resection obtains superior outcomes.[18],[19]

Pilonidal sinus is a persistent inflammatory disorder caused by hair fragments penetrating into the skin. 0.7% of the population are affected by this condition. Pilonidal sinus is a difficult condition that causes suffering and embarrassment to its victims. Despite it being rare and atypical, pilonidal sinus could also occur in the auricular region. Although pilonidal sinus has been reported in a number of uncommon areas of the body besides the sacrococcygeal region, this is the second case of pilonidal sinus that we are aware of in the preauricular region that is reported in the literature.[20]


  Conclusion Top


Thus, we present a very rare case of pilonidal sinus in the preauricular region. Only two cases have been reported till date in the literature, and our case is the second one to be reported in the preauricular region. Histopathological confirmation is necessary for detecting the presence of hair follicle and keratin and for confirming the diagnosis. Management includes complete excision of the tract and regular periodic checkup to ensure that there is no recurrence.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the legal guardian has given consent for images and other clinical information to be reported in the journal. The guardian understands that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Tan T, Constantinides H, Mitchell TE. The preauricular sinus: A review of its aetiology, clinical presentation and management. Int J Pediatr Otorhinolaryngol 2005;69:1469-74.  Back to cited text no. 1
    
2.
Aronsohn RS, Batsakis JG, Rice DH, Work WP. Anomalies of the first branchial cleft. Arch Otolaryngol 1976;102:737-40.  Back to cited text no. 2
    
3.
Aird I. Ear-pit (congenital aural and preauricular fistula). Edinb Med J 1946;53:498-507.  Back to cited text no. 3
    
4.
Ewing MR. Congenital sinuses of the external ear. J Laryngol Otol 1946;61:18-23.  Back to cited text no. 4
    
5.
Tsai FJ, Tsai CH. Birthmarks and congenital skin lesions in Chinese newborns. J Formos Med Assoc 1993;92:838-41.  Back to cited text no. 5
    
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Nofsinger YC, Tom LW, LaRossa D, Wetmore RF, Handler SD. Periauricular cysts and sinuses. Laryngoscope 1997;107:883-7.  Back to cited text no. 6
    
7.
Dunham B, Guttenberg M, Morrison W, Tom L. The histologic relationship of preauricular sinuses to auricular cartilage. Arch Otolaryngol Head Neck Surg 2009;135:1262-5.  Back to cited text no. 7
    
8.
Johnston MC, Pratt RM. A developmental approach to teratology. In: Berry CL, Poswilla DE, editors. Teratology, Trends and Applications. Berlin: Springer-Verlag; 1975.  Back to cited text no. 8
    
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Moore KL. The Developing Human: Clinically Orientated Embryology. Philadelphia: W.B. Saunders; 1973.  Back to cited text no. 9
    
10.
Salih AM, Kakamad FH. Preauricular pilonidal sinus: The first reported case. Int J Case Rep Images 2016;7:162-4.  Back to cited text no. 10
    
11.
Song-Hwan O, So IK, Kim JH. Clinical features of preauricular sinus and recurrence rate of supra-auricular approach. Indian J Otol 2018;24:91-4.  Back to cited text no. 11
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12.
Congdon ED, Rowhanavongse S, Vara-Misara P. Human congenital auricular and Juxta-auricular fossae, sinuses and scars (including the so-called aural and auricular fistulae) and the bearing of their anatomy upon the theories of their genesis. Am J Anat 1932;51:439-63.  Back to cited text no. 12
    
13.
Sharma A, Taneja V, Mehra M, Khandelwal P, Banerjee M. Surgical excision of preauricular sinus. Indian J Otol 2019;25:31.  Back to cited text no. 13
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14.
Scheinfeld NS, Silverberg NB, Weinberg JM, Nozad V. The preauricular sinus: A review of its clinical presentation, treatment, and associations. Pediatr Dermatol 2004;21:191-6.  Back to cited text no. 14
    
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Gur E, Yeung A, Al-Azzawi M, Thomson H. The excised preauricular sinus in 14 years of experience: Is there a problem? Plast Reconstr Surg 1998;102:1405-8.  Back to cited text no. 15
    
16.
Leung AK, Robson WL. Association of preauricular sinuses and renal anomalies. Urology 1992;40:259-61.  Back to cited text no. 16
    
17.
Lam HC, Soo G, Wormald PJ, Van Hasselt CA. Excision of the preauricular sinus: A comparison of two surgical techniques. Laryngoscope 2001;111:317-9.  Back to cited text no. 17
    
18.
Prasad S, Grundfast K, Milmoe G. Management of congenital preauricular pit and sinus tract in children. Laryngoscope 1990;100:320-1.  Back to cited text no. 18
    
19.
Baatenburg de Jong RJ. A new surgical technique for treatment of preauricular sinus. Surgery 2005;137:567-70.  Back to cited text no. 19
    
20.
Salih AM, Hassan SH, Hassan MN, Fatah ML, Kakamad FH, Salih BK, et al. Post auricular pilonidal sinus; a rare case with a brief review of literature. International Journal of Surgery Open 2022:100489.  Back to cited text no. 20
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]



 

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