Journal of Otology & RhinologyISSN: 2324-8785

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Case Report, J Otol Rhinol Vol: 4 Issue: 1

Pilonidal Sinus of Nasal Dorsum: A Common Disease at Uncommon Site

Sagar S Gaurkar1*, Prasad T Deshmukh2, Shraddha Jain2 and Minal S Gupta3
1Assistant Professor, Dept of ENT, JNMC, Sawang(m), Wardha, India
2Professor, Dept of ENT, JNMC, Sawang(m), Wardha, India
3Ressident, Dept of ENT, JNMC, Sawang(m), Wardha, India
*Corresponding author : Dr. Sagar M Gaurkar
29 Sagar, Snehal Nagar, Ward No. 7, Wardha, Maharashtra 442001, India
E-mail: [email protected]
Received: April 26, 2014 Accepted: November 05, 2014 Published: January 07, 2015
Citation: Gaurkar SS, Deshmukh PT, Jain S, Gupta MS (2015) Pilonidal Sinus of Nasal Dorsum: A Common Disease at Uncommon Site. J Otol Rhinol 4:1. doi:10.4172/2324-8785.1000202

Abstract

Pilonidal Sinus of Nasal Dorsum: A Common Disease at Uncommon Site

Though most commonly found in sacrococcygeal region, pilonidal sinus, it can also be seen in rare localizations like umbilicus, forehead, scalp, clitoris interdigital area, penis, abdomen, neck and axilla. Occurrence of this lesion in the nasal pyramid and dorsum is all the more uncommon.

A 45 year old male, a known hypertensive presented to ENT OPD of AVBRH with complaints of swelling over the nasal dorsum since birth. Local examination revealed a deformed external nasal pyramid with a swelling located in midline over the nose at junction of bony cartilaginous dorsum. A sinus opening was seen over the swelling with hair coming out through it. The lesion was reported histopathologically as a pilonidal sinus.

Keywords: Pilonidal Sinus; Nasal Dorsum

Introduction

Pilonidal sinus was first reported by Anderson et al. [1] in 1847 as a hair detected in sacrococcygeal ulcer. This entity was defined and christened, as pilonidal sinus, for the first time by Hodges et al. [2] etymologically derived from Latin Pilus-Hair, Nidus –nest. Generally this lesion arises in midline in a skin dimple in relation to tip of the coccyx. Commonly seen in young males, condition is hypothesized to be acquired chronic inflammatory condition surfacing as pilonidal cyst or sinus, due mainly to hair trapped beneath the area [3].
Though most commonly found in sacrococcygeal region, it can also be seen in rare localizations like umbilicus [4], forehead [5], scalp [6], clitoris [7] interdigital area [8], penis, abdomen, neck and axilla [9-12]. Occurrence of this lesion in the nasal pyramid and dorsum is all the more uncommon. Apart from being rare, it creates a very ticklish situation wherein differentiation between nasal dermoid and pilonidal sinus become difficult [13]. Here we are reporting a case of pilonidal sinus at the nasal dorsum principally for its rarity.

Case Report

A 45 year old male, a known hypertensive presented to ENT OPD of AVBRH with complaints of swelling over the nasal dorsum since birth. The swelling did not produce any symptom till the age of 10 years when he sustained trauma to nose. Following this, discharge and extrusion of hairs from the sinus opening ensued. As fallout of the same trauma, he had two major episodes of infection over the nasal dorsum. Trauma also led to formation of pit on midline of dorsum of nose with increase in number and length of hair extruding from the sinus opening. Patient used to frequently trim the hair to avoid social embarrassment. There was no previous history of any surgical intervention.
Local examination revealed a deformed external nasal pyramid with a swelling located in midline over the nose at junction of bony cartilaginous dorsum. A sinus opening was seen over the swelling with hair coming out through it. Swelling was non-tender with a depression present over it. Nose examination including anterior rhinoscopy was normal so also the X-ray of nasal bone. CT Sinogram revealed small sinus tract of 4-5 mm on inferior aspect of nasal ridge communicating with the external surface with no intracranial extensions. Other general investigations were unremarkable.
The lesion was excised with an elliptical incision around the depression to include the opening of sinus. Intraoperatively, we found the tract branching into two with one going towards the nasal bone superiorly and the other moving inferiorly 1 cm above the nasal tip. Nasal bones were found to be normal. The pit was lined by a sac filled with loose, fragmented hairs. Subcutaneous sutures were given over the nasal dorsum for better aesthetic outcome. The wound healed uneventfully (Figure 1).
Figure 1: Elliptical incision around the depression including the opening of sinus.
Histopathological examination of the tissue revealed a sinus tract extending into the underlying dermis. The tract was lined by keratinized stratified squamous epithelium and filled with loose, fragmented hair shafts. No dermal adnexae were seen with evidence of sweat glands, hair follicles and sebaceous glands. The lesion was reported histopathologically as a pilonidal sinus (Figure 2).
Figure 2: Histopathological examination of the tissue.

Discussion

Three cases of pilonidal sinus over the nasal dorsum have been reported so far in the world literature [14,15]. We are reporting this otherwise common entity at rare site.
In the beginning congenital theory has been suggested for the pathogenesis of pilonidal sinus but afterwards acquired theory gained roots and now firmly accepted by most of the surgeon .In the sacrococcygeal region early reports favoured congenital origin but it was unequivocally rejected by clinicians in 2nd half of 20th century. Congenital theory was advocated for some of the cases reported at nasal dorsum. Implantation of hair in diseased or surgically traumatized skin has also been described [13].
This lesion has a male preponderance [13,16] and typically seen in hirsute white males. Blendes [13] and Black population are less prone to development of the lesion due to fine hair in former and curly hair in later [16]. Generally pilonidal sinus appears after puberty because there was no stiff body hair till then. Mayo et al. [4] indicated that the hair may curl back on themselves and pierces the surrounding skin with their distal end first, by growth forces while Oryu et al. felt penetration by shed hair as the beginning point of disease [9].
Consensus has been reached about presence of loose hair being essential for diagnosis of this condition on histopathology but not about the lining of the sinus. In the sacrococcygeal region the sinus is lined by squamous epithelium [13] which may become thin and flat due to episodes of infection [17]. Malignant degeneration of pilonidal sinus is a rare complication occurring in approximately 0.1% of patients with chronic untreated or recurrent pilonidal sinus [18,19]. Among the malignant degeneration of pilonidal sinus most of the cases are squamous cell carcinoma and biological behaviour of malignancy in pilonidal sinus is more aggressive than the squamous cell carcinoma elsewhere.
As far as the pilonidal sinus on the nasal dorsum, a diagnostic dilemma exists between the pilonidal sinus and dermoid sinus cyst. Generally dermoid sinus cyst appear within eighteen months of birth [20] while pilonidal sinus are adult onset in origin .Both have hair protruding through the sinus opening but extrusion of cheesy material is only seen in dermoid cyst [20]. The exclusive and exquisite feature of pilonidal sinus is not being associated with adenxal element and has fragmented hair shaft lying loose within the lumen [21]. Final court of appeal to diagnose this entity being histopathological study.

Conclusion

Pilonidal sinus is not very uncommon disease especially in males and in sacrococcygeal region. However on nasal dorsum, pilonidal sinus is rare and hence we felt it merits reporting. Though preferred site is sacrococcygeal region, one should be alive to the possibility of pilonidal sinus at other location also. Another important attribute of this condition is its potential for malignant transformation. Any hair bearing lesion over the nasal dorsum should prod clinician to investigate for this exotic entity.

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