Unusual presentation of more common disease/injury

CASE REPORT

Pilonidal sinus involving the nasal bridge: a rare manifestation Montasir Junaid,1 Sadaf Qadeer Ahmed,2 Maliha Kazi,2 Naeem Sultan Ali3 1

Department of Otolaryngology, Jinnah Medical and Dental College, Karachi, Pakistan 2 Department of Otolaryngology, Aga Khan University Hospital, Karachi, Pakistan 3 Department of Otolaryngology, Aga Khan University, Dar-ul-Salaam, United Republic of Tanzania Correspondence to Dr Maliha Kazi, [email protected] Accepted 21 June 2015

SUMMARY Pilonidal sinus is very commonly associated with the sacrococcygeal area, but its presence within the head and neck is still unknown to many. Once diagnosed, it is easy to treat and should, therefore, be kept in mind as a possibility when coming across a discharging sinus swelling. We share our experience of two cases of pilonidal sinus presenting over the nasal bridge and their management.

BACKGROUND Pilonidal sinus, a well-known acquired entity, was first described by Herbert Mayo in 1833.1 The term itself was coined by Hodge in 1880, and is derived from the Latin words ‘pilus’ (hair) and ‘nidus’ (nest). Pilonidal sinus describes specific subcutaneous hair-containing lesions occurring most commonly in the sacrococcygeal region and having a tendency to recur.1 2 These lesions mostly occur in people in their late teens to early 20s. Rarely do they involve people over the age of 45 years.3 Although this condition can be considered as one of the most well-known, controversies still prevail regarding its development aetiology. There is a constant debate about whether it is a congenital or an acquired condition. While initial hypotheses favoured the congenital theory, recent evidence proves otherwise. One of the most diagnostic signs for the presence of a pilonidal sinus is the occurrence of free hairs within the sinus tract. This finding, along with others, has led many to believe that pilonidal disease is indeed an acquired condition.3 While the majority occur in the postanal region, pilonidal sinuses have been reported in a variety of atypical sites including the axilla, perineum, suprapubic region, umbilicus, interdigital space and even a mid-thigh amputation stump.4–6 Within the head and neck region, their presence has also been reported on the scalp and ear.7 8 They have, in addition, been reported to present, though rarely, on the nose.9 10 We report two similar atypical cases.

patient underwent surgical removal of the swelling under general anaesthesia in a nearby hospital, 6 months prior to presenting to us. After having remained well for a couple of months, the wound later began discharging purulent material. When the patient presented to us, there were effectively two swellings: one on the nasal bridge and a second near the right medial canthus. There was obvious erythema connecting the two lesions. On deep palpation, both lesions were tender and purulent discharge expelled out from them. Convinced that it was an improperly addressed pilonidal sinus, the patient’s parents were counselled. After informed consent, the patient was scheduled for surgical removal of the sinus.

TREATMENT As there were two openings, two separate incisions were used to address: (1) the sinus on the centre of the nasal bridge and (2) the sinus adjacent to the medial canthus of the right eye (figure 1). The sinus tracts were traced along their pathway, which had multiple hair follicles, found in clusters. Interestingly, the tracts of the two openings combined into one around the left lateral border of the nasal bridge. The sinus was traced along its path and excised. Both incisions were closed with nonabsorbable sutures (figures 2 and 3).

OUTCOME AND FOLLOW-UP The patient was started on Gram-positive coverage antibiotics. She was discharged the next day with antibiotics and reviewed a week later; she had good wound healing and scar formation. The final histopathological report showed squamous epithelium lining the draining sinus along with hair shafts in

CASE PRESENTATION

To cite: Junaid M, Ahmed SQ, Kazi M, et al. BMJ Case Rep Published online: [ please include Day Month Year] doi:10.1136/ bcr-2015-209948

A 13-year-old girl was referred to our department with a history of a discharging sinus on the nasal bridge for the past 9 months. Starting a year back as a small swelling on her nasal bridge, it gradually continued to increase in size. In the following months, it became associated with a thin, mucoid, non-blood stained discharge. The discharge was intermittent and, occasionally, foul smelling. The

Figure 1 Two separate incisions performed to address each opening of the pilonidal sinus.

Junaid M, et al. BMJ Case Rep 2015. doi:10.1136/bcr-2015-209948

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Unusual presentation of more common disease/injury

Figure 4

Figure 2 Complete excision of the sinus after tracing the tract. clusters within the specimen. It was confirmed as pilonidal sinus.

CASE PRESENTATION 2 The case 2 patient was a 24-year-old grocery shop owner. He had a small swelling on his nasal bridge since birth. It was accompanied by intermittent pain and discharge, and was a social embarrassment to the patient. On closer inspection, tufts of hair were seen protruding from the lesion with expiration of clear fluid on deep digital palpation. The diagnosis of pilonidal sinus was considered and after informed consent, the patient was scheduled for surgery.

TREATMENT AND FOLLOW-UP Diagnosed with pilonidal sinus, the patient underwent surgery of the sinus tract under general anaesthesia. The lesion was

Figure 3 Both incisions closed with non-absorbable sutures. 2

Healed incision scar after 3 months.

found to extend up to the nasal bone and to have multiple hair follicles. The superficial cortex of the nasal bridge also had to be curetted to prevent recurrence. The sinus tract was traced and sinus excised. The wound was closed with non-absorbable sutures and the patient was discharged the next day on standard antibiotics with Gram-positive coverage. The histopathological report showed sinuses lined with squamous epithelium and multiple hair follicles were found within the tracts, indicating pilonidal sinus. The patient presented for follow-ups and 3 months later had a well-healed contracted scar (figure 4).

DISCUSSION Pilonidal sinuses typically involve the sacrococcygeal region and plenty has been written about their presence in that region. However, there are reports of their occurrence at other atypical hair-growing sites such as the axilla, umbilicus, anterior perineal region and scalp. Not surprisingly, due to their relation to hair growth, they have also been reported to occur in the interdigital spaces of barbers and animal handlers. As far as the condition’s aetiology is concerned, there is a congenital and an acquired theory for its development. When initially described in the mid-19th century, it was thought to be congenital in nature. Most of the experts favour the acquired theory as the cause of pathogenesis. The acquired theory dictates that pilonidal sinus develops secondary to hormones, trauma, infection and friction.11 12 A general consensus on one favoured aetiological theory has yet to be achieved. In our case series, the first patient had the condition for a year, whereas the second patient stated that the swelling had been present since birth. While there have been controversies regarding the exact aetiological origin, it has been stated that hairs penetrate through the dilated hair follicles, puncturing the intact skin, to stand erect. The negative pressure thus created further leads to hair being sucked into the sinus. Presence of a curved, concaved surface favours the creation of this negative pressure, a fact that leads to a large number of pilonidal sinuses developing in the sacrococcygeal region. With a male predominance, the presenting age is usually between 15 and 24 years. It is most commonly seen in hirsute and white males. The incidence of pilonidal sinus is less in those of African origin and blondes. As there is no stiff hair before puberty, pilonidal sinuses usually form later.2–13 It has been found to be associated with a positive family history, a sedentary occupation and obesity as well as local irritation or trauma.14 Junaid M, et al. BMJ Case Rep 2015. doi:10.1136/bcr-2015-209948

Unusual presentation of more common disease/injury Neither of our patients had any history of trauma at their sites, nor did they shave the region concerned. While many individuals remain asymptomatic, symptomatic occurrences of both acute and chronic duration may be noted. Acutely, the classical presentation involves a painful, swollen, erythematous abscess, which may or may not drain, and can form a secondary tract if drainage to the surface is blocked. On the contrary, the chronic presentation involves a single or branching abscess containing loose hair. This variant is usually due to prolonged neglect of symptoms but can also occur in spite of appropriate treatment.15 Diagnosis of a pilonidal sinus is based primarily on the history and examination of the site. Imaging does not play a vital role in its diagnosis. Treatment options are numerous. Dogru et al16 used crystallised phenol application to treat pilonidal sinus, with a less than 1% recurrence rate within the first year. Use of cauterisation, alcohol injection and simple phenol solution has been mentioned in the literature, but an optimal treatment option has not been formed out of them. Surgical treatment follows the principle of excision and thorough cleansing of hair and debris. Senapati et al17 reported a recurrence-free rate of around 90%, with debridement and a lay open technique. This was also supported by Al-Hassan et al, when he reported a mean healing time of 13 weeks and recurrence rate of 12% using the lay open technique. However, excision of a pilonidal sinus with primary closure has showed an even shorter healing time.18 This treatment procedure is mostly opted for in smaller pilonidal sinus diseases, where primary closure is possible due to approximation of the skin edges without any tension, as was possible in both our patients. Both patients had their wounds closed primarily with minimal scarring on subsequent follow-ups. Nevertheless, the use of various kinds of flaps as part of the surgery has been mentioned time and again. Skin flaps, V-Y flaps and rhomboid flaps have been used to cover the defects created. In 1963, Alexander Alexandrovich Limberg defined a classic rhomboid flap using paper models to cover skin defects. His procedure was first used for pilonidal sinus treatment by Azab et al.19 Bascom et al mentioned using advancement flap as part of surgery in 149 patients. They reported a recurrence rate of 16%.20 More famously, Karidakis described an advancement flap used to repair the wound, which reduced the depth of the wound and reduced hair accumulation, and, in turn, reduced mechanical irritation. His method reported a recurrence rate of 1%.21 However, recurrence rates depend on various factors. Hull and Wu3 state that the presence of hair, undebrided tissue and tension along the suture line are some of the important factors leading to recurrence of the condition. Despite the proposal of various conservative and surgical cures for the condition, an agreement for the optimal cure of the disease is yet to be formulated. Pilonidal sinus disease involving the nose generally requires complete excision followed by primary closure, if small in size. For larger sized defects, an advancement flap is required. However, the treatment option depends on the surgeon’s preference, the anatomical site and the size of the pilonidal sinus, among other factors. Pilonidal sinus, though a common occurrence, is still unknown in the head and neck. Nevertheless, it should be

Junaid M, et al. BMJ Case Rep 2015. doi:10.1136/bcr-2015-209948

included in the differential diagnosis of subcutaneous lesions within the head and neck while looking for a diagnosis. It can prove to be symptomatically frustrating, but once correctly identified, it is easy to manage and treat.

Learning points ▸ Pilonidal sinus in the head and neck is a well-recognised entity. ▸ Knowledge regarding its diagnosis and treatment is vital despite it being a rare condition. ▸ Various surgical options to treat the disease are available, with advancement flaps favoured.

Contributors MJ was the primary surgeon and was involved in the operations and follow-ups. SQA helped in the surgeries and postoperative follow-ups. MK wrote the manuscript for submission. NSA wrote parts of the manuscript and reviewed it. Competing interests None declared. Patient consent Obtained. Provenance and peer review Not commissioned; externally peer reviewed.

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Junaid M, et al. BMJ Case Rep 2015. doi:10.1136/bcr-2015-209948

Pilonidal sinus involving the nasal bridge: a rare manifestation.

Pilonidal sinus is very commonly associated with the sacrococcygeal area, but its presence within the head and neck is still unknown to many. Once dia...
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