Pilonidal sinus of atypical areas: presentation and management

Abstract

ABSTRACT
Introduction: Pilonidal sinus (PNS) is an inflammatory disorder due
to hair penetrating the epidermis. It occurs usually in sacrococcygeal
region. Clinical course and management of PNS in atypical
areas has never been addressed in a systematic review.
Methods: Google scholar was used to identify articles describing
pilonidal sinus in any place other than the sacro-coccygeal area.
Results: From 37 papers, there were reports of 302 cases of pilonidal
sinus occurring in various sites other than sacro-coccygeal area. 253
cases (85%) were male. The age of presentation ranged from 16 to
70 years with average of 24.3 years. The most common atypical sites
of pilonidal sinus was umbilicus (91%), followed by hand (4%).
Conclusion: Umbilical PNS is the most common one. Pain, swelling
and discharge is the usual presentation. Apart from umbilical PNS
which is mostly managed conservatively, PNS of atypical sites is
managed by excision and primary repair.

Full text

Pilonidal sinus of atypical areas: presentation and
management
Salih AM1,
Kakamad FH2,3,
Essa RA3,4 ,
Aziz MS3,5,
Salih RQ3,6 ,
Mohammed SH3,6,
Saeed YA4,
Habibullah IJ4,
Muhialdeen AS4,
Othman S3,
Mohameed HA3
REVIEW ARTICLE
Corresponding author: 2F. Hussein
Kakamad, Faculty of Medical Sciences,
School of Medicine, Department
Cardiothoracic and Vascular Surgery,
University of Sulaimani,
François Mitterrand Street,
Sulaymaniyah, Iraq; Email:
fahmi.hussein@univsul.edu.iq
1,3,4,5,6 see pages 12, 13
Reviewers: DS, AW
Date of submission: 28/12/2016
INTRODUCTION
Pilonidal sinus (PNS) is an inflammatory disorder caused by hair penetrating the epidermis.
Granulation tissue lines a blind tract, which ends in a cavity containing pus (1). It is clinically
manifested by a classical inflammatory pattern: local pain, heat, tenderness and erythema (2). It
may present with purulent discharge (3). It is commonly identified in drivers involving the sacro-
coccygeal region (4).
Several methods have been proposed and practiced for management of PNS including
removing the hair and cotton-like dirt in an outpatient fashion and excision with or without
ABSTRACT
Introduction: Pilonidal sinus (PNS) is an inflammatory disorder due
to hair penetrating the epidermis. It occurs usually in sacro-
coccygeal region. Clinical course and management of PNS in atypical
areas has never been addressed in a systematic review.
Methods: Google scholar was used to identify articles describing
pilonidal sinus in any place other than the sacro-coccygeal area.
Results: From 37 papers, there were reports of 302 cases of pilonidal
sinus occurring in various sites other than sacro-coccygeal area. 253
cases (85%) were male. The age of presentation ranged from 16 to
70 years with average of 24.3 years. The most common atypical sites
of pilonidal sinus was umbilicus (91%), followed by hand (4%).
Conclusion: Umbilical PNS is the most common one. Pain, swelling
and discharge is the usual presentation. Apart from umbilical PNS
which is mostly managed conservatively, PNS of atypical sites is
managed by excision and primary repair.
Keywords: pilonidal sinus, unusual area, atypical area, umbilical,
scalp.
DOI:

Pilonidal Sinus Journal (2017) 3(1), 8 - 15
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PSJ (2017) 3(1): 8-15.
9
reconstruction. Both primary closure and allowing for secondary healing with daily dressing are
reported (5-7). General, regional or local anesthesia can be used (7). PNS of atypical areas
started to be increasingly reported in literature in form of case reports and small case series (5-
8).
Atypical areas include groin, interdigital web, umbilicus, nose, inter mammary areas, supra-pubic
area, clitoris, prepuce, penis, occiput, and on the feet. Clinical course and management of PNS in
atypical area has never been addressed in a systematic review. The current study highlights the
presentation and management of pilonidal sinus in atypical areas.
METHODS
Google Scholar engine was searched for the key words (pilonidal sinus of unusual area, pilonidal
sinus of atypical area and pilonidal sinus case report). The search found 155 items. From which,
40 papers were excluded by title because either they were duplicate or unrelated to the subject.
From the remaining 115, 75 articles excluded because they did not fulfill the inclusion criteria.
The inclusion criteria were any article which mentioned presentation and management provided
that the pilonidal sinus occurred in any place other than the sacro-coccygeal area. The data were
re-analyzed and presented in tables for some variables and others are presented without re-
analysis.
RESULTS
From 40 papers, 302 cases of pilonidal sinus were reported occurring in sites other than sacro-
coccygeal area. From which, 255 cases (84%) were male. The age of presentation ranged from 16
to 70 years with an average of 24.3 years. The most common atypical site of pilonidal sinus was
umbilicus (272 cases; 90%) followed by hand (12 cases; 4%). Table 1 shows the reported atypical
sites of pilonidal sinus with their frequency.

Pilonidal Sinus Journal (2017) 3(1), 8 - 15
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PSJ (2017) 3(1): 8-15.
10
References
%
No. of cases
Site
3-5,9-17
90.0
272
umblicus
6,7,18-24
3.9
12
hand
2,25,26
1.7
5
scalp
27-29
1.0
3
intermammary
1,30-32
1.3
4
perianal
33,34
0.7
2
face
35
0.3
1
periareola
36
0.3
1
penis
37
0.3
1
clitoris
38
0.3
1
prepuce
100
302
Total
Tale 1: Atypical sites of pilonidal sinus.
The most common presentation of umbilical pilonidal sinus was discharge, swelling and pain
(85%) followed by only discharge (11.8%) associated with bleeding (2.3%). Removal of hair with
daily dressing was the most frequent treatment modality (73.5%) which was performed without
anesthesia. Otherwise umbilectomy under general anesthesia was associated with a recurrence
rate of 2%.
Discharging sinus was the most common presentation of the interdigital and hand pilonidal
sinuses (90%) which were most commonly treated by surgical excision under general anesthesia
(80%). Scalp pilonidal sinus presented with subcutaneous nodules in 4 cases (80%). All were
treated by excision but one patient needed craniotomy.
Intermammary PNS presented with pain and discharge typically treated by excision and primary
repair. Perineal PNS presented with pain on defecation with discharge, treated with drainage
and left for secondary intention. Penile PNS presented with abscess formation treated by
excision and primary closure. Clitoris PNS presented with swelling and pain treated with excision
and primary closure. Prepuce PNS presented with swelling and discharge treated with excision
and primary closure.
DISCUSSION
Eryilmaz et al. identified the following risk factors for umbilical PNS: young age, male sex,
hairiness, and poor personal hygiene. The same team highlighted the most common presenting
symptoms as purulent discharge, umbilical mass, bloody discharge, and pain with frequency of
23%, 26% 69%, 100%, respectively (11). The current review found that discharge, swelling and

Pilonidal Sinus Journal (2017) 3(1), 8 - 15
Cite this Article as: Salih A, Kakamad F, Essa R. et al. "Pilonidal sinus of atypical areas: presentation and management".
PSJ (2017) 3(1): 8-15.
11
pain occur in 85%, only discharge in 11%, and bloody discharge in 2.3% of cases with umbilical
PNS. The key difference between umbilical PNS and sacro-coccygeal PNS is the method of
management.
In sacro-coccygeal PNS, conservative management has minimal role and the condition requires
excision (drainage if there is collection) with either off midline primary closure or daily dressing
having left the wound for the secondary healing (1). However, umbilical PNS can be successfully
treated by conservative measures provided that the hair is removed and daily dressings applied
(3-5,9-17). Removal of hair in umbilical PNS does not even need local anesthesia (3-5,8-17).
Sacro-coccygeal PNS is a recurring condition with recurrence rates of 15% being often reported,
while PNS in atypical areas (including umbilical) has lower recurrence rate as low as 2%
(11,21,33).
Six cases of umbilical PNS were reported to be recurrent. 4 cases were treated conservatively
(2% recurrence) and 2 cases (3% recurrence) underwent surgery (10,30). This suggests that both
conservative and surgical modalities may have the same rate of recurrence – although this
conclusion is strongly rejected by some (11). A randomized controlled trial with high statistical
power is necessary to overcome this controversy.
While dealing with umbilical PNS, other pathologies such as Sister Joseph nodule, umbilical
hernia, endometriosis, urachal cyst, epidermoid cyst and pyogenic granuloma should not be
neglected as the differential diagnosis. Preoperative imaging may help in this regard (9).
Barber's disease is another name for PNS of the hand (6). While sacro-coccygeal PNS occurs
often in drivers, Barber’s disease occurs in hair dressers, sheep shearers, dog groomers and cow
milkers (6). It is reported more commonly among male hair dressers (6). Higher incidence of
Barber's disease among male gender might be attributed to more frequent female obsessive
personality regarding cleansing of interdigital webs (6) or that most barbers are male.
Scalp PNS is reported in teenagers and children ranging between 1 to 20 years with average 10
years of age. This differentiates scalp PNS from other types of PNS which tend to occur in older
adults (average 23 years old). Excision under local anesthesia is the most common modality of
treatment with no report of recurrences (2,25,26). Borges et al reported a scalp pilonidal sinus in
a 20-year-old male with intracranial extension which needed craniotomy under general
anesthesia. This may alert doctors to investigate scalp PNS before intervention searching for
intracranial extension. Plain antero-posterior skull x-ray may be the first investigation, followed
by brain Computed Tomography (2).
Perianal PNS occurs in adults older than typical PNS (average 34 years old) (31). Excision under
regional anesthesia is the usual therapeutic approach (1,29,30). Doll et al reported a 61-year-old

Pilonidal Sinus Journal (2017) 3(1), 8 - 15
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PSJ (2017) 3(1): 8-15.
12
lady presenting with intermittent anal pain for 10 years due to endoanal PNS treated by opening
the cavity and leaving for healing by secondary intension (32).
Intermammary PNS presented with pain and discharge treated by excision and primary repair.
Perineal PNS presented with pain on defecation with discharge, treated with drainage and was
left for healing by secondary intention. Penile PNS presented with abscess formation treated by
excision and primary closure. Clitoris PNS presented with swelling and pain - treated with
excision and primary closure. Prepuce PNS presented with swelling and discharge - treated with
excision and primary closure (29,36-38).
As expected, PNS of atypical areas is a rare presentation. Umbilical PNS is the most common -
pain, swelling and discharge is the usual presentation. Apart from umbilical PNS which is mostly
managed conservatively, PNS of atypical sites is managed by excision and primary repair.
ACKNOWLEDGMENTS
N/A
MULTIMEDIA
N/A
CONTRIBUTION AND AFFILIATION
Salih AM1,
Kakamad FH2,3,
Essa RA3,4 ,
Aziz MS3,5,
Salih RQ3,6 ,
Mohammed SH3,6,
Saeed YA4,
Habibullah IJ4,
Muhialdeen AS4,
Othman S3,
Mohameed HA3
1. Faculty of Medical Sciences, School of Medicine, Department Surgery, University of Sulaimani,
François Mitterrand Street, Sulaimani, Kurdistan Region, Iraq

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2. Faculty of Medical Sciences, School of Medicine, Department Cardiothoracic and Vascular
Surgery, University of Sulaimani, François Mitterrand Street, Sulaimani, Kurdistan Region, Iraq
3. Bioscience Center, Goran Street, Sulaimani, Kurdistan region, Iraq
4. Sulaimani Teaching Hospital, Sulaimani, Kurdistan region, Iraq
5. Daik laboratory, Goran Street, Sulaimani, Kurdistan region, Iraq
6. University of Sulaimani, Faculty of Science & Science Education, School of Science, Biology
Department, Raperin Street, Sulaimani, Kurdistan region, Iraq
Salih, Kakamad and Essa: substantial contribution to the concept and design, drafting the
manuscript and final revision and approval of the manuscript.
Aziz, Salih, Mohammed and Saeed: literature review, drafting the manuscript and final revision
and approval of the manuscript.
Habibullah, Muhialdeen, Othman and Mohameed: extraction of data, analysis of data, drafting
the manuscript and final revision and approval of the manuscript.
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