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Case Reports and Images in Surgery

 
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Case Report
 
A case report of endoanal pilonidal sinus
Abdulwahid M. Salih1, F. H. Kakamad2
1Faculty of Medical Sciences, School of Medicine/Department Surgery/ University of Sulaimani, François Mitterrand Street, Sulaymaniyah, Iraq.
2Faculty of Medical Sciences, School of Medicine, Department Cardiothoracic and Vascular Surgery, University of Sulaimani, François Mitterrand Street, Sulaymaniya, Iraq.

Article ID: 100024Z12AS2016
doi:10.5348/Z12-2016-24-CR-16

Address correspondence to:
Fahmi H. Kakamad
Faculty of Medical Sciences, School of Medicine/Department Cardiothoracic and Vascular Surgery
University of Sulaimani/ François Mitterrand Street
Sulaymaniyah
Iraq

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How to cite this article:
Salih AM, Kakamad FH. A case report of endoanal pilonidal sinus. J Case Rep Images Surg 2016;2:60–62.


Abstract
Introduction: Pilonidal sinus is a disease in which chronic inflammatory condition occurs due to involution of hair fragments into the skin. Endoanal pilonidal sinus is an extremely rare condition with only 10 cases have been reported in literature. We present a case of endoanal pilonidal sinus with literature review.
Case Report: A 39-year-old male with perianal discharge and itching for six months duration. Clinical examination showed an external opening at 11 o'clock. Fistulectomy was performed. The histopathological examination confirmed endoanal pilonidal sinus.
Conclusion: Although extremely rare, pilonidal sinus may occur in the anal canal, especially in those patients with previous operation. Fistulectomy with open tract is an effective treatment.

Keywords: Endoanal pilonidal sinus, Hair containing sinus, Pilonidal fistula, Pilonidal sinus


Introduction

Pilonidal sinus is a common medical problem that accounts for about 15% of anal suppurations [1]. It is a condition with chronic inflammation associated with the involution of hair into the skin. It occurs commonly in the sacrococcygeal area [2]. However, it may also occur in other areas rarely like umbilicus, nose, suprapubic area, groin, interdigital web, axilla, clitoris, prepuce, or penis [3]. The onset of pilonidal sinus is rare before puberty and beyond the age of forty [4]. It occurs three times more in male than female [4]. It clinically presents as mass, pain, cellulitis and redness [5].

Endoanal pilonidal sinus is an extremely rare condition with only 10 cases have been reported in literatures [6][7][8][9][10][11]. Although previous anal operation accounts for etiology of some cases, its exact cause remains unknown [11]. We present a case of endoanal pilonidal sinus with literature review.


Case Report

A 39-year-old male with history of surgically treated pilonidal sinus and anal fistula before two years, presented with perianal discharge and itching for six months duration associated with pain especially during defecation. Clinical examination showed an external opening at 11 o'clock about 3.5 cm distant from anal verge with indurations and surrounding skin excoriation. Transperineal ultrasonography revealed single fistula tract extending proximally into the anal canal and merging with its wall about 16 millimeters from anal verge. The sonographer concluded intersphincteric, low type, fistula-in-ano. Under spinal anesthesia, fistulectomy was performed, the tract left open. The histopathological examination showed chronic inflammation containing scattered foreign body, multinucleated giant cells centered on a tract which was lined by thick benign stratified squamous epithelium and contained several free hair shafts, features of endoanal pilonidal sinus (Figure 1). After five weeks of follow-up, patient was comfortable and free on symptoms.


Cursor on image to zoom/Click text to open image
Figure 1: Microscopic examination showed chronic inflammation containing scattered foreign body, multinucleated giant cells centered on the tract confirming pilonidal sinus.



Discussion

The origin of pilonidal disease is not well understood. There are two theories for its pathogenesis: congenital and acquired theories. However, the majority of opinion favors the acquired theory [12]. In general, at least three conditions should be present for a pilonidal sinus to develop: First is hair in the skin. Second, some sort of wrinkled skin, like the natal cleft or a scar. The third condition is a mixture of hormonal and hygienic problem [13]. It usually presents as pain, local inflammation and redness [5].

Endoanal pilonidal sinus which was previously called pilonidal fistula, is a pilonidal sinus with an opening into anal canal [14]. There are several ways for sinus to be driven into the anal canal:

  • Spread of sacrococcygeal pilonidal diseases into the perianal area.
  • Less known theories are that the pilonidal cysts reach through the penetration of hairs into an open anal fissures or that hairs can get into the anal canal as they do in other areas of the body.
  • The most updated theory regards the acquired condition after anal operation in which hairs may penetrate the underlining tissues through the healing wound or via developing scar [11].

The main presenting features of the endoanal pilonidal sinus are discharge, inching and pain. Surgical fistulectomy with primary repair is the corner stone of the management. To our knowledge, there are only 10 reported cases in literatures. Nine of them occurred in male and one occurred in a woman. The age range of the patients was 23–58 years. Eight of them presented with recurrent purulent discharge. Two of the cases were symptomless and they were accidentally found during a surgical intervention to manage hemorrhoids. None of the reported patients had found hairs in the anal canal by themselves. As in this case, the sinus was singular in nine of them while other patient had double endoanal sinuses. Regarding the surgical intervention, in five cases the cyst and the tracts were opened and left for secondary intention healing and on the other four cases the lesion was thoroughly removed. Two of the reported cases had the same initial presentation as ours. All three patients were initially surgically diagnosed as cases of anal fistula. While others diagnosed as pilonidal sinus initially. Later on during surgical correction and histopathological examination, they were fount that to be cases of endoanal fistula [6] [7][8][9][10][11].


Conclusion

Although extremely rare, pilonidal sinus may occur in the anal canal. The main risk factor is previous anal operation. Fistulectomy with open tract appears to be a good treatment option for healing with a very low rate of recurrence.


References
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  2. Brebbia G, Boni L, Dionigi G, et al. Surgical site infections in day surgery settings. Surg Infect (Larchmt) 2006;7 Suppl 2:S121–3.   [CrossRef]   [Pubmed]    Back to citation no. 2
  3. Sunkara A, Wagh D, Harode S. Intermammary pilonidal sinus. Int J Trichology 2010 Jul;2(2):116–8.   [CrossRef]   [Pubmed]    Back to citation no. 3
  4. Søndenaa K, Andersen E, Nesvik I, Søreide JA. Patient characteristics and symptoms in chronic pilonidal sinus disease. Int J Colorectal Dis 1995;10(1):39–42.   [CrossRef]   [Pubmed]    Back to citation no. 4
  5. Richardson HC. Intermammary pilonidal sinus. Br J Clin Pract 1994 Jul-Aug;48(4):221–2.   [Pubmed]    Back to citation no. 5
  6. Wilson E, Failes DG, Killingback M. Pilonidal sinuses of the anal canal: report of a case. Dis Colon Rectum 1971 Nov-Dec;14(6):468–70.   [CrossRef]   [Pubmed]    Back to citation no. 6
  7. Weston SD, Irving SS. Pilonidal cyst of the anal canal: Case report. Dis Colon Rectum 1963;6(2):139–41.   [Pubmed]    Back to citation no. 7
  8. Weston SD, Irving SS. Pilonidal cyst of the anal canal: Case report. Dis Colon Rectum 1963;6(2):139–41.   [Pubmed]    Back to citation no. 8
  9. Ortiz H, Marti J, DeMiguel M, Carmona A, Cabañas IP. Hair-containing lesions within the anal canal. Int J Colorectal Dis 1987 Aug;2(3):153–4.   [CrossRef]   [Pubmed]    Back to citation no. 9
  10. Alrawashdeh W, Ajaz S, Hammond TM, Porrett TR, Lunniss PJ. Primary anal pilonidal disease. Colorectal Dis 2008 Mar;10(3):303–4.   [CrossRef]   [Pubmed]    Back to citation no. 10
  11. Talinia C, Fracaroa GB, Araújoa ACF, dos Reis Limab DM, Westphalen AP. Endoanal pilonidal sinus: case report and literature review. J Coloproctol (Rio J.) 2015;35(3):175–7.   [CrossRef]    Back to citation no. 11
  12. Bascom J. Pilonidal disease: long-term results of follicle removal. Dis Colon Rectum 1983 Dec;26(12):800–7.   [CrossRef]   [Pubmed]    Back to citation no. 12
  13. Nalbant Ak O, Nalbant E. Pilonidal Sinus of the Scalp: A Case Report and Review of the Literature. Balkan Med J 2011;28:445–7.   [Pubmed]    Back to citation no. 13
  14. Vallance S. Pilonidal fistulas mimicking fistulas-in-ano. Br J Surg 1982 Mar;69(3):161–   [CrossRef]   [Pubmed]    Back to citation no. 14

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Author Contributions
Abdulwahid M. Salih – Substantial contributions to conception and design, Acquisition of data, Analysis and interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
F. H. Kakamad – Analysis and interpretation of data, Revising it critically for important intellectual content, Final approval of the version to be published
Guarantor of submission
The corresponding author is the guarantor of submission.
Source of support
None
Conflict of interest
Authors declare no conflict of interest.
Copyright
© 2016 Abdulwahid M. Salih et al. This article is distributed under the terms of Creative Commons Attribution License which permits unrestricted use, distribution and reproduction in any medium provided the original author(s) and original publisher are properly credited. Please see the copyright policy on the journal website for more information.



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