Original Article

Review Article Arch Clin Exp Surg 2016;5:228-232 Archives of Clinical doi:10.5455/aces.20151019114717 Experimental Surgery

Increased of Langerhans Cells in Smokeless Tobacco-Associated Oral Mucosal Lesions Pilonidal sinus disease - Etiological factors, pathogenesis and clinical features Kazim Duman1, Yavuz Ozdemir2, Ozgur Dandin3 Érica Dorigati de Ávila1, Rafael Scaf de Molon2, Melaine de Almeida Lawall1, Renata Bianco 1 1 Consolaro , Alberto Consolaro ABSTRACT

‘Pilonidal sinus’ disease, which is most commonly seen in reproductive populations, such as young adults - mostly in males who are in their twenties - is actually a controversial disease in that there is no consensus on its many facets. It is some- times seen as an infected draining from an opening or a lesion extending to the perineum. It may also present as a draining fstula opening to skin. In terms of etiological factors, various theories (main theories being congenital and 1Bauru Dental School acquired)Abstract have been established since it was frst described, no universal understanding achieved. A long and signifcant University of São Paulo post-operative care period with diferent lengths of recovery depending on the type of operation are quite prevalent with Bauru–SP, Brazil regardsObjective: to recurrence To evaluate and complication the changes status. inIn orderthe number to prevent of recurrence Langerhans and improve Cells the(LC) quality observed of life, etiological in the epithelium and of predisposing factors as well as clinical features of sacrococcygeal should be well known, a detailed difer- 2 smokeless tobacco (SLT-induced) lesions. Araraquara Dental School ential diagnosis should be made, and a suitable and timely intervention should be performed. It was aimed here to explain São Paulo State University theMethods: etiological factors, Microscopic pathogenesis sections and clinical from featuresbiopsies of carriedthe disease out that in themay buccalpresent withmucosa various of clinical twenty symptoms. patients, who were Araraquara-SP, Brazil Keychronic words: users Pilonidal of smokelesssinus disease, tobacco clinic, etiology, (SLT), pathogenesis were utilized. For the control group, twenty non-SLT users of SLT with normal mucosa were selected. Te sections were studied with routine coloring and were immunostained Received: February 05, 2012 Accepted: February 29, 2012 Introductionfor S-100, CD1a, Ki-67 and p63. Tese data were statisticallying to skin [1].analyzed In this by article, the Student’s the aim was t-test to explain to investigate the the Arch Clin Exp Surg 2012;X: X-X ‘Pilonidaldiferences sinus’ in the disease, expression most commonlyof immune seen markers in inetiological normal mucosafactors, pathogenesis and in SLT-induced and clinical leukoplakia features of lesions. DOI: 10.5455/aces.20120229052919 reproductiveResults: populationsTere was a like signi youngfcant adults diference - mostly in thethe immunolabeling disease that may of present all markers with various between clinical normal symp -mucosa in males who are in their twenties - is actually a rather toms and impairs life quality and satisfaction [2]. Corresponding author and SLT-induced lesions (p<0.001). Te leukoplakia lesions in chronic SLT users demonstrated a signifcant Érica Dorigati de Avila controversial disease in that there is still no consensus Etiology increase in the number of Langerhans cells and in the absence of epithelial dysplasia. Departamento de Estomatologia on its treatment, probability of its recurrence is high, Although various theories have been established da Faculdade de Odontologia de and Conclusion:its care takes a Tlonge increasetime and in is thecostly. number It is mostly of these cellssince represents it was frst the described, initial stage no of consensus leukoplakia. has been Bauru an acquired,Key words: chronic Smokeless and in tobacco,fammatory leukoplakic condition lesions, af- cancer,reached. langerhans cells, chewing tobacco. Universidade de São Paulo (USP) fecting the sacrococcygeal region in most cases. It is According to the supporters of congenital theory, Avenida Alameda Octávio sometimes seen as an infected abscess draining from pilonidal sinus disease occurs because of residual epi- Pinheiro Brizola, 9-75, 17012-901 an openingIntroduction or a lesion extending to the perineum. As theliumcontact in the spinal with canal the and oral skin mucosaor when hair and fol - creates a Bauru–SP, Brasil [email protected] well, it may also be observed as a draining fstula open- licles entermore to interspacealkaline formedenvironment, by incomplete its products fusion may Among tobacco users, there is a false be- Author afliations : Department of General Surgery, 1Elazig Military Hospital, Elazig, Turkey 2Gulhane Military Medical Academy, Haydarpasa Teaching Hospital, Uskudar, Istanbul, Turkey 3Bursa Military Hospital, Bursa, Turkey even be more aggressive to tissue [5]. Te Correspondencelief that SLT : Kazim is Duman,safe becauseMD, Department it of Generalis not Surgery, burned, Elazig Military Hospital, Elazig, Turkey. e-mail: [email protected] Received / Accepted : July 02, 2015 / August 03, 2015 percentage of SLT users is lower compared which leads many people to quit cigaretes and start using SLT [1]. However, SLT con- to cigarete users; however, usage is increasing tains higher concentrations of nicotine than among young individuals and it is therefore a cigaretes and, in addition, nearly 30 carci- signifcant and disturbing danger [6,7]. nogenic substances, such as tobacco-specifc Initial studies on the efects of SLT on the N-nitrosamines (TSNA), which is formed oral mucosa demonstrated the formation of during the aging process of the tobacco, [2-4] white lesions induced by chronic exposure to and which presents high carcinogenic poten- tobacco, characterized by epithelial thicken- tial. Moreover, because the tobacco has direct ing, increased vascularization, collagen altera- 229 Duman K et al. occurring as a result of a defective union of the skin Pathogenesis layer during the early embryonic period. On this basis, Pilonidal sinus is a chronic infammatory disease from the time of Fere (1878) to Stone (1951) and even that develops between the sacrococcygeal joint and until today, a number of authors have established four . It is mainly composed of 2 canals - one canal diferent theories: medullary canal theory, dermal in- that begins from the sinus orifce that opens to skin clusion theory, beak glands theory and sex gland theory and moves upwards subcutaneously for 2-5 cm, called [3]. Tese theories, recently having just few supporters, the primary canal. Canals which connect the canal have never been proven and failed to explain pilonidal located posterior to primary canal with the skin are diseases located in the regions other than the sacrococ- called secondary canals. Resultant initially cygeal area [4,5]. Tese authors have been known to form in secondary canals and then primary canals. Te misdiagnose certain diseases, such as medullary canal primary canal is lined by squamous epithelium, while fstulas and hydroadenitis, important in the diferential secondary canals are lined by granulation tissue. Hairs diagnosis of pilonidal disease. In addition, successive freely located in the primary canal and secondary ca- clustering of hairs with the same length, thickness, col- nals contain hair follicles, leukocytes, hemosiderin- our and position could not be explained by this theory containing macrophages but not sweat from sebaceous [6, 7]. Cubic epithelial lining of the inner wall of con- glands [10]. In a typical case, hairs gush outwards from genital pilonidal disease undermines this theory [8]. the sinus opening, and unlike the anal fstula, these si- Te main characteristic of pilonidal sinus is squamous nuses usually move upwards and towards the sacrum, epithelium lining its inner wall. mostly unable to reach to the bone and end as a blind According to the supporters of acquired theory, and enlarged cavity. Tis cavity is macroscopically pilonidal disease occurs as a result of infammation seen as a cystic structure. Hairs may appear as they that results from foreign body reaction occurring gush outwards from the sinus or may be frst seen in secondary to the entry of hairs in the subdermal area the wound afer it is opened. When these hairs are ex- afer trauma. Supporters of this theory also suggest amined, they are seen as structures with thin edges as various other theories to explain the occurrence of the hairs shed. However, in a study by Kooistra, only this mechanism. According to Bascom, hair folli- half of the cases were found to have hairs in the wound cles become infected under the efect of sex steroids [11, 12]. when they are flled with keratin during puberty, this Bascom suggested that the origin of pilonidal sinus infection forming an abscess tract. Hairs in this tract disease was pits could not be present without them. pierce the skin while they are still atached to it and Tey might best be seen in the midline when the glu- are pulled into deeper regions under the vacuuming teus is opened to both sides. Pits with diameters reach- efect of gluteal muscles to form a sinus [7]. Te vacu- ing up to 1-3 mm are formed by hair follicles atached uming efect is mostly observed in the siting position. to the skin with the help of the power of the vacuum For this reason, it is suggested that this disease is more efect. Te weakest point of the skin is pierced and common in people that sit for long periods of time. the abscess process begins. Bascom also put forth that However, this disease is not very common in patients keratin is important in the pathogenesis of pilonidal confned to bed and elderly people who spend their sinus disease, such that keratin shed from the stratum time mostly siting during the day. Karydakis sug- corneum enters into the pits with the efect of pressure gests that hairs move towards the follicle and enter and compression, contributing to the formation of an into deeper regions like a drill. Te frst entry occurs abscess and infammation by anaerobic bacteria. Te through a weak part of the skin and other hairs come analogy is akin to keratin acting much like the match through afer [9]. Tis theory is validated by the fact making the fre [7, 13]. that the disease occurs in interdigital areas, the um- Further, Bascom proposed that in addition to hair bilicus, clitoris and anal canal, and it recurs even afer follicles, cut or shed hairs also had roles in pathogen- radical excisions. esis. Rather than the type and quality of the hair, the Archives of Clinical and Experimental Surgery Year 2016 | Volume 5 | Issue 4 | 228-232 Pilonidal Sinus; etiology, pathogenez, clinic 230

Figure 1. Asymptomatic form. Figure 3. Chronic disease form.

Figure 2. Acute abscess form. Figure 4. Recurrent pilonidal sinus disease. place where hair grows is important. It does not make Clinical Findings any diference whether it is located either above or un- Pilonidal disease has 3 diferent clinical presentations: der the skin, but hairs piercing the skin facilitate the 1. Asymptomatic form establishment of the infection. Karydakis formulated 2. Acute abscess form the idea that hair shedding, piercing ability and afect- 3. Chronic disease form ability of the skin in the natal clef as an important triad r Recurrent complex pilonidal sinus disease; in the pathophysiology of pilonidal sinus disease [14]. r Chronic pilonidal sinus disease. For this reason, surgeons want to leave a fat surface in 1- Asymptomatic form: patients have no com- the area, rather than a nook surface to which hairs may plaints and are incidentally diagnosed. One or more atach and accumulate. pits are located at the midline, about 5 cm from anus, Compression and deoxygenation, accepted as an- are detected (Figure 1). In a study by Efaiha and Abra- other important pathophysiological cause of pilonidal cian, the ratio of these patients among all patients with disease. Natural compression and depth of natal groove pilonidal sinus was found to be 11% [16]. prevents the drainage of abscess located in deeper re- 2- Acute abscess form: severe pain, swelling and gions and above all, they keep healing oxygen away redness are present in the sacrococcygeal region (Fig- from the area. Oxygen cannot be utilized for oxidative ure 2). Fever and leukocytosis may be present. It is killing, which is an important defence mechanism, and the most common cause of hospital admission [17]. so disease recovery becomes difcult [15]. Another study showed that 50% of patients presented www.acesjournal.org Archives of Clinical and Experimental Surgery 231 Duman K et al.

Discussion Te literature suggests that malignant degeneration - especially squamous and varicose carcinomas - may occasionally develop on the basis of disease with long- standing drainage and laceration [22]. It is recommend- ed that wide excision should be performed for chronic, non-healing, ulcerated and rapidly growing lesions and involved inguinal lymph nodes should be biopsied. In the cases with metastatic lymph node involvement, re- currence rate is 50%, 5 year survival is around 50% and prognosis is poor. Chemoradiotherapy may be added

Figure 5. Post-operative appearance. to the treatment regimen in these cases [23]. Pilonidal sinus disease should be diferentiated with this form [18]. Te indurated area containing ab- from those diseases which involve the sacrococcygeal scess gradually grows and accumulated malodorous region [24]; fuid spontaneously drains or is surgically drained. Af- r pruritis ani; ter this stage, conversion to the chronic form begins. r -furuncle, gangrenosum; Microbiological examinations of drainage fuids mostly r periproctitis and perianal abscess and fstulas; reveal anaerobic proliferation (Escherichia coli) [19]. r tuberculosis granuloma, actinomycosis Other common microorganisms are known to be Pro- r osteomyelitis drained by sinuses, teus, beta hemolytic , Pseudomonas and r , S. aureus, respectively. Tere are also studies that sug- r spina bifda, medullary canal fstulas gests S. aureus and Streptococcus were more common r , postanal dermoid . [20]. In a study by Sondenaa et al., the growing micro- It is very important for prognosis to make a difer- organisms in abscess cultures were anaerobes (mainly ential diagnosis among these aforementioned diseases E. Coli) in 77%, aerobes in 4% and mixed microorgan- that have diferent diagnostic and therapeutic protocols isms in 17% of the patients [21]. (Figure 5). 3- Chronic Disease Form: Chronic Pilonidal Sinus Long and important post-operative care periods Disease: named afer the period of abscess drainage. and diferent lengths of recovery depending on the Intermitent drainage and pain are present (Figure 3). type of the operation are quite discouraging in terms of Mild tenderness and induration is present upon palpa- recurrence and complication status. In order to prevent tion. Tere may be one or more than one sinus orifces recurrences and improve the quality of life, etiologi- on which hairs may grow. Skin may have shrinkage and cal and predisposing factors, and also clinical features scarring secondary to drainage. of sacrococcygeal pilonidal disease, should be made Chronic Recurrent Pilonidal Sinus Disease: recur- well known, a detailed diferential diagnosis should be rent abscess atacks following drainage lead to the for- made, and a suitable and timely intervention should be mation of independent and irregular orifces and scars. performed. It is the name of this condition. As a result of these Confict of interest statement acute exacerbations, irregular fstulas sometimes move Te authors have no conficts of interest to declare. towards the anal canal and rectum to cause the forma- References tion of complete fstulas and the sacrum may even be 1. Mentes O, Oysul A, Harlak A, Zeybek N, Kozak O, infltrated because of extension of sinus tissue. It is a Tufan T. Ultrasonography accurately evaluates the condition which may cause psychological problems dimension and shape of the pilonidal sinus. Clinics that negatively afects one’s social life results in a de- 2009;64:189-92. crease in quality of life (Figure 4). 2. Duman K, Ozdemir Y, Yucel E, Akin ML. Compar- Archives of Clinical and Experimental Surgery Year 2016 | Volume 5 | Issue 4 | 228-232 Pilonidal Sinus; etiology, pathogenez, clinic 232

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