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Treatment of adolescent pilonidal disease with a new modification to the Limberg flap: Symmetrically rotated rhomboid excision and lateralization of the Limberg flap technique
Journal of Pediatric Surgery (2013) 48, 1744–1749
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Treatment of adolescent pilonidal disease with a new modification to the Limberg flap: Symmetrically rotated rhomboid excision and lateralization of the Limberg flap technique☆ Çağatay Evrim Afşarlar ⁎, Engin Yılmaz, Ayşe Karaman, İbrahim Karaman, İsmet Faruk Özgüner, Derya Erdoğan, Yusuf Hakan Çavuşoğlu, Haşim Ata Maden Dr. Sami Ulus Children's Hospital, Department of Pediatric Surgery, Ankara, Turkey Received 26 July 2012; revised 19 January 2013; accepted 21 January 2013
Key words: Pilonidal disease; Adolescent; Obesity; Limberg flap; Modified Limberg flap
Abstract Background/Purpose: Pilonidal disease is a common and frustrating problem among adolescents due to its high recurrence rate. The rhomboid excision and Limberg flap techniques promise successful results, but the lower part of the incision left on the intergluteal sulcus is prone to recurrences. Consequently, we have developed a new modification to this technique and have designed a descriptive prospective study to evaluate its efficiency. Methods: We conducted this prospective study between March 2011 and March 2012. All of the patients who were operated on for sacrococcygeal pilonidal disease were included in this study. The surgical procedure primarily consisted of symmetrically rotated (clockwise) rhomboid excision and lateralization of the Limberg flap in order to keep the inferior corner of the suture line apart from the intergluteal sulcus. Results: A total of 15 adolescents (8 boys and 7 girls) were included in the study group. Of the patients, 47% were normal, 13% were overweight, and 40% were obese. Five patients were operated on under general anesthesia, and 10 were operated on under spinal anesthesia. The length of the flap margins ranged from 2.5 to 7 cm (median = 4 cm). The median duration of hospitalization was 5 days, and the median duration of suction drainage was 4 days. The median postoperative follow-up period was 4 months (ranging from 1 to 12 months), and we did not encounter any wound infection or recurrent disease during this period. Only one patient had wound hematoma as a result of drain breakdown and was treated with wound care without any additional complications. Conclusion: Although the number of patients in this study was small and the follow-up period was short, we obtained satisfactory results without any recurrence by performing a symmetrically rotated rhomboid excision and lateralized Limberg flap procedure. © 2013 Elsevier Inc. All rights reserved.
☆ This study was presented at the 19th International Pediatric Colorectal Club Meeting in Rome, June 11–12, 2012. ⁎ Corresponding author. Babür Cad., No: 44, 06080, Altındağ, Ankara, Turkey. Fax: + 312 317 03 53. E-mail address: [email protected] (Ç.E. Afşarlar).
0022-3468/$ – see front matter © 2013 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jpedsurg.2013.01.029
Pilonidal disease (PD) is a common and frustrating problem among adolescents due to its high recurrence rate. The disease was first named by Hodges in 1880 from the
A new modification to the Limberg flap for pilonidal disease Latin words pilus (hair) and nidus (nest) and was long considered to have a congenital etiology [1]. Today, PD is commonly accepted as an acquired lesion in the natal cleft that results from burrowing of loose hair shafts into the distended hair follicles, which subsequently leads to an infected site and forms a suppurative sinus filled with hair [1–5]. In the pediatric surgery literature, various surgical procedures including incision and drainage, curettage of the cavity, unroofing sinus tracts with marsupialization, excision with packing, excision with vacuum-assisted closure (VAC) and excision with primary closure and flap techniques have been reported for the treatment of adolescent PD with different recurrence rates [1,4–12]. In recent decades, better understanding of the risk factors for PD, particularly the importance of the depth of natal cleft and avoiding midline incisions, has promoted the use of the rhomboid excision with Limberg flap closure, which has significantly decreased the recurrence rate of PD [5,13]. Although the rhomboid excision and Limberg flap techniques promise successful results, the recurrence rate following Limberg flap procedures has been reported to range from 0% to 7% [5,14] and has primarily been encountered at the lower portion of the incision left on the intergluteal sulcus [14–17]. Following a case of a recurrent PD disease early after a Limberg flap procedure, we developed a new modification to the rhomboid excision and Limberg flap technique and designed a descriptive prospective study to evaluate the results of this new technique.
1. Materials and methods We conducted a prospective study in our department between March 2011, when we first began to perform this procedure, and March 2012. After obtaining institutional ethics committee approval and informed consent from the parents and patients, the patient's age, gender, body mass index (BMI), duration of symptoms, disease exacerbations, previous history of surgical procedures, history of abscess, and prior culture results were recorded. Following the Limberg flap procedure, the type of anesthesia utilized, duration of hospitalization, duration of suction drainage, postoperative complications and length of follow-up were recorded. All of the patients who were operated on for sacrococcygeal pilonidal disease were included in this study. Patients who were lost to follow-up in the preoperative period were excluded from this study. In the case of acute suppurative PD, patients were treated with sulbactam/ampicillin for 10 days with or without abscess drainage and were reevaluated for surgery following medical treatment. As soon as the cellulitis and acute inflammation resolved, an elective surgery was planned. One staff surgeon (ÇEA) supervised all of the operations. All of the patients were re-examined the day before the surgery. The surgical area was marked, and patients were
1745 advised to clean the area with depilatories and to take a bath. The anesthesiologists determined the type of anesthesia after a discussion with the parents. On the operative day, a rectal cleansing enema was given 4 h before surgery.
1.1. Surgical procedure All the procedures were performed in the prone position under general or spinal anesthesia. A single dose of sulbactam/ampicillin (1 g IV) was administered to patients immediately before the skin incision was made. The surgical area was exposed by lateral traction of the buttocks with adhesive tapes and was cleaned with 10% povidone–iodine. The extent of the sinus tract was determined with methylene blue injection through the sinus orifices. The excision area was delineated according to the sinus orifice localizations and the expanse of natal cleft in order to excise all the diseased tissue and to achieve a flattened natal cleft. The rhomboid excision area was marked by the rotation of the rhombus symmetrically clockwise to achieve 1–2 cm of lateralization in the inferior corner of the rhombus. The borders of the Limberg flap were also marked (Fig. 1A). Following skin incision with a scalpel, the diseased tissue was dissected with diathermy down to the presacral and gluteal fascia and was excised with meticulous hemostasis (Fig. 1B). If any visible sinus tracts were encountered, the excision was further extended into the subcutaneous tissue without any additional skin incision. The Limberg flap was created from non-diseased tissue along the right inferior border of the rhomboid. The flap was elevated off the gluteal fascia to achieve a tensionless rotation with careful dissection to avoid damaging the feeding arteries located in the inferior aspect of the flap (Fig. 1C, D). After releasing the traction tapes, a suction drain placed on the presacral fascia was brought out several centimeters away from the left superior border of the wound, and the Limberg flap was secured with deep, interrupted 2-0 polyglactin sutures passing through the flap and the defect margins. The subcutaneous layer was approximated with 3-0 polyglactin interrupted sutures, and the skin was closed with 4-0 polypropylene sutures (Fig. 1E).
1.2. Postoperative care and follow-up Oral intake was started at postoperative hour 4. Patients were encouraged to ambulate 8 h following the surgery, but were advised to limit extension of the sacral region until they felt completely free of pain and tension. The suction drain was removed after the drainage decreased to 10 cc/day, and patients received sulbactam–ampicillin (1 g per dose IV, three times a day) during this period. Following removal of the suction drain, patients were hospitalized for one additional day to observe the wound and then were discharged. The skin stitches were removed on postoperative day 14, and patients were advised not to sit directly on their buttocks until the third postoperative week. Additionally,
1746
Ç.E. Afşarlar et al.
Fig. 1 (A) Operative design with drawing of symmetrically rotated rhombus and the donor site for Limberg flap. (B) Rhomboid excision is completed down to the presacral and gluteal fascia. (C) Limberg flap is prepared for transposition. (D) Following the release of adhesive tapes, a tensionless flap is ready for closure. (E) Postoperative appearance after skin closure.
patients were instructed on self-hygiene, avoiding contact sports for three months, and the use of depilatories for 3 months. Patients were called for monthly follow-up examinations in the first 3 months postoperation and for quarterly follow-up examinations thereafter until a year had passed.
1.3. Statistical analysis Statistical analyses were performed with the SPSS Statistics for Windows version 15 package software (SPSS Inc., Chicago, IL, USA). Continuous parameters were expressed as medians with ranges, and analyzed with the Mann–Whitney U test. Correlations were investigated using the Kruskal–Wallis test. Categorical variables were analyzed with the Chi square test or, where appropriate, the Fisher's exact test. A p-value of b 0.05 was considered to be statistically significant.
2. Results A total of 15 adolescents, including 8 boys (53%) and 7 girls (47%), composed the study group. The age distribution of the patients ranged from 12 years to 18 years (median = 14 years). The median ages of the boys and girls were 13.6 (12.2 to 16.5) and 15.6 (13.7 to 17.4) years, respectively, and the girls were significantly older than the boys (p = 0.049). The median BMI of the patients was 25 (18.6 to 37). Of the patients, 47% (n = 7) were normal, 13% (n = 2) were overweight, and 40% (n = 6) were obese. The number of
pilonidal sinus orifices ranged from 1 to 4. Sixty-seven percent of the patients had 1 sinus orifice, 13% had three sinus orifices, and 20% of the patients had 4 sinus orifices. Prolonged duration of the disease significantly increased the number of sinus orifices (p = 0.009), but the BMI index and gender did not affect the number of sinus orifices (p N 0.05). Of the patients, 33% (n = 5) had one and 67% (n = 10) had two episodes of PD exacerbations within the median period of 2 months (1 to 12 months) prior to surgery. Although 83% (n = 5) of the obese patients had two episodes of PD exacerbations, no significant difference was found between BMI and the number of PD exacerbations (p N 0.05). Six patients (40%) had pilonidal abscess preoperatively, and the median time between abscess drainage and surgery was 27 days (range = 14 to 60 days). Staphylococcus aureus (n = 2) and Streptococcus strains (n = 2) were encountered in the abscess cultures, and 2 of the cultures yielded no growth. Two patients (13%) had a previous history of excision and primary closure of PD in our department, and their recurrent disease was observed within 1 month and two months after previous surgery, respectively. Five patients (33%) were operated on under general anesthesia, and 10 (67%) were operated on under spinal anesthesia. The median operative time was 90 min (range from 60 to 120 min). The length of the flap margins ranged from 2.5 to 7 cm (median = 4 cm), and the rhomboid excision area ranged from 5 to 41 cm 2 (median = 13.2 cm2). The BMI, gender, and the increased number of sinus orifices did not affect the length of flap margins or the rhomboid excision area (p N 0.05). The median duration of hospitalization was 5 days (4 to 14 days), and the median duration of suction drainage was 4 days (3–10 days).
A new modification to the Limberg flap for pilonidal disease The median postoperative follow-up period was 4 months (ranging from 1 to 12 months), and we did not encounter any wound infection or recurrent disease during this period. Only one patient (6.7%) had wound hematoma as a result of the suction drain breakdown and was treated with wound care without any additional complications.
3. Discussion Pilonidal disease is an acquired disease in young adults and adolescents. The incidence of adolescent PD is reported to be 2.63/1000 [18]. Although adults with PD are reported to have a 3–4:1 male to female ratio [19], various studies concerning adolescent PD have indicated a significantly increased female ratio similar to our results (male:female ratio, 8:7) [6,7,10,11,20], which is attributed to the early onset of puberty in females [4,6]. In our study group, the females were significantly older than the males. Endocrine changes with the initiation of puberty play critical roles in the etiology of the PD. The secretions of the sebaceous glands become more viscous, accumulation of keratin distends the hair follicles, and rapid body growth particularly in the gluteal region stretches the hair follicles in the midline [2,21]. In addition to these physiological changes during puberty, moisture in the depth of the natal cleft provides an environment for the bacterial overgrowth that results in skin maceration. All of these changes, together with a vacuum effect of the buttocks, facilitate the burrowing of loose hair shafts into the hair follicles and initiate the PD [4,13]. Implantation of the loose hair initially causes a foreign body reaction, and bacterial infection subsequently complicates the disease. Finally, the distended and infected follicle ruptures into the subcutaneous tissue and forms an infected sinus cavity [4,22]. Even though this theory clarifies the pathophysiology of PD, it is unclear why some adolescents have the disease but most do not. Other predisposing factors must exists, and for years, hirsutism, poor hygiene, obesity and a deep natal cleft have been proposed as etiologic agents for adolescent PD [4,22,23]. Hair insertion is the essential cause of the disease, and many authors have suggested its prevention as a possible solution [3,6,13,22]. Electrolysis epilation, depilatories, shaving of the area and finally laser epilation have been used for this purpose [22]. Although all of our patients used depilatories during the preoperative and postoperative course (for 3 months), laser epilation is a permanent solution to hirsutism. Obesity is another risk factor for both primary and recurrent PDs because the natal cleft in obese individuals is generally deep and wet, which renders the skin vulnerable to hair penetration [24]. Of the adolescents with PD, 50%– 57.1% have been reported to be overweight and obese in terms of BMI [4,21]. In our study, we used BMI for age according to BMI percentile charts [25] and, similarly, 53% of our patients were overweight or obese. No significant
1747 difference was found between BMI indexes and preoperative PD exacerbations, but 83% of the obese patients had 2 episodes of PD exacerbations prior to surgery in our study. Although Çubukçu et al. [24] have indicated that patients with high BMI had significantly higher recurrence rates of PD, we did not encounter any recurrent PD with the use of our new treatment technique. Various surgical options have been proposed for the treatment of PD due to the disease's tendency to recur. The vast majority of the studies in the literature focus on adult PD. Only a few studies on adolescent PD have been reported and have demonstrated varied success and recurrence rates. Recently reported series indicate the high recurrence rate following classical procedures. Recurrence rates for excision with packing, with marsupialization, and with primary closure were 22% to 26.5%, 23%, and 25%, respectively [10–12]. Almost all the postoperative recurrences and complications were encountered in the midline. The classical procedures do not change the depth of the natal cleft, an important predisposing factor of the disease; thus, such procedures are associated with high recurrence and morbidity rates [14]. Since Karydakis and Bascom emphasized the principles of off-midline closure and flattening of the natal cleft owing to the importance of the depth of the natal cleft, the recurrence rate of the PD has significantly decreased [3,13]. Although the Karydakis flap and Bascom cleft-lift procedure have low reported recurrence rates (0% to 5.8%), these techniques may fail in those with branching fistulas far from midline, extending to each side of the buttocks [5,26]. The rhomboid excision and Limberg flap transposition (Fig. 2A) have many benefits including the ability to aggressively excise extensive and complex disease, easier defect closure with a tension-free rotational flap, flattening of the natal cleft with bulky, well-vascularized tissue and reduced recurrence rates (0%–7%) as well as reduced infection rates (1%–13%) [5,14]. Additionally, rhomboid excision and Limberg flap transposition reduce postoperative pain, the healing period, and early wound complications such as wound dehiscence, maceration, and wound infection [14]. The Limberg flap technique may be regarded as disadvantageous in terms of cosmetic appearance; however, in our study, though the 47% (n = 7) of the patients were female, none of the patients complained about the surgical scar. The only weak point of the classical rhomboid excision and Limberg flap transposition is that the lower pole of the flap stays in the intergluteal sulcus, and all of the recurrences are encountered at this site [14–16]. To contend with this problem, Mentes et al. [15] modified the Limberg flap technique by tailoring the rhomboid excision asymmetrically to place the lower pole of the flap 1–2 cm lateral to the inferior midline (Fig. 2B), and they obtained 0% recurrence rate and 0.8% wound infection rate without any dehiscence or flap necrosis. Several studies have been reported following Mentes et al. concerning asymmetric rhomboid excision and Limberg flap transposition in the same fashion, and they have indicated 0%–5.4% recurrence rates, 1.46%–
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Ç.E. Afşarlar et al.
Fig. 2 (A) Demonstration of classical Limberg flap. (B) Modification of Menteş et al. with asymmetrical rhomboid excision. (C) Modification of Kaya et al. with completely lateralized rhomboid excision. (D) Our new modification with symmetrically rotated rhomboid excision and lateralized Limberg flap.
3.9% wound infection rates and 0%–1.3% wound dehiscence rates, respectively [14,16,17,26]. Kaya et al. [27] have reported another modification of Limberg flap technique. Instead of lateralizing the inferior pole of the rhomboid excision, they completely lateralized the rhomboid excision and transposed the Limberg flap (Fig. 2C) resulting in a 4.2% recurrence, 5.3% wound infection rate and 1.1% wound dehiscence rate. Although these modifications have satisfactory results when compared with the vast majority of the literature, our study demonstrates that there is no need to perform asymmetrical or completely lateralized rhomboid excisions that result in excessive healthy tissue loss. Additionally, a completely lateralized rhomboid excision technique may fail to excise all of the diseased tissue because only a small portion of the excision area stays in the midline and cannot be applicable to the complicated PD with branching fistulas extending to each side of the buttocks. Our new modification of Limberg flap provides satisfactory lateralization of the inferior pole of the flap, centers the diseased area for complete excision and flattens the natal cleft by symmetrically rotating the rhomboid excision clockwise during flap transposition (Fig. 2D). Using this modification, we obtained a 0% recurrence rate without any wound infections or dehiscence. The surgical treatment of adolescent PD should include resection of all diseased tissue, flattening the natal cleft and avoiding midline closure. Our modification of the Limberg flap technique meets these goals with the additional advantage of reduced recurrence, wound dehiscence and infection rates. Longer follow-up evaluations will be required to determine whether these initial results persist over time.
References [1] Golladay ES, Wagner CW. Pediatric pilonidal disease: a method of management. South Med J 1990;83:922-4. [2] Bascom J. Pilonidal disease: origin from follicles of hairs and results of follicle removal as treatment. Surgery 1980;87:567-72. [3] Karydakis GE. Easy and successful treatment of pilonidal sinus after explanation of its causative process. Aust N Z J Surg 1992;62:385-9. [4] Arda IS, Güney LH, Sevmiş S, et al. High body mass index as a possible risk factor for pilonidal sinus disease in adolescents. World J Surg 2005;29:469-71.
[5] Yamout SZ, Caty MG, Lee YH, et al. Early experience with the use of rhomboid excision and Limberg flap in 16 adolescents with pilonidal disease. J Pediatr Surg 2009;44:1586-90. [6] Serour F, Somekh E, Krutman B, et al. Excision with primary closure and suction drainage for pilonidal sinus in adolescent patients. Pediatr Surg Int 2002;18:159-61. [7] Morden P, Drongowski RA, Geiger JD, et al. Comparison of Karydakis versus midline excision for treatment of pilonidal sinus disease. Pediatr Surg Int 2005;21:793-6. [8] Caniano DA, Ruth B, Teich S. Wound management with vacuumassisted closure: experience in 51 pediatric patients. J Pediatr Surg 2005;40:128-32. [9] Bütter A, Emran M, Al-Jazaeri A, et al. Vacuum-assisted closure for wound management in the pediatric population. J Pediatr Surg 2006;41:940-2. [10] Nasr A, Ein SH. A pediatric surgeon's 35-year experience with pilonidal disease in a Canadian children's hospital. Can J Surg 2011;54:39-42. [11] Fike FB, Mortellaro VE, Juang D, et al. Experience with pilonidal disease in children. J Surg Res 2011;170:165-8. [12] Gendy AS, Glick RD, Hong AR, et al. A comparison of the cleft lift procedure vs wide excision and packing for the treatment of pilonidal disease in adolescents. J Pediatr Surg 2011;46:1256-9. [13] Bascom J, Bascom T. Failed pilonidal surgery: new paradigm and new operation leading to cures. Arch Surg 2002;137:1146-50. [14] Akin M, Leventoglu S, Mentes BB, et al. Comparison of the classic Limberg flap and modified Limberg flap in the treatment of pilonidal sinus disease: a retrospective analysis of 416 patients. Surg Today 2010;40:757-62. [15] Mentes BB, Leventoglu S, Cihan A, et al. Modified Limberg transposition flap for sacrococcygeal pilonidal sinus. Surg Today 2004;34:419-23. [16] Cihan A, Mentes BB, Tatlicioglu E, et al. Modified Limberg flap reconstruction compares favourably with primary repair for pilonidal sinus surgery. ANZ J Surg 2004;74:238-42. [17] Cihan A, Ucan BH, Comert M, et al. Superiority of asymmetric modified Limberg flap for surgical treatment of pilonidal disease. Dis Colon Rectum 2006;49:244-9. [18] Yücesan S, Dindar H, Olcay I, et al. Prevalence of congenital abnormalities in Turkish school children. Eur J Epidemiol 1993;9:373-80. [19] Horwood J, Hanratty D, Chandran P, et al. Primary closure or rhomboid excision and Limberg flap for the management of primary sacrococcygeal pilonidal disease? A meta-analysis of randomized controlled trials. Colorectal Dis 2012;14:143-51. [20] Lee SL, Tejirian T, Abbas MA. Current management of adolescent pilonidal disease. J Pediatr Surg 2008;43:1124-7. [21] Golladay ES. Outpatient adolescent surgical problems. Adolesc Med Clin 2004;15:503-20. [22] Lukish JR, Kindelan T, Marmon LM, et al. Laser epilation is a safe and effective therapy for teenagers with pilonidal disease. J Pediatr Surg 2009;44:282-5. [23] Chamberlain JW, Vawter GF. The congenital origin of pilonidal sinus. J Pediatr Surg 1974;9:441-4.
A new modification to the Limberg flap for pilonidal disease [24] Cubukçu A, Gönüllü NN, Paksoy M, et al. The role of obesity on the recurrence of pilonidal sinus disease in patients, who were treated by excision and Limberg flap transposition. Int J Colorectal Dis 2000;15: 173-5. [25] Neyzi O, Günöz H, Furman A, et al. Weight, height, head circumference and body mass index references for Turkish children. Turk Pediatr J 2008;51:1-14.
1749 [26] Can MF, Sevinc MM, Hancerliogullari O, et al. Multicenter prospective randomized trial comparing modified Limberg flap transposition and Karydakis flap reconstruction in patients with sacrococcygeal pilonidal disease. Am J Surg 2010;200:318-27. [27] Kaya B, Eris C, Atalay S, et al. Modified Limberg transposition flap in the treatment of pilonidal sinus disease. Tech Coloproctol 2012;16: 55-9.
www.elsevier.com/locate/jpedsurg
Treatment of adolescent pilonidal disease with a new modification to the Limberg flap: Symmetrically rotated rhomboid excision and lateralization of the Limberg flap technique☆ Çağatay Evrim Afşarlar ⁎, Engin Yılmaz, Ayşe Karaman, İbrahim Karaman, İsmet Faruk Özgüner, Derya Erdoğan, Yusuf Hakan Çavuşoğlu, Haşim Ata Maden Dr. Sami Ulus Children's Hospital, Department of Pediatric Surgery, Ankara, Turkey Received 26 July 2012; revised 19 January 2013; accepted 21 January 2013
Key words: Pilonidal disease; Adolescent; Obesity; Limberg flap; Modified Limberg flap
Abstract Background/Purpose: Pilonidal disease is a common and frustrating problem among adolescents due to its high recurrence rate. The rhomboid excision and Limberg flap techniques promise successful results, but the lower part of the incision left on the intergluteal sulcus is prone to recurrences. Consequently, we have developed a new modification to this technique and have designed a descriptive prospective study to evaluate its efficiency. Methods: We conducted this prospective study between March 2011 and March 2012. All of the patients who were operated on for sacrococcygeal pilonidal disease were included in this study. The surgical procedure primarily consisted of symmetrically rotated (clockwise) rhomboid excision and lateralization of the Limberg flap in order to keep the inferior corner of the suture line apart from the intergluteal sulcus. Results: A total of 15 adolescents (8 boys and 7 girls) were included in the study group. Of the patients, 47% were normal, 13% were overweight, and 40% were obese. Five patients were operated on under general anesthesia, and 10 were operated on under spinal anesthesia. The length of the flap margins ranged from 2.5 to 7 cm (median = 4 cm). The median duration of hospitalization was 5 days, and the median duration of suction drainage was 4 days. The median postoperative follow-up period was 4 months (ranging from 1 to 12 months), and we did not encounter any wound infection or recurrent disease during this period. Only one patient had wound hematoma as a result of drain breakdown and was treated with wound care without any additional complications. Conclusion: Although the number of patients in this study was small and the follow-up period was short, we obtained satisfactory results without any recurrence by performing a symmetrically rotated rhomboid excision and lateralized Limberg flap procedure. © 2013 Elsevier Inc. All rights reserved.
☆ This study was presented at the 19th International Pediatric Colorectal Club Meeting in Rome, June 11–12, 2012. ⁎ Corresponding author. Babür Cad., No: 44, 06080, Altındağ, Ankara, Turkey. Fax: + 312 317 03 53. E-mail address: [email protected] (Ç.E. Afşarlar).
0022-3468/$ – see front matter © 2013 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jpedsurg.2013.01.029
Pilonidal disease (PD) is a common and frustrating problem among adolescents due to its high recurrence rate. The disease was first named by Hodges in 1880 from the
A new modification to the Limberg flap for pilonidal disease Latin words pilus (hair) and nidus (nest) and was long considered to have a congenital etiology [1]. Today, PD is commonly accepted as an acquired lesion in the natal cleft that results from burrowing of loose hair shafts into the distended hair follicles, which subsequently leads to an infected site and forms a suppurative sinus filled with hair [1–5]. In the pediatric surgery literature, various surgical procedures including incision and drainage, curettage of the cavity, unroofing sinus tracts with marsupialization, excision with packing, excision with vacuum-assisted closure (VAC) and excision with primary closure and flap techniques have been reported for the treatment of adolescent PD with different recurrence rates [1,4–12]. In recent decades, better understanding of the risk factors for PD, particularly the importance of the depth of natal cleft and avoiding midline incisions, has promoted the use of the rhomboid excision with Limberg flap closure, which has significantly decreased the recurrence rate of PD [5,13]. Although the rhomboid excision and Limberg flap techniques promise successful results, the recurrence rate following Limberg flap procedures has been reported to range from 0% to 7% [5,14] and has primarily been encountered at the lower portion of the incision left on the intergluteal sulcus [14–17]. Following a case of a recurrent PD disease early after a Limberg flap procedure, we developed a new modification to the rhomboid excision and Limberg flap technique and designed a descriptive prospective study to evaluate the results of this new technique.
1. Materials and methods We conducted a prospective study in our department between March 2011, when we first began to perform this procedure, and March 2012. After obtaining institutional ethics committee approval and informed consent from the parents and patients, the patient's age, gender, body mass index (BMI), duration of symptoms, disease exacerbations, previous history of surgical procedures, history of abscess, and prior culture results were recorded. Following the Limberg flap procedure, the type of anesthesia utilized, duration of hospitalization, duration of suction drainage, postoperative complications and length of follow-up were recorded. All of the patients who were operated on for sacrococcygeal pilonidal disease were included in this study. Patients who were lost to follow-up in the preoperative period were excluded from this study. In the case of acute suppurative PD, patients were treated with sulbactam/ampicillin for 10 days with or without abscess drainage and were reevaluated for surgery following medical treatment. As soon as the cellulitis and acute inflammation resolved, an elective surgery was planned. One staff surgeon (ÇEA) supervised all of the operations. All of the patients were re-examined the day before the surgery. The surgical area was marked, and patients were
1745 advised to clean the area with depilatories and to take a bath. The anesthesiologists determined the type of anesthesia after a discussion with the parents. On the operative day, a rectal cleansing enema was given 4 h before surgery.
1.1. Surgical procedure All the procedures were performed in the prone position under general or spinal anesthesia. A single dose of sulbactam/ampicillin (1 g IV) was administered to patients immediately before the skin incision was made. The surgical area was exposed by lateral traction of the buttocks with adhesive tapes and was cleaned with 10% povidone–iodine. The extent of the sinus tract was determined with methylene blue injection through the sinus orifices. The excision area was delineated according to the sinus orifice localizations and the expanse of natal cleft in order to excise all the diseased tissue and to achieve a flattened natal cleft. The rhomboid excision area was marked by the rotation of the rhombus symmetrically clockwise to achieve 1–2 cm of lateralization in the inferior corner of the rhombus. The borders of the Limberg flap were also marked (Fig. 1A). Following skin incision with a scalpel, the diseased tissue was dissected with diathermy down to the presacral and gluteal fascia and was excised with meticulous hemostasis (Fig. 1B). If any visible sinus tracts were encountered, the excision was further extended into the subcutaneous tissue without any additional skin incision. The Limberg flap was created from non-diseased tissue along the right inferior border of the rhomboid. The flap was elevated off the gluteal fascia to achieve a tensionless rotation with careful dissection to avoid damaging the feeding arteries located in the inferior aspect of the flap (Fig. 1C, D). After releasing the traction tapes, a suction drain placed on the presacral fascia was brought out several centimeters away from the left superior border of the wound, and the Limberg flap was secured with deep, interrupted 2-0 polyglactin sutures passing through the flap and the defect margins. The subcutaneous layer was approximated with 3-0 polyglactin interrupted sutures, and the skin was closed with 4-0 polypropylene sutures (Fig. 1E).
1.2. Postoperative care and follow-up Oral intake was started at postoperative hour 4. Patients were encouraged to ambulate 8 h following the surgery, but were advised to limit extension of the sacral region until they felt completely free of pain and tension. The suction drain was removed after the drainage decreased to 10 cc/day, and patients received sulbactam–ampicillin (1 g per dose IV, three times a day) during this period. Following removal of the suction drain, patients were hospitalized for one additional day to observe the wound and then were discharged. The skin stitches were removed on postoperative day 14, and patients were advised not to sit directly on their buttocks until the third postoperative week. Additionally,
1746
Ç.E. Afşarlar et al.
Fig. 1 (A) Operative design with drawing of symmetrically rotated rhombus and the donor site for Limberg flap. (B) Rhomboid excision is completed down to the presacral and gluteal fascia. (C) Limberg flap is prepared for transposition. (D) Following the release of adhesive tapes, a tensionless flap is ready for closure. (E) Postoperative appearance after skin closure.
patients were instructed on self-hygiene, avoiding contact sports for three months, and the use of depilatories for 3 months. Patients were called for monthly follow-up examinations in the first 3 months postoperation and for quarterly follow-up examinations thereafter until a year had passed.
1.3. Statistical analysis Statistical analyses were performed with the SPSS Statistics for Windows version 15 package software (SPSS Inc., Chicago, IL, USA). Continuous parameters were expressed as medians with ranges, and analyzed with the Mann–Whitney U test. Correlations were investigated using the Kruskal–Wallis test. Categorical variables were analyzed with the Chi square test or, where appropriate, the Fisher's exact test. A p-value of b 0.05 was considered to be statistically significant.
2. Results A total of 15 adolescents, including 8 boys (53%) and 7 girls (47%), composed the study group. The age distribution of the patients ranged from 12 years to 18 years (median = 14 years). The median ages of the boys and girls were 13.6 (12.2 to 16.5) and 15.6 (13.7 to 17.4) years, respectively, and the girls were significantly older than the boys (p = 0.049). The median BMI of the patients was 25 (18.6 to 37). Of the patients, 47% (n = 7) were normal, 13% (n = 2) were overweight, and 40% (n = 6) were obese. The number of
pilonidal sinus orifices ranged from 1 to 4. Sixty-seven percent of the patients had 1 sinus orifice, 13% had three sinus orifices, and 20% of the patients had 4 sinus orifices. Prolonged duration of the disease significantly increased the number of sinus orifices (p = 0.009), but the BMI index and gender did not affect the number of sinus orifices (p N 0.05). Of the patients, 33% (n = 5) had one and 67% (n = 10) had two episodes of PD exacerbations within the median period of 2 months (1 to 12 months) prior to surgery. Although 83% (n = 5) of the obese patients had two episodes of PD exacerbations, no significant difference was found between BMI and the number of PD exacerbations (p N 0.05). Six patients (40%) had pilonidal abscess preoperatively, and the median time between abscess drainage and surgery was 27 days (range = 14 to 60 days). Staphylococcus aureus (n = 2) and Streptococcus strains (n = 2) were encountered in the abscess cultures, and 2 of the cultures yielded no growth. Two patients (13%) had a previous history of excision and primary closure of PD in our department, and their recurrent disease was observed within 1 month and two months after previous surgery, respectively. Five patients (33%) were operated on under general anesthesia, and 10 (67%) were operated on under spinal anesthesia. The median operative time was 90 min (range from 60 to 120 min). The length of the flap margins ranged from 2.5 to 7 cm (median = 4 cm), and the rhomboid excision area ranged from 5 to 41 cm 2 (median = 13.2 cm2). The BMI, gender, and the increased number of sinus orifices did not affect the length of flap margins or the rhomboid excision area (p N 0.05). The median duration of hospitalization was 5 days (4 to 14 days), and the median duration of suction drainage was 4 days (3–10 days).
A new modification to the Limberg flap for pilonidal disease The median postoperative follow-up period was 4 months (ranging from 1 to 12 months), and we did not encounter any wound infection or recurrent disease during this period. Only one patient (6.7%) had wound hematoma as a result of the suction drain breakdown and was treated with wound care without any additional complications.
3. Discussion Pilonidal disease is an acquired disease in young adults and adolescents. The incidence of adolescent PD is reported to be 2.63/1000 [18]. Although adults with PD are reported to have a 3–4:1 male to female ratio [19], various studies concerning adolescent PD have indicated a significantly increased female ratio similar to our results (male:female ratio, 8:7) [6,7,10,11,20], which is attributed to the early onset of puberty in females [4,6]. In our study group, the females were significantly older than the males. Endocrine changes with the initiation of puberty play critical roles in the etiology of the PD. The secretions of the sebaceous glands become more viscous, accumulation of keratin distends the hair follicles, and rapid body growth particularly in the gluteal region stretches the hair follicles in the midline [2,21]. In addition to these physiological changes during puberty, moisture in the depth of the natal cleft provides an environment for the bacterial overgrowth that results in skin maceration. All of these changes, together with a vacuum effect of the buttocks, facilitate the burrowing of loose hair shafts into the hair follicles and initiate the PD [4,13]. Implantation of the loose hair initially causes a foreign body reaction, and bacterial infection subsequently complicates the disease. Finally, the distended and infected follicle ruptures into the subcutaneous tissue and forms an infected sinus cavity [4,22]. Even though this theory clarifies the pathophysiology of PD, it is unclear why some adolescents have the disease but most do not. Other predisposing factors must exists, and for years, hirsutism, poor hygiene, obesity and a deep natal cleft have been proposed as etiologic agents for adolescent PD [4,22,23]. Hair insertion is the essential cause of the disease, and many authors have suggested its prevention as a possible solution [3,6,13,22]. Electrolysis epilation, depilatories, shaving of the area and finally laser epilation have been used for this purpose [22]. Although all of our patients used depilatories during the preoperative and postoperative course (for 3 months), laser epilation is a permanent solution to hirsutism. Obesity is another risk factor for both primary and recurrent PDs because the natal cleft in obese individuals is generally deep and wet, which renders the skin vulnerable to hair penetration [24]. Of the adolescents with PD, 50%– 57.1% have been reported to be overweight and obese in terms of BMI [4,21]. In our study, we used BMI for age according to BMI percentile charts [25] and, similarly, 53% of our patients were overweight or obese. No significant
1747 difference was found between BMI indexes and preoperative PD exacerbations, but 83% of the obese patients had 2 episodes of PD exacerbations prior to surgery in our study. Although Çubukçu et al. [24] have indicated that patients with high BMI had significantly higher recurrence rates of PD, we did not encounter any recurrent PD with the use of our new treatment technique. Various surgical options have been proposed for the treatment of PD due to the disease's tendency to recur. The vast majority of the studies in the literature focus on adult PD. Only a few studies on adolescent PD have been reported and have demonstrated varied success and recurrence rates. Recently reported series indicate the high recurrence rate following classical procedures. Recurrence rates for excision with packing, with marsupialization, and with primary closure were 22% to 26.5%, 23%, and 25%, respectively [10–12]. Almost all the postoperative recurrences and complications were encountered in the midline. The classical procedures do not change the depth of the natal cleft, an important predisposing factor of the disease; thus, such procedures are associated with high recurrence and morbidity rates [14]. Since Karydakis and Bascom emphasized the principles of off-midline closure and flattening of the natal cleft owing to the importance of the depth of the natal cleft, the recurrence rate of the PD has significantly decreased [3,13]. Although the Karydakis flap and Bascom cleft-lift procedure have low reported recurrence rates (0% to 5.8%), these techniques may fail in those with branching fistulas far from midline, extending to each side of the buttocks [5,26]. The rhomboid excision and Limberg flap transposition (Fig. 2A) have many benefits including the ability to aggressively excise extensive and complex disease, easier defect closure with a tension-free rotational flap, flattening of the natal cleft with bulky, well-vascularized tissue and reduced recurrence rates (0%–7%) as well as reduced infection rates (1%–13%) [5,14]. Additionally, rhomboid excision and Limberg flap transposition reduce postoperative pain, the healing period, and early wound complications such as wound dehiscence, maceration, and wound infection [14]. The Limberg flap technique may be regarded as disadvantageous in terms of cosmetic appearance; however, in our study, though the 47% (n = 7) of the patients were female, none of the patients complained about the surgical scar. The only weak point of the classical rhomboid excision and Limberg flap transposition is that the lower pole of the flap stays in the intergluteal sulcus, and all of the recurrences are encountered at this site [14–16]. To contend with this problem, Mentes et al. [15] modified the Limberg flap technique by tailoring the rhomboid excision asymmetrically to place the lower pole of the flap 1–2 cm lateral to the inferior midline (Fig. 2B), and they obtained 0% recurrence rate and 0.8% wound infection rate without any dehiscence or flap necrosis. Several studies have been reported following Mentes et al. concerning asymmetric rhomboid excision and Limberg flap transposition in the same fashion, and they have indicated 0%–5.4% recurrence rates, 1.46%–
1748
Ç.E. Afşarlar et al.
Fig. 2 (A) Demonstration of classical Limberg flap. (B) Modification of Menteş et al. with asymmetrical rhomboid excision. (C) Modification of Kaya et al. with completely lateralized rhomboid excision. (D) Our new modification with symmetrically rotated rhomboid excision and lateralized Limberg flap.
3.9% wound infection rates and 0%–1.3% wound dehiscence rates, respectively [14,16,17,26]. Kaya et al. [27] have reported another modification of Limberg flap technique. Instead of lateralizing the inferior pole of the rhomboid excision, they completely lateralized the rhomboid excision and transposed the Limberg flap (Fig. 2C) resulting in a 4.2% recurrence, 5.3% wound infection rate and 1.1% wound dehiscence rate. Although these modifications have satisfactory results when compared with the vast majority of the literature, our study demonstrates that there is no need to perform asymmetrical or completely lateralized rhomboid excisions that result in excessive healthy tissue loss. Additionally, a completely lateralized rhomboid excision technique may fail to excise all of the diseased tissue because only a small portion of the excision area stays in the midline and cannot be applicable to the complicated PD with branching fistulas extending to each side of the buttocks. Our new modification of Limberg flap provides satisfactory lateralization of the inferior pole of the flap, centers the diseased area for complete excision and flattens the natal cleft by symmetrically rotating the rhomboid excision clockwise during flap transposition (Fig. 2D). Using this modification, we obtained a 0% recurrence rate without any wound infections or dehiscence. The surgical treatment of adolescent PD should include resection of all diseased tissue, flattening the natal cleft and avoiding midline closure. Our modification of the Limberg flap technique meets these goals with the additional advantage of reduced recurrence, wound dehiscence and infection rates. Longer follow-up evaluations will be required to determine whether these initial results persist over time.
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[5] Yamout SZ, Caty MG, Lee YH, et al. Early experience with the use of rhomboid excision and Limberg flap in 16 adolescents with pilonidal disease. J Pediatr Surg 2009;44:1586-90. [6] Serour F, Somekh E, Krutman B, et al. Excision with primary closure and suction drainage for pilonidal sinus in adolescent patients. Pediatr Surg Int 2002;18:159-61. [7] Morden P, Drongowski RA, Geiger JD, et al. Comparison of Karydakis versus midline excision for treatment of pilonidal sinus disease. Pediatr Surg Int 2005;21:793-6. [8] Caniano DA, Ruth B, Teich S. Wound management with vacuumassisted closure: experience in 51 pediatric patients. J Pediatr Surg 2005;40:128-32. [9] Bütter A, Emran M, Al-Jazaeri A, et al. Vacuum-assisted closure for wound management in the pediatric population. J Pediatr Surg 2006;41:940-2. [10] Nasr A, Ein SH. A pediatric surgeon's 35-year experience with pilonidal disease in a Canadian children's hospital. Can J Surg 2011;54:39-42. [11] Fike FB, Mortellaro VE, Juang D, et al. Experience with pilonidal disease in children. J Surg Res 2011;170:165-8. [12] Gendy AS, Glick RD, Hong AR, et al. A comparison of the cleft lift procedure vs wide excision and packing for the treatment of pilonidal disease in adolescents. J Pediatr Surg 2011;46:1256-9. [13] Bascom J, Bascom T. Failed pilonidal surgery: new paradigm and new operation leading to cures. Arch Surg 2002;137:1146-50. [14] Akin M, Leventoglu S, Mentes BB, et al. Comparison of the classic Limberg flap and modified Limberg flap in the treatment of pilonidal sinus disease: a retrospective analysis of 416 patients. Surg Today 2010;40:757-62. [15] Mentes BB, Leventoglu S, Cihan A, et al. Modified Limberg transposition flap for sacrococcygeal pilonidal sinus. Surg Today 2004;34:419-23. [16] Cihan A, Mentes BB, Tatlicioglu E, et al. Modified Limberg flap reconstruction compares favourably with primary repair for pilonidal sinus surgery. ANZ J Surg 2004;74:238-42. [17] Cihan A, Ucan BH, Comert M, et al. Superiority of asymmetric modified Limberg flap for surgical treatment of pilonidal disease. Dis Colon Rectum 2006;49:244-9. [18] Yücesan S, Dindar H, Olcay I, et al. Prevalence of congenital abnormalities in Turkish school children. Eur J Epidemiol 1993;9:373-80. [19] Horwood J, Hanratty D, Chandran P, et al. Primary closure or rhomboid excision and Limberg flap for the management of primary sacrococcygeal pilonidal disease? A meta-analysis of randomized controlled trials. Colorectal Dis 2012;14:143-51. [20] Lee SL, Tejirian T, Abbas MA. Current management of adolescent pilonidal disease. J Pediatr Surg 2008;43:1124-7. [21] Golladay ES. Outpatient adolescent surgical problems. Adolesc Med Clin 2004;15:503-20. [22] Lukish JR, Kindelan T, Marmon LM, et al. Laser epilation is a safe and effective therapy for teenagers with pilonidal disease. J Pediatr Surg 2009;44:282-5. [23] Chamberlain JW, Vawter GF. The congenital origin of pilonidal sinus. J Pediatr Surg 1974;9:441-4.
A new modification to the Limberg flap for pilonidal disease [24] Cubukçu A, Gönüllü NN, Paksoy M, et al. The role of obesity on the recurrence of pilonidal sinus disease in patients, who were treated by excision and Limberg flap transposition. Int J Colorectal Dis 2000;15: 173-5. [25] Neyzi O, Günöz H, Furman A, et al. Weight, height, head circumference and body mass index references for Turkish children. Turk Pediatr J 2008;51:1-14.
1749 [26] Can MF, Sevinc MM, Hancerliogullari O, et al. Multicenter prospective randomized trial comparing modified Limberg flap transposition and Karydakis flap reconstruction in patients with sacrococcygeal pilonidal disease. Am J Surg 2010;200:318-27. [27] Kaya B, Eris C, Atalay S, et al. Modified Limberg transposition flap in the treatment of pilonidal sinus disease. Tech Coloproctol 2012;16: 55-9.