www.elsevier.com/locate/jpedsurg

Journal of Pediatric Surgery (2009) 44, 1586–1590

Early experience with the use of rhomboid excision andLimberg flap in 16 adolescents with pilonidal diseaseSani Z. Yamout⁎, Michael G. Caty, Yi-Horng Lee, Stanely T. Lau,Mauricio A. Escobar, Philip L. Glick

Division of Pediatric Surgery, Department of Surgery, University at Buffalo, Women and Children’s Hospital of Buffalo,Buffalo, NY, USA

Received 25 July 2008; revised 4 November 2008; accepted 5 November 2008

CT

0d

Key words:Pilonidal disease;Limberg flap, Adolescents

AbstractBackground: Rhomboid excision with Limberg flap (RELF) repair has been shown to be effective in themanagement of pilonidal disease (PD) in adults. Wide excision allows complete removal of diseasedtissue, and the rotational flap allows tensionless coverage as well as helps flatten the natal crease, whichis believed to contribute to the recurrence of PD.Methods: This study is a retrospective review of all adolescents who underwent excision of pilonidaldisease using RELF at a single institution for a period of 18 months.Results: Sixteen adolescents with PD were treated with RELF during this period. All procedures werecompletedwith no intraoperative complications.Mean operative time and hospital staywere 92 ± 30minutesand 1.8 ± 0.29 days, respectively. Mean follow-up was 11 ± 6.0 months. One patient had recurrence of hisdisease, and one needed prolonged wound care after wound breakdown. Six others had minor complicationsincluding 4 patients (25%) who had superficial wound separation that resolved promptly with dressingchange. One patient had a superficial wound infection. One patient had residual pain.Conclusion:Rhomboid excision with Limberg flap is effective in the management of PD in adolescents. The6% recurrence rate is similar to that reported in the adult literature. Despite the limitations of this study, thelow morbidity, hospital stay, and recurrence rate noted with our initial experience are very encouraging.© 2009 Elsevier Inc. All rights reserved.

Pilonidal sinus disease is a chronic disorder of the disease process, surgical procedures have been modified so

sacrococcygeal area. It is a challenging entity to managebecause of the morbidity and high recurrence rates associatedwith its treatment. The multiple surgical procedures availablefor therapy suggest that an optimal technique is still lacking.With better understanding of the pathophysiology behind this

⁎ Corresponding author. Division of Pediatric Surgery, Women andhildren's Hospital of Buffalo, 219 Bryant Street, Buffalo, NY 14222, USAel.: +1 716 430 3242, +1 716 878 7301; fax: +1 716 888 3850.E-mail address: syamout@kaleidahealth.org (S.Z. Yamout).

022-3468/$ – see front matter © 2009 Elsevier Inc. All rights reserved.oi:10.1016/j.jpedsurg.2008.11.033

that both the diseased tissue is removed and the predisposingfactors are decreased. The advantages of rhomboid tissueexcision with Limberg rotational flap (RELF) repair includethe ability to aggressively excise diseased tissue andaccomplish tensionless wound coverage using a skin andsoft tissue rotational flap from the gluteal region. In addition,the bulky flap flattens the gluteal cleft. The deep gluteal cleftis believed to play a central role in disease recurrence bymaintaining a moist environment that predisposes to skinmaceration, bacterial overgrowth, and ingrowth of hair. The

1587Rhomboid excision & limberg flap in pilonidal disease

use of the RELF repair has shown great promise in the adultpopulation [1-4]; however, this technique is not welldocumented in the pediatric surgery literature.

In this study, we present our experience with RELFtechnique for the management of pilonidal sinus disease inadolescents in a pediatric surgery practice.

1. Materials and methods

This is a single institution, retrospective review of alladolescent patients who underwent RELF repair of sacro-coccygeal pilonidal sinus disease at the Division of PediatricSurgery at the Women and Children's Hospital of Buffalo(Buffalo, NY) between February 2006, at the time when thisprocedure was first performed at this institution, andDecember 2007. Before this period, most patients withpilonidal disease were treated with drainage alone ordrainage followed by excision and marsupialization orprimary closure. All patients who underwent RELF duringthis period were included. Two staff surgeons (PLG, YHL)performed all the operation, with most procedures per-formed by the senior author (69%). Although no majorvariations in operative technique or perioperative manage-ment existed between the 2 operating surgeons, no specificattempts to standardize management were made. Theexperience of both staff surgeons was limited to patientspresented in this study. Institutional review board approvalwas obtained (institutional review board no. 2259) beforedata collection. Sixteen patients were identified and theircharts reviewed. Patients were diagnosed as having pilonidaldisease if they were found to have clinical evidence ofinflammation or infection with hair burrowing in midlinesinus pits in the sacrococcygeal area. Patients who presentedwith infected pilonidal sinuses were treated with 7 to 10 daysof cephalexin (10mg/Kg/dose) after drainage if necessary.These patients were then reevaluated for signs of persistentinfection. The criteria for the determination of resolutionof infection were the absence of cellulitis and purulentdrainage from the wound site. Definitive repair was delayeduntil all signs of infection had resolved and the woundscompletely healed; a minimum of 8 weeks of infection-freetime was preferred in the staged procedure.

1.1. Procedure

All procedures are performed under general anesthesia. Asingle dose of intravenous cefazolin (25 mg/kg per dose) isadministered to patients within 30 minutes of the skinincision. Patients are placed in the prone position beforeshaving and cleaning the sacrococcygeal area with povidone-iodine. Methylene blue is then injected into all sinus tracts toguide complete excision. Once the extent of the disease isdelineated, the area to be excised is included within arhomboid form drawn on the skin (Fig. 1A). A piece of

cardboard is then placed on the skin over the rhomboid tomake an exact template, and this is used to outline theborders of the Limberg rotational flap (Fig. 1B). Thediseased area is then excised by extending the incisionsdown to the level of the presacral and gluteal fascia (Fig. 1C).If any methylene blue–stained sinus tracts are encountered,the excision is extended further to include them to avoidleaving residual disease and then the rotational flap isredrawn if necessary. Once all visible disease is excised, theLimberg flap is prepared by extending the skin incisions tothe gluteal fascia. The flap is then elevated off the fascia toallow for tensionless rotation (Fig. 1D). This is followed byinserting a 10F suction drain through the skin proximally andlaying it deep to the flap (Fig. 1E). The wound is then closedwith a single layer of full thickness nonabsorbable sutures(Fig. 1F). A field block is then performed by injectingbupivacaine (2.5 mg/kg per dose) into the surrounding tissue.Patients are placed on oral cephalexin (10 mg/kg/dose) untiltheir drain is removed 1 week later, to help prevent woundinfections. Patients are discharged on oral acetaminophen/hydrocodone on the day of surgery or the next day. Stoolsofteners are strongly encouraged to avoid constipation.Patients are instructed to avoid sitting for 2 weeks andinstead to lay on their side or abdomen and avoid trauma tothat region for 4 to 8 weeks, after which they can return tofull unrestricted activity. Postoperatively, no attempts atpreventing hair regrowth at the site of repair were made.Sutures are removed 2 weeks postoperatively, after whichfollow-up consisted of telephone calls to check on progressor on an as needed basis in the office. Information regardingthe duration of operation and hospital stay, early and latewound complications, and recurrence was collected.

2. Results

Fifty-four patients were treated for pilonidal disease over aperiod of 22 months. Most of the patients were treated witheither drainage alone or drainage followed by excision andmarsupialization or primary closure. Sixteen patients weretreated with RELF during this period. There were 9 males and7 females, with a mean age of 16 ± 1.9 years (range, 13-19 years). Median follow-up was 11 ± 6.0 months (range,5-22 months). Two patients had undergone previous excisionand repair of PD; one with primary closure and the other byZ-plasty technique. Fifteen patients initially presented withsoft tissue infection, 12 of whom required incision anddrainage to control the infection before staged definitiverepair, and the rest were treated with oral antibiotics only. Theaverage time from treatment of infection to definitive repairwas 134 ± 85 days (range, 41-305 days).

All procedures were successfully completed with nointraoperative complications. The average size of resultantdefect of 15 of the patients after excision of all grosslyinvolved tissue was 20.5 ± 14.4 cm2 (range, 5-64 cm2). The

Fig. 1 A, A rhombus is drawn around the tissue to be excised; B, the Limberg flap (X) is outlined adjacent to the rhombus; C, the diseasedtissue is excised to the level of the fascia; D, the Limberg flap is rotated into the defect; E, a drain is placed under the flap; and F, final view ofthe repair.

1588 S.Z. Yamout et al.

size of the defect in one patient was not documented. Themean operative time was 92 ± 30 minutes (range, 58-160minutes) with a mean hospital stay of 1.8 ± 0.29 days (range,1-2 days). On follow-up, most patients did well with nomajor complications. One patient had disease recurrence 18months postoperatively. Recurrence was defined as anyclinical or pathologic evidence of pilonidal disease (inflam-mation with hair shafts) after definitive repair. Therecurrence occurred over the scar at the distal aspect of theflap; it was not clear if this recurrence was within the flap ornot. The patient was treated with reexcision and wound care.One patient required prolonged wound care for woundbreakdown. The patient was examined in the operating roomtwice with no evidence of disease recurrence. He wasinitially treated with wet to dry dressing changes. Calciumalginate dressing was also used in an attempt to facilitate

wound healing. He subsequently required reexcision andhyperbaric oxygen therapy at another institution before thewound completely healed 14 months later. The cause ofwound breakdown was not clear but could have beenbecause of unrecognized trauma or wound infection. Sixpatients (37.5%) had minor complications. Of those, 4 (25%)had minimal superficial skin separation that resolvedpromptly with wet to dry dressing change, 1 patient hadcellulitis around the wound that was treated with oralantibiotics and did not require a drainage procedure, and 1patient had residual pain when last seen 3 months post-operatively. Unfortunately, she was lost to follow-up.

Six patients (37.5%) in this study were overweight, asdefined by a body mass index above the 95th percentilefor age and sex. Of the 8 complications, 3 (37.5%) occur-red in overweight patients and consisted of 2 superficial

Table 1 Outcome of RELF in adults

Study n Follow-up(mo)

Recurrence(%)

Woundbreakdown (%)

Superficial woundseparation (%)

Woundinfection (%)

Residualpain (%)

Arumugam et al [2] 53 24 7 10 13 13 -Urhan et al [1] 102 36 5 0 3 1 20Eryilmaz et al [3] 63 25 3 0 0 5 -Akca et al [10] 100 28 0 0 0 2 -

1589Rhomboid excision & limberg flap in pilonidal disease

wound separations and 1 recurrence. There were nowound complications in the one patient who had type Idiabetes mellitus.

3. Discussion

The pathophysiology of pilonidal disease is attributed toloose hair that burrows through moist and macerated skininto the subcutaneous tissue under the mechanical action ofthe rolling of the buttock. The hair is believed to penetrate theskin through a primary sinus and then exits throughsecondary sinuses lateral to midline, secondary to infection[4]. Procedures designed to treat disease without addressingthe primary problem have been shown to have highrecurrence rates. Incision and drainage is usually requiredto treat acutely infected pilonidal sinuses; however, incisionand drainage alone is associated with up to a 92% recurrencerate [5]. Excision of diseased tissue, with or withoutmarsupialization, and packing has a recurrence rate of upto 46% and is associated with high morbidity related to long-term management of an open wound [6-8]. Attempts atprimary closure of wounds after resection of pilonidaldisease have also been shown to be associated with a similarhigh recurrence rate reaching 38% [8-10]. Primary closure isalso associated with high wound complication such aswound infection and breakdown, which occur in up to 78%of cases [6,11,12]. Wound complications can be particularlyproblematic when wide excision is necessary because of themorbidity associated with the large residual defect.

The introduction of excision with flap repair techniques,such as asymmetric excision with advancement flap cover-age (Karydakis flap), V-Y advancement flaps, and the Z-plasty techniques, have had a significant impact ondecreasing recurrence rates. These procedures, however,are not without shortcomings. The Karydakis flap is aneffective technique when dealing with limited disease. Itrelies on a relatively restricted incision that may not be aseffective when dealing with more complex disease [13]. Thedisadvantage of the V-Yadvancement flap is that it results ina midline scar, which in theory places the patients atincreased risk for recurrence. Despite the theoretical risk forincreased recurrence rates, studies have shown promisingresults with this technique, particularly when dealing withrecurrent pilonidal disease [13,14]. The Z-plasty technique is

associated with a 20% rate of flap tip necrosis at the delicateends of the transposed flaps [15]. The introduction of theRELF technique, with its low recurrence and woundcomplication rates, has had promising results in the adultliterature [1-3,10] (Table 1).

The RELF repair has many of the characteristics central toa successful treatment. Specifically, it allows the surgeon tocompletely excise diseased tissue with little concern for thesize and the safe coverage of the residual defect. Theabundant tissue available from the gluteal region, regardlessof body habitus, allowed tensionless coverage of all defectsthat resulted from our excision, the area of one of which was64 cm2. The ability to aggressively excise all diseased tissueis one of the main advantages of this procedure. Oncediseased tissue is excised, the bulky flap that is rotated intothe defect flattens the natal crease, eliminating one of themain predisposing factors to recurrence. The above char-acteristics are the main reasons behind the low recurrencerate noted with this procedure. Despite that we did notincorporate routine shaving of hair in our treatmentalgorithm, as is described in some studies in the adultpopulation, the 6% rate of recurrence noted in our study iscomparable to the 0% to 7% rate of recurrence reported inadult surgical literature [2,3,10].

Another advantage of the tensionless closure is that itresults in minimal pain and discomfort to patients. This isreflected by the short postoperative hospital stay. Althoughmost of our patients where admitted for overnight stay, 3patients (19%) were managed on an outpatient basis becausetheir pain was well controlled with oral analgesics. Theaverage stay of 1.8 days compares favorably with the 3 to 8-day average stay reported in the adult literature [3,12]. As wegain experience with this procedure, we are becoming moreaggressive with day surgery management of our patients.

Wound complications are another factor that plagues themanagement of pilonidal disease. The RELF, throughimmediate and tensionless coverage of the wound with awell-vascularized flap, has resulted in a low rate of woundcomplications. Studies in the adult population have shown awound breakdown rate of up to 10% [2,10]. Empirically, ourpatients are instructed to avoid sitting for 2 weeks and refrainfrom strenuous activity for up to 2 months to decreasetraction forces and trauma to the wound and help reducewound complications. In our study, only one patient had amajor wound breakdown requiring long-term wound care.The cause of this breakdown was not clear but could have

1590 S.Z. Yamout et al.

been because of unrecognized trauma or infection. Fourpatients had minimal superficial wound separation thatresolved promptly with dressing change. In one case, thewound edges separated immediately after removal of the skinsutures. Since then, we have modified our technique toinclude interrupted subcutaneous absorbable sutures to helpmaintain wound integrity after removal of the skin sutures.One patient in our study had a superficial wound infection.This was treated successfully with oral antibiotics on anoutpatient basis. Another patient had residual pain 3 monthspostoperatively. Unfortunately, the current condition of thatpatient is not known because she was lost to follow-up.

Rhomboid excision with Limberg flap is an effectivemethod for the management of pilonidal disease inadolescents. It is easily learned and is a valuable techniqueavailable to the pediatric surgeon, especially when dealingwith complex and extensive pilonidal disease. Although thenumber of patients was limited, the results of our initialexperience with this procedure are encouraging and closelyresemble those of larger studies in the adult surgicalliterature. The follow-up period in our study was relativelyshort, and further studies with a larger patient population anda longer follow-up period are needed to better define the roleof the use of RELF in the management of pilonidal disease inthe adolescent population.

References

[1] Urhan MK, Kucukel F, Topgul K, et al. Rhomboid excision andLimberg flap for managing pilonidal sinus: results of 102 cases. DisColon Rectum 2002;45:656-9.

[2] Arumugam PJ, Chandrasekaran TV, Morgan AR, et al. The rhomboidflap for pilonidal disease. Colorectal Dis 2003;5:218-21.

[3] Eryilmaz R, Sahin M, Alimoglu O, et al. Surgical treatment ofsacrococcygeal pilonidal sinus with the Limberg transposition flap.Surgery 2003;134:745-9.

[4] Karydakis GE. Easy and successful treatment of pilonidal sinus afterexplanation of its causative process. Aust N Z J Surg 1992;62:385-9.

[5] Goodall P. The aetiology and treatment of pilonidal sinus. A review of163 patients. Br J Surg 1961;49:212-8.

[6] Lee S, Tejirian T, Abbas M. Current management of adolescentpilonidal disease. J Pediatr Surg 2008;43:1124-7.

[7] Weinstein MA, Rubin RJ, Salvati EP. The dilemma of pilonidaldisease: pilonidal cystotomy, reappraisal of al old technique. Dis ColonRectum 1977;20:287-9.

[8] Sondenaa K, Nesvik I, Andersen E, et al. Recurrent pilonidal sinusafter excision with closed or open treatment: final results of arandomized trial. Eur J Surg 1996;162:237-40.

[9] Kronborg O, Christensen K, Zimmermann-Nielsen C. Chronicpilonidal disease: a randomized trial with a complete 3-year follow-up. Br J Surg 1985;72:303-4.

[10] Akca T, Colak T, Ustunsoy B, et al. Randomized clinical trialcomparing primary closure with the Limberg flap in the treatment ofprimary sacrococcygeal pilonidal sinus. Br J Surg 2005;92:1081-4.

[11] Sondenaa K, Anderson E, Soreide JA. Morbidity and short term resultsin the randomized trial of open compared with closed treatment ofchronic pilonidal sinus. Eur J Surg 1992;158:351-5.

[12] Abu Galala KH, Salam IM, Abu Samaan KR, et al. Treatment ofpilonidal sinus by primary closure with a transposed rhomboid flapwith deep suturing: a prospective randomized clinical trail. Eur J Surg1999;165:468-72.

[13] Schoeller T, Wechselberger G, Otto A, et al. Definite surgical treatmentor complicated recurrent pilonidal disease with a modified fasciocu-taneous V-Y advancement flap. Surgery 1997;121:258-63.

[14] Khatri VP, Espinosa MH, Amin AK. Management of recurrentpilonidal sinus by simple V-Y fasciocutaneous flap. Dis Colon Rectum1994;37:1232-5.

[15] Bose G, Candy J. Radical cure for pilonidal sinus by Z-Plasty. Am JSurg 1970;120:783-6.