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ORIGINAL ARTICLE
Year : 2023 | Volume
: 31
| Issue : 1 | Page : 8-12
Does surgical treatment for complicated pilonidal cyst disease in the sacral region affect anal sphincter functions
Mustafa Akyurek1, Caghan Benli1, Mustafa Kaya2, Ali Surmeli3
1 Department of Plastic Reconstructive and Aesthetic Surgery, Canakkale Onsekiz Mart University, Canakkale, Turkey
2 Department of Surgical Oncology, Dr. Ersin Arslan EAH, Gaziantep, Turkey
3 Department of Gastroinstestinal Surgery, University of Health Sciences, Haydarpasa Numune Training and Research Hospital, Istanbul, Turkey
Correspondence Address:
Dr. Mustafa Akyurek
Canakkale Onsekiz Mart University Hospital, Canakkale 17100
Turkey
Source of Support: None, Conflict of Interest: None
DOI: 10.4103/tjps.tjps_44_22
Introduction: Pilonidal cyst disease is characterized by chronic abscess, discharge, and sinus openings. The anal sphincter functions of the patients, who previously undergone recurrent surgical treatment, were compared using a surgical approach. Patients were divided into two groups by whether perianal soft tissues were preserved in the surgery. Materials and Methods: Between 2014 and 2019, 21 male patients who had either undergone insufficient surgery or had complications with multiple sinus openings applied to our clinic. Excision and reconstruction of the patients were performed. The coccyx, dorsal part of sacrum, anococcygeal raphe, and ischiorectal fossa were additionally included in the excision for patients with biopsy reports of squamous cell carcinoma. All patients underwent rectoanal manometry to evaluate anal sphincter function. Preoperative, postoperative 5th week, and 6th-month anal manometry results were recorded and analyzed by the Shapiro − Wilk test and independent t-test. Results: The anal sphincter function regresses in the 5th week after the reconstruction and improves in the 6 month. The results of the independent t-test, which were used to compare the anal manometry results for each group, showed that the difference in the anal sphincter pressure of the two groups was statistically significant (P < 0.05). If the perianal soft tissues were excised, anal sphincter manometry shows poor results. Discussion: The anal sphincter function, which regresses in the 5th week after the reconstruction and improves in the 6 months, depends on the scar tissue occurring in the perianal connective tissue, while the softening caused by the maturation of the scar in the last phase of wound healing improves the function.
Keywords: Anal manometry, pilonidal cyst, sphincter function
Pilonidal cyst disease, which was first described by Hodge in 1880, is a pathology characterized by chronic abscess, discharge, and sinus openings. Pilonidal cyst disease occurs most frequently in the superior intergluteal cleft and in young adult males. The etiology is linked to younger age, male gender, obesity (body mass index >25 kg/m2), a deep gluteal cleft, lack of hygiene, being hairy, and Mediterranean ethnicity.[1],[2] The prevalence of the disease is 6.6% in Turkey.[3],[4] In patients who are not treated correctly, there is a possibility of recurrence, sinus formation, and malignant transformation arising from the pilonidal cyst. The aim of this study is to discuss the prognosis and functional results of 21 cases that presented to our clinic with complicated recurrent pilonidal cyst disease. The anal sphincter functions of the patients were compared using a surgical approach. Patients were divided into two groups by whether perianal soft tissues were preserved in the surgical treatment.
Materials and MethodsBetween 2014 and 2019, 21 male patients who had either undergone insufficient surgery or had complications with multiple sinus openings applied to our clinic. Their ages ranged from 33 to 52, with a mean age of 41.4. Incisional biopsies were taken from open wounds and scar tissue around the pilonidal cysts and sent for the pathological examination. Preoperative anal manometry was performed and recorded. To perform unconstrained postoperative infection control for the patient, the laparoscopic loop sigmoidostomy procedure was performed by the general surgery department in patients who were diagnosed with squamous cell carcinoma (SCC) or were suspect, during clinical evaluation, for inadequate tumor-free margin excision. Once tumor-free margins were obtained, reconstructions were performed for all cases by the plastic reconstructive and esthetic surgery (PRAS) department. Excision and reconstruction of non-SCC diagnosed patients were performed in a single stage by the PRAS department. To determine the excision margin for non-SCC diagnosed patients, methylene blue dye was applied to the area around the opening of the sinus. The postoperative periodic follow-ups were performed in conjunction with the general surgery department. In the postoperative 5th week, and again in the 6th month, anal manometry was performed and recorded to evaluate the functional results of the operations. The patients were followed up for a mean of 4 years. Follow-ups were performed by physical examination, and contrast computed tomography (CT) was undertaken at 3-month intervals.
All patients underwent rectoanal manometry, by means of 8-water perfusion, to evaluate anal sphincter function (Dynosmart (mode 1), Menfis (medica) Italy). Preoperative, postoperative 5th week, and postoperative 6th month anal manometry results were recorded and analyzed using IBM SPSS software (IBM Corp., Armonk, N. Y., USA). The Shapiro − Wilk test and independent t-test were run for the analysis of the data.
ResultsSix patients were diagnosed with SCC and seven patients with pseudoepithelial hyperplasia. The operations were performed under spinal anesthesia. Excision and reconstruction procedures were performed in the same session for patients with a high risk of malignancy and those with pseudoepithelial hyperplasia. Excisions for these patients were completed with the excision of the perianal soft tissue in the deep plane. Both patient groups were reconstructed with two musculocutaneous flaps with a V-Y advancement pattern from the gluteal regions. Six of the patients' final pathology reports indicated SCC, while the others had pseudoepithelial hyperplasia and/or chronic inflammation. The patients were hospitalized for 5 days in the postoperative period, hemovac drains were removed at the end of the 5th day, and they were then discharged. No infection, wound dehiscence, or discharge was observed during the follow-up. At the end of the 3rd week, activities such as sitting and driving were allowed.
The coccyx, dorsal part of sacrum, anococcygeal raphe, and ischiorectal fossa were additionally included in the excision for patients with the biopsy reports of SCC. If these parts were included in the excision, the patients were grouped as the perianal soft-tissue excised group. Reconstruction was postponed to the second session since tumor-free surgical margins could not be assured in the first session. At the end of the excision, the posterior parts of the anus muscles and the ampulla of the rectus were left exposed. Following the excisions, the anal sphincter and erectile functions of all patients were normal. Wound care was performed with moist dressings until a definitive pathologic diagnosis was obtained. In the postoperative 2nd week, the excision margins were concluded to be tumor-free, and the patients were reconstructed with either the Limberg flap (including contralateral), the bilateral V-Y closure, or the musculocutaneous rotation flap. While the digital rectal examination showed adequate contraction in all patients in the early postoperative examination, in the postoperative 5th week controls, the digital rectal examination indicated weakening in the anal sphincter function, and rectoanal manometry was ordered to evaluate the results objectively. In the postoperative 6th month, anal sphincter functions were determined as sufficient and the sigmoidostomy was closed. The erectile functions of the patients did not decrease in the early or late postoperative periods.
Patients diagnosed with SCC were referred to the oncology clinic after wound healing was completed. The patients who refused to receive radiotherapy were followed up with clinical and radiological examinations. One SCC-diagnosed patient died during the 3rd month, while the other five patients diagnosed with SCC had a mean of 4 years follow-up. Four of them did not show any local recurrence or distant metastasis, but one of them showed recurrence at the 1-year follow-up.
Anal sphincter examination of patients in the preoperative period showed that the resting anal canal pressure mean, which reflects to a large extent the internal sphincter pressure, was 56.10 mmHg (normal: 40–70 mmHg). If we exclude the two patients whose anal manometry measurement could not be followed up in the postoperative period, the preoperative mean was 55.79 mmHg. During the anal sphincter examination of patients in the postoperative 5th week, the sphincter muscles were found to be weakened. The resting anal canal pressure mean, which reflects to a large extent the internal sphincter pressure, was 34.79 mmHg. If the perianal soft tissue was excised during the debridement, we found that internal sphincter pressure had decreased to 30.89 mmHg, while the rest of the patients, with non-excised perianal soft tissue, showed 38.3 mmHg internal sphincter pressure.
In the postoperative 6th month, mean resting anal canal pressure had increased to 45.89 mmHg. While the perianal soft-tissue excision group showed 41.67 mmHg, the rest of the patients, with nonexcised perianal soft tissue, showed 49.7 mmHg resting anal canal pressure. In the voluntary squeeze test, which reflects the external sphincter pressure, the mean sphincter pressure increased to 94.26 mmHg. The perianal soft-tissue excision group and the patients with nonexcised perianal soft tissue showed 88.33 mmHg and 99.6 mmHg, respectively. Normally, external sphincter pressure is expected to exceed 100 mmHg; however, in the perianal soft-tissue excision group, the maximum voluntary squeeze pressure was 98 mmHg; hence, in these patients, it was considered to be slightly low. In the cough test, the anal sphincter pressure mean was 97.1 mmHg and 76.44 mmHg for the perianal soft-tissue excision group and the patients with nonexcised perianal soft tissue, respectively. The mean cough test pressure for all patients was 87.32 mmHg. Normally, in the cough test, anal sphincter pressure is expected to exceed the external sphincter pressure; however, it was measured as lower, especially in the perianal soft-tissue excision patients. In light of these anal manometry measurement results, partial injury of the external anal sphincter was suspected. Endoanal ultrasonography was therefore performed on all patients. In the endoanal ultrasonography, a partial defect was assessed in the subcutaneous portion of the external sphincter. These defects were evaluated as injuries in the subcutaneous portion of the external anal sphincter that did not impair a patient's quality of life.
For the data analysis, two patients were excluded from the data pool since one had died during the 3rd month postoperatively, and the colostomy reversal had been unsuccessful for the other. Therefore, anal sphincter functional measurement results were not available for those two patients. Data from the remaining 19 patients were evaluated using the SPSS software. The results of the Shapiro − Wilk test showed that the data had a normal distribution, and hence, the parametric independent t-test was used to compare the anal manometry results of the two groups for whom different surgical approaches had been used [Table 1]. The results of the independent t-test, which were used to compare the anal manometry results for each group, showed that the difference in the anal sphincter pressure of the two groups was statistically significant (P < 0.05) [Table 2].
Table 1: The results of the Shapiro-Wilk test showed that the data had a normal distribution
Table 2: The results of the independent t-test, which were used to compare the anal manometry results for perianal soft tissue excised and nonexcised group Discussion Cases of squamous cell cancer arising from pilonidal cysts are rare. In the literature review, using the PubMed biomedical database, 83 case reports were found between 2000 and 2018.[5] This condition is called a Marjolin ulcer and it takes from 6 to 42 years from the onset of the inflammation to the diagnosis.[6] In our clinical study, the patients who were diagnosed with pseudoepithelial hyperplasia had already had the disease for a mean of 10 years, while in the cases diagnosed with SCC, the patients had each undergone at least five different ineffective surgeries and the disease had existed for at least 12 years. Neglect by patients can lead to this problem as an incorrect or incomplete surgical procedures.
The effectiveness of chemotherapy and radiotherapy is limited in patients with SCC arising from pilonidal cysts. The main treatment is the excision of the lesion with a wide tumor-free surgical margin.[7] In our clinical experience, the tumor-free excision margin needs to be 2 cm. The base margin was determined by evaluating preoperative contrast tomography. In cases where the radiologic bone invasion was detected, the coccyx, the posterior part of the sacrum, and perianal soft tissue were included in the excision. Prophylactic inguinal lymphadenectomy is not recommended in patients with SCC arising from pilonidal cyst. In a small series study, the incidence of inguinal lymph metastasis was found to be 14%.[8] In addition, prophylactic lymph node dissection may cause lymphedema in the lower extremities.
As the area with tumor suspicion increases, the possibility of missing the tumor in the biopsy samples increases. In addition, there is a possibility of another type of malignancy that arises from pseudoepithelial hyperplasia, which can be falsely identified as SCC.[9] In order to plan the operation precisely and to define the surgical margins, we took incisional biopsy samples before the surgery. The patients whose incisional biopsy results indicated pseudoepithelial hyperplasia were treated as though for SCC and the tumor-free excision margin was determined at 2 cm. In addition, distant metastasis screening was performed in these patients before surgery. In the postoperative period, patients with pseudoepithelial hyperplasia were followed up as frequently as the patients who were diagnosed with SCC.
For the reconstruction, each patient was evaluated individually for their particular needs and healing process. Since all of our patients had experienced recurrence and more than one operation, primary closure was not an option. We performed the Limberg flap (including contralateral), bilateral V-Y closure, and musculocutaneous rotation flap according to the particular patient's reconstructive need [Figure 1]. The blood supply to the Limberg flap comes from many small arteries; the majority of which originate from the lateral sacral and superior gluteal arteries and their perforators.[10] During the excision and debridement of the pilonidal cyst, if either the superior gluteal artery perforators, the lateral sacral artery perforators, or the piriformis muscle was damaged, bilateral V-Y closure or musculocutaneous rotation flap was used for reconstruction according to the size and location of the defect. Under large debridement and defect conditions, bilateral V-Y closure or musculocutaneous rotation flaps are raised from unnamed small perforators. Since reconstruction choices depend on excision and debridement outcomes, optimal reconstruction methods are evaluated during surgery. Horwood et al. show that the Limberg flap is superior to primary closure with lower wound dehiscence and surgical site infection.[11] In addition, the Limberg flap is found to be superior in pilonidal cyst disease reconstruction by having lower acute abscess rates.[10]
Figure 1: Bilateral V-Y closure for reconstruction after excision of squamous cell cancerUsing the gluteus maximus muscle to cover large sacral defects is very practical and successful. According to the Mathes–Nahai classification, the gluteus maximus muscle is classified as type III, namely as having two dominant pedicles, only one of which is necessary to supply the muscle; therefore, it can be elevated individually whether inferior or superior gluteal artery-vein-based. In addition, the fascio-cutaneous advancement flap with V-Y closure can be successfully used to cover pressure sores in the sacral region. In all of our cases, excisions were made at 90° angles to the base and, where necessary, including the posterior part of the sacrum. Therefore, the defect to be covered is deeper and wider than the defect formed after the debridement of pressure sores. In our reconstruction selection, the superior gluteal artery-based gluteus maximus muscle was included in the flaps to prevent the formation of dead space under the flaps and to provide a better blood supply to the skin.
Creating a stoma for fecal diversion in patients with planned surgical intervention due to Fournier gangrene, full-thickness burns in the perineum, or a complicated perianal fistula positively affects wound care in the perianal region during the postoperative period. To prevent fecal contamination of the wound and to have better infection control, wound care was performed for 2 weeks. Consequently, the reconstruction of the tumor-free surgical margin was left to a second session.
The use of prophylactic antibiotherapy is still a controversial subject. Mavros et al. showed that there is no difference in the healing outcomes between single-dose prophylaxis and no prophylaxis.[12] Prophylactic antibiotherapy was not used in this study. In addition, there is no difference in wound healing time by using negative pressure wound therapy after the surgery.[13] Removing body hair can decrease the recurrence rate of pilonidal cyst disease after surgery.[14]
To assess the success of pilonidal sinus treatment, different factors can be evaluated, such as healing time and process, complications during healing, recurrence rate, time to return to normal life, and number of operations needed to achieve cure.[15] In this study, we focused on recurrent complicated pilonidal cysts, and as a novel approach, we evaluated the functional outcomes of the patients.
Certain surgical considerations are necessary in the perianal region due to the abundance of fibroelastic tissue. Normally, the connective tissue network moves together with the sphincter muscles. However, scar formation stiffens the web; hence, it cannot follow muscle movements. Serious deformities may develop if the scar contracts, with rigid scars and deformities preventing normal sphincter function. “Stretching” of the sphincter involves stretching of the connective tissue networks rather than the stretching of the muscle fibers. While the mesh can be easily stretched, the many small tears left in the fibroelastic fibers of the web usually heal with scar formation and dysfunction. Removing the perianal soft tissue during debridement causes rigid scars and contractions in the perianal region. The impaired function does not necessarily result in incontinence, but it can contribute to this situation.[16] Since the removal of perianal soft tissue causes serious outcomes, extending the healing process and preventing single-stage closure and reconstruction, if we do need to debride these soft tissues, patients must be informed clearly about the process. In particular, patients who have undergone primary debridement in another clinic must be given information about the healing process since the patient might associate scar formation and fibrosis in connective tissue sequalae, such as anal sphincter dysfunction, with the reconstruction operation. However, these are late complications and presentations of the primary debridement, occurring when the maturation of the scar is completed.
Maturation of the scar tissue occurs during the last phase of wound healing. During this period, rigid scars become soft and obtain their final form. In our cases diagnosed with squamous cell cancer, an open area, of approximately 120° at the posterior part of the anus, was observed after excision [Figure 2]. The open area was treated with a moist dressing for 2 weeks, and it was covered with granulation tissue before the reconstruction operation. In conclusion, the anal sphincter function, which regresses in the 5th week after the reconstruction and improves in the 6 months, depends on the scar tissue occurring in the perianal connective tissue, while the softening caused by the maturation of the scar in the last phase of wound healing improves the function. However, none of the patients showed a higher postoperative value in their anal manometry measurements than preoperatively. The healthcare providers and the patient must agree unambivalently about the healing process, and patients must be informed about the possible temporary or permanent anal sphincter dysfunction due to scar tissue. Informing patients has important implications for patient compliance and legal obligations.
Figure 2: Typical view after excision of patients diagnosed squamous cell cancer, an open area, of approximately 120° at the posterior part of the anusHighlights
Pilonidal cysts disease patients were divided into two groups by whether perianal soft tissues were preserved in the surgical treatmentAll patients underwent rectoanal manometry to evaluate anal sphincter functionAnal sphincter function regresses in the 5th week after the reconstruction and improves in the 6th monthSoftening due to maturation of the scar in the wound healing improves the function.Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
References
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