Squamous cell carcinoma arising from chronic sacrococcygeal pilonidal disease: a case report

Abstract

BackgroundSacrococcygeal pilonidal disease is a chronic, well-recognized entity, characterized by the recurrent formation of an abscess or draining sinus over the sacrococcygeal area. It is one of the most common surgical problems. Rarely, chronic inflammation and recurrent disease leads to malignant transformation, most commonly to squamous cell carcinoma (SCC).Case presentationWe describe an extremely unusual case of SCC developing in a 60-year-old patient with a chronic pilonidal sinus complicated by an anal fistula. After wide surgical excision of the pilonidal sinus and fistulas and because of the poor healing process 6 months later, colonoscopy and a percutaneous fistulography were performed, revealing an anal canal-pilonidal fistula. Patient was treated with a more radical surgical resection with a prophylactic loop colostomy, but healing was not accelerated. Multiple biopsies were then taken from the surgical site at the time, which revealed the development of SCC. CT and MRI imaging techniques revealed SCC partial invasion of the coccyx and sacrum. As a result, aggressive surgical approach was decided. Histological examination revealed moderately to poorly differentiated SCC, and the patient was treated with adjuvant radiation therapy postoperatively. Nine months later, recurrence was found in the sacrum and para-aorta lymph nodes and the patient died shortly after. We discuss the clinical features, pathogenesis, treatment options, and prognosis of this rare malignant transformation.ConclusionsThe development of SCC in chronic pilonidal disease is a rare but serious complication. Symptoms are usually attributed to the sacrococcygeal pilonidal disease (SPD), and diagnosis is often made late by histological examination of biopsies. Malignant transformation should be suspected in chronic SPD with recurrent episodes of inflammation, repeated purulent discharge, poor healing, and chronic complex fistulas.

Karydakis Flap: A preferred procedure for Sacrococcygeal Pilonidal Sinus Disease (SPNSD).

“…Loose hair within sacrococcygeal area due to friction force between buttocks tend to invade the moist natal cleft skin leading to formation of abscess and sinus, so complete excision of the sinuses is necessary to avoid recurrence. 7 Many surgical procedures have been described in the literature to treat this disease but every technique has its own pros and cons. These procedures can vary from simple wide excision and thus leaving the wound open for secondary healing, to simple excision with midline closure, asymmetrical closure, marsupialization, Bascom repair and different flaps procedures like W plasty, Z plasty, VY plasty, Limberg flap and modified limberg flap.…”

Would Marjolin see it coming? Two unusual cases of squamous cell carcinoma

“…There is a paucity of literature describing Marjolin's ulcer cases in the setting of pressure sores of spina bifida patients or epidermal inclusion cysts [ 11 , 20 , 24 , 25 ]. Hence, the natural history of the ulcerated tumors presented in these two cases remains a mystery.…”

“…There are numerous Treatment options for low risk SCC tumors [ 26 , 27 ] including surgical excision, cryotherapy, electrosurgery, topical treatment, radiation and photodynamic therapy. The risk of recurrence and metastasis is important for determining the appropriate treatment [ [13] , [14] , [15] , [16] , [17] , [18] , [19] , [20] , [21] , [22] , [23] ]. Marjolin’s ulcer falls into the category of high risk tumors.…”

“…In these cases, surgery is still the cornerstone of treatment with adjuvant chemotherapy and radiation if necessary. Although the prognosis is excellent for low risk tumors, high risk lesions are associated with increased mortality and require lifelong monitoring, such as the two cases presented here [ [17] , [18] , [19] , [20] , [21] , [22] , [23] , [24] ].…”

“…The patient specific treatment plan is based on a risk assessment for local recurrence and metastasis [ [13] , [14] , [15] , [16] ]. Regional lymph node involvement and metastases are rare and associated with increased mortality [ [17] , [18] , [19] , [20] , [21] ]. Although the five-year cure rate is greater than 90%, all patients require long term oncologic follow up even after successful treatment.…”