Two staged surgical treatment of a low rectal tumor presented with Fournier’s gangrene: A case report

Introduction: Rectal cancer presenting with Fournier’s gangrene (FG) is a very rare life­ threatening surgical emergency that presents a challenge for clinicians for diagnosis and management. Case Report: We report case of a 57­year­old male patient having a low rectal tumor and presenting with Fournier’s gangrene. The patient first underwent surgical debridement and fecal diversion for treatment of Fournier’s gangrene. Following a fast and successful wound healing with the application of a vacuum assisted wound closure device, surgical removal of the rectal tumor was performed on the 11th day. After receiving chemotherapy and radiotherapy and one year of follow­up, the patient is currently disease free. Conclusion: In our opinion a fecal diversion is necessary in case of Fournier’s gangrene not only to control the source of infection but in cases of Fournier’s gangrene also to provide a chance of applying a vacuum assisted wound closure device which dramatically shortens the duration of wound contraction and granulation in Fournier’s gangrene. After this initial effort is accomplished a definitive surgery can be successfully implemented.


INTRODUCTION
Rectal cancer is the third most common cancer in the Western world. It usually presents with pain, rectal bleeding, tenesmus or alteration in bowel habits [1]. However, rectal cancer may not always present with such classical symptoms. Presence of tumor perforation below the peritoneal reflection might cause a clinical picture resembling Fournier's gangrene (FG). [2,3] Fournier's gangrene is a necrotizing fasciitis of the perineal, perianal or genital areas and associated with high morbidity and mortality. Although the presentation of rectal cancer in the form of Fournier's gangrene is a rare phenomenon, this lifethreatening condition requires urgent, radical surgical debridement and administration of broad spectrum antibiotics [4,5]. In the majority of previously reported colorectal tumor cases presenting with FG, the first goal of the therapy was achieving infection control with surgical debridement and eventually fecal diversion. However, despite appropriate treatment only few patients have survived and undergone chemo or radiotherapy without tumor resection [6][7][8][9]. Through this case report, we highlight the importance of recognizing and managing of FG early in the setting of low rectal cancer. We present a twostage surgical strategy for the treatment of FG and its successful outcome after one year clinical followup.

CASE REPORT
A 57yearold male patient was referred to our hospital for severe scrotal swelling and pain. Patient had no history of a systemic disease, substance abuse or any other condition that could alter his immune status. Physical examination on admission revealed high fever (38, 7°C), hypotension (80/60 mmHg) and tachycardia (130/min). The skin of the scrotum and perineum was swollen. The patient had an edema that had spread in the inguinal regions bilaterally and extended up word and laterally along the inner surface of the left thigh. A foulsmelling purulent discharge from the necrotic skin in the scrotum was also present ( Figure 1). Digital rectal examination revealed an irregular mass filling the left side of the lower rectum that was three cm in length and extending to the dentate line distally. Additionally, patient had grade IV internal hemorrhoids with thrombosis. The laboratory tests showed high white blood cell count (22x10 3 /mm 3 ), low hemoglobin (7.8 g/dL) and low albumin (1.8 g/dL).
Following the diagnosis of FG, broad spectrum antibiotics for empiric treatment were commenced and the patient underwent emergency surgery. All necrotic tissue was removed quickly and aggressively ( Figure 2). Both testes were preserved and left in place. The removal of the tumor was deferred to a later date. Because of the patient's septic shock and poor general condition. A laparotomy was performed for fecal diversion by a low midline incision. Following a fast abdominal exploration which revealed no abnormal finding above the peritoneal reflection, sigmoid colon was diverted by a linear stapler (Autosuture Endo GIA, Covidien, Mansfield, MA, USA) and an end sigmoid colostomy was performed to ensure the infections source control.
The patient was followed in the intensive care unit for the first two postoperative days. Extubation was done the next morning of surgery and oral nutrition was allowed in the evening of first day. On the first postoperative day in intravenous infusion 50 mL, 20% human albumin every 12 h was started and continued for three days. A referred was sent to infection diseases clinic on postoperative day2 and the previously given empiric antibiotics (imipenemcilastatin sodium 500 mg intravenously every 6 h, metronidazole 500 mg intravenously every 8 h and clindamycin 900 mg every 8 h) were continued for 10 days according to their recommendations.
In addition to bedside minor debridement, vacuum assisted wound closure device was applied on the postoperative day2 with local anesthesia (Figure 3).
The polyurethane sponge of this device was changed every other day and the wound was regularly inspected. After four applications of this sponge, on postoperative day10, the defect on the pelvic floor was completely covered with a clean granulation tissue (Figure 4).
Meanwhile the patient underwent total colonoscopy, abdominal computed tomography scan and pelvic magnetic resonance imaging. Biopsies obtained at colonoscopy confirmed the diagnosis of rectal adenocarcinoma. Tomography and magnetic resonance imaging revealed no findings which would contraindicate the resection. Patient's general condition was fine and he could undergo the Miles procedure on postoperative day11. The space between sacrum and posterior wall of the rectum where mesorectum should exist was hardened with fibrotic tissue but the dissection was carried out as in total mesorectal excision. The defect on perineum and both testes was closed by the skin flaps derived from the medial surfaces of both thighs following the abdominoperineal resection ( Figure  5). Patient's postoperative course was uneventful. The  pelvic suction drains were removed on postoperative day3 and the patient was discharged on postoperative day14.
In the histopathological report, the tumor was found to be stage T3NX, moderately differentiated adenocarcinoma with negative surgical margins. Despite through gross examination no lymph nodes could be identified in the resected specimen histologically. The patient was referred to medical and radiation oncology clinics where he received six cycles of adjuvant chemotherapy with fluorouracil and folinic acid. During the second cycle of chemotherapy, pelvic radiotherapy was also given as adjuvant. After one year of followup, the patient is currently disease free as evidenced by computed abdominal tomography and total colonoscopy.

DISCUSSION
In majority of the previously reported colorectal tumor cases presenting with FG, only few patients have survived the infection and undergone further chemo or radiotherapy without any tumor resection. Of these, in only one exceptional paper reported by Carr et al. in 2010, a case presenting with a very similar clinical picture to our patient plus having a diagnose of rectal carcinoma underwent aggressive debridement and abdominoperineal resection simultaneously [10]. The patient survived, but Carr et al. underlined that the surgical decision of abdominoperineal resection was challenging in the presence of such a serious infection.
We did not choose to go through an abdominoperineal resection at first operation as the patient was showing signs and symptoms of severe septic shock. Moreover, we did not have a confirmed diagnosis of rectal malignancy to justify such a radical surgery.
It was unusual that pathologists could not identify any lymph node in the gross specimen. But at the first debridement the soft tissue surrounding the rectum was removed circumferentially and there were barely any soft tissue left till the proximal section of the sacrum   near promontorium. We think that this could be a reason for not finding any lymph nodes in gross specimens.
Although there are controversies for fecal diversion in the treatment of FG, if a preventive colostomy is planned, it should be performed during the initial debridement [11]. In our opinion, a fecal diversion was necessary not only to control the source of the infection but also for the ease of implementing a vacuum assisted wound closure device which accelerated the formation of a healthy granulation tissue safely [12].

CONCLUSION
In this case report the successful outcome of a two staged surgical approach for the treatment of a rectal tumor with Fournier's gangrene is presented. We think that a fecal diversion is necessary to control the source of infection and to provide a chance of applying a vacuum assisted wound closure device which dramatically shortens the duration of wound contraction and granulation. After this initial surgery, a through and definitive surgery can be successfully performed. *********

Author Contributions
Osman

Guarantor
The corresponding author is the guarantor of submission.