台灣癌症醫誌 (J. Cancer Res. Pract.) 28(1),41-46, 2012 Case Report A 42-year-old man presented with a four-day hisjournal homepage:www.cos.org.tw/web/index.asp Necrotizing Fasciitis of the Lower Limb Secondary to Perforated Colon Cancer Min-Yao Chuang 1, Chien-Kuo Liu 2, Te-Yang Huang 1 * 1 Department of Orthopaedic Surgery, Mackay Memorial Hospital, Taipei, Taiwan 2 Division of Colorectal Surgery, Department of Surgery, Mackay Memorial Hospital, Taipei, Taiwan Abstract. Perforated colon cancer commonly occurs intraperitoneally and seldom presents without abdominal pain, better enabling the clinician to make a timely and accurate diagnosis. On the other hand, necrotizing fasciitis of the lower limb from perforated colon cancer is rare and difficult to diagnose. Clinically, presentation of the above two conditions together is extremely unusual and typically difficult to treat. We report a case of necrotizing fasciitis of the left lower limb in a middle aged male that is the result of perforated colon cancer. 病例報告 Keywords : necrotizing fasciitis, colon cancer, bowel perforation 結腸癌破裂造成的下肢壞死性筋膜炎 莊閔堯 1 劉建國 2 黃德揚 1 * 1 馬偕紀念醫院骨科 2 馬偕紀念醫院大腸直腸外科 中文摘要大腸癌破裂常發生於腹膜內, 但少見無伴隨腹痛症狀 另一方面, 大腸癌造成的下肢壞死性筋膜炎為罕見且難以診斷的疾病 臨床上合併上述兩種狀況為極稀有且難以治療的 在此報告一位以左下肢壞死性筋膜炎為初始表徵的大腸癌破裂中年男性患者 關鍵字 : 壞死性筋膜炎 大腸癌 腸道破裂 INTRODUCTION Necrotizing fasciitis is a fulminant gangrene which arises from synergistic polymicrobial infection with aerobic and anaerobic bacteria [1,2]. It most commonly occurs after a dermal wound, and its development is accelerated in immunosuppressed patients, or in cases of cancer or diabetes [2,3]. Rare cases of necrotizing fasciitis due to underlying bowel disease have been reported. However, the course of disease is often unfavorable, particularly when bowel perforation occurs [4]. We report an unusual case with perforated colon cancer presenting as necrotizing fasciitis of the lower limb. CASE REPORT
42 M. Y. Chuang et al./jcrp 28(2012) 41-46 tory of pain and swelling in his left thigh and lower leg, who lacked any symptoms of bowel habit change, anorexia, abdominal pain, or body weight loss. The patient s brother sent him to our emergency room with initial clinical signs as follows: afebrile at 36.0 C, tachycardia at 129 beats/min, and normotension at 155/71 mm-hg. The medial and posterior portion of his left lower limb were erythematous, with apparent tenderness. Palpable crepitus was also noted. The abdomen was soft without tenderness or rebounding pain. Laboratory investigation showed a white blood cell count of 60,600/mm 3 with 70% neutrophils and 15% bandemia, and a high C-reactive protein level of 45.61 mg/dl. Plain X-ray revealed extensive gas in the soft tissues of his left thigh and down below the left knee. The presence of gas was dominant in the subcutaneous tissue and deep within the lower limb, delineating a longitudinal structure (Figure 1). We performed emergency exploratory surgery under the preliminary impression of necrotizing fasciitis. A long longitudinal incision was made from the medial aspect of the patient s left thigh to the posterior aspect of the left calf. During the operation, an offensive gas-forming infection of the deep fascia extending between the muscles of the medial compartment of the left thigh and the superficial posterior compartment of lower left leg was disclosed. All of the involved skin, fascia and muscle components were excised by Fillet procedure (Figure 2). Blunt dissection of the proximal aspect of the wound showed a tract around the inguinal area which communicated with the pelvis, and an abdominal source of infection was suspected. However, due to his unstable blood *Corresponding author: Te-Yang Huang M.D. * 通訊作者 : 黃德揚醫師 Tel: +886-2-28098324 Fax: +886-2-25433642 E-mail: doctorcat@gmail.com pressure and noted critical condition, the patient was sent to the intensive care unit, where the wound was kept open with wet gauzing. Microbiological testing showed profuse growth of Escherichia coli, beta- Streptococcus non-abd and anaerobes. Additionally, pathology confirmed necrotizing fasciitis, and the patient was treated with broad-spectrum antibiotics. In order to evaluate the patient s intraabdominal condition, a complete abdominal computerized tomography (CT) was performed. This testing showed that the colonic luminal structure disappeared at the left side lower abdomen, with an accumulation of abnormal air density extending to the left inguinal region, suggesting perforation of the descending colon (Figure 3A, 3B). Four days after the first operation, follow-up laparotomy revealed a tumor wrapped in the omentum found in the left lower quadrant of the patient s abdomen. Due to the location of the tumor, it was difficult to obtain an adequate biopsy at the time; however, a transverse-colon loop colostomy was performed. Then, a sigmoidoscopic biopsy revealed an adenocarcinoma of the descending colon. Seven weeks after the patient was admitted, a left hemicolectomy with Hartmann s procedure was performed. Pathology revealed moderately differentiated mucinous adenocarcinoma without lymph nodes metastasis, classified as T4N0M0. Ten weeks after the initial orthopaedic surgery, the patient s thigh wound was covered with split-thickness skin grafts and locally advanced skin flap. Finally, the patient was discharged and could walk with crutches. DISCUSSION The incidence of perforated colon cancer ranges from 3% to 10% [5]. However, perforation without acute abdominal symptoms and signs, as seen in this patient, is rare. Typically, perforated colon cancer is found intraperitoneally, and rarely extends into the retroperitoneum [6]. Patients with retroperitoneal infection commonly present with vague complaints of
M. Y. Chuang et al./jcrp 28(2012) 41-46 43 Figure 1. Left thigh and lower leg radiograph shows subcutaneous and intermuscular gas (arrows) pain in the lower abdomen, flank, back, hip or thigh, intestinal distension, abdominal mass, general weakness, and fever [7]. Additional symptoms may include weight loss, malaise, hip and back motor power weakness, chills, scoliosis and anorexia. It has been noted that only 20% of all retroperitoneal infections involved a positive psoas sign [8], which may explain the lack of clinical symptoms in this patient. However, necrotizing fasciitis of the thigh due to the spread from perforated colorectal cancer is extremely rare. Only five such cases had been reported in previously published articles [6,9-12]. To our knowledge, a report of colon cancer perforation causing necrotizing fasciitis of the thigh and the calf similar to our case had not been previously presented. The large amount of gas present in the tissues of
44 M. Y. Chuang et al./jcrp 28(2012) 41-46 Figure 2. Photographs show extensive debridement of the left thigh medial compartment and lower leg posterior compartment A the lower limb was also speculated to be secondary to the escape of bowel gas, and not solely due to the presence of gas-forming organisms [13]. Several routes have been described as conduit pathways for retroperitoneal lesions to the lower extremities [14]. The psoas sheath, femoral sheath, femoral canal, sacrosciatic notch, and obturator foramen may all serve as routes of entry [15-17]. Extravasated air from the perforated bowel may travel along these routes as well, and contribute to emphysema produced by bacterial metabolism [17]. The location of the infection may provide some clues to help identify its route of spread. In one review of thigh abscesses secondary to abdominal infections, the majority of these abscesses were located in the anterior and medial aspects of the thigh, indicating spread via the psoas and femoral sheath, and the obturator foramen; but a small number of posterior abscesses have been documented as well, indicating that spread can occur via the sacrosciatic notch [14]. According to the clinical findings, CT images, and intraoperative gross observation in our patient, we hypothesize that the infection was transmitted by direct extension from the retroperitoneal area, and that the bacteria traveled along the psoas or femoral sheath and passed through the obturator foramen B Figure 3. Axial view of CT images shows (A) disappearance of colonic luminal structure with a hypodense mass (arrow) over the left lower quadrant of the abdomen, and (B) fluid and gas (arrow) tracking from the retroperitoneum into the intramuscular plane of the grossly enlarged left thigh to the lower limb. Perforated colorectal cancer exposes the patient to a double risk: tumor spread or sepsis secondary to peritonitis and, less commonly, pelvic inflammation, which can be complicated by perineal cellulitis or necrotizing fasciitis. The surgical treatment of perforated
M. Y. Chuang et al./jcrp 28(2012) 41-46 45 colon cancer is usually resection of the colon with colostomy [18]. In a retrospective analysis of 1551 colorectal cancers [19], 3.3% of the patients had localized perforation, and 61% of these patients had localized abscess. In that study, the overall operative mortality was 12%. In cases of colorectal cancer complicated by necrotizing cellulitis, Chow et al. have reported an operative mortality rate of 50% [4]. Necrotizing fasciitis is a progressive, rapidly spreading infection affecting the subcutaneous fat, the superficial fascia (a cardinal feature), and the deep fascia (to a varying degree). It can be idiopathic, secondary to trauma of the skin, such as insert bites, acupuncture, needle injection, surgical wound infection, and intra-abdominal infections. General risk factors include immunosuppression, malignancy, diabetes, malnutrition, alcoholism, obesity, chronic alcoholism, drug abuse and old age. The causative organism may be aerobic, anaerobic or mixed flora. Group A betahemolytic Streptococcus species or a Clostridium species are frequently the initiating bacteria. The presence of gas-forming organisms leads to classically described subcutaneous emphysema. Necrotizing fasciitis can be difficult to recognize in the early stages, but is rapidly progressing and has an elevated mortality of 20% to 40% [20], especially in the perineal location originating from an intestinal source [4]. The affected area is initially very painful, but otherwise lacks visible change. The tissues become erythematous, swollen and necrotic with palpable crepitus, and the patient typically suffers from complicating septic shock. Two distinctive clinical entities of necrotizing fasciitis are recognized: hyperacute and subacute variants [21]. Hyperacute necrotizing fasciitis usually manifests as a rapidly progressing infection with resultant multi-organ failure due to group A streptococcus or clostridium species infection. Contrarily, polymicrobial infection is implicated in subacute necrotizing fasciitis that represents a slowly evolving disease with less fulminant systemic complications. Our patient was classified as subacute group according to the clinical course and microbiological examination. We suggest that is the reason the patient postponed medical assistance for four days. This delay further enabled the bacteria to translocate along the thigh to the lower leg, a process which has been rarely described in the literature. In order for clinicians to successfully confront perforated colon cancer, surgical treatment must be urgent and aggressive, and effectively combine broad-spectrum antibiotics. Fascial necrosis is typically more advanced on exploration than the external appearance suggests. Reconstruction can be accomplished at a later date, allowing aggressive initial surgery to be adequately performed. The additional use of hyperbaric oxygen therapy remains controversial, notwithstanding that some authors reported it reduced the mortality rate from 66% to 23% [22]. In our case, the patient did receive hyperbaric oxygen therapy. This case emphasizes the importance of considering a rare underlying cause of necrotizing fasciitis. Despite the lack of clinical signs at presentation, involvement of the inguinal area and stool smelling of pus would suggest an abdominal source of sepsis. A CT scan could have facilitated better operative planning and treatment counseling for the patient. In conclusion, perforated colon cancer usually occurs intraperitoneally. Retroperitoneal perforation is rare and difficult to diagnose due to the lack of clinical symptoms. On the other hand, necrotizing fasciitis is a rare condition that demands prompt diagnosis and surgical exploration. Treatment needs to be aggressive and urgent; debridement and reconstruction can be accomplished at a later date. Taking into account the compounded risk caused by both perforated colon cancer and necrotizing fasciitis presenting together as in this case, it is crucial for clinicians to consider possible rare underlying causes to enhance their diagnostic efficiency, which can further improve overall patient survival rates as well.
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