Necrotizing Fasciitis with Non-Clostridial Gas Gangrene Due to the Rare Complication of Cecal Cancer Perforation

盲腸惡性腫瘤破裂併發罕見壞死性筋膜炎併非梭菌氣性壞疽

謝松育(Sung-Yu Hsieh) ； 石敏言(Min-Yen Shi) ； 李建智(Jian-Jr Lee)

《臺灣整形外科醫學會雜誌》 27卷3期 (2018/09) Pp. 221-230

摘要

Background: Plastic surgeons must often treat necrotizing fasciitis in daily practice. However, there are pitfalls and cautions that they should be aware of. Aim and Objectives: We introduce a rare case of cecal cancer presenting as necrotizing fasciitis with non-clostridial gas gangrene of the bilateral buttocks and thighs, and discuss this condition based on a review of the related literature. Materials and Methods: A 49-year-old male came to our emergency department by himself due to severe left leg weakness for 2 days. Mild lower abdominal tenderness and ecchymosis with crepitus over the bilateral buttocks to upper thighs were noted. Lab data revealed a normal white blood cell count with severe bandemia and an elevated C-reactive protein (CRP) level. Plain film abdominal x-ray and abdominal computed tomography revealed wall thickening around the ileocecal valve with 6 cm of surrounding gas, as well as a massive volume of free air (approximately 25×20×8 cm^3), in the subcutaneous to intramuscular spaces from the lower abdomen to the bilateral buttocks, thighs, and knees. A fasciotomy of the bilateral buttocks and thighs and an abdominal exploratory laparotomy were performed by a plastic surgeon and general surgeon simultaneously. Septic shock developed during the operation. During the fasciotomy, considerable amounts of subcutaneous and intramuscular air and right gluteal muscle necrosis were found without obvious pus accumulation. During the laparotomy, cecal perforation with dirty ascites extending to retroperitoneum was found, and a right colectomy with ileostomy was performed. Multiple organ failure progressed in the intensive care unit, and the patient expired the next morning. The surgical pathology report indicated cecal mucinous adenocarcinoma, and the blood culture showed Propionibacterium acnes in 2 sets. Furthermore, the pus culture showed Klebsiella pneumoniae and coagulase-negative Staphylococcus. Results: A review of the literature in the PUBMED database conducted using keywords revealed reports on 15 cases of cecal cancer and necrotizing fasciitis of the limbs which were similar to our case. The common features of these cases were the presence of a non-traumatic, undiagnosed occult malignancy, rapid disease progression, and early mortality after the first clinical visit. The anatomical route between the peritoneum and extremities plays an important role in this kind of infection. Gas gangrene is typically caused by clostridia infection, while mixed infection may also cause it. Treatments include surgical intervention, the administration of intravenous antibiotics, and hyperbaric oxygen therapy, and should be performed as soon as possible. We found a tendency toward better outcomes, including longer periods of survival, for the cases from the 21st century due to better use of the relevant diagnostic tools, which led, in turn, to earlier treatment. Conclusion: In conclusion, necrotizing fasciitis with gas gangrene of the lower extremities originating from cecal perforation is rare but has still been reported in the literature sporadically. Moreover, similar cases of necrotizing fasciitis with gas gangrene of limbs due to undiagnosed cecal cancer have also been reported. The dramatically rapid progress of the disease is usually appalling and difficult to control. In this case, we encountered dramatic spreading of non-clostridial gas gangrene of the thigh, with a total free air volume of 4 liters, that later proved to be result of cecal cancer as revealed by laparotomy. Immediate surveys and appropriate procedures for fasciitis should be executed in such cases.

關鍵字

colon cancer ； hollow organ perforation ； necrotizing fasciitis ； gas gangrene

並列摘要

背景：整形外科醫師在日常執業中常會遇到壞死性筋膜炎的處理。我們必須特別小心某些陷阱和注意事項。目的及目標：介紹一個罕見的雙側臀部至下肢的罕見壞死性筋膜炎合併大量非梭菌氣性壞疽的病例與基於文獻回顧的討論。材料及方法：49歲男性因為兩天來左下肢嚴重無力自行至急診求診。同時發現有些微的下腹疼痛與臀部下肢發紅與皮下撚髮音。實驗室檢查發現白血球指數升高合併桿狀核粒細胞增多症與C反應蛋白升高。腹部放射線檢查與電腦斷層檢查發現迴結腸瓣處腸壁增厚與腹腔自由空氣約六公分、下腹部至雙側臀部與大腿的皮下層至肌肉層大量約25×20×8立方公分的自由空氣。雙側臀部與下肢的筋膜切開術與開腹探查術同時進行。在手術中，發生敗血性休克。筋膜切開後發現大量氣體與臀大肌壞死，但沒有膿狀分泌物聚集。開腹探查術發現大腸穿孔與大量髒腹水，於是執行了右結腸切除術併腸造口。術後病人在加護病房有多重器官衰竭的症狀。病人在次日早上死亡。病理報告為盲腸黏液性惡性腫瘤。血液培養發現痤瘡丙酸桿菌，傷口培養發現肺炎克雷伯氏菌與凝固酶陰性葡萄球菌。結果：使用了PUBMED關鍵字搜尋，整理發現了15個病例與我們相似有盲腸腫瘤合併肢體的壞死性筋膜炎。這些病例同樣有著非創傷性、未診斷的潛伏惡性腫瘤、快速的疾病進展與首次求診後早期死亡的特徵。腹腔與肢體間的解剖構造在此類感染案例上有其重要性。氣性壞疽通常是由梭狀桿菌引起，但多種病原體合併感染也有可能造成。治療包括外科介入、靜脈抗生素給予與高壓氧治療，並且要在可能的情況下盡速執行。我們發現在本世紀被報告的病例有較好的預後或是較長一點的存活時間，推測是因為使用診斷工具的進步，能更快進行治療。結論：總結來說，下肢壞死性筋膜炎合併氣性壞疽起源於盲腸破裂是罕見病例，但依然可在文獻搜尋中零星見到相似病例。同樣的特徵如壞死性筋膜炎合併氣性壞疽合併未診斷的盲腸惡性腫瘤依然有被報導過。我們遇到這一個戲劇性擴張共約四公升的氣性壞疽病例。應該要立即檢查配合適當的處置。