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© 1999 The Journal of Bone and Joint Surgery, Inc. Pericapsular Fistula of the Hip After Radiation Therapy and Resection of a Rectal Carcinoma. A Case Report*  , BALTIMORE, MARYLANDInvestigation performed at the Johns Hopkins University School of Medicine, Baltimore
Enterocutaneous fistulae between the colon and the hip have been described as devestating complications of total hip arthroplasty and arthrodesis of the hip2,13,14,16. There have been numerous reports of fistulae involving pelvic structures following pelvic radiation therapy3,4,6,7,9. We report the case of a patient in whom a large perirectal abscess and a fistula between the rectum and the trochanteric region of the left hip developed thirty-two months after the patient was managed with a low anterior resection followed by adjuvant chemotherapy and radiation therapy for the treatment of rectal cancer. The fistula initially was treated with irrigation and débridement of the trochanteric region, and the wound healed by secondary intention. The fistula recurred three years later, at twhich time the patient was successfully managed with irrigation and débridement of the hip followed by perineal drainage, intestinal resection, and closure with pedicled muscle flaps. To the best of our knowledge, this is the first reported case of a colocutaneous fistula complicating the treatment of colorectal cancer in a patient who had no history of operations on the hip.
In January 1992, a sixty-eight-year-old man had a low anterior resection of the distal end of the colon and the rectum with end-to-end anastomosis for the treatment of high rectal cancer. Pathological examination revealed stage-III adenocarcinoma with invasion of the muscularis propria, involvement of three preirectal lymph nodes, and no evidence of distant metastasis (T2N1MO)1. The patient therefore received adjuvant chemotherapy with 5-fluorouracil (5-FU) and radiation therapy (a total dose of 5000 centigray administered to the pelvis and a boost of 1000 centigray administered to the presacral space)15. The patient also had a history of type-II diabetes mellitus, chronic renal insufficiency, hypertension, and coronary artery disease. He had no history of inflammatory bowel disease or perirectal disease. In June 1993, the patient was seen because of pain in the pelvis and bleeding from the rectum. Sigmoidoscopy revealed radiation-induced proctitis and a mucosal ulcer in the distal aspect of the rectum. The ulcer was treated with daily Rowasa (mesalamine) enemas. At the time of the six-month follow-up, the symptoms were markedly decreased. A colonoscopy with biopsy demonstrated a slowly granulating ulcer and no evidence of recurrence of the tumor. In June 1994, the patient was seen because of a fever as well as severe pain in the rectum. A computed tomography scan demonstrated a perirectal abscess with extravasation of air and contrast medium into the adjacent soft tissue. The abscess cavity was drained, and a descending end colostomy with a Hartmann's (blind rectal) pouch was created to divert the fecal stream. This colostomy was placed superior to the level of the previous colorectal anastomosis. The postoperative course was uneventful, and pathological examination of intraoperative specimens revealed no recurrence of the tumor. In November 1994, the patient returned with pain in the left hip, and a diagnosis of trochanteric bursitis was made. The patient was managed with injection of a steroid into the left bursa, but after three weeks the pain had increased. A magnetic resonance imaging scan revealed a focal area of fluid in and around the trochanteric bursa, a finding that was thought to be consistent with an abscess or a hematoma; there was no evidence of a fistula or a perirectal abscess. Two days later, the patient was unable to bear weight on the left lower extremity and had a fever, an elevated erythrocyte sedimentation rate (120 millimeters per hour) (normal, four to thirty millimeters per hour) and a white blood-cell count of 13,000 per cubic millimeter (1.30 X 109 per liter) (normal, 4500 to 11,000 per cubic millimeter [0.45 to 1.10 X 109 per liter]). He was admitted to the hospital immediately and was taken to the operating room, where irrigation and débridement was performed through a standard lateral incision over the greater tuberosity. The skin, subcutaneous tissues, and tensor fasciae latae were incised sharply, revealing thick, purulent, foul-smelling material that drained freely from underneath this fascial layer. The incision in the tensor fasciae latae was then extended proximally and distally, and an abscess cavity, four centimeters in diameter, was found just distal to the greater trochanter, at the level of the conjoint tendon. The entire wound was inspected thoroughly, and no additional abscesses were identified. After copious irrigation and placement of a drain into the abscess cavity, the wound was packed open. Fecal material was noted in the drain on the first postoperative day. The diagnosis of an enterocutaneous fistula was made, and no additional studies were done at this time. Cultures of intraoperative specimens were positive for Proteus mirabilis, Enterococcus faecium, Streptococcus viridans, and Bacteroides species. The patient was managed with frequent changes of dressing and open packing as well as with intravenous administration of ticarcillin-clavulanate (3.1 grams every eight hours for fourteen days), followed by a six-week course of clindamycin (300 milligrams taken orally three times a day on an outpatient basis). At the time of follow-up, the hip wound was well healed and and the patient had had no additional episodes of pain in the hip and no fever or chills. For the next three years, the patient remained essentially asymptomatic, but acute flare-ups of proctitis necessitated close observation periodically. Endoscopic evaluations were performed in August 1995 and May 1996, and the examinations of biopsy specimens that were obtained from the site of a chronic rectal ulcer were negative for tumor. In December 1997, the patient returned because of mild discomfort in the left hip. A magnetic resonance imaging scan showed a moderate amount of fluid in the area of the trochanteric bursa without evidence of joint effusion, and abscess, or a fistula. The pain resolved without treatment. One month later, severe pain developed in the left hip and the patient was unable to bear weight. Cultures of blood were positive for coagulase-negative Staphylococcus, and the white blood-cell count was 15,200 per cubic millimeter (15.2 X 109 per liter) with 33 percent band forms. Cultures of specimens from the left hip joint, obtained under the guidance of ultrasonography, were negative. Radiographs made after a Hypaque (diatrizoate meglumine) enema revealed a large sinus tract extending from the rectum to the region of the left hip, with large collections of fluid posterior to the acetabulum and tracking down the thigh (Fig. 1). An empirical course of intravenous administration of ciprofloxacin (400 millgrams every twelve hours), metronidazole (500 milligrams every ninety-six hours), and ampicillin (one gram every ninety-six hours) was begun. A magnetic resonance imaging scan confirmed the presence of a fistula immediately posterior and adjacent to the joint capsule of the left hip (Fig. 2), and the patient was taken to the operating room for irrigation and débridement. The wound was packed open, and intraoperative cultures of grossly purulent material showed growth of Escherichia coli, Enterococcus faecium, and Bacteroides fragilis. Two additional irrigations and débridements with open packing of the wound were followed by a final irrigation and débridement with closure of the wound over a closed suction drain. During the latter procedure, sigmoidoscopy revealed necrosis of the rectum at six centimeters from the anal verge, with communication to a large pelvic abscess. Irrigation and débridement of this abscess was performed through a second, precoccygeal incision. The perineal wound was connected to sump suction. A sinogram, made after injection of Hypaque through the presacral drain, showed communication to the rectum with no demonstrable fistula tract to the region of the left hip or thigh. The drains in the left hip were then removed. Cultures of specimens from the left hip were positive for coagulase-negative Staphylococcus and Enterococcus faecium and cultures of specimens from the pelvic abscess were positive for Enterococcus faecium and Candida albicans. The patient was instructed to begin a two-week course of amoxicillin-clavulanate (500 milligrams taken orally twice daily) and was discharged to home with the presacral drain in place.
A workup for metastatic disease revealed no evidence of local or distant recurrence. Flexible sigmoidoscopy and radiographs made after a repeat Hypaque enema revealed complete disruption of the colorectal anastomosis with communication into a large abscess cavity. The defunctioned colon was resected in two stages, performed approximately one month apart. During the first stage, the perineal region was explored through the precoccygeal incision and any remnants of the rectum and colon as well as any devitalized tissue that could be visualized were removed. The second stage involved an abdominal incision with removal of the portions of the rectum and the distal aspect of the colon that were not accessible through the perineal incision. The abscess cavity was further debrided, and the resulting soft-tissue defects within the irradiated field were filled with pedicled rotational muscle flaps. A right rectus abdominus flap was rotated down into the pelvis through a markedly fibrose pelvic inlet. The perineal defect was closed over a left gracilis rotation flap. At the time of the eight-month follow-up, in January 1999, the patient was doing well; he was walking normally and had normal stomach and urinary function.
Irradiation of the pelvis is widely used for primary and adjuvant treatment of a variety of extraperitoneal malignant lesions, including those of the rectum, the prostate, and the uterine cervix. Whereas acute toxicity resulting from pelvic irradiation usually resolves with temporary interruption of therapy, late complications often present more challenging problems to the patient or clinician. The overall risk of major complications among patients receiving pelvic radiation therapy has been reported to be as high as 12.9 percent at ten years6, and such injuries have been associated with severe morbidity and a high rate of mortality3,4,6,17. Importantly, damage resulting from radiation therapy may be both cumulative and progressive and therefore may not become apparent until years after the initial exposure17. The most common symptoms related to radiation-induced proctitis include fecal urgency and frequency and are due to chronic ischemia and fibrosis of the rectum3. Rectal bleeding is also common. More sever pelvic complications include intestinal obstruction and perforation, formation of fistulae, diseases of the genitourinary tract, arterial occlusion, venous thromboembolism, and fractures of the femoral neck and pelvis3,4,7,9. The prevalence of fistulae following pelvic irradiation has decreased with the use of modern principles of treatment (for example, lower total dosages of radiation, narrower portal widths, and increased surveillance of patients)3. In 1969, DeCosse et al. reported a 29 percent prevalence of pelvic fistulae in 100 patients who had had pelvic irradiation4. More recently, however, Eifel et al. reported that the overall risk of a fistula was 3.1 percent at twenty years in patients who had had pelvic irradiation for the treatment of cervical cancer; forty-seven fistulae had occured in forty-one patients, in a total series of 1784 patients6. These fistulae included vesicovaginal, rectovaginal, enterovaginal, enterouterine, and enterovesicular communications. Anastomosis-related fistulae are more common in patients receiving adjuvant radiation for the treatment of rectal cancer. In the Stockholm-I and II trials of preoperative irradiation for the treatment of rectal cancer, the overall rate of fistulae among patients who had received preoperative irradiation (4.8 percent; twenty-four of 500) was substantially higher than that among patients who had been randomized to operative treatment alone (1.9 percent; ten of 527)9. Levin et al. recently reported that a fistula developed between the sigmoid colon and the hip of a patient who had been managed with irradiation for the treatment of a rectosigmoid carcinoma after a previous total hip replacement14. Other authors have reported on patients in whom the development of a fistula between the hip and the gastrointestinal tract had complicated a total hip arthroplasty or an arthrodesis2,13,16. Trochanteric bursitis is a clinical syndrome that is characterized by pain, swelling, erythema, and tenderness over the proximal aspect of the thigh and the greater trochanter18. Although the diagnosis is based on clinical findings, magnetic resonance imaging is sensitive for the detection of collections of fluid in the trochanteric bursae and has been utilized to support this diagnosis. Typical findings on magnetic resonance imaging scans include high signal intensity in the bursa on T2-weighted or other fluid-enhancing sequences18. In the present report, the patient came to us twice because of pain in the left hip and the initial findings on clinical examination and magnetic resonance imaging were suggestive of trochanteric bursitis. On both occasions, the patient had mild symptoms in the hip, which initially improved but subsequently progressed to severe pain with systemic symptoms. The present report further suggests that fistulae between the bowel and the pericapsular region of the hip should be suspected in patients who have pain in the hpi and a history of irradiation or operative treatment of the pelvis, regardless of any history of operations on the hip. Enterocutaneous fistulae can have devestating consequences if they are not recognized and treated early with the performance of débridement and decompression of the tract. Mixed infections with Escherichia coli and Bacteroides fragilis substantially alter the defense mechanisms of the host through the elaboration of specific virulent factors such as endotoxin and capsular polysaccharide10. Multiple débridements of the hip and pelvis facilitate removal of all nonviable tissue and eradication of the infection. In the present report, open drainage of both the rectal abscess and the wound in the thigh ensured decompression of the fistula tract and diversion of the infection away from the hip. To prevent recurrence, it was essential to remove all remaining rectal tissue as well as the Hartmann's pouch. Transposition of pedicled muscle flaps allowed the chronically infected dead space to be filled with healthy, non-irradiated muscle. Adjuvant postoperative chemotherapy and radiation therapy have been shown to markedly decrease local recurrence and to improve survival among patients who have stage-II and III rectal cancer5,8,12. More recently, a preoperative combination of chemotherapy and radiation therapy has been utilized before the radical resection of rectal tumors that were similarly staged (with use of endorectal ultrasonography)15. Moreover, preoperative radiation therapy alone has been shown to decrease local recurrence and to increase overall survival compared with operative treatment alon in patients who have resectable rectal tumors19. Although the oncological benefits of adjuvant radiation therapy have led to the incorporation of the modality into most protocols for the treatment of rectal cancer, infections and critical alterations in bowel function have been reported11. Although rare, colocutaneous fistulae between the gastrointestinal tract and the pericapsular region of the hip can be particularly devestating and therefore must be considered in patients who have trochanteric pain and a history of irradiation or operative treatment of the pelvis.
*No benefits in any form have been received or will be received from a commercial party related directly or indirectly to the subject of this article. No funds were received in support of this study.
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