Rectalepidural Fistula Complicating Crohn's Enterocolitis

DAVID WEST M.D., THOMAS R. RUSSELL, M.D.,* MARTIN BROTMAN, M.D.,t

West D, Russell TR, Brotman M. Rectalepidural fistula complicating Crohn's enterocolitis. Dis Colon Rectum 1983;26:622-624.

A previously unreported complication of a patient with Crohn's entero- colitis and internal fistulation is presented. The patient presented with meningeal signs in the lumbosacral region, fever, and sepsis. Compu- terized axial tomography revealed air in the epidural space, and a presumptive diagnosis of rectalepidural fistula was made. Surgical management included a diverting end sigmoid colostomy and pre- sacral drainage. [Key words: Crohn's disease; Rectalepidural fistula]

SINCE THE INITIAL DESCRIPTION of regional enteritis, as d e s c r i b e d by C r o h n a n d h i s c o l l e a g u e s i n 1932,1

n u m e r o u s ar t ic les h a v e r e p o r t e d m u l t i p l e c o m p l i c a t i o n s .

C o m p l i c a t i o n s a re u s u a l l y d e s c r i b e d as i n t e s t i n a l o r

e x t r a - i n t e s t i n a l m a n i f e s t a t i o n s , w i t h i n t e r n a l f i s tu l iza-

t i o n b e i n g a f r e q u e n t l y r e p o r t e d o c c u r r e n c e . W e h a v e

r ecen t ly t r ea ted a 31 -yea r -o ld w h i t e m a n w i t h l o n g -

s t a n d i n g C r o h n ' s d isease , k n o w n p r e v i o u s l y to i n v o l v e

b o t h the s m a l l i n t e s t i n e a n d the s i g m o i d c o l o n , as w e l l as

the p r o x i m a l r e c t u m . T h i s p a t i e n t p r e s e n t e d w i t h m e n i n -

gi t is , s e c o n d a r y to a r e c t a l e p i d u r a l f is tula .

R e p o r t o f a C a s e

The patient is a 31-year-old man with a 15 year history of Crohn's disease involving the distal small intestine, sigmoid colon, and rectum. Barium-enema examination two years prior to admission had demon- strated severe localized disease of the sigmoid colon and rectosigmoid juncture with a fistula between the sigmoid segment and a posterior sinus tract originating at the proximal rectum (Fig. 1). Symptoms of Crohn's disease had been minimal and stable on his oral medications-- prednisone, 12.5 mg every other day, azathioprine, 50 mg every day, and sulfasoxazole, 500 mg three times daily. He was transferred to this medical center from an outlying hospital with a 17-day history of back pain, fever and sepsis.

Received for publication February 28, 1983. Address reprint requests to Dr. Russell: Department of Surgery,

Presbyterian Hospital, Box 7999, San Francisco, California 94120.

From the *Department of Surgery, and SDivision of Gastroenterology, Department of Medicine, Presbyterian

Hospital of Pacific Medical Center, San Francisco, California

One week prior to admission to the referral hospital, the patient complained of an upper respiratory infection manifested by coryza, sore throat, and lever as high as 105~ Shortly after resolution of the respiratory symptoms, he developed severe lumbosacral pain inten- sified by forward flexion, continued fever, and vomiting. There were no urinary or new gastrointestinal symptoms.

On admission to the hospital, abnormal physical findings were limited to supersacral and perispinal tenderness in the lumbosacral area without erythema or warmth of the overlying skin, and marked tenderness of the posterior rectum without evidence of a mass. Admis- sion laboratory data revealed a WBC of 3,200/cu, mm, with a differen- tial of 65 segmented cells, 34 bands, 1 lymphocyte, and 1 monocyte. Urinalysis revealed 20-30 WBC's/hpf. The hemoglobin, SMA 12, and electrolytes were within normal limits. Cultures of blood and urine were sterile.

Radiographs of the chest and abdomen and selected views of L5 and S 1 intervertebral spaces were normal. Abdominal ultrasound examina- tion and bone scan were normal. A gallium scan showed increased uptake in the area of the sacrum. A presumptive diagnosis of para- spinal abscess secondary to Crohn's disease involvement of the rectum was made. Azathioprine was discontinued, and clindamycin was begun parenterally. The patient was then transferred to the Gastro- enterology Service at Presbyterian Hospital, San Francisco.

Examination following transfer revealed normal vital signs with the exception of a sinus tachycardia at 120 per minute. There was exquisite presacral tenderness and marked tenderness on percussion over the sacrum posteriorly. There was no skin erythema or warmth over the sacrum. Range of back motion was normal except for forward bending, which was limited to about 45 degrees. Straight leg raising induced pain at 30 degrees bilaterally. There was no evidence of paraspinus muscle spasm. Deep tendon reflexes were normal, Babinski signs were absent. Deep and superficial sensation and motor strength were normal.

Laboratory data revealed hemoglobin, 12.5 percent; hematocrit, 35.5 percent; WBC, 10,4000 cumm with 84 segmented cells, two bands, nine lymphocytes, and five monocytes with toxic granulations present; urinalysis negative; ESR 91 and serum chemistries normal, except for alkaline phosphatase of 135 (normal 35-105).

A 72-hour gallium scan confirmed the area of increased uptake over the sacrum. Radiographs of the lumbosacral spine, pelvis, and sacroi- liac joints were normal. Computerized axial tomography of the lumbo- sacral spine (Figs. 2 and 3) revealed air surrounding the thecal sac from the level of the body of L4 into the sacrum, with a collection of air extending along the sacral nerve canal to the left. There was also a thin

0012-3706/83/0900/0622/$00.95 �9 American Society of Colon and Rectal Surgeons

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Volume 26 Number 9 R E C T A L E P I D U R A L FISTULA 623

FIG. 2. C T scan demons t ra t ing air in epidural space (white square).

FIG. 3. C T scan demonst ra t ing air in epidm'al space.

FIG. 1. Bar ium enema denionstrat ing s inus tract extending poste- riorly from proximal rectum.

line of air ex tending from the posterior rectum into the mass of pre- sacral soft tissue. It was concluded that the pat ient had extension of the previously noted fistula from the posterior rectum to epidural space, without evidence of an epidural abscess.

Cefoxitin and tobramyxin were commenced. Under general anes- thesia, the perineal area was examined and reveaIed no acute inflam- mation. Digital rectal examina t ion revealed indura t ion and stenosis of the lower rectal segment with an area of umbi l ica t ion in the posterior rectum. Proctoscopy revealed marked inf lammatory changes. Narrow- ing at 8 cm from the anus precluded further advance of the ins t rument . Explorat ion of the abdominal cavity th rough a midl ine incision was then performed, revealing inf lammatory changes of the distal 2 feet of i leum compatible grossly with Crohn ' s disease. T h e r ight and trans- verse colon appeared grossly normal; the distal s igmoid colon was severely adhesed to the bladder anteriorly. Th i s was taken down with sharp dissection. Examina t ion of the rectum revealed indura t ion and in f lammatory changes. It was noted that the fistula was distal to the poin t of dissection. An end s igmoid colostomy and separate mucous fistula were performed and the abdomen closed. T h e pat ient was placed in the l i tho tomy posit ion, the presacral space entered by incis- ing the anococcygeal l igament, and presacral space opened widely. Air was heard to escape from the presacral area. Drains were placed and secured.

The patient 's postoperative course was excellent, with immediate cessation of fever and pain. Fol low-up C T scan (Figs. 4-5) revealed absence of the previously documented epidural air. He was discharged on the tenth postoperative day. He continues well 12 m o n t h s later.

FIG. 4. C T scan demons t ra t ing absence of epidural air in postopera- tive period.

FIG, 5. C T scan demons t ra t ing absence of epidural air postopera- tively.

Dis. Col. 8c Rect. 624 WEST, ET AL. September 1983

Discussion

A l t h o u g h more than 100 c o m p l i c a t i o n s of C r o h n ' s disease have been repor ted in the l i terature , 2-4 e p i d u r a l f i s tu la has no t been p rev ious ly repor ted. T e p l i c k 5 has repor ted a case of sacral os teomyel i t i s c o m p l i c a t i n g C r o h n ' s disease of the colon . T h e d e v e l o p m e n t of poste- r ior rectal f i s tu l iza t ion w i th a presacra l mass, fo l lowed by severe low back p a i n a n d evidence of sepsis, shou ld aler t the phys i c i an to a c o m p l i c a t i o n such as is be ing repor ted here. G a l l i u m and C T scans are then indicated. Conserva- tive surgical m a n a g m e n t w i th d ivers ion of fecal s t ream and presacral d ra inage shou ld fo l low p rompt ly .

References

1. Crohn BD, Ginsberg L, Oppenheimer GD. Regional ileitis: a pathologic and clinical entity. JAMA 1932, 99:1323-8.

2. Kirsner JB. The local and systemic complications of inflammatory bowel disease. JAMA 1980; 242:1177-83.

3. Greenstein A J, Janowitz HD, Sachar DB. The extra-intestinal com- plications of Crohn's disease and ulcerative colitis: a study of 700 patients. Medicine (Baltimore) 1976;55:401-12.

4. Rankin GB, Watts, HD, Melynk CS, et al. National Cooperative Crohn's Disease Study: extra-intestinal manifestations and peri- anal complications. Gastroenterology 1979;77:914-20.

5. Teplic SK. Retrorectal space. In: Greenbaum EI, ed. Radiographic atlas of colon disease. Chicago: Yearbook Medical Publishers, 1980:477-84.

Announcement

THE AMERICAN COLLEGE OF GASTROENTEROLOGY

The 48th Annual Scientific Meeting of the American College of Gastroen- terology will be held October 24-26, 1983 and the Annual Postgraduate Gastroenterology Course, entitled "Peptic Ulcer Disease-1983" will be held October 27-28, 1983 at the Biltmore Hotel, Los Angeles, California. For more information write: American College of Gastroenterology, 13 Elm Street, Manchester, Massachusetts 01944 or call (617) 927-8330.