Successful Treatment of a Benign Solitary Rectal Ulcer by Temporary Diverting Sigmoidostomy: Report
of a Case*
M. STAVOROVSKY,M.D., t S. r~VEINTROUB,M.D., t J. RATAN, M.D., + P. ROZEN, M.D. + From the Municipal Government Medical Center, lchilov Hospital, Tel-Aviv, Israel
SOLITARY
BENIGN
ULCERS
Of
the
large
bowel are uncommon. Since the first description by Cruveilhier in 1830, 2 about 130 benign ulcers of the colonl, 2, s and about 88 benign ulcers of the rectum 3, 4,6, 7 have been reported. T h e diagnosis of benign rectal ulcer is made by excluding specific causes, especially" malignancy. T h e r e is no consistently successful conservative local or systemic therapy, while radical surgery (especially when the ulcer is in the lower part of the rectum) is sometimes undesirable because of the danger of incontinence. A case of successful m a n a g e m e n t of such a rectal ulcer by use of a temporary diverting sigmoidostomy is described.
Fro. I.
Colonoscopic view of rectal ulcer (short
arrow), with stenotic rectal l u m e n
(long arrow).
Report o f a C a s e e n e m a e x a m i n a t i o n s d e m o n s t r a t e d the ulcer, with a stenotic area j u s t p r o x i m a l to it, strongly suggestive of m a l i g n a n c y (Fig. 2). T h e r e m a i n d e r of the colon was n o r m a l . Altogether, 18 biopsies were taken by different e x a m i n e r s at various sessions. T h e biopsy specimens from the base a n d all edges of the ulcer a n d the stenotic area above revealed only non-specific chronic i n f l a m m a t o r y changes; there was no evidence of m a l i g n a n c y (Fig. 3). Special stains for tuberculosis, f u n g i and parasites were negative. Multiple stool e x a m i n a t i o n s were negative for parasites and p a t h o g e n i c bacteria. Serologic tests for syphilis, l y m p h o g r a n u l o m a v e n e r e u m , a n d amebiasis, a n d skin tests for tuberculosis a n d fungi, were negative, as was g u i n e a pig inoculation for tuberculosis. T h e s e negative findings suggested the diagnosis of a solitary chronic benign rectal ulcer causing stenosis by scar formation. D u r i n g the n e x t 12 m o n t h s the p a t i e n t received steroids by suppository, e n e m a , a n d finally, orally: Salazopyrinew orally a n d in the form of suppositories; a course of t r e a t m e n t with metronidazole,~ and various b r o a d - s p e c t r u m antibiotics. All medi-
A 40-year-old u n m a r r i e d w o m a n h a d an 18m o n t h history of tenesmus, rectal bleeding, a n d discharge of m u c u s a n d pus. She h a d no history of previous illness, a n d denied recent changes in bowel habits, the use of enemas, or any other rectal t r a u m a . Results of physical e x a m i n a t i o n were n o r m a l except that the rectal e x a m i n a t i o n revealed a firm ulcer on the posterior wall of the r e c t u m about 8 cm above the anus. Rectoscopy disclosed a single, deep, well-defined ulcer, 2 cm in diameter, with a grayish base surr o u n d e d by a h y p e r e m i c e d e m a t o u s edge. Just p r o x i m a l to the ulcer the bowel l u m e n was narrowed, firm to touch, b u t allowed passage of the rectoscope (Fig. 1). No other mucosal lesion was found. Fiberoptic colonoscopy of the left colon above the stenosis disclosed no abnormality. Gynecologic e x a m i n a t i o n revealed retroflexion of the uterus. R o u t i n e blood tests, chest x-ray and liver scan disclosed no abnormality. T w o b a r i u m * Received for publication August 23, 1976. t D e p a r t m e n t of Surgery. + D e p a r t m e n t of Gastroenterology. Address reprint requests to Dr. Stavorovsky: H e a d of Surgical D e p a r t m e n t "C", Ichilov Hospital, TelAviv, israel.
w P h a r m a c y A.B., Uppsala, Sweden. ~[ Flagyl "Specia," France.
347 Dis. Col. & Rect. May-June, 1977
Volume 20 Number 4
348
STAVOROVSKY, ET AL.
Dis. CoL & Rect. May-June, 1977
Fro. 2- Barium-enema study, demonstrating the stenotic area (black arrow) above the rectal ulcer (white arrow), strongly suggestive of malignancy. Anteroposterior and lateral views. cation failed to produce any subjective or objective improvement and, in fact, the ulcer and stenosis prog-ressed until the patient was readmitred. The recta| ulcer was deeper and the associated stenosis now prevented passage of a rectoscope,
FIG. 3. FnucOSa.
Conservative therapy having failed, it became imperative to choose a suitable surgical procedure. Owing to the low location of this deep, but benign, ulcer, whose local excision could damage the functioning of the anal sphincter, we elected to perform a diverting sigmoidostomy.
Histologic section of the ulcer edge, showing chronic inflammatory tissue and normal rectal
Volume 20 9Number 4
SIGMOIDOSTOMY FOR R E C T A L ULCER
349
Fro. 4. Barium-enema study (anteroposterior and lateral views) following healing of the rectal ulcer tnd dilatation of the stenotic area. Apart from slight n a r r o w i n g (arrows), no abnormality can be seen.
After bowel preparation with neomycin, a lower laparotomy was performed. No abnormality was found in the abdominal cavity apart from some thickening of the left lateral wall of the distal rectum, and no l y m p h a d e n o p a t h y was found. A double-loop sigmoidostomy was performed. T h e postoperative course was uneventful and the patient was discharged in good condition. Repeated rectoscopic examinations revealed complete healing of the ulcer within a month. However, the stenosis persisted. T h i s was dilated with metal bougies over a period of the next three months, until a 22-mm dilator could be passed freely. Four m o n t h s later no recurrence of the ulcer or change of the narrowed area was seen on rectoscopic or barium-enema examination (Fig. 4). It was now felt safe to close the sigTnoidostomy, and normal bowel movements appeared on the fifth postoperative day. T h e patient has been u n d e r observation for 18 months. She is asymptomatic, with regular bowel movements without tenesmus, rectal bleeding, or mucous discharge. A recent rectoscopic examination disclosed no abnormality apart from slight narrowing of the rectal lumen 8 cm above the anus.
Discussion
Since the first description of a solitary ulcer of the colon by Cruveilhier, 2 this entity has been variously named simple, solitary, benign, nonspecific, idiopathic, or stercoral ulcer of the colon. These ulcers have been well documented by Smithwick et al., 9 Butsch e t al., I Madigan and Morson,7 a n d Lazarovitch e t al. s About 88 cases of
benign rectal ulcers have been described. T h e lesion is most frequently seen in women 20 to 50 years of age:, 6, 7 T h e ulcer may appear in various parts of the colon and may be solitary or multiple, as were those in 30 per cent of Madigan and Morson's cases. 7 It may manifest acutely or pursue a chronic course. 9 Rectal ulcers are usually chronic, although rarely t h e r e is massive bleeding.7 I t appears that benign rectal ulcers occur most frequently on the anterior wall 7 to 10 cm from the anus, and are 1 to 4 cm in diameter:, 4 T h e base is pale, covered with a grayish pseudomembrane, and is well demarcated from the surrounding mucosa, which appears normal. I t is occasionally accompanied by stenosis. 7 Histologically, the base is formed by nonspecific granulation tissue, covered by inflammatory e x u d a t e . 3, 4, 6
T h e etiology of benign colonic or rectal ulcers is unknown, nor is it clear whether they have a common etiology. Madigan and Morson,7 in their series of 68 cases, did not establish a relationship to occupation, social class, race, smoking, or drinking. Selye e t al.S by producing ischemia to the colon or rectum, succeeded, in the presence of
350
Dis. Col. & Rect. May-June, 1977
STAVOROVSKY, E T AL.
constipation, in causing necrotic ulcers in 50 per cent of their experimental animals. These experiments may explain the occurrence of stercoral ulcers in the elderly. T h e presence of such ulcers in young people who have regular bowel habits or tendency to diarrhea7 and the frequent occurrence o[ rectal bleeding tend to negate this ischemicconstipation theory. Also, in the majority of patients no evidence of histologic or clinical vascular abnormality was found. T h e presence of various strains of cocci at the bases of the ulcers may suggest a pyogenic etiology,1 although others 4 accept a viral cause. It is possible that the organisms are secondary, thus reflecting both these theories. Some investigators 4, 7 consider foreign bodies or fecaliths to be responsible for the ulcers. T h e possibility of self-inflicted trauma has been raised, since 20 per cent of patients d i s t a l l y remove feces.7 However, the ulcers may be higher than 10 cm from the anus, and this does not account for the presence of multiple ulcers. Psychosomatic stress, which is responsible for many cases of u p p e r gastrointestinal ulceration, was shown by Madigan and Morson7 to be absent in 88 per cent of their patients. These patients have histories of left lower abdominal pain or rectal discomfort for some years. T h e y have a tendency to diarrhea, and sometimes intermittent rectal bleeding and a discharge of mucus or pus. T h e diag-nosis is confirmed after excluding malignancy and specific etiologic factors such as syphilis, tuberculosis, lymphogranuloma venereum, amebiasis, ulcerative colitis, proctitis, and Crohn's disease. W i t h the advent of coionoscopy, it is probably wise to examine the entire colon to exclude the presence of a pathologic process in another location. Spontaneous healing may rarely occur. 4, 7 Conventional therapy, which may include topical and systemic administration of corticosteroids, antibiotics, and salicylazosulfa-
pyridine, is usually unsuccessful. Resection, local excision, or diverting sigmoidostomy has been used, although there have been recurrences after these treatments. 4, 7 In the case of our patient, we felt that resection or local excision of the ulcer would result in incontinence, in view of the fact that excision of the associated stenotic segment would be necessary. By diverting the fecal stream we obtained rapid healing of the ulcer within a month, following which it was possible to dilate the stenotic rectal segment. Follow up of more than 18 months showed no recurrence, indicating that this operation may be useful in selected cases of benign solitary ulcer situated in the lower part of the rectum.
Summary A rare case of a solitary benign rectal ulcer in which conservative medical therapy was unsuccessful is described. Diversion of the fecal stream by temporary sigmoidostomy resulted in rapid healing of the ulcer. T h i s entity presents diagnostic and therapeutic problems, which are discussed. T h e use of temporary sigmoidostomy in such cases is recommended.
References 1. Butsch JL, Dockerty MB, McGill DB, et al: "Solitary" nonspecific ulcers of the colon. Arch Surg 98: 171, 1969 2. CruveilMer J: Q u o t e d by Butsch JL, Dockerty MB, McGill DB, et al 1 3. Haskell B, R o v n e r H: Solitary ulcer of the rectum. Dis Colon R e c t u m 8: 333, 1965 4. J a l a n KN, B r u n t P W , Maclean N. et al: Benign solitary ulcer of the r e c t u m - - a report oE 5 cases. Scand J Gastroenterol 5: 143, 1970 5. Lazarovitch I, Michowitz M, L o w e n t h a l M, et al: Nonspecific ulcers of the cecum: Report of two cases. Dis Colon R e c t u m 17: 381, 1974 6. L o c k h a r t - M u m m e r y HE: Solitary ulcer of t h e rectum. Proc R Soc Med 57: 403, 1964 7. Madigan M R , Morson BC: Solitary ulcer of the rectum. G u t 10: 871, 1969 8. Selye H, W i n a n d y G, G a b b i a n i G: Production a n d prevention of stercoraI ulcers in the rat. A m J. Pathol 48: 299, 1966 9. Smithwick W, A n d e r s o n RP, Ballinger W F tI: Nonspecific ulcer of the colon. Arch Surg 97: 133, 1968
of a Case*
M. STAVOROVSKY,M.D., t S. r~VEINTROUB,M.D., t J. RATAN, M.D., + P. ROZEN, M.D. + From the Municipal Government Medical Center, lchilov Hospital, Tel-Aviv, Israel
SOLITARY
BENIGN
ULCERS
Of
the
large
bowel are uncommon. Since the first description by Cruveilhier in 1830, 2 about 130 benign ulcers of the colonl, 2, s and about 88 benign ulcers of the rectum 3, 4,6, 7 have been reported. T h e diagnosis of benign rectal ulcer is made by excluding specific causes, especially" malignancy. T h e r e is no consistently successful conservative local or systemic therapy, while radical surgery (especially when the ulcer is in the lower part of the rectum) is sometimes undesirable because of the danger of incontinence. A case of successful m a n a g e m e n t of such a rectal ulcer by use of a temporary diverting sigmoidostomy is described.
Fro. I.
Colonoscopic view of rectal ulcer (short
arrow), with stenotic rectal l u m e n
(long arrow).
Report o f a C a s e e n e m a e x a m i n a t i o n s d e m o n s t r a t e d the ulcer, with a stenotic area j u s t p r o x i m a l to it, strongly suggestive of m a l i g n a n c y (Fig. 2). T h e r e m a i n d e r of the colon was n o r m a l . Altogether, 18 biopsies were taken by different e x a m i n e r s at various sessions. T h e biopsy specimens from the base a n d all edges of the ulcer a n d the stenotic area above revealed only non-specific chronic i n f l a m m a t o r y changes; there was no evidence of m a l i g n a n c y (Fig. 3). Special stains for tuberculosis, f u n g i and parasites were negative. Multiple stool e x a m i n a t i o n s were negative for parasites and p a t h o g e n i c bacteria. Serologic tests for syphilis, l y m p h o g r a n u l o m a v e n e r e u m , a n d amebiasis, a n d skin tests for tuberculosis a n d fungi, were negative, as was g u i n e a pig inoculation for tuberculosis. T h e s e negative findings suggested the diagnosis of a solitary chronic benign rectal ulcer causing stenosis by scar formation. D u r i n g the n e x t 12 m o n t h s the p a t i e n t received steroids by suppository, e n e m a , a n d finally, orally: Salazopyrinew orally a n d in the form of suppositories; a course of t r e a t m e n t with metronidazole,~ and various b r o a d - s p e c t r u m antibiotics. All medi-
A 40-year-old u n m a r r i e d w o m a n h a d an 18m o n t h history of tenesmus, rectal bleeding, a n d discharge of m u c u s a n d pus. She h a d no history of previous illness, a n d denied recent changes in bowel habits, the use of enemas, or any other rectal t r a u m a . Results of physical e x a m i n a t i o n were n o r m a l except that the rectal e x a m i n a t i o n revealed a firm ulcer on the posterior wall of the r e c t u m about 8 cm above the anus. Rectoscopy disclosed a single, deep, well-defined ulcer, 2 cm in diameter, with a grayish base surr o u n d e d by a h y p e r e m i c e d e m a t o u s edge. Just p r o x i m a l to the ulcer the bowel l u m e n was narrowed, firm to touch, b u t allowed passage of the rectoscope (Fig. 1). No other mucosal lesion was found. Fiberoptic colonoscopy of the left colon above the stenosis disclosed no abnormality. Gynecologic e x a m i n a t i o n revealed retroflexion of the uterus. R o u t i n e blood tests, chest x-ray and liver scan disclosed no abnormality. T w o b a r i u m * Received for publication August 23, 1976. t D e p a r t m e n t of Surgery. + D e p a r t m e n t of Gastroenterology. Address reprint requests to Dr. Stavorovsky: H e a d of Surgical D e p a r t m e n t "C", Ichilov Hospital, TelAviv, israel.
w P h a r m a c y A.B., Uppsala, Sweden. ~[ Flagyl "Specia," France.
347 Dis. Col. & Rect. May-June, 1977
Volume 20 Number 4
348
STAVOROVSKY, ET AL.
Dis. CoL & Rect. May-June, 1977
Fro. 2- Barium-enema study, demonstrating the stenotic area (black arrow) above the rectal ulcer (white arrow), strongly suggestive of malignancy. Anteroposterior and lateral views. cation failed to produce any subjective or objective improvement and, in fact, the ulcer and stenosis prog-ressed until the patient was readmitred. The recta| ulcer was deeper and the associated stenosis now prevented passage of a rectoscope,
FIG. 3. FnucOSa.
Conservative therapy having failed, it became imperative to choose a suitable surgical procedure. Owing to the low location of this deep, but benign, ulcer, whose local excision could damage the functioning of the anal sphincter, we elected to perform a diverting sigmoidostomy.
Histologic section of the ulcer edge, showing chronic inflammatory tissue and normal rectal
Volume 20 9Number 4
SIGMOIDOSTOMY FOR R E C T A L ULCER
349
Fro. 4. Barium-enema study (anteroposterior and lateral views) following healing of the rectal ulcer tnd dilatation of the stenotic area. Apart from slight n a r r o w i n g (arrows), no abnormality can be seen.
After bowel preparation with neomycin, a lower laparotomy was performed. No abnormality was found in the abdominal cavity apart from some thickening of the left lateral wall of the distal rectum, and no l y m p h a d e n o p a t h y was found. A double-loop sigmoidostomy was performed. T h e postoperative course was uneventful and the patient was discharged in good condition. Repeated rectoscopic examinations revealed complete healing of the ulcer within a month. However, the stenosis persisted. T h i s was dilated with metal bougies over a period of the next three months, until a 22-mm dilator could be passed freely. Four m o n t h s later no recurrence of the ulcer or change of the narrowed area was seen on rectoscopic or barium-enema examination (Fig. 4). It was now felt safe to close the sigTnoidostomy, and normal bowel movements appeared on the fifth postoperative day. T h e patient has been u n d e r observation for 18 months. She is asymptomatic, with regular bowel movements without tenesmus, rectal bleeding, or mucous discharge. A recent rectoscopic examination disclosed no abnormality apart from slight narrowing of the rectal lumen 8 cm above the anus.
Discussion
Since the first description of a solitary ulcer of the colon by Cruveilhier, 2 this entity has been variously named simple, solitary, benign, nonspecific, idiopathic, or stercoral ulcer of the colon. These ulcers have been well documented by Smithwick et al., 9 Butsch e t al., I Madigan and Morson,7 a n d Lazarovitch e t al. s About 88 cases of
benign rectal ulcers have been described. T h e lesion is most frequently seen in women 20 to 50 years of age:, 6, 7 T h e ulcer may appear in various parts of the colon and may be solitary or multiple, as were those in 30 per cent of Madigan and Morson's cases. 7 It may manifest acutely or pursue a chronic course. 9 Rectal ulcers are usually chronic, although rarely t h e r e is massive bleeding.7 I t appears that benign rectal ulcers occur most frequently on the anterior wall 7 to 10 cm from the anus, and are 1 to 4 cm in diameter:, 4 T h e base is pale, covered with a grayish pseudomembrane, and is well demarcated from the surrounding mucosa, which appears normal. I t is occasionally accompanied by stenosis. 7 Histologically, the base is formed by nonspecific granulation tissue, covered by inflammatory e x u d a t e . 3, 4, 6
T h e etiology of benign colonic or rectal ulcers is unknown, nor is it clear whether they have a common etiology. Madigan and Morson,7 in their series of 68 cases, did not establish a relationship to occupation, social class, race, smoking, or drinking. Selye e t al.S by producing ischemia to the colon or rectum, succeeded, in the presence of
350
Dis. Col. & Rect. May-June, 1977
STAVOROVSKY, E T AL.
constipation, in causing necrotic ulcers in 50 per cent of their experimental animals. These experiments may explain the occurrence of stercoral ulcers in the elderly. T h e presence of such ulcers in young people who have regular bowel habits or tendency to diarrhea7 and the frequent occurrence o[ rectal bleeding tend to negate this ischemicconstipation theory. Also, in the majority of patients no evidence of histologic or clinical vascular abnormality was found. T h e presence of various strains of cocci at the bases of the ulcers may suggest a pyogenic etiology,1 although others 4 accept a viral cause. It is possible that the organisms are secondary, thus reflecting both these theories. Some investigators 4, 7 consider foreign bodies or fecaliths to be responsible for the ulcers. T h e possibility of self-inflicted trauma has been raised, since 20 per cent of patients d i s t a l l y remove feces.7 However, the ulcers may be higher than 10 cm from the anus, and this does not account for the presence of multiple ulcers. Psychosomatic stress, which is responsible for many cases of u p p e r gastrointestinal ulceration, was shown by Madigan and Morson7 to be absent in 88 per cent of their patients. These patients have histories of left lower abdominal pain or rectal discomfort for some years. T h e y have a tendency to diarrhea, and sometimes intermittent rectal bleeding and a discharge of mucus or pus. T h e diag-nosis is confirmed after excluding malignancy and specific etiologic factors such as syphilis, tuberculosis, lymphogranuloma venereum, amebiasis, ulcerative colitis, proctitis, and Crohn's disease. W i t h the advent of coionoscopy, it is probably wise to examine the entire colon to exclude the presence of a pathologic process in another location. Spontaneous healing may rarely occur. 4, 7 Conventional therapy, which may include topical and systemic administration of corticosteroids, antibiotics, and salicylazosulfa-
pyridine, is usually unsuccessful. Resection, local excision, or diverting sigmoidostomy has been used, although there have been recurrences after these treatments. 4, 7 In the case of our patient, we felt that resection or local excision of the ulcer would result in incontinence, in view of the fact that excision of the associated stenotic segment would be necessary. By diverting the fecal stream we obtained rapid healing of the ulcer within a month, following which it was possible to dilate the stenotic rectal segment. Follow up of more than 18 months showed no recurrence, indicating that this operation may be useful in selected cases of benign solitary ulcer situated in the lower part of the rectum.
Summary A rare case of a solitary benign rectal ulcer in which conservative medical therapy was unsuccessful is described. Diversion of the fecal stream by temporary sigmoidostomy resulted in rapid healing of the ulcer. T h i s entity presents diagnostic and therapeutic problems, which are discussed. T h e use of temporary sigmoidostomy in such cases is recommended.
References 1. Butsch JL, Dockerty MB, McGill DB, et al: "Solitary" nonspecific ulcers of the colon. Arch Surg 98: 171, 1969 2. CruveilMer J: Q u o t e d by Butsch JL, Dockerty MB, McGill DB, et al 1 3. Haskell B, R o v n e r H: Solitary ulcer of the rectum. Dis Colon R e c t u m 8: 333, 1965 4. J a l a n KN, B r u n t P W , Maclean N. et al: Benign solitary ulcer of the r e c t u m - - a report oE 5 cases. Scand J Gastroenterol 5: 143, 1970 5. Lazarovitch I, Michowitz M, L o w e n t h a l M, et al: Nonspecific ulcers of the cecum: Report of two cases. Dis Colon R e c t u m 17: 381, 1974 6. L o c k h a r t - M u m m e r y HE: Solitary ulcer of t h e rectum. Proc R Soc Med 57: 403, 1964 7. Madigan M R , Morson BC: Solitary ulcer of the rectum. G u t 10: 871, 1969 8. Selye H, W i n a n d y G, G a b b i a n i G: Production a n d prevention of stercoraI ulcers in the rat. A m J. Pathol 48: 299, 1966 9. Smithwick W, A n d e r s o n RP, Ballinger W F tI: Nonspecific ulcer of the colon. Arch Surg 97: 133, 1968
