Cavernous hemangioma of the rectum and rectosigmoid colon

{papers of the ScknLifk sessions CAVERNOUS HEMANGIOMA OF THE RECTUM AND RECTOSIGMOID COLON ALAN A. JAQUES, M .D.

(By Invitation)

Rockville Centre, New York ARGE cavernous hemangiomas of the rectum or sigmoid colon have been reported twenty-five times .',',',6 The tumor may be single or multiple, submucous, intramuscular or subserous, or all layers may be involved . It may be localized to the bowel or it may extend into adjacent tissue . Small or massive rectal hemorrhage is usual and there is generally tenesmus and the passage of mucus . Several lesions have been treated as hemorrhoids before the proper diagnosis was made . Clusters of phleboliths on x-ray may suggest hemangioma . Treatment in reported cases has been by cautery, irradiation, surgical exclusion and resection . Forty per cent of treated patients are reported to have died of their disease . Kausch obtained a cure with a five-stage surgical procedure .' Sawyer did a Mikulicz resection of a sigmoid hemangioma .' Hunt resected the rectum and rectosigmoid colon 4 . Babcock and Jonas resected the rectum and rectosigmoid colon and succeeded in preserving the sphincter and its function .' The present case was reported as a cure by Bancroft in 1931 . 2

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CASE REPORT

B . L ., a thirty-nine year old business man, was first admitted to South Nassau Communities Hospital on December 4, 1951, in moderately severe shock and with signs of advanced diffuse peritonitis . Ten days prior to admission he had had an attack of severe lower abdominal pain and diarrhea which gradually subsided . Four hours before admission severe abdominal pain was experienced and x-ray showed free gas in both subphrenic spaces . Past history revealed that rectal bleeding was first apparent at fifteen months of age . Operations for hemorrhoids were performed at four, six and ten years of age! Rectal bleeding conNovember, 1 95 2

tinned intermittently and the patient remained anemic with weakness and exertional dyspnea . When the patient was seventeen years of age, Drs . F . W . Bancroft and Harvey Stone did independent sigmoidoscopies and diagnosed "angioma ." Ten inches of mucosa were inspected and greatly dilated and engorged veins were seen which "stand out like blood splotches against the pale mucous membrane ." 2 There was also the appearance of two internal hemorrhoids . On April io, 193o, Dr . Bancroft ligated the superior hemorrhoidal vein and injected it distally with io cc . Of 40 per cent sodium salicylate . At the same operation a high sigmoidostomy was established . The surfaces of the "sigmoid and rectosigmoid were purple in color with distended tortuous veins up to about ten inches of the end of the descending colon . The line of demarcation between normal and abnormal bowel was distributed over an area of two inches ." Two, seven and eleven months after operation proctoscopy showed complete disappearance of veins previously seen and rectal bleeding completely ceased . In March, 1931, the colostomy was closed and a cecostomy established . Dr . Bancroft states, "While there were still a few dilated veins, the greater number appeared thrombosed and it seemed that the vessels on the surface had greatly diminished in number ." The serosa was normally pink . Since the operation previously described the patient had taken mineral oil daily and had found it necessary to eliminate from his diet certain foods which produced cramps and diarrhea . Every two or three years rectal vessels were injected . Three years after operation the patient began to have occasional bloody stools and increased rectal and low abdominal pain after meals . These complaints have been more marked during the past two years . 507

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Jaques

Hemangioma of Rectum

Mucosa and submucosa showing large and small sinuses . FIG . 2 .

FIG . I . Semi-diagrammatic sketch to illustrate thickness of wall, invasion of all layers by hemangioma, extension of disease to and below the level of the sphincters and bullous character of mucosa with congestion and occasional ulceration of hemangiomatous sinus .

At the first stage operation, December 4, 1951, there was diffuse, moderately advanced peritonitis originating from a ruptured abscess . The abscess was 8 cm . in diameter and densely adherent under omentum, ileum and angulated sigmoid colon . The distal half of the sigmoid colon was the site of a firm rubbery tumor 9 .5 cm . in diameter . Under the serosa of the entire circumference of the bowel was a complete network of tortuous, distended, bluish purple vessels 1 to 3 mm . In diameter . The junction of normal and involved bowel was the site of marked narrowing and a large perforation . The perforation was exteriorized and drains placed . Brisk hemorrhage from mildly traumatized bowel could be immediately controlled by gentle pressure and permanently arrested by oxyeel gauze . At the second stage operation, December 20th, a transverse colostomy was performed with the establishment of two separate stomas .

The liver, transverse colon and small bowel were normal in appearance . On December 3oth a sigmoidoscope was passed i o inches without difficulty . The mucosa was edematous and mottled dark and light red . Over about twothirds of the surface huge dilated vessels could be seen . These veins were continuous with dilated veins in the hemorrhoidal area . However, simple anoscopic examination yielded the appearance of a complete rosette of internal hemorrhoids . The patient was discharged on January 1, 1952, and regained good general health but continued to pass some bloody mucus . His habitual tenesmus and low abdominal pain became tolerable with the use of banthine . Sigmoidoscopy performed on February loth by Dr. Stuart T. Ross showed no essential change . X-ray at this time showed a normal bony pelvis and many small calcific deposits within the pelvis . The third stage operation was performed on February 15th, at which time the abdominal portion of the tumor was as noted ten weeks before . There was no hemangioma in the mesosigmoid and its vessels were unaltered except by previous surgery . The bulk of the tumor was below the peritoneal reflection and was densely adherent to the bladder, prostate and urethra . Cleavage planes were obtained with difficulty but there was no apparent invasion of the urinary tract or periosteum . Cystoscopy and ureteral catheterization were perAmerican Journal of Surgery

Jaques

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Hemangioma of Rectum

FIG . 3 . Hemangioma of the bowel wall in the region of the muscularis . FIG . 4 . Hemangioma under serosa of epiploic fat .

formed in the operating room and a Miles abdominoperineal resection of the rectum and lower sigmoid was done . No unexpected hemorrhage was encountered but there was a constant ooze from the hemangiomatous tissue and control was possible only by pressure and packing . An estimated 750 cc of blood loss was replaced by I,ooo cc . of citrated blood . A permanent sigmoidostomy was made . The perineum was drained and closed . Pathologic report revealed the following : The specimen consisted of an anus, rectum and distal sigmoid measuring 34 cm . in length and I I cm . in its greatest diameter 7 cm . above the rectum . The distal two-thirds of the bowel showed raised pink or dark red irregularities of the mucosal and serosal surfaces o .2 to 2 cm . in diameter . Along the cut margins of the bowel were thin-walled vascular channels containing fresh and organized thrombi extensively distributed through the submucous and subserous layers and in the epiploic fat . Similar vascular spaces were found at the end of and i cm . distal to the external sphincter . (Figs . I to 4 .) The diagnosis was benign cavernous hemangioma of the anus, rectum and rectosigmoid colon . At the fourth stage operation on March 6th closure of transverse colostomy was done . The patient was discharged on March I8th . Convalescence from all procedures was relatively uneventful except for some delay in closing of the perineal sinus . One month after discharge

November, 1952

from the hospital the patient started a new and active business which he is continuing at present . He is completely free from complaints for the first time in years and is well adjusted to his colostomy . CONCLUSION

A case of cavernous hemangioma of the rectum and rectosigmoid colon has been presented . Twenty years ago remission had been obtained by injecting a sclerosing solution into the superior hemorrhoidal veins but the tumor had since re-established itself completely . The possibility of an erroneous diagnosis of hemorrhoids is emphasized . Since fatal hemorrhage is a constant threat, complete extirpation is recommended whenever possible . REFERENCES i . BABCOCK, W . and JONAS, K. C . Hemangioma of colon . Am . J. Surg ., 8o : 854 -759, 1 950 . 2 . BANCROFT, F . W . Hemangioma of the sigmoid and colon . Ann . Surg ., 94 : 828-838, 1931 . 3 . GENTRY, R. W . et al . Vascular malformations and tumors of the gastro-intestinal tract . Internal . Abstr. Surg ., 88 : 281 - 323, 1949. 4- HUNT, V . C. Hemangioma of the large bowel . Surgery, to : 65i-66o, 1941 . 5 . KAUSC1, W. Ueber Varicose and Cavernose des Mastdarms. Verbandl . d . deutscb . Gesellsch . Cbir ., 43 : 243-2 45, 19146 . OPPENHEIM, A . and O'BRIEN, J . P . Unusual rectal perirectal tumors palpable by rectal examinations . Am . J. Surg., 79 : 302-311, 1950 . 7 . SAWYER, C . F . Hemangioma of colon . Arcb . Surg., 39 :987-991, 1939.