Summary
A 13-year-old girl with Prader-Willi syndrome was admitted to our hospital with an 18-month history of anal bleeding and mucus discharge on defecation.
Physical examination revealed obesity, hypogonadism, hypotonia and hypomentia. On digital examination, a nodular mass was palpated on the right wall of the amupulla recti, which was suspected to be carcinoma on a barium enema study. Proctoscopic examination revealed a large, irregular ulceration with white slough at the base, surrounded by the nodular and lumpy mucosa.
The lesion was excised by the abdomino-anal pull-through method. The resected specimen showed a lesion of large, shallow, irregular ulcer, 5.0 x 2.2 cm in size. Microscopic examination revealed obliterated lamina propria by fibroblasts and muscle fibers derived from the muscularis mucosae, and misplaced cystic dilated glands in the submucosa at the margin of the ulcer. The gross and microscopic appearances are identical to those of “solitary ulcer of the rectum” described by Madigan and others, and similar to those of “colitis cystica profunda” described by Goodall and others. According to these findings, this lesion was diagnosed as solitary ulcer of the rectum.
In the present report, the relationship between solitary ulcer of the rectum and colitis cystica profunda was discussed.
Similar content being viewed by others
References
Allen Ms: Hamartomatous inverted polyps of the rectum. Cancer 19: 257–265, 1966
Ballas M, et al: localized colitis cystica profunda. Arch Surg 103: 406–408, 1971
Barlow D, et al: Simple ulcer of the caecum, colon and rectum. Br J Surg28: 575–581, 1941
Barner JL: Colitis cystica profunda. Radiology 89: 435–437, 1967
Barron ME: Simple nonspecific ulcer of the colon. Arch Surg 17: 355–407, 1928
Black HC, et al: Localized colitis cystica profunda, a benign lesion simulating malignancy Am Surgeon 38: 237–239, 1972
Burt CAV, et al: Colitis cystica profunda concurrent with and differentiated from mucinous adenocarcinoma: report of a case. Dis Colon Rectum 13: 460–469, 1970
Capehart RJ, et al: Colitis cystica profunda: clinical and histological differentiation of mucinous adenocarcinoma from colitis cystica profunda. J Kans Med Soc 72: 385–386, 1971
Carstens PHB, et al: Colitis cystica profunda. Acta pathol Microbiol Scandinav 75: 273–281, 1969
Clark JF, et al: Coltitis cystica profunda in an adenoma (adenomatous polyp): report of a case. Dis Colon Rectum 13: 387–389, 1970
Cruveilhier J: Ulcere chronique du rectum. In Anatomie Pathologique du corps humain. Vol 2, livr 25, maladies du rectum, p 4, J B Balliere, Paris
Epstein SE, et al: Colitis cystica Profunda. Am J Clin pathol 45: 186–281, 1966
Fechner RE: Polyp of the colon possessing features of colitis cystica profunda. Dis Colon Rectum 10: 359–364, 1967
Ghani A: Colitis cystica profunda. Br J Surg 57: 596–598, 1970
Goligher JC: Surgery of the anus rectum and colon. Balliere Tindall, London, 1975, P 1051–1052
Goodall HB, et al: Colitis cystica profunda. J Pathol Bact 73: 33–42, 1957
Grant KB, et al: Colitis cystica profunda. A lesion of increasing significance to radiologist and pathologist. Radiology 89: 110–111, 1967
Green GI, et al: Colitis cystica profunda. Am J Surg 127: 749–752, 1974
Haskell B, et al: Solitary ulcer of the rectum. Dis Colon Rectum 8: 333–336, 1965
Herman AH, et al: Colitis cystica profunda: Localized, Segmental and diffuse. Arch Surg 106: 337–341, 1973
Horimi T, et al: Two cases of solitary rectal ulcer: treated by amputation of the rectum. Stomach and Intestine 13(6): 843–848, 1978 (Jpn)
Howard RJ, et al: Colitis cystica profunda. Surgery 69: 306–308, 1971
Ino M, et al: Colitis cystica profunda. J Clin Surg 31(10): 1372–1375, 1976 (Jpn)
Kobayashi Y: Colitis cystica profunda. Stomach and Intestine 8(2): 164, 1973 (Jpn)
Kon J, et al: Colitis cystica profunda and solitary ulcer of the rectum. Koseisho Tokutei -shikkan, Tokuhatsusei Chokan-shogai chosa Kenkyuhan Gyosekishu, 31–39, Mar 1977
Lloyd -Davies OV: From 29
Lockhart-Mummery HE: Solitary ulcer of the rectum. Proc Roy Soc Med 75: 403, 1964
Madigan MR: Solitary ulcer of the rectum. Proc Roy Soc Med 57: 403, 1964
Madigan MR, et al: Solitary ulcer of the rectum. Gut 10: 871–881, 1969
Mason AY, et al: Surgical access to the rectum: A transsphincteric exposure. Proc Roy Soc Med Suppl, 63,1
Matsuda Y, et al: Clinicopathological investigations of rectal ulcer; with special reference to solitary ulcer of the rectum and colitis cystica profunda. Koseisho Tokutei-shikkan, Tokuhatusei Chokan-shogai Chosa Kenkyuhan Gyosekishu, 24–28, Mar 1977 (Jpn)
Matsumoto K, et al: Three cases of solitary ulcer of the rectum. J P S S 36(6): 736–742, 1975 (Jpn)
Muldoon JP, et al: Colitis cystica profunda: report of two cases. Dis Colon Rectum 11: 220–222, 1968
Murakami H, et al: Two cases of nonspecific ulcer of the colon and rectum. Surg Diag Treat 14(5): 603–607, 1972 (Jpn)
Nagasako K, et al: Colitis cystica profunda: Report of a case in which differentiation from rectal cancer was difficult. Dis Colon Rectum 20(7): 618–624, 1977
Nakahara A, et al: Two cases of solitary ulcer of the rectum. Prog Digest Endoscopy 7(12): 195–198, 1975 (Jpn)
Nakamura T, et al: A case report of nonspecific ulcer of the colon. J J G S 7(6): 626–629, 1974 (Jpn)
O’Brien SE, et al: Colitis cystica profunda: a rare lesion of the colon. Can J Surg 14: 53–55, 1971
Okuyama A, et al: PRADER-WILLI syndrome: report of two cases. Jpn J Urol 67(7): 548–557, 1976 (Jpn)
Rutter RP, et al: The solitary ulcer syndrome of the rectum. Clinics in gastroenterology 4: 505–530, 1975
Sasaki K, et al: Endoscopy of the Rectum & Colon. “Rectal Ulcer”. Prog Digest Endoscopy 3(12): 139–141, 1973 (Jpn)
Schweiger M, et al: Solitary-ulcer syndrome of the Rectum. It’s association with occult rectal prolapse. Lancet 1(8004): 170–171, Jan 22, 1977
Scruggs FL, et al: Colitis cystica profunda. South Med J 61: 618–620, 1968
Sullivan JJ, et al: Localized submucosal mucous cysts of the rectum (colitis cystica profunda). Med J Aust 1: 133–135, 1968
Takada Y, et al: A case of colitis cystica profunda. Kitazato Medicine 6: 115–120, 1976 (Jpn)
Takano M, et al: A typical case of solitary ulcer of the rectum. Stomach and Intesine 8(4): 505–508, 1973 (Jpn)
Talerman A: Enterogenous cysts of the rectum (colitis cystica profunda). Br J Surg 58: 643–647, 1971
Tasaka K, et al: Colitis cystica profunda. Stomach and intestine 11(4): 503–508, 1976 (Jpn)
Wannarache N, et al: Hypogonadism in PraderWilli Syndrome. Am J Mental Deficiency 79(5): 592–603, 1975
Wayte DM, et al: Colitis cystica profunda. Am J Clin Patho 148: 159–169, 1967
Yamaoka I, et al: Solitary ulcer of the rectum: report of three cases. Prog Digest Endoscopy 10(6): 260–263, 1977 (Jpn)
Yoshida T, et al: Colitis cystica profunda: report of a case. Stomach and Intestine 15(3): 311–316, 1980 (Jpn)
Yoshioka H, et al: A case of nonspecific ulcer of the rectum to be difficult to defferentiate from the rectal cancer on X-ray examination. Clin Radiology 23(8): 875–879, 1978 (Jpn)
Young VT, et al: Diagnostic features of colloidadenocarcinoma of anal ducts and colitis cystica profunda. South Med J 65: 1035–1039, 1972
Zellweger H, et al: Syndrome of Hypotonia-Hypomentia-Hypogonadism-Obesity (HHHO) or prader-Willi Syndrome. Am J Dis Child 115(5): 588–598, 1968
Author information
Authors and Affiliations
Rights and permissions
About this article
Cite this article
Misumi, A., Sera, Y., Matsuda, M. et al. Solitary ulcer of the rectum: report of a case and review of the literature. Gastroenterol Jpn 16, 286–294 (1981). https://doi.org/10.1007/BF02815810
Received:
Accepted:
Issue Date:
DOI: https://doi.org/10.1007/BF02815810