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PubMed1992

Lack of association among typical congenital hypertrophy of the retinal pigment epithelium, adenomatous polyposis, and Gardner syndrome.

BACKGROUND: It has been recently documented that multiple bilateral pigmented lesions at the level of the retinal pigment epithelium may be an indicator of patients with familial adenomatous polyposis who are prone to develop intestinal cancer, particularly if there is a positive family history of these intestinal disorders. Although atypical, such lesions have been called congenital hypertrophy of the retinal pigment epithelium (CHRPE). This study was undertaken to determine whether the typical lesions of CHRPE, seen frequently by ophthalmologists, also were indicators of familial adenomatous polyposis. METHODS: Review of charts and follow-up studies were performed on all patients diagnosed and coded as having solitary CHRPE or its multifocal variant (congenital grouped pigmentation; bear tracks). Patients and their physicians were contacted by telephone to complete a detailed questionnaire designed to detect signs or symptoms of familial adenomatous polyposis or Gardner syndrome among these patients with CHRPE and their relatives. RESULTS: Of the 132 patients with previously diagnosed CHRPE, there were none with familial adenomatous polyposis, Gardner syndrome, or intestinal cancer, and only one patient had a history of intestinal polyps. Among more than 2000 of their blood relatives, only 20 had intestinal polyposis or colonic cancer (1%). This is much lower than would be expected from a survey of patients with the typical fundus lesions seen with familial adenomatous polyposis. CONCLUSIONS: It appears that solitary CHRPE and congenital grouped pigmentation differ clinically from the multiple pigmented lesions seen with familial adenomatous polyposis and that patients with these conditions, as well as their relatives, are not at a greater risk of developing intestinal cancer.

Adenomatous Polyposis Coli↗
PubMed1976

Nature of juvenile polyps in the large intestine.

Forty-five juvenile polyps were found among approximately 390 mucosal polyps of the large intestine. Various degrees of epithelial pseudostratification were found among juvenile polyps and the larger-sized polyps of both juvenile type and adult one tended to have a more advanced degree of epithelial pseudostratification. The glands of both juvenile polyps and adult ones revealed a closely resembled or almost identical pattern of size-distribution-pattern of the glandular size in both normal mucosae and hyperplastic mucosae of long-standing ulcerative colitis. Transition of juvenile polyps into adult ones was suggested by the cases with increase in numbers of glands and their orifices to the surface of the polyp and there were some polyps showing an intermediate form between juvenile polyps and adult ones. On the basis of histologic characteristics and age incidence of the patients, it may be said that a juvenile polyp is an immature variant of an adult polyp.

Adolescent↗
PubMed1992

[Improvement in the resection of intestinal Peutz-Jeghers polyps].

Polyps in patients with Peutz-Jeghers syndrome (PJS) are found in the large and small bowels. In the past, intestinal polyps were treated mainly by multiple abdominal operations. The authors reported resecting 259 polyps in 13 patients with PJS by laparotomy plus colonoscopy via small bowel incision for small intestines, and/or plus colonoscopy via the anus for ileum and large bowel polyps at a time combined with periodic colonoscopy via the anus for colorectal polyps. The largest polyp resected in this way was 10 x 9 x 8 cm. As many as 46 polyps were resected by snare polypectomy at a single colonoscopic attempt. Using the above-mentioned methods, multiple intestinal polyps can be resected more thoroughly, and postoperative recurrence and complication became less frequent.

Adolescent↗
PubMed1989

Mesenteric fibromatosis in Gardner's syndrome.

Gardner's syndrome, first described in the 1950s, is a composite clinical entity characterized by intestinal polyps, osteomas, fibromas, and epidermal cysts. A case of mesenteric fibromatosis in Gardner's syndrome is presented. Whereas most of the literature on this subject stresses the significance of the intestinal polyposis and its premalignant potential, in this paper the serious significance of the intraabdominal fibromas is stressed. This case demonstrates an unusual complication of erosion of this tumor into the small intestine, resulting in multiple intestinal fistulae with abscess formation. Surgical management of this problem necessitated resection of the fibroma and involved small intestine. A review of the literature is presented.

Adult↗
PubMedDate not supplied

Small-intestinal involvement in familial polyposis diagnosed by operative intestinal fiberscopy: report of four cases.

Operative intestinal fiberscopy, in which the duodenal fiberscope was introduced during laparotomy for colectomy in familial polyposis via the enterotomy opening, permitted the demonstration of small intestinal polyps in six of seven consecutive cases. Four of the six patients had adenomatous polyps in the proximal jejunum, including one patient with the concomitant presence of ileal adenomas. Polyposis due to lymphoid hyperplasia in the terminal ileum was found in three patients. Preoperative upper gastrointestinal surveys revealed adenomas in the duodenums of all seven patients, adenomas in the gastric antrum in three, and multiple hamartomas in the gastric corpus in two. Thus, in familial polyposis or Gardner's syndrome, more or less the entire gastrointestinal tract seems to be involved and the term "gastrointestinal polyposis" seems to describe these conditions.

Adenoma↗
PubMed1979

Desmoid tumors and mesenteric fibromatosis in Gardner's syndrome: report of kindred 109.

Kindred 109, from which Gardner's syndrome was first described, now has 224 members, 28 of whom have inherited the syndrome. Among the group of 28, desmoid tumors or mesenteric fibromatosis or both have developed in eight (29%). In four patients, the fibrous dysplasia appeared to originate in postsurgical abdominal incisional scars; in two of these cases, the mesenteric involvement resulted in death. In one patient, the process was present in the mesentery at the original operation. In three patients, the fibrous dysplastic lesions were extraincisional, with no known trauma associated with their occurrence.

Abdominal Muscles↗
PubMed1977

Diet and metabolism: large-bowel cancer.

Epidemiological data demonstrate that colon cancer incidence is associated mainly with high dietary fat consumption. Studies in metabolic epidemiology indicate that high fat intake influences both the amount and type of bile acids and neutral sterols and intestinal microflora acting on these compounds which may contain tumorigenic activity for the colon. This is compatible with the results of studies comparing populations with high or low risk for the colon cancer and patients with colon cancer.

Bacteria↗
PubMed1975

Epidermoid cysts, polyposis coli and Gardner's syndrome.

One hundred and ninety-six members of 15 families with Gardner's syndrome were investigated to determine the type of skin cyst that is part of this syndrome. These were shown to be epidermoid cysts and not pilar cysts or steatocystoma multiplex. They were solitary or multiple and seldom large and disfiguring. The skin cysts often occurred before the intestinal polyps were detectable. The presence of epidermoid cysts in children should be an indication for sigmoidoscopy when the child reaches the age of 14 years, and at 3-yearly intervals thereafter up to the age of 30 years, whether or not there is a family history of polyposis coli. At present it is not possible to say if Gardner's syndrome is the same as, or different from, familial polyposis. Until all patients with colonic polyps have a full clinical examination, looking for skin cysts and osteomas, X-rays of the skull and long bones to detect osteomas and dental X-rays for abnormalities of the teeth this question will remain unanswered.

Adolescent↗