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PubMedDate not supplied

[Juvenile recto-colonic polyposis. Apropos of a case in Madagascar].

The authors relate the first case of juvenile polyposis coli noticed in Madagascar. It is a sporadic case developing a serious clinic picture with an accidentally fatal end in a 7 years old Malagasy girl. They recall the principal epidemiological, anatomopathological, clinic and evolutive features which are typical of this polyposis. They wish that the way to medical progress will soon lead to better knowledge of the place of the intestinal polyposis in African countries.

Child↗
PubMed1990

[Gardner syndrome. A report of 3 cases in members of the same family].

The aim of the present report is to describe the overall mandibular and extramandibular lesions correlated to Gardner's syndrome which were found in the same family (mother and two daughters). In two cases (mother and one daughter) mandibular lesions consisted of osteoma. In the latter, these were associated with composite odontoma. In the other daughter the lesions consisted of desmoid fibroma. The two daughters revealed single colonic polyposis whereas the mother was affected by multiple colonic polyposis and subcutaneous lipoma. Such a constellation of lesions may be attributed to Gardner's syndrome, a Mendelian hereditary syndrome which is often incomplete. In fact, intestinal polyposis can only be associated with one of the various extraintestinal manifestations of the syndrome. The recognition of the syndrome is important given the high percentage of intestinal polyp-adenocarcinoma associations.

Adolescent↗
PubMed1969

Juvenile polyps with cachexia. Report of an infant and comparison with Cronkhite-Canada syndrome in adults.

Juvenile polyps occur in adults and infants; where multiple, they may be complicated by progressive cachexia with hypoalbuminemia and electrolyte depletion. We report a fatal case of multiple juvenile polyposis with cachexia, alopecia, and megalocephaly presenting in a 9-month-old infant, and review 2 additional cases in infancy. A similar syndrome in adults had been described by Cronkhite and Canada, with intestinal polyposis, nail dystrophy, hyperpigmentation, and alopecia. Histological examination of a polyp from one case of Cronkhite-Canada syndrome suggests a juvenile rather than adenomatous pathology. Multiple juvenile polyps can cause devastating enteropathy and cachexia.

Adenomatous Polyposis Coli↗
PubMed1997

Juvenile polyposis of the stomach: clinicopathological features and its malignant potential.

AIMS: To clarify a clinical entity of juvenile polyposis of the stomach compared with generalised juvenile gastrointestinal polyposis. METHODS: The clinicopathological features of juvenile polyposis dominantly involving the stomach at initial presentation were reviewed in 12 patients (three new patients and nine from the literature). These were compared with 29 cases of generalised juvenile gastrointestinal polyposis. RESULTS: There were three men and nine women with juvenile polyposis of the stomach, aged 10-63 years. Hypoproteinaemia was present in nine patients, anaemia in seven, and a family history of intestinal polyposis in seven. No patient presented with a congenital abnormality. During the observation period, two patients developed colonic juvenile polyps. Gastric polyps invariably affected the antrum and extended to the fundus, eventually becoming more numerous, larger, and more pedunculated. Ten patients required gastrectomy for associated malignancy or uncontrolled protein losing gastropathy. Histological examinations of the resected specimens demonstrated neoplastic tissue arising from juvenile polyps in four of the 12 patients. Atypism in these mixed polyps varied from adenoma to well or moderately differentiated adenocarcinoma. CONCLUSIONS: Juvenile polyposis of the stomach has malignant potential, and may be a separate entity from generalised juvenile gastrointestinal polyposis.

Adenocarcinoma↗
PubMed1987

Peutz-Jeghers polyposis associated with carcinoma of the digestive organs. Report of three cases and review of the literature.

Three cases of Peutz-Jeghers polyposis with carcinoma of the digestive organs are studied. Although mucocutaneous pigmentation was not present in two of the three patients, the features of intestinal polyposis are consistent with those of Peutz-Jeghers syndrome. One patient had a carcinoma of the pancreas and the other two had carcinomas with colonic Peutz-Jeghers polyps. Previous reports on carcinomas associated with Peutz-Jeghers syndrome are reviewed. An unusual location in the gastrointestinal tract, together with occurrence at an early age, characterize the carcinoma in Peutz-Jeghers syndrome. In Japanese patients, the large bowel is the site of the greatest number of carcinomas. On the other hand, Western patients showed a relatively even distribution. A possible surveillance protocol for early detection of gastrointestinal carcinoma in patients with Peutz-Jeghers syndrome is discussed.

Adenocarcinoma↗
PubMed2004

Activation of nuclear hormone receptor peroxisome proliferator-activated receptor-delta accelerates intestinal adenoma growth.

We treated Apc(min) mice, which are predisposed to intestinal polyposis, with a selective synthetic agonist of peroxisome proliferator-activated receptor-delta (PPAR-delta). Exposure of Apc(min) mice to the PPAR-delta ligand GW501516 resulted in a significant increase in the number and size of intestinal polyps. The most prominent effect was on polyp size; mice treated with the PPAR-delta activator had a fivefold increase in the number of polyps larger than 2 mm. Our results implicate PPAR-delta in the regulation of intestinal adenoma growth.

Adenoma↗
PubMed1971

Familial focal facial dermal dysplasia.

Familial focal facial dermal dysplasia is a disorder, autosomal dominant in its inheritance, characterized by areas of puckered skin at the temples with other areas of the face sometimes affected. The large family reported has 26 known affected individuals. The family considers "cancer of the stomach" as an hereditary trait, but their histories suggest some abdominal carcinoma, such as intestinal polyposis.

Abdominal Neoplasms↗
PubMed1980

[Main forms and stages in the development of diffuse polyposis of the large intestine (problems of classification)].

Diffuse polyposis of the large intestine is a hereditary disease involving mostly young people and characterized by a high index of malignization. Previously proposed various classifications of the disease do not reflect completely the numerous important aspects of this pathology. On the basis of macro- and miroscopic studies on 186 operation preparations of the large intestine removed for diffuse polyposis 4 groups of the disease were distinguished: I--the largest group including polyposis with predominance of the cell proliferation processes (various kinds of adenomas--147 observations), II--polyposis with prevalence of secretion processes (juvenile polyps--24 observations), III--polyposis with both signs (10 observations); in this mixed group juvenile polyps alternated with glandular and glandular-villous polyps; group IV included 5 observations with Peuts-Jeghers syndrome.

Humans↗
PubMed1997

Recent developments in the management of Peutz-Jeghers syndrome in childhood.

A combined surgical and endoscopic approach for managing extensive polyposis in a child with Peutz-Jeghers syndrome (PJS) is presented. Application of this technique offers the following advantages over conventional surgical practice -- 1) an accurate assessment can be made of the extent of intestinal polyposis; 2) small polyps can be easily removed endoscopically; 3) endoscopy can direct the operating surgeon to selected enterotomy sites for open excision of larger polyps and 4) multiple laparotomy, extensive intestinal resection(s) and the potential threat of short-gut syndrome in PJS patients may be avoided.

Adolescent↗
PubMed1979

[A case contribution to the Gardner syndrome].

As to its course the Gardner syndrome is similar to a chronic disease and has the following symptoms: 1. Fibro-epithelial tumours of the skin with first symptoms in early childhood. 2. Osteomes, osteofibromas, desmoids mainly located in the cranial region. 3. Disturbances of the dental development in the form of tooth retentions, persistence of milk teeth and supernumerary tooth germs. 4. Intra- and retroperitoneal or intramesenteric fibromatoses which scarcely become clinically manifest. 5. Intestinal polyposis occurring in most cases only in the third decade of life. The tumours of the skin, of the connective tissue and of the bone are always benign, whereas the intestinal polypi show a considerable tendency toward malignant degeneration. For this reason, the prognosis of the Gardner syndrome must be considered unfavourable. On the basis of a case report, the clinical symptoms and findings in the maxillofacial region are outlined.

Adult↗
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↗
PubMed1980

Metaplastic polyps and polyposis of the colorectum.

Five hundred and fifty-four colorectal metaplastic polyps have been studied histologically. Whilst most lesions were small and sessile, 16.1% measured greater than 0.5 cm in diameter and 0.9% were greater than 1 cm. The larger polyps were frequently pedunculated and occasionally showed a tubulo-villous or villous pattern. A structural similarity between the larger metaplastic polyps and colorectal adenomas is illustrated and the importance of the distinction of metaplastic from dysplastic epithelium in the differentiation of these lesions is stressed. Other unusual features of metaplastic polyps are described. Evidence is given to suggest that males have a greater propensity to develop metaplastic polyps than females. A search for men adenomas, but relatively frequent (20.8%) in juvenile polyps. Finally, seven patients with multiple metaplastic polyps of the colorectum are described, in whom a diagnosis of adenomatous polyposis had been made at some stage in their management. Six of the seven patients were males and the mean age at presentation was 37.4 years. Larger metaplastic polyps were frequent in these cases. The necessity for histological confirmation in all cases of intestinal polyposis is stressed, and the possibility that 'metaplastic polyposis' is a pathological entity is discussed.

Adenoma↗
PubMed2005

Potential role of flavonoids in the prevention of intestinal neoplasia: a review of their mode of action and their clinical perspectives.

Intestinal neoplasia (adenomas and carcinomas) can possibly be prevented by a diet rich in vegetables and fruits, treatment with aspirin and other nonsteroidal antiinflammatory drugs, and early colonoscopic removal of adenomas. Ballast, fiber, and secondary plant products could play a major role in colon cancer prevention. Recently there has been much experimental work in vitro and in vivo about flavonoids as inducers of bioprevention. Flavonoids are secondary plant products with a wide variety of beneficial biological properties, and they possess anticarcinogenic, antimutagenic, and antioxidative modes of actions. Flavonoids are the main components of a healthy diet containing fruits and vegetables and are concentrated especially in tea, apples, and onions. We will focus this review on flavonoids which are derived from tea products such as proanthocyanidins (green tea) and flavons (camomille tea). Oral supplementation with bioflavonoids derived from tea could be used in humans to prevent growth of intestinal neoplasia such as adenomatous polyps of the colon. Flavonoids are a large group of natural compounds of which only a few have been used in animal models, cell cultures, and enzyme studies to inhibit mutagenic and carcinogenic events. Their clinical mode of action was evaluated by epidemiological studies, but no intervention studies in humans have been performed so far. In vitro flavonoids can bind electrophils, inactivate oxygen radicals, prevent lipid peroxidation, and inhibit DNA oxidation. In cell cultures they increase the rate of apoptosis, inhibit cell proliferation, and angiogenesis. In vivo they can induce the activities of protective enzymes (conjugating enzymes such as glutathione transferases and glucuronosyl transferases) of the intestine and the liver. In models of intestinal polyposis, flavonoids suppress polyp formation. Some epidemiological studies show a protective effect of flavonoids contained in fruits, vegetables, and tea. Flavonoid mixtures of tea origin supplied as nutritional supplements could be studied as a new way of bioprevention of intestinal neoplasia (colon adenomas and cancer). Therefore, a controlled, randomized clinical study should be performed to evaluate the efficacy of flavonoids.

Animals↗
PubMed2005

Bridging the BMP and Wnt pathways by PI3 kinase/Akt and 14-3-3zeta.

BMP, PTEN and Wnt/beta-catenin pathways are the three signaling pathways that control normal development and regeneration of the intestine, and contribute to intestinal polyposis when aberrant inactivation or activation occurs in each of these pathways. Using genetic targeting of BMPR1A in mice, we show that inactivation of BMP signaling results in multiple polyps due to an increased number of crypts and stem cells, accompanied by enhanced Wnt signaling in all proliferating intestine cells. However the increased transcriptional activity of Wnt effecter protein, beta-catenin, is found primarily in intestine stem cells (ISCs). Concurrently, PTEN, an inhibitor of PI3K/Akt pathway, is also primarily inactivated in the ISCs, leading to activation of Akt. Thus, Akt may contribute to activation of beta-catenin in ISCs in coordination with Wnt signaling. By conducting a proteomic analysis of the beta-catenin complex, we show that 14-3-3zeta exists in the beta-catenin complex and facilitates activation of beta-catenin by Akt, which, intriguingly, appears to be predominantly in ISCs. Thus, we propose that BMP signaling plays a role in inhibition of ISC self-renewal through suppression of Wnt/beta-catenin signaling in ISC, and this cross-talk is bridged, at least in part, through the PTEN/Akt pathway and further enforced by 14-3-3zeta.

14-3-3 Proteins↗
PubMed1981

[Radiological aspect of Peutz-Jeghers syndrome: considerations on a case].

The authors present their experience on a case of Peutz-Jeghers syndrome. Initially described in 1921, the association of intestinal polyposis with pigmentation of the skin and mucous membranes has been called the Peutz-Jeghers syndrome; the pigment, which is melanin, usually involves the lips and mouth and at times the face and extremities. The syndrome appears to be a hereditable disease. Polyps may be present in the stomach, small bowel and colon, but are most frequent in the small bowel, where they produce intussusception or bleeding. Our patient, a 35-year-old man, presented abdominal pain and recurrent intestinal bleeding. Our aim was to evidence radiological signs of this syndrome. the radiographic examination of small bowel, showing multiple polyps and signs of intussusception, definitely confirmed the diagnosis. The Authors finally discuss the possibility of other syndromes associated with gastrointestinal polyposis such as familial polyposis, Cronkhite-Canada syndrome, Gardner syndrome etc.

Adult↗
PubMed1979

[Gardner's syndrome. three patients with varying manifestations (author's transl)].

Fully developed Gardner's syndrome is characterised by the triad of intestinal polyposis, osteomas and multiple soft tissue tumours. Absence of any one of these manifestations results in a bisymptomatic form. We describe three patients, one woman and two men aged 40, 46 and 51 years. In two patients the triad was fully expressed, but with varying organ involvement. The third patient was of the bisymptomatic type with osseous and intestinal lesions.

Adult↗
PubMed1988

Villous tumors of the duodenum.

Records of 32 patients with 34 villous and tubulovillous adenomas of the duodenum, treated at the Cleveland Clinic over the past 21 years, were reviewed. Twenty-two patients (69%) had complete resection of the adenoma; the incidence of malignancy was 47%. Five patients underwent a Whipple procedure; 4 patients had segmental resection of the duodenum; 12 had wide local excision of the adenoma; 1 had both a segmental resection and a local excision for two separate adenomas; and 5 patients had endoscopic excision alone. The remaining five patients underwent exploratory laparotomy alone or with palliative bypass procedures. A 28% recurrence rate was observed, all of these after segmental resection, local excision, or endoscopic excision. The highest recurrence rate was associated with local excision. The 2- and 5-year survival rates for patients with adenomas containing invasive cancer were 22% and 0%, respectively, compared to 87% and 87%, respectively, for benign adenomas (including those with carcinoma in situ). Twenty-two per cent of patients had intestinal polyposis syndromes. Duodenal adenomas were diagnosed a mean of 17 years after colectomy for polyposis, indicating the need for continued surveillance in these patients.

Adenocarcinoma↗