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      Nonsyndromic cleft lip and palate, gastric cancer and tooth agenesis

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          Abstract

          Background

          To determine the frequency of nonsyndromic cleft lip and/or palate (NSCL/P) in first-degree relatives and to analyze the prevalence of tooth agenesis in patients with gastric cancer.

          Material and Methods

          This cross-sectional, observational, case-control study included 798 patients attended at hospital Santa Casa in Montes Claros, Minas Gerais and Alfa Institute of Gastroenterology of the Federal University of the Minas Gerais. Information on basic demographic data and tooth agenesis of both groups and their family history of NSCL/P in first-degree relatives were evaluated. The collected information was stored in a database and analyzed using statistical program SPSS® version 21.0 and the values with p<0.05 were considered statistically significant.

          Results

          Of the 798 patients, 113 (14.16%) consisted of the case group and 685 of the control group (85.84%). Non-Caucasian males were the most affected, although no differences among the groups were detected. Of all participants (n=798), 66 (8.27%) presented tooth agenesis and 25 (3.13%) presented oral cleft in first degree relative.

          Conclusions

          Our results no found increase in the frequency of tooth agenesis in patients with gastric cancer and in the frequency of NSCL/P in the first-degree relatives of patients with gastric cancer.

          Key words:Nonsyndromic cleft lip and/or palate, tooth agenesis, gastric cancer.

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          Most cited references26

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          Mutations in AXIN2 cause familial tooth agenesis and predispose to colorectal cancer.

          Wnt signaling regulates embryonic pattern formation and morphogenesis of most organs. Aberrations of regulation of Wnt signaling may lead to cancer. Here, we have used positional cloning to identify the causative mutation in a Finnish family in which severe permanent tooth agenesis (oligodontia) and colorectal neoplasia segregate with dominant inheritance. Eleven members of the family lacked at least eight permanent teeth, two of whom developed only three permanent teeth. Colorectal cancer or precancerous lesions of variable types were found in eight of the patients with oligodontia. We show that oligodontia and predisposition to cancer are caused by a nonsense mutation, Arg656Stop, in the Wnt-signaling regulator AXIN2. In addition, we identified a de novo frameshift mutation 1994-1995insG in AXIN2 in an unrelated young patient with severe tooth agenesis. Both mutations are expected to activate Wnt signaling. The results provide the first evidence of the importance of Wnt signaling for the development of dentition in humans and suggest that an intricate control of Wnt-signal activity is necessary for normal tooth development, since both inhibition and stimulation of Wnt signaling may lead to tooth agenesis. Our findings introduce a new gene for hereditary colorectal cancer and suggest that tooth agenesis may be an indicator of cancer susceptibility.
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            Gastric cancer: epidemiologic aspects.

            A multifactorial and multistep model of gastric cancer (GC) is currently accepted, according to which different environmental and genetic factors are involved at different stages in the cancer process. The aim of this article is to review the most relevant information published on the relative contribution of genetic and environmental factors. Large meta-analyses confirmed the association between IL8, IL10, TNF-b, TP53 and PSCA, while genetic variation at different genes such as XPG, PLCE1, HFE, ERCC5, EZH2, DOC2, CYP19A1, ALDH2, and CDH1 have been reported to be associated with GC risk. Several microRNAs have also been associated with GC and their prognosis. Cohort studies have shown the association between GC and fruit, flavonoid, total antioxidant capacity, and green tea intake. Obesity was associated with cardia GC, heme iron intake from meat with GC risk. Several large meta-analyses have confirmed the positive association of GC with salt intake and pickled foods and the negative association with aspirin use. © 2013 John Wiley & Sons Ltd.
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              Familial gastric cancer: guidelines for diagnosis, treatment and periodic surveillance.

              Hereditary diffuse gastric cancer (HDGC) is a relatively rare disorder, with a mutated CDH1 gene as the only known cause. Carriers of a germline mutation in CDH1 have a lifetime risk of >80% of developing diffuse gastric cancer. As periodic gastric surveillance is of limited value in detecting early stages of HDGC, prophylactic gastrectomy is advised for this patient group. Little is known about other types of familial gastric cancer. The Dutch working group on hereditary gastric cancer has formulated guidelines for various aspects of medical management for families and individuals at high risk of developing gastric cancer, including criteria for referral, classification, diagnostics, and periodic gastric surveillance. These guidelines are not limited to HDGC and are therefore partially complementary to the guidelines on hereditary diffuse gastric cancer of the international gastric cancer linkage consortium (IGCLC 2010). In order to optimize the care and increase the knowledge on hereditary gastric cancer it is important to centralize medical care for these patients. National and international collaboration is warranted to improve the quality of research by increasing the size of study cohorts.
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                Author and article information

                Journal
                Med Oral Patol Oral Cir Bucal
                Med Oral Patol Oral Cir Bucal
                Medicina Oral S.L.
                Medicina Oral, Patología Oral y Cirugía Bucal
                Medicina Oral S.L.
                1698-4447
                1698-6946
                January 2018
                24 December 2017
                : 23
                : 1
                : e44-e48
                Affiliations
                [1 ]School of Medicine, State University of Montes Claros, Unimontes, Montes Claros, Minas Gerais, Brazil
                [2 ]School of Dentistry, State University of Montes Claros, Unimontes, Montes Claros, Minas Gerais, Brazil
                [3 ]Department of Oral Diagnosis, School of Dentistry, State University of Campinas, FOP-Unicamp, Piracicaba, São Paulo, Brazil
                [4 ]Alfa Institute of Gastroenterology, Federal University of the State of Minas Gerais, UFMG, Belo Horizonte, Minas Gerais, Brazil
                [5 ]Center for the Rehabilitation of Craniofacial Anomalies, University of José do Rosário Vellano, Unifenas, Alfenas, Minas Gerais, Brazil
                Author notes
                State University of Montes Claros School of Medicine Bário street, 258, Edgar Pereira Montes Claros, Minas Gerais Brazil, Zip Code: 39400-167 , E-mail: mfleitef@ 123456gmail.com

                Conflict of interest statement: The authors state that they have no conflicts of interest.

                Article
                22132
                10.4317/medoral.22132
                5822538
                29274157
                3ab1f1f5-eca2-496f-bdba-ab9688c95d99
                Copyright: © 2018 Medicina Oral S.L.

                This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 9 October 2017
                : 10 August 2017
                Categories
                Research
                Oral Medicine and Pathology

                Surgery
                Surgery

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