Funds are requested to support a one-day workshop on issues related to the design and analysis of etiologic studies for oral facial clefts (OFC). The workshop will be held on April 19, 1998, immediately prior to the 55th Annual meeting of the American Cleft Palate-Craniofacial Association (ACPA), at the Renaissance Harbour Place Hotel in Baltimore, Maryland. The workshop will be attended by approximately 40 members of the International Consortium for Oral Clefts Genetics (ICOCG). This consortium is composed of molecular and statistical geneticists, epidemiologists and clinicians who are united by their efforts to identify the causes of OFC. The members of ICOCG recognize that efforts to understand the etiology of OFC have been hampered by small study samples and lack of comparability between studies. Hence, the goal of the proposed workshop is to develop guidelines for data collection and analysis that will facilitate collaborative research ventures, and increase the comparability of results from independent studies. The proposed workshop will focus on two major topics: 1) ICOCGs role in the international research arena; and 2) standardization of data collection and analysis. The international role of ICOCG will be discussed by three invited speakers (Drs. Barmes, Mossey and Shaw). The standardization of the field will be topically discussed in four working groups. These topics will be addressed by the development of guidelines in four specific areas: 1) diagnostic/phenotype assessment; 2) molecular genetic studies; 3) collection and analysis of epidemiologic data; and 4) collection and analysis of genetic data. Following this workshop, ICOCG will advise the NIDR, and other interested agencies/programs, on issues related to the design and analysis of etiologic studies for OFC. Specifically, the guidelines developed during this workshop will be provided as a unifying framework for future OFC studies. This framewor will facilitate pooling of data from independent studies, meta-analyses and replication of study findings. Hence, it will reduce both the number of independent investigations and the time required to delineate the causes of, and preventive strategies for OFC. Ultimately, this will translate into decreased spending of U.S. tax dollars on OFC research, and a reduction in OFC-related costs to the healthcare system.
