A six-center international study of treatment outcome in patients with clefts of the lip and palate: Part 1. Principles and study design.

This article describes the design of an intercenter comparative study of treatment outcome in the treatment of children with a unilateral complete cleft of the lip and palate. The rationale and aims of this study are defined and treatment schemes of the participating centers are described. The findings are presented in a series of three papers (Parts 2, 3, and 4) dealing with the comparison of craniofacial form, dental arch relationships, and nasolabial appearance. In Part 5, conclusions and general recommendations regarding future research are discussed.

A six-center international study of treatment outcome in patients with clefts of the lip and palate: Part 2. Craniofacial form and soft tissue profile.

The craniofacial morphology and the soft tissue profile were evaluated in this part of the intercenter study of the European Cleft Lip and Palate Research Group. The sample was comprised of cephalometric x-rays of the full cohort of 151 cases from the six European cleft palate centers. The facial morphology in complete unilateral cleft lip and palate patients was evaluated by means of roentgen cephalometry. Approximately 25 consecutive cases from each of six European cleft palate centers were compared. Only one center showed notable and consistent differences from the others. A contributing factor for these differences may be an inconsistent treatment regimen with many surgeons involved. Analysis of the soft tissue profile between the centers showed more pronounced differences than analysis of the skeletal profile. The treatment outcome in centers with more complex or expensive programs was no better than those centers using simpler management approaches.

A six-center international study of treatment outcome in patients with clefts of the lip and palate: Part 3. Dental arch relationships.

One hundred and forty-nine dental casts of subjects with complete unilateral clefts of the lip and palate from six European cleft palate centers were assessed by means of the Goslon Yardstick. The Yardstick proved capable of discriminating between the quality of the dental arch relationships between the six centers. Two centers showed especially poor results. Three centers obtained satisfactory results although differing surgical techniques were used in these centers. One of the centers showing satisfactory dental arch relationships employed a more complex and expensive treatment program than the other two centers, which both used simpler centralized treatment regimens.

A six-center international study of treatment outcome in patients with clefts of the lip and palate: Part 4. Assessment of nasolabial appearance.

One hundred and fifteen frontal and profile photographs of the nasolabial area of subjects with complete unilateral clefts of the lip and palate from six European centers were assessed. Four components of the nasolabial area were rated separately by a panel of judges using a five-point scale of attractiveness. The Tukey multiple comparison test showed significant differences between the centers. The relative position of the six centers in this study followed a similar pattern to their respective positions in the cephalometric and dental cast studies.

A six-center international study of treatment outcome in patients with clefts of the lip and palate: Part 5. General discussion and conclusions.

Part 5 is the final part of a series of five articles reporting on an international, multicenter clinical audit of treatment outcome for complete UCLP. A number of recommendations for the methodology of future studies is made especially with respect to entry criteria, sample size, assumptions of homogeneity, and the reproducibility and validity of outcome measures. The findings of the present study regarding clinical procedures are presented tentatively, and improvement and extension of the methodology are required. It appears, however, that acceptable results can be achieved by different programs and ultimately clinical choices may be based on factors such as complexity, costs, and demands of treatment. Standardization, centralization, and the participation of high volume operators were associated with good outcomes, and nonstandardization and the participation of low volume operators with poor outcomes. Therapeutic factors associated with good outcomes were the employment of a vomer flap to close the anterior palate, and poor outcomes with primary bone grafting and with active presurgical orthopedics.

Dominantly inherited cleft lip and palate in two families.

Two families with non-syndromic cleft lip and cleft palate are described. The linear pattern of inheritance through several generations is difficult to explain by conventional multifactorial models. The pedigrees strengthen the suggestion that a dominantly inherited mutation exists with a major influence on clefting of the lip and palate alone.

A long-term study of rapid maxillary expansion and bone grafting in cleft lip and palate patients.

Rapid maxillary expansion followed by iliac crest bone grafting was a common procedure in cleft lip and palate patients until a few years ago. This study is based on patients who had rapid expansion in the early 1960s to late 1970s. The object of the study was to assess the long-term stability of the expansion. Over 80 sets of records were used. 65 per cent of the patients had iliac crest bone grafting at the completion of the expansion. Transverse measurements of the maxillary arch both anteriorly and posteriorly, showed that bone grafting did not alter the overall pattern of relapse. In cases where all posterior retention had been removed, the relapse was almost complete, whether bone grafted or not.

The Goslon Yardstick: a new system of assessing dental arch relationships in children with unilateral clefts of the lip and palate.

The Goslon (Great Ormond Street, London and Oslo) Yardstick is a clinical tool that allows categorization of the dental relationships in the late mixed and or early permanent dentition stage into five discrete categories. Cases are allocated to these categories on a value judgment basis by reference to the anchor groups of the Goslon Yardstick. The categorization was sufficiently sensitive to distinguish the treatment results at different centers in this study. It is proposed that the Goslon Yardstick should facilitate cross-center studies.