One-stage repair of unilateral cleft lip and palate. Dentofacial treatment outcome.
A scientific essay in Medical Sciences
DOCTORAL THESIS defended in public on 8th of June 2011
Chapter 1 is a brief introduction of the subject of cleft lip and palate to the reader. Etiology and epidemiology of the cleft deformity, including Polish epidemiological data, are discussed first. Although the prevalence of cleft lip and palate is relatively low (1 - 2 per 1000 live births), there is a considerable number of children with clefts requiring comprehensive and prolonged treatment worldwide. Complete unilateral cleft lip and palate (UCLP) is one of the most challenging to treat types of the cleft deformity. Facial morphology and treatment strategies in UCLP, including the rationale of employing a 3-stage surgical protocol with delayed hard palate closure as practiced in the Radboud University Nijmegen, are subsequently discussed. Then, the overview and history of a 1-stage simultaneous repair of UCLP is presented. This is followed by a description of the Warsaw approach - a protocol based on 1-stage simultaneous closure practiced in the Warsaw Cleft Centre at the Institute of Mother and Child, Warsaw, Poland. Finally, the methodology of evaluation of treatment outcome in UCLP - single-center versus intercenter research - is explored.
In chapter 2 the results of evaluation of midfacial morphology following a 1-stage repair of UCLP are presented. In comparison with non-cleft Polish children, the maxilla in prepubertal subjects with cleft was found shortened, retruded and rotated posteriorly and the maxillary incisors were retroclined. The amount of deviation of midfacial morphology in the 1-stage group was, however, comparable with that found in published reports that examined children with UCLP treated with different methods.
Chapter 3 describes the results of evaluation of mandibular morphology and spatial position following 1 -stage simultaneous repair of UCLP. The mandible in prepubertal children was found to be retruded and at a larger inclination to the cranial base as compared with controls. Both total mandibular length and length of the mandibular body were smaller by 2 mm than in the control group, whereas height of the ramus and gonial angle were similar in both groups. It was concluded, however, that, as was the case with midfacial morphology, the amount of deviation in the 1-stage group was comparable with that found in other published reports.
In chapter 4 the results of within-the-center evaluation of dental arch relationship in a sample of 28 consecutive cases are presented. Two examiners rated the dental arch relationship with the GOSLON Yardstick on the basis of photographs. It was found that 57% of patients were assigned the GOSLON 1 or 2 categories (very good and good outcome), 32% were rated 3 (average outcome), and 11% were rated 4 or 5 (poor and very poor outcome). It was concluded that dental arch relationship following one-stage repair was comparable with the results of other centers with a favorable treatment outcome.
The results of the Warsaw-Oslo inter-center comparison of dental arch relationship are resented in chapter 5. The dental models of two samples of 61 consecutively treated patients, matched regarding age and gender, were evaluated with the GOSLON Yardstick by a panel consisting of 4 examiners. The study models were given random numbers to blind their origin. The intra- and inter-rater agreement was high. No difference in dental arch relationship between Warsaw and Oslo groups was found (mean GOSLON score = 2.68 and 2.65 for Warsaw and Oslo samples, respectively). The distribution of the Goslon grades was similar in both groups. It was concluded that the dental arch relationship following 1-stage repair (Warsaw protocol) was comparable with the outcome of the Oslo Cleft Team’s protocol.
The results of the Warsaw-Nijmegen inter-center comparison of dental arch relationship are described in chapter 6. The dental casts of 61 consecutively treated children were assigned random numbers to blind their origin. 4 raters graded dental arch relationship and palatal morphology using the EUROCRAN Index. The intra- and inter-rater agreement was moderate to very good. Dental arch relationship in the Warsaw 1-stage sample was less favorable than in Nijmegen 3-stage group (mean scores 2.58 and 1.97 for 1-stage and 3-stage samples, respectively;p < 0.000). Palatal morphology in the 1-stage sample was more favorable than in the 3-stage group (mean scores 1.79 and 1.96 for 1-stage and 3-stage samples, respectively;p = 0.047). It was concluded that the dental arch relationship following 1-stage repair (Warsaw protocol) was less favorable than that following a 3-stage protocol, whereas the palatal morphology in the Warsaw sample was more favorable than in the Nijmegen sample.
Chapter 7 describes nasolabial esthetics after a 1-stage (Warsaw group, 41 boys and 19 girls) or a 3-stage (Nijmegen group, 30 boys and 18 girls) treatment protocol. 4 components of the nasolabial appearance: nasal form, nasal deviation, mucocutaneous junction, and profile view were assessed by 4 raters with the aid of a 5-grade esthetic index of Asher-McDade. Nasal form was judged as the least esthetic in both groups and graded 3.1 (SD = 1.1) and 3.2 (SD = 1.1). Nasal deviation, mucocutaneous junction, and profile view were scored from 2.1 (SD = 0.8) to 2.3 (SD = 1.0) in both groups. Treatment outcome following the Nijmegen and Warsaw protocols was comparable. Neither overall, nor any of the 4 components of the nasolabial appearance showed inter-center difference (p > 0.1). It was concluded that the nasolabial appearance following the Warsaw and Nijmegen protocols was comparable.
Finally, in chapter 8, a general discussion of the methodological problems encountered during this investigation is presented. The overall outcome of the Warsaw protocol is also critically discussed and limitations of this study - emphasized. This chapter ends with suggestions for further research, particularly an evaluation of speech and a final assessment of the outcome once craniofacial growth is complete.