Oral Maxillofac Surg DOI 10.

1007/s10006-013-0425-2

ORIGINAL ARTICLE

Facial growth evaluation of complete unilateral cleft lip and palate operated patients: a cleft reference center in Paraba, Brazil, using the GOSLON yardstick
Rosa Helena Wanderley Lacerda & Terumi Okada Ozawa & Tnia Braga Ramos & Paulo Germano Cavalcanti Furtado & Lindalva Alves de Oliveira & Andressa Feitosa Bezerra de Oliveira

Received: 13 February 2013 / Accepted: 1 July 2013 # Springer-Verlag Berlin Heidelberg 2013

Abstract Objectives To evaluate the surgical outcomes of patients with complete unilateral cleft lip and palate (CUCLP) operated on by a single surgeon of the cleft reference center of the Lauro Wanderley University Hospital at the Federal University of Paraiba. Methods Forty-four individuals' dental casts diagnosed with CUCLP, born between 1995 and 2002, mean age of 11 years, were evaluated by three calibrated orthodontic specialists and scored by the Great Ormond Street, London and Oslo (GOSLON) yardstick on two occasions. The scores were compared with those observed in other centers around the world. The Kappa test was applied to evaluate the intra- and inter-examiner agreement. Descriptive statistics was applied for the GOSLON yardsticks core. Results The mean GOSLON score was 2.75. For the GOSLON yardstick, 43.2 % of the sample presented scores 1 and 2, 31.8 % had score 3, and 25 % were with scores 4 and 5. There was very

good intra- and inter-examiner Kappa agreement in the application of the GOSLON yardstick. Conclusions The data suggest favorable outcomes, with 75 % of cases with no need of orthognathic surgery. The Kappa values confirmed the high reproducibility of the GOSLON yardstick. Keywords GOSLON yardstick . Cleft lip and palate . Maxillary growth

Introduction Cleft lip and palate, a common malformation [4, 26, 29] and causes variable magnitudes of impairment to the face (middle face growth and facial esthetics) and velopharyngeal function (speech), affecting the individual's life from a psychosocial standpoint [7]. This impairment is directly related to primary cheiloplasty and palatoplasty surgery performed in early childhood [10, 22, 27, 29]. Complete unilateral cleft lip and palate (CUCLP), concomitantly affecting the lip and palate, is more prevalent [22, 26] and causes more severe deformity, demanding more complex and longer treatment protocols [26]. The search for an ideal protocol in the care of patients with clefts have encouraged researchers [5, 9, 18, 20, 25] to compare their results between cleft centers all over the world, aiming to contribute to the analysis of quality and awareness and encourage professionals which protocols should be followed [30]. Several evaluation methods of dental occlusion in individuals with complete unilateral cleft lip and palate are described in the literature [8, 11, 17, 23]. In 1987, Mars et al. [16] published a method called Great Ormond Street, London and Oslo

R. H. W. Lacerda (*) : T. B. Ramos : P. G. C. Furtado : L. A. de Oliveira Cleft Center, HULW, UFPB, Joo Pessoa, Brazil e-mail: rhelenawanderley@msn.com T. O. Ozawa Dental Division, HRAC-USP, Bauru, So Paulo, Brazil T. O. Ozawa UNESP, Araraquara, So Paulo, Brazil A. F. B. de Oliveira Morphology Department, Federal University of Paraiba, Joo Pessoa, Brazil A. F. B. de Oliveira Pediatric Dentistry, Pernambuco University, Recife, Brazil

Oral Maxillofac Surg

35.00% 30.00% 25.00% 20.00% 15.00% 10.00% 5.00% 0.00% G1 G2 G3 G4 G5

Fig. 1 Graph representing the GOSLON scores of cases treated in the cleft center at HULW, Joo Pessoa. The x axis represents groups 1 to 5 of the GOSLON score, and the y axis indicates the percentage of cases per group

Table 1 Intra-examiner reproducibility Examiner Ex 1 Ex 2 Ex 3 Kappa value K =0.990 K =0.978 K =0.974 Standard Error of Kappa 0.010 0.015 0.018

(GOSLON) yardstick based on the anteroposterior, transverse, and vertical evaluation of dental casts [15, 19]. The dental casts are scored into five groups according to the occlusal characteristics and respective prognoses as follow: group 1, positive overjet, with normal or lingual inclination of maxillary incisors, no crossbite or open bite, satisfactory morphology of the maxillary arch with excellent prognosis; group 2, positive overjet, with normal or buccal inclination of maxillary incisors, unilateral crossbite or tendency to crossbite, and tendency to open bite adjacent to the cleft with good prognosis; group 3, edge relationship with normal or buccal inclination of maxillary incisors or negative overjet with lingually inclined incisors and tendency to open bite adjacent to the cleft with regular prognosis; group 4, negative overjet, with normal or buccal inclination of maxillary incisors and tendency to uni/bilateral crossbite and to open bite at the cleft area with poor prognosis; and group 5, negative overjet with buccal inclination of maxillary incisors with bilateral crossbite and altered morphology of the maxillary arch with very poor prognosis [16]. The GOSLON yardstick was widely accepted by the scientific community and is used in most studies in the literature [3, 10, 14, 18, 19, 28] due to its simplicity and high reproducibility. Mossey et al. [20] highlighted that this methodology requires professional experience as orthodontic correction can mask deformities.

In Brazil, the first study using the GOSLON yardstick was conducted in 2001 [22] to evaluate the outcomes of the Rehabilitation of Craniofacial Anomalies Hospital at So Paulo University (HRAC-USP) in Bauru City. This hospital is also a reference center for the cleft lip and palate treatment in Brazil. The aim of this study was to evaluate the maxillary growth outcomes of all individuals with CUCLP, born between 1995 and 2002, that attended the cleft center of the Lauro Wanderley University Hospital at the Federal University of Paraiba (HULW-UFPB) using the GOSLON yardstick.

Material and methods Ethical aspects This cross-sectional observational study was approved by the IRB of the HULW-UFPB under protocol no. CEP/HULW 461/10, cover no. 356967, and CAA no. 0372.0.126.000-10. Sample The sample composed of all patients, born from 1995 to 2002, that attended the orthodontic clinic of the HULW-UFPB cleft center presenting complete unilateral cleft lip and palate. The diagnosis of the CUCLP was confirmed by the previous medical records and preoperative photographs. The exclusion criteria were patients with other types of clefts, with associated syndromes, operated in other centers, or previously submitted to any orthodontic or orthopedic procedure. A total of 44

Fig. 2 Graph representing scores of the GOSLON scores of cases treated in the cleft center at HULW, Joo Pessoa grouped as GOSLON 1+2, GOSLON 3 and GOSLON 4+5, and mean GOSLON score found

0.5 0.45 0.4 0.432

mdia= 2.75

0.35 0.3

0.318 0.25 G1+G2

HULW (n=44)

0.25
0.2 0.15 0.1 0.05 0

G3
G4+G5

Oral Maxillofac Surg Table 2 Inter-examiner reproducibility Examiners First evaluation Ex1 Ex2 Ex1 Ex3 Ex2 Ex3 Second evaluation Ex1 Ex2 Ex1 Ex3 Ex2 Ex3 Kappa value Standard error of Kappa

K =0.912 K =0.918 K =0.964 K =0.964 K =0.885 K =0.926

0.028 0.029 0.019 0.019 0.032 0.024

examiners, two were from HULW-UFPB cleft center and the third was from the HRAC-USP, a reference center in Brazil. The intra- and inter-examiner agreement was evaluated by the Kappa statistics, interpreted as described by Altman [1]. Statistical analysis The data were analyzed by descriptive techniques.

Results The results for the GOSLON yardstick score are described in Figs. 1 and 2. Among the 44 patients, 43.2 % (19 patients) scored 1 and 2 with excellent and good prognosis, 31.8 % (14 patients) had score 3 with regular prognosis requiring more complex orthodontic management, and 25 % (11 patients) received scores 4 and 5 with predicted need of orthognathic surgery. The mean GOSLON score was 2.75. The Kappa values for intra-examiner agreement ranged from 0.974 to 0.990 (Table 1), indicating very good agreement, as well as the inter-examiner Kappa values, which ranged from 0.885 to 0.964 (Table 2).

consecutive patients' dental casts (22 males and 22 females) were selected with an age range from 7 to 15 years. Surgical protocol The surgical protocol of the HULW-UFPB cleft center comprises cheiloplasty surgery at 3 months of age by the Millard technique and palatoplasty at 12 months by the von Langenbeck technique without preoperative orthopedics. All patients were operated by the same surgeon, with a mean of 75 new cases per year, including all types of clefts. Due to geographic and low socioeconomic conditions, this study sample presented variations in the age of the primary surgery, with mean age of 11 months for the cheiloplasty and 33.41 months for the palatoplasty. Dental cast analysis For the outcomes evaluation, the pre-orthodontic treatment dental casts were scored twice according to the GOSLON yardstick. Three calibrated examiners, specialists in Orthodontics and with experience, in cleft lip and palate treatment score the dental casts by the GOSLON yardstick, but not trained by the GOSLON team. The dental casts were evaluated by each examiner at different times. Among the

Discussion The present sample number in this study was similar to other studies [2, 9, 10, 12, 13], and the 44 cases were consecutive and representative of the results obtained in the HULW-UFPB cleft center. The intra- and inter-examiner agreement considered very good by Altman [1] was the same as the most studies using this method [6, 9, 21, 28]. This highlights the importance of the examiner's calibration, the specialists experience in clefts treatment [20]. For this study, the analysis agreement of the experienced examiner from other cleft reference center avoided bias in the outcomes.

Fig. 3 Graph representing the GOSLON scores found in the six centers of Eurocleft [15] and respective means

Mdia

Centro B (n=27) Centro E (n=30) Centro A (n=24) Centro F (n=19) Centro C (n=24) Centro D (n=27) 0 0.16 0.2 0.31 0.34

0.6 0.56 0.46 0.37 0.29 0.35 0.4 0.6

0.3 0.36 0.45 0.32 0.37 0.49 0.8

0.1 0.08 0.09

2.47 2.59 2.64 3.03 3.04 3.46

GOSLON 1+2 GOSLON 3 GOSLON 4+5

1.2

Oral Maxillofac Surg Fig. 4 Graph representing the GOSLON scores found in the five centers of Americleft [9] and respective means
Mdia

Centro C (n=38)

2.63

Centro E (n=35)

3.18
GOSLON 1+2
GOSLON 3

Centro D (n=38)

3.32

GOSLON 4+5
Centro A (n=18)

3.38

Centro B (n=40) 0 0.2 0.4 0.6 0.8 1

3.66
1.2

The GOSLON yardstick demonstrates favorable outcomes in which 43.2 % of cases will require only simple orthodontic treatment, 31.8 % will need more complex orthodontic treatment, and 25 % will require orthodonticsurgical treatment, i.e., with unfavorable growth prognosis. This results may be related to the fact that only one surgeon was involved who has a large experience and uses the same protocol for all surgery [6, 24], as well as not using primary bone grafts [9]. Thus, it may explain why other centers, with more than one surgeon using different protocols, have poorer outcomes. Comparing this study to the outcomes of other multicenter studies as Eurocleft ([15], Fig. 3) and Americleft ([9], Fig. 4), the percentage of cases with scores 1 and 2 is similar to center A of Eurocleft, but lower than centers B and E of this study and center A of Americleft. These lower results can be explained by the different protocol of the primary surgery used in these centers compared to the present study. However, this worse result should be analyzed bearing in mind the lower socioeconomic and geographic problems of the demographic region were the patient's live. The lower percentage of cases with orthodontic-surgical prognosis (25 %) compared to centers F, C, and D of Eurocleft and centers E, D, A, and B of Americleft were seen as satisfactory for the studied center as compared to the others.

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