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Journal of Orthodontics, Vol. 29, No. 2, 113-118, June 2002
© 2002 British Orthodontic Society


Scientific Section

A cephalometric inter-centre comparison of growth in children with cleft lip and palate

M. J. Gaukroger1, J. H. Noar1, R. Sanders2 and G. Semb3

1 Eastman Dental Institute for Oral Health Care Sciences, London, UK
2 Mount Vernon Hospital, Middlesex, UK
3 University Dental Hospital of Manchester, Manchester, UK

Correspondence: Maren J. Gaukroger, Eastman Dental Institute for Oral Health Care Sciences, University College London, 256 Gray's Inn Road, London WC1X 8LD, UK. E-mail: mjgaukroger{at}hotmail.com.

Abstract

Aim: To examine whether the treatment provided by the Mount Vernon Cleft Team produces craniofacial growth outcomes comparable with that of the Oslo Team.

Location: Mount Vernon Hospital, Middlesex, UK.

Design: A retrospective cephalometric investigation.

Subjects: Seventy-five Mount Vernon children and 150 Oslo children with complete unilateral or bilateral clefts of the lip and palate

Method: The subjects were matched for age, gender, and cleft type, and their radiographs were digitized. The radiographs from each site were grouped according to patient age (9–11 or 14–16) and cleft classification (bilateral/unilateral). Patients with associated craniofacial anomalies were excluded from the study.

Results: Of the four variables studied (SNA, SNPg, NGn, sNANsPG) significant differences in maxillary growth were noted for bilateral and unilateral cleft groups at 14–16 years of age. The soft tissue profile was significantly flatter in bilateral and unilateral Mount Vernon cases at 14–16 years. The craniofacial growth exhibited by the Mount Vernon patients demonstrated 3.9–5.1 degrees reduction in maxillary prominence with respect to the Oslo sample. The bilateral cases from Mount Vernon had greater anterior face heights at 14–16 years.

Conclusion: The treatment provided by the Mount Vernon Cleft team leads to a reduced maxillary prominence in children aged 14–16 years compared with the Oslo sample. This reduction is statistically significant in unilateral cleft lip and palate.

Key words: Cephalometry, inter-centre comparison, treatment outcome, unilateral and bilateral complete cleft lip and palate

Introduction

It has been documented that children with repaired complete clefts of the lip and palate suffer adverse maxillary growth.1Go It appears that scarring produced by the primary surgical repair is a major aetiological factor.2Go Whatever the mechanism growth impairment becomes progressively apparent as patients reach maturity.3Go The Clinical Standards Advisory Group (CSAG), reported that compared with some centres in Europe, many aspects of cleft care in the UK are inadequate.4Go The Report suggested that there should be a common database made available for comparative audit studies on all cleft patients. Difficulties arise in comparing growth from samples of cases described in the literature, treated by different methods, due to potential biases. Thus, randomized controlled trials are the ideal in terms of research for comparing specific clinical methods. However, for obtaining an impression of the outcome of the overall package of care, including surgical protocols and proficiency, retrospective inter-centre studies are considered to be acceptable, providing certain criteria are met.5Go This study aims to compare the craniofacial growth of patients with unilateral and bilateral cleft lip and palate treated at Mount Vernon Hospital, Middlesex and Rikshospitalet, Oslo, Norway. It examines the hypothesis that craniofacial growth outcomes for children treated at the two centres are the same.

Materials and method

The study groups consisted of 150 patients with unilateral complete cleft lip and palate (UCLP), 50 from Mount Vernon and 100 from Oslo, and 75 patients with bilateral complete cleft lip and palate (BCLP), 25 from Mount Vernon and 50 from Oslo. Children with associated craniofacial anomalies were excluded from the study. Those with incomplete clefts and radiographs of poor quality were also excluded. The patients were divided into two groups aged 9–11 and 14–16 years. The Oslo and Mount Vernon Groups were matched for age, gender, and cleft type, and a description of the material is presented in Table 1Go. The resultant small numbers in the bilateral group decrease the power of the statistical comparisons. For this reason double matching was undertaken.


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Table 1 Age, gender, and cleft types of groups.
 
The Oslo protocol
Pre-surgical orthopaedics have never been performed in Oslo. The surgical protocol was as follows: patients with UCLP had lip closure (Millard technique) and hard palate closure using a single layer vomer flap at 3 months of age. Patients with BCLP had lip (straight line technique) and hard palate closure with a single layer vomer flap done in two stages, one side was closed at 3 months and the other at about 4–6 weeks later. The posterior palate was closed at 18 months using a modified von Langenbeck technique. All patients have had alveolar bone grafting in the mixed dentition. Secondary surgery was undertaken on an individual basis.

The Mount Vernon protocol
Pre-surgical orthopaedics was not employed at Mount Vernon, but in babies with wider clefts a reduction in cleft width was encouraged using neonatal lip adhesion. This technique was introduced to the protocol in the late 1980s and has therefore been employed in some of the 10-year-old children studied. Patients with UCLP then had lip closure with the Millard approach and a vomer flap prior to 3 months of age.

Patients with BCLP also had lip adhesion where clefts were wide, followed by straight-line closure and vomer flap. The posterior palate was closed at 4–12 months using the Wardill–Kilner pushback. Alveolar bone grafting was undertaken prior to completion of root formation of the maxillary canines. At Mount Vernon too, secondary surgery was undertaken on an individual basis.

Oslo was chosen as the reference centre because it has a large database, which meant that double matching could be undertaken. This study design would increase the statistical power of the comparison.6Go A total of 50 unilateral cleft children were analysed and comparisons made with 100 Oslo children. A group of 25 bilateral cleft children were analysed and compared with 50 Oslo views. The landmarks used were the following:

For linear measurements the distance measured was divided by the magnification factor for each unit to allow direct comparison. All cephalograms were digitized by one individual (MJG).

Forty radiographs were digitized a second time 2 weeks later to enable the cephalometric measurement error of the operator to be calculated (Table 2Go).


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Table 2 Cephalometric measurement error.
 
This paper seeks to examine the following four variables that were chosen to give a broad overview of sagittal, vertical, and soft tissue growth for clinical use. Soft tissue ANB was not employed as the study is retrospective, and it was felt that soft tissue B point may be affected by open or closed lip posture.

Results.

Student t-tests were used to compare the two samples. The results as mean values, standard deviations, confidence intervals, and P-values are tabulated in Tables 3–6GoGoGoGo.


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Table 3 Mean values, standard deviations, confidence intervals, and P-values for 10-year-olds (UCLP).
 

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Table 4 Mean values, standard deviations, confidence intervals, and P-values for 15-year-olds (UCLP).
 

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Table 5 Mean values, standard deviations, confidence intervals, and P-values for 10-year-olds (BCLP).
 

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Table 6 Mean values, standard deviations, confidence intervals, and P-values for 15 year olds (BCLP).
 
No significant differences were found in the prominence of the mandible (SNPg) for unilateral or bilateral cleft groups. The 15-year-old bilateral cases showed significant differences in facial heights (NGn). Maxillary prominence (SNA) was significantly reduced in 15-year-olds with unilateral (P < 0.0001) cleft children under the care of the Mount Vernon team. The 15-year-old bilateral cleft lip and palate cases also showed a reduction in maxillary prominence. This reduction cannot be considered significant (P = 0.0243) in view of the multiple comparisons made. The soft tissue profile was similar in the 10-year-old group for unilateral cases but tended to be flatter amongst 15-year-old unilateral cases (P = 0.0021), and also in 10- and 15-year-old bilateral Mount Vernon patients.

Discussion

Clinical audit in cleft care should ideally include the inter-related outcomes of facial growth, nasolabial appearance, dental arch relationships, speech, hearing, and burden of care, and can be achieved in a number of ways:

  1. Comparisons of records of cohorts of consecutive cases from different centres as in the Eurocleft and Scandcleft studies.11–Go17Go. To some extent this approach allows prospective planning for standardized record collection and blinded panel analysis to minimize analysis bias.
  2. Comparison (preferably blinded) of the records of one centre with an archive of consecutive matched cases from another6,Go18Go and as proposed in the establishment of a European reference archive.19Go
  3. Comparison of a team's records with an agreed set of normative values.20Go
  4. Comparison with published reports already in the literature. This last method is probably the least reliable.3Go

This study has analysed the practice of one cleft centre over a 20 year period under the care of one cleft team. The numbers reported are small despite the fact that the centre has been one of the busier cleft units in the UK, and has had an organized and committed team approach for many years. The collection of longitudinal data, however, requires extreme rigour and depends not only on the cleft team members, but also on other members of staff, such as dental technicians, secretaries, and others involved in the storage and filing of information within a hospital. These difficulties, together with patients and their families moving area during the long treatment period, makes record collection extremely difficult. The Oslo cohort was chosen as this represents a centre with an established database that has been used in a number of previous inter-centre comparisons.

The Mount Vernon cohort was assessed for facial growth at ten and fifteen years of age as this relates to the recommendations made by the Clinical Standards Advisory Group (CSAG) for milestone records.

As in the present study, the retrusive maxilla of UK patients compared with those of some other North European centres has been observed before.4,Go6,Go13,Go18,Go21,Go22Go In this study the differences may be attributable to the earlier timing of surgery or to differences in surgical protocol, i.e. the use of the Wardill–Kilner pushback. The maxillary prominence of the 10-year-old Mount Vernon children compares well with the Bristol UCLP patients of the same age.18Go It has also been suggested that there are differences in the craniofacial growth of Norwegian and British children.23Go

Conclusions

The results demonstrated in this study show that there are no significant differences in facial profile and A–P position of the maxilla or mandible as measured on lateral cephalograms for either unilateral or bilateral cleft lip and palate patients between the Mount Vernon sample and the Oslo sample at 10 years.

Patients at age 15 years with UCLP and BCLP treated at Mount Vernon had flatter facial profile, and reduced maxillary prominence compared with a matched group of patients treated by the Oslo team. This difference was significant in the unilateral cleft lip and palate patients (P < 0.0001).

Acknowledgments

The authors would like to thank Marie Pinkstone, Cleft Coordinator, for her assistance with data collection and the Restoration of Appearance and Function Trust (RAFT) for their support.

References

1 Semb G, Shaw WC. Facial growth in orofacial clefting disorders. In Turvey TA, Vig KWL, Fonseca RJ, editors. Facial clefts and craniosynostosis, Philadelphia: WB Saunders Co; 1996. pp 28–56.

2 Mars M, Houston WJB. A preliminary study of facial growth and morphology in unoperated male unilateral cleft lip and palate subjects over 13 years of age. Cleft Palate J 1990; 27: 7–10.[Medline]

3 Semb G, Shaw WC. Facial growth after different methods of surgical intervention in patients with cleft lip and palate. Acta Odontol Scand 1998; 56: 352–5.[Medline]

4 Clinical Standards Advisory Group. Cleft Lip and/or Palate. London: HMSO 1998.

5 Roberts CT, Semb G, Shaw WC. Strategies for the advancement of surgical methods in cleft lip and palate. Cleft Palate Craniofac J 1991; 28: 141–9.[Medline]

6 MacKay F, Bottomley J, Semb G, Roberts CT. A two center study of dentofacial development in the five year old child with unilateral cleft lip and palate. Cleft Palate Craniofac J 1994; 31: 372–5.[Medline]

7 Bjørk A. The relationship of the jaws to the cranium. In Lundström A, editor. Introduction to orthodontics. London: McGraw-Hill; 1960. pp 104–40.

8 Mølsted K. Kraniofacial morfologi hos børn med komplet unilateral læbe og ganespalte, Master Dissertation. Copenhagen: Royal Dental College; 1987.

9 Larson O, Nordin K-E, Nylén B, Eklund G. Early bone grafting in complete cleft lip and palate cases following maxillofacial orthopaedics. II. The soft tissue development from seven to thirteen years of age. Scand J Plast Reconstr Surg 1983; 17: 51–62.[Medline]

10 Wisth PJ. Changes in the soft tissue profile of Norwegian children from age four to ten years. A roentgen cephalometric study. Thesis. University of Bergen; 1971.

11 Friede H, Enemark H, Semb G, Paulin G, Abyholm F, Bolund S, Lilja J, Östrup L. Craniofacial and occlusal characteristics in unilateral cleft lip and palate patients from four Scandinavian centres. Scand J Plast Reconstr Hand Surg 1991; 25: 269–76.[Medline]

12 Shaw WC, Asher-McDade C, Brattström V, Dahl E, Mars M, McWilliam J, Mølsted K, Plint DA, Prahl-Andersen B, Semb G, The RPS. A six-centre international study of treatment outcome in patients with clefts of the lip and palate: Part 1. Principles and study design. Cleft Palate Craniofac J 1992; 29: 393–7.[Medline]

13 Mølsted K, Asher-McDade C, Brattström V, Dahl E, McWilliam J, Plint DA, Prahl-Andersen B, Semb G, Shaw WC, The RPS. A six-centre international study of treatment outcome in patients with clefts of the lip and palate: Part 2. Craniofacial form and soft tissue profile, Cleft Palate Craniofac J 1992; 29: 398–404.[Medline]

14 Asher-McDade C, Brattström V, Dahl E, McWilliam J, Mølsted K, Plint DA, Prahl-Andersen B, Semb, G, Shaw WC, The RPS. A six-center international study of treatment outcome in patients with clefts of the lip and palate: Part 4. Assessment of nasolabial appearance, Cleft Palate Craniofac J 1992; 29: 409–12.[Medline]

15 Enemark H, Friede H, Paulin G, Semb G, Abyholm F, Bolund S, Lilja J, Östrup L. Lip and nose morphology in patients with unilateral cleft lip and palate from four Scandinavian centres. Scand J Plast Reconstr Hand Surg 1993; 27: 41–7.[Medline]

16 Garattini G, Semb G, Brusati R, Abyholm F, Shaw WC. A comparison of dental arch relationships between consecutive cases of unilateral cleft lip and palate treated by the Milan method and the Oslo method. The Sixth European Craniofacial Congress, Manchester, 1999 (abstract).

17 Grunwell P, Brøndsted K, Henningsson G, Jansonius K, Karling J, Meijer M, Ording U, Wyatt R, Vermeij-Zieverink E, Sell D. A six-centre international study of the outcome of treatment of patients with clefts of the lip and palate: the results of a cross-linguistic investigation of cleft palate speech. Scand J Plast Reconstr Hand Surg 2000; 34: 219–29.[Medline]

18 Roberts-Harry D, Semb G, Hathorn I, Killingback N. Facial growth in patients with unilateral clefts of the lip and palate: A Two-Center Study. Cleft Palate Craniofac J 1996; 33: 489–93.[Medline]

19 Shaw WC, Semb G, Nelson P, Brattström V, Mølsted K, Prahl-Andersen B. The Eurocleft Project 1996–2000. Standards of care for cleft lip and palate in Europe. Amsterdam: IOS Press; 2000.

20 Bearn DR. Outcome assessment in unilateral cleft lip and palate. PhD Thesis, University of Manchester; 2000.

21 Mars M, Plint DA, Houston WJB, Bergland O, Semb G. The Goslon Yardstick: a new system of assessing dental arch relationships in cleft lip and palate patients. Cleft Palate J 1987; 24: 314–22.[Medline]

22 Morris DO, Semb G, Roberts-Harry DP. A comparative study of facial growth in 10-year old children with unilateral cleft lip and palate (UCLP). The Sixth European Craniofacial Congress, Manchester; 1999 (abstract).

23 Trenouth MJ, Laitung G, Naftel AJ. Differences in cephalometric reference values between five centres: relevance to the Eurocleft Study. Br J Oral Maxillofac Surg 1999; 37: 19–24.[Medline]

Received May 3, 2001; accepted October 3, 2001





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