Effect of primary surgery for cleft lip and palate on mid-facial growth

Bruish Journai ofOra/ and Maxdlofacial Surger). (1997) 35.6-10 0 1997 The British Association of Oral and Maxillofacial Surgeons

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Effect of primary surgery for cleft lip and palate on mid-facial growth A. F. Markus*,

D. S. Precious?

* Maxillofacial Surgery, Poole Hospital, Poole, Dorset, UK; 7 Maxillofacial Halifax, Nova Scotia, Canada

Surgery, Dalhousie University,

SUMMARY. Surgery for cleft lip and palate is known to have an effect on growth and development of the midface. This paper studies the outcomes in 34 consecutive lo-year-old patients with unilateral cleft lip and palate. Clinical observations of the importance of both surgical technique and the influence of cranial base morphology on maxillo-mandibular position are discussed.

deciduous canine teeth, as early as 8 weeks in utero,g it still behaves as a skeletal unit as defined by Moss.” Indeed, the premaxillary-maxillary suture can be identified even after growth has ceased, albeit only in the mid-line (Fig. 4).” Factors important in the development of the premaxilla are also important in development of the mid-face as a whole.2 Therefore, it is essential that the relationship between the nasolabial muscles and the premaxilla through the intermediary of the median septal system is correct (Fig. 5).” Failure to establish this relationship deprives the premaxilla and, therefore, the mid-face as a whole, of one of the main factors responsible for global development of the face. Facial symmetry must also be considered in the process of restoring the connection between the musculature and the premaxilla. If it is not, the inherent asymmetry in unilateral cleft lip and palate, and the symmetrical elongation in bilateral clefts, is perpetuated. The effect of palatal surgery must also be considered.13 A wide range of techniques exist, including one-stage and two-stage palatoplasties, intra-velar veloplasties and the use of vomerine mucosa to create the nasal layer in closure of the hard palate. If a significant adverse effect on growth is to be avoided, not only is it necessary to carefully reconstruct the muscles of the soft palate but also to minimize scarring. Medial displacement of the mucoperiosteum overlying the palatal shelves should be avoided because this complex tissue layer has an important influence on palatal growth in the vertical, transverse and sagittal planes.3 It has also been postulated that vomerine mucosal flaps result in a reduction in vertical height of the maxilla, retropositioning of the maxilla and over closure of the mandible.r4 The effects on growth of the vomerine flap are both complex and difficult to prove, a fact often reflected in conflicting conclusions in the literature. For example, Shaw et a1.15 considered that vomerine mucosal flaps were associated with good outcomes, but many of the same authors in this paper subsequently came to the opposite conclusion.‘6,17 The purpose of this study was to assess anterior

INTRODUCTION An important cause of the deformities in cleft lip and palate patients is displacement and underdevelopment of the divided parts. Whether the global deformity is due to true hypoplasia, hypofunction and attendant hypodevelopment, or a combination of both, the principal surgical goal is the same: to establish good function through careful muscle reconstruction which in turn will permit optimum subsequent growth and development of the facial skeleton. This principle is important both in primary and secondary cleft corrections because good function is a prerequisite to good facial aesthetics. The importance of reconstruction of all the muscles of facial expression and not just the orbicularis oris has previously been highlighted,im3 these muscles being either directly or indirectly involved in the cleft. In particular, the nasolabial muscles form a confluence with the three heads of orbicularis oris, inserting into the mid-line in the region of antero-inferior part of the nasal septal cartilage and anterior nasal spine.4 The importance of muscle reconstruction is further strengthened by histological fetal studies aged between 6 weeks and 25 weeks.’ These studies confirmed that in the cleft fetus the failure of normal muscle insertions to develop led to identifiable deformities of the nasal cartilages at a very early stage as well as abnormal development of the bone and periosteum (Figs l-3). Further corroboration of the importance of muscle function with the aid of electromyographic and magnetic resonance studies by Joo&~ confirmed the difference in muscle activity between clefts repaired by functional cheilorhinoplasty as opposed to a repair confined largely to orbicularis oris. It is important when considering methods of cleft repair, to remember the pivotal role of the premaxilla. By definition, a cleft separates areas of primary bone growth as well as separating those primary areas from secondary growth centres.8 Although it can be demonstrated that the premaxilla becomes united to the maxilla laterally, in the region of the developing 6

Effect

Fig.

1

of primary

surgery

for cleft

lip and palate

on mid-facial

growth

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Fig. 3 Fig. 1 - Non-cleft fetus - 20 weeks: symmetrical and purposeful organization. Note muscle fibres inserting into nasal sill. Reproduced with kind permission from Precious DS (who \~#as supported by Talmant JC and Kardjiev L). The surgical relevance of labioseptopremaxillary anatomy in normal and cleft lip/palate human fetuses. 50th Anniversary Meeting of the American Cleft Palate-Craniofacial Association. Pittsburgh, I9 -24 April 1993. Fig. 2 - Cleft lip fetus - 19 weeks: cleft side. Reproduced with permission from Precious DS (who was supported by Tdlmant and Kardjiev L). The surgical relevance of labioseptopremaxillary anatomy in normal and cleft lip,‘palate human fetuses. 50th Anniversary Meeting of the American Cleft Palate-Craniofacial Association. Pittsburgh, 19-24 April 1993.

Fig. 2

arch width, cranial predisposition to facial type, incisor overjet and coincidence of skeletal and dental mid-lines in 34 lo-year-old children with complete unilateral cleft lip and palate. METHODS

Lateral cephalograms were taken and the Delaire craniofacial analysisl’ used to assess the underlying cranial predisposition of the patients. Measurements were also taken from study models to assess transverse dimensions of the palate at a dento-alveolar level in the region of the canines and maxillary tuberosities. Together, radiographs and models were used to assess incisor relationship, not only as a measure of normal occlusal relationship at a dentoalveolar level but also as a meaningful, easily measured linear assessment of forward development of the mid-face. Assessment of the coincidence of the maxillary and mandibular skeletal and dental mid-lines was used as an indicator of facial symmetry. Impressions were all taken in alginate mixed to the same proportions and models were cast in plaster stone (Kaffir D mix) within 1 h. All cephalograms were taken by the same radiographer and on the same machine to minimize radiographic error. All

kind JC

Fig. 3 - Cleft lip fetus 19 weeks: non-cleft side. Reproduced with kind permission from Precious DS (who was assisted by Talmant JC and Kardjiev L). The surgical relevance of labioseptopremaxillary anatomy in normal and cleft lip/palate human fetuses. 50th Anniversary Meeting of the American Cleft Palate-Craniofacial Association. Pittsburgh. 19-24 April 1993.

radiographs were traced and all models measured 2 weeks apart and by two observers, independently. The method error, which is the standard deviation of errors in individual measurements, was tested according to Dahlberg’s formula. The maximum acceptable error in this study was 1 mm and 2’ to maintain confidence at the 5% level. The study involved the recall of 100 consecutive patients who had had primary surgery carried out over a 12-month period and who were aged 10 at the time of examination. Of those patients with adequate records, 47 were excluded (either because they had inadequate records or they had commenced orthodontic treatment). Of the remaining 53 patients, 34 had a complete unilateral cleft of the lip and palate, 11 had a cleft of the hard and/or soft palate and 8 had a bilateral cleft. This study was confined to those patients with unilateral cleft lip and palate.

RESULTS Anterior arch and intertuherosity width (Table 1)

The inter-canine and inter-tuberosity dimensions were used, as these points allowed comparison with other cleft studies and values for non-cleft children of the

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same age. lg The variations between cleft and noncleft were of no statistical significance. Measurement of forward growth and facial symmetry (Table 2) The incisor overjet, as an easily assessed linear measurement of forward development was considered within the context of the underlying cranial base. Mandibular and maxillary mid-line coincidence was also assessed. DISCUSSION

Fig. 4 - (A) Premaxillary-maxillary suture in a dry skull in a subject in the mixed dentition phase. (B) Nasal view of persistent premaxillary-maxillary suture in an adult patient undergoing a Le Fort I downfracture osteotomy.

Fig. 5 - Complete cleft of lip and alveolus - 20 weeks: deviation of nasal septum.

Table 1 -Arch

width (mm)

Anterior

Group

Posterior

34.11 k4.2 32.13k5.4

Non-cleft This study

41.76k6.3 44.45+55

The cephalometric assessment must make reference to the underlying cranial base.20 Irrespective of the cleft deformity, patients who have a cranial predisposition to a Class II facial type should not be grouped with those who have a cranial predisposition to a Class I facial type if misleading comparisons are to be avoided. The results of this study show that it is possible to achieve development more closely approaching normal in the patient with a cleft. They contrast well with the results of the study by Joos7 comparing the different outcomes between functional and nonfunctional surgery practised by two different surgeons in the same unit. In this study, in which the measure of forward mid-facial growth, the dento-alveolar relationship was compared to the underlying cranial predisposition, 80% developed an Angle Class III relationship following non-functional surgery (in this case Millard lip repair and a modified von Langenbeck palatoplasty). The results for functional repair were almost the exact opposite. Further confirmation of this trend was evident in subsequent papers by Joos8 and Adcock and Markus, both being studies of 5-year-old children, Nicolau22 studied the relationship between the orbicularis oris muscle and all the other muscles involved in the cleft deformity. He examined some of the more commonly used techniques for closure of the cleft lip and noted that none of them recognized the importance of the nasolabial muscles, which were placed in an anatomically incorrect position. The work of Delaire’ and Joo@,~ confirmed the importance of reconstruction of all the nasolabial muscles if one is to encourage development at both the primary and secondary growth centres and only in this way can normal function occur. Not only is this confirmed in the papers already cited but further corroboration comes from Horswill and Pospisilz3 in their study of nasal form in which it was shown that nasal symmetry was significantly better following functional cheilorhinoplasty than following the classic rotation-advancement technique. It is also worth considering the influence of other factors in repair, in particular those relating to cleft palate, notably the medial transposition of palatal mucosal flaps and the use of vomerine mucosa. Murison and Pigottz4 outlined their experiences with a modified von Langenbeck repair of the cleft palate not dissimilar

Effect Table 2 -Cranial Cranial Class Class Class (Delaire

predisposition,

incisor

predisposition I II III

relationship

and mid-line

Incisor 11 16 7

Cheilorhinoplasty,

33’%) 47’%, 20% Delaire

Class Class Class

of primary coincidence

surgery in 34 UCLP

relationship I II III

for cleft

lip and palate

80% 1 1‘%I 9%

growth

9

patients

Maxillaryimandibular 27 4 3

on mid-facial

Coincident Deviation Deviation

to non-cleft to cleft

mid-lines 28 5 I

82.x4 14.7% 3’%,

Palatoplasty)

to the method adopted by Malek25 in which the lateral relieving incisions are medial to the neurovascular bundle but in which a small posterior vomerine flap is used to stabilize the soft palate. The effect of the Malek repair was assessed by Ross.*~ Cephalometric assessment in lo-year-olds demonstrated excellent mid-facial development. However, facial symmetry, i.e. midline coincidence of dental skeletal and soft tissue structures, was not assessed. The method of palatal repair advocated by Delaire is not dissimilar to Malek’s, but does not involve the use of any vomerine mucosal flaps. It would seem that the results in this study confirm similarly excellent results to those of Malek. It is interesting to note that the timing of lip closure by both Delaire and Malek is, at 6 months, late compared to the increasingly commonly practised neonatal closure at 2-3 days. In conclusion, whilst the evidence points to successful outcomes using this method, it is important that the next stages of growth, between the ages of 10 and

Fig. 6 -(A) Cleft lip and palate ~ 5 months: showing deviation premaxilla. (B) Cleft lip and palate - 5 years: after functional repair, showing mid-line symmetry.

of

15 years, are carefully monitored and that the outcome at the end of this period be assessed (Fig. 6). Acknowledgements The authors for guidance manuscript.

wish to express their thanks to Professor Jean Delaire and to Mrs Maureen Peters for preparation of this

References 1. Delaire J. La cheilo-rhinoplastie primaire pour fente labiomaxillaire congenitale unilaterale. Rev Stomatol Chir Maxillofac 1975: 76: 193. 2. Markus AF, Delaire J, Smith WP. Facial Balance in Cleft Lip and Palate. I: normal development and cleft palate. Br J Oral Maxillofac Surg 1992; 30: 287-295. 3. Markus AF, Delaire J, Smith WP. Facial Balance in Cleft Lip and Palate. II: cleft lip and secondary deformities. Br J Oral Maxillofac Surg 1992; 30: 296-304. 4. Delaire J, Feve R, Chateau J-P. Coutay D, Tulasne JF. Anatomie et physiologie des muscles et du frein de la levre superieure. Rev Stomatol Chir Maxillofac 1977; 78: 93. 5. Precious DS, Delaire J. Clinical Observations of Cleft Lip and Palate. Oral Surg Oral Med Oral Pathol 1993; 75: I41 -~I 5 1. 6. Joos U. The importance of muscular reconstruction in the treatment of cleft lip and palate. Stand J Plast Reconstr Surg 1987; 21: 109. 7. Joos U. Muscle reconstruction in primary cleft lip surgery. J Cranio-maxillofac Surg 1989: 17: S- 10. 8. Joos U. Skeletal growth after muscular reconstruction for cleft lip, alveolus and palate. Br J Oral Maxillofac Surg 1995: 33: 139.-144. 9. Thibault H. Contribution a I’etude du premaxillaire. Thesis: University of Nantes 1978. 10. Moss ML. The primary role of functional matrices in facial growth. Am J Orthod 1969; 55: 566. 11. Smith WP, Markus AF, Delaire J. Primary closure of the cleft alveolus: a functional approach. Br J Oral Maxillofac Surg 1995; 33: 156-165. 12. Delaire J, Precious DS. Interaction of the development of the nasal septum, the nasal pyramid and the face. Int J Pediatr Otorhinolaryngol 1987; 12: 31 l-326. 13. 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. 14. Delaire J, Precious DS. Avoidance of the use of vomerine mucosa in primary surgical management of velopalatine clefts. Oral Surg Oral Med Oral Pathol 1985; 60: 5899597. 15. Shaw WC, Dahl E, Asher-McDade C ef trl. Six centre international study of treatment outcome in patients with clefts of the lip and palate: part 5. General discussion and conclusions. Cleft Palate-Craniofac J 1992; 29: 4 133418. 16. Molsted K, Dahl E, Brattstrom V. McWilliam J. Semb G. A six centre international study of treatment outcome in patients with clefts of the lip and palate: evaluation of maxillary asymmetry. Cleft Palate-Craniofac J 1993: 30: 22228. 17. Enemark H, Fried H. Paulin G et crl. Lip and nose morphology in patients with unilateral cleft lip and palate from four Scandinavian centres. Stand J Plast Reconstr Surg Hand Surg 1993; 27: 41-47. 18. Precious DS. Delaire J. Surgical considerations in patients

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with cleft deformities. In: Bell WH (ed.) Modern Practice in Orthognathic and Reconstructive Surgery; Vol 1. Philadelphia: W B Saunders, 1992. Bhatia SN, Leighton BC. A Manual of Facial Growth. Oxford: Oxford University Press, 1993. Ross RB. My Friend the Cranial Base: Why is it so normal? Cleft Palate J 1993; 30: 511-512. Adcock S, Markus AF. Maxillary growth in 5 year olds following functional cleft surgery. Br J Oral Maxillofac Surg 1996; (in press). Nicolau PJ. The orbicularis muscle: a functional approach to its repair in the cleft lip. Br J Plast Surg 1983; 36: i41-153. Horswill BB. Posnisil OA. Nasal svmmetrv after nrimarv cleft lip repair: compaiison between DGaire cheilorhinoplasty and modified rotation-advancement. J Oral Maxillofac Surg 1995; 53: 1025-1030. Murison MSC, Pigott RW. Medial Langenbeck: experience of a modified von Langenbeck repair of the cleft palate. A preliminary report. Br J Plast Surg 1992; 45: 4544459. Malek R, Psaume J. Nouvelle conception de la chronologie et de la technique chirurgicale du traitment des fentes labiopalatines. Ann Chir Plast 1983; 28: 237. Ross RB. Growth of the facial skeleton following the Malek repair for unilateral cleft lip and palate. Cleft PalateCraniofacial J 1995; 32: 1944198.

The Authors A.F. Markus Consultant Maxillofacial Surgery Poole Hospital Longfleet Road Poole Dorset BH15 2JB UK D.S. Precious Professor and Chair Maxillofacial Surgery Dalhousie University Halifax Nova Scotia Canada B311 355 Correspondence and requests for offprints to A.F. Markus Paper received 26 January 1996 Accepted 2 October 1996