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Secondary bonegrafting in unilateral cleft lip palate patients: indications and treatment procedure
Int. J. Oral Surg, 1985: 14: 2-10 (Key words: cleft, residual; fistula, oro-nasal; bone graft; transplantation bone; cleft lip, palate; surgery, oraf)
Secondary bonegrafting in unilateral cleft lip palate patients: indications and treatment procedure HANS ENEMARK, ERIK KRANTZ-SIMONSEN AND JAN ERIK SCHRAMM Aarhus Cleft Palate Clinic, Taleinstituttet, and Department of Oral Surgery, Kommunehospitalet, Aarhus, Denmark
A consecutive series of 62 complete unilateral cleft lip and palate patients were investigated with respect to indication for a secondary bone grafting. 2 major indications for secondary bone grafting were established. One was a symptomatic oro-nasal fistula, the other one being a bony defect in the alveolar process, which could impair orthodontic treatment and prosthodontic rehabilitation in the cleft area. The average age for bonegrafting was 12 years. After surgery, fistula-related discomfort was eliminated and speech disorders weredrastically reduced. Orthodontic uprightning of teeth and correction of mid-line deviations indicated in 50% of the patients were facilitated. In 10% of the cases with extensive defects in the alveolar process, the graft enhanced the possibility for later prosthodontic treatment. Secondarily, periodontal conditions improved. When bonegrafting was performed before canine eruption, total dental rehabilitation by orthodontic treatment was often sufficient, thus decreasing the need for later prosthodontic restoration of the cleft area. Patients who had surgery after eruption of the cleft side canine exhibited more complications. The optimal treatment sequence therefore appears to be transversal expansion of the maxilla in the late mixed dentition, followed by bone grafting. Maxillary expansion must be retained until final orthodontic and prosthodontic treatment is carried out. ABSTRACT -
(Received for publication 6 June 1983, accepted 19 February 1984)
The original purpose of bonegrafting was to stabilize the maxilla with a secondary operation 22 •2 5 and to provide bone inthe alveolar process of the maxilla, so that teeth adjacent to the cleft could erupt'", In 1958, SCHRUDDE & STELLMACH 2 3 recommended, bone grafting to be performed as a primary
procedure, and the short-term results appeared promising. However, later evaluation of these patients indicated maxillary growth retardation. FRIEDE & JOHANSSON 1 5 demonstrated almost normal development in the cleft area for 90% of the patients and tooth eruption into the graft. However, the
SECONDARY BONE GRAFTING IN CLEFTS
3
primary surgery interfered that much with tion is dependent upon bone in the cleft maxillary growth that they recommended area. discontinuance of primary bonegrafting. In theory, if growth of the upper jaw Material and methods should be completely free of surgical in- A consecutive series of 62 complete unilat eral fluence (apart from the inevitable primary cleft lip and palate patients representing all pasoft tissue repair), bone grafting would not tients born with tbis malformation in the western part of Denmark between 1964 and 1967 were be possible until the age of 18-20. During examined. All patients had their lip repaired at 2 the last 10 years different points-of-view months of age by one plastic surgeon, who used a have evolved concerning the optimal timing Tennison procedure. At the same time, the hard of the bone grafting procedure" 7.11. Nor- palate was repaired with double vomerine flaps. 22 mon ths of age, the soft palate was closed mal growth in the maxilla is sutural and At using a Wardill palatoplasty. With this type of appositional, and between the ages of 6 and surgical procedure, a residual oro-nasal fistula in 7 years, most transverse maxillary growth the anterio r pa rt of the palate often remained. has occurred". If bone grafting is delayed The fistula was always situated in the alveolar until after this age, a possible transverse process from the bottom of the alveolobuccal sulcus extending into the anterior part of the hard growth retardation can be compensated for palate (Fig. 1). This often slit-like deformity may by orthodontic treatment. not be easily observed, since the lateral segment of The two major indications for bone graft- the maxilla is usually collapsed before orthodoning are a symptomatic oro-nasal fistula and tic treatment is started. A diagnostic method to if a fistula is present, is to administrate a bony defect in the alveolar process impair- determine air from a dental syringe to the anterior part of ing orthodontic and prosthodontic treat- the palate. In cases with oro-nasal communicment. A fistula can cause patient discomfort ation, the patient wil\ feel a tickling in the nasal in regurgitation of food through it, and also cavity, always in the cleft side only. In 1972, we started secondary bone grafting on influences speech. The alveolar process pa tients with clefts using auto-transplantation of bony defect may restrict orthodontic treat- cancellous iliac bone to the maxillary bone defect, ment and complicate final rehabilitation of basically using the same procedure as described by BoYNE & SANDS7• At first, bone grafting was the cleft area. For some years, our surgeons closed oro- delayed until the eruption of permanent teeth, based upon our experience from more than nasal fistulae with soft tissue only. However, but, 350 secondary bone graftings, the age of surgery our experience with this procedure showed has gradually been lowered. PEDERSEN & ENEtoo many failures. BOWERS & GRUBER 6 also MARK 19 indicated the optimal time for bone mentioned closure of an oro-nasal fistula as grafting as being between 10 and 12 years of age, being suprisingly difficult. AABYHOLM et al? before the canine on the cleft side has erupted . The treatment sequence for patients with uniand RINTALA21 also had to operate more lateral cleft lip and palate is commenced with a than once on every forth patient before transvers al maxillary expansion to correct the closure of a fistula was obtained. However, lateral crossbite and to provide the surgeon with more space for the bonegraft. many of their subjects were adult patients. In the bone grafting procedure, the cleft is The second indication for bone grafting is dissected free, and a nasal diaphragma is estaba deficienc y of bone in the alveolar process. lished. A " molded" piece of compact bone is Intra-oral examination may indicate a placed at the nasal floor of the cleft. The compact moderate defect, but an X-ray of the area bone is fastened by drilling a small retention will always reveal a defect larger than ex- furrow in the lateral bony parts of the cleft. The defect of the alveolar process is filled with canpected. Besides, the mid-line of the upper cellous bone, as is the palatal defect. In this jaw is very often displaced towards the non- manner, the high palatal vault is flattened, procleft side. Correction of the mid-line devia- viding more tissue for palatal soft tissue closure.
4
ENEMARK, KRANTZ-SIMONSEN AND SCHRAMM
Fig. 1. (a) Left-side complete unilateral cleft lip palate patient demonstrating a slit-like oro-nasal fistula. (b) The X-ray reveals the bone defect. (c) Before maxillary expansion, the fistula has been asymptomatic. After 3 months of orthodontic expansion, the fistula is easily seen.
It is important that facial soft tissue suturing is also performed without tension". If there is insufficient gingival tissue, it may be necessary to add a cheek flap. If a cheek flap is used, a secondary gingival plasty has to be expected. Following completion of suturing, the operated area is covered with Surgicelv and a dental plate, which is underlined with wound paste. The palatal appliance is wired to the teeth. One month post-operatively, the appliance is removed. The dental plate is meant to protect the wound and bone graft, as well as preventing formation of a hematoma. The appliance also retains the expanded maxilla. After disengagement from the teeth, the dental plate is used as a removable retainer for another month. After complete healing of the palate, the pre-operative maxillary appliance is re-inserted until permanent teeth are fully erupted and the orthodontic treatment can be completed.
Results Table 1 illustrates that 89% of the subjects
had an oro-nasal fistula. Most fistulae were slit-like deformities in the alveolar process, extending into the anterior part of the hard palate, where a fistula hole, 1-2 mm in largest diameter, occurred. There was no
Table 1. Frequency, size and symptoms of fistulae for 62 consecutive C-UCLP patients before transversal expansion of the maxilla
no fistula fistula 1-2 mm fistula with symptoms 28 asymptomatic fistulae 22 fistulae 3-5 mm fistula with symptoms 4 asymptomatic fistula I total
N
(%)
7
50
11 81
5
8
62
100
5
SECONDARY BONE GRAFTING IN CLEFTS
connection between size of fistula and the degree of patient discomfort. 52% complained of regurgitation of especially fluid food into the nasal cavity. Following orthodontic maxillary transversal expansion, an additional 37% of the subjects exhibited fistula symptoms. All fistulae were verified by probing at the time of surgery. When about half of the investigated group had been operated upon with a secondary bone grafting, J0RGENSEN & ENEMARK 1 6 in a preliminary investigation studied the influence of fistula on speech performance. It is of differential diagnostic importance to ensure the existance of a velopharyngel competence. Therefore patients with incompetence and flaps were excluded. 21 patients qualified for the study and were studied before the fistula closure and bone graft procedure. U sing a standardized speech test, including conversation speech samples, 13 patients exhibited nasal distortion on (s) sound. Subjects were re-evaluated approximately 6 months after the surgical procedure. The same speech test was carried out by the same speech pathologist. Only one subject was then noted to continue exhibition of nasal escape of air in the production of (s). In this study, 43% of the speech problems were related to the presence of the oro-nasal fistula (Table 2).
Table 2. Indications for secondary bone grafting in 62 complete UCLP patients
fistula patient discomfort speech disorders alveolar process defect orthodontic indication prosthodontic indication
N
(%)
32
26
52 43
31
50
6
10
Orthodontic indications for bone grafting were found in 50% of the patients. The orthodontic treatment was initiated once permanent incisors had erupted in malposition. The initial treatment was simply to correct inversions and forced bites. At the same time, lateral cross-bites were corrected. Uprightning of tipped incisors and correction of mid-line deviations were delayed until the gap in the alveolar process had been provided with bone. Bone grafting has until now been carried out in 56 of the 62 patients. 3 patients with a slit-like fistula did not want to be operated upon. In another 3 patients, a Le Fort I operation was planned, and in theese cases, the bone grafting has been delayed to after the osteotomy has been performed. The patients average age at the bonegrafting procedure in this sample was 12 years (Table 3). 23 patients were
Table 3. Time of operation in relation to canine eruption and need for later bridgework Orthodontical1y closed dental arch
Need for later bridgework
N
x=10 7 (86-11 3)
14
9
23
x=13 4
8
25
33
22
34
56
Age at operation operation before eruption of cleft-side canine operation after eruption of cleft-side canine total
(11°_15 1 1 )
x=12 1
(86-15 11 )
6
ENEMARK, KRANTZ-SIMONSEN AND SCHRAMM
operated on before and 33 patients after eruption of the cleft-side canine. In 40% of the patients, the orthodontic treatment was finished without the need for later prosthodontic restoration of the cleft area (Fig. 2). In t of these 40%, the bone grafting had been performed before canine eruption. In contrast, 72% of patients having surgery after canine eruption needed later prosthodontic treatment. The difference between the pre- and post-canine eruption group with respect to need for later prosthodontic bridgework was statistically sig-
Fig. 2. (a) C-UCLP patient ready for bone grafting after transversal maxillary expansion, and before eruption of the cleft-side canine. (b) Same patient 6 years post-operative after orthodontic treatment.
nificant (Table 3) at the 1% level of confidence (X 2=9 .41). In 10% ofthe investigated group, a large alveolar process defect was found. When the lack of alveolar bone is that extensive, it is technically very difficult to restore the alveolar process by prosthodontic means. With proper timing of the bone grafting , the newly formed bone seemed to be stabilized in its new function of tooth-bearing bone by eruption of teeth down through the graft (Fig. 3). As vertical growth of the upper jaw to a large extent is appositional", the cleft area was further stimulated by eruption of teeth. Following full eruption and orthodontic treatment, the teeth could unrestrictedly be situated with parallel crowns and appropriate diastemas ready for a later bridgework. Furthermore, the periodontal conditions were improved by the bone graft. The former irregular topography of the cleft area was levelled, whereby the possibility for proper oral hygiene was improved. Besides, an oro-nasal communication most often caused a gingival inflammation in the cleft area, due to secretion from the nasal cavity. After closure of the fistula, bridgework thereby obtained a better prognosis. Apart from insignificant losses of small pieces of bone chips, no grafts were lost. Complications were found in canine retention, external root resorptions and secondary resorptions of already healed bonegrafts. In 5 patients, operated upon before canine eruption, the path of eruption changed after the bone grafting and resulted in canine retention. The average age for these operations was 9.6 years - compared to the average age of 10.7 years for all patients operated upon before canine eruption. Therefore, surgery should not be performed too early in the mixed dentition. In 2 patients operated upon after full eruption of the cleft-side canine, external root resorption was discovered at the canine 3 to 4 years after surgery. The resorption was
SECONDARY BONE GRAFTING IN CLEFTS
7
Fig. 3. (a, b) C-UCLP patient with a large alveolar process defect. (c) After initial orthodontic
correction of incisor rotation and cross-bite. (d) The cleft-side canine has erupted down through the bone graft. (e, f) After orthodontic treatment ready for later bridgework.
situated at the facial surface at the cementoenamel junction. Both teeth were lost. This complication should be avoided by covering the canine at surgery by lyophilizied bones. In another 2 patients, a secondary resorption of bone was observed 1 to 2 years after surgery. In both patients, the oral surgeon had used a cheek flap, resulting in lack of attached gingiva at the facial surface of the
alveolar process. X-rays 6 months postoperatively showed a healed reorganized bonegraft, but 1 to 2 years later, a secondary partial bone resorption was observed under the atypical "cheek-flap gingiva".
Discussion As demonstrated in Table 2, most patients
8
ENEMARK, KRANTZ-SIMONSEN AND SCHRAMM
had more than one indication for a secondary bone grafting. 89% had a fistula which contributed to either discomfort or speech disorders. This frequency in complete unilateral cleft lip and palate patients appears higher than the 20 to 50% that has been previously reported in the literature 1 , 1 2 . 1 7 . The difference in occurence may be due to heterogeneity in the samples or differences in surgical procedures. Although the double vomerine flap procedure may leave a fistula, there appears to be less surgical interference in the pre-maxilla. ENEMARlC et aT. 1 3 demonstrated in the temporary dentition a large number of lateral cross-bites, but fewer anterior cross-bites than in comparable groups reported by PRUZANSKY & ADuss2 0 and BERGLAND 3 . Similar findings have been reported when DAHL et aT. 1 0 compared Danish and Norwegian groups. The relatively small number of anterior cross-bites in our patients may be interpreted as the surgical procedure creating less sagittal and vertical growth retardation in the premaxilla. In this series of patients, 52% complained of discomfort from the fistula before maxillary expansion. In 152 patients with fistulae, AABYHOLM et al? found 74% with symptoms requiring surgical intervention. Although STARK2 4 has reported that a fistula of less than 5 mm would contribute to neither regurgitation of food, nor to nasal speech, our findings are not in agreement. We found no essential relationship between the size of the fistula and the abovementioned symptoms, which is probably due to the fact that the anatomy of the area is highly irregular and that a depressed alar cartilage may reduce nasal escape of air. 43% of our patients had speech disorders which were related to a fistula, most often I to 2 mm in largest diameter. This finding is similar to VAN DEMARK 2 6 , who studied Danish patients, and found that those patients with fistulae exhibited more severe ratings of articulation. Thus it appears to us that for
children with a fistula contributing to eating or speech problems, either temporary obturation or surgery should be considered. Temporary obturation calls for good patient co-operation and good mouth hygiene. Besides, gingival inflammation is most often seen in the cleft area, due to secretion from the nasal cavity. In these cases, the temporary plate should be discharged well ahead of surgical closure. The optimal time for bone grafting and fistula closure appears to be between 10 and 12 years of age. This age is in agreement with BoYNE & SANDS 7 and DEEB et aT,u. The latter related surgical age to root formation of the canine. He found spontaneous eruption of the canine in 27% of his patients, and obtained the best results when land i of the canine root had developed, usually between 9 to 12 years of age. AABYHOLM et aT.2 recommended that bone grafting take place well ahead of canine eruption so that the canine could be guided down into the grafted area. They reported that in 80% of the patients, an almost normal alveolar process was achieved, and that most often the space in the alveolar process was closed by following orthodontic treatment, among other things, by using a Delaire mask. In our material using conventional intra-oral orthodontic appliances, we achieved a closed dental arch in 40% of our patients. From a technical point-of-view, a closed dental arch could have been achieved in a higher % of patients; however, aesthetically, facial appearance is often improved by sagittal expansion of the maxillary teeth, thereby opening up for prosthodontic bridgework in the cleft area. There are few complications mentioned in the literature concerning bone grafting, with the exception of canine retention and lost transplants. Both DEEB et aT. l l and AAByHOLM et al." indicated that the canine erupts down through the transplants without radiological changes in root or crown. In this
SECONDARY BONE GRAFTING IN CLEFTS
material, we found 2 patients with external root resorption on the facial surface of the canine at the cemento-enamal junction, Furthermore, we have observed 7 additional patients who have exhibited the same problem. All patients had surgery after canine eruption. When a more normal bony alveolar base is formed, there is better support for the lip and the depressed alar base on the cleft side. EpSTEIN et aI. 14 treated an aesthetic indication in 36% of their patients by reconditioning a depressed alar base in unilateral clefts. They also dealt with a functional indication in 40% of unilateral and bilateral cleft patients by unification of the maxilla. In bilateral clefts, there is no doubt that the premaxilla will be stabilized with bone grafting, but in our experience, a bonegraft will not prevent relapse of a transversally expanded maxilla. If a post-operative retention appliance is not used, patients will develop a cross-bite which is difficult to treat. We therefore favour a moderate approach in bonegrafting, since, if the procedure is performed just before canine eruption, the retention period will not be unduly long before complete orthodontic treatment can be commenced and finalized.
References 1. AABYHOLM, F. E., BORCHGREVINK, H. C. & ESKELAND, G.: Palatal fistulae following cleft palate surgery. Scand. J. Plast. Reconstr. Surg. 1979: 13: 295-300. 2. AABYHOLM, F. E., BERGLAND, O. & SEMB, G.: Secondary bone grafting of alveolar clefts. Scand. J. Plast. Reconstr. Surg, 1981: 15: 127140. 3. BERGLAND, 0.: Changes in cleft palate malocclusion after the introduction of improved surgery. Trans. Europ. Orthod. Soc. 1967: 43: 383-397. 4. BJORK, A & SKlELLER, V.: Growth in width of the maxilla studied by the implant method. Scand. Plast. Reconstr. Surg. 1974: 8: 26-33. 5. BJORK, A. & SKlELLER, V.: Postnatal growth and development of the maxillary complex.
9
Factors affecting the growth of the midface. McNamara, J. A, Jr. (ed.). Monograph 6, Craniofacial growth series, Center for Human growth and development. The University of Michigan, Ann Arbor 1976. 6. BOWERS, D. G. & GRUBER, H.: Use of acrylic obturators to protect suture lines in the hard palate. Plast. reconstr. Surg. 1973: 51: 98-101. 7. BoYNE, P. J. & SANDS, N. R.: Secondary bone grafting of residual alveolar and palatal clefts. J. Oral Surg. 1972: 30: 87-92. 8. BOYNE, P. J.: Personal communication, 1982. 9. BROUDE, D. 1. & WAITE, D. E.: Secondary closure of alveolar defects. OralSurgery 1974: 37: 829-840. 10. DAHL, E., HAMUSARDOTTffi, B. & BERGLAND, 0.: A comparison of occlusions in two groups of children whose clefts were repaired by three surgical procedures. Cleft Palate J. 1981: 18: 122-127. 11. DEEB, M., MESSER, L. B., LEHNERT, M. V., HEBDA, T. & WAITE, D. E.: Canine eruption into grafted bone in maxillary alveolar cleft defects. Cleft Palate J. 1982: 19: 9-16. 12. DRILLlEN, C. M., FUGRAM, T. T. S. & WILKINSON, E. M.: The causes and natural history of cleft lip and palate. E. and S. Livingstone, Edinburgh and London 1966, pp. 159-162. 13. ENEMARK, H., GREISEN, O. & J0RGENSEN, J.: The maxilla in cleft, lip and palate patients. 2nd Internal. Congress on Cleftpalate. Copenhagen 1973. 14. EpSTEIN, L. 1., DAVIS, W. B. & THOMPSON, L.: Delayed bone grafting in cleft palate patients. Plast. Reconstr. Surg. 1970: 46: 363-367. 15. FRIEDE, H. & JOHANSON, B.: A follow-up study of cleft children treated with primary bone grafting. Scand. J. Plast. Reconstr. Surg. 1974: 8: 1-16. 16. J0RGENSEN, J. & ENEMARK, H.: Signatismus nasalis in cleft lip and palate. Proceedings of 17th International Congress ofIALP, Copenhagen 1977, pp. 623-626. 17. LINDSAY, W. K.: Von Langenbeck paltorrhaphy. Cleft lip and palate 1971, Little, Brown and Company, Boston 1971, pp. 393---403. 18. NORDIN, K. E. & JOHANSSON, B.: Freie Knochen transplantation bei Defekten im Alveolarkamm usw. Fortsehr. Kief, - Ges. Chir. (Stuttgart) 1955: 1: 168-171. 19. PEDERSEN, S. & ENEMARK, H.: A comparative study of secondary and late secondary bonegrafting in patients with recidual cleft defects. Int. J. Oral Surg. 1985: 14: in press. 20. PRUZANSKY, S. & Anuss, H.: Arch form and the decidous occlusion in complete unilateral
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ENEMARK, KRANTZ-SIMONSEN AND SCHRAMM
clefts. Cleft Palate J. 1964: 1: 411-418. 21. RINTALA, A. E.: Surgical closure of palatal fistulae. Scand. J. Plast. Reconstr. Surg. 1980: 14: 235-238. 22. SCHMID, E.: Die Annaherung der Kieferstiimpfe bei Lippen-Kiefer-Gaumenspalten; ihre schadliche Folgen und Vermeidung. Fortschr. Kief. - Ges. - Chir. (Stuttgart) 1955: 1: 37-39. 23. SCHRUDDE, J. & STELLMACH, R.: Die primate Osteoplastik der Defekte des Kieferbogens bei Lippen-Kiefer-Gaumenspalten am Saugling. Zentralblatt far Chirurgie 1958: 14: 849-859. 24. STARK, R. B.: Reconstructive plastic surgery. CONVERSE, J. M. (ed.), W. B. Saunders, Philadelphia 1964, p. 1425.
25. STELLMACH, R.: Die funktionskieferorthopadische Behandlung der Kiefer-deformitaten bei Lippen-Kiefer-Gaumenspalten im Sauglingsalter. Fortsch. Kief. - Ges. - Chir. 1955: 16: 247-255. 26. VAN DEMARK, D. R.: Assessment ofarticulation for children with cleft palate. Cleft Palate J. 1974: 11: 200-208. Address: Hans Enemark Arhus Cleft Palate Clinic Taleinstituttet Finsensgade 12A 8000 Aarhus C Denmark
Secondary bonegrafting in unilateral cleft lip palate patients: indications and treatment procedure HANS ENEMARK, ERIK KRANTZ-SIMONSEN AND JAN ERIK SCHRAMM Aarhus Cleft Palate Clinic, Taleinstituttet, and Department of Oral Surgery, Kommunehospitalet, Aarhus, Denmark
A consecutive series of 62 complete unilateral cleft lip and palate patients were investigated with respect to indication for a secondary bone grafting. 2 major indications for secondary bone grafting were established. One was a symptomatic oro-nasal fistula, the other one being a bony defect in the alveolar process, which could impair orthodontic treatment and prosthodontic rehabilitation in the cleft area. The average age for bonegrafting was 12 years. After surgery, fistula-related discomfort was eliminated and speech disorders weredrastically reduced. Orthodontic uprightning of teeth and correction of mid-line deviations indicated in 50% of the patients were facilitated. In 10% of the cases with extensive defects in the alveolar process, the graft enhanced the possibility for later prosthodontic treatment. Secondarily, periodontal conditions improved. When bonegrafting was performed before canine eruption, total dental rehabilitation by orthodontic treatment was often sufficient, thus decreasing the need for later prosthodontic restoration of the cleft area. Patients who had surgery after eruption of the cleft side canine exhibited more complications. The optimal treatment sequence therefore appears to be transversal expansion of the maxilla in the late mixed dentition, followed by bone grafting. Maxillary expansion must be retained until final orthodontic and prosthodontic treatment is carried out. ABSTRACT -
(Received for publication 6 June 1983, accepted 19 February 1984)
The original purpose of bonegrafting was to stabilize the maxilla with a secondary operation 22 •2 5 and to provide bone inthe alveolar process of the maxilla, so that teeth adjacent to the cleft could erupt'", In 1958, SCHRUDDE & STELLMACH 2 3 recommended, bone grafting to be performed as a primary
procedure, and the short-term results appeared promising. However, later evaluation of these patients indicated maxillary growth retardation. FRIEDE & JOHANSSON 1 5 demonstrated almost normal development in the cleft area for 90% of the patients and tooth eruption into the graft. However, the
SECONDARY BONE GRAFTING IN CLEFTS
3
primary surgery interfered that much with tion is dependent upon bone in the cleft maxillary growth that they recommended area. discontinuance of primary bonegrafting. In theory, if growth of the upper jaw Material and methods should be completely free of surgical in- A consecutive series of 62 complete unilat eral fluence (apart from the inevitable primary cleft lip and palate patients representing all pasoft tissue repair), bone grafting would not tients born with tbis malformation in the western part of Denmark between 1964 and 1967 were be possible until the age of 18-20. During examined. All patients had their lip repaired at 2 the last 10 years different points-of-view months of age by one plastic surgeon, who used a have evolved concerning the optimal timing Tennison procedure. At the same time, the hard of the bone grafting procedure" 7.11. Nor- palate was repaired with double vomerine flaps. 22 mon ths of age, the soft palate was closed mal growth in the maxilla is sutural and At using a Wardill palatoplasty. With this type of appositional, and between the ages of 6 and surgical procedure, a residual oro-nasal fistula in 7 years, most transverse maxillary growth the anterio r pa rt of the palate often remained. has occurred". If bone grafting is delayed The fistula was always situated in the alveolar until after this age, a possible transverse process from the bottom of the alveolobuccal sulcus extending into the anterior part of the hard growth retardation can be compensated for palate (Fig. 1). This often slit-like deformity may by orthodontic treatment. not be easily observed, since the lateral segment of The two major indications for bone graft- the maxilla is usually collapsed before orthodoning are a symptomatic oro-nasal fistula and tic treatment is started. A diagnostic method to if a fistula is present, is to administrate a bony defect in the alveolar process impair- determine air from a dental syringe to the anterior part of ing orthodontic and prosthodontic treat- the palate. In cases with oro-nasal communicment. A fistula can cause patient discomfort ation, the patient wil\ feel a tickling in the nasal in regurgitation of food through it, and also cavity, always in the cleft side only. In 1972, we started secondary bone grafting on influences speech. The alveolar process pa tients with clefts using auto-transplantation of bony defect may restrict orthodontic treat- cancellous iliac bone to the maxillary bone defect, ment and complicate final rehabilitation of basically using the same procedure as described by BoYNE & SANDS7• At first, bone grafting was the cleft area. For some years, our surgeons closed oro- delayed until the eruption of permanent teeth, based upon our experience from more than nasal fistulae with soft tissue only. However, but, 350 secondary bone graftings, the age of surgery our experience with this procedure showed has gradually been lowered. PEDERSEN & ENEtoo many failures. BOWERS & GRUBER 6 also MARK 19 indicated the optimal time for bone mentioned closure of an oro-nasal fistula as grafting as being between 10 and 12 years of age, being suprisingly difficult. AABYHOLM et al? before the canine on the cleft side has erupted . The treatment sequence for patients with uniand RINTALA21 also had to operate more lateral cleft lip and palate is commenced with a than once on every forth patient before transvers al maxillary expansion to correct the closure of a fistula was obtained. However, lateral crossbite and to provide the surgeon with more space for the bonegraft. many of their subjects were adult patients. In the bone grafting procedure, the cleft is The second indication for bone grafting is dissected free, and a nasal diaphragma is estaba deficienc y of bone in the alveolar process. lished. A " molded" piece of compact bone is Intra-oral examination may indicate a placed at the nasal floor of the cleft. The compact moderate defect, but an X-ray of the area bone is fastened by drilling a small retention will always reveal a defect larger than ex- furrow in the lateral bony parts of the cleft. The defect of the alveolar process is filled with canpected. Besides, the mid-line of the upper cellous bone, as is the palatal defect. In this jaw is very often displaced towards the non- manner, the high palatal vault is flattened, procleft side. Correction of the mid-line devia- viding more tissue for palatal soft tissue closure.
4
ENEMARK, KRANTZ-SIMONSEN AND SCHRAMM
Fig. 1. (a) Left-side complete unilateral cleft lip palate patient demonstrating a slit-like oro-nasal fistula. (b) The X-ray reveals the bone defect. (c) Before maxillary expansion, the fistula has been asymptomatic. After 3 months of orthodontic expansion, the fistula is easily seen.
It is important that facial soft tissue suturing is also performed without tension". If there is insufficient gingival tissue, it may be necessary to add a cheek flap. If a cheek flap is used, a secondary gingival plasty has to be expected. Following completion of suturing, the operated area is covered with Surgicelv and a dental plate, which is underlined with wound paste. The palatal appliance is wired to the teeth. One month post-operatively, the appliance is removed. The dental plate is meant to protect the wound and bone graft, as well as preventing formation of a hematoma. The appliance also retains the expanded maxilla. After disengagement from the teeth, the dental plate is used as a removable retainer for another month. After complete healing of the palate, the pre-operative maxillary appliance is re-inserted until permanent teeth are fully erupted and the orthodontic treatment can be completed.
Results Table 1 illustrates that 89% of the subjects
had an oro-nasal fistula. Most fistulae were slit-like deformities in the alveolar process, extending into the anterior part of the hard palate, where a fistula hole, 1-2 mm in largest diameter, occurred. There was no
Table 1. Frequency, size and symptoms of fistulae for 62 consecutive C-UCLP patients before transversal expansion of the maxilla
no fistula fistula 1-2 mm fistula with symptoms 28 asymptomatic fistulae 22 fistulae 3-5 mm fistula with symptoms 4 asymptomatic fistula I total
N
(%)
7
50
11 81
5
8
62
100
5
SECONDARY BONE GRAFTING IN CLEFTS
connection between size of fistula and the degree of patient discomfort. 52% complained of regurgitation of especially fluid food into the nasal cavity. Following orthodontic maxillary transversal expansion, an additional 37% of the subjects exhibited fistula symptoms. All fistulae were verified by probing at the time of surgery. When about half of the investigated group had been operated upon with a secondary bone grafting, J0RGENSEN & ENEMARK 1 6 in a preliminary investigation studied the influence of fistula on speech performance. It is of differential diagnostic importance to ensure the existance of a velopharyngel competence. Therefore patients with incompetence and flaps were excluded. 21 patients qualified for the study and were studied before the fistula closure and bone graft procedure. U sing a standardized speech test, including conversation speech samples, 13 patients exhibited nasal distortion on (s) sound. Subjects were re-evaluated approximately 6 months after the surgical procedure. The same speech test was carried out by the same speech pathologist. Only one subject was then noted to continue exhibition of nasal escape of air in the production of (s). In this study, 43% of the speech problems were related to the presence of the oro-nasal fistula (Table 2).
Table 2. Indications for secondary bone grafting in 62 complete UCLP patients
fistula patient discomfort speech disorders alveolar process defect orthodontic indication prosthodontic indication
N
(%)
32
26
52 43
31
50
6
10
Orthodontic indications for bone grafting were found in 50% of the patients. The orthodontic treatment was initiated once permanent incisors had erupted in malposition. The initial treatment was simply to correct inversions and forced bites. At the same time, lateral cross-bites were corrected. Uprightning of tipped incisors and correction of mid-line deviations were delayed until the gap in the alveolar process had been provided with bone. Bone grafting has until now been carried out in 56 of the 62 patients. 3 patients with a slit-like fistula did not want to be operated upon. In another 3 patients, a Le Fort I operation was planned, and in theese cases, the bone grafting has been delayed to after the osteotomy has been performed. The patients average age at the bonegrafting procedure in this sample was 12 years (Table 3). 23 patients were
Table 3. Time of operation in relation to canine eruption and need for later bridgework Orthodontical1y closed dental arch
Need for later bridgework
N
x=10 7 (86-11 3)
14
9
23
x=13 4
8
25
33
22
34
56
Age at operation operation before eruption of cleft-side canine operation after eruption of cleft-side canine total
(11°_15 1 1 )
x=12 1
(86-15 11 )
6
ENEMARK, KRANTZ-SIMONSEN AND SCHRAMM
operated on before and 33 patients after eruption of the cleft-side canine. In 40% of the patients, the orthodontic treatment was finished without the need for later prosthodontic restoration of the cleft area (Fig. 2). In t of these 40%, the bone grafting had been performed before canine eruption. In contrast, 72% of patients having surgery after canine eruption needed later prosthodontic treatment. The difference between the pre- and post-canine eruption group with respect to need for later prosthodontic bridgework was statistically sig-
Fig. 2. (a) C-UCLP patient ready for bone grafting after transversal maxillary expansion, and before eruption of the cleft-side canine. (b) Same patient 6 years post-operative after orthodontic treatment.
nificant (Table 3) at the 1% level of confidence (X 2=9 .41). In 10% ofthe investigated group, a large alveolar process defect was found. When the lack of alveolar bone is that extensive, it is technically very difficult to restore the alveolar process by prosthodontic means. With proper timing of the bone grafting , the newly formed bone seemed to be stabilized in its new function of tooth-bearing bone by eruption of teeth down through the graft (Fig. 3). As vertical growth of the upper jaw to a large extent is appositional", the cleft area was further stimulated by eruption of teeth. Following full eruption and orthodontic treatment, the teeth could unrestrictedly be situated with parallel crowns and appropriate diastemas ready for a later bridgework. Furthermore, the periodontal conditions were improved by the bone graft. The former irregular topography of the cleft area was levelled, whereby the possibility for proper oral hygiene was improved. Besides, an oro-nasal communication most often caused a gingival inflammation in the cleft area, due to secretion from the nasal cavity. After closure of the fistula, bridgework thereby obtained a better prognosis. Apart from insignificant losses of small pieces of bone chips, no grafts were lost. Complications were found in canine retention, external root resorptions and secondary resorptions of already healed bonegrafts. In 5 patients, operated upon before canine eruption, the path of eruption changed after the bone grafting and resulted in canine retention. The average age for these operations was 9.6 years - compared to the average age of 10.7 years for all patients operated upon before canine eruption. Therefore, surgery should not be performed too early in the mixed dentition. In 2 patients operated upon after full eruption of the cleft-side canine, external root resorption was discovered at the canine 3 to 4 years after surgery. The resorption was
SECONDARY BONE GRAFTING IN CLEFTS
7
Fig. 3. (a, b) C-UCLP patient with a large alveolar process defect. (c) After initial orthodontic
correction of incisor rotation and cross-bite. (d) The cleft-side canine has erupted down through the bone graft. (e, f) After orthodontic treatment ready for later bridgework.
situated at the facial surface at the cementoenamel junction. Both teeth were lost. This complication should be avoided by covering the canine at surgery by lyophilizied bones. In another 2 patients, a secondary resorption of bone was observed 1 to 2 years after surgery. In both patients, the oral surgeon had used a cheek flap, resulting in lack of attached gingiva at the facial surface of the
alveolar process. X-rays 6 months postoperatively showed a healed reorganized bonegraft, but 1 to 2 years later, a secondary partial bone resorption was observed under the atypical "cheek-flap gingiva".
Discussion As demonstrated in Table 2, most patients
8
ENEMARK, KRANTZ-SIMONSEN AND SCHRAMM
had more than one indication for a secondary bone grafting. 89% had a fistula which contributed to either discomfort or speech disorders. This frequency in complete unilateral cleft lip and palate patients appears higher than the 20 to 50% that has been previously reported in the literature 1 , 1 2 . 1 7 . The difference in occurence may be due to heterogeneity in the samples or differences in surgical procedures. Although the double vomerine flap procedure may leave a fistula, there appears to be less surgical interference in the pre-maxilla. ENEMARlC et aT. 1 3 demonstrated in the temporary dentition a large number of lateral cross-bites, but fewer anterior cross-bites than in comparable groups reported by PRUZANSKY & ADuss2 0 and BERGLAND 3 . Similar findings have been reported when DAHL et aT. 1 0 compared Danish and Norwegian groups. The relatively small number of anterior cross-bites in our patients may be interpreted as the surgical procedure creating less sagittal and vertical growth retardation in the premaxilla. In this series of patients, 52% complained of discomfort from the fistula before maxillary expansion. In 152 patients with fistulae, AABYHOLM et al? found 74% with symptoms requiring surgical intervention. Although STARK2 4 has reported that a fistula of less than 5 mm would contribute to neither regurgitation of food, nor to nasal speech, our findings are not in agreement. We found no essential relationship between the size of the fistula and the abovementioned symptoms, which is probably due to the fact that the anatomy of the area is highly irregular and that a depressed alar cartilage may reduce nasal escape of air. 43% of our patients had speech disorders which were related to a fistula, most often I to 2 mm in largest diameter. This finding is similar to VAN DEMARK 2 6 , who studied Danish patients, and found that those patients with fistulae exhibited more severe ratings of articulation. Thus it appears to us that for
children with a fistula contributing to eating or speech problems, either temporary obturation or surgery should be considered. Temporary obturation calls for good patient co-operation and good mouth hygiene. Besides, gingival inflammation is most often seen in the cleft area, due to secretion from the nasal cavity. In these cases, the temporary plate should be discharged well ahead of surgical closure. The optimal time for bone grafting and fistula closure appears to be between 10 and 12 years of age. This age is in agreement with BoYNE & SANDS 7 and DEEB et aT,u. The latter related surgical age to root formation of the canine. He found spontaneous eruption of the canine in 27% of his patients, and obtained the best results when land i of the canine root had developed, usually between 9 to 12 years of age. AABYHOLM et aT.2 recommended that bone grafting take place well ahead of canine eruption so that the canine could be guided down into the grafted area. They reported that in 80% of the patients, an almost normal alveolar process was achieved, and that most often the space in the alveolar process was closed by following orthodontic treatment, among other things, by using a Delaire mask. In our material using conventional intra-oral orthodontic appliances, we achieved a closed dental arch in 40% of our patients. From a technical point-of-view, a closed dental arch could have been achieved in a higher % of patients; however, aesthetically, facial appearance is often improved by sagittal expansion of the maxillary teeth, thereby opening up for prosthodontic bridgework in the cleft area. There are few complications mentioned in the literature concerning bone grafting, with the exception of canine retention and lost transplants. Both DEEB et aT. l l and AAByHOLM et al." indicated that the canine erupts down through the transplants without radiological changes in root or crown. In this
SECONDARY BONE GRAFTING IN CLEFTS
material, we found 2 patients with external root resorption on the facial surface of the canine at the cemento-enamal junction, Furthermore, we have observed 7 additional patients who have exhibited the same problem. All patients had surgery after canine eruption. When a more normal bony alveolar base is formed, there is better support for the lip and the depressed alar base on the cleft side. EpSTEIN et aI. 14 treated an aesthetic indication in 36% of their patients by reconditioning a depressed alar base in unilateral clefts. They also dealt with a functional indication in 40% of unilateral and bilateral cleft patients by unification of the maxilla. In bilateral clefts, there is no doubt that the premaxilla will be stabilized with bone grafting, but in our experience, a bonegraft will not prevent relapse of a transversally expanded maxilla. If a post-operative retention appliance is not used, patients will develop a cross-bite which is difficult to treat. We therefore favour a moderate approach in bonegrafting, since, if the procedure is performed just before canine eruption, the retention period will not be unduly long before complete orthodontic treatment can be commenced and finalized.
References 1. AABYHOLM, F. E., BORCHGREVINK, H. C. & ESKELAND, G.: Palatal fistulae following cleft palate surgery. Scand. J. Plast. Reconstr. Surg. 1979: 13: 295-300. 2. AABYHOLM, F. E., BERGLAND, O. & SEMB, G.: Secondary bone grafting of alveolar clefts. Scand. J. Plast. Reconstr. Surg, 1981: 15: 127140. 3. BERGLAND, 0.: Changes in cleft palate malocclusion after the introduction of improved surgery. Trans. Europ. Orthod. Soc. 1967: 43: 383-397. 4. BJORK, A & SKlELLER, V.: Growth in width of the maxilla studied by the implant method. Scand. Plast. Reconstr. Surg. 1974: 8: 26-33. 5. BJORK, A. & SKlELLER, V.: Postnatal growth and development of the maxillary complex.
9
Factors affecting the growth of the midface. McNamara, J. A, Jr. (ed.). Monograph 6, Craniofacial growth series, Center for Human growth and development. The University of Michigan, Ann Arbor 1976. 6. BOWERS, D. G. & GRUBER, H.: Use of acrylic obturators to protect suture lines in the hard palate. Plast. reconstr. Surg. 1973: 51: 98-101. 7. BoYNE, P. J. & SANDS, N. R.: Secondary bone grafting of residual alveolar and palatal clefts. J. Oral Surg. 1972: 30: 87-92. 8. BOYNE, P. J.: Personal communication, 1982. 9. BROUDE, D. 1. & WAITE, D. E.: Secondary closure of alveolar defects. OralSurgery 1974: 37: 829-840. 10. DAHL, E., HAMUSARDOTTffi, B. & BERGLAND, 0.: A comparison of occlusions in two groups of children whose clefts were repaired by three surgical procedures. Cleft Palate J. 1981: 18: 122-127. 11. DEEB, M., MESSER, L. B., LEHNERT, M. V., HEBDA, T. & WAITE, D. E.: Canine eruption into grafted bone in maxillary alveolar cleft defects. Cleft Palate J. 1982: 19: 9-16. 12. DRILLlEN, C. M., FUGRAM, T. T. S. & WILKINSON, E. M.: The causes and natural history of cleft lip and palate. E. and S. Livingstone, Edinburgh and London 1966, pp. 159-162. 13. ENEMARK, H., GREISEN, O. & J0RGENSEN, J.: The maxilla in cleft, lip and palate patients. 2nd Internal. Congress on Cleftpalate. Copenhagen 1973. 14. EpSTEIN, L. 1., DAVIS, W. B. & THOMPSON, L.: Delayed bone grafting in cleft palate patients. Plast. Reconstr. Surg. 1970: 46: 363-367. 15. FRIEDE, H. & JOHANSON, B.: A follow-up study of cleft children treated with primary bone grafting. Scand. J. Plast. Reconstr. Surg. 1974: 8: 1-16. 16. J0RGENSEN, J. & ENEMARK, H.: Signatismus nasalis in cleft lip and palate. Proceedings of 17th International Congress ofIALP, Copenhagen 1977, pp. 623-626. 17. LINDSAY, W. K.: Von Langenbeck paltorrhaphy. Cleft lip and palate 1971, Little, Brown and Company, Boston 1971, pp. 393---403. 18. NORDIN, K. E. & JOHANSSON, B.: Freie Knochen transplantation bei Defekten im Alveolarkamm usw. Fortsehr. Kief, - Ges. Chir. (Stuttgart) 1955: 1: 168-171. 19. PEDERSEN, S. & ENEMARK, H.: A comparative study of secondary and late secondary bonegrafting in patients with recidual cleft defects. Int. J. Oral Surg. 1985: 14: in press. 20. PRUZANSKY, S. & Anuss, H.: Arch form and the decidous occlusion in complete unilateral
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ENEMARK, KRANTZ-SIMONSEN AND SCHRAMM
clefts. Cleft Palate J. 1964: 1: 411-418. 21. RINTALA, A. E.: Surgical closure of palatal fistulae. Scand. J. Plast. Reconstr. Surg. 1980: 14: 235-238. 22. SCHMID, E.: Die Annaherung der Kieferstiimpfe bei Lippen-Kiefer-Gaumenspalten; ihre schadliche Folgen und Vermeidung. Fortschr. Kief. - Ges. - Chir. (Stuttgart) 1955: 1: 37-39. 23. SCHRUDDE, J. & STELLMACH, R.: Die primate Osteoplastik der Defekte des Kieferbogens bei Lippen-Kiefer-Gaumenspalten am Saugling. Zentralblatt far Chirurgie 1958: 14: 849-859. 24. STARK, R. B.: Reconstructive plastic surgery. CONVERSE, J. M. (ed.), W. B. Saunders, Philadelphia 1964, p. 1425.
25. STELLMACH, R.: Die funktionskieferorthopadische Behandlung der Kiefer-deformitaten bei Lippen-Kiefer-Gaumenspalten im Sauglingsalter. Fortsch. Kief. - Ges. - Chir. 1955: 16: 247-255. 26. VAN DEMARK, D. R.: Assessment ofarticulation for children with cleft palate. Cleft Palate J. 1974: 11: 200-208. Address: Hans Enemark Arhus Cleft Palate Clinic Taleinstituttet Finsensgade 12A 8000 Aarhus C Denmark