Treatment Variables Affecting Facial Growth in. Unilateral Cleft Lip and Palate. Part 3: Alveolus Repair and

Treatment Variables Affecting Facial Growth in Complete Unilateral Cleft Lip and Palate. Part 3: Alveolus Repair and Bone Grafting Facial growth in a sample of 439 males with unilateral complete cleft lip and palate was evaluated. There were 213 subjects with some form of alveolus repair and 226 subjects with no alveolus repair. The results indicated that surgical repair of the alveolus in unilateral cleft lip and palate resulted in a deficiency in the vertical growth of the anterior maxilla. If, in addition, a bone graft or periosteoplasty was used, the vertical growth deficiency was slightly worse and lower face height was greater, so that facial vertical proportions were poor. There was also a slight anteroposterior maxillary deficiency following bone grafting. Late bone grafts in the mixed dentition have the same vertical effect as theinfant bone grafts. These findings should be applied to individuals at risk for poor facial growth. Infants born with unilateral cleft lip and palate have certain urgent requirements to enable them to achieve reasonably normal function and facial esthetics. There must be surgical reconstruction of the lip and the soft palate, and there is great advantage to repairing the hard palate. Truly successful lip and palate surgery was, for a long time, a difficult undertaking, and these areas received the major attention. As the results of surgery improved and orthodontists and speech pathologists became more directly involved in cleft habilitation, dental arch form and the cleft of the alveolus assumed more importance. Many surgeons repaired the alveolus with soft tissue flaps of various kinds while repairing the lip. This closed off the anterior oral cavity from the nasal, and subsequent hard and soft palate repair completed the reconstruction. There were some sporadic attempts at using bone in the repairs. Koberg (1973) and Witsenburg (1985) have published excellent reviews. Presurgical orthopaedics and infant alveolar bone grafting appeared on the scene at about the same time. McNeil (1950) with presurgical orthopaedics and Schmid (1955) and Nordin and Johanson (1955) with bone grafting were the early proponents of arch manipulation followed by fusion of the segments in a much more normal configuration than was present at birth. They reasoned that collapse of the maxilla would be prevented, and improved growth and dental occlusion would follow. Different kinds of bone in different configurations have been used to partially or completely fill the alveolus, the basal bone to the piriform fossa, and even part of the hard palate. Skoog in Uppsala (Hellquist and 33 Ponten, 1979) developed a periosteoplasty procedure that induced bone to bridge the alveolar gap. The results were variable. Friede and Johanson (1982) reported well documented studies. which found that patients who had received their type of alveolar bone grafts in infancy developed a severe maxillary retrognathia and vertical deficiency that worsened with age, and they soon discontinued the procedure. They later noted that 50 percent of the cases required orthognathic surgery to achieve adequate occlusion and facial appearance. Robertson and Jolleys (1983) had a similar experience, noting in addition that the grafts deteriorated to a small strut of bone, with no evidence of tooth migration into it. Rosenstein et al (1982) and Nordin et al (1983) continued to use primary bone grafts and were not dissatisfied with the maxillary growth that resulted. The Nordin study indicated that a "*Ttraction'' orthopaedic device improved the results considerably. Ross (1970) pointed out that the alveolus and area of the maxilla in which the bone graft is placed is not a site of maxillary growth (Enlow, 1968), so that anteroposterior maxillary development should not be affected if grafts are confined to the alveolar area. Grafting could prevent medial collapse of the bony segments, but would have no effect on the dentoalveolar medial collapse caused by residual scar tissue from the palate surgery. The additional scar tissue from the bone graft, however, could interfere with vertical development of the adjacent alveolar process and alter the eruptive direction of the teeth. Friede and Johanson (1982) felt that the poor development they observed was caused by

34 Cleft Palate Journal, January 1987, Vol. 24 No. 1 interference with the maxillary-vomerine suture. Dahl et al (1981) found that some techniques of vomeroplasty caused an increased incidence of crossbite in the primary dentition. They concluded that variationin the lip repair technique used may also have influenced the results, but far less than the vomeroplasty procedures. The purpose of this study was to analyze the facial growth of children who had the cleft alveolus and anterior maxilla repaired by different surgical techniques, in order to determine whether facial growth would be enhanced or inhibited by these procedures. SAMPLE The sample was drawn from the 538 male subjects with complete unilateral cleft lip and palate described in detail in the initial paper of this series. ' Many centers repaired the cleft alveolus using a variety of flap designs, and "borrowing" mucosa from areas of the cheek, alveolar ridge, vomer, and anterior palate. It was difficult and arbitrary to classify these techniques into precise subgroups, and the numbers became too few to be trustworthy. Four groups of patients, however, were identified within the sample: Group 1: alveolus repair with a bone graft in infancy; Group 2: alveolus repair with a bone graft in childhood; Group 3: alveolus repair by soft tissue; almost always with a vomer flap at some stage; and Group 4: no alveolus repair. The sample from Marburg and the Unoperated cases were deleted to avoid introducing irrelevant growth factors. A total sample of 213 cases with some form of repair was available to compare with 226 cases having had no alveolus repair. The latter group consisted of Toronto Base (136 subjects), Lindsay (56 subjects), Manchester (20 subjects) and Toronto Orthopaedic (14 subjects) cases. Alveolar Bone Grafts in Infancy There were three Centers with cases of primary bone grafting to the alveolar cleft: Goteborg (21 cases); Stockholm (23 cases), and Chicago (16 cases). A fourth Center, Uppsala (23 cases), used a technique of periosteoplasty that resulted in bony union between the maxillary segments. Two approaches to the periosteoplasty technique were available from Uppsala, but were found to give essentially the same results and were pooled. Only successful cases were submitted for analysis, that is, cases where a ridge of bone was present across the cleft. A total of 83 cases was available for analysis. Alveolar Bone Grafting in Childhood There were many cases that received an alveolar bone graft in childhood as a secondary procedure. The entire Oslo sample and some of the Auckland, Perth, and Malmo samples had received this treatment. Also, many cases from these and other Centers were bone grafted in the permanent dentition, presumably after final orthodontic treatment. Growth effects were studied using a group where the graft was inserted prior to 11.0 years of age and adequate follow-up radiographs were available at least 3 years after the bone graft had been placed. The sample consisted of 28 cases (16 from Oslo, 10 from Auckland, and 2 from Malmo) with a mean age of 8.4 years at the time of grafting. The 10 Auckland cases had been grafted between 4 years, 4 months and 7 years, 6 months of age, with a mean of 5.8 years. The 16 Oslo cases had been grafted between 9 years and 10 years 11 months of age, with a mean of 10.0 years. The two Malmo cases were grafted at 8 years and 10 years of age. A group bone grafted between 11 and 13 years of age was too small to be reliable (16 cases), and growth effects at these ages would be difficult to determine. The small group bone grafted after 13 years of age was not classified as having been bone grafted, but appeared either in the soft tissue alveolus repair or the nonrepaired groups depending on their earlier management. Almost all of these subjects were grafted in the late teenage years. Soft-Tissue Alveolus Repair A sample of 84 cases in which the alveolus and often some of the anterior hard palate were repaired by means of Burian, Muir, labial, alveolar, or vomer flaps were available. There were 35 cases from Copenhagen, 24 from Auckland, 16 from Zurich, and 9 from Perth. All but a few had a vomer flap as part of the repair. METHOD A complete description of the method was provided in the initial article of this series. Early Bone Grafting FINDINGS Measurements on the four samples of early bone grafting are presented in Table 1. As previously noted, Goteborg had a particularly extensive bone grafting procedure that resulted in

Ross, ALvEOLUS REPAIR AND BONE GRAFTING 35 TABLE 1 Measurements of 83 Cases That Had Received Alveolar Bone Grafts in Infancy from Uppsala, Chicago, Stockholm, and Goteborg with a Normal Sample Provided for Comparison Data Uppsala Chicago Stockholm Subtotal Goteborg Normal Sample Size 23. 16 19 62 21 54 Mean Age (yr) 11.4 10.8 10.9 10.6 10.9 10.8 Cranial Base Angle (N-S-Ba) 47.7 46.7 49.0 47.8 49.4 51.4 Proportion (N-S/N-S-Ba) 61.3 61.7 61.9 61.7 60.6 61.1 Length (N-Ba) 104.4 108.1 107.3 105.8 100.4 107.6 Area (cm2)* 10.8 10.9 11.4 10.9 10.9 12.2 Adjusted Linear Measurements Pharynx (Ba-PMP)* 40.8 39.7 40.7 40.5 39.8 41.6 Maxilla (PMP-ANS)* 48.6 49.3 49.2 48.9 46.2 52.5 Maxilla (PMP-A Pt)* 43.9 44.8 44.4 44.4 40.3 47.2 Ba-ANS* 89.4 89.0 89.9 89.6 86.0 94.1 Ba-A Pt* 84.3 84.2 85.0 84.7 79.8 88.7 Ba-UI* 88.6 88.0 88.6 88.3 83.8 95.7 Ba-LI* 86.7 85.6 86.4 86.3 83.8 91.5 Overjet 1.9 2.4 2.2 2.0 0.0 4.2 N-ANS* 45.1 46.3 43.8 44.8 43.1 48.3 Vertical proportion 41.1 43.1 40.5 41.4 39.6 45.0 ANS-UI/N-Men 25.8 25.3 26.6 25.9 26.1 25.5 Men-LI/N-Men 36.1 35.6 36.1 36.0 370 35.4 Maxilla area (cm2)* 19.7 21.2 20.9 20.3 18.0 23.2 Mandible (Co-Gn)* 104.5 101.3 103.8 102.8 102.7 104.6 Angles Ba-N-ANS 63.4 62.8 64.0 63.5 58.6 69.0 Ba-N-A Pt 57.4 57.2 58.2 57.8 52.3 62.2 Ba-N-UI 59.2 58.6 59.3 59.0 55.6 64.5 Ba-N-LI 58.0 57.0 57.9 57.7 55.9 62.0 Ba-N-B Pt 56.0 54.7 56.3 55.6 54.8 58.2 Ba-N-Pog 56.9 55.4 57.2 56.4 56.2 58.6 N-UI-UI apex 15.8 16.2 16.3 15.6 20.6 17.5 SNA 75.5 74.8 76.2 75.7 71.4 81.6 SNB 74.1 72.3 74.6 73.6 73.9 77.8 ANB 1.4 2.6 1.6 2.1-2.5 4.0 Mandibular plane 57.0 55.3 54.7 56.1 55.4 57.8 Nasal bone angle 101.0 99.5 103.7 101.2 104.2 110.0 + Toronto Standards Maxilla (Ba-N-ANS) -O.7-1.5-0.2-0.8-5.6 4.7 Mandible (Ba-N-Pog) 0.3-1.1 0.7-0.2-0.3 2.1 AP Jaw (ANS-N-Pog) -0.9-0.4-0.9-0.6-35.3 2.6 AP Jaw (ANB) -0.9 -O.1-1.0-0O.7-35.1 1.4 Maxilla height* -2.3-0.9-3.4-2.2-4.1 1.2 Lower face height 1.4 0.2 1.6 1.4 2.9-4.0 * Raw linear measurements were adjusted by dividing by the cranial base length (N-Ba) and multiplying by 100. extreme growth attenuation of the maxilla. The other three samples (Chicago, Stockholm, and Uppsala) showed less growth interference (Fig. 1, Table 1). Measurements presented in parenthesis with +TS indicate the deviation (plus or minus) from the Toronto Standards. Vertical Relations. Anterior maxillary height (N-ANS) for the Chicago sample (-0.9 mm TS) was greatest followed by Uppsala (-2.1 TS), while Stockholm and Goteborg samples had the least anterior maxillary height (-2.9, -4.1 TS). All of the samples had measurements that were markedly below the Toronto Standards. Posterior maxillary height (R-PMP) was greatest in the Stockholm sample. For lower face height, the Chicago sample (0.2 TS) was equal to the Toronto Standard and less than the Uppsala (1.4 TS), Stockholm (1.7 TS), and Goteborg (2.9 TS) samples. The greater wer values indicated excessively long lower faces for the size of the mandibles. Facial vertical proportions were thus very good for the Chicago sample, with midface comprising 43.1 percent of the facial height. The normal sample midface was 45.0 percent of the facial height at this age. The Uppsala and Stockholm samples had quite poor facial vertical proportions at 41.1 percent and 40.7 percent, and the Goteborg sample had the most severe proportions at only 39.6 percent. Anteroposterior Relations. Differences in maxillary length (PMP-A Pt) other than the marked reduction in the Goteborg sample, were negligible. The Chicago sample had a slightly longer maxillary length, but it was posteriorly positioned (PMP-Ba less), as illustrated in Figure 1. Maxillary protrusion (Ba-N-ANS) showed the Stockholm sample to be the most favorable at

36 Cleft Palate Journal, January 1987, Vol. 24 No. 1 UCLP Alveolus Repair Infant Bone Grafts Uppsala Sample 23 males 11.4 yr Chicago Sample ------ 16 males 10.8 yr _c Stockholm Sample -- --- we" 19 males 10.9 yr see" f was ]."tn. / di 2; \! I NJ FIGURE 1 Three moderate infant bone graft samples are superimposed on the cranial base (N-Ba) and scaled to the same size. - only 0.2 degrees below the Toronto Standard, with the Uppsala sample -O0.7 degrees, Chicago sample -1.5 degrees, and the Goteborg sample a severe -5.6 degrees. The alveolus to basal bone measurement (ANS-N-A Pt), which indicates the amount of alveolar process retrusion on the basal bone, showed no difference in any sample. Mandibular protrusion and size were least in the Chicago sample, (1.4 degrees and 3.2 mm less than the Uppsala sample, 1.8 degrees and 2.5 mm less than the Stockholm sample, and 0.8 degrees and 1.4 mm less than the Goteborg sample). There were no differences, except for the Goteborg sample, in overall anteroposterior jaw relations, although the Chicago sample was very slightly more favorable than the Stockholm and Uppsala samples. The maxillary and mandibular incisors (ANS-N-UI, ANS-N-LI) were in identical positions in all samples. The overjet was identical for three of the samples (1.9 mm in the Uppsala sample, 2.4 mm in the Chicago sample, 2.2 mm in the Stockholm sample,) but Goteborg had a 0.0 mm overjet. The total maxillary area was identical for the Chicago and Stockholm samples (21.2 and 20.9 cm), less in the Uppsala sample (19.7 cm?) and much reduced in the Goteborg sample (18.0 cm). The cranial base proportions and area were very close in all four samples, although registration point and orbitale were forward in the Uppsala sample (Fig. 1). One interesting observation was the identical position of the posterior soft palate. {s In summary, then, there were minor disparities between three of the samples, with the fourth, (Goteborg) showing severe growth effects. It should be noted that the Chicago group had better overall facial proportions than the other three, with none of the key measurement differences attaining statistical significance from the sample with unrepaired alveolus. Excluding Goteborg and combining the three ''moderates'', (Table 1) provided evidence that at age 10.6 years there was a modest maxillary retrusion and less desirable jaw relations compared to the Toronto Standards. More serious was a marked decrease in anterior maxillary vertical height (-2.2 mm TS) and an increase in lower face height, so that the overall vertical proportions were poor. The maxilla comprised only 41.4 percent of the facial height compared to the normal 45 percent and the 43 percent earlier noted in the better cleft samples. This group of 62 ''moderate'' infant bone graft cases, excluding the Goteborg cases, will be used for comparisons with other methods of managing the cleft alveolus. Growth patterns for the four subsamples are shown in Figure 2 and Table 2. In general, there was less maxillary retrusion with age, so that the values reached thetoronto Standards, while jaw relations remained approximately the same. An- - terior maxillary vertical height remained relatively the same in the Uppsala and Chicago samples, but worsened in the Stockholm and Goteborg groups. Lower face height showed little change, except in the Goteborg group. The vertical proportions remained the same in the three moderate groups at 41.4 percent and worsened in the Goteborg sample to 38.7 percent. The growth patterns as visualized in Figure 2 were virtually identical, with the degree of maxillary protrusion usually the only difference. The Uppsala and Stockholm samples maintained maxillary position particularly well. The Goteborg sample benefitted from a vertical mandibular growth direction, but the Stockholm sample had a horizontal pattern. The Uppsala and Chicago groups showed moderately vertical mandibular growth. It is interesting to compare Figure 2 with the Normal and the Copenhagen samples' growth patterns from the initial paper in this series (Fig. 16A and B, Part 1). In the Normal pattern the maxillary incisor apex moved down and forward, while in all cleft samples it moved down and back. The mandibular plane closed slightly in Normals, but not in any of the cleft samples. It should also be noted that growth conformed

Ross, ALVEOLUS REPAIR AnD BONE GRAFTING 37 UCLP Alveolus Repair Chicago Sample (infant bone graft) - 15 males 10.2 yr 14.7 yt ----- UCLP Alveolus Repair Goteborg Sample (infant bone graft) - 21 males 10.9 yr 16.0 yr ----- UCLP Alveolus Repair Stockholm Sample (infant bone graft) - 23 males 10.1 yr \ UCLP Alveolus Repair 14.4 yr --- -- Uppsala Sample (infant periosteoplasty) - 23 males 11.4 yr 14.7 yr --- -- UCLP Alveolus Repair Infant Bone Graft - 61 males (Chicago, Stockholm, and Uppsala samples) \ 10.6 yr ----- 14.6 yr --- -- FIGURE 2 Growth patterns of the infant bone graft samples are superimposed on the cranial base (N-Ba) and scaled to the same size. to the Toronto Standards during this period, with no indication of worsening. If anything, maxillary anteroposterior development improved slightly. Whatever the growth effects related to the infant bone grafting were, they acted early and thereafter remained stable. Alveolar Bone Grafting at Age 4 to 10 Years The measurements (Table 3) and plots (Fig. 3) for the 28 cases that were bone grafted in childhood are presented as changes from 10 to 15 years of age. There was a mild improvement in the ANB angle due to the marked vertical growth

38 Cleft Palate Journal, January 1987, Vol. 24 No. 1 TABLE 2 Facial Growth Patterns Following Infant Bone Grafting - Uppsala Sample Chicago Sample Stockholm Sample Goteborg Sample Subtotalt (N=23) (N=15) (N=23) (N=21) (N=62) Age (yrs) Age (yr8) Age (yrs) Age (yrs) Age (yrs) Measurements 11.4 14.7 Change 10.2 14.7 Change 10.1 14.4 Change 10.9 160 Change 10.6 14.5 Change '+ Toronto Standard Maxilla (Ba-N-ANS) -0.7 -O.1 0.6-1.16-1l1 0.5-02 0.5 07-5.6-57 -O.1-0.8 -O.1 0.7 Mandible (Ba-N-Pog) 0.3 06 03 _-I1.1 -O0.5 06 02 08 06-0.3-1.1-0.8-0.2 0.4 0.6 AP Jaw (ANS-N-Pog) -1.0-08 0.2-05 -07-02 -04-04 0.0-5.3-5.1 02-06 -0.5 0.1 AP Jaw (ANB) -0.9 -O07 02-0.3-0.2 0.1-0.5-0.4 O.I1-5.1-4.2-0.9 -O07-04 0.3 Maxilla height* -2.3-2.3 0.0-0.5-0.3 Q2-2.8-37 -0.9-4.1-5.3-1.2-2.2-2.3 -O.1 Lower face height 1.4 18 0.4 0.4 00-0.4 2.3 2.1-0.2 2.9 4.5 1.6 1.4 1.4 00 Vertical proportion** 41.1 40.8-0.3 43.0 4310 0.0 40.6 40.2-0.4 39:6 38.7 O:9 41.4 41.2 -O.2 * Adjusted to cranial base length ** Toronto Standards not available { Subtotal excludes the Goteborg sample. TABLE 3 Standards Alveolar Bone Grafting in Childhood: Difference from Youngest to Oldest Records, + Toronto Age AP Jaws (degrees) Maxilla Lower Face Vertical Range Maxilla Height Height Proportion Sample Number (yr) (degrees) Basal ANB (mm) Height (mm) (%) Graft at 4 to 10 yr 28 10.1-15.4 0.0 0.4 0.7-0.8 2.1-1.1 Auckland, graft at 4 to 7 yr 10 11.9-16.7 -O.1-0.3 0.5-0.6 1.0-1.3 Auckland, no bone graft 24 11.1-13.4 1.0 0.4 0.6 0.8 0.5 0.2 Oslo, graft at 9 to 10 yr 16 9.7-14.4 e 0.9 0.6-0.8 2.8-1.0 Oslo, graft at 9 to 12 yr 25 9.7-15.0 0.3 0.8 0.6-1.2 2.2-0.9 Copenhagen, late bone graft 13 9.9-18.2-0.5-0.6 0.4 -O.7 0.4-0.8 Copenhagen, no bone graft 22 9.9-18.1 0.3 0.5 1.2 -O0.7 0.7-0.6 of the mandible. There was a relative decrease. in the anterior maxillary height and a very large increase over the Toronto Standard in lower face \\ height. Thus, the vertical proportions of the face UCLP Alveolus Repair \<\\ worsened as the maxillary contribution decreased FIGURE 3 Growth pattern of the 4- to 10-year-old bone grafted sample. Plots of Young and Old groups are superimposed on the cranial base (N-Ba) and scaled to the same size. Bone Graft (4-10 years) - 28 males \j\ to an. unsatleactory 41 6 perce t N This sample consisted in part of a group of 10 1g; a I \\ Auckland cases grafted between 4 and 8 years of age. They were compared with the 24 nongrafted cases from Auckland who were otherwise identically managed. There were differences at age 11 (Fig. 4), and subsequent growth patterns were worse in the grafted cases. The maxilla became more retrusive by 1.1 degrees in the grafted cases; maxillary height worsened by 1.4 mm, and the overall vertical proportions worsened by 1.5 percent. These were differences that occured after the age of 11 years, many years after the bone grafting. Another part of the sample consisted of 16 Oslo children who had alveolar bone grafts between 9.0 and 10.9 years of age. Maxillary anteroposterior development was not favorable at the time the grafts were placed, but they showed mild improvement in anteroposterior jaw relations at age 14. This was not because of improved maxillary growth, but rather because of a vertical mandibular growth pattern (Table 3 and

Ross, ALvEOLUS REPAIR AnD BONE GRAFTING 39 Auckland Sample _ \ No bone graft group --------- \ 24 males 11.1 yr \ Bone graft group -- -- -- -- 10 males 11.9 yr Figure 7. Both anteroposterior and vertical data were virtually identical to the Toronto Standard at both ages. The vertical facial proportions showed a 42.9 degree maxilla contribution. Comparison of All Methods FIGURE 4 Plots of 10 Auckland cases bone grafted at 4 to 8 years of age and 24 Auckland cases not bone grafted. Fig. 5), which also increased the lower face height by 2.8 mm more than the Toronto Standard. The maxillary vertical height became more deficient, and therefore the maxillary contribution dropped by 1.0 percent. The Oslo sample of 25 children were all bone grafted by 12 years of age, and comparing the total to the 16 children grafted under 11 years of age showed that there was virtually no difference in the growth changes between them (Table 3). Finally, there were 13 individuals from Copenhagen who received late bone grafts in adolescence, and they were compared with the 22 from Copenhagen who did not receive bone grafts. Anteroposterior jaw relations were worse in the nongraft cases at 9.9 years of age, but by 18 years the values were identical. The vertical relations were identical throughout the period studied. Infant Versus Childhood (4 to 10 years) Bone Graft Groups. The differences noted in Table 5 and Figure 8 were so slight that these two groups were pooled. Anterior vertical height was slightly decreased in the plot of the infant bone graft cases, although the age difference may account for this, since in comparison with the Toronto Standard (Table 5) the anterior maxillary height deficiencies were just over 2 mm in each group. A slightly increased posterior maxillary height was noted in the infant graft group. All Bone Grafting Groups Versus Soft Tissue Repair Group. Tables 5 and 6 and Figure 9 provide a comparison of these two groups. The soft tissue repair group had more favorable maxillary protrusion (p <0.02) by 1.3 degrees and slightly better jaw relations by 0.9 degrees both in ANS-N-Pog (not significant) and ANB (p <0.04) angles. Maxillary vertical growth was slightly better by 0.7 mm (not significant) and lower face height was reduced by 1.9 mm (p <0.02) so that better vertical facial proportions resulted (a 42.2 percent maxillary contribution compared to 41.3 percent, p <0.002). Again, there was an increased posterior maxillary height in the bone grafting sample (Fig. 9). \ \ \\ UCLP Alveolus Repair ) \ A Oslo sample (late bone graft) - 25 males 9.7 yr --------- 15.0 yr -- --- -- " /* Soft Tissue Alveolus Repair The findings for the soft tissue repair of the alveolus are presented in Table 4 and Figure 6. Maxillary height was deficient by 1.1 mm at age 9.9 years and became slightly worse by 14.6 years. Lower face height remained proportional, and the overall vertical proportions remained the same with 42.2 percent maxillary contribution to the total. Maxillary protrusion was favorable and improved slightly, and the anteroposterior jaw relations were equal to the Toronto Standard. No Alveolus Repair The measurements and plot for the sample with no alveolus repair are presented in Table 4 and FIGURE 5 Growth pattern of the Oslo late bone graft sample. Plots of Young and Old groups are superimposed on the cranial base (N-Ba) and scaled to the same size.

_- 40 Cleft Palate Journal, January 1987, Vol. 24 No. 1 TABLE 4 Soft Tissue Alveolus Repair and No Alveolus Repair in Young and Older Group Soft Tissue Repair vs Soft Tissue Repair No Alveolus Repair No Repair Data Young Old Difference Young Old Difference Difference Sample Size 84 84 206 198 84:198 Mean Age (yr) 9.9 14.6 4.7 9.0 14.8 5.8 14.6:14.8 Cranial Base Angle (N-S-Ba) 47.7 48.1 0.4 47.5 48.6 1.1-0.5 Proportion (N-S-Ba/N-S) 61.9 61.2-0.7 61.9 60.7-1,.2 0.5 Length (N-Ba) 104.3 110.1 5.8 103.2 112.0 8.8-1.9 Area (cm) Adjusted Linear Measurements Pharynx (Ba-PMP)* 40.1 39.5-0.6 40.1 39.4-0.7 0.1 Maxilla (PMP-ANS)* 50.7 50.6 10.1 50.6 50.2-0.4 0.4 Maxilla (PMP-A Pt)* 46.0 45.0-1.0 46.5 44.7-1.8 0.3 Ba-ANS* 90.8 90.1-0.7 90.7 89.6-1.1 0.5 Ba-A Pt* 85.7 84.1-1.6 86.3 83.7-2.6 0.4 Ba-UI* 87.1 88.3 1.2 86.0 88.2 2.2 0.1 Ba-LI* 85.9 86.9 1.0 86.5 87.6 1.1 -O0.7 Ba-B Pt* 88.5 89.8 1.3 88.9 90.7 1.8-0.9 Ba-Pog* 95.9 99.2 3.3 95.2 99.6 4.4-0.4 N-ANS* 45.4 47.0 1.6 45.8 48.7 2.9-1.7 Vertical Proportion 42.4 42.4 0.0 42.6 42.9 0.3-0.5 ANS-UI/N-Men 25.4 24.5-0.9 25.1 24.2-0.9 0.3 Men-LI/N-Men 36.1 36.2 0.1 35.7 35.8 0.1 0.4 Maxilla area (cm )* 20.3 22.4 2.1 20.0 23.1 3.1 -O0.7 Mandible (Co-Gn)* 102.2 106.5 4.3 101.4 107.9 6.5-1.4 Angles Ba-N-ANS 65.1 64.2-0.9 64.9 63.5-1.4 0.7 Ba-N-A Pt 58.9 57.2-1.7 59.7 56.8-2.9 0.4 Ba-N-UI 58.2 58.8 0.6 57.3 58.2 0.9 0.6 Ba-N-LI 57.6 58.1 0.5 58.0 58.2 0.2 -O.1 Ba-N-B Pt 55.9 56.1 0.2 56.0 55.9-0.1 0.2 Ba-N-Pog 56.9 57.8 0.9 56.6 57.3 0.7 0.5 SNA 76.8 75.6-1.2 77.4 75.6-1.8 0.0 SNB 73.7 74.4 0.9 73.7 74.7 1.0-0.3 ANB 3.1 1.2-1.9 3.7 0.9-2.8 0.3 Mandibular plane 54.4 53.6-0.8 55.7 56.6 0.9-3.0 Nasal bone angle< 102.5 105.5 3.0 101.0 104.9 3.9 0.6 + Toronto Standards Maxilla (Ba-N-ANS) 0.7 1.1 0.4 0.2 0.4 0.2 0.7 Mandible (Ba-N-Pog) 0.5 0.8 0.3 0.1 0.3 0.2 0.5 AP Jaw (ANS-N-Pog) 0.2 0.2 0.0 0.2 0.0-0.2 0.2 AP Jaw (ANB) 0.1 0.5 0.4 0.2 0.3 0.1 0.2 Maxilla height* -1.1-1.6-0.5 -O.1 0.0 0.1-1.6 Lower face height -0.3-0.6-0.3 0.1 0.2 0.1-0.8 * Raw linear measurements were adjusted by dividing by the cranial base length (N-Ba) and multiplying by 100. Soft Tissue Repair Group Versus No Repair Group. Tables 5 and 6 and Figure 10 provide a comparison of these two groups. The unrepaired group had slightly less maxillary protrusion (not significant), but jaw relations and ANB angles were the same in the two groups. The maxillary height was significantly greater (by 1.6 mm, p <0.001), and lower face height was slightly greater (not significant), so that vertical facial proportions in the two groups were 42.9 percent maxillary contribution in the unrepaired against 42.2 percent in the repaired group (p <0.05). Groups with All Repairs Versus No Repair Group. Tables 5 and 6 and Figure 11 provide a comparisonof these two groups. The only measurement differences favored the unrepaired group in anterior maxillary height (1.9 mm), maxillary area, and facial vertical proportions (1.1 percent). All were highly significant with p <0.001. The plots were virtually identical except for anterior maxillary height and the mandibular and occlusal plane adjustments to it. DISCUSSION Infant bone grafting, as represented by the four centers in this sample, caused growth attenuation of the maxilla in the two planes of space analyzed. The anteroposterior length and protrusion of the maxilla were reduced, as was anterior maxillary height. There were consequent mandibular adjustments that increased lower face height and produced poor vertical proportions of the face. The Goteborg sample was particularly affected and demonstrated the manner and ex-

Ross, ALVEOLUS REPAIR AND BONE GRAFTING 41 UCLP Alveolus Repair Soft Tissue Repair - 84 males 9.9 yr 14.6 yr -- --- -- -- \\ UCLP Alveolus Repair \ \\ W \ \ R No Repair Group - 206 males 9.0 yr 15.8 yr -- --- -- \ \ \ FIGURE 6 Growth pattern of the soft tissue alveolus repair sample. Plots of Young and Old groups are superimposed on the cranial base (N-Ba) and scaled to the same size. tent to which maxillary growth can be affected. The other bone graft cases had much less effect (the Chicago sample very little), but it was in the same direction, so that the nature if not the extent of the adverse growth influence was expressed. It was surprising to note that the groups bone grafted in childhood had essentially the same morphology by age 15 years as the infant bone graft group, with virtually identical measurements (Table 5). The Auckland cases in the group were grafted earlier, at 4 to 7 years of age, and showed a worsening in anteroposterior relations compared to the nongrafted Auckland cases. The Oslo cases had unsatisfactory maxillary development prior to the placing of the bone graft at 9 to 11 years of age. Maxillary an- FIGURE 7 Growth pattern of the sample with no alveolus repair. Plots of Young and Old groups are superimposed on the cranial base (N-Ba) and scaled to the same size. teroposterior growth thereafter was favorable, indicating that the initial surgery in infancy was probably responsible for the growth attenuation. The Oslo cases were unusual, in that the hard palate was repaired very early, at 3 months of age, and this could be the important variable. If these interpretations are correct, it appears that bone grafting prior to age 8 or 9 is detrimental to anteroposterior development of the maxilla, but there is no apparent effect with bone grafting after this age. All samples of childhood bone grafting had unfavorable vertical relations, although it took some years for these changes to develop. The 4- to 11- year old bone grafted group had reasonably good vertical facial proportions at 10.6 years (the maxillary contribution to the TABLE 5 A Comparison of All Methods of Alveolar Repair and No Repair Infant Bone Graft All Bone Soft Tissue All No Difference: All Data Bone Graft at 4-10 yr Graft Repair Repairs Repair vs No Repair Number 62 28 90 84 174 198 Age 14.5 15.4 14.8 14.6 14.7 14.8 + Toronto Standards Maxilla (Ba-N-ANS) -O.1 -O.1-0.2 1.1 0.4 0.4 0.0 Mandible (Ba-N-Pog) 0.4 0.4 0.4 0.8 0.6 0.3-0.3 AP Jaw (ANS-N-Pog) -O0.5-0.5-0.6 0.3-0.2 0.1 0.3 AP Jaw (ANB) -0.4 0.1-0.4 0.5 0.1 0.3 0.2 Maxilla height* -2.3-2.1-2.3-1.6-1.9 0.0 1.9 Lower face height 1.4 0.8 1.3-0.6 0.3 0.2 0.1 Vertical proportion** 41,2 41.6 41.3 42.4 41.8 42.9 1.1 * Raw measurements adjusted by dividing by cranial base length (N-Ba) ** Toronto Standards not available

42 Cleft Palate Journal, January 1987, Vol. 24 No. 1 UCLP Alveolus Repair \ Infant Bone Graft Group 62 males 14.5 yr Bone Graft Group (4 to 10 years) ------ 28 males 15.4 yr l \ Alveolus Repair Soft Tissue Repair Group 84 males 14.6 yr All Bone Grafts ----- -- 90 males 14.8 yr / FIGURE 8 Infant bone graft sample compared to the sample bone grafted at 4 to 10 years of age. Plots are superimposed on the cranial base (N-Ba) and scaled to the same size. FIGURE 9 All cases bone grafted from birth to 10.9 years compared to the soft tissue alveolus repair. Plots are superimposed on the cranial base (N-Ba) and scaled to the same size. total face height was 42.7 percent) but this deteriorated to 41.6 percent by 15.4 years. The infant bone graft group measured 41.4 percent at 10.6 years and changed very little, becoming 41.2 percent at 14.5 years. The Oslo measurements from the first paper in this series revealed that (a) anterior maxillary height worsened form 9.7 years to 15.0 years (more in this group than in any other of the 14 Centers except Goteborg), (b) lower face height increased more than in any other Center, and (c) the vertical facial proportions worsened more than in any other Center (tied with Goteborg). This appears to confirm that anterior maxillary vertical growth is inhibited following bone grafting, even as late as 9 to 11 years of age. An older group grafted at 11 to 13 years had the same vertical deficiencies, so it appears that only if bone grafting is postponed until age 15 or later can the vertical growth effects be avoided. The question arises as to whether it is the placing (or induction) of bone across the alveolar cleft that causes the growth distortion, or whether the associated soft tissue repair, usually involving a vomer flap, is responsible for a major proportion of the growth disturbance. This study indicates that repair of the alveolus is mainly responsible for the vertical deficiency of the anterior maxilla, and that the deficiency is only slightly worsened by the placement of a bone graft. The bone graft apparently induces additional growth changes, particularly in lower face height and anteroposterior maxillary development. The clinical importance of these findings could be debated. Early alveolus repair, with or TABLE 6 Significance of Differences in Selected Measurements Between Bone Grafted (BG), Soft Tissue Alveolus Repair (ST), All Repair (All) and No Repair (NR) Groups Significance of Difference (p) Measurements BG vs ST BG vs NR ST vs NR ALL vs NR Maxillary length (PMP-A Pt) 0.001 0.001 N N Maxillary protrusion (Ba-N-ANS) 0.02 N N N ANS-N-Pog angle N* N N N ANB angle 0.04 N N N N-ANS N 0.001 0.001 0.001 LFH 0.02 N N N Vertical proportions 0.002 0.001 0.05 0.001 Maxillary area N 0.001 0.001 0.001 * N = not significant

Ross, ALvEOLUS REPAIR AnD BoNnE GrRaArTING ' 43 Alveolus Repair No Repair Group 198 males 14.8 yr Soft Tissue Repair Group 84 males 14.6 yr Alveolus Repair No Repair Group 198 males 14.8 yr All Repairs Grouped ------ 174 males 14.7yr FIGURE 10 Soft tissue repair sample compared to the no alveolus repair sample. Plots are superimposed on the cranial base (N-Ba) and scaled to the same size, without bone grafting, may affect local dental features or cause alterations in the transverse dimensions of the maxilla. These could not be determined with the material available for this study. Orthodontic expansion and subsequent retention should be facilitated with an unrepaired alveolus, free from the binding effect of scar tissue. There are intraoral esthetic advantages to repair, both in dental appearance and the absence of fistulas. Facial esthetics, however, must be adversely influenced by the midface vertical deficiency noted in repaired alveolus cases, both in facial vertical proportions and in the effect on the position of the base of the columellar. The clinical significance of reduced maxillary vertical height and abnormal facial proportions in the vertical dimension can only be hypothesized at present. One characteristic of the male with a unilateral cleft lip and palate is the appearance of a short midface and long lower face. The differences between noncleft cases, the four or five best cleft samples, and the poor cleft samples is approximately 2 percent for each, at a 45- percent, 43-percent and 41-percent maxillary contribution to the total (data from the first paper in this series). A study is underway to determine how accurately facial appearance reflects the underlying skeletal values above and what proportions are detectable to observers. Another result of reduced maxillary vertical height is the altered form of the nose. It is unlikely that overall nose growth is affected by alveolar surgery, but the vertical position of the FIGURE 11 All cases with alveolus repaired compared to those with no alveolus repair. Plots are superimposed on the cranial base (N-Ba) and scaled to the same size. anterior nasal spine determines the position of the base of the columella. If the maxilla is deficient in vertical growth, the base of the columella is raised, giving a more horizontal columellar plane and a reduced nasolabial angle. In individuals with unilateral clefts, this angle is almost 90 degrees instead of the approximately 110 degrees found in noncleft individuals. This horizontal position of the columella is almost certainly related to the midface vertical and anteroposterior deficiency. There is thus the distinct possibility that repair of the alveolus, especially by means of infant and early childhood bone grafting, has a very detrimental effect on facial esthetics in individuals with unilateral cleft lip and palate. Given the many esthetic problems these individuals have, a very good argument could be advanced for delaying alveolus repair at least until the mixed dentition when secondary bone grafting offers such great clinical benefits. At that time, an evaluation of vertical proportions might identify cases that would be adversely affected by alveolus repair and bone grafting. In addition to esthetic considerations, there are difficulties in establishing good functional jaw relations, even with orthodontic treatment, for many children with unilateral cleft lip and palate. It might be well, therefore, to avoid bone grafting until the mixed dentition, and further delay it to age 10 or 11 in individuals with unfavorable anteroposterior growth patterns. Bone grafting should probably be avoided completely

44 Cleft Palate Journal, January 1987, Vol. 24 No. 1 in openbite cases, although this is a suggestion not based directly on the data available. In the same way that different individuals respond in different ways to a given procedure, there must be variations in procedures which will affect growth more orless than the data presented here indicate. If clinicians are convinced that alveolus repair and bone grafting are valuable treatment procedures, then an effort should be made to investigate or develop techniques which will provide the full bony reconstruction of the alveolar defect, but will not have adverse effects. Some of the procedures in this study, while promising, involved samples which were too small or too young to serve as evidence of a satisfactory method. SUMMARY The findings of this study indicate that: - 1. Early repair ofthe cleft alveolus by any means has a detrimental effect on maxillary growth. 2. Bone grafting in infancy may cause growth attenuation of the maxilla in length and height, and may induce compensatory changes in the lower face that adversely alter facial vertical proportions. There were variations noted with different surgical techniques. 3. Bone grafting in children from 4 to 10 years of age has almost the same effect as infant bone grafting, although there is no apparent effect on the anteroposterior development of the maxilla following bone grafting at age 9 or later. 4. Bone grafting in the late mixed dentition adversely affects the vertical dimensions of the anterior maxilla and indirectly the lower face. 5. Soft tissue repair of the alveolus results in a deficiency in vertical growth of the anterior maxilla. Anteroposterior relations, however, are not affected. 6. These findings should be applied to individuals at risk for poor facial growth. Since this is impossible to determine accurately at an early age, clinicians should consider avoiding bone grafting or alveolus repair until the child is well into the mixed dentition, or later. References ABYHOLM FE, BERGLAND O, SEMB G. Secondary bone grafting of alveolar clefts. Scand J Plast Reconstr Surg 1981; 15:127. BERGLAND O. Changes in cleft palate malocclusion after the introduction of improved surgery. Eur Orthod Soc Trans 1967; 43:383. BoYyNE PJ, SaNDs NR. Secondary bone grafting of residual alveolar and palatal clefts. J Oral Surg 1972; 30:87. DAHL E, HAanUsArRDOTTER B, BERGLAND O. A comparison of occlusions in two groups of children whose clefts were repaired by three different surgical procedures. Cleft Palate J 1981; 18:122. Entow DH. The human face. New York: Hoeber, 1968. FRIEDE H, JOHANSON B. Adolescent facial morphology of early bone grafted cleft lip and palate patients. Scand J Plast Reconstr Surg 1982; 16:41. HELLQUIST R, PONTEN B. The influence of infant periosteoplasty on facial growth and dental occlusion from five to eight years of age in cases of complete unilateral cleft lip and palate. Scand J Plast Reconstr Surg 1979; 13:305. KoBERG WR. Present view on bone grafting in cleft palate. J Maxillofac Surg 1973; 1:185. McNEm CK. Orthodontic procedures in the treatment of congenital cleft. Dent Rec 1950; 70:125. NoORDIN KE, JOHANSON B. Freie Knochentransplantation bei Defekten im Alveolarkamm nach kiefer-orthopadischer Einstellung der Maxilla bei Lippen-Kiefer-Gaumenspalten. Fortschr. Kiefer-u. Gesichts-chir., Bd. 1. Stuttgart: Thieme, 1955. ROBERTSON MRE, JOLLEYS A. An 11-year follow-up of the effects of early bone grafting in infants born with complete clefts of the lip and palate. Br J Plast Surg 1983; 36:438. RosENsTEIN SW, MunroE CW, KERNAHAN DA, JaAcoBson BN, GrirFitH BH, BAUER BS. The case for early bone grafting in cleft lip and palate. Plast Reconstr Surg 1982; 70:297. Ross RB. The clinical implications of facial growth in cleft lip and palate. Cleft Palate J 1970; 7:37. SCHMID E. Die Annaherung der Kieferstumpfe bei Lippen- Kiefer-Gaumenspalten; ihre schadlichen Folgen und Vermeidung. Fortschr. Kiefer-u. Gesichts-chir., Bd. 1. Stuttgart: Thieme, 1955. WITsENBURG B. The reconstruction of anterior residual bone defects in patients with cleft lip, alveolus and palate: a review. J Maxillofac Surg 1985; 13:197.