The Cleft Palate–Craniofacial Journal 00(00) pp. 000–000 Month 2015 Ó Copyright 2015 American Cleft Palate–Craniofacial Association
ORIGINAL ARTICLE Secondary Alveolar Bone Grafting (CLEFTSiS) 2007–2010 Michael Paterson, B.D.S., Jennifer Rae, B.D.S., Paul Paterson, B.D.S.(Hons.), M.F.D.S., R.C.P.S.(Glasg.), Toby Gilgrass, B.D.S., M.Sc., F.D.S.(Orth.), Mark Devlin, F.R.C.S.Ed.(O.M.F.S.), F.R.C.S.(Glasg.), F.D.S.R.C.P.S., Grant McIntyre, Ph.D., M.Orth., F.D.S.(Orth.) Objective: To determine whether alveolar bone graft outcomes for unilateral and bilateral cleft lip and palate patients have continued to improve since the reorganization of cleft services in Scotland in 2000. Design: Retrospective analysis of postoperative anterior occlusal radiographs. Patients and Participants: Eighty-one of 106 patients who were eligible for alveolar bone grafting between 2007 and 2010 had suitable postoperative radiographs available. Interventions: Twenty-seven percent of the patients (n ¼ 22) had presurgical orthodontic intervention. All patients underwent alveolar bone grafting with bone harvested from the iliac crest. Main Outcome Measures: The Kindelan bone-fill index was used to evaluate success. Weighted kappa statistics were used to assess intra- and interobserver reproducibility. A comparison was made with results from 2000 to 2004 to assess any improvement. Chi-square tests (or Fisher exact test) were used to determine whether outcomes differed depending on the laterality of the cleft, use of presurgical expansion, or age at bone grafting. Results: Interobserver scoring agreement was good (weighted kappa ¼ .383). Intraobserver reproducibility was greater (weighted kappas of .835 and .620). Success was achieved in 99% of bone grafts, compared with 76% in the period from 2000 to 2004 (P , .001). There was no statistically significant relationship between the laterality of the cleft (P ¼ 1.000), use of presurgical expansion (P ¼ 1.000), or age at time of bone grafting and outcome (P ¼ .259). Conclusion: Scottish secondary alveolar bone graft outcomes improved during 2007 to 2010 in comparison to the 2000 to 2004 results. KEY WORDS:
bone graft, cleft lip, outcome, palate
Secondary alveolar bone grafting for patients with a cleft involving the maxillary alveolus was first advocated by Boyne and Sands (1972, 1976) and has transformed the dental management of children with a cleft involving the maxillary alveolus (Bergland et al., 1986a, 1986b).
The grafting procedure is usually undertaken around the age of 9 to 11 years to coincide with dental development, most notably of the cleft side permanent canine (Bergland et al., 1986a). The lateral incisor is often absent on the cleft side. However, earlier grafting may be considered when it is present, of reasonable form, and on the mesial side of the cleft (Kalaaji et al., 1996; Enemark et al., 2001). Once erupted through the graft, the canine maintains the alveolar bone within the grafted region, as without functional stimulation, the bone rapidly resorbs (Feichtinger et al., 2007). The erupted canine has adequate periodontal support to allow orthodontic movement of the tooth (Dewinter et al., 2003), and orthodontic space closure can be considered for many children, obviating the need for a final prosthesis (Ramstad and Semb, 1997). Prebone graft orthodontic intervention has, in some studies, led to improved outcomes (McIntyre and Devlin, 2010) and may be used to correct anterior and some posterior crossbites where they are considered due to cleft segment collapse rather than skeletal discrepancy. Irrespective of the crossbite correction, the main aim of the orthodontic intervention is to improve surgical access to the floor of the nose to allow closure prior to graft placement (Kindelan and Roberts-Harry, 1999).
Mr. Paterson is Vocational Dental Practitioner, NHS Highland. Ms. Rae is Vocational Dental Practitioner, NHS Dumfries & Galloway. Mr. Paterson is third-year medical student, University of Dundee. Mr. Gilgrass is Consultant Cleft Orthodontist, West of Scotland Cleft Team, Royal Hospital for Sick Children, Glasgow, United Kingdom, and affiliated with University of Glasgow, Honorary Clinical Senior Lecturer. Mr. Devlin is Cleft and Maxillofacial Surgeon, West of Scotland Cleft Team, Royal Hospital for Sick Children, Glasgow, United Kingdom, and affiliated with University of Glasgow, Honorary Clinical Senior Lecturer. Dr. McIntyre is Consultant in Orthodontics, University of Dundee, Honorary Senior Lecturer. Presented at the Cleftsis National Meeting, Perth Royal Infirmary Lecture Theatre, March 29, 2012. Submitted March 2014; Revised August 2014; Accepted November 2014. Address correspondence to: Mr. Michael Paterson, C/O Mr. Toby Gilgrass, Consultant Orthodontist, Glasgow Dental Hospital & School, 378 Sauchiehall St, Glasgow, Scotland, G2 3JZ. E-mail: [email protected]. DOI: 10.1597/14-093 0
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The operation is carried out under general anesthesia. Local mucoperiosteal flaps are raised around the alveolar cleft to define the bony defect fully. Important steps within the procedure include careful repair of the nasal floor, filling of the bony defect with wellcondensed autogenous cancellous bone chips, minimizing the time between harvest and placement, and a water-tight soft tissue closure around the graft with well-vascularized keratinized local flaps to allow tooth eruption. Although a number of potential autogenous donor sites have been used for repairing the alveolar defect, including mandible (Koole et al., 1989; Enemark et al., 2001), tibia (Kalaaji et al., 2001), and cranium (LaRossa et al., 1995; Denny et al., 1999), the iliac crest remains the most commonly utilized and is used for procedures within the CLEFTSiS surgical service. Conventional post–bone graft radiographs are scanned and entered into the electronic patient record (EPR), and the outcomes are scored and included in the CLEFTSiS annual report (available at http://www.cleftsis.scot.nhs.uk/ ). McIntyre and Devlin (2010) investigated cases completed between 2000 and 2004 and noted record collection during the annual audit period was variable, with the availability of records being at times problematic. This has led to more coordinated methods of data collection through designated clinics for grafting in some centers within Scotland, with the aim of better record collection. The Clinical Standards Advisory Group (CSAG) investigation into the status of cleft care in the United Kingdom in 1996 identified that the success rate for alveolar bone grafting was 58% (Williams et al., 2001). Following reorganization of cleft services within the United Kingdom, a more recent study examining grafts carried out in 2006 found an improvement in success rates to 85% (Revington et al., 2010). McIntyre and Devlin (2010) reported that for Scotland, grafts carried out between 2000 and 2004 had a success rate of 76%. While this was better than the findings of CSAG, it still left room for improvement, given that Revington et al. (2010) and others achieved .90% success (Hall and Posnick, 1983; Bergland et al., 1986a; Kalaaji et al., 1996; Demas and Sotereanos, 1988; Lilja et al., 2000; Jia et al., 2006). Reorganization of cleft care in Scotland has differed from England and Wales, where services related to cleft lip and palate are fully commissioned. The establishment of the first National Managed Clinical Network for Cleft Services in Scotland (CLEFTSiS) took place in 2000 and continues to the present day. Through the intervening years, there has been a progressive reduction in the numbers of surgeons undertaking alveolar bone grafting as a drive to increase the volume of grafting carried out by each operator. In Scotland, the number of surgeons in 2000 to 2004 was nine (McIntyre and Devlin, 2010), and by 2007, this had reduced to three.
Study Objectives 1. To compare the secondary alveolar bone graft outcomes from 2007 to 2010 with those from 2000 to 2004. 2. To determine whether outcomes differed depending on the laterality of the cleft (unilateral/bilateral), use of presurgical expansion, or age at time of bone grafting. 3. To reassess the availability on the EPR of records for the assessment of alveolar bone graft outcomes. MATERIALS
AND
METHODS
The ExceliCare EPR (ExceliCare, AxSys Technology, Glasgow, UK) database was used to access pre- and postoperative alveolar bone graft radiographic records of patients treated within the CLEFTSiS network between January 1, 2007, and December 31, 2010. The radiographs were accessed via the EPR and scored for radiographic success by two independent observers. Information regarding type of cleft, operating surgeon, use of presurgical orthodontics, age at grafting, and postcode were also collected. The principles of the Declaration of Helsinki were followed throughout. A total of 106 patients were eligible for alveolar bone grafting in the 4-year cohort studied. One patient elected not to undergo surgery. Each patient had one surgeon carrying out their bone graft. Postoperative occlusal radiographs were available for 82 patients, of which 1 was deemed unreadable. The 81 remaining patient records were anonymized. The radiographs were collated into a single document and scored by two consultant orthodontists, one based in Glasgow and the other based in Dundee. This eliminated any opportunity for collusion. Scoring took place on two separate occasions per observer, 2 weeks apart. Where image quality presented a problem, the contrast and brightness were altered by an individual independent to the scorers to determine the location and quantity of the bone graft. The radiographs were then scored using the Kindelan bone-fill index (Kindelan et al., 1997; Kindelan and Roberts-Harry, 1999). This is a four-grade scale in which grade 1 signifies .75% bony fill, grade 2 is 50% to 75% bony fill, grade 3 is ,50% bony fill, and grade 4 means no complete bony bridge. Grades 1 and 2 were deemed successful, grade 3 represented a partial failure, and grade 4 was considered a total failure. The success of alveolar bone grafting can be determined using a number of other clinical and radiographic measures (Bergland et al., 1986a; Long et al., 1995; Witherow et al., 2002). However, these indices are designed to be used once the permanent maxillary canine has erupted, often a significant time after the initial graft. Where the outcome is poor and additional bone grafting is deemed necessary, the success of this further graft may be jeopardized by this
Paterson et al., SECONDARY ALVEOLAR BONE GRAFTING (CLEFTSIS) 2007–2010
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TABLE 1 Intra- and Interobserver Unweighted Kappa Scores for Observers 1 and 2*
Observer 1 Observer 2
Observer 1
Observer 2
0.831 (0.711–0.951) 0.363 (0.170–0.556)
0.363 (0.170–0.556) 0.587 (0.420–0.753)
delay (Dickinson et al., 2008), unless these indices are modified for earlier use (Williams et al., 2003). The Kindelan bone-fill index (Kindelan et al., 1997) can be applied any time after bony integration has taken place by comparing the preoperative and postoperative anterior occlusal radiograph. Indeed, the cleft canine does not need to be erupted for the Kindelan index to be used and, because radiographic success is determined at an early stage, repeat bone grafting can still be undertaken within the optimal time period. Although there is little evidence to suggest that a good early Kindelan outcome score relates to clinical success (Seike et al., 2012), it remains the most commonly used method of bone graft outcome assessment in the United Kingdom. With the advent of cone beam computed tomography within the CLEFTSiS network, the number of preoperative anterior occlusal radiographs taken has been reduced. Although this prevents direct comparison between pre- and postoperative radiographs, recent work suggests that this may not be critical in determining an outcome score using the Kindelan bone-fill index (Dobbyn et al., 2012). Intraobserver reproducibility of the Kindelan scoring was measured using the kappa statistic (Landis and Koch, 1977). Then, another kappa statistic was calculated to assess interobserver reproducibility. Where disagreement existed on a particular set of radiographs, a third consensus score was discussed and agreed over the telephone. A chi-square test of independence was used to compare the data from 2007 to 2010 with that obtained by McIntyre and Devlin (2010) for 2000 to 2004. The data were categorized as either success (score 1 or 2) or failure (score 3 or 4) of bone graft before undertaking this test. When considering the comparison of outcomes of unilateral and bilateral clefts, it is important to note that the side with the worst score in bilateral cases was used for comparison. When comparing age at time of bone grafting, the patients were categorized either as younger than 11 years or greater than or equal to 11 years. Eleven years was TABLE 2 Kindelan Bone-Fill Index Outcomes Obtained in This Study and Results From McIntyre and Devlin (2010) Grade 1
Grade 2
Grade 3
Grade 4
Total
2000–2004 Number of patients Percentage
36 32
50 44
26 23
2 2
114
2007–2010 (this study) Number of patients Percentage
49 60
31 38
1 1
0 0
81
FIGURE 1 Chart showing percentage of patients allocated each Kindelan bone-fill index score. The white columns depict results from this study (2007 to 2010). Black represents data from McIntyre and Devlin (2010), who reported on 2000 to 2004. Note the large increase in the percentage of patients achieving grade 1 in this study.
chosen as this is the upper limit of the normal age range during which to perform bone grafting. Chi-square tests (or Fisher exact test) were used to determine whether outcomes differed depending on the laterality of the cleft (unilateral/ bilateral), use of presurgical expansion, or age at time of bone grafting. Data regarding the quality of the primary palatal repair performed on each patient were not available. Related to this, no account was taken of the preoperative size of the clefts as radiographic analysis alone could not give sufficiently accurate data. RESULTS Table 1 shows the intra- and interobserver unweighted kappa scores for observers 1 and 2. Interobserver scoring agreement was good, with a kappa of .363. The weighted kappa value for interobserver agreement was .383. Intraobserver reproducibility was better with scores of .831 and .587 (weighted values .835 and .620, respectively). We shall now consider the Kindelan bone-fill index outcomes obtained for the 81 patients included in this study, alongside the results from McIntyre and Devlin (2010). Success is defined as having a Kindelan score of 1 or 2. Overall, for this study (n ¼ 81), bone grafting was successful in 99% of cases (n ¼ 80). This compares with the 76% success rate in the data set from 2000 to 2004. This is a significant improvement (P ¼ .000006). It is also notable that the percentage of total failures has fallen from 2% (n¼ 2) in 2000 to 2004 to 0% in this study. These results are shown in Table 2. Figure 1 helps demonstrate the trend of continued improvement in outcomes. In particular, it is notable that the percentage of patients scoring grade 1 on the Kindelan index has risen from 32% in 2000 to 2004 to 60% in 2007 to 2010.
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TABLE 3
Effect of Laterality of the Cleft, Use of Presurgical Expansion, and Age at Time of Bone Grafting on Outcome* Percentage Success
Unilateral Bilateral Presurgical maxillary expansion No presurgical expansion Age ,11 y Age 11 y
98.5 100 100 98.3 100 95.2
(66 (14 (22 (58 (60 (20
of of of of of of
67) 14) 22) 59) 60) 21)
P Value 1.000* 1.000* .259*
Percentage With Kindelan Score of 1 61.2 57.1 55.4 62.7 63.3 52.4
(41 of 67) (8 of 14) (12 of 22) (37 of 59) (38 of 60) (11 of 21)
P Value .778 .504 .377
* Fisher exact test used.
There was no statistically significant relationship between the laterality of the cleft (P ¼ 1.000), use of presurgical expansion (P ¼ 1.000), or age at time of bone grafting (P ¼ .259) and outcomes. In light of the high success rate seen in this study, it was decided to recategorize the data into those patients having a successful bone graft with Kindelan score 1 and those having worse outcomes. This was done to determine any trends missed by examining only success or failure. The results of this analysis are presented in Table 3, and although the P values are lower, none are significant at P , .05. DISCUSSION This study reports the radiographic outcomes of secondary alveolar bone grafting in Scotland for patients with clefts involving the alveolus (and where the records were available in the EPR) for the years 2007 to 2010. It is notable that the data set presented is incomplete, with 82 of 106 patients having postoperative occlusal radiographs (77%). This is disappointing but is similar to the percentage achieved by Revington et al. (2010) of 70% in their U.K. national audit. Seventy-seven percent represents a significant improvement on the previous report for CLEFTSiS (McIntyre and Devlin, 2010), which found access to postoperative radiographs in only 46%. It is notable that in the CSAG study, there was a similar significant attrition bias, with only 49% of postoperative radiographs being available (CSAG, 1998). The improvements may be due the dissemination of the results from the previous study and the reconfiguration of the service in some areas resulting in postoperative radiographs being taken in a single location. However, the system fundamentally relies on scanning (conventional films) and uploading patient information. Although, in England and Wales, this would routinely be the job of the coordinator, this facility is not available for much of Scotland. It is unknown what effect the missing radiographs would have had; this should be taken into consideration when viewing the results of this investigation. As alluded to earlier, another limitation of this study is the lack of data on the quality of the primary palatal repair or preoperative dimensions of the cleft. As such, these factors are not taken into account when assessing success of secondary bone grafting. The radiologic method used for assessment of the grafts in this cohort was the anterior occlusal radiograph. A
significant number of studies have also used the anterior occlusal radiograph as the standard assessment tool (Hall and Posnick, 1983; Bergland et al., 1986a, 1986b; Enemark et al., 1987; Demas and Sotereanos, 1988; Amanat and Langdon, 1991; Kortebein et al., 1991; Freihofer et al., 1993; LaRossa et al., 1995; Long et al., 1995; Kalaaji et al., 1996; Tan et al., 1996; Kindelan et al., 1997; CSAG, 1998; Montecinos et al., 1998; Denny et al., 1999; Kindelan and Roberts-Harry, 1999; Opitz et al., 1999; Aurouze et al., 2000; Jia and Mars, 2000; Lilja et al., 2000; Mao et al., 2000; Kalaaji et al., 2001; Williams et al., 2001; Jia et al., 2002; Witherow et al., 2002; Schultze-Mosgau et al., 2003; Nightingale et al., 2003; Williams et al., 2003; Clarkson et al., 2005; Trindade et al., 2005; Bayerlein et al., 2006; Jia et al., 2006; Rawashdeh and Nimri, 2007; Matic and Power, 2008; McIntyre and Devlin, 2010; Francis et al., 2013). Within the CLEFTSiS network, attempts have been made to reduce the variability in quality of radiographs produced between centers by a descriptive protocol for radiograph production. Only one radiograph was deemed unsuitable for assessment within the present cohort. This is similar to the work of McIntyre and Devlin (2010), who reported ,1% and Revington et al. (2010) with 1.4%. It also compares favorably with that of the CSAG report (1998), which found 14% to be unsuitable. The index used in the present study was the Kindelan bone-fill index (Kindelan et al., 1997), as that not only allows direct comparison with McIntyre and Devlin (2010) but also was used in the national study by Revington et al. (2010). Examining the weighted kappa statistics for inter- and intraobserver reproducibility revealed the former to be .383 and the latter two as .835 and .620. This suggests a moderate/good level of agreement between the observers used in this study, and the values are similar to those of other studies (Kindelan et al., 1997; Nightingale et al., 2003; Revington et al., 2010; Dobbyn et al., 2012). A 99% success rate (Kindelan grades 1 and 2) represents a significant improvement in comparison with McIntyre and Devlin’s (2010) study into the years 2000 through 2004, which reported a 76% success rate (P , .001). This result is also an improvement on the results from the CSAG (1998) study, although caution should be used with this comparison as they used the modified Bergland Index (Williams et al., 2003). The result also compares favorably with those studies having success rates of .90% (Hall and Posnick, 1983; Bergland et al., 1986a; Demas and Sotereanos, 1988;
Paterson et al., SECONDARY ALVEOLAR BONE GRAFTING (CLEFTSIS) 2007–2010
Kalaaji et al., 1996; Newlands, 2000; Jia et al., 2006) and can be viewed as a performance indicator of continuing improvement in cleft services in Scotland over recent years. Since 2000 to 2004, significant changes in the Scottish alveolar bone graft service have taken place. Previously, there were nine surgeons carrying out alveolar bone graft surgery across Scotland. This was reduced in 2007 to three dedicated surgeons, with surgeons and orthodontists improving the coordination of bone graft delivery through dedicated bone graft clinics and the extraction of any unwanted teeth in the surgical area 3 months in advance of bone graft surgery. These factors may have contributed to an improvement in successful bone grafting outcomes and are in accordance with the finding of improved success rates with high-volume operators (Nightingale et al., 2003) and those with greater experience (Kalaaji et al., 1996). Attempting to examine factors that may affect successful outcomes is difficult with an overall success rate of 99%. However, when considering the results pertaining to unilateral and bilateral clefts, the data suggest that unilateral clefts have better outcomes. This is evidenced by 61.2% of unilateral cases achieving a Kindelan score of 1 compared with only 57.1% of bilateral cases. A similar small percentage difference in outcomes between unilateral and bilateral cases was found by Revington et al. (2010), but as in this study (P ¼ .778), no significant difference was found between the two. This is in accordance with other studies (Bergland et al., 1986b; Long et al., 1995; Kindelan and Roberts-Harry, 1999; Newlands, 2000; McIntyre and Devlin, 2010). Perhaps a larger data set would allow a more significant result to be established. An interesting trend exists for the use of presurgical maxillary expansion. Contrary to McIntyre and Devlin’s (2010) findings, it appears that those patients without maxillary expansion had better outcomes (62.7% obtaining a score of 1 compared with 55.4% for those undergoing expansion). Again, this apparent trend is not statistically significant. Finally, those patients younger than 11 years at the time of alveolar bone grafting were found to have a 100% success rate (Kindelan score of 1 or 2), whereas those 11 years or older had only 95.2% success. Although this difference was not statistically significant, it replicates the findings of Lee et al. (1995), Kalaaji et al. (1996), CSAG (1998), Kindelan and Roberts-Harry (1999), and Williams et al. (2003), who found increasing age a predictor of poorer outcome. CONCLUSIONS Scottish secondary alveolar bone graft outcomes improved during 2007 to 2010 in comparison with the 2000 to 2004 results. There were no significant relationships between laterality of the cleft, use of presurgical expansion, or age at time of bone grafting on secondary alveolar bone graft outcomes. Only 77% of the patient cohort studied had
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Matic DB, Power SM. Evaluating the success of gingivoperiosteoplasty versus secondary bone grafting in patients with unilateral clefts. Plast Reconstr Surg. 2008;121:1343–1353. McIntyre GT, Devlin MF. Secondary alveolar bone grafting (CLEFTSiS) 2000–2004. Cleft Palate Craniofac J. 2010;47:66–72. Montecinos G, Almeida A, Heilenkotter E, Almeida V, Cabezas G. ¨ Iliac bone for secondary grafting in residual alveolar clefts. Cir Pediatr. 1998;11:97–101. Newlands LC. Secondary alveolar bone grafting in cleft lip and palate patients. Br J Oral Maxillofac Surg. 2000;38:488–491. Nightingale C, Witherow H, Reid FD, Edler R. Comparative reproducibility of three methods of radiographic assessment of alveolar bone grafting. Eur J Orthod. 2003;25:35–41. Opitz C, Meier B, Stoll C, Subklew D. Radiographic evaluation of the transplant bone height in patients with clefts of the lip/alveolus/ palate after secondary bone grafting. J Orofac Orthop. 1999;60:383– 391. Ramstad T, Semb G. The effect of alveolar bone grafting on the prosthodontics/reconstructive treatment of patients with unilateral complete cleft lip and palate. Int J Prosthodont. 1997;10:156–163. Rawashdeh MA, Al Nimri KS. Outcome of secondary alveolar bone grafting before and after eruption of the canine in Jordanian patients with cleft lip and palate. J Craniofac Surg. 2007;18:1331– 1337. Revington PJ, McNamara C, Mukarram S, Perera E, Shah HV, Deacon SA. Alveolar bone grafting: results of a national outcome study. Ann R Coll Surg Engl. 2010;92:643–646. Schultze-Mosgau S, Nkenke E, Schlegel AK, Hirschfelder U, Wiltfang J. Analysis of bone resorption after secondary alveolar cleft bone grafts before and after canine eruption in connection with orthodontic gap closure or prosthodontic treatment. J Oral Maxillofac Surg. 2003;61:1245–1248. Seike T, Hashimoto I, Matsumoto K, Tanaka E, Nakanishi H. Early postoperative evaluation of secondary bone grafting into the alveolar cleft and its effects on subsequent orthodontic treatment. J Med Invest. 2012;59:152–165. Tan AE, Brogan WF, McComb HK, Henry PJ. Secondary alveolar bone grafting—five-year periodontal and radiographic evaluation in 100 consecutive cases. Cleft Palate Craniofac J. 1996;33:513–518. Trindade IK, Mazzottini R, Silva Filho OG, Trindade IE, Deboni MC. Long-term radiographic assessment of secondary alveolar bone grafting outcomes in patients with alveolar clefts. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2005;100:271–277. Williams AC, Bearn D, Mildinhall S, Murphy T, Sell D, Shaw WC, Murray JJ, Sandy JR. Cleft lip and palate care in the United Kingdom—the Clinical Standards Advisory Group (CSAG) Study. Part 2: dentofacial outcomes and patient satisfaction. Cleft Palate Craniofac J. 2001;38:24–29. Williams A, Semb G, Bearn D, Shaw W, Sandy J. Prediction of outcomes of secondary alveolar bone grafting in children born with unilateral cleft lip and palate. Eur J Orthod. 2003;25:205–211. Witherow H, Cox S, Jones E, Carr R, Waterhouse N. A new scale to assess radiographic success of secondary alveolar bone grafts. Cleft Palate Craniofac J. 2002;39:255–260.
ORIGINAL ARTICLE Secondary Alveolar Bone Grafting (CLEFTSiS) 2007–2010 Michael Paterson, B.D.S., Jennifer Rae, B.D.S., Paul Paterson, B.D.S.(Hons.), M.F.D.S., R.C.P.S.(Glasg.), Toby Gilgrass, B.D.S., M.Sc., F.D.S.(Orth.), Mark Devlin, F.R.C.S.Ed.(O.M.F.S.), F.R.C.S.(Glasg.), F.D.S.R.C.P.S., Grant McIntyre, Ph.D., M.Orth., F.D.S.(Orth.) Objective: To determine whether alveolar bone graft outcomes for unilateral and bilateral cleft lip and palate patients have continued to improve since the reorganization of cleft services in Scotland in 2000. Design: Retrospective analysis of postoperative anterior occlusal radiographs. Patients and Participants: Eighty-one of 106 patients who were eligible for alveolar bone grafting between 2007 and 2010 had suitable postoperative radiographs available. Interventions: Twenty-seven percent of the patients (n ¼ 22) had presurgical orthodontic intervention. All patients underwent alveolar bone grafting with bone harvested from the iliac crest. Main Outcome Measures: The Kindelan bone-fill index was used to evaluate success. Weighted kappa statistics were used to assess intra- and interobserver reproducibility. A comparison was made with results from 2000 to 2004 to assess any improvement. Chi-square tests (or Fisher exact test) were used to determine whether outcomes differed depending on the laterality of the cleft, use of presurgical expansion, or age at bone grafting. Results: Interobserver scoring agreement was good (weighted kappa ¼ .383). Intraobserver reproducibility was greater (weighted kappas of .835 and .620). Success was achieved in 99% of bone grafts, compared with 76% in the period from 2000 to 2004 (P , .001). There was no statistically significant relationship between the laterality of the cleft (P ¼ 1.000), use of presurgical expansion (P ¼ 1.000), or age at time of bone grafting and outcome (P ¼ .259). Conclusion: Scottish secondary alveolar bone graft outcomes improved during 2007 to 2010 in comparison to the 2000 to 2004 results. KEY WORDS:
bone graft, cleft lip, outcome, palate
Secondary alveolar bone grafting for patients with a cleft involving the maxillary alveolus was first advocated by Boyne and Sands (1972, 1976) and has transformed the dental management of children with a cleft involving the maxillary alveolus (Bergland et al., 1986a, 1986b).
The grafting procedure is usually undertaken around the age of 9 to 11 years to coincide with dental development, most notably of the cleft side permanent canine (Bergland et al., 1986a). The lateral incisor is often absent on the cleft side. However, earlier grafting may be considered when it is present, of reasonable form, and on the mesial side of the cleft (Kalaaji et al., 1996; Enemark et al., 2001). Once erupted through the graft, the canine maintains the alveolar bone within the grafted region, as without functional stimulation, the bone rapidly resorbs (Feichtinger et al., 2007). The erupted canine has adequate periodontal support to allow orthodontic movement of the tooth (Dewinter et al., 2003), and orthodontic space closure can be considered for many children, obviating the need for a final prosthesis (Ramstad and Semb, 1997). Prebone graft orthodontic intervention has, in some studies, led to improved outcomes (McIntyre and Devlin, 2010) and may be used to correct anterior and some posterior crossbites where they are considered due to cleft segment collapse rather than skeletal discrepancy. Irrespective of the crossbite correction, the main aim of the orthodontic intervention is to improve surgical access to the floor of the nose to allow closure prior to graft placement (Kindelan and Roberts-Harry, 1999).
Mr. Paterson is Vocational Dental Practitioner, NHS Highland. Ms. Rae is Vocational Dental Practitioner, NHS Dumfries & Galloway. Mr. Paterson is third-year medical student, University of Dundee. Mr. Gilgrass is Consultant Cleft Orthodontist, West of Scotland Cleft Team, Royal Hospital for Sick Children, Glasgow, United Kingdom, and affiliated with University of Glasgow, Honorary Clinical Senior Lecturer. Mr. Devlin is Cleft and Maxillofacial Surgeon, West of Scotland Cleft Team, Royal Hospital for Sick Children, Glasgow, United Kingdom, and affiliated with University of Glasgow, Honorary Clinical Senior Lecturer. Dr. McIntyre is Consultant in Orthodontics, University of Dundee, Honorary Senior Lecturer. Presented at the Cleftsis National Meeting, Perth Royal Infirmary Lecture Theatre, March 29, 2012. Submitted March 2014; Revised August 2014; Accepted November 2014. Address correspondence to: Mr. Michael Paterson, C/O Mr. Toby Gilgrass, Consultant Orthodontist, Glasgow Dental Hospital & School, 378 Sauchiehall St, Glasgow, Scotland, G2 3JZ. E-mail: [email protected]. DOI: 10.1597/14-093 0
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The operation is carried out under general anesthesia. Local mucoperiosteal flaps are raised around the alveolar cleft to define the bony defect fully. Important steps within the procedure include careful repair of the nasal floor, filling of the bony defect with wellcondensed autogenous cancellous bone chips, minimizing the time between harvest and placement, and a water-tight soft tissue closure around the graft with well-vascularized keratinized local flaps to allow tooth eruption. Although a number of potential autogenous donor sites have been used for repairing the alveolar defect, including mandible (Koole et al., 1989; Enemark et al., 2001), tibia (Kalaaji et al., 2001), and cranium (LaRossa et al., 1995; Denny et al., 1999), the iliac crest remains the most commonly utilized and is used for procedures within the CLEFTSiS surgical service. Conventional post–bone graft radiographs are scanned and entered into the electronic patient record (EPR), and the outcomes are scored and included in the CLEFTSiS annual report (available at http://www.cleftsis.scot.nhs.uk/ ). McIntyre and Devlin (2010) investigated cases completed between 2000 and 2004 and noted record collection during the annual audit period was variable, with the availability of records being at times problematic. This has led to more coordinated methods of data collection through designated clinics for grafting in some centers within Scotland, with the aim of better record collection. The Clinical Standards Advisory Group (CSAG) investigation into the status of cleft care in the United Kingdom in 1996 identified that the success rate for alveolar bone grafting was 58% (Williams et al., 2001). Following reorganization of cleft services within the United Kingdom, a more recent study examining grafts carried out in 2006 found an improvement in success rates to 85% (Revington et al., 2010). McIntyre and Devlin (2010) reported that for Scotland, grafts carried out between 2000 and 2004 had a success rate of 76%. While this was better than the findings of CSAG, it still left room for improvement, given that Revington et al. (2010) and others achieved .90% success (Hall and Posnick, 1983; Bergland et al., 1986a; Kalaaji et al., 1996; Demas and Sotereanos, 1988; Lilja et al., 2000; Jia et al., 2006). Reorganization of cleft care in Scotland has differed from England and Wales, where services related to cleft lip and palate are fully commissioned. The establishment of the first National Managed Clinical Network for Cleft Services in Scotland (CLEFTSiS) took place in 2000 and continues to the present day. Through the intervening years, there has been a progressive reduction in the numbers of surgeons undertaking alveolar bone grafting as a drive to increase the volume of grafting carried out by each operator. In Scotland, the number of surgeons in 2000 to 2004 was nine (McIntyre and Devlin, 2010), and by 2007, this had reduced to three.
Study Objectives 1. To compare the secondary alveolar bone graft outcomes from 2007 to 2010 with those from 2000 to 2004. 2. To determine whether outcomes differed depending on the laterality of the cleft (unilateral/bilateral), use of presurgical expansion, or age at time of bone grafting. 3. To reassess the availability on the EPR of records for the assessment of alveolar bone graft outcomes. MATERIALS
AND
METHODS
The ExceliCare EPR (ExceliCare, AxSys Technology, Glasgow, UK) database was used to access pre- and postoperative alveolar bone graft radiographic records of patients treated within the CLEFTSiS network between January 1, 2007, and December 31, 2010. The radiographs were accessed via the EPR and scored for radiographic success by two independent observers. Information regarding type of cleft, operating surgeon, use of presurgical orthodontics, age at grafting, and postcode were also collected. The principles of the Declaration of Helsinki were followed throughout. A total of 106 patients were eligible for alveolar bone grafting in the 4-year cohort studied. One patient elected not to undergo surgery. Each patient had one surgeon carrying out their bone graft. Postoperative occlusal radiographs were available for 82 patients, of which 1 was deemed unreadable. The 81 remaining patient records were anonymized. The radiographs were collated into a single document and scored by two consultant orthodontists, one based in Glasgow and the other based in Dundee. This eliminated any opportunity for collusion. Scoring took place on two separate occasions per observer, 2 weeks apart. Where image quality presented a problem, the contrast and brightness were altered by an individual independent to the scorers to determine the location and quantity of the bone graft. The radiographs were then scored using the Kindelan bone-fill index (Kindelan et al., 1997; Kindelan and Roberts-Harry, 1999). This is a four-grade scale in which grade 1 signifies .75% bony fill, grade 2 is 50% to 75% bony fill, grade 3 is ,50% bony fill, and grade 4 means no complete bony bridge. Grades 1 and 2 were deemed successful, grade 3 represented a partial failure, and grade 4 was considered a total failure. The success of alveolar bone grafting can be determined using a number of other clinical and radiographic measures (Bergland et al., 1986a; Long et al., 1995; Witherow et al., 2002). However, these indices are designed to be used once the permanent maxillary canine has erupted, often a significant time after the initial graft. Where the outcome is poor and additional bone grafting is deemed necessary, the success of this further graft may be jeopardized by this
Paterson et al., SECONDARY ALVEOLAR BONE GRAFTING (CLEFTSIS) 2007–2010
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TABLE 1 Intra- and Interobserver Unweighted Kappa Scores for Observers 1 and 2*
Observer 1 Observer 2
Observer 1
Observer 2
0.831 (0.711–0.951) 0.363 (0.170–0.556)
0.363 (0.170–0.556) 0.587 (0.420–0.753)
delay (Dickinson et al., 2008), unless these indices are modified for earlier use (Williams et al., 2003). The Kindelan bone-fill index (Kindelan et al., 1997) can be applied any time after bony integration has taken place by comparing the preoperative and postoperative anterior occlusal radiograph. Indeed, the cleft canine does not need to be erupted for the Kindelan index to be used and, because radiographic success is determined at an early stage, repeat bone grafting can still be undertaken within the optimal time period. Although there is little evidence to suggest that a good early Kindelan outcome score relates to clinical success (Seike et al., 2012), it remains the most commonly used method of bone graft outcome assessment in the United Kingdom. With the advent of cone beam computed tomography within the CLEFTSiS network, the number of preoperative anterior occlusal radiographs taken has been reduced. Although this prevents direct comparison between pre- and postoperative radiographs, recent work suggests that this may not be critical in determining an outcome score using the Kindelan bone-fill index (Dobbyn et al., 2012). Intraobserver reproducibility of the Kindelan scoring was measured using the kappa statistic (Landis and Koch, 1977). Then, another kappa statistic was calculated to assess interobserver reproducibility. Where disagreement existed on a particular set of radiographs, a third consensus score was discussed and agreed over the telephone. A chi-square test of independence was used to compare the data from 2007 to 2010 with that obtained by McIntyre and Devlin (2010) for 2000 to 2004. The data were categorized as either success (score 1 or 2) or failure (score 3 or 4) of bone graft before undertaking this test. When considering the comparison of outcomes of unilateral and bilateral clefts, it is important to note that the side with the worst score in bilateral cases was used for comparison. When comparing age at time of bone grafting, the patients were categorized either as younger than 11 years or greater than or equal to 11 years. Eleven years was TABLE 2 Kindelan Bone-Fill Index Outcomes Obtained in This Study and Results From McIntyre and Devlin (2010) Grade 1
Grade 2
Grade 3
Grade 4
Total
2000–2004 Number of patients Percentage
36 32
50 44
26 23
2 2
114
2007–2010 (this study) Number of patients Percentage
49 60
31 38
1 1
0 0
81
FIGURE 1 Chart showing percentage of patients allocated each Kindelan bone-fill index score. The white columns depict results from this study (2007 to 2010). Black represents data from McIntyre and Devlin (2010), who reported on 2000 to 2004. Note the large increase in the percentage of patients achieving grade 1 in this study.
chosen as this is the upper limit of the normal age range during which to perform bone grafting. Chi-square tests (or Fisher exact test) were used to determine whether outcomes differed depending on the laterality of the cleft (unilateral/ bilateral), use of presurgical expansion, or age at time of bone grafting. Data regarding the quality of the primary palatal repair performed on each patient were not available. Related to this, no account was taken of the preoperative size of the clefts as radiographic analysis alone could not give sufficiently accurate data. RESULTS Table 1 shows the intra- and interobserver unweighted kappa scores for observers 1 and 2. Interobserver scoring agreement was good, with a kappa of .363. The weighted kappa value for interobserver agreement was .383. Intraobserver reproducibility was better with scores of .831 and .587 (weighted values .835 and .620, respectively). We shall now consider the Kindelan bone-fill index outcomes obtained for the 81 patients included in this study, alongside the results from McIntyre and Devlin (2010). Success is defined as having a Kindelan score of 1 or 2. Overall, for this study (n ¼ 81), bone grafting was successful in 99% of cases (n ¼ 80). This compares with the 76% success rate in the data set from 2000 to 2004. This is a significant improvement (P ¼ .000006). It is also notable that the percentage of total failures has fallen from 2% (n¼ 2) in 2000 to 2004 to 0% in this study. These results are shown in Table 2. Figure 1 helps demonstrate the trend of continued improvement in outcomes. In particular, it is notable that the percentage of patients scoring grade 1 on the Kindelan index has risen from 32% in 2000 to 2004 to 60% in 2007 to 2010.
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TABLE 3
Effect of Laterality of the Cleft, Use of Presurgical Expansion, and Age at Time of Bone Grafting on Outcome* Percentage Success
Unilateral Bilateral Presurgical maxillary expansion No presurgical expansion Age ,11 y Age 11 y
98.5 100 100 98.3 100 95.2
(66 (14 (22 (58 (60 (20
of of of of of of
67) 14) 22) 59) 60) 21)
P Value 1.000* 1.000* .259*
Percentage With Kindelan Score of 1 61.2 57.1 55.4 62.7 63.3 52.4
(41 of 67) (8 of 14) (12 of 22) (37 of 59) (38 of 60) (11 of 21)
P Value .778 .504 .377
* Fisher exact test used.
There was no statistically significant relationship between the laterality of the cleft (P ¼ 1.000), use of presurgical expansion (P ¼ 1.000), or age at time of bone grafting (P ¼ .259) and outcomes. In light of the high success rate seen in this study, it was decided to recategorize the data into those patients having a successful bone graft with Kindelan score 1 and those having worse outcomes. This was done to determine any trends missed by examining only success or failure. The results of this analysis are presented in Table 3, and although the P values are lower, none are significant at P , .05. DISCUSSION This study reports the radiographic outcomes of secondary alveolar bone grafting in Scotland for patients with clefts involving the alveolus (and where the records were available in the EPR) for the years 2007 to 2010. It is notable that the data set presented is incomplete, with 82 of 106 patients having postoperative occlusal radiographs (77%). This is disappointing but is similar to the percentage achieved by Revington et al. (2010) of 70% in their U.K. national audit. Seventy-seven percent represents a significant improvement on the previous report for CLEFTSiS (McIntyre and Devlin, 2010), which found access to postoperative radiographs in only 46%. It is notable that in the CSAG study, there was a similar significant attrition bias, with only 49% of postoperative radiographs being available (CSAG, 1998). The improvements may be due the dissemination of the results from the previous study and the reconfiguration of the service in some areas resulting in postoperative radiographs being taken in a single location. However, the system fundamentally relies on scanning (conventional films) and uploading patient information. Although, in England and Wales, this would routinely be the job of the coordinator, this facility is not available for much of Scotland. It is unknown what effect the missing radiographs would have had; this should be taken into consideration when viewing the results of this investigation. As alluded to earlier, another limitation of this study is the lack of data on the quality of the primary palatal repair or preoperative dimensions of the cleft. As such, these factors are not taken into account when assessing success of secondary bone grafting. The radiologic method used for assessment of the grafts in this cohort was the anterior occlusal radiograph. A
significant number of studies have also used the anterior occlusal radiograph as the standard assessment tool (Hall and Posnick, 1983; Bergland et al., 1986a, 1986b; Enemark et al., 1987; Demas and Sotereanos, 1988; Amanat and Langdon, 1991; Kortebein et al., 1991; Freihofer et al., 1993; LaRossa et al., 1995; Long et al., 1995; Kalaaji et al., 1996; Tan et al., 1996; Kindelan et al., 1997; CSAG, 1998; Montecinos et al., 1998; Denny et al., 1999; Kindelan and Roberts-Harry, 1999; Opitz et al., 1999; Aurouze et al., 2000; Jia and Mars, 2000; Lilja et al., 2000; Mao et al., 2000; Kalaaji et al., 2001; Williams et al., 2001; Jia et al., 2002; Witherow et al., 2002; Schultze-Mosgau et al., 2003; Nightingale et al., 2003; Williams et al., 2003; Clarkson et al., 2005; Trindade et al., 2005; Bayerlein et al., 2006; Jia et al., 2006; Rawashdeh and Nimri, 2007; Matic and Power, 2008; McIntyre and Devlin, 2010; Francis et al., 2013). Within the CLEFTSiS network, attempts have been made to reduce the variability in quality of radiographs produced between centers by a descriptive protocol for radiograph production. Only one radiograph was deemed unsuitable for assessment within the present cohort. This is similar to the work of McIntyre and Devlin (2010), who reported ,1% and Revington et al. (2010) with 1.4%. It also compares favorably with that of the CSAG report (1998), which found 14% to be unsuitable. The index used in the present study was the Kindelan bone-fill index (Kindelan et al., 1997), as that not only allows direct comparison with McIntyre and Devlin (2010) but also was used in the national study by Revington et al. (2010). Examining the weighted kappa statistics for inter- and intraobserver reproducibility revealed the former to be .383 and the latter two as .835 and .620. This suggests a moderate/good level of agreement between the observers used in this study, and the values are similar to those of other studies (Kindelan et al., 1997; Nightingale et al., 2003; Revington et al., 2010; Dobbyn et al., 2012). A 99% success rate (Kindelan grades 1 and 2) represents a significant improvement in comparison with McIntyre and Devlin’s (2010) study into the years 2000 through 2004, which reported a 76% success rate (P , .001). This result is also an improvement on the results from the CSAG (1998) study, although caution should be used with this comparison as they used the modified Bergland Index (Williams et al., 2003). The result also compares favorably with those studies having success rates of .90% (Hall and Posnick, 1983; Bergland et al., 1986a; Demas and Sotereanos, 1988;
Paterson et al., SECONDARY ALVEOLAR BONE GRAFTING (CLEFTSIS) 2007–2010
Kalaaji et al., 1996; Newlands, 2000; Jia et al., 2006) and can be viewed as a performance indicator of continuing improvement in cleft services in Scotland over recent years. Since 2000 to 2004, significant changes in the Scottish alveolar bone graft service have taken place. Previously, there were nine surgeons carrying out alveolar bone graft surgery across Scotland. This was reduced in 2007 to three dedicated surgeons, with surgeons and orthodontists improving the coordination of bone graft delivery through dedicated bone graft clinics and the extraction of any unwanted teeth in the surgical area 3 months in advance of bone graft surgery. These factors may have contributed to an improvement in successful bone grafting outcomes and are in accordance with the finding of improved success rates with high-volume operators (Nightingale et al., 2003) and those with greater experience (Kalaaji et al., 1996). Attempting to examine factors that may affect successful outcomes is difficult with an overall success rate of 99%. However, when considering the results pertaining to unilateral and bilateral clefts, the data suggest that unilateral clefts have better outcomes. This is evidenced by 61.2% of unilateral cases achieving a Kindelan score of 1 compared with only 57.1% of bilateral cases. A similar small percentage difference in outcomes between unilateral and bilateral cases was found by Revington et al. (2010), but as in this study (P ¼ .778), no significant difference was found between the two. This is in accordance with other studies (Bergland et al., 1986b; Long et al., 1995; Kindelan and Roberts-Harry, 1999; Newlands, 2000; McIntyre and Devlin, 2010). Perhaps a larger data set would allow a more significant result to be established. An interesting trend exists for the use of presurgical maxillary expansion. Contrary to McIntyre and Devlin’s (2010) findings, it appears that those patients without maxillary expansion had better outcomes (62.7% obtaining a score of 1 compared with 55.4% for those undergoing expansion). Again, this apparent trend is not statistically significant. Finally, those patients younger than 11 years at the time of alveolar bone grafting were found to have a 100% success rate (Kindelan score of 1 or 2), whereas those 11 years or older had only 95.2% success. Although this difference was not statistically significant, it replicates the findings of Lee et al. (1995), Kalaaji et al. (1996), CSAG (1998), Kindelan and Roberts-Harry (1999), and Williams et al. (2003), who found increasing age a predictor of poorer outcome. CONCLUSIONS Scottish secondary alveolar bone graft outcomes improved during 2007 to 2010 in comparison with the 2000 to 2004 results. There were no significant relationships between laterality of the cleft, use of presurgical expansion, or age at time of bone grafting on secondary alveolar bone graft outcomes. Only 77% of the patient cohort studied had
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