Retrospective Review of 99 Patients With Secondary Alveolar Cleft Repair

J Oral Maxillofac Surg 68:1283-1289, 2010

Retrospective Review of 99 Patients With Secondary Alveolar Cleft Repair Lisa L. Miller, DMD, MD,* Daniel Kauffmann, BS,† Dane St. John, DMD, MD,‡ Deli Wang, PhD,§ John H. Grant III, MD,储 and Peter D. Waite, MPH, DDS, MD¶ Purpose: The purpose of the present review was to evaluate the protocol and technique used in a large

population of patients with cleft lip and palate when secondary grafting is performed during the early mixed dentition stage, as determined by eruption of the central incisor. In the United States, most investigators have recommended alveolar grafting at the 9- to 11-year age range or before eruption of the permanent canines. Materials and Methods: An institutional review board-approved chart review of 99 patients undergoing alveolar cleft bone grafting during a 7-year period at a single institution was performed. Data were collected regarding demographics, operative time, length of hospitalization, follow-up time, complications, and additional procedures performed. The cases were divided by patient age into 2 groups: group 1, aged 6 to 8 years (n ⫽ 61); and group 2, aged 9 years and older (n ⫽ 38). Statistical analysis was performed for various comparisons in the study. Results: The average operative time for groups 1 and 2 was 86 and 103 minutes, respectively. The complication rate, length of stay, and follow-up time between the 2 groups was not statistically significant at the P ⫽ .05 significance level. Conclusions: We recommend earlier bone grafting at or before the eruption of the central incisor, rather than delaying until the cuspid tooth root is 25% formed. We believe this will provide better bone support for the dentition, a decreased burden of treatment for the patient, and improved quality of life. Performing the procedure at this time can lead to decreased operative times, with comparable postoperative outcomes. This is a US government work. There are no restrictions on its use. Published by Elsevier Inc on behalf of the American Association of Oral and Maxillofacial Surgeons. J Oral Maxillofac Surg 68:1283-1289, 2010 Surgery involving the patient with cleft lip and palate must balance the need for a functional and esthetic outcome against the potential increased restriction of normal maxillary growth and development. The timing of the alveolar bone graft is often quite variable *Former Resident, Department of Oral and Maxillofacial Surgery, University of Alabama at Birmingham, Birmingham, AL; Currently, Private Practice, Oral and Facial Surgery of Alabama, Birmingham, AL. †Public Health Student, University of Alabama at Birmingham, Birmingham, AL. ‡Former Resident, Department of Oral and Maxillofacial Surgery, University of Alabama at Birmingham, Birmingham, AL; Currently, Staff Surgeon, Department of Oral and Maxillofacial Surgery, United States Army, El Paso, TX. §Assistant Professor, Northwestern University Feinberg School of Medicine, Children’s Memorial Hospital, Chicago, IL. 储Associate Professor, Department of Surgery, Division of Plastic and Reconstructive Surgery, University of Alabama at Birmingham, Birmingham, AL.

but is most often related to the development of the permanent maxillary canine root. The primary goals for reconstruction of the secondary alveolar cleft deformity include 1) closing the oral nasal fistula, 2) providing bone for eruption and support of anterior ¶Professor and Chairman, Department of Oral and Maxillofacial Surgery, University of Alabama at Birmingham, Birmingham, AL. Address correspondence and reprint requests to Dr Waite: Department of Oral and Maxillofacial Surgery, SDB 419, 1530 Third Avenue South, Birmingham, AL 35294-0007; e-mail: pwaite@uab. edu This is a US government work. There are no restrictions on its use. Published by Elsevier Inc on behalf of the American Association of Oral and Maxillofacial Surgeons 0278-2391/6806-0011$36.00/0 doi:10.1016/j.joms.2009.09.106

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1284 teeth, 3) unification of the maxillary segments, 4) support to the base of the nose, and 5) normal ridge form for prosthetic reconstruction, including osseointegrated implants.1-3 The criteria used for evaluating long-term success are 1) restoration of the alveolar bone height and width, 2) eruption and periodontal health of the permanent incisor and canine teeth, 3) adequate attached gingiva adjacent to the cleft, and 4) successful placement of implant-supported restorations.1,4-7 Although other sources of autogenous bone have been attempted, iliac cancellous bone is most commonly used owing to its abundance, ease of access, and superior outcomes.8-11 The concerns associated with iliac crest harvesting have focused primarily on the possible effects on growth, gait disturbances, hematoma, and donor site morbidity. Most of these complications can be minimized with a careful surgical technique that uses a limited incision, minimal stripping of the musculature on the crest, meticulous hemostasis, carefully layered wound closure with reapproximation of the cartilage cap, adequate postoperative pain control, and early ambulation. The timing of alveolar bone grafting has traditionally been divided into primary and secondary stages with primary grafting performed after lip repair but before repair of the palate.12,13 Secondary grafting has been defined as early secondary at 2 to 5 years, early mixed dentition at 6 to 8 years, late mixed dentition at 9 to 12 years, and late secondary grafting if done after the age of 13.14 In the United States, most investigators recommend alveolar grafting at the 9- to 11-year age range or before eruption of the permanent canines. Numerous studies have demonstrated greater success rates when grafting before canine eruption compared with delayed grafting.15-23 Earlier grafting has been advocated when the lateral or central incisor is developing in an attempt to prevent eruption into a residual cleft, which could jeopardize the health of the tooth. Often, waiting for canine root development jeopardizes the periodontal support of the central and lateral incisors, which collapse into the cleft.1 When one root surface of the incisor has become exposed, bone reattachment is nearly impossible to achieve.16 At the University of Alabama at Birmingham, we have preferred to reconstruct the alveolar defect using iliac bone, timing the surgery according to maxillary central incisor development. If possible, we elect to perform grafting in these patients just before the central incisor begins eruption, which is well before 50% of the cuspid root has developed. We believe that timing of alveolar grafting should be determined by the developing dentition rather than chronologic age. Thus, we prefer that patients undergo evaluation before exfoliation of the primary central incisors. The purpose of the present study was to evaluate the

RETROSPECTIVE REVIEW OF ALVEOLAR CLEFT REPAIR

protocol, technique, and results for early alveolar cleft bone grafting at a single institution.

Materials and Methods The present retrospective review of 99 patients undergoing alveolar cleft bone grafting from 2000 to 2008 was approved by our institutional review board. The information included relevant hospital records, operative reports, clinic notes, and films. The data extracted included demographic information, perioperative factors, complication rates, and outcome data of the protocol used at our institution. Of the 99 patients, 71 had appropriate imaging available, and data were collected regarding the presence or absence of the permanent incisors. The retained primary dentition adjacent to the cleft were extracted at least 4 weeks before bone grafting to allow for adequate healing of the keratinized mucosa. This improved the gingival flap and facilitated a watertight closure over the cleft site during repair (Fig 1). A horizontal advancement flap of keratinized tissue provides better periodontal support around the incisors and canine teeth than a vestibular finger flap. The pediatric technique used to harvest the anterior iliac crest bone graft was as follows. A minimal skin incision 2 to 3 cm long was placed 1 cm lateral to the iliac crest posterior to the anterior superior iliac spine (Fig 2). The incision was carried down through the skin, subcutaneous tissue, and fascia to the cartilaginous cap overlying the crest. Meticulous attention was taken to perform minimal stripping of the musculature on the crest. The perichondrium was not

FIGURE 1. Healed mucosa after extraction of primary dentition adjacent to cleft. Miller et al. Retrospective Review of Alveolar Cleft Repair. J Oral Maxillofac Surg 2010.

MILLER ET AL

FIGURE 2. Conservative 2-cm incision for iliac crest bone graft harvest. Miller et al. Retrospective Review of Alveolar Cleft Repair. J Oral Maxillofac Surg 2010.

elevated, and the cartilage was not detached. An osteotome was then used to divide the cartilage along the iliac crest parallel to the long axis of the ridge. Two perpendicular horizontal cuts were made with an osteotome, allowing the cartilage to be split open, gaining access to the cancellous bone. The medial portion of the cartilage cap and periosteum were raised in a continuous layer for exposure and access to the cancellous bone of the ilium. A malleable retractor was placed into the subperiosteal plane to retract and protect the peritoneum against potential

FIGURE 3. Harvesting cancellous graft from anterior iliac crest. Miller et al. Retrospective Review of Alveolar Cleft Repair. J Oral Maxillofac Surg 2010.

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FIGURE 4. Closure of incision and subcutaneous local anesthetic pump in place. Miller et al. Retrospective Review of Alveolar Cleft Repair. J Oral Maxillofac Surg 2010.

perforation through the medial cortex. Curettes were used to harvest the desired amount of cancellous bone (Fig 3). The surgical site was copiously irrigated, and any potential sources of active bleeding addressed. Microfibrillar collagen was placed into the marrow cavity to aid in hemostasis. The cartilaginous cap was reapproximated with 2-0 polyglactin suture, and the wound was closed in a layered fashion. A local anesthetic subcutaneous infusion pump providing 0.25% Marcaine was placed into the wound on the medial aspect of the iliac bone (Fig 4). No drain or pressure dressing was used. Repair of the alveolar cleft began with infiltration of 1% lidocaine with 1:100,000 epinephrine on the buccal and palatal aspect of the anterior maxilla. Two full-thickness mucoperiosteal advancement flaps were initially developed along the gingival sulcus on the labial side extending into the cleft. An incision was made further into the cleft, separating the nasal mucosa from the gingiva. A similar incision was performed on the palate. Careful flap elevation began with a sharp periosteal elevator along the labial surface of the alveolus identifying the piriform aperture. The nasal mucosa was elevated off the lateral wall of the nose and separated from the oral mucosa. The buccal flaps were elevated above the piriform rim. The nasal mucosa was reflected into the nose and the periosteum out of the cleft so that new bone could be grafted directly onto the bone. Next, 2-0 silk sutures were placed at the gingival papilla of the tissue adjacent to the cleft to facilitate advancement during closure and prevent tissue maceration. A transition was created, separating the nasal and oral mucosa, so

1286 the bone had proper containment. The bone graft was contained by the nasal floor mucosa, the palatal mucosa, and the labial flap. The labial flap was generously undermined just below the mucosa up into the lip, incising the periosteum so the flap was closed without tethering the vestibule. Closure of the nasal floor mucosa was performed in a simple interrupted manner with 4-0 polyglactin suture on a J needle. The bone graft was tightly packed into the alveolar defect, and a layer of demineralized bone matrix was placed over the graft to aid in cohesion of the cancellous bone (Fig 5). Keratinized gingival mucosa was advanced from the posterior to provide healthy future periodontium (Fig 6). The oral mucosa closure was tension free with 4-0 polyglactin suture in an interrupted manner with reapproximation of the papilla. A horizontal mattress suture was placed over the alveolar ridge where all the incisions come together. The patients were admitted for 24-hour observation and discharged the following day after removal of the anesthetic pump. The patients were instructed to eat a soft diet for 1 week. The activity restrictions included no kicking balls, lifting weights, school physical education, or swimming. Bathing was allowed after 24 hours. Patients could resume normal activities within 2 weeks. Figure 7 shows the final ridge formation 2 years after grafting. We used descriptive statistics such as the mean, median, and range to describe continuous variables, including age and operative time, and used proportions to describe categorical variables such as gender, ethnicity, complications, and so forth. A 2-sample t

RETROSPECTIVE REVIEW OF ALVEOLAR CLEFT REPAIR

FIGURE 6. Horizontal advancement and closure over bone graft. Miller et al. Retrospective Review of Alveolar Cleft Repair. J Oral Maxillofac Surg 2010.

test was conducted to compare difference in age, operative time, and length of hospital stay. A ␹2 test or Fisher’s exact test was used to study the associations for the categorical variables of interest. McNemar’s test was used to test associations of different missing teeth among patients in the study. Cochran-MantelHaenszel test was used to study associations between different teeth and their missing status after controlling the stratum factor such as the cleft side of missing teeth. Because of the exploratory property of the

FIGURE 5. Bone graft packed into alveolar cleft defect. Silk sutures identified and protected delicate gingival papilla.

FIGURE 7. Final ridge form approximately 2 years after grafting to left alveolus.

Miller et al. Retrospective Review of Alveolar Cleft Repair. J Oral Maxillofac Surg 2010.

Miller et al. Retrospective Review of Alveolar Cleft Repair. J Oral Maxillofac Surg 2010.

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MILLER ET AL

Surgery Time

Pt Age Distribution

100

103.1

105 100

61 50

38

Group 1

95

Group 2

85

Group 1

80

Group 2

0

86.2

90

75 Time (mins)

FIGURE 8. Patient distribution. Miller et al. Retrospective Review of Alveolar Cleft Repair. J Oral Maxillofac Surg 2010.

FIGURE 10. Operative time stratified by group. Miller et al. Retrospective Review of Alveolar Cleft Repair. J Oral Maxillofac Surg 2010.

present study, the resulting P values were not adjusted for multiple comparisons. We used P ⫽ .05 as the statistically significant level.

Results Group 1 included 61 patients who underwent grafting before complete eruption of the permanent central incisors, and group 2 included 38 patients who had undergoing grafting later (Fig 8). The mean age of all patients was 8.8 years (range, 6 to 37). Of the 91 patients, 62 were males and 37 were females; the demographic breakdown was 75 white, 11 black, 4 Asian, and 1 Russian. Of the 91 patients, 32 had a bilateral cleft defect, 43 had a left cleft defect, and 24 had a right cleft defect (Fig 9). The operative time ranged from 47 to 191 minutes. The average operative time for groups 1 and 2 was 86.2 and 103.1 minutes, respectively (Fig 10). The average length of stay for all patients was 1.06 days (range, 1 to 2). All patients were seen postoperatively and the length of follow-up varied from 1 week to 52 months. The postoperative complications for the 2 groups were compared. Overall, only minor complications were associated with this procedure, and no statisti-

cally significant differences were found between the 2 groups. In group 1, 9.8% of patients developed complications, including pyogenic granuloma in 2, residual fistula in 2, residual alveolar defect without fistula in 1, and hip wound dehiscence in 1. In group 2, 13.2% of patients developed minor postoperative complications, including residual fistula in 3, residual alveolar cleft defect without fistula in 1, gingival hyperplasia in 1, and fibroma in 1. No statistically significant differences (P ⬍ .05) were found between groups 1 and 2 in gender, ethnicity, cleft type, hospital stay, length of follow-up, or complication rate. A statistically significant difference was found for the operative time between the 2 groups (P ⫽ .0025). The operative time was reduced approximately 17 minutes in group 1 compared with group 2. The incisor teeth (central and lateral incisors) were noted in 71 patients. In our cleft population, it was found that the incidence of the right lateral incisor missing was statistically significant (P ⫽ .0018), regardless of the type of cleft (left, right, or bilateral). Also, when looking at the probability of missing multiple teeth, the lateral incisors were more likely to be missing than the centrals; this difference was also statistically significant (P ⫽ .0025).

Cleft Type

Discussion

60 40 20

43 24

32

Le ft Right Bila te ra l

0 FIGURE 9. Distribution of cleft type. Miller et al. Retrospective Review of Alveolar Cleft Repair. J Oral Maxillofac Surg 2010.

The factors that contribute to the timing of alveolar cleft grafting, such as chronologic and dental age, vary. However, the state of the developing dentition should be the primary factor to assist in making this decision. Performing the graft before eruption of the permanent canines is generally the latest time to provide optimal outcomes. It would be preferable to perform the graft before eruption of the central or lateral incisors to increase the retention and gingival health of these teeth. Therefore, it is ideal for the oral and maxillofacial surgeon to evaluate the patient and

1288 radiographs before the mixed dentition stage to appropriately determine the timing of surgery. The most significant change we established at our institution during the past several years has been the evaluation for alveolar grafting before exfoliation of the primary central incisors.24 This has decreased the number of cases of late secondary grafting and increased the number of cases performed during the early mixed dentition stage. If the permanent incisors are missing, surgery should be deferred to allow for growth of the maxilla and root development of the canine. It is important to stress that the timing of surgery is dependent on the patient’s dental development, not their chronologic age. The results from our large patient population has demonstrated that it is preferable to perform the graft of the alveolus at an earlier developmental stage. The morbidity and operative time associated with earlier grafting is less. We have not seen any long-term adverse outcomes associated with the harvesting of the anterior iliac crest in the younger patient. The patients have tolerated the procedure well and are typically back to regular activities sooner. We believe younger patients have a quicker convalescence. None of the patients in our study population had any longterm gait disturbances related to graft harvest, consistent with the findings from other publications.25,26 Most postoperative pain after iliac crest bone grafting is thought to be secondary to stripping of the periosteal and muscular attachments that are painful during ambulation.8 Our surgical technique is conservative, and we do not perform aggressive dissection of the periosteum or muscular attachments on the ilium. For several years, we have used a local anesthetic infusion pump to provide 1 mL/hour of local anesthetic for 24 hours postoperatively. We believe this helps decrease postoperative pain, decreasing the need for narcotics, and providing earlier ambulation and discharge from the hospital. We are performing a randomized trial to further elucidate the utility of this device. A significant difference between the 2 groups was the decreased operative time in the younger patients. This can be attributed to the ease of iliac graft harvest, the simple access to the alveolus without malpositioned teeth, and the decreased cleft size in the younger patient. A shorter operative/anesthesia time is also safer for the patients. In the current economic environment, in which reduced cost measures are needed, it is beneficial to perform a procedure more efficiently with a decreased operative time. By following our protocol, we believe that the permanent dentition will have improved bony support and periodontal health. By extracting the deciduous teeth adjacent to the cleft 4 weeks before grafting, a full-thickness flap of keratinized mucosa can be

RETROSPECTIVE REVIEW OF ALVEOLAR CLEFT REPAIR

developed and facilitate primary closure over the cleft defect. This prevents additional loss of vestibular height and maintains keratinized tissue around the erupting incisors. The incisors will help stimulate the alveolar and graft bone growth while erupting into the oral cavity.1 One limitation of our review was that we did not have definitive information regarding the long-term outcome and development of the anterior incisors in our patients. This will take many years and closer dental follow-up. It is common for a permanent lateral incisor to be missing on the cleft side.27 When studying the tooth data available, it was interesting that our findings revealed a statistically significant difference for the right lateral incisor to be missing, regardless of the side of the cleft, and not the left lateral incisor. This might have resulted from a sampling error owing to the small sample size in this subset of patients. We plan to continue following this same subset of 99 patients with alveolar cleft and re-examine them in the future. We will then review our data to assist in determining whether early grafting improves retention of the anterior incisors and/or replacement and whether any effect is present on anteroposterior growth of the maxilla. The patient with cleft lip and palate has a complex skeletal deformity that usually requires multiple procedures, starting in infancy, and continuing through puberty, to help restore normal esthetics and function. We believe that by grafting earlier, restoring the maxillary arch form, and providing bone for anterior incisor eruption, the overall treatment burden might be decreased, including less surgical and orthodontic intervention.

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MILLER ET AL 9. Kortebein MJ, Nelson CL, Sadove AM: Retrospective analysis of 135 secondary alveolar cleft grafts using iliac or calvarial bone. J Oral Maxillofac Surg 49:493, 1991 10. Sadove MA, Nelson CL, Eppley BL, et al: Evaluation of calvarial and iliac donor sites in alveolar cleft grafting. Cleft Palate J 27:225, 1990 11. Kraut RA: The use of allogenic bone for alveolar cleft grafting. Oral Surg Oral Med Oral Pathol 64:278, 1987 12. Eppley BL: Alveolar cleft bone grafting (part I): Primary bone grafting. J Oral Maxillofac Surg 54:74, 1996 13. Rosenstein SW: Early bone grafting of alveolar cleft deformities. J Oral Maxillofac Surg 61:1078, 2003 14. Kazemi A, Stearns JW, Fonseca RJ: Secondary grafting in the alveolar cleft patient. Oral Maxillofac Surg Clin North Am 14:477, 2002 15. Dempf R, Teltzrow T, Kramer FJ, et al: Alveolar bone grafting in patients with complete clefts: A comparative study between secondary and tertiary bone grafting. Cleft Palate Craniofac J 39:18, 2002 16. Turvey TA, Vig K, Moriarty J, et al: Delayed bone grafting in the cleft maxilla and palate: A retrospective multidisciplinary analysis. Am J Orthod 86:244, 1984 17. Hall HD, Posnick JC: Early results of secondary bone grafts in 106 alveolar clefts. J Oral Maxillofac Surg 41:289, 1983 18. Troxell JB, Fonseca RJ, Osbon DB: A retrospective study of alveolar cleft grafting. J Oral Maxillofac Surg 40:721, 1982 19. Tan AES, Brogan WF, McComb HK, et al: Secondary alveolar bone grafting—Five year periodontal and radiographic evalua-

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23. 24. 25. 26.

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