The Journal of Craniofacial Surgery • Volume 25, Number 4, July 2014

Brief Clinical Studies

manner despite being a benign tumor, given their potential to lead to complications such as compressive neuropathy of the affected peripheral nerves. It is important to note, however, that complete excision of the lesion also carries the risk for severe complications such as sensory and motor dysfunction. Thus, a more conservative management including decompression may be warranted in patients with LFH. Although LFH typically involves peripheral nerves of the upper extremities, several cases have been reported, which involved the peroneal nerve of the ankle or foot, the sciatic nerve, the cervical nerve plexus of neck, and even the distal branch of a cranial nerve. Hence, a careful diagnostic workup and development of an appropriate surgical approach are recommended, even for benignappearing lesions. To the best of our knowledge, this is the second case report of LFH arising along a cranial nerve. The minimal neural degeneration observed in this case suggests that this may have been an early lesion involving small branches of the cranial nerve. Additional case reports will be helpful to better characterize the pathogenic mechanisms of this disease. In summary, LFHs are extremely rare benign tumorous condition mostly developing along the branches of peripheral nerves. Because of the complexity of the anatomic relationship with nerve structure, an accurate diagnosis and proper treatment, such as partial decompression, should be carefully made to avoid irreversible neurologic complications.

REFERENCES 1. Razzaghi A, Anastakis DJ. Lipofibromatous hamartoma: review of early diagnosis and treatment. Can J Surg 2005;48:394–399 2. Weiss SW, Goldblum JR, Enzinger FM. Enzinger and Weiss’s soft tissue tumors. 4th ed. St Louis,: Mosby, 2001 3. Fandridis EM, Kiriako AS, Spyridonos SG, et al. Lipomatosis of the sciatic nerve: report of a case and review of the literature. Microsurgery 2009;29:66–71 4. Hirakawa E, Miki H, Kobayashi S, et al. Lipofibromatous hamartoma of nerve in the foot. Acta Pathol Jpn 1993;43:265–267 5. Berti E, Roncaroli F. Fibrolipomatous hamartoma of a cranial nerve. Histopathology 1994;24:391–392

Stabilization of Premaxilla Repositioned During Secondary Bone Grafting in Complete Bilateral Cleft Lip and Palate Patients Hossein Behnia, DMD, MS,* Abolhasan Mesgarzadeh, DMD, MS,† Azita Tehranchi, DDS, MS,‡ Golnaz Morad, DDS,‡ Sahand Samieerad, DMD, MS,§ Farnaz Younessian, DDS, MS‡ Abstract: Secondary bone grafting simultaneous to premaxillary repositioning is a well-recognized surgical procedure for the management of bilateral cleft lip and palate patients. Proper stabilization of the repositioned premaxilla is considered as a key factor for the success of secondary bone grafting because the mobility of the premaxillary segment jeopardizes graft integration. This case series reports a reliable method of premaxillary stabilization that incorporated the intrasurgical application of resin bone cement to cover and reinforce the arch bars

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or orthodontic brackets applied on the maxillary teeth. Occlusal loads were reduced by application of posterior bite blocks on the mandibular teeth. The stabilization method was performed on 7 patients (5 women and 2 men) with a mean age of 12.4 years. During postsurgery followups, the repositioned premaxillary segments did not show mobility in any of the patients. The palatal fistulae were completely closed. Panoramic radiographies taken 2 months after surgery demonstrated acceptable graft integration. The patients have now been followed up to 5 years. No evidence of relapse has been observed. This technique seemed to be undemanding, included minimal laboratory procedure, and maintained the labial mucosa overlying the repositioned segment intact. Key Words: Bilateral cleft lip and palate, premaxillary retropositioning, secondary bone grafting, maxillary stabilization

S

econdary bone grafting is a well-recognized procedure for the management of bilateral cleft lip and palate (BCLP) patients. The treatment is primarily aimed to unite the maxillary segments, close the oronasal fistula, and provide adequate volume of bone for tooth eruption.1 Protrusion of the premaxillary segment, a common feature in BCLP patients, might however complicate bone grafting.2 The condition often necessitates surgical repositioning of the protrusive premaxilla to facilitate bone grafting and optimize the final treatment outcomes.3,4 The appropriate timing of premaxilla repositioning in relation to the bone grafting surgery is a matter of controversy. Although a 1-stage procedure including simultaneous maxillary repositioning and bone grafting is indicated to be highly techniquedemanding5, it seems to be the preferred approach among several surgeons.2,3,6 Proper stabilization of the retropositioned premaxilla is considered as a key factor for the success of secondary bone grafting because the mobility of the premaxillary segment endangers the blood supply and therefore jeopardizes graft integration.3 Internal fixation of the maxillary bone segment with miniplates and bone screws provides rigid stability. Yet, it poses detrimental effects on blood supply and tooth bacteria.2,7,8 This problem is also of concern when orthodontic arch wires and surgical screws are used for fixation of the repositioned segments.9 The most widely used method of premaxilla stabilization includes occlusal splints, generally fabricated before the surgery. Occlusal splints might be held in place by means of orthodontic wires with or without bone screws2–5,8 or cements.7,10 This method has the advantage of reducing occlusal forces exerted on the From the *Dentofacial Deformities Research Center, Research Institute of Dental Sciences, Department of Oral and Maxillofacial Surgery, School of Dentistry, Shahid Beheshti University of Medical Sciences, Tehran, Iran; †Iranian Association of Implantology, Tehran, Iran; ‡Dental Research Center, Research Institute of Dental Sciences, Department of Orthodontics, School of Dentistry, Shahid Beheshti University of Medical Sciences, Tehran, Iran; §Oral and Dental Disease Research Center, Department of Oral and Maxillofacial Surgery, School of Dentistry, Kerman University of Medical Sciences, Kerman, Iran. Received June 20, 2013. Accepted for publication February 8, 2014. Address correspondence and reprint requests to Hossein Behnia, DMD, MS, Department of Oral and Maxillofacial Surgery, Faculty of Dentistry, Shahid Beheshti University of Medical Science, Daneshjou Boulevard, Evin, Tehran, Iran 19839; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000865

© 2014 Mutaz B. Habal, MD

Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.

The Journal of Craniofacial Surgery • Volume 25, Number 4, July 2014

Brief Clinical Studies

FIGURE 3. Orthodontic brackets/arch bars were further reinforced by methyl methacrylate bone cement.

patients, with a mean age of 12 years and 4 months (range, 10–15 y) at the time of surgery. All of the surgical procedures were performed by the senior author (H.B.). Patients with any craniofacial syndrome or previous bony surgical interventions were not considered.

Presurgical Orthodontic Therapy All patients had unerupted or malpositioned canines on both sides (Figs. 1A–H). In cases of canine impaction, surgeries were scheduled for the time when two thirds of the canine roots were formed. Presurgical orthodontic expansion of the maxilla was performed in all patients by means of quad-helix devices to eliminate the posterior crossbite. This preoperative expansion widened the existing oronasal fistulae, which improve the surgical access to the cleft defect. Moreover, any tooth rotation and malpositioning in permanent dentitions was corrected until tooth prematurities were removed and acceptable arch coordination was achieved. Position of the teeth adjacent to the cleft was thoroughly evaluated via cone beam computed tomography scans. Whenever, a risk of root displacement into the cleft defect existed, the correction of tooth position and angulation was postponed to postsurgical orthodontic phase.

Surgical Method

Seven BCLP patients underwent premaxilla repositioning and simultaneous bilateral bone grafting with autogenous iliac cancellous bone from 2005 to 2011. The subjects include 5 female and 2 male

A stab incision was made over the vomer bone to approach the vomerine-premaxillary spur. The premaxillary segment was cut free from its attachment to the vomer bone using an osteotome. Mobilization of the premaxillary segment provided adequate visibility for closure of the nasal mucosa (Fig. 2). The mobilized premaxilla was then repositioned to the optimal position, which immediately improves the sagittal and vertical intermaxillary relations. Palatal closure was achieved by suturing the anterior edge of the palatal flap to the posterior edge of the premaxillary mucosa. No approximation was performed. Whenever the available soft tissue was not sufficient for complete palatal closure, finger flaps from the buccal mucosa were used. Concomitant to premaxillary repositioning, a second surgical team harvested spongious bone chips from the iliac crest. After maxillary stabilization, harvested bone chips were packed into the remaining defects on the buccal side (Fig. 3). The procedure was terminated by closure of the buccal flap (4–0 vicryl sutures; Ethicon, Somerville, NJ). Orthodontic brackets and wires fixed on the maxillary posterior teeth and the teeth present in the premaxillary segment were further reinforced by methyl methacrylate bone cement. Patients in whom presurgical orthodontic treatment did not include orthodontic brackets received arch bars during surgery, which were then covered by bone cement for additional reinforcement (Fig. 4).

FIGURE 2. The premaxillary segment was detached from the vomer bone and mobilized for retropositioning.

FIGURE 4. Harvested bone chips were packed into the remaining defects on the buccal side.

FIGURE 1. A–H, Extraoral and intraoral photographs of a 6-year-old boy with repaired BCLP.

premaxillary segment. Yet, it requires presurgical fabrication and impedes efficient mastication and hygiene control.10 Aiming to optimize the premaxilla stabilization technique, this case series reports a reliable method that incorporated the intrasurgical application of resin cement to cover the orthodontic brackets or arch bars applied on the maxillary teeth and a posterior bite block on the mandibular teeth. This stabilization method, eliminating the prefabrication of an occlusal splint, intended to improve function, comfort, and patient compliance during the fixation period while reducing the pressure of occlusal loads.

MATERIALS AND METHODS

© 2014 Mutaz B. Habal, MD

Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.

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The Journal of Craniofacial Surgery • Volume 25, Number 4, July 2014

Brief Clinical Studies

FIGURE 5. Composite blocks etched on the maxillary posterior teeth for the removal of occlusal loads on the grafted area in a 15-year-old male patient. Impacted canines erupted into the grafted area.

Postoperative antibiotic prescription included cephazoline (1 g every 6 h) and gentamycin (500 mg every 8 h). Patients were instructed to mouth rinse with chlorhexidine mouthwash and to use soft diet for 2 to 3 weeks after surgery. To avoid occlusal trauma to the bone grafts, at surgery, the patients received previously prepared posterior bite plates, inserted at the lower arch. In cases of poor patient compliance, instead of posterior bite plates, the upper posterior teeth were etched on the occlusal surface on which composite blocks were placed (Fig. 5). During the 2-month period for integration of the grafts, the occlusion of the patients were monitored closely, and posterior bite plates or composite blocks were gradually removed from the occlusal surface to avoid any possible tooth intrusion. The applied bone cement was removed after approximately 4 months by means of rotary instrument and surgical burs, without any significant complication.

FIGURE 7. Intraoral view at the time of (A) presurgical orthodontic treatment and (B) after arch coordination. Missing lateral incisors are temporarily substituted via the removable appliance until definite treatment with dental implants.

No evidence of premaxilla mobility was observed, and all grafts were integrated successfully. For promising outcomes to be obtained with bone grafting, proper stabilization of the premaxilla and reduction of occlusal loads on the retropositioned premaxillary segment and bone grafts are imperatives.2,3 In the presented method, while the premaxillary segment was stabilized using bone cement over orthodontic brackets/arch bars, patients were provided with posterior bite blocks in the mandible or composite blocks on the maxillary posterior teeth for the elimination of occlusal contacts. Stability of the premaxillary segment is occasionally provided via application of miniplates, arch wires, and bone screws, although not generally suggested because of the negative impact on the premaxillary segment’s blood supply.2,7–9 On the other hand, most commonly advocated methods of premaxillary

Postsurgical Orthodontic Therapy At this time, the orthodontic treatment was continued to achieve arch coordination. With the presence of bone graft filling the cleft defect, any tooth malpositioning adjacent to the cleft was corrected. Impacted canines were left to erupt spontaneously into the bone graft (Fig. 5). These tooth movements also provided stability for the bone graft. Missing teeth were either replaced via implant or prosthesis, depending on the individual’s comprehensive orthodontic treatment plan regimen.

RESULTS In all patients, the overall outcome of surgery was deemed successful. The repositioned premaxillary segments did not show mobility as a graft failure. The palatal fistulae showed complete closure. Panoramic radiographies taken 2 months after surgery demonstrated acceptable graft integration. As shown in Figures 6A and B, graft consolidation continued during the postoperative orthodontic treatment. The patients have now been followed up to 5 years. Arch coordination has been completed in 3 patients (Figs. 7A, B). No evidence of relapse has been observed so far (Figs. 8A–H). The treatment is still ongoing in patients in whom correction of interarch relationship was required via further surgeries.

DISCUSSION This study introduced a reliable technique for stabilization of the repositioned premaxilla during bone graft surgery in BCLP patients.

FIGURE 6. Comparison of (A) the preoperative and (B) 1-year postoperative cone beam computed tomography scans of a 14-year-old male patient demonstrate graft consolidation.

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FIGURE 8. A–H, Extraoral and intraoral photographs of the same patient after 2 years of secondary graft surgery.

© 2014 Mutaz B. Habal, MD

Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.

The Journal of Craniofacial Surgery • Volume 25, Number 4, July 2014

stabilization include occlusal splints for the simultaneous reduction of occlusal loads.2–5 Occlusal splints are generally fixed in place by means of orthodontic wires and bone screws. Penetration of labial mucosa and periosteum overlying the repositioned segment by bone screws can hamper blood supply to the repositioned bone segment leading to improper healing and necrosis. Moreover, it might damage the unerupted teeth within the anterior segment or interfere with their eruption.8 Carlini et al2 reported 50 BCLP patients in whom the premaxillary segment was retropositioned simultaneous to bone grafting and was stabilized using an acrylic splint, a bone screw, and orthodontic wires. Premaxilla necrosis was observed in 2 patients and was assumed to be due to poor hygiene and contamination. The bone screw inserted in between the 2 central incisors might also be a contributing factor of failure. Moreover, bite splints should be fabricated before operation, necessitating a cast model surgery stage. This process does not allow for intrasurgical adjustments should the premaxilla had to be positioned different to that indicated during cast surgery. A number of protocols have been suggested with occlusal splints, which eliminate the need for wires and screws. Oyama et al10 used glass ionomer cement for the fixation of prefabricated splints on the maxillary teeth in 6 patients. The authors reported sufficient premaxilla stability. Miyasaka et al7 also fabricated a removable device consisting of a palatal bite plate and an anterior tray, which could be filled with soft resin during surgery, providing stability to the retropositioned premaxilla. Although the authors reported promising results with these methods, both require a splint fabrication stage before surgery. In contrast to routine stabilization procedures, this new technique seems to be a simple noncomplicated modality and less technique sensitive. Thus, it may be a potential alternative approach for proper fixation of the premaxilla while maintaining the labial mucosa overlying the repositioned segment intact. It is well proven that the surgical scar tissue is a major interfering component of the normal growth of the midface in named population, but regarding the disabilities of remained cleft defect, these surgical interventions are not avoidable.11 The possible growth retardation tendency and relapse over time does not hamper the obvious advantages of secondary bone grafting in cleft patients.12 Thus, delaying the timing of the surgery and application of more conservative, not technique-sensitive stabilizing approaches might minimize the inhibitory effect of the surgery on future maxillary growth.5 This study concludes the restricted number of first patients treated by this new method of stabilization of secondary bone graft. Considering the age range of these patients, it is recommended to design a longitudinal study to investigate the possible growth effect of the applied technique on midfacial growth of the samples in sagittal and vertical dimensions. As other studies revealed the possible growth impairment by different premaxilla repositioning procedures combined with bone grafting, the possible growth retardation effect should not be interpreted as a consequence of applied stabilization technique.12 To precisely clarify the effect of this newly presented stabilization technique, the dimensional changes and the rate of orthognathic surgery for maxillary advancement based on objective criterion in the presented cases should be compared with a control group of bilateral cleft patients who underwent routine secondary bone graft and stabilization procedures.

CONCLUSIONS The method of premaxillary stabilization introduced in this study provided adequate premaxilla stability. The technique was undemanding and included minimal laboratory procedure. Moreover, it allowed for proper mastication and maintenance of hygiene.

REFERENCES 1. Guo J, Li C, Zhang Q, et al. Secondary bone grafting for alveolar cleft in children with cleft lip or cleft lip and palate. Cochrane Database Syst Rev 2011;6:CD008050

Brief Clinical Studies

2. Carlini JL, Biron C, Gomes KU, et al. Surgical repositioning of the premaxilla with bone graft in 50 bilateral cleft lip and palate patients. J Oral Maxillofac Surg 2009;67:760–766 3. Aburezq H, Daskalogiannakis J, Forrest C. Management of the prominent premaxilla in bilateral cleft lip and palate. Cleft Palate Craniofac J 2006;43:92–95 4. Scott JK, Webb RM, Flood TR. Premaxillary osteotomy and guided tissue regeneration in secondary bone grafting in children with bilateral cleft lip and palate. Cleft Palate Craniofac J 2007;44:469–475 5. Iino M, Sasaki T, Kochi S, et al. Surgical repositioning of the premaxilla in combination with two-stage alveolar bone grafting in bilateral cleft lip and palate. Cleft Palate Craniofac J 1998;35:304–309 6. Akita S, Hirano A. Usefulness of simultaneous pre-maxillary osteotomy and bone grafting in the bilateral clefts. J Craniofac Surg 2006;17:291–296 7. Miyasaka M, Akamatsu T, Yamazaki A, et al. A new device for fixing the premaxilla with osteotomy in a child with complete bilateral cleft lip and palate in the mixed dentition period—a preliminary case report. Tokai J Exp Clin Med 2008;33:21–27 8. Singh M, Flood T, Mahmood R, et al. Stabilisation of premaxillary segment in bilateral cleft palate during alveolar bone graft. Br J Oral Maxillofac Surg 2012;50:678–679 9. Bohman P, Yamashita DD, Baek SH, et al. Stabilization of an edentulous premaxilla for an alveolar bone graft: case report. Cleft Palate Craniofac J 2004;41:214–217 10. Oyama T, Yoshimura Y, Onoda M, et al. Stabilization of a mobile premaxilla by cementing a bite splint with 1-stage bilateral alveolar bone grafting. J Craniofac Surg 2008;19:1705–1707 11. Gesch D, Kirbschus A. Complete cleft lip and palate without bone grafting and its effect on craniofacial morphology in three planes. Ann Anat 2004;186:531–537 12. Geraedts CT, Borstlap WA, Groenewoud JM, et al. Long-term evaluation of bilateral cleft lip and palate patients after early secondary closure and premaxilla repositioning. Int J Oral Maxillofac Surg 2007;36:788–796

Facial Asymmetry With Enlarged Frontal Sinus and Hyperplasia of the Cranial, Nasal, and Mandible Bones Rie Taguchi, MD, Masanobu Yamashita, MD, Shigehiko Kawakami, MD Abstract: The conditions of facial asymmetry are caused by congenital or acquired diseases, and several unclassifiable syndromes with unknown etiologies exist. In this report, a case of facial asymmetry with enlarged frontal sinus and hyperplasia of the frontal cranial bone and nasal bone is presented. Although the etiology of the facial malformation was clear, it was thought that the cause of the enlarged frontal sinus was related From the Department of Plastic and Reconstructive Surgery, Kanazawa Medical University, Ishikawa, Japan. Received January 16, 2014. Accepted for publication February 8, 2014. Address correspondence and reprint requests to Rie Taguchi, MD, Department of Plastic and Reconstructive Surgery, Kanazawa Medical University, 1-1 Daigaku, Uchinada, Kahoku, Ishikawa 9200293 Japan; E-mail: [email protected] The authors have no funding to disclose. The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000866

© 2014 Mutaz B. Habal, MD

Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.

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Stabilization of premaxilla repositioned during secondary bone grafting in complete bilateral cleft lip and palate patients.

Secondary bone grafting simultaneous to premaxillary repositioning is a well-recognized surgical procedure for the management of bilateral cleft lip a...
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