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noma. The benign but focally atypical cytologic appearance of the cells and the maintenance of the acinar architecture separate necrotizing sialometaplasia from either squamous cell carcinoma or mucoepidermoid carcinoma.14 The lesions undergo spontaneous recovery in 2 to 3 months. It is a painful lesion that requires treatment other than to relieve pain. Necrotizing sialometaplasia usually does not recur. The importance of this clinical entity lies in its clinical and histologic similarity to a malignant lesion because it can cause unnecessary treatment ranging from conservative excision to maxillectomy.15 In conclusion, we report a case of palatal clear cell carcinoma with necrotizing sialometaplasia in the subsequent excision material, a finding that has not been previously reported. FIGURE 4. Adjacent hyperplastic mucosal epithelium with squamous metaplasia of the seromucous glands with preservation of lobular morphology (hematoxylin-eosin, original magnification 40).

fibrillary acidic protein, actin, and vimentin. The other salivary gland neoplasms of the oral cavity having clear cells of the component should be kept in mind for the differential diagnosis. These include pleomorphic adenoma, myoepithelioma, and low-grade mucoepidermoid carcinoma. Specific histopathologic findings of clear cellYrich tumors of the salivary gland and their respective immunostaining patterns will help differentiate HCCC from these tumors.9 Pleomorphic adenoma may have a clear-cell component, but it has bland nuclear features and chondromyxoid stroma. In addition, smooth muscle actin and S100 immunostains, which indicate myoepithelial origin, may be useful in suspected cases.10 Hyalinizing clear cell carcinoma usually consists of a pure population of clear cells, whereas mucoepidermoid carcinoma consists of mucin-containing cells, squamous cells, and intermediate cells.10,11 Another important entity to be kept in mind regarding differential diagnosis is metastatic clear cell renal carcinoma. The usual rich vascularity of renal cell carcinoma showing immunopostivity for vimentin and CD10 are not seen in HCCC.11 Clear cell carcinoma, which occurs almost exclusively in minor salivary glands, is a rare low-grade carcinoma. It is one of the entities included in the latest World Health Organization classification of tumors of the salivary gland. Although wide excision is the most preffered method of treatment in clear cell carcinomas, in some cases, neck dissection and radiotherapy should be performed. Adverse biologic behavior ranges from multiple local recurrences to the nodal or distant metastases.12 Wide local excision was applied to our patient, and necrotizing sialometaplasia, which is also a rare entity, was seen at the subsequent excision biopsy specimen. Necrotizing sialometaplasia most commonly involves the minor salivary glands of the palate. The lesion can be painful or painless and presents as a craterlike ulcer. Involvement of the palate usually appears as an unilateral ulcer on the posterior hard palate or other sites including the maxillary sinus, retromolar site, lower lip, tongue, and buccal mucosa. The frequency is 2 to 3 times more in males than in females.10,13 The most widely accepted theory regarding the development of necrotizing sialometaplasia is the ischemia of the blood vessels, leading to infarction of the gland tissues. The factors believed to lead to ischemia are trauma, surgical applications, intubation, administration of local anesthetics, smoking, alcohol and cocaine use, and infection.13 In our case, a local anesthetic, the use of expired or long-term use of anesthetic drugs also seems to be a possible etiologic factor. Early microscopic findings are coagulation necrosis of the salivary gland acini, an inflammatory response, and pseudoepitheliomatous hyperplasia, whereas squamous metaplasia of ducts and reactive fibrosis can be seen in late lesions. Generally, the findings are vascular proliferation, prominent inflammatory infiltrate, and partial necrosis of salivary glands, associated with regeneration and squamous metaplasia of the adjacent duct and acini. Two of the most important differential diagnosis include squamous cell carcinoma and mucoepidermoid carci-

REFERENCES 1. Manoharan M, Othman NH, SamsudNn AR. Hyalinizing clear cell carcinoma of minor salivary gland: case report. Braz Dent J 2002;13:66Y69 2. Michal M, Skalova A, Simpson RH. Clear cell malignant myoepithelioma of the salivary glands. Histopathology 1996;28:309Y315 3. Kauzman A, Tabet JC, Stiharu TI. Hyalinizing clear cell carcinoma: a case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2011;112:26Y34 4. Berho M, Huvos AG. Central hyalinizing clear cell carcinoma of the mandible and the maxilla. Clinicopathologic study of two cases with an analysis of the literature. Hum Path 1999;30:101Y105 5. Solar AA, Schmidt BL, Jordan RC. Hyalinizing clear cell carcinoma: case series and comprehensive review of the literature. Cancer 2009;115:75Y83 6. Fechner RE. Necrotizing sialometaplasia: a source of confusion with carcinoma of the palate. Am J Clin Pathol 1977;67:315Y317 7. Pagni F, Za`rate AF, Urbanski SJ. Necrotizing sialometaplasia of bronchial mucosa. Int J Surg Pathol 2010;18:648 8. Krishna S, Ramnarayan BK. Necrotizing sialometaplasia of palate: a case report. Imaging Sci Dent 2011;41:35Y38 9. Masilamani S, Rao S, Chirakkal P. Hyalinising clear cell carcinoma of the base of tongue: a distinct and rare entity. Indian J Pathol Microbiol 2011;54:167Y169 10. Wenig BM. Atlas of Head and Neck Pathology. 2nd ed. New York, NY: Saunders Elsevier, 2008 11. Rezende RB, Drachenberg CB, Kumar D, et al. Differential diagnosis between monomorphic clear cell adenocarcinoma of salivary glands and renal (clear) cell carcinoma. Am J Surg Pathol 1999;23: 1532Y1538 12. Ponniah I, SureshKumar P, Karunakaran K. Clear cell carcinoma of minor salivary glandVcase report. Ann Acad Med Singapore 2007;36: 857Y860 13. Randhawa T, Varghese I, Shameena P. Necrotizing sialometaplasia of tongue. J Oral Maxillofac Pathol 2009;13:35Y37 14. Oliveira Alves MG, Kitakawa D, Carvalho YR. Necrotizing sialometaplasia as a cause of a nodule in the hard palate : a case report. J Med Case Rep 2011;5:406 15. Carlson DL. Necrotizing sialometaplasia: a practical approach to the diagnosis. Arch Pathol Lab Med 2009;133: 692Y698

Sagittal Mandibular Osteotomy for Removal of Intraosseous Lesion Ju´lio Ce´sar Silva de Oliveira, DDS,* Idelmo Rangel Garcia Jr, DDS, MSc,* Willian Morais de Melo, DDS, MSc,* Saulo de Matos Barbosa, DDS,Þ Paulo Maria Santos Rabeˆlo Jr, DDS, MSc,þ Eider Guimara˜es Bastos, DDS, MScþ Abstract: The ramus sagittal split osteotomy or mandibular body is an established technique for correction of dentofacial deformities

* 2014 Mutaz B. Habal, MD

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

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but can have an accurate indication in cases requiring surgical access to remove lesions or more teeth included in the region of the mandibular angle. The main advantages of this technique are the possibility of preservation of the inferior alveolar nerve bundle and significant reduction in postoperative morbidity. In this article, the authors show a case in which the sagittal osteotomy of the mandible was used to gain access for removal of a lesion (complex odontoma). Key Words: Sagittal split osteotomy, odontoma, mandibular nerve, surgical management

T

he most common and versatile osteotomy for the treatment of dentofacial deformities is the bilateral sagittal split osteotomy of the mandible.1 This technique has become more prominent in 1957,2 which could be conducted, retreats or advances, depending on the surgical planning. This osteotomy had several suggested changes until the procedure became so popular, but many of them are based on an individual surgeon’s preference, and their effect on the outcome of the osteotomy is questionable. This type has the advantages of less surgical morbidity and especially the protection of the inferior alveolar neurovascular bundle. However, it has disadvantages such as the need for general anesthesia and a certain complex technique to be performed well.3 Although the main indication is the correction of dentofacial deformities,4,5 including prognathism, outdated, facial asymmetry, and small anterior open bites, the bilateral sagittal split osteotomy can also be easily indicated for the removal of cysts and bone tumors and teeth located near the lower alveolar nerve. In this article, we will briefly review the literature on the use of bilateral sagittal split osteotomy of the mandible for removal of lesions located in the mandibular ramus through a clinical case.

CLINICAL REPORT This is the case of a 20-year-old female patient who presented at the School of Dentistry, Federal University of Maranha˜o, for evaluation of the possibility of removal of third molars. The patient did not have any health problems, whether systemic or local. After clinical intraoral evaluation, absence of the elements in question was noted. Radiographic examination was requested preoperatively, a panoramic radiograph, and a large radiopaque mass in the region of the mandibular branch on the right side was found (Fig. 1A). Dental elements were present in 18, 28, and 38. The patient did not complain of pain, there was no increase in volume that could have repercussions in the face, and there was no limitation of mouth opening. From the *Department of Surgery and Integrated Clinic, Ara0atuba Dental School, Univ Estadual Paulista Ju´lio de Mesquita Filho Ara0atubaYUNESP, Sa˜o Paulo, Brazil; †Oral and Maxillofacial Surgery Division, Pedro Ernesto University Hospital, Rio de Janeiro, Brazil; and ‡Department of Dentistry II, Federal University of the Maranha˜o, Sa˜o Luı´s, Brazil. Received October 1, 2013. Accepted for publication October 28, 2013. Address correspondence and reprint requests to Ju´lio Ce´sar Silva de Oliveira, DDS, Department of Surgery and Integrated Clinic, University of the State of Sa˜o Paulo, Rua Jose´ Bonifa´cio 1193, CEP: 16015-050, Ara0atuba, Sa˜o Paulo, Brazil; E-mail: [email protected] Ju´lio Ce´sar Silva de Oliveira, DDS, is an MSc student; Willian Morais de Melo, DDS, MSc, is a PhD student; and Saulo de Matos Barbosa, DDS, is a resident in oral and maxillofacial surgery. The authors report no conflicts of interest. Copyright * 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000519

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FIGURE 1. A, Panoramic radiograph showing the bone mass in the region of the right mandibular angle. B, Sagittal view cone beam computed tomography, showing the proximity of the injury to the inferior alveolar nerve. C, Three-dimensional reconstruction of the patient’s jaw; we can notice a slight asymmetry of the right side over the left side.

To foster a better surgical planning, we requested a cone beam computed tomography and three-dimensional reconstruction (Figs. 1B, C), which noted that the lesion was surrounded by a thin radiolucent zone. Preoperative occlusion was in stable condition (Fig. 2A). Because the inferioralveolar nerveinjurywasveryclose,thisregionisdifficulttoreach to perform a common enucleation, and performance of sagittal mandibular osteotomy was chosen in agreement with the patient to provide adequate access for the removal of the lesion, thus preserving the nerve. After tracheal intubation, we gained full access to the mucoperiosteum in the region involved, then detached the periosteum, and then performed sagittal split osteotomy with a surgical saw and copious irrigation with 0.9% saline and supplemented with chisel and hammer, but there was a fracture of the buccal cortex, which fortunately caused no major intraoperative problems. We proceeded to the total removal of the lesion and, enjoying the surgical time, extracted 18, 28, and 38 (Fig. 2B). After this phase, the patient was positioned, locking the jaw by means of locking screws (Fig. 2C), restoring the preoperative occlusion. The bone stumps were repositioned and fixed with 1 plate and 4 screws through the 2.0 system (Fig. 3A). The incision was closed with resorbable thread, and the lock jaw was released. The patient did not complain of paresthesia of the inferior alveolar nerve, which ensured the success of the surgical indication with the total removal of the lesion (Fig. 3B) without complications and resulted in a similar occlusion postoperatively (Fig. 4). The diagnosis was complex odontoma, after histopathologic analysis. The patient was monitored for 3 years without occlusal alteration, complaints of pain, and paresthesia and with jaw movements preserved.

DISCUSSION The bilateral sagittal split osteotomy is indicated, first, to possibly move the lower jaw to correct any types of deviations and deformities, within a certain limit. Furthermore, the sagittal split osteotomy may be

FIGURE 2. A, Preoperative occlusion. B, Macroscopic aspect of the lesion and the third molars removed. C, Lock jaw transoperative.

FIGURE 3. A, Intraoperative image after osteotomy fixation with a plate system 2.0. B, Postoperative radiographic image showing the total removal of the lesion.

* 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 3, May 2014

Brief Clinical Studies

REFERENCES

FIGURE 4. Image of intraoral occlusion 45 days postoperatively without infection or other complications, similar to preoperative occlusion.

used as means of access for removal of deeply impacted teeth and damage, primarily in young patients, in that the lesion is completely across the alveolar bone4,6 so that the inferior alveolar nerve is preserved, besides preventing the need for bone grafting.7 Excision of lesions of this size for enucleation typical in hard-toreach places usually requires the removal of large amounts of bone tissue,5,6 which can cause significant bone defects, fractures, or unwanted damage to the inferior alveolar nerve, resulting in increased postoperative morbidity.3,4,8 In a review, we found 11 articles using the sagittal split osteotomy of the mandible for removal of intraosseous lesions (Table 1). Note the predominance of odontomas among lesions that were removed using this surgical technique and the case presented in this article. Scolozzi et al,3 2007, conducted a study correlating the Le Fort I osteotomy and sagittal used for the removal of cysts in the maxilla and the mandible, respectively. The authors concluded that Le Fort I osteotomy and mandibular sagittal provide excellent surgical exposure for the safe removal of intraosseous cysts of the jaws and in particular cases in which the use of conventional techniques could increase the risk for complications or result in a significant loss of bone mass tissue. However, this technique can have potential complications inherent such as excessive swelling and bleeding, intraoperative trauma to the temporomandibular joint, and damage to the inferior alveolar nerve, although in most cases these are temporary and not disabling,3 and unfavorable fractures4 that can result in infection, sequestration of fragments, delayed union, malunion, or fibrous union.1 In the articles obtained, there was no citation regarding complications, but in this case, there was an undesirable vestibular cortex fracture during division of the jaw, which fortunately brought no major problems during the surgery and during the postoperative period. Consistent with the literature, we can conclude that the sagittal osteotomy of the mandible, when properly indicated and performed, shows to be very versatile and efficient in providing surgical access to remove lesions and included more teeth in the region of the mandibular angle, and the benefits are much greater than the risks.

TABLE 1. Articles Found in the Literature Related to the Removal of Lesions Using Sagittal Split Osteotomy of the Mandible Author and Year

Type of Lesion

9

Mahmood et al 2013 Orbach10 2008 Scolozzi et al3 2007

Schwannoma Ossifying fibroma 2 Dentigerous cysts and 2 odontogenic keratocyst tumors Odontoma Odontoma Myxoma Odontoma Myxoma Odontoma Odontoma Odontogenic keratocyst tumor

Casap et al8 2006 Guven11 1999 Wong6 1992 Wong7 1989 Petti et al12 1987 Frame4 1986 Barnard5 1983 Rittersma and van Gool13 1979

1. Cillo JE, Stella JP. Selection of sagittal split ramus osteotomy technique based on skeletal anatomy and planned distal segment movement: current therapy. J Oral Maxillofac Surg 2005;63:109Y114 2. Trauner R, Obwegeser H. The surgical correction of mandibular prognathism and retrognathia with consideration of genioplasty: part I. Surgical procedures to correct mandibular prognathism and reshaping of the chin. Oral Surg Oral Med Oral Pathol 1957;10:677Y689 3. Scolozzi P, Lombardi T, Jaques B. Le Fort I type osteotomy and mandibular sagittal osteotomy as a surgical approach for removal of jaw cysts. J Oral Maxillofac Surg 2007;65:1419Y1426 4. Frame JW. Surgical excision of a large complex composite odontome of the mandible. Br J Oral Maxillofac Surg 1986;24:47Y51 5. Barnard D. Surgical access to a complex composite odontome by sagittal splitting of the mandible. Br J Oral Surg 1983;21:44Y48 6. Wong GB. Large odontogenic myxoma of the mandible treated by sagittal ramus osteotomy and peripheral ostectomy. J Oral Maxillofac Surg 1992;50:1221Y1224 7. Wong GB. Surgical management of a large, complex mandibular odontoma by unilateral sagittal split osteotomy. J Oral Maxillofac Surg 1989;47:179Y182 8. Casap N, Zeltser R, Abu-Tair J, et al. Removal of a large odontoma by sagittal split osteotomy. J Oral Maxillofac Surg 2006;64:1833Y1836 9. Mahmood L, Demian N, Weinstock YE, et al. Mandibular nerve schwannoma resection using sagittal split ramus osteotomy. J Oral Maxillofac Surg 2013;71:1861Y1872 10. Orbach S. Ossifying fibroma of mandibular ramus treated with sagittal splitting osteotomy. N Y State Dent J 2008;74:34Y36 11. Guven O. An unusual treatment with sagittal split osteotomy: report of a case involving an odontoma. Int J Adult Orthodon Orthognath Surg 1999;14:163Y166 12. Petti NA, Weber FL, Miller MC. Resection of a mandibular myxoma via a sagittal ramus osteotomy. J Oral Maxillofac Surg 1987;45:793Y795 13. Rittersma J, van Gool AV. Surgical access to multicystic lesions, by sagittal splitting of the lower jaw. J Maxillofac Surg 1979;7:246Y250

Heminasal Agenesis: A Reconstructive Challenge Mark Fisher, BA, Jonathan Zelken, MD, Richard J. Redett, MD Abstract: Heminasal agenesis is a rare congenital malformation often associated with deformities of the eyes and lacrimal system, midface, and proboscis lateralis. Reconstruction is especially challenging because of missing lining, cartilage, and skin. We present a case of heminasal agenesis in a 5-year-old girl with concomitant hypertelorism, coloboma of the eyelids, and maxillary hypoplasia. The patient underwent facial bipartition for hypertelorism correction and cantilever bone graft. A forehead flap was designed using an anaplastic model from the patient’s twin sister. Cartilage harvested From the Departments of Plastic and Reconstructive Surgery, Johns Hopkins University School of Medicine, Baltimore, Maryland. Received October 4, 2013. Accepted for publication November 13, 2013. Address correspondence and reprint requests to Richard J. Redett, MD, Department of Plastic and Reconstructive Surgery, Johns Hopkins School of Medicine, Bloomberg 7314B, 1800 Orleans St, Baltimore, MD 21287; 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.0000000000000542

* 2014 Mutaz B. Habal, MD

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

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Sagittal mandibular osteotomy for removal of intraosseous lesion.

The ramus sagittal split osteotomy or mandibular body is an established technique for correction of dentofacial deformities but can have an accurate i...
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