The Journal of Craniofacial Surgery
& Volume 25, Number 3, May 2014
a silver point that had remained in the maxilla when a tooth (probably 27) had been extracted. In patient 2, the major component was iron. On the basis of the major component, shape, region of migration, and the patient?s clinical history, the foreign body was considered to be an iatrogenic migration of a medical tool, such as a suture needle.
DISCUSSION There have been many reports that have described the migration of a foreign body in the head and neck region, and there have been various migration pathways reported, such as dental treatment, accidents, during meals, and as a result of self-injurious behavior. Regarding the foreign bodies, the needle used for the injection of dental anesthesia was the most commonly reported agent. Augello et al10 reported that the cause of injection needle fracture was generally an inadequate technique or using too thin of needles and that most of the accidents involving these injections occurred during inferior alveolar nerve block. They concluded that observing basic rules of treatment could help to avoid these kinds of accidents. This can also be said about other dental treatments. Awad et al11 evaluated the retained root fragments, impacted teeth, foreign bodies, radiolucencies, radiopacities, mental foramina at or near the crest of the residual alveolar ridge, and maxillary sinus proximity to the crest of the residual alveolar ridge from OPGs in 271 patients who were edentulous in 1 or both arches. A foreign body was found in 6 of these patients. In our cases, the foreign bodies were detected incidentally on OPGs. These findings suggest the importance of OPG examinations for screening the maxilla and the mandible. It has been reported that CT is the most efficacious examination for detecting foreign bodies.12,13 Computed tomography was used with a stent that enclosed the radiopaque materials to determine the exact position of the foreign body because there was no useful anatomic landmark in patient 1. The stent enabled the exact position of the foreign body to be revealed, which made the removal of the object during surgery easier. On the other hand, there were residual teeth around the foreign body in patient 2. Therefore, the identification of the position was possible without a stent. Computed tomographic examination with a stent that encloses radiopaque materials is considered to be an effective way to reveal the exact position of a foreign body when there is no anatomic landmark around the foreign body. Kili0 et al14 reported the case of a patient who experienced pain during a magnetic resonance imaging (MRI) examination. A migrated metallic foreign body, which had not manifested any symptoms for 28 years, moved into his eyeball because of magnetic force. The authors concluded that it was important to confirm that no metallic bodies, including medical implants and foreign bodies, are present before performing an MRI examination. Both foreign bodies in the current study were observed to have reached the maxillary sinus according to CT sections. It was considered that the foreign bodies might have moved into the maxillary sinus if an MRI examination had been performed. Moreover, the finding and removal of foreign bodies were beneficial for these patients because the formation of an oroantral fistula or severe inflammation may be induced when an infection occurs. The migration pathways in the current study were speculated on the basis of the componential analysis using an x-ray fluorescence spectrometer and the patients? clinical histories. The revelation of foreign bodies and their migration pathways into the jaw bone can help clinicians avoid injuring patients during further examinations (eg, MRI), can improve the patients? health, and can also contribute to reducing iatrogenic accidents.
Brief Clinical Studies
REFERENCES 1. Passi S, Sharma N. Case report unusual foreign bodies in the orofacial region. Case Rep Dent [published ahead of print July 9, 2012] doi:10.1155/2012/191873. 2. Mohanavalli S, David JJ, Gnanam A. Rare foreign bodies in oro-facial regions. Indian J Dent Res 2011;22:713Y715 3. Bodet Agustı´ E, Viza Puiggro´s I, Romeu Figuerola C, et al. Foreign bodies in maxillary sinus. Acta Otorrinolaringol Esp 2009;60:190Y193 4. de Jong AL, Moola F, Kramer D, et al. Foreign bodies of the hard palate. Int J Pediatr Otorhinolaryngol 1998;43:27Y31 5. Nariai Y, Yanai C, Kondo S, et al. A fish bone in the tongue: a report of a case. Asian J Oral Maxillofac Surg 2010;22:30Y32 6. Pigott DC, Buckingham RB, Eller RL, et al. Foreign body in the tongue: a novel use for emergency department ultrasonography. Ann Emerg Med 2005;45:677Y679 7. Rangeeth BN, Moses J, Reddy NV. Self-injurious behavior and foreign body entrapment in the root canal of a mandibular lateral incisor. J Indian Soc Pedod Prev Dent 2011;29:S95YS98 8. Tay AB. Long-standing intranasal foreign body: an incidental finding on dental radiograph: a case report and literature review. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2000;90:546Y549 9. Kittle PE, Aaron GR, Jones HL, et al. Incidental finding of an intranasal foreign body discovered on routine dental examination: case report. Pediatr Dent 1991;13:49Y51 10. Augello M, von Jackowski J, Gra¨tz KW, et al. Needle breakage during local anesthesia in the oral cavityVa retrospective of the last 50 years with guidelines for treatment and prevention. Clin Oral Investig 2011;15:3Y8 11. Awad EA, Al-Dharrab A. Panoramic radiographic examination: a survey of 271 edentulous patients. Int J Prosthodont 2011;24:55Y57 12. Wilson WB, Dreisbach JN, Lattin DE, et al. Magnetic resonance imaging of nonmetallic orbital foreign bodies. Am J Ophthalmol 1988;105:612Y617 13. de Santana Santos T, Avelar RL, Melo AR, et al. Current approach in the management of patients with foreign bodies in the maxillofacial region. J Oral Maxillofac Surg 2011;69:2376Y2382 14. Kili0 A, Avcu S, TekBn S, et al. MRI-induced migration of retained metallic foreign body in the eye. Ophthalmic Surg Lasers Imaging 2010;9:1Y3
Two Rare Entities in the Same Palate Lesion: Hyalinizing-Type Clear Cell Carcinoma and Necrotizing Sialometaplasia Rabia Bozdo?an Arpaci, MD,* Tuba Kara, MD,* Canan Porgali, MD,* Ebru Serinsoz, MD,* Ayse Polat, MD,* Yusuf Vayisoglu, MD,Þ Cengiz Ozcan, MDÞ Abstract: Hyalinizing clear cell carcinoma is a low-grade malignant epithelial neoplasm of the salivary glands. The tumor has epithelial cells and lacks myoepithelial cells. Necrotizing sialometaplasia is a benign, self-limiting lesion of the salivary glands. The clinical and From the Departments of *Pathology, and †Otorhinolaryngology, Mersin University Medical School, Mersin, Turkey. Received September 27, 2013. Accepted for publication October 28, 2013. Address correspondence and reprint requests to Rabia Bozdo?an Arpaci, MD, Department of Pathology, Mersin University Medical School, 33100, Mersin, Turkey; 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.0000000000000517
* 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
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histologic features mimic those of mucoepidermoid carcinoma or squamous cell carcinoma. The importance of these entities are the rarity of both of them and their potential to be misdiagnosed as other lesions. Pathologists and clinicians should be aware of these entities to prevent misdiagnosis. This is the first clinical report of 2 rare and consecutive different entities of the same location on the hard palate to our knowledge. Key Words: Clear cell, carcinoma, palate, necrotizing sialometaplasia
H
yalinizing clear cell carcinoma (HCCC) is an uncommon tumor and has been described recently as a neoplasm that mainly affects the oral cavity of an adult woman. The tumor is composed of characteristic areas of clear cells and hyalinization of the stroma. It is characterized by hyalinizing stroma and its clear-appearing cytoplasm. Hyalinizing clear cell carcinoma is composed only of epithelial cells. These cells are positive for cytokeratin (CK) but negative for S100 protein and smooth muscle actin immunohistochemically. This immunohistochemical staining characteristic of the tumor distinguishes it from other salivary gland tumors that have a predominant population of clear cell component.1,2 Because of the rarity of recurrence and metastases, it is considered to be a low-grade neoplasm.3 The tumor is usually misdiagnosed as poorly differentiated carcinoma, squamous cell carcinoma, acinic cell carcinoma, mucoepidermoid carcinoma and epithelial-myoepithelial carcinoma, as well as primary or metastatic clear cell neoplasms, including various odontogenic clear cell tumors, other primary intraosseous salivary gland tumors, and metastatic clear cell adenocarcinomas from other organs, particularly the kidney.1,4,5 Necrotizing sialometaplasia presents as a benign, self-healing process of the hard and soft palates characterized by deeply seated ulcers.6 This benign condition has been also reported in the paranasal sinuses, lung, breast, trachea, larynx, hypopharynx, and oral cavity; also, other sites of the body that contain elements of salivary gland has been reported.7 The lesion may be mistaken for malignant diseases both clinically and microscopically. The correct diagnosis of necrotizing sialometaplasia is important because of results in unnecessary surgery.8 We report a rare case of HCCC in a patient who had a swelling in the palate with a 3-month duration. In addition to the typical features of HCCC in this case, areas of cystic degeneration and necrotizing sialometaplasia were seen histologically on the specimen that underwent a subsequent excision, which was performed 12 days after the original surgery.
& Volume 25, Number 3, May 2014
FIGURE 2. Large polygonal cells with abundant clear cytoplasm and distinct cell borders (hematoxylin-eosin, original magnification 200).
physical examination. There was no other palpable mass. The result of systemic examination was otherwise normal. An incision biopsy of the mass was sent for a frozen section for diagnostic purposes, which was reported as mucoepidermoid carcinoma. Grossly, the tumor was solid with cystic areas, grayish white in color, and 2.5 2 cm in size. Microscopically, the tumor was composed of large polygonal cells with abundant clear cytoplasm (Fig. 2) and distinct cell borders with a few cells having eosinophilic cytoplasm. There was a a large area of cystic degeneration. The stroma was desmoplastic-sclerotic and showed mucoid-myxoid areas. These clear cells were negative for mucin and were seen as diastasesensitive while positive for periodic asit schiff with intracytoplasmic material. The cells were arranged in islands and trabeculae with stroma showing areas of hyalinization (Fig. 3). The tumor has shown perineural infiltration, and the number of mitosis was less than 1 per 10 high-power field. Immunohistochemically, these cells were positive for CK while negative for p63 and S100 protein. A definite diagnosis of HCCC was made. The resection material showed that the tumor was close to the margin, and the patient was operated on again. The subsequent excision was performed after 12 days from the initial operation. Microscopically, the biopsy results showed no evidence of any neoplastic changes; the histologic examination of the lesion showed moderate inflammatory infiltrate and mild partial necrosis of the salivary gland. Results of the histopathologic examination revealed an adjacent hyperplastic mucosal epithelium, squamous metaplasia, and pseudoepitheliomatous hyperplasia of the adjacent overlying epithelium and associated squamous metaplasia of the seromucous glands with preservation of lobular morphology (Fig. 4). These findings were consistent with early necrotizing sialometaplasia.
DISCUSSION
A 58-year-old man was referred to otorhinolaryngology clinic because of a swelling in the palate. It began as a swelling 3 months earlier and gradually increased to its present size of 3 3 cm. It had a cystic apperance and there was no ulceration (Fig. 1) as seen in the
Hyalinizing clear cell carcinoma usually presents as an asymptomatic slow-growing and painless submucosal mass. Results of histopathologic examination of clear cell carcinoma of the oral cavity show monomorphic clear cells arranged in sheets, nests, cords, organoid, and single cells. Ducts and glandlike spaces are not identified. The hyalinizing thick bands of collagen subtype separates the clusters of tumor cells into solid lobules. Clearing of the cytoplasm is caused by the presence of glycogen. This feature is shown by periodic asit schiff stain with and without diastase. The tumor cells are also negative for mucicarmine stain.9,10 The tumor is immunoreactive to CK and variable staining with S100, glial
FIGURE 1. This figure shows a nodular swelling in the palate of the patient.
FIGURE 3. Clear cells were arranged in islands and trabeculae with stroma showing areas of hyalinization (hematoxylin-eosin, original magnification 100).
CLINICAL REPORT
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* 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
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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& Volume 25, Number 3, May 2014
a silver point that had remained in the maxilla when a tooth (probably 27) had been extracted. In patient 2, the major component was iron. On the basis of the major component, shape, region of migration, and the patient?s clinical history, the foreign body was considered to be an iatrogenic migration of a medical tool, such as a suture needle.
DISCUSSION There have been many reports that have described the migration of a foreign body in the head and neck region, and there have been various migration pathways reported, such as dental treatment, accidents, during meals, and as a result of self-injurious behavior. Regarding the foreign bodies, the needle used for the injection of dental anesthesia was the most commonly reported agent. Augello et al10 reported that the cause of injection needle fracture was generally an inadequate technique or using too thin of needles and that most of the accidents involving these injections occurred during inferior alveolar nerve block. They concluded that observing basic rules of treatment could help to avoid these kinds of accidents. This can also be said about other dental treatments. Awad et al11 evaluated the retained root fragments, impacted teeth, foreign bodies, radiolucencies, radiopacities, mental foramina at or near the crest of the residual alveolar ridge, and maxillary sinus proximity to the crest of the residual alveolar ridge from OPGs in 271 patients who were edentulous in 1 or both arches. A foreign body was found in 6 of these patients. In our cases, the foreign bodies were detected incidentally on OPGs. These findings suggest the importance of OPG examinations for screening the maxilla and the mandible. It has been reported that CT is the most efficacious examination for detecting foreign bodies.12,13 Computed tomography was used with a stent that enclosed the radiopaque materials to determine the exact position of the foreign body because there was no useful anatomic landmark in patient 1. The stent enabled the exact position of the foreign body to be revealed, which made the removal of the object during surgery easier. On the other hand, there were residual teeth around the foreign body in patient 2. Therefore, the identification of the position was possible without a stent. Computed tomographic examination with a stent that encloses radiopaque materials is considered to be an effective way to reveal the exact position of a foreign body when there is no anatomic landmark around the foreign body. Kili0 et al14 reported the case of a patient who experienced pain during a magnetic resonance imaging (MRI) examination. A migrated metallic foreign body, which had not manifested any symptoms for 28 years, moved into his eyeball because of magnetic force. The authors concluded that it was important to confirm that no metallic bodies, including medical implants and foreign bodies, are present before performing an MRI examination. Both foreign bodies in the current study were observed to have reached the maxillary sinus according to CT sections. It was considered that the foreign bodies might have moved into the maxillary sinus if an MRI examination had been performed. Moreover, the finding and removal of foreign bodies were beneficial for these patients because the formation of an oroantral fistula or severe inflammation may be induced when an infection occurs. The migration pathways in the current study were speculated on the basis of the componential analysis using an x-ray fluorescence spectrometer and the patients? clinical histories. The revelation of foreign bodies and their migration pathways into the jaw bone can help clinicians avoid injuring patients during further examinations (eg, MRI), can improve the patients? health, and can also contribute to reducing iatrogenic accidents.
Brief Clinical Studies
REFERENCES 1. Passi S, Sharma N. Case report unusual foreign bodies in the orofacial region. Case Rep Dent [published ahead of print July 9, 2012] doi:10.1155/2012/191873. 2. Mohanavalli S, David JJ, Gnanam A. Rare foreign bodies in oro-facial regions. Indian J Dent Res 2011;22:713Y715 3. Bodet Agustı´ E, Viza Puiggro´s I, Romeu Figuerola C, et al. Foreign bodies in maxillary sinus. Acta Otorrinolaringol Esp 2009;60:190Y193 4. de Jong AL, Moola F, Kramer D, et al. Foreign bodies of the hard palate. Int J Pediatr Otorhinolaryngol 1998;43:27Y31 5. Nariai Y, Yanai C, Kondo S, et al. A fish bone in the tongue: a report of a case. Asian J Oral Maxillofac Surg 2010;22:30Y32 6. Pigott DC, Buckingham RB, Eller RL, et al. Foreign body in the tongue: a novel use for emergency department ultrasonography. Ann Emerg Med 2005;45:677Y679 7. Rangeeth BN, Moses J, Reddy NV. Self-injurious behavior and foreign body entrapment in the root canal of a mandibular lateral incisor. J Indian Soc Pedod Prev Dent 2011;29:S95YS98 8. Tay AB. Long-standing intranasal foreign body: an incidental finding on dental radiograph: a case report and literature review. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2000;90:546Y549 9. Kittle PE, Aaron GR, Jones HL, et al. Incidental finding of an intranasal foreign body discovered on routine dental examination: case report. Pediatr Dent 1991;13:49Y51 10. Augello M, von Jackowski J, Gra¨tz KW, et al. Needle breakage during local anesthesia in the oral cavityVa retrospective of the last 50 years with guidelines for treatment and prevention. Clin Oral Investig 2011;15:3Y8 11. Awad EA, Al-Dharrab A. Panoramic radiographic examination: a survey of 271 edentulous patients. Int J Prosthodont 2011;24:55Y57 12. Wilson WB, Dreisbach JN, Lattin DE, et al. Magnetic resonance imaging of nonmetallic orbital foreign bodies. Am J Ophthalmol 1988;105:612Y617 13. de Santana Santos T, Avelar RL, Melo AR, et al. Current approach in the management of patients with foreign bodies in the maxillofacial region. J Oral Maxillofac Surg 2011;69:2376Y2382 14. Kili0 A, Avcu S, TekBn S, et al. MRI-induced migration of retained metallic foreign body in the eye. Ophthalmic Surg Lasers Imaging 2010;9:1Y3
Two Rare Entities in the Same Palate Lesion: Hyalinizing-Type Clear Cell Carcinoma and Necrotizing Sialometaplasia Rabia Bozdo?an Arpaci, MD,* Tuba Kara, MD,* Canan Porgali, MD,* Ebru Serinsoz, MD,* Ayse Polat, MD,* Yusuf Vayisoglu, MD,Þ Cengiz Ozcan, MDÞ Abstract: Hyalinizing clear cell carcinoma is a low-grade malignant epithelial neoplasm of the salivary glands. The tumor has epithelial cells and lacks myoepithelial cells. Necrotizing sialometaplasia is a benign, self-limiting lesion of the salivary glands. The clinical and From the Departments of *Pathology, and †Otorhinolaryngology, Mersin University Medical School, Mersin, Turkey. Received September 27, 2013. Accepted for publication October 28, 2013. Address correspondence and reprint requests to Rabia Bozdo?an Arpaci, MD, Department of Pathology, Mersin University Medical School, 33100, Mersin, Turkey; 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.0000000000000517
* 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
Brief Clinical Studies
histologic features mimic those of mucoepidermoid carcinoma or squamous cell carcinoma. The importance of these entities are the rarity of both of them and their potential to be misdiagnosed as other lesions. Pathologists and clinicians should be aware of these entities to prevent misdiagnosis. This is the first clinical report of 2 rare and consecutive different entities of the same location on the hard palate to our knowledge. Key Words: Clear cell, carcinoma, palate, necrotizing sialometaplasia
H
yalinizing clear cell carcinoma (HCCC) is an uncommon tumor and has been described recently as a neoplasm that mainly affects the oral cavity of an adult woman. The tumor is composed of characteristic areas of clear cells and hyalinization of the stroma. It is characterized by hyalinizing stroma and its clear-appearing cytoplasm. Hyalinizing clear cell carcinoma is composed only of epithelial cells. These cells are positive for cytokeratin (CK) but negative for S100 protein and smooth muscle actin immunohistochemically. This immunohistochemical staining characteristic of the tumor distinguishes it from other salivary gland tumors that have a predominant population of clear cell component.1,2 Because of the rarity of recurrence and metastases, it is considered to be a low-grade neoplasm.3 The tumor is usually misdiagnosed as poorly differentiated carcinoma, squamous cell carcinoma, acinic cell carcinoma, mucoepidermoid carcinoma and epithelial-myoepithelial carcinoma, as well as primary or metastatic clear cell neoplasms, including various odontogenic clear cell tumors, other primary intraosseous salivary gland tumors, and metastatic clear cell adenocarcinomas from other organs, particularly the kidney.1,4,5 Necrotizing sialometaplasia presents as a benign, self-healing process of the hard and soft palates characterized by deeply seated ulcers.6 This benign condition has been also reported in the paranasal sinuses, lung, breast, trachea, larynx, hypopharynx, and oral cavity; also, other sites of the body that contain elements of salivary gland has been reported.7 The lesion may be mistaken for malignant diseases both clinically and microscopically. The correct diagnosis of necrotizing sialometaplasia is important because of results in unnecessary surgery.8 We report a rare case of HCCC in a patient who had a swelling in the palate with a 3-month duration. In addition to the typical features of HCCC in this case, areas of cystic degeneration and necrotizing sialometaplasia were seen histologically on the specimen that underwent a subsequent excision, which was performed 12 days after the original surgery.
& Volume 25, Number 3, May 2014
FIGURE 2. Large polygonal cells with abundant clear cytoplasm and distinct cell borders (hematoxylin-eosin, original magnification 200).
physical examination. There was no other palpable mass. The result of systemic examination was otherwise normal. An incision biopsy of the mass was sent for a frozen section for diagnostic purposes, which was reported as mucoepidermoid carcinoma. Grossly, the tumor was solid with cystic areas, grayish white in color, and 2.5 2 cm in size. Microscopically, the tumor was composed of large polygonal cells with abundant clear cytoplasm (Fig. 2) and distinct cell borders with a few cells having eosinophilic cytoplasm. There was a a large area of cystic degeneration. The stroma was desmoplastic-sclerotic and showed mucoid-myxoid areas. These clear cells were negative for mucin and were seen as diastasesensitive while positive for periodic asit schiff with intracytoplasmic material. The cells were arranged in islands and trabeculae with stroma showing areas of hyalinization (Fig. 3). The tumor has shown perineural infiltration, and the number of mitosis was less than 1 per 10 high-power field. Immunohistochemically, these cells were positive for CK while negative for p63 and S100 protein. A definite diagnosis of HCCC was made. The resection material showed that the tumor was close to the margin, and the patient was operated on again. The subsequent excision was performed after 12 days from the initial operation. Microscopically, the biopsy results showed no evidence of any neoplastic changes; the histologic examination of the lesion showed moderate inflammatory infiltrate and mild partial necrosis of the salivary gland. Results of the histopathologic examination revealed an adjacent hyperplastic mucosal epithelium, squamous metaplasia, and pseudoepitheliomatous hyperplasia of the adjacent overlying epithelium and associated squamous metaplasia of the seromucous glands with preservation of lobular morphology (Fig. 4). These findings were consistent with early necrotizing sialometaplasia.
DISCUSSION
A 58-year-old man was referred to otorhinolaryngology clinic because of a swelling in the palate. It began as a swelling 3 months earlier and gradually increased to its present size of 3 3 cm. It had a cystic apperance and there was no ulceration (Fig. 1) as seen in the
Hyalinizing clear cell carcinoma usually presents as an asymptomatic slow-growing and painless submucosal mass. Results of histopathologic examination of clear cell carcinoma of the oral cavity show monomorphic clear cells arranged in sheets, nests, cords, organoid, and single cells. Ducts and glandlike spaces are not identified. The hyalinizing thick bands of collagen subtype separates the clusters of tumor cells into solid lobules. Clearing of the cytoplasm is caused by the presence of glycogen. This feature is shown by periodic asit schiff stain with and without diastase. The tumor cells are also negative for mucicarmine stain.9,10 The tumor is immunoreactive to CK and variable staining with S100, glial
FIGURE 1. This figure shows a nodular swelling in the palate of the patient.
FIGURE 3. Clear cells were arranged in islands and trabeculae with stroma showing areas of hyalinization (hematoxylin-eosin, original magnification 100).
CLINICAL REPORT
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* 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
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-
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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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