Brief Clinical Studies
The Journal of Craniofacial Surgery • Volume 25, Number 6, November 2014
27. Moystad A, Mork-Knutsen BB, Bjornland T. Injection of sodium hyaluronate compared to a corticosteroid in the treatment of patients with temporomandibular joint osteoarthritis: a CT evaluation. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2008;105:53–60 28. Sakamoto I, Yoda T, Tsukahara H. Clinical studies of arthrocentesis of the temporomandibular joint—analysis of clinical findings in patients with a good outcome. Jpn J Oral Maxillofac Surg 1996;42:808–812 29. Emshoff R. Clinical factors affecting the outcome of arthrocentesis and hydraulic distension of the temporomandibular joint. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2005;100:409–414 30. Krogstad BS, Jokstad A, Dahl BL, et al. Relationships between risk factors and treatment outcome in a group of patients with temporomandibular disorders. J Orofac Pain 1996;10:48–53 31. Wexler GB, Steed PA. Psychological factors and temporomandibular outcomes. Cranio 1998;16:72–77 32. Corvol M, Blanchard O, Tsagris L. Bone and cartilage responsiveness to sex steroid hormones. J Steroid Biochem Mol Biol 1992;43:415–418 33. Melby MK, Lock M, Kaufert P. Culture and symptom reporting at menopause. Hum Reprod Update 2005;11:495–512
Salivary Duct Carcinoma in the Mandible Shuang Shi, MS,* Qi-Gen Fang, MS,† Changfu Sun, MD† Abstracts: We reported 1 case of salivary duct carcinoma (SDC) in the mandible. The patient complained of pain and a growing mass in the right submandibular area for approximately 2 months. On clinical examination, there was a mass under the right angle of the mandible with a size of approximately 3 3 cm, a smooth surface, a poor activity, and a hard texture. Panoramic radiograph revealed poorly circumscribed area. Computed tomography presented mandible central destruction. Biopsy examination showed a malignant tumor that originated in the central epithelium of the mandible. An operation of unilateral selective neck dissection and mandible subtotal ectomy was performed. Postoperative pathology reported SDC. The patient received postoperative radiation and stayed alive at last follow-up without disease recurrence. Ablative resection and postoperative radiotherapy were the standard treatment stratagem for SDC, but trastuzumab therapy might play a key role in treating the disease in future. Key Words: Salivary duct carcinoma, mandible disease, trastuzumab therapy
S
alivary duct carcinoma (SDC) is an aggressive neoplasm characterized by nerve infiltration, lymph metastasis, and poor
From the *Department of Pediatric Dentistry, and †Department of Oromaxillofacial-Head and Neck Surgery and Department of Oral and Maxillofacial Surgery, School of Stomatology, China Medical University, Shenyang, Liaoning, PR China. Received November 8, 2013. Accepted for publication January 14, 2014. Address correspondence and reprint requests to Chang-Fu Sun, MD, Department of Oral and Maxillofacial Surgery, School of Stomatology, China Medical University, No.117, Nanjing North St, Heping District, Shenyang, Liaoning 110002, PR China; E-mail: [email protected] The authors report no conflicts of interest. The first 2 authors contributed equally to this work. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000764
e598
prognosis.1,2 It has a male predominance and usually arises in the parotid gland, followed by the submandibular gland, but the involved site of the mandibular gland is very few; to our knowledge, only 2 cases have been reported.3,4 In this paper, we report a new case of mandibular SDC.
PATIENT A 47-year-old male patient complained of pain and a growing mass in the right submandibular area for approximately 2 months. On clinical examination, there was a mass under the right angle of the mandible with a size of approximately 3 3 cm, a smooth surface, a poor activity, and a hard texture. There were no significant abnormalities intraorally. Panoramic radiograph revealed poorly circumscribed area. Computed tomography presented mandible central destruction, and no significant lesions were found in the parotid and submandibular glands. We performed a biopsy of the lesion, and histological examination showed a malignant tumor that originated in the central epithelium of the mandible. Therefore, we conducted an operation of unilateral selective neck dissection and mandible subtotal ectomy. Postoperative pathology reported salivary duct carcinoma (SDC). The patient received postoperative radiation and stayed alive at last follow-up without disease recurrence. Trastuzumab therapy may play a key role in treating the disease.
DISCUSSION Salivary duct carcinoma was first introduced in 1968 by Kleinsasser et al,5 whose histological picture strongly resembled that of breast ductal carcinoma. Generally, SDC is a highly malignant tumor and occurs most often in men older than 40 years. The most commonly involved site is the parotid gland, followed by the submandibular gland.6,7 Cases of SDC originating in sublingual gland and minor salivary gland are few, and the mandible as the site of origin is rarer. To the best of our knowledge, the current case is the third one. Salivary duct carcinoma has a characteristic of early regional lymph node metastasis. Salovaara et al8 found that 56% of their patients had pathological cervical lymph node metastasis and that the metastasis was more often in the primary tumor staged as T2 or higher. Additionally, Guzzo et al9 reported that 15 of 26 patients had pathologic nodal involvements and lymphatic spread may be related to T stage, and more, the authors found that nodal metastases could be regarded as a negative prognostic factor. Kim et al6 described that 74.3% of the patients showed pathologic nodal involvement at the time of diagnosis and that the pathologic node metastasis was correlated with distant metastasis. Salivary duct carcinoma has another characteristic of distant metastasis. The common sites of distant metastases were liver, lung, bone, brain, and skin.9 Jaehne et al7 presented that 48% of the patients developed distant disease metastasis and that the most frequency site was lung (30%). Salovaara et al8 reported that 6 of 25 patients had distant metastases and that the most common site for metastasis was bone. Salivary duct carcinoma may arise de novo or as a relatively common malignant component of a carcinoma ex pleomorphic adenoma.1 The former usually manifests as a rapidly growing mass with nerve infiltration, but the latter may have a history of a longstanding mass with recent enlargement. Previous studies have shown that there was a relatively better prognosis in carcinoma ex pleomorphic adenoma than in SDC. The differential diagnoses for SDC were metastatic squamous cell carcinoma, metastatic breast cancer, melanoma, and oncocytic carcinoma.2 Suzuki et al3 thought that the lesion, which was most difficult to distinguish from SDC, was poorly differentiated mucoepidermoid carcinoma. In the current case, all these lesions were excluded by preoperative routine examinations and histological investigation. © 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 6, November 2014
Ablative resection and postoperative radiotherapy are still the standard treatment stratagem for SDC. However, the result is not satisfactory. Salovaara8 reported their 2- and 5-year overall and disease-specific survival rates as 66%, 41% and 75, 55%. Guzzo9 described the observed 2- and 5-year overall survival rates as 42.3% and 11.5%. New treatments were needed. Nabili et al10 found that trastuzumab was effective in treating HER-2-positive SDC and concluded that given the immunohistochemical similarities between SDC and breast duct carcinoma, patients with fluorescence in situ hybridization-positive HER-2/neu should be considered for trastuzumab therapy. Similarly, Jaspers et al11 used androgen deprivation therapy to treat 10 patients with androgen receptor–positive SDC; the outcome was really promising, but the sample was small. More large prospective studies are needed.
REFERENCES 1. Thompson LD. Salivary duct carcinoma. Ear Nose Throat J 2012;91:356–359 2. Wee DT, Thomas AA, Bradley PJ. Salivary duct carcinoma: what is already known, and can we improve survival? J Laryngol Otol 2012;126:S2–S7 3. Suzuki H, Hashimoto K. Salivary duct carcinoma in the mandible: report of a case with immunohistochemical studies. Br J Oral Maxillofac Surg 1999;37:67–69
Brief Clinical Studies
4. Kikuchi Y, Hirota M, Iwai T, et al. Salivary duct carcinoma in the mandible: a case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007;103:e41–e46 5. Kleinsasser O, Klein HJ, Hübner G. Salivary duct carcinoma. A group of salivary gland tumors analogous to mammary duct carcinoma. Arch Klin Exp Ohren Nasen Kehlkopfheikd 1968;192:100–105 6. Kim JY, Lee S, Cho KJ, et al. Treatment results of post-operative radiotherapy in patients with salivary duct carcinoma of the major salivary glands. Br J Radiol 2012;85:e947–e952 7. Jaehne M, Roeser K, Jaekel T, et al. Clinical and immunohistologic typing of salivary duct carcinoma: a report of 50 cases. Cancer 2005;103:2526–2533 8. Salovaara E, Hakala O, Bäck L, et al. Management and outcome of salivary duct carcinoma in major salivary glands. Eur Arch Otorhinolaryngol 2013;270:281–285 9. Guzzo M, Di Palma S, Grandi C, et al. Salivary duct carcinoma: clinical characteristics and treatment strategies. Head Neck 1997;19:126–133 10. Nabili V, Tan JW, Bhuta S, et al. Salivary duct carcinoma: a clinical histologic review with implications for trastuzumab therapy. Head Neck 2007;29:907–912 11. Jaspers HC, Verbist BM, Schoffelen R, et al. Androgen receptor–positive salivary duct carcinoma: a disease entity with promising new treatment options. J Clin Oncol 2011;29:e473–e476
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
e599
The Journal of Craniofacial Surgery • Volume 25, Number 6, November 2014
27. Moystad A, Mork-Knutsen BB, Bjornland T. Injection of sodium hyaluronate compared to a corticosteroid in the treatment of patients with temporomandibular joint osteoarthritis: a CT evaluation. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2008;105:53–60 28. Sakamoto I, Yoda T, Tsukahara H. Clinical studies of arthrocentesis of the temporomandibular joint—analysis of clinical findings in patients with a good outcome. Jpn J Oral Maxillofac Surg 1996;42:808–812 29. Emshoff R. Clinical factors affecting the outcome of arthrocentesis and hydraulic distension of the temporomandibular joint. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2005;100:409–414 30. Krogstad BS, Jokstad A, Dahl BL, et al. Relationships between risk factors and treatment outcome in a group of patients with temporomandibular disorders. J Orofac Pain 1996;10:48–53 31. Wexler GB, Steed PA. Psychological factors and temporomandibular outcomes. Cranio 1998;16:72–77 32. Corvol M, Blanchard O, Tsagris L. Bone and cartilage responsiveness to sex steroid hormones. J Steroid Biochem Mol Biol 1992;43:415–418 33. Melby MK, Lock M, Kaufert P. Culture and symptom reporting at menopause. Hum Reprod Update 2005;11:495–512
Salivary Duct Carcinoma in the Mandible Shuang Shi, MS,* Qi-Gen Fang, MS,† Changfu Sun, MD† Abstracts: We reported 1 case of salivary duct carcinoma (SDC) in the mandible. The patient complained of pain and a growing mass in the right submandibular area for approximately 2 months. On clinical examination, there was a mass under the right angle of the mandible with a size of approximately 3 3 cm, a smooth surface, a poor activity, and a hard texture. Panoramic radiograph revealed poorly circumscribed area. Computed tomography presented mandible central destruction. Biopsy examination showed a malignant tumor that originated in the central epithelium of the mandible. An operation of unilateral selective neck dissection and mandible subtotal ectomy was performed. Postoperative pathology reported SDC. The patient received postoperative radiation and stayed alive at last follow-up without disease recurrence. Ablative resection and postoperative radiotherapy were the standard treatment stratagem for SDC, but trastuzumab therapy might play a key role in treating the disease in future. Key Words: Salivary duct carcinoma, mandible disease, trastuzumab therapy
S
alivary duct carcinoma (SDC) is an aggressive neoplasm characterized by nerve infiltration, lymph metastasis, and poor
From the *Department of Pediatric Dentistry, and †Department of Oromaxillofacial-Head and Neck Surgery and Department of Oral and Maxillofacial Surgery, School of Stomatology, China Medical University, Shenyang, Liaoning, PR China. Received November 8, 2013. Accepted for publication January 14, 2014. Address correspondence and reprint requests to Chang-Fu Sun, MD, Department of Oral and Maxillofacial Surgery, School of Stomatology, China Medical University, No.117, Nanjing North St, Heping District, Shenyang, Liaoning 110002, PR China; E-mail: [email protected] The authors report no conflicts of interest. The first 2 authors contributed equally to this work. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000764
e598
prognosis.1,2 It has a male predominance and usually arises in the parotid gland, followed by the submandibular gland, but the involved site of the mandibular gland is very few; to our knowledge, only 2 cases have been reported.3,4 In this paper, we report a new case of mandibular SDC.
PATIENT A 47-year-old male patient complained of pain and a growing mass in the right submandibular area for approximately 2 months. On clinical examination, there was a mass under the right angle of the mandible with a size of approximately 3 3 cm, a smooth surface, a poor activity, and a hard texture. There were no significant abnormalities intraorally. Panoramic radiograph revealed poorly circumscribed area. Computed tomography presented mandible central destruction, and no significant lesions were found in the parotid and submandibular glands. We performed a biopsy of the lesion, and histological examination showed a malignant tumor that originated in the central epithelium of the mandible. Therefore, we conducted an operation of unilateral selective neck dissection and mandible subtotal ectomy. Postoperative pathology reported salivary duct carcinoma (SDC). The patient received postoperative radiation and stayed alive at last follow-up without disease recurrence. Trastuzumab therapy may play a key role in treating the disease.
DISCUSSION Salivary duct carcinoma was first introduced in 1968 by Kleinsasser et al,5 whose histological picture strongly resembled that of breast ductal carcinoma. Generally, SDC is a highly malignant tumor and occurs most often in men older than 40 years. The most commonly involved site is the parotid gland, followed by the submandibular gland.6,7 Cases of SDC originating in sublingual gland and minor salivary gland are few, and the mandible as the site of origin is rarer. To the best of our knowledge, the current case is the third one. Salivary duct carcinoma has a characteristic of early regional lymph node metastasis. Salovaara et al8 found that 56% of their patients had pathological cervical lymph node metastasis and that the metastasis was more often in the primary tumor staged as T2 or higher. Additionally, Guzzo et al9 reported that 15 of 26 patients had pathologic nodal involvements and lymphatic spread may be related to T stage, and more, the authors found that nodal metastases could be regarded as a negative prognostic factor. Kim et al6 described that 74.3% of the patients showed pathologic nodal involvement at the time of diagnosis and that the pathologic node metastasis was correlated with distant metastasis. Salivary duct carcinoma has another characteristic of distant metastasis. The common sites of distant metastases were liver, lung, bone, brain, and skin.9 Jaehne et al7 presented that 48% of the patients developed distant disease metastasis and that the most frequency site was lung (30%). Salovaara et al8 reported that 6 of 25 patients had distant metastases and that the most common site for metastasis was bone. Salivary duct carcinoma may arise de novo or as a relatively common malignant component of a carcinoma ex pleomorphic adenoma.1 The former usually manifests as a rapidly growing mass with nerve infiltration, but the latter may have a history of a longstanding mass with recent enlargement. Previous studies have shown that there was a relatively better prognosis in carcinoma ex pleomorphic adenoma than in SDC. The differential diagnoses for SDC were metastatic squamous cell carcinoma, metastatic breast cancer, melanoma, and oncocytic carcinoma.2 Suzuki et al3 thought that the lesion, which was most difficult to distinguish from SDC, was poorly differentiated mucoepidermoid carcinoma. In the current case, all these lesions were excluded by preoperative routine examinations and histological investigation. © 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 6, November 2014
Ablative resection and postoperative radiotherapy are still the standard treatment stratagem for SDC. However, the result is not satisfactory. Salovaara8 reported their 2- and 5-year overall and disease-specific survival rates as 66%, 41% and 75, 55%. Guzzo9 described the observed 2- and 5-year overall survival rates as 42.3% and 11.5%. New treatments were needed. Nabili et al10 found that trastuzumab was effective in treating HER-2-positive SDC and concluded that given the immunohistochemical similarities between SDC and breast duct carcinoma, patients with fluorescence in situ hybridization-positive HER-2/neu should be considered for trastuzumab therapy. Similarly, Jaspers et al11 used androgen deprivation therapy to treat 10 patients with androgen receptor–positive SDC; the outcome was really promising, but the sample was small. More large prospective studies are needed.
REFERENCES 1. Thompson LD. Salivary duct carcinoma. Ear Nose Throat J 2012;91:356–359 2. Wee DT, Thomas AA, Bradley PJ. Salivary duct carcinoma: what is already known, and can we improve survival? J Laryngol Otol 2012;126:S2–S7 3. Suzuki H, Hashimoto K. Salivary duct carcinoma in the mandible: report of a case with immunohistochemical studies. Br J Oral Maxillofac Surg 1999;37:67–69
Brief Clinical Studies
4. Kikuchi Y, Hirota M, Iwai T, et al. Salivary duct carcinoma in the mandible: a case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007;103:e41–e46 5. Kleinsasser O, Klein HJ, Hübner G. Salivary duct carcinoma. A group of salivary gland tumors analogous to mammary duct carcinoma. Arch Klin Exp Ohren Nasen Kehlkopfheikd 1968;192:100–105 6. Kim JY, Lee S, Cho KJ, et al. Treatment results of post-operative radiotherapy in patients with salivary duct carcinoma of the major salivary glands. Br J Radiol 2012;85:e947–e952 7. Jaehne M, Roeser K, Jaekel T, et al. Clinical and immunohistologic typing of salivary duct carcinoma: a report of 50 cases. Cancer 2005;103:2526–2533 8. Salovaara E, Hakala O, Bäck L, et al. Management and outcome of salivary duct carcinoma in major salivary glands. Eur Arch Otorhinolaryngol 2013;270:281–285 9. Guzzo M, Di Palma S, Grandi C, et al. Salivary duct carcinoma: clinical characteristics and treatment strategies. Head Neck 1997;19:126–133 10. Nabili V, Tan JW, Bhuta S, et al. Salivary duct carcinoma: a clinical histologic review with implications for trastuzumab therapy. Head Neck 2007;29:907–912 11. Jaspers HC, Verbist BM, Schoffelen R, et al. Androgen receptor–positive salivary duct carcinoma: a disease entity with promising new treatment options. J Clin Oncol 2011;29:e473–e476
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
e599
