Journal of the Egyptian National Cancer Institute (2015) xxx, xxx–xxx
Cairo University
Journal of the Egyptian National Cancer Institute www.elsevier.com/locate/jnci www.sciencedirect.com
Case Report
Small cell carcinoma of the lung in a treated case of Myoepithelial carcinoma of the tongue – Report of a rare case with illustrated review of the literature Bhanuprasad Venkatesulu, Supriya Mallick *, Archana George, Suman Bhasker Department of Radiation Oncology, All India Institute of Medical Sciences, New Delhi, India Received 16 March 2015; revised 5 May 2015; accepted 3 June 2015
KEYWORDS Myoepithelial carcinoma; Tongue; Small cell carcinoma lung
Abstract Myoepithelial carcinoma has rarely been reported in the oral cavity and oropharynx. We found only 6 cases of myoepithelioma of the tongue reported till date. Two cases had a benign myoepithelioma; four had epithelial-Myoepithelial carcinoma. The present case had malignant myoepithelioma, a distinct entity from other histologies. ª 2015 Production and hosting by Elsevier B.V. on behalf of National Cancer Institute, Cairo University. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-ncnd/4.0/).
Introduction
Case report
Myoepithelial cells are ectodermal origin cells found in secretary organs like salivary glands, breast, skin, and kidney [1,2]. Sheldon was the first to identify Myoepithelial tumours as a distinct salivary gland tumour entity [3]. Myoepitheliomas are unusual and uncommon tumours and constitute only 1–1.5% of all salivary gland tumours [4]. In the literature till now only one case of Myoepithelial carcinoma of the base of the tongue has been described. Rationale in reporting this 22 year old patient is to illustrate the diagnostic and therapeutic dilemma encountered.
A 22 year old male patient presented to our multidisciplinary head and neck cancer clinic with complaints of non healing ulcer over the posterior part of left side of the tongue for 6 months, difficulty in swallowing for 4 months, dull aching pain, non radiating for 4 months. The patient did not give any history of smoking, alcoholism, sharp teeth or oral sexual habits. He had no history of malignancy in the family. He had a performance status of ECOG-One [European cooperative oncology group]. Oral examination and laryngoscopy revealed a 4 * 3 cm sized ulcer-proliferative growth on the posterior 1/3rd of the tongue on the left side and extending to the base of the tongue. Neck node examination was found normal. A biopsy from the edge of the ulcer with normal tissue was taken and was reported as poorly differentiated carcinoma and positive for cytokeratin. Hence, a final diagnosis was achieved as carcinoma tongue, CT2 N0 M0. The patient was planned for
* Corresponding author. Tel.: +91 9899448450; fax: +91 11 26589243. E-mail address: [email protected] (S. Mallick). Peer review under responsibility of The National Cancer Institute, Cairo University.
http://dx.doi.org/10.1016/j.jnci.2015.06.002 1110-0362 ª 2015 Production and hosting by Elsevier B.V. on behalf of National Cancer Institute, Cairo University. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/). Please cite this article in press as: Venkatesulu B et al. Small cell carcinoma of the lung in a treated case of Myoepithelial carcinoma of the tongue – Report of a rare case with illustrated review of the literature, J Egyptian Nat Cancer Inst (2015), http://dx.doi.org/10.1016/j.jnci.2015.06.002
2 wide local excision and ipsilateral selective neck dissection followed by adjuvant radiotherapy. The patient underwent transoral laser excision of the left posterior 1/3rd and base of tongue tumour with supra-omohyoid neck dissection and tracheostomy under general anaesthesia. Post operative recovery was uneventful. The histopathology revealed a mass size of 4 * 3 cm. Microscopic examination revealed a poorly differentiated malignant tumour. Tumour cells were arranged in diffuse sheets. There was evidence of perineural invasion. Tumour cells were focally immune positive for cytokeratin (CK), epithelial membrane antigen (EMA), smooth muscle actin (SMA) and vimentin and negative for synaptophysin, NeuIV chromogranin leucocyte common antigen (LCA), desmin, Myogenin, and MIC-2, HMB-45, CD54, GFAP and S-100 protein (Fig. 1). Features were in favour of Myoepithelial carcinoma. None of the nine dissected lymph nodes revealed evidence of metastasis. Subsequently, the patient received postoperative radiotherapy 50 Gray in 25 fractions over 5 weeks with 5 fractions per week by the 3DCRT technique. The radiotherapy was planned in Eclipse treatment planning system with a 6 MV photon beam. The patient tolerated the treatment well with no treatment interruptions and no major toxicity reported. The patient was kept on regular follow up with clinical examination every 3 months and CECT face and neck repeated every 6 months. After three and half years of symptom free interval he presented with chest pain and breathlessness for 1 month. Chest X-ray was ordered and opacity in left upper and middle zone was found. Contract enhanced computed tomography (CECT chest and face and neck) was done which revealed postoperative changes with surgical drops at the base of the tongue and moderate pleural effusion on the left side with a partial collapse of the underlying lung showing patchy consolidation fibrosis without any evidence of mediastinal lymphadenopathy. Patient was evaluated in an pulmonary medicine clinic and advised CT guided biopsy of the lung mass. Microscopy revealed small cell carcinoma of the lung with areas of necrosis. Tumour cells were immune-positive for synaptophysin, chromogranin and focally for cytokeratin, MIB1 labelling index is approximately 90%. Subsequently he was started on chemotherapy with cisplatin 30 mg/m2 and etoposide 100 mg/m2 intravenous (IV)
B. Venkatesulu et al. day one through day three repeated every 3 weekly for six cycles. At the time of writing this case report the patient was being planned for thoracic radiation and prophylactic cranial irradiation. Discussion Squamous cell carcinomas are the most common histology of the base of the tongue with >90% of patients diagnosed with SCC. Human papilloma virus has been found to be an aetiological factor for tumours of the oropharynx. Myoepithelial carcinoma may arise from minor salivary glands of the base of the tongue. According to World Health Organization histological classification of salivary gland tumours Myoepithelial carcinoma has been recognized as a distinct entity from 1991 and accorded a morphology code of 8982/3 of the International classification of disease for oncology (ICO-O) [5]. The very low historic incidence is probably due to their recent recognition as a separate tumour entity. Myoepithelial carcinoma is known to occur in partial, submandibular, sublingual, minor salivary gland, hard palate, breast, lung, soft tissue, kidney, paranasal sinus, trachea, nasal cavity and lacrimal gland [6–8]. Myoepithelial cells exhibit 4 main cell morphologies: spindle (most common), epithelioid, plasmacytoid and clear cells (least common). Myoepithelial cells are found usually to be positive for cytokeratin (AEI/AE3, CK 5/6, Cam 52, CK-7 and CK-14) and vimentin (positive in neoplastic Myoepithelial cell and negative in normal Myoepithelial cells. Neoplastic transformation of Myoepithelial cells results in loss or modification of their smooth muscle phenotype resulting in positivity of S100 calponin, smooth muscle actin (SMA), muscle specific actin (MSA), smooth muscle myosin, p53 protein and glial fibrillary acidic protein (GFAP). Myoepithelial cells are typically negative for carcino-embryonic antigen (CEA), signifying no tubular differentiation and E-cadherin expression also detected in myoepithelioma [9–11]. Our patient was focally immune positive for cytokeratin, epithelial membrane antigen, smooth muscle actin and vimentin. Malignant myoepithelioma are a distinct entity from epithelial Myoepithelial neoplasms and should not be confused to entertain a diagnosis of malignant myoepithelioma. Cytokeratin and other Myoepithelial markers including smooth muscle actin,
Figure 1 (A) Photomicrograph showing tumour arranged in diffuse sheets and infiltrating into the skeletal muscle (10·). (B) Tumour cells show scant rim of cytoplasm, vesicular nucleus, prominent eosinophilic nucleus and moderate degree of anisonucleosis (40·) (both H&E stain).
Please cite this article in press as: Venkatesulu B et al. Small cell carcinoma of the lung in a treated case of Myoepithelial carcinoma of the tongue – Report of a rare case with illustrated review of the literature, J Egyptian Nat Cancer Inst (2015), http://dx.doi.org/10.1016/j.jnci.2015.06.002
Small cell carcinoma of lung in a treated Table 1
3
Pathological and immunochemistry details of reported cases.
S. No.
Author
Histopathology
Immunohistochemistry
Diagnostic imaging used
1
Casas et al. (2001) [12] Puri et al. (2004) [13] Kei Woo et al. (2005) [14] Kumai et al. (2006) [15] DeMatos et al. (2009) [16] Peters et al. (2010) [17] Present case (2014)
Myoepithelioma
S-100+, GFAP + Muscle specific actin+
No imaging stage NA
Epithelial Myoepithelial tumour Myoepithelioma
Cytokeratin (AE1/3) negative smooth muscle actin+, CK+ S-100+, vimentin + Leu7, p63+
Stage CT3 N0M0 (only clinical examination) Imaging used stage NA
Epithelial Myoepithelial tumour Epithelial Myoepithelial tumour Epithelial Myoepithelial carcinoma Malignant myoepithelioma
Cytokeratin+, Smooth muscle actin+, S100+ Cytokeratin + Smooth muscle actin+
Stage* CT2 N0M0 (MRI** used)
2 3 4 5 6 7 * **
P53 + CK 5/6 + CK34 + SMA + AEI/ AE3+ Cytokeratin + EMA + SMA + Vimentin+
CT1 N0M0 (MRI used) CT2 N0M0 (MRI used) CT2 N0M0 no imaging
Staging done according to the American Joint Committee on Cancer Research, AJCC 2008 (TNM staging). MRI – magnetic resonance imaging.
Table 2
Clinical features, treatment details and end status details of reported cases.
S.No.
Author
Site of lesion
Age/sex
Treatment modality
Follow up
Recurrence
1
Casas et al. (2001) [12] Puri et al. (2004) [13] Kei Woo et al. (2005) [14] Kumai et al. (2006) [15] de Matos et al. (2009) [16] Peters et al. (2010) [17] Present case (2014)
Midline cystic tongue lesion Base of tongue Midline dorsal lesion
65/M
Complete resection
10 months
No recurrence at last follow up
48/M
Neo-adjuvant Chemotherapy cisplatin, 5 fluorouracil · 2 cycles fi 66 Gy RT EL excision
14 months
No recurrence at last follow up
14 months
No recurrence at last follow up
Base of tongue Ventral surface of tongue Base of tongue Base of tongue
76/M
19 months
No recurrence at last follow up
7 years
Recurrence at 4 years, No further recurrence at 7 years
NR
No recurrence at last follow up
42 months
No recurrence but second new primary in the lung (small cell carcinoma)
2 3
4 5
6 7
19/F
48/F
60/F 22/M
Surgery (subtotal glossectomy, b/l neck dissection, and reconstruction) 1st surgery Excision biopsy
End surgery WLE RT 60 Gy/30#/ 6 weeks Transoral laser surgery + supraondyoid neck dissection fi RT 50 GY/25#
GFAP, CD10 calponin and smooth muscle myosin hearing heavy chain are required for diagnosis. We found only 6 cases of myoepithelioma of the tongue reported till date. Two cases had a benign myoepithelioma; four had epithelial-Myoepithelial carcinoma. The present case had malignant myoepithelioma, a distinct entity from other histology. The pathological and immuno-histochemistry details of the reported cases have been summarized in Table 1 [12–17]. Table 2 summarizes the clinical, treatment details and end results of the reported cases [12–17]. In the management of tumour due to its enigmatic presentation and rarity different modalities of surgery and radiochemotherapy have been applied. There was a concurrence for surgery in the form of wide local excision for 4 patients and radiotherapy was used in 3 patients. Since the present patient had the largest follow up of 42 months and in head and neck cancer maximum recurrence occured within 6 months of treatment completion, we may claim that initial
surgery followed by a radiation dose of 50 Gy could be sufficient for adequate local control. Another unusual aspect of our case was after 3 and [half] years of symptom free interval, patients developed a second new primary of small cell carcinoma of the lung [18,19]. There have been case reports of myoepithelioma of the lung. But in the present case immunohistochemistry was positive for synaptophysin, chromogranin and cytokeratin, and does not favour myoepithelioma. Whether Myoepithelial malignancy of the tongue and small cell carcinoma of the lung have really any cytogenetic or molecular association or was it just a coincidence is not known. Conclusion What is the rationale in reporting those rare cases? In a high volume tertiary referral hospital like ours, unknown cases
Please cite this article in press as: Venkatesulu B et al. Small cell carcinoma of the lung in a treated case of Myoepithelial carcinoma of the tongue – Report of a rare case with illustrated review of the literature, J Egyptian Nat Cancer Inst (2015), http://dx.doi.org/10.1016/j.jnci.2015.06.002
4 are seen more frequently with a catalogue of individual experiences and a database could be built in respect to behaviour and treatment options, so a standard surgical approach and radiation dose could be devised for this uncommon and rarely diagnosed neoplasm. Disclosures The authors have nothing to disclose. Conflict of interest The authors have no conflict of interest. References [1] Dardick I, van Nostrand AW, Phillips MJ. Histogenesis of salivary gland pleomorphic adenoma (mixed tumor) with an evaluation of the role of the myoepithelial cell. Hum Pathol 1982;13(1):62–75. [2] Batsakis JG. Myoepithelioma. Ann Otol Rhinol Laryngol 1985;94:523–4. [3] Sheldon WH. So-called mixed tumors of the salivary glands. Arch Pathol 1943;35:1–20. [4] Ellis GL, Auclair PL. Myoepithelioma. Atlas of tumor pathology: tumors of the salivary glands. 3rd ed. Washington, DC: Armed Forces Institute of Pathology; 1996. p. 57–68. [5] Seifert G, Sobin LH. Myoepithelioma. World Health Organization International histological classification of tumours: histological typing of salivary gland tumours. 2nd ed. Berlin, Germany: Springer-Verlag; 1991. p. 20–1. [6] Kumai Y, Ogata N, Yumoto E. Epithelial-myoepithelial carcinoma in the base of the tongue: a case report. Am J Otolaryngol Head Neck Surg 2006;27:58–60. [7] Chetty R. Intercalated duct hyperplasia: possible relationship to epithelial-myoepithelial carcinoma hybrid tumours of salivary gland. Histopathology 2000;37:260–3.
B. Venkatesulu et al. [8] Senis L, Sahuquillo E, Davo´ R, Hamad P, Floria LM, Baquero M. Carcinoma epitelial-mioepitelial de glaˆndulassalivales: comportamiento, diagno´stico y tratamiento. Med Oral 2002;7:391–5. [9] Dardick I, van Nostrand AW. Myoepithelial cells in salivary gland tumors – revisited. Head Neck Surg 1985;7(5):395–408. [10] Hornick JL, Fletcher CD. Cutaneous myoepithelioma: a clinicopathologic and immunohistochemical study of 14 cases. Hum Pathol 2004;35(1):14–24. [11] Dardick I, Thomas MJ, van Nostrand AW. Myoepithelioma – new concepts of histology and classification: a light and electron microscopic study. Ultrastruct Pathol 1989;13(2–3):187–224. [12] De Las Casas LE, Hoerl HD, Oberley TD, Hafez GR, Sempf JM, Shalkham JE, et al. Myoepithelioma presenting as a midline cystic lesion. Diagn Cytopathol 2001;24:403–7. [13] Puri T, Singh K, Sharma DN, Khurana. Epithelialmyoepithelial carcinoma of the base of tongue: pathology and management. Indian J Cancer 2004;41:138–40. [14] Woo VL, Angiero F, Fantasia JE. Myoepithelioma of tongue. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2005;99:581–9. [15] Kumai Y, Ogata N, Yumoto E. Epithelial-myoepithelial carcinoma in the base of the tongue: a case report. Am J Otolaryngol 2006;27:58–60. [16] Matos FR, Miranda JL, Mesquita AT, Santos CR, Freitas Rde A. Epithelial-myoepithelial carcinoma in the ventral surface of the tongue. Braz J Otorhinolaryngol 2010;76(4):540. [17] Peters P, Repanos C, Earnshaw J, Stark P, Burmeister B, McGuire L, et al. Epithelial-myoepithelial carcinoma of the tongue base: a case for the case report and review of the literature. Head Neck Oncol 2010;2:4. [18] Dardick I. Myoepithelioma: definitions and diagnostic criteria. Ultrastruct Pathol 1995;19:335–45. [19] Higashiyama M, Kodama K, Yokouchi H, Takami K, Kabuto T, Tsuji N, et al. Myoepithelioma of the lung: report of two cases and review of the literature. Lung Cancer 1998;20:47–56.
Please cite this article in press as: Venkatesulu B et al. Small cell carcinoma of the lung in a treated case of Myoepithelial carcinoma of the tongue – Report of a rare case with illustrated review of the literature, J Egyptian Nat Cancer Inst (2015), http://dx.doi.org/10.1016/j.jnci.2015.06.002
Cairo University
Journal of the Egyptian National Cancer Institute www.elsevier.com/locate/jnci www.sciencedirect.com
Case Report
Small cell carcinoma of the lung in a treated case of Myoepithelial carcinoma of the tongue – Report of a rare case with illustrated review of the literature Bhanuprasad Venkatesulu, Supriya Mallick *, Archana George, Suman Bhasker Department of Radiation Oncology, All India Institute of Medical Sciences, New Delhi, India Received 16 March 2015; revised 5 May 2015; accepted 3 June 2015
KEYWORDS Myoepithelial carcinoma; Tongue; Small cell carcinoma lung
Abstract Myoepithelial carcinoma has rarely been reported in the oral cavity and oropharynx. We found only 6 cases of myoepithelioma of the tongue reported till date. Two cases had a benign myoepithelioma; four had epithelial-Myoepithelial carcinoma. The present case had malignant myoepithelioma, a distinct entity from other histologies. ª 2015 Production and hosting by Elsevier B.V. on behalf of National Cancer Institute, Cairo University. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-ncnd/4.0/).
Introduction
Case report
Myoepithelial cells are ectodermal origin cells found in secretary organs like salivary glands, breast, skin, and kidney [1,2]. Sheldon was the first to identify Myoepithelial tumours as a distinct salivary gland tumour entity [3]. Myoepitheliomas are unusual and uncommon tumours and constitute only 1–1.5% of all salivary gland tumours [4]. In the literature till now only one case of Myoepithelial carcinoma of the base of the tongue has been described. Rationale in reporting this 22 year old patient is to illustrate the diagnostic and therapeutic dilemma encountered.
A 22 year old male patient presented to our multidisciplinary head and neck cancer clinic with complaints of non healing ulcer over the posterior part of left side of the tongue for 6 months, difficulty in swallowing for 4 months, dull aching pain, non radiating for 4 months. The patient did not give any history of smoking, alcoholism, sharp teeth or oral sexual habits. He had no history of malignancy in the family. He had a performance status of ECOG-One [European cooperative oncology group]. Oral examination and laryngoscopy revealed a 4 * 3 cm sized ulcer-proliferative growth on the posterior 1/3rd of the tongue on the left side and extending to the base of the tongue. Neck node examination was found normal. A biopsy from the edge of the ulcer with normal tissue was taken and was reported as poorly differentiated carcinoma and positive for cytokeratin. Hence, a final diagnosis was achieved as carcinoma tongue, CT2 N0 M0. The patient was planned for
* Corresponding author. Tel.: +91 9899448450; fax: +91 11 26589243. E-mail address: [email protected] (S. Mallick). Peer review under responsibility of The National Cancer Institute, Cairo University.
http://dx.doi.org/10.1016/j.jnci.2015.06.002 1110-0362 ª 2015 Production and hosting by Elsevier B.V. on behalf of National Cancer Institute, Cairo University. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/). Please cite this article in press as: Venkatesulu B et al. Small cell carcinoma of the lung in a treated case of Myoepithelial carcinoma of the tongue – Report of a rare case with illustrated review of the literature, J Egyptian Nat Cancer Inst (2015), http://dx.doi.org/10.1016/j.jnci.2015.06.002
2 wide local excision and ipsilateral selective neck dissection followed by adjuvant radiotherapy. The patient underwent transoral laser excision of the left posterior 1/3rd and base of tongue tumour with supra-omohyoid neck dissection and tracheostomy under general anaesthesia. Post operative recovery was uneventful. The histopathology revealed a mass size of 4 * 3 cm. Microscopic examination revealed a poorly differentiated malignant tumour. Tumour cells were arranged in diffuse sheets. There was evidence of perineural invasion. Tumour cells were focally immune positive for cytokeratin (CK), epithelial membrane antigen (EMA), smooth muscle actin (SMA) and vimentin and negative for synaptophysin, NeuIV chromogranin leucocyte common antigen (LCA), desmin, Myogenin, and MIC-2, HMB-45, CD54, GFAP and S-100 protein (Fig. 1). Features were in favour of Myoepithelial carcinoma. None of the nine dissected lymph nodes revealed evidence of metastasis. Subsequently, the patient received postoperative radiotherapy 50 Gray in 25 fractions over 5 weeks with 5 fractions per week by the 3DCRT technique. The radiotherapy was planned in Eclipse treatment planning system with a 6 MV photon beam. The patient tolerated the treatment well with no treatment interruptions and no major toxicity reported. The patient was kept on regular follow up with clinical examination every 3 months and CECT face and neck repeated every 6 months. After three and half years of symptom free interval he presented with chest pain and breathlessness for 1 month. Chest X-ray was ordered and opacity in left upper and middle zone was found. Contract enhanced computed tomography (CECT chest and face and neck) was done which revealed postoperative changes with surgical drops at the base of the tongue and moderate pleural effusion on the left side with a partial collapse of the underlying lung showing patchy consolidation fibrosis without any evidence of mediastinal lymphadenopathy. Patient was evaluated in an pulmonary medicine clinic and advised CT guided biopsy of the lung mass. Microscopy revealed small cell carcinoma of the lung with areas of necrosis. Tumour cells were immune-positive for synaptophysin, chromogranin and focally for cytokeratin, MIB1 labelling index is approximately 90%. Subsequently he was started on chemotherapy with cisplatin 30 mg/m2 and etoposide 100 mg/m2 intravenous (IV)
B. Venkatesulu et al. day one through day three repeated every 3 weekly for six cycles. At the time of writing this case report the patient was being planned for thoracic radiation and prophylactic cranial irradiation. Discussion Squamous cell carcinomas are the most common histology of the base of the tongue with >90% of patients diagnosed with SCC. Human papilloma virus has been found to be an aetiological factor for tumours of the oropharynx. Myoepithelial carcinoma may arise from minor salivary glands of the base of the tongue. According to World Health Organization histological classification of salivary gland tumours Myoepithelial carcinoma has been recognized as a distinct entity from 1991 and accorded a morphology code of 8982/3 of the International classification of disease for oncology (ICO-O) [5]. The very low historic incidence is probably due to their recent recognition as a separate tumour entity. Myoepithelial carcinoma is known to occur in partial, submandibular, sublingual, minor salivary gland, hard palate, breast, lung, soft tissue, kidney, paranasal sinus, trachea, nasal cavity and lacrimal gland [6–8]. Myoepithelial cells exhibit 4 main cell morphologies: spindle (most common), epithelioid, plasmacytoid and clear cells (least common). Myoepithelial cells are found usually to be positive for cytokeratin (AEI/AE3, CK 5/6, Cam 52, CK-7 and CK-14) and vimentin (positive in neoplastic Myoepithelial cell and negative in normal Myoepithelial cells. Neoplastic transformation of Myoepithelial cells results in loss or modification of their smooth muscle phenotype resulting in positivity of S100 calponin, smooth muscle actin (SMA), muscle specific actin (MSA), smooth muscle myosin, p53 protein and glial fibrillary acidic protein (GFAP). Myoepithelial cells are typically negative for carcino-embryonic antigen (CEA), signifying no tubular differentiation and E-cadherin expression also detected in myoepithelioma [9–11]. Our patient was focally immune positive for cytokeratin, epithelial membrane antigen, smooth muscle actin and vimentin. Malignant myoepithelioma are a distinct entity from epithelial Myoepithelial neoplasms and should not be confused to entertain a diagnosis of malignant myoepithelioma. Cytokeratin and other Myoepithelial markers including smooth muscle actin,
Figure 1 (A) Photomicrograph showing tumour arranged in diffuse sheets and infiltrating into the skeletal muscle (10·). (B) Tumour cells show scant rim of cytoplasm, vesicular nucleus, prominent eosinophilic nucleus and moderate degree of anisonucleosis (40·) (both H&E stain).
Please cite this article in press as: Venkatesulu B et al. Small cell carcinoma of the lung in a treated case of Myoepithelial carcinoma of the tongue – Report of a rare case with illustrated review of the literature, J Egyptian Nat Cancer Inst (2015), http://dx.doi.org/10.1016/j.jnci.2015.06.002
Small cell carcinoma of lung in a treated Table 1
3
Pathological and immunochemistry details of reported cases.
S. No.
Author
Histopathology
Immunohistochemistry
Diagnostic imaging used
1
Casas et al. (2001) [12] Puri et al. (2004) [13] Kei Woo et al. (2005) [14] Kumai et al. (2006) [15] DeMatos et al. (2009) [16] Peters et al. (2010) [17] Present case (2014)
Myoepithelioma
S-100+, GFAP + Muscle specific actin+
No imaging stage NA
Epithelial Myoepithelial tumour Myoepithelioma
Cytokeratin (AE1/3) negative smooth muscle actin+, CK+ S-100+, vimentin + Leu7, p63+
Stage CT3 N0M0 (only clinical examination) Imaging used stage NA
Epithelial Myoepithelial tumour Epithelial Myoepithelial tumour Epithelial Myoepithelial carcinoma Malignant myoepithelioma
Cytokeratin+, Smooth muscle actin+, S100+ Cytokeratin + Smooth muscle actin+
Stage* CT2 N0M0 (MRI** used)
2 3 4 5 6 7 * **
P53 + CK 5/6 + CK34 + SMA + AEI/ AE3+ Cytokeratin + EMA + SMA + Vimentin+
CT1 N0M0 (MRI used) CT2 N0M0 (MRI used) CT2 N0M0 no imaging
Staging done according to the American Joint Committee on Cancer Research, AJCC 2008 (TNM staging). MRI – magnetic resonance imaging.
Table 2
Clinical features, treatment details and end status details of reported cases.
S.No.
Author
Site of lesion
Age/sex
Treatment modality
Follow up
Recurrence
1
Casas et al. (2001) [12] Puri et al. (2004) [13] Kei Woo et al. (2005) [14] Kumai et al. (2006) [15] de Matos et al. (2009) [16] Peters et al. (2010) [17] Present case (2014)
Midline cystic tongue lesion Base of tongue Midline dorsal lesion
65/M
Complete resection
10 months
No recurrence at last follow up
48/M
Neo-adjuvant Chemotherapy cisplatin, 5 fluorouracil · 2 cycles fi 66 Gy RT EL excision
14 months
No recurrence at last follow up
14 months
No recurrence at last follow up
Base of tongue Ventral surface of tongue Base of tongue Base of tongue
76/M
19 months
No recurrence at last follow up
7 years
Recurrence at 4 years, No further recurrence at 7 years
NR
No recurrence at last follow up
42 months
No recurrence but second new primary in the lung (small cell carcinoma)
2 3
4 5
6 7
19/F
48/F
60/F 22/M
Surgery (subtotal glossectomy, b/l neck dissection, and reconstruction) 1st surgery Excision biopsy
End surgery WLE RT 60 Gy/30#/ 6 weeks Transoral laser surgery + supraondyoid neck dissection fi RT 50 GY/25#
GFAP, CD10 calponin and smooth muscle myosin hearing heavy chain are required for diagnosis. We found only 6 cases of myoepithelioma of the tongue reported till date. Two cases had a benign myoepithelioma; four had epithelial-Myoepithelial carcinoma. The present case had malignant myoepithelioma, a distinct entity from other histology. The pathological and immuno-histochemistry details of the reported cases have been summarized in Table 1 [12–17]. Table 2 summarizes the clinical, treatment details and end results of the reported cases [12–17]. In the management of tumour due to its enigmatic presentation and rarity different modalities of surgery and radiochemotherapy have been applied. There was a concurrence for surgery in the form of wide local excision for 4 patients and radiotherapy was used in 3 patients. Since the present patient had the largest follow up of 42 months and in head and neck cancer maximum recurrence occured within 6 months of treatment completion, we may claim that initial
surgery followed by a radiation dose of 50 Gy could be sufficient for adequate local control. Another unusual aspect of our case was after 3 and [half] years of symptom free interval, patients developed a second new primary of small cell carcinoma of the lung [18,19]. There have been case reports of myoepithelioma of the lung. But in the present case immunohistochemistry was positive for synaptophysin, chromogranin and cytokeratin, and does not favour myoepithelioma. Whether Myoepithelial malignancy of the tongue and small cell carcinoma of the lung have really any cytogenetic or molecular association or was it just a coincidence is not known. Conclusion What is the rationale in reporting those rare cases? In a high volume tertiary referral hospital like ours, unknown cases
Please cite this article in press as: Venkatesulu B et al. Small cell carcinoma of the lung in a treated case of Myoepithelial carcinoma of the tongue – Report of a rare case with illustrated review of the literature, J Egyptian Nat Cancer Inst (2015), http://dx.doi.org/10.1016/j.jnci.2015.06.002
4 are seen more frequently with a catalogue of individual experiences and a database could be built in respect to behaviour and treatment options, so a standard surgical approach and radiation dose could be devised for this uncommon and rarely diagnosed neoplasm. Disclosures The authors have nothing to disclose. Conflict of interest The authors have no conflict of interest. References [1] Dardick I, van Nostrand AW, Phillips MJ. Histogenesis of salivary gland pleomorphic adenoma (mixed tumor) with an evaluation of the role of the myoepithelial cell. Hum Pathol 1982;13(1):62–75. [2] Batsakis JG. Myoepithelioma. Ann Otol Rhinol Laryngol 1985;94:523–4. [3] Sheldon WH. So-called mixed tumors of the salivary glands. Arch Pathol 1943;35:1–20. [4] Ellis GL, Auclair PL. Myoepithelioma. Atlas of tumor pathology: tumors of the salivary glands. 3rd ed. Washington, DC: Armed Forces Institute of Pathology; 1996. p. 57–68. [5] Seifert G, Sobin LH. Myoepithelioma. World Health Organization International histological classification of tumours: histological typing of salivary gland tumours. 2nd ed. Berlin, Germany: Springer-Verlag; 1991. p. 20–1. [6] Kumai Y, Ogata N, Yumoto E. Epithelial-myoepithelial carcinoma in the base of the tongue: a case report. Am J Otolaryngol Head Neck Surg 2006;27:58–60. [7] Chetty R. Intercalated duct hyperplasia: possible relationship to epithelial-myoepithelial carcinoma hybrid tumours of salivary gland. Histopathology 2000;37:260–3.
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Please cite this article in press as: Venkatesulu B et al. Small cell carcinoma of the lung in a treated case of Myoepithelial carcinoma of the tongue – Report of a rare case with illustrated review of the literature, J Egyptian Nat Cancer Inst (2015), http://dx.doi.org/10.1016/j.jnci.2015.06.002
