OTPOL-114; No. of Pages 3 otolaryngologia polska xxx (2013) xxx–xxx
Available online at www.sciencedirect.com
ScienceDirect journal homepage: www.elsevier.com/locate/otpol
Case report/Kazuistyka
Basaloid squamous cell carcinoma of the oropharynx: Comparison of two cases and review of the literature Sien Hui Tan 1,*, Aun Wee Chong 1, Abdul Rahman Nazarina 2, Narayanan Prepageran 1 1 2
Department of Otolaryngology, Faculty of Medicine, University Malaya, Kuala Lumpur, Malaysia Department of Pathology, Faculty of Medicine, University Malaya, Kuala Lumpur, Malaysia
article info
abstract
Article history:
We describe a rare case of BSCC in the retromolar trigone with only 3 other cases in the
Received: 09.09.2013
literature and also compare the differences between 2 cases of BSCC in terms of presen-
Accepted: 16.09.2013
tation and progression. The first patient had a markedly slower progression with painful
Available online: xxx
retromolar trigone swelling over 4 months without nodal metastases. In contrast, the
Keywords: Basaloid squamous cell carcinoma
ting extremely rapid progression. Our management was tailored accordingly with the
second patient gave a 2-week history of neck swelling with nodal involvement, indica-
Squamous cell carcinoma Retromolar trigone Tonsil Oropharynx
first patient undergoing intraoral excision of tumour and adjuvant radiotherapy while the other had bilateral neck dissection with postoperative chemoradiotherapy. Both patients achieved good outcome and are free from disease. © 2013 Polish Otorhinolaryngology - Head and Neck Surgery Society. Published by
Introduction
Elsevier Urban & Partner Sp. z o.o. All rights reserved.
Case reports Case 1
Basaloid squamous cell carcinoma (BSCC), a rare variant of squamous cell carcinoma (SCC), is believed to originate from a totipotential primitive cell in the basal layer of the surface epithelium or from salivary glands ducts [1]. To the best of our knowledge, BSCC of the retromolar trigone is extremely uncommon with only 3 cases in the literature [2–4]. We describe the fourth case of BSCC in the retromolar trigone and another case of BSCC in the tonsil, as well as compare their differences.
A 54-year-old man presented to the Otolaryngology Department with a 4 months history of painful swelling at the left retromolar trigone. He had a 60 pack-year smoking history with occasional alcohol consumption and no known illness. Clinical examination revealed an irregular tumour of the left retromolar trigone and a mobile left level I lymph node. Flexible nasal endoscopy was unremarkable. Computed tomography (CT) reported left submandibular lymph nodes
* Corresponding author at: Department of Otolaryngology, Faculty of Medicine, University Malaya, Lembah Pantai, 50603 Kuala Lumpur, Malaysia. Tel.: +60 379492062; fax: +60 379556963. E-mail address: [email protected] (S.H. Tan). 0030-6657/$ – see front matter © 2013 Polish Otorhinolaryngology - Head and Neck Surgery Society. Published by Elsevier Urban & Partner Sp. z o.o. All rights reserved.
http://dx.doi.org/10.1016/j.otpol.2013.09.003 Please cite this article in press as: Tan SH, et al. Basaloid squamous cell carcinoma of the oropharynx: Comparison of two cases and review of the literature. Otolaryngol Pol. (2013), http://dx.doi.org/10.1016/j.otpol.2013.09.003
OTPOL-114; No. of Pages 3
2
otolaryngologia polska xxx (2013) xxx–xxx
with the largest measuring 2.2 cm 1.8 cm and no bony erosion. Positron emission tomography-computed tomography (PET-CT) scan showed an active lesion at the left retromolar trigone and palate with no distant metastases. The patient underwent intraoral en bloc excision of the tumour. The histopathological report showed a moderately differentiated BSCC and fine needle aspiration (FNA) of the cervical node revealed no malignant cells. The patient was clinically staged as T4N2M0. He received adjuvant radiotherapy, and achieved a good response.
Case 2 A 61-year-old man with hypertension presented to the Otolaryngology Department with a 2 weeks history of painful right neck swelling. He was a non-smoker and drank alcohol occasionally. Clinical examination revealed an ulcerative left tonsillar mass and fixed bilateral level III lymph nodes. Flexible nasal endoscopy was unremarkable. CT reported multiple bilateral enlarged cervical lymph nodes, with the largest on the left side measuring 2.3 cm 2 cm and the right, measuring 2.8 cm 2 cm. There was no bony erosion or metastases. The histopathological report showed a moderately differentiated BSCC and FNA of the cervical nodes revealed dysplastic squamous epithelium. The patient was clinically staged as T1N2cM0. He underwent bilateral neck dissection with postoperative chemoradiotherapy, and achieved a good outcome.
Discussion BSCC, first described by Wain et al. [1], is found predominantly in men aged between 60 to 80 years. Tobacco and alcohol abuse are significant risk factors. BSCC has a strong predilection for the pyriform sinus, base of tongue, tonsil and supraglottic larynx. The 2005 WHO blue book [5] defined BSCC as a high grade, variant of SCC composed of both basaloid and squamous component. There is frequent comedo-type necrosis (Fig. 1) and prominent peripheral palisading (Fig. 2). However, histological diagnosis can be challenging and immunochemistry is required for accurate diagnosis. BerEP4 (Fig. 3), which was used in our institution, is a valuable diagnostic marker with an overall accuracy of up to 77% [6]. The differential diagnosis of BSCC includes adenoid cystic carcinoma, adenosquamous carcinoma and neuroendocrine carcinoma. Nevertheless, there are several distinguishing features to differentiate BSCC from the rest. Adenoid cytic carcinoma has a myoepithelial component and lacks squamous differentiation whereas true ducto-glandular differentiation and intracellular mucin are found only in adenosquamous carcinoma [5]. Neuroendocrine markers such as chromogranin, synaptophysin, muscle-specific actin and glial fibrillary acid protein help discern BSCC from neuroendocrine carcinoma [5]. Interestingly, there were dissimilarities in our described cases. The first patient had a markedly slower progression over 4 months without nodal metastases. In contrast, the
Fig. 1 – Comedonecrosis with basaloid and squamous admixtures (haematoxylin eosin, original magnification 40T)
second patient gave a 2-week history, indicating extremely rapid progression. Although BSCC is reported as an aggressive tumour with lymph nodes involvement and metastases, we should note that each case is unique and may not follow a typical course. There is ongoing debate over the prognosis of BSCC compared to SCC. Some studies [1, 7] described a poorer prognosis for BSCC whereas others reported similar outcomes for both [8, 9]. Intriguingly, there is evidence [8] to suggest that human papilloma virus (HPV) is a crucial prognostic factor for oropharyngeal BSCC signifying a better prognosis. Aggressive multimodality treatment is usually recommended with a combination of surgery, radiotherapy and chemotherapy. However, recent literature [10] has emphasized the propensity for HPV-positive BSCC in the oropharynx and the radiosensitivity of HPV-positive disease. Our management was tailored accordingly with the first patient
Fig. 2 – Focal peripheral palisading (arrow) (haematoxylin eosin, original magnification 40T)
Please cite this article in press as: Tan SH, et al. Basaloid squamous cell carcinoma of the oropharynx: Comparison of two cases and review of the literature. Otolaryngol Pol. (2013), http://dx.doi.org/10.1016/j.otpol.2013.09.003
OTPOL-114; No. of Pages 3 otolaryngologia polska xxx (2013) xxx–xxx
3
Association (Declaration of Helsinki) for experiments involving humans; EU Directive 2010/63/EU for animal experiments; Uniform Requirements for manuscripts submitted to Biomedical journals.
references/pi smiennictwo
Fig. 3 – Ber-EP4 expression in BSCC (haematoxylin eosin, original magnification 100T)
receiving radiotherapy while the other underwent neck dissection with chemoradiotherapy. In summary, BSCC of the oropharynx can vary in terms of presentation and progression. We must consider these factors and not merely adopt a uniformly aggressive approach.
Authors' contributions/Wkład autorów According to order.
Conflict of interest/Konflikt interesu None declared.
Financial support/Finansowanie None declared.
Ethics/Etyka The work described in this article has been carried out in accordance with The Code of Ethics of the World Medical
[1] Wain SL, Kier R, Vollmer RT, Bossen EH. Basaloid-squamous carcinoma of the tongue, hypopharynx and larynx: report of 10 cases. Hum Pathol 1986;17(11):1158–1166. [2] Campos MS, Modolo F, De Oliveira JS, Pinto-Junior DS, De Sousa SC. Atypical presentation of oral basaloid squamous cell carcinoma. J Contemp Dent Pract 2009;10 (2):98–104. [3] Rachel JR, Kumar NS, Jain NS. Basaloid squamous cell carcinoma of retromolar trigone: a case report with review of the literature. J Oral Maxillofac Pathol 2011;15(2): 192–196. [4] Jain K, Kaur H, Madhushankari GS. Basaloid squamous cell carcinoma in retromolar ridge area: a rare case report. Int J Oral Maxillofac Pathol 2011;2(3):27–31. [5] Cardesa A, Zidar N, Ereno C. Basaloid squamous cell carcinoma. In: Barnes L, Eveson JW, Reichart P, Sidransky D, editors. Pathology and genetics of head and neck tumours. Kleihues P, Sobon LH, series editors. World Health Organization classification of tumours. Lyon, France: IARC Press; 2005. [6] Winters R, Naud S, Evans MF, Trotman W, Kasznica P, Elhosseiny A. Ber-EP4, CK1, CK7 and CK14 are useful markers for basaloid squamous carcinoma: a study of 45 cases. Head Neck Pathol 2008;2:265–271. [7] Bahar G, Feinmesser R, Popovtzer A, Ulanovsky D, Nageris B, Marshak G, et al. Basaloid squamous carcinoma of the larynx. Am J Otolaryngol 2003;24:204–208. [8] Banks ER, Frierson Jr HF, Mills SE, George E, Zarbo RJ, Swanson PE. Basaloid squamous cell carcinoma of the head and neck: a clinicopathologic and immunohistochemical study of 40 cases. Am J Surg Pathol 1992;16:939–946. [9] Woolgar JA, Lewis Jr JS, Devaney KO, Rinaldo A, Coskun HH, Takes RT, et al. Basaloid squamous cell carcinoma of the upper aerodigestive tract: a single squamous cell carcinoma subtype or two distinct entities hiding under one histologic pattern? Eur Arch Otorhinolaryngol 2011; 268:161–164. [10] Chernock RD, Lewis Jr JS, Zhang Q, El-Mofty SK. Human papillomavirus-positive basaloid squamous cell carcinomas of the upper aerodigestive tract: a distinct clinicopathologic and molecular subtype of basaloid squamous cell carcinoma. Hum Pathol 2010;41: 1016–1023.
Please cite this article in press as: Tan SH, et al. Basaloid squamous cell carcinoma of the oropharynx: Comparison of two cases and review of the literature. Otolaryngol Pol. (2013), http://dx.doi.org/10.1016/j.otpol.2013.09.003
Available online at www.sciencedirect.com
ScienceDirect journal homepage: www.elsevier.com/locate/otpol
Case report/Kazuistyka
Basaloid squamous cell carcinoma of the oropharynx: Comparison of two cases and review of the literature Sien Hui Tan 1,*, Aun Wee Chong 1, Abdul Rahman Nazarina 2, Narayanan Prepageran 1 1 2
Department of Otolaryngology, Faculty of Medicine, University Malaya, Kuala Lumpur, Malaysia Department of Pathology, Faculty of Medicine, University Malaya, Kuala Lumpur, Malaysia
article info
abstract
Article history:
We describe a rare case of BSCC in the retromolar trigone with only 3 other cases in the
Received: 09.09.2013
literature and also compare the differences between 2 cases of BSCC in terms of presen-
Accepted: 16.09.2013
tation and progression. The first patient had a markedly slower progression with painful
Available online: xxx
retromolar trigone swelling over 4 months without nodal metastases. In contrast, the
Keywords: Basaloid squamous cell carcinoma
ting extremely rapid progression. Our management was tailored accordingly with the
second patient gave a 2-week history of neck swelling with nodal involvement, indica-
Squamous cell carcinoma Retromolar trigone Tonsil Oropharynx
first patient undergoing intraoral excision of tumour and adjuvant radiotherapy while the other had bilateral neck dissection with postoperative chemoradiotherapy. Both patients achieved good outcome and are free from disease. © 2013 Polish Otorhinolaryngology - Head and Neck Surgery Society. Published by
Introduction
Elsevier Urban & Partner Sp. z o.o. All rights reserved.
Case reports Case 1
Basaloid squamous cell carcinoma (BSCC), a rare variant of squamous cell carcinoma (SCC), is believed to originate from a totipotential primitive cell in the basal layer of the surface epithelium or from salivary glands ducts [1]. To the best of our knowledge, BSCC of the retromolar trigone is extremely uncommon with only 3 cases in the literature [2–4]. We describe the fourth case of BSCC in the retromolar trigone and another case of BSCC in the tonsil, as well as compare their differences.
A 54-year-old man presented to the Otolaryngology Department with a 4 months history of painful swelling at the left retromolar trigone. He had a 60 pack-year smoking history with occasional alcohol consumption and no known illness. Clinical examination revealed an irregular tumour of the left retromolar trigone and a mobile left level I lymph node. Flexible nasal endoscopy was unremarkable. Computed tomography (CT) reported left submandibular lymph nodes
* Corresponding author at: Department of Otolaryngology, Faculty of Medicine, University Malaya, Lembah Pantai, 50603 Kuala Lumpur, Malaysia. Tel.: +60 379492062; fax: +60 379556963. E-mail address: [email protected] (S.H. Tan). 0030-6657/$ – see front matter © 2013 Polish Otorhinolaryngology - Head and Neck Surgery Society. Published by Elsevier Urban & Partner Sp. z o.o. All rights reserved.
http://dx.doi.org/10.1016/j.otpol.2013.09.003 Please cite this article in press as: Tan SH, et al. Basaloid squamous cell carcinoma of the oropharynx: Comparison of two cases and review of the literature. Otolaryngol Pol. (2013), http://dx.doi.org/10.1016/j.otpol.2013.09.003
OTPOL-114; No. of Pages 3
2
otolaryngologia polska xxx (2013) xxx–xxx
with the largest measuring 2.2 cm 1.8 cm and no bony erosion. Positron emission tomography-computed tomography (PET-CT) scan showed an active lesion at the left retromolar trigone and palate with no distant metastases. The patient underwent intraoral en bloc excision of the tumour. The histopathological report showed a moderately differentiated BSCC and fine needle aspiration (FNA) of the cervical node revealed no malignant cells. The patient was clinically staged as T4N2M0. He received adjuvant radiotherapy, and achieved a good response.
Case 2 A 61-year-old man with hypertension presented to the Otolaryngology Department with a 2 weeks history of painful right neck swelling. He was a non-smoker and drank alcohol occasionally. Clinical examination revealed an ulcerative left tonsillar mass and fixed bilateral level III lymph nodes. Flexible nasal endoscopy was unremarkable. CT reported multiple bilateral enlarged cervical lymph nodes, with the largest on the left side measuring 2.3 cm 2 cm and the right, measuring 2.8 cm 2 cm. There was no bony erosion or metastases. The histopathological report showed a moderately differentiated BSCC and FNA of the cervical nodes revealed dysplastic squamous epithelium. The patient was clinically staged as T1N2cM0. He underwent bilateral neck dissection with postoperative chemoradiotherapy, and achieved a good outcome.
Discussion BSCC, first described by Wain et al. [1], is found predominantly in men aged between 60 to 80 years. Tobacco and alcohol abuse are significant risk factors. BSCC has a strong predilection for the pyriform sinus, base of tongue, tonsil and supraglottic larynx. The 2005 WHO blue book [5] defined BSCC as a high grade, variant of SCC composed of both basaloid and squamous component. There is frequent comedo-type necrosis (Fig. 1) and prominent peripheral palisading (Fig. 2). However, histological diagnosis can be challenging and immunochemistry is required for accurate diagnosis. BerEP4 (Fig. 3), which was used in our institution, is a valuable diagnostic marker with an overall accuracy of up to 77% [6]. The differential diagnosis of BSCC includes adenoid cystic carcinoma, adenosquamous carcinoma and neuroendocrine carcinoma. Nevertheless, there are several distinguishing features to differentiate BSCC from the rest. Adenoid cytic carcinoma has a myoepithelial component and lacks squamous differentiation whereas true ducto-glandular differentiation and intracellular mucin are found only in adenosquamous carcinoma [5]. Neuroendocrine markers such as chromogranin, synaptophysin, muscle-specific actin and glial fibrillary acid protein help discern BSCC from neuroendocrine carcinoma [5]. Interestingly, there were dissimilarities in our described cases. The first patient had a markedly slower progression over 4 months without nodal metastases. In contrast, the
Fig. 1 – Comedonecrosis with basaloid and squamous admixtures (haematoxylin eosin, original magnification 40T)
second patient gave a 2-week history, indicating extremely rapid progression. Although BSCC is reported as an aggressive tumour with lymph nodes involvement and metastases, we should note that each case is unique and may not follow a typical course. There is ongoing debate over the prognosis of BSCC compared to SCC. Some studies [1, 7] described a poorer prognosis for BSCC whereas others reported similar outcomes for both [8, 9]. Intriguingly, there is evidence [8] to suggest that human papilloma virus (HPV) is a crucial prognostic factor for oropharyngeal BSCC signifying a better prognosis. Aggressive multimodality treatment is usually recommended with a combination of surgery, radiotherapy and chemotherapy. However, recent literature [10] has emphasized the propensity for HPV-positive BSCC in the oropharynx and the radiosensitivity of HPV-positive disease. Our management was tailored accordingly with the first patient
Fig. 2 – Focal peripheral palisading (arrow) (haematoxylin eosin, original magnification 40T)
Please cite this article in press as: Tan SH, et al. Basaloid squamous cell carcinoma of the oropharynx: Comparison of two cases and review of the literature. Otolaryngol Pol. (2013), http://dx.doi.org/10.1016/j.otpol.2013.09.003
OTPOL-114; No. of Pages 3 otolaryngologia polska xxx (2013) xxx–xxx
3
Association (Declaration of Helsinki) for experiments involving humans; EU Directive 2010/63/EU for animal experiments; Uniform Requirements for manuscripts submitted to Biomedical journals.
references/pi smiennictwo
Fig. 3 – Ber-EP4 expression in BSCC (haematoxylin eosin, original magnification 100T)
receiving radiotherapy while the other underwent neck dissection with chemoradiotherapy. In summary, BSCC of the oropharynx can vary in terms of presentation and progression. We must consider these factors and not merely adopt a uniformly aggressive approach.
Authors' contributions/Wkład autorów According to order.
Conflict of interest/Konflikt interesu None declared.
Financial support/Finansowanie None declared.
Ethics/Etyka The work described in this article has been carried out in accordance with The Code of Ethics of the World Medical
[1] Wain SL, Kier R, Vollmer RT, Bossen EH. Basaloid-squamous carcinoma of the tongue, hypopharynx and larynx: report of 10 cases. Hum Pathol 1986;17(11):1158–1166. [2] Campos MS, Modolo F, De Oliveira JS, Pinto-Junior DS, De Sousa SC. Atypical presentation of oral basaloid squamous cell carcinoma. J Contemp Dent Pract 2009;10 (2):98–104. [3] Rachel JR, Kumar NS, Jain NS. Basaloid squamous cell carcinoma of retromolar trigone: a case report with review of the literature. J Oral Maxillofac Pathol 2011;15(2): 192–196. [4] Jain K, Kaur H, Madhushankari GS. Basaloid squamous cell carcinoma in retromolar ridge area: a rare case report. Int J Oral Maxillofac Pathol 2011;2(3):27–31. [5] Cardesa A, Zidar N, Ereno C. Basaloid squamous cell carcinoma. In: Barnes L, Eveson JW, Reichart P, Sidransky D, editors. Pathology and genetics of head and neck tumours. Kleihues P, Sobon LH, series editors. World Health Organization classification of tumours. Lyon, France: IARC Press; 2005. [6] Winters R, Naud S, Evans MF, Trotman W, Kasznica P, Elhosseiny A. Ber-EP4, CK1, CK7 and CK14 are useful markers for basaloid squamous carcinoma: a study of 45 cases. Head Neck Pathol 2008;2:265–271. [7] Bahar G, Feinmesser R, Popovtzer A, Ulanovsky D, Nageris B, Marshak G, et al. Basaloid squamous carcinoma of the larynx. Am J Otolaryngol 2003;24:204–208. [8] Banks ER, Frierson Jr HF, Mills SE, George E, Zarbo RJ, Swanson PE. Basaloid squamous cell carcinoma of the head and neck: a clinicopathologic and immunohistochemical study of 40 cases. Am J Surg Pathol 1992;16:939–946. [9] Woolgar JA, Lewis Jr JS, Devaney KO, Rinaldo A, Coskun HH, Takes RT, et al. Basaloid squamous cell carcinoma of the upper aerodigestive tract: a single squamous cell carcinoma subtype or two distinct entities hiding under one histologic pattern? Eur Arch Otorhinolaryngol 2011; 268:161–164. [10] Chernock RD, Lewis Jr JS, Zhang Q, El-Mofty SK. Human papillomavirus-positive basaloid squamous cell carcinomas of the upper aerodigestive tract: a distinct clinicopathologic and molecular subtype of basaloid squamous cell carcinoma. Hum Pathol 2010;41: 1016–1023.
Please cite this article in press as: Tan SH, et al. Basaloid squamous cell carcinoma of the oropharynx: Comparison of two cases and review of the literature. Otolaryngol Pol. (2013), http://dx.doi.org/10.1016/j.otpol.2013.09.003
