Clear cell variant of oral squamous cell carcinoma: case report and review

Mukkanwar, Rutuja Narsing; Palaskar, Sangeeta; Pawar, Rasika; Shah, Darshana Rajesh

doi:10.4322/acr.2021.388

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Clinical Case Report and Review • Autops. Case Rep. 12 • 2022 • https://doi.org/10.4322/acr.2021.388 copy

Clear cell variant of oral squamous cell carcinoma: case report and review

Authorship SCIMAGO INSTITUTIONS RANKINGS

Abstract

The clear cell variant of Oral Squamous Cell Carcinoma (OSCC) is an uncommon histological variant. Kuo first discovered it in the skin, and Frazier et al. found it in the oral cavity. We know of only nine cases of clear cell variant of OSCC reported in the literature till now. The present case is in a 60-year-old male patient with an ulcer on the left posterolateral border of the tongue. The patient had a history of chewing tobacco for 22 years. Clinical examination showed features of malignant ulcer associated with pain and discomfort. Histopathological examination revealed sheets and islands of atypical epithelial cells with clear cytoplasm, nuclear and cellular pleomorphism, and few keratin pearls in the connective tissue suggesting OSCC. Various special stains were performed to identify clear cells. Periodic Acid Schiff-Diastase (PAS-D) and Mucicarmine stains showed positive and negative reactions in clear cells, respectively. Immunohistochemical (IHC) analysis for cytokeratin (AE1/AE3) showed diffuse positivity in clear cells and other epithelial cells. Based on special stains and IHC markers, we confirmed the diagnosis as a clear cell variant of OSCC. This variant is rare and presents diagnostic challenges. It is said to be aggressive in nature. More such cases should be reported to understand its biological behavior and prognosis.

Keywords
Carcinoma, Squamous Cell; Mouth Neoplasms; Sarcoma, Clear Cell; Squamous Cell Carcinoma of Head and Neck

Content of cytoplasm	Accumulation of intra-cytoplasmatic structures	Artifactual changes in the cytoplasm
Mucopolysaccharides, immature zymogen granules	Water, glycogen, mucin	Fixation and histologic techniques
Intermediate filaments	Lipids
Paucity of cytoplasmatic organelles	Phagocytized foreign material
	Hydropic degeneration

Ref	Age/ Sex	Site and Presentation	Etiology	IHC Markers	Staining	Prognosis
Kumar et al.¹⁵15 Kumar K, Shetty DC, Wadhwan V, Gupta P. Synchronous oral squamous cell carcinomas with unusual histopathological feature. J Oral Maxillofac Pathol. 2012;16(3):420-4. http://dx.doi.org/10.4103/0973-029X.102506. PMid:23248478. http://dx.doi.org/10.4103/0973-029X.1025...	70/ F	Lobulated growth 1: Maxillary anterior region in between central incisors Lobulated growth 2: Edentulous area of mandibular right first molar	Pan chewing with areca nut, lime, and tobacco	Positive – EMA, CK8, CK18	Negative – PAS, Mucicarmine	Died within 2 months
	70/ F		Pan chewing with areca nut, lime, and tobacco	Negative – Vimentin, S-100, HMB 45	Negative – PAS, Mucicarmine	Died within 2 months
Frazier et al.¹¹11 Frazier JJ, Sacks H, Freedman PD. Primary glycogen-rich clear cell squamous cell carcinoma of the mandibular gingiva. Oral Surg Oral Med Oral Pathol Oral Radiol. 2012;114(5):e47-51. http://dx.doi.org/10.1016/j.oooo.2012.01.033. PMid:23083485. http://dx.doi.org/10.1016/j.oooo.2012.01...	59/ F	Exophytic lesion on the left mandibular gingiva extending from the buccal vestibule to floor of the mouth; Unhealed extraction socket	–	–	Positive – PAS	Lost to follow-up
	59/ F		–	–	Negative – PAS diastase, Mucicarmine	Lost to follow-up
Nainani et al.¹⁶16 Nainani P, Singh HP, Paliwal A, Nagpal N. A rare case report of clear cell variant of oral squamous cell carcinoma. J Clin Diagn Res. 2014;8(12):QD07-09. http://dx.doi.org/10.7860/JCDR/2014/11536.5339. PMid:25654003. http://dx.doi.org/10.7860/JCDR/2014/1153...	52/ M	Irregular diffuse erythematous ulcero-proliferative growth on the left side of buccal mucosa.	No tobacco/ alcohol history	Positive – CK8, CK18	Negative – PAS, Mucicarmine, Oil red O	Died within 3 months
	52/ M		No tobacco/ alcohol history	Negative – Vimentin, S-100	Negative – PAS, Mucicarmine, Oil red O	Died within 3 months
Romañach et al.¹⁷17 Romañach M, Canedo N, Cortezzi E, Abrahão A, Cabral M, Agostini M. Clear cell variant of oral squamous cell carcinoma. Oral Surg Oral Med Oral Pathol Oral Radiol. 2014;118(6):e195. http://dx.doi.org/10.1016/j.oooo.2014.05.102. http://dx.doi.org/10.1016/j.oooo.2014.05...	60/ F	Ulcerated swelling in the posterior buccal mucosa extending to the soft palate.	–	Positive – CKAE1/AE3, p63	Positive – PAS	No recurrence after 12 months of surgery
	60/ F		–	Negative – Vimentin, CD10	Positive – PAS	No recurrence after 12 months of surgery
Kaliamoorthy et al.⁵5 Kaliamoorthy S, Sethuraman V, Ramalingam SM, Arunkumar S. A rare case of clear cell variant of oral squamous cell carcinoma. J Nat Sci Biol Med. 2015;6(1):245-7. http://dx.doi.org/10.4103/0976-9668.149209. PMid:25810675. http://dx.doi.org/10.4103/0976-9668.1492...	35/ F	Non-healing ulcer involving left posterior lateral border of the tongue and lingual vestibule	No tobacco/ alcohol history	Positive – CK AE1/ AE3	Negative – PAS, Mucicarmine	–
	35/ F		No tobacco/ alcohol history	Negative – Vimentin, EMA, HMB 45.	Negative – PAS, Mucicarmine	–
Devi et al.¹⁸18 Devi A, Kamboj M, Singh V, Singh S. Clear-cell variant of squamous cell carcinoma in maxilla as primary lesion: a rare case. J Oral Maxillofac Pathol. 2017;21(3):425-8. http://dx.doi.org/10.4103/jomfp.JOMFP_180_16. PMid:29391719. http://dx.doi.org/10.4103/jomfp.JOMFP_18...	55/ M	an ulcerated swelling on Left posterior region of maxilla	–	Positive – CK, EMA	Negative – PAS, Mucicarmine	–
	55/ M	an ulcerated swelling on Left posterior region of maxilla	–	Negative – S-100, Vimentin	Negative – PAS, Mucicarmine	–
Khoury et al.¹⁹19 Khoury ZH, Bugshan A, Lubek JE, Papadimitriou JC, Basile JR, Younis RH. Glycogen-rich clear cell squamous cell carcinoma originating in the oral cavity. Head Neck Pathol. 2017;11(4):552-60. http://dx.doi.org/10.1007/s12105-017-0812-3. PMid:28342060. http://dx.doi.org/10.1007/s12105-017-081...	66/ F	Ulcerative mass of the left lateral tongue extending anteriorly to the floor of the mouth	–	Positive – Pancytokeratin, CK5/6, and p63	Positive – PAS	3 months after surgery, metastasis to left lung
	66/ F		–	Negative − S-100, Calponin, SMA	Positive – PAS	3 months after surgery, metastasis to left lung
Kakoti et al.¹⁰10 Kakoti LM, Mahanta D, Sharma JD, Chowdhury Z. Clear-cell squamous cell carcinoma: an uncommon variant of very common malignancy in the head and neck. Int J Oral Health Sci. 2018;8(2):136-9. http://dx.doi.org/10.4103/ijohs.ijohs_23_18. http://dx.doi.org/10.4103/ijohs.ijohs_23...	59/ M	Exophytic growth in the right upper jaw	–	Positive – CK	Negative – PAS	–
	59/ M	Exophytic growth in the right upper jaw	–	Negative – S-100, EMA	Negative – PAS	–
Ramani et al.¹²12 Ramani P, Gheena S, Karunagaran M, Hannah R. Clear-cell variant of oral squamous cell carcinoma: a rare entity. J Oral Maxillofac Pathol. 2021;25(4, Suppl. 1):S22-6. http://dx.doi.org/10.4103/jomfp.JOMFP_295_20. PMid:34083965. http://dx.doi.org/10.4103/jomfp.JOMFP_29...	42/ F	a soft-tissue growth with erythematous and non-scrapable irregular white patches on the left alveolar mucosa in relation to 35, 36, and 37	–	Positive – CK	Negative – PAS, Mucicarmine	Lost to follow-up
	42/ F		–	Negative – S-100, EMA, SMA, CD 117	Negative – PAS, Mucicarmine	Lost to follow-up
Index case	60/ M	Non-healing ulcer on posterolateral border of tongue	Tobacco chewing	Positive – CK	Positive – PAS diastase	Lost to follow-up
Index case	60/ M	Non-healing ulcer on posterolateral border of tongue	Tobacco chewing	Positive – CK	Negative – Mucicarmine	Lost to follow-up

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E-mail: autopsy.hu@gmail.com

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[1] Authors’ contributions: Rutuja Narsing Mukkanwar contributed to writing and editing the manuscript, Sangeeta Palaskar contributed to conceptualization, writing, designing and reviewing of manuscript. Rasika Pawar and Darshana Rajesh Shah contributed to design the manuscript, reviewing and editing of manuscript. All authors read and approved the final manuscript.