Abstract
Carcinoma cuniculatum (CC) is a rare clinicopathologic variant of squamous cell carcinoma. Histologically, it is characterized by invasive growth of bland, acanthotic, and keratinizing squamous epithelium that forms multiple rabbit burrow-like, keratin-filled crypts and sinuses. We present a 51-year-old male smoker with CC of the left vocal cord. The tumor was staged T1a and the patient was disease-free 12 months after surgery. To our knowledge, this is the fourth case of CC of the larynx reported in the English literature and the first, due to its early diagnosis, where radical surgery was not performed. We highlight the necessity for awareness of this entity and coordination between otolaryngologists, radiologists, and pathologists for early diagnosis and organ-sparing surgical treatment.
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References
Aird I, Johnson HD, Lennox B, Stansfeld AG. Epithelioma cuniculatum: a variety of squamous carcinoma peculiar to the foot. Br J Surg. 1954;42:245–50. https://doi.org/10.1002/bjs.18004217304.
Kao GF, Graham JH, Helwig EB. Carcinoma cuniculatum (verrucous carcinoma of the skin): a clinicopathologic study of 46 cases with ultrastructural observations. Cancer. 1982;49:2395–403. https://doi.org/10.1002/1097-0142(19820601)49:11%3c2395::aid-cncr2820491129%3e3.0.co;2-3.
Datar UV, Kale A, Mane D. Oral carcinoma cuniculatum: a new entity in the clinicopathological spectrum of oral squamous cell carcinoma. J Clin Diagn Res. 2017;11:37–9. https://doi.org/10.7860/JCDR/2017/23437.9226.
Griffith CC, Goodstein LA, Tuluc M, Prestipino A, Cognetti D, Spiegel J, et al. A rare case of carcinoma cuniculatum involving the larynx in association with a saccular cyst. Head Neck Pathol. 2015;9:421–5. https://doi.org/10.1007/s12105-014-0603-z.
Flieger S, Owinski T. Epithelioma cuniculatum an unusual form of mouth and jaw neoplasm. Czas Stomatol. 1977;30:395–401.
Eisenberg E, Rosenberg B, Krutchkoff DJ. Verrucous carcinoma: a possible viral pathogenesis. Oral Surg Oral Med Oral Pathol. 1985;59:52–7. https://doi.org/10.1016/0030-4220(85)90115-x.
Kahn JL, Blez P, Gasser B, Weill-Bousson M, Vetter JM, Champy M. Carcinoma cuniculatum. Apropos of 4 cases with orofacial involvement. Rev Stomatol Chir Maxillofac. 1991;92:27–33.
Delahaye JF, Janser JC, Rodier JF, Auge B. Cuniculatum carcinoma. 6 cases and review of the literature. J Chir. 1994;131:73–8.
Petersen CS, Sjolin KE, Rosman N, Lindeberg H. Lack of human papillomavirus DNA in carcinoma cuniculatum. Acta Derm Venereol. 1994;74:231–2. https://doi.org/10.2340/0001555574231232.
Huault M, Laroche C, Levy J, Laxenaire A, Roucayrol AM, Scheffer P. Epithelioma cuniculatum. Apropos of a case in the anterior gingiva with involvement of the mandibular symphyseal bone and reconstruction using a fibular osteocutaneous flap and integrated implants. Rev Stomatol Chir Maxillofac. 1998;99:143–8.
Allon D, Kaplan I, Manor R, Calderon S. Carcinoma cuniculatum of the jaw: a rare variant of oral carcinoma. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2002;94:601–8. https://doi.org/10.1067/moe.2002.126913.
Puxeddu R, Cocco D, Parodo G, Mallarini G, Medda M, Brennan PA. Carcinoma cuniculatum of the larynx: a rare clinicopathological entity. J Laryngol Otol. 2008;122:1118–23. https://doi.org/10.1017/S0022215107000163.
Thavaraj S, Cobb A, Kalavrezos N, Beale T, Walker DM, Jay A. Carcinoma cuniculatum arising in the tongue. Head Neck Pathol. 2012;6:130–4. https://doi.org/10.1007/s12105-011-0270-2.
Sun Y, Kuyama K, Burkhardt A, Yamamot H. Clinicopathological evaluation of carcinoma cuniculatum: a variant of oral squamous cell carcinoma. J Oral Pathol Med. 2012;41:303–8.
Padilla RJ, Murrach VA. Carcinoma cuniculatum of the oral mucosa: a potentially underdiagnosed entity in the absence of clinical correlation. Oral Surg Oral Med Oral Pathol Oral Radiol. 2014;118:684–93. https://doi.org/10.1016/j.oooo.2014.08.011.
Sivapathasundhara B, Kavitha B, Padmapriya VM. Carcinoma cuniculatum of the alveolar mucosa: a rare variant of squamous cell carcinoma. Head Neck Pathol. 2019;13:652–5. https://doi.org/10.1007/s12105-018-0938-y.
Johnson N, Franceschi S, Ferlay J, et al. Squamous cell carcinoma. In: Barnes EL, Eveson JW, Reichart P, Sidransky D, editors., et al., World Health Organization classification of tumours. Pathology and genetics of head and neck tumours. Geneva: WHO/IARC Press; 2005. p. 165–73.
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Marcotullio, D., Zoccali, F., Gilardi, A. et al. Carcinoma Cuniculatum of the Larynx. Head and Neck Pathol 15, 1308–1312 (2021). https://doi.org/10.1007/s12105-020-01264-7
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DOI: https://doi.org/10.1007/s12105-020-01264-7