Imaging Sci Dent.  2018 Dec;48(4):233-244. 10.5624/isd.2018.48.4.233.

Oral carcinoma cuniculatum, an unacquainted variant of oral squamous cell carcinoma: A systematic review

Affiliations
  • 1Oral Pathology Department, Faculty of Dentistry, October 6 University, Egypt. aminafouad83@gmail.com
  • 2Oral Radiology Department, Faculty of Dentistry, Modern University for Technology and Information, Egypt.
  • 3Oral and Maxillofacial Radiology Department, Faculty of Dentistry, Cairo University, Egypt.

Abstract

PURPOSE
Oral carcinoma cuniculatum is a rare well-differentiated variant of oral squamous cell carcinoma. The purpose was to systematically review its unique features to differentiate it from other variants as verrucous carcinoma, papillary squamous cell carcinoma and well-differentiated squamous cell carcinoma.
MATERIALS AND METHODS
A systematic review was performed using MEDLINE, Dentistry and Oral Sciences Source and PubMed databases and any existing articles related to the research subject missed in the search strategy to screen ones reporting cases occurring exclusively in the oral cavity in English literature. Variables analyzed included clinical, etiologic, imaging, histopatholgical features, treatment, follow-up and survival rates.
RESULTS
From 229 hits, 17 articles with 43 cases were included in the systematic review. Clinically it showed a female predilection with pain and/or ulceration of a relatively long duration and exudation being the most common symptoms. Histologically, it showed more endophytic features comprising well-differentiated squamous epithelium with absent or minimal cytological atypia and multiple keratin filled crypts or cuniculus. Inflammatory stromal reaction and discharging abscesses were reported in most of the cases. Bone destruction was predominant in most imaging features. Complete surgical resection with a safety margin was the treatment of choice in most of the cases with few recorded recurrence cases.
CONCLUSION
Apprehensive knowledge of oral carcinoma cuniculatum unique features is essential to avoid its misdiagnosis and provide proper treatment especially for recurrent cases.

Keyword

Mouth; Carcinoma, Squamous Cell; Carcinoma, Verrucous

MeSH Terms

Abscess
Carcinoma, Squamous Cell*
Carcinoma, Verrucous
Cuniculidae
Dentistry
Diagnostic Errors
Epithelial Cells*
Epithelium
Female
Follow-Up Studies
Humans
Mouth
Recurrence
Research Subjects
Survival Rate
Ulcer

Figure

  • Fig. 1 CARE statement and checklist, 2013.

  • Fig. 2 PRISMA flowchart of studies included and excluded in each steps.


Cited by  1 articles

Management and treatment of four cases of oral carcinoma cuniculatum
Constantinos Mourouzis, Iordanis Toursounidis, Christos Eftychiadis, Georgios Rallis
J Korean Assoc Oral Maxillofac Surg. 2024;50(1):35-40.    doi: 10.5125/jkaoms.2024.50.1.35.


Reference

1. 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:ZD37–ZD39.
2. Aird I, Johnson HD, Lennox B, Stansfeld AG. Epithelioma cuniculatum a variety of squamous carcinoma peculiar to the foot. Br J Surg. 1954; 42:245–250.
Article
3. Goh GH, Venkateswaran K, Leow PC, Loh KS, Thamboo TP, Petersson F. Carcinoma cuniculatum of the esophagus and tongue: report of two cases, including TP53 mutational analysis. Head Neck Pathol. 2014; 8:261–268.
Article
4. Lawrence-Brown MM, Gollow IJ, Lam TP, Frost FA. Carcinoma cuniculatum of the abdominal wall. Med J Aust. 1984; 140:668–669.
5. Barreto JE, Velazquez EF, Ayala E, Torres J, Cubilla AL. Carcinoma cuniculatum: a distinctive variant of penile squamous cell carcinoma: report of 7 cases. Am J Surg Pathol. 2007; 31:71–75.
6. Smyth M, Jaaback K, Tabrizi SN, Garland SM, Yin H, Scurry J. Carcinoma cuniculatum of the cervix. Pathology. 2014; 46:353–355.
Article
7. Kotwal M, Poflee S, Bobhate S. Carcinoma cuniculatum at various anatomical sites. Indian J Dermatol. 2005; 50:216–220.
8. Flieger S, Owiński T. Epithelioma cuniculatum an unusual form of mouth and jaw neoplasm. Czas Stomatol. 1977; 30:395–401.
9. Sun Y, Kuyama K, Burkhardt A, Yamamoto H. Clinicopathological evaluation of carcinoma cuniculatum: a variant of oral squamous cell carcinoma. J Oral Pathol Med. 2012; 41:303–308.
Article
10. Pons Y, Kerrary S, Cox A, Guerre A, Bertolus C, Gruffaz F, et al. Mandibular cuniculatum carcinoma: apropos of 3 cases and literature review. Head Neck. 2012; 34:291–295.
Article
11. Barnes L, Eveson J, Reichart P, Sidransky D. World Health Organization classification of tumours. Pathology and genetics of head and neck tumours. Lyon: WHO IARC Press;2005. p. 163–175.
12. Devaney KO, Ferlito A, Rinaldo A, El-Naggar AK, Barnes L. Verrucous carcinoma (carcinoma cuniculatum) of the head and neck: what do we know now that we did not know a decade ago? Eur Arch Otorhinolaryngol. 2011; 268:477–480.
Article
13. Padilla RJ, Murrah 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; 118:684–693.
Article
14. Moher D, Liberati A, Tetzlaff J, Altman DG. PRISMA Group. Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. Ann Intern Med. 2009; 151:264–269.
Article
15. Gagnier JJ, Kienle G, Altman DG, Moher D, Sox H, Riley D, et al. The CARE guidelines: consensus-based clinical case report guideline development. J Clin Epidemiol. 2014; 67:46–51.
Article
16. Burkhardt A. Verrucous carcinoma and carcinoma cuniculatum - forms of squamous cell carcinoma? Hautarzt. 1986; 37:373–383.
17. 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.
18. Delahaye JF, Janser JC, Rodier JF, Auge B. Cuniculatum carcinoma. 6 cases and review of the literature. J Chir (Paris). 1994; 131:73–78.
19. 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; 99:143–148.
20. Gassler N, Helmke B, Schweigert HG, Hassfeld S, Otto HF, Flechtenmacher C. Carcinoma cuniculatum of the oral cavity. A contribution to the differential diagnosis of potentially malignant papillary lesions of mouth mucosa. Pathologe. 2002; 23:313–317.
21. Neill S, Calnan C, Rahim G. Carcinoma cuniculatum. Clin Exp Dermatol. 1984; 9:309–311.
Article
22. Lozzi GP, Peris K. Carcinoma cuniculatum. CMAJ. 2007; 177:249–251.
Article
23. Prasad SG, Kaur K, Gupta S. Carcinoma cuniculatum: a review. Indian J Dent Sci. 2012; 4:87–89.
24. Kubik MJ, Rhatigan RM. Carcinoma cuniculatum: not a verrucous carcinoma. J Cutan Pathol. 2012; 39:1083–1087.
Article
25. 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; 94:601–608.
Article
26. Heasman P, Smith D, Martin I, Soames J. Carcinoma cuniculatum presenting as a gingival lesion. Perio. 2005; 2:199–203.
27. Raguse JD, Menneking H, Scholmann HJ, Bier J. Manifestation of carcinoma cuniculatum in the mandible. Oral Oncol Extra. 2006; 42:173–175.
Article
28. Kruse AL, Graetz KW. Carcinoma cuniculatum: a rare entity in the oral cavity. J Craniofac Surg. 2009; 20:1270–1272.
29. Hutton A, McKaig S, Bardsley P, Monaghan A, Parmar S. Oral carcinoma cuniculatum in a young child. J Clin Pediatr Dent. 2010; 35:89–94.
Article
30. Sepòlveda I, Spencer L, Platin E, Bravo F. Maxillary cuniculatum carcinoma: a case report and literature review. Int J Odontostomatol. 2012; 6:281–284.
31. Suzuki J, Hashimoto S, Watanabe K, Takahashi K, Usubuchi H, Suzuki H. Carcinoma cuniculatum mimicking leukoplakia of the mandibular gingiva. Auris Nasus Larynx. 2012; 39:321–325.
Article
32. Thavaraj S, Cobb A, Kalavrezos N, Beale T, Walker DM, Jay A. Carcinoma cuniculatum arising in the tongue. Head Neck Pathol. 2012; 6:130–134.
Article
33. Fonseca FP, Pontes HA, Pontes FS, de Carvalho PL, Sena-Filho M, Jorge J, et al. Oral carcinoma cuniculatum: two cases illustrative of a diagnostic challenge. Oral Surg Oral Med Oral Pathol Oral Radiol. 2013; 116:457–463.
Article
34. Shakil M, Mohtesham I, Jode M, Prabhu V. Carcinoma cuniculatum of the oral cavity- a rare entity. J Adv Med Dent Sci. 2014; 2:124–126.
35. Shapiro MC, Wong B, O'Brien MJ, Salama A. Mandibular destruction secondary to invasion by carcinoma cuniculatum. J Oral Maxillofac Surg. 2015; 73:2343–2351.
Article
36. Shay S, Choy W, Christensen RE, St John MA. Extensive carcinoma cuniculatum of the mandible. Am J Otolaryngol. 2015; 36:446–450.
Article
37. Knobler RM, Schneider S, Neumann RA, Bodemer W, Radlwimmer B, Aberer E, et al. DNA dot-blot hybridization implicates human papillomavirus type 11-DNA in epithelioma cuniculatum. J Med Virol. 1989; 29:33–37.
Article
38. Wastiaux H, Dreno B. Recurrent cuniculatum squamous cell carcinoma of the fingers and virus. J Eur Acad Dermatol Venereol. 2008; 22:627–628.
Article
39. Chen D, Goldblum JR, Landau M, Rice TW, Pai RK, Xiao SY, et al. Semiquantitative histologic evaluation improves diagnosis of esophageal carcinoma cuniculatum on biopsy. Mod Pathol. 2013; 26:806–815.
Article
40. Goh AM, Coffill CR, Lane DP. The role of mutant p53 in human cancer. J Pathol. 2011; 223:116–126.
Article
41. Stransky N, Egloff AM, Tward AD, Kostic AD, Cibulskis K, Sivachenko A, et al. The mutational landscape of head and neck squamous cell carcinoma. Science. 2011; 333:1157–1160.
Article
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