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A distinct entity

The lesion looked like a wart but was, in fact, something far more unusual
Published:October 19, 2012DOI:https://doi.org/10.1016/j.ajog.2012.10.864

      Case notes

      A 74-year-old woman presented with a 9-month history of a white slow-growing warty lesion on her vulva (Figure 1). She reported mild pruritus, but she had not experienced any significant pain or lower urinary tract symptoms. Several antiwart creams were prescribed but none produced a response. A deep surgical biopsy led to a final diagnosis of carcinoma cuniculatum (CC) (FIGURE 2, FIGURE 3).
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      Figure thumbnail gr1
      FIGURE 1The patient had a white slow-growing warty lesion on her vulva.
      El Ahmed. Lesion looked like a wart, but was something far more unusual. Am J Obstet Gynecol 2013.
      Figure thumbnail gr2
      FIGURE 2Deep biopsy of the lesion showing the proliferative deep infiltration of the Malpighian layer. Hematoxylin and eosin ×4.
      El Ahmed. Lesion looked like a wart, but was something far more unusual. Am J Obstet Gynecol 2013.
      Figure thumbnail gr3
      FIGURE 3Characteristic tunnels of this condition give the shape of rabbit burrows. Hematoxylin and eosin ×10.
      El Ahmed. Lesion looked like a wart, but was something far more unusual. Am J Obstet Gynecol 2013.

      Conclusions

      CC is a rare subtype of squamous cell or epidermoid carcinoma, which presents clinically as a slow-growing verrucous exophytic mass with local malignancy and chronic inflammation. It usually occurs in the oral cavity, the genital area, or on the sole of the foot. Lymph node involvement is uncommon. The etiology of this tumor is not well defined, and diagnosis is particularly difficult, requiring multiple biopsies and close clinical correlation before a definite conclusion can be reached.
      • Delahaye J.F.
      • Janser J.C.
      • Rodier J.F.
      • Auge B.
      Cuniculatum carcinoma 6 cases and review of the literature.
      Histologic assessment shows proliferative deep infiltration of the Malpighian layer with an aspect of normal cell maturation. Tunnels are hollowed out, producing the shape of rabbit burrows (cuniculatum).
      The nomenclature and definition of CC is not uniform, and although some authors have equated it with verrucous carcinoma, it is actually a distinct entity. CC displays hybrid features of squamous cell carcinoma and verrucous carcinoma. In contrast to verrucous carcinoma, CC has at least focally jagged borders, as well as a propensity to invade more deeply, involving the corporal bodies. Unlike squamous cell carcinoma, CC is rarely associated with lymphatic or distant metastases, despite deep invasion of the skin.
      • Lau P.
      • Li Chang H.H.
      • Gomez J.A.
      • Erdeljan P.
      • Srigley J.R.
      • Izawa J.I.
      A rare case of carcinoma cuniculatum of the penis in a 55-year-old.
      Patients with CC are managed with surgery, usually without lymph node excision. Radiotherapy is contraindicated because it has been associated with anaplastic transformation in patients with CC.
      • Sun Y.
      • Kuyama K.
      • Burkhardt A.
      • Yamamoto H.
      Clinicopathological evaluation of carcinoma cuniculatum: a variant of oral squamous cell carcinoma.
      The patient underwent radical vulvectomy, which produced margins that were free of disease. She is followed-up on a regular basis. No recurrence of the disease has been observed.

      References

        • Delahaye J.F.
        • Janser J.C.
        • Rodier J.F.
        • Auge B.
        Cuniculatum carcinoma.
        J Chir (Paris). 1994; 131: 73-78
        • Lau P.
        • Li Chang H.H.
        • Gomez J.A.
        • Erdeljan P.
        • Srigley J.R.
        • Izawa J.I.
        A rare case of carcinoma cuniculatum of the penis in a 55-year-old.
        Can Urol Assoc J. 2010; 5: E129-E132
        • 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