The authors have indicated no significant interest with commercial supporters.T richilemmal carcinoma is a rare, cutaneous adnexal malignant tumor deriving from the outer root sheath of hair follicles. This rare cutaneous cancer usually occurs on the sun-exposed areas of the elderly, 1 although it can appear as multiple lesions on non-sun-exposed skin. 2 Clinically, it may be mistaken for squamous cell carcinoma, basal cell carcinoma, nodular melanoma, or keratoacanthoma. It has been shown to be a malignant form of a trichilemmoma. 3 Trichilemmal carcinoma generally has a nonaggressive course. Boscaino and colleagues reported that, after excision of the lesions in seven patients, there was no recurrence of the tumor within a 2-month to 4-year follow-up. 1 Trichilemmal carcinoma poorly mimics differential squamous cell carcinoma, and its biological behavior is unpredictable because it rarely can produce distant metastasis. 4 Most of the time there is a unique lesion that presents a papulonodular aspect with possible keratosis or ulceration on the top of it. Simple excision with adequate safety margin is a safe, low-cost, effective mode of treatment. 5 The appropriate treatment also includes wide resection and postoperative follow-up of the patient to facilitate early diagnosis of the recurrence and distant metastasis. 4The clinical behavior of the trichilemmal carcinoma appears to be relatively indolent, despite its cytologically malignant appearance. Local recurrence after wide excision is rare, and metastasis has not been reported. 6 It typically manifests as a single, superficial nodule measuring less than 2 cm that commonly undergoes ulceration. 7 We present here a case of local recurrence of trichilemmal carcinoma three times after excision.
Case ReportWe describe the case of a 20-year-old white man who, as of 2008, had a 5-year history of a persistent lesion on his scalp and had been operated twice for this tumor before referral to our center. The first and second operations were performed 2 years and 6 months, respectively, before the referral. He had no pathologic research report of the first excision material. The second excision material, which was taken 6 months before application to our center, was reported as trichilemmal carcinoma with involvement of the margins of resection. Upon being examined at our center, he was found to have a solitary subcutaneous exophytic, hyperkeratotic, indurated papulonodular, 3-cm-wide lesion without erythema and ulceration. He had no lymphadenopathy and no history of any other tumor or long-time sun exposure. At his first application to our center, wide surgical tumor excision and reconstruction with rotation flap was performed. The pathology result was reported as malignant trichilemmal carcinoma with tumor-free margins.