Multiple Recurrence of Trichilemmal Carcinoma of the Scalp in a Young Adult

Abstract

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.

Trichilemmal carcinoma: an unusual presentation of a rare cutaneous lesion

“…Some clinically aggressive examples of possible TC have, however, been reported. Allee et al 16 and Kulahci et al 17 reported cases of multiple tumour recurrence while Knoeller et al noted a case of lymphatic and skeletal metastasis 18. While these cases may well represent atypically aggressive cases of TC, it again raises questions over histopathological diagnostic accuracy.…”

Recurrent Proliferating Trichilemmal Tumor of the Vulva

“…Proliferating trichilemmal cyst likely originates from trichilemmal cysts and typically leads to trauma or inflammation [5]. The distribution of lesions tends to suggest that sunlight plays an important role in the development of this malignancy [11]. Most trichilemmal tumors have been known to occur in heat-bearing and/or sun-exposed areas of elderly persons, without a sex predilection [12].…”

“…Proliferating trichilemmal tumor generally has a nonaggressive course and can be treated with wide excision with the demonstration of clear margins [11]. Complete surgical excision and accurate follow-up in all cases of PTT are recommended because local relapses and metastatic spread are possible and the biological behavior of this neoplasm seems to be difficult to predict [11].…”

“…Complete surgical excision and accurate follow-up in all cases of PTT are recommended because local relapses and metastatic spread are possible and the biological behavior of this neoplasm seems to be difficult to predict [11]. Deep or perineural invasion and failure to respond to multiple surgical excisions have also been reported [16].…”

“…Malignant PTT, which must be differentiated from PTT, is a rare skin tumor that affects mainly older women and is most frequently observed on the scalp [11]. Differential diagnosis should be made on the basis of mitotic rate, cytological and architectural atypia, necrosis, stromal infiltration, and ploidy [17].…”