Trichoblastic carcinoma arising in trichoepithelioma in Brooke–Spiegler syndrome

Leelamma, Jayalakshmy Payippat; Sundaram, Sankar; Kandasamy, Sangeetha

doi:10.4103/ijpm.ijpm_639_17

Cited by 5 publications

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“…Their overall behaviour appears to be indolent. In contrast, high‐grade trichoblastic carcinomas present as large and often ulcerated nodular tumours, occurring sporadically or as malignant transformation of trichoepitheliomas in patients with Brooke–Spiegler syndrome 1,2,8–12 . Histopathologically, they are characterised by expansile nodular growth of undifferentiated carcinoma arising in a background of a benign trichoblastoma or trichoepithelioma.…”

Section: Discussionmentioning

confidence: 99%

“…The anatomical distribution is wide but, in contrast to previous case reports, there appears to be a predilection for the head and neck area, and the majority of tumours are sporadic. Rarely, they develop in patients with Brooke–Spiegler syndrome as malignant transformation of longstanding trichoepithliomas 1,2 . The histopathological hallmark of these tumours is the presence of an undifferentiated carcinoma within the dermis and subcutis in the background of a benign trichoblastoma or trichoepithelioma 1,2,8–11 .…”

Section: Discussionmentioning

confidence: 99%

“…Rarely, they develop in patients with Brooke–Spiegler syndrome as malignant transformation of longstanding trichoepithliomas 1,2 . The histopathological hallmark of these tumours is the presence of an undifferentiated carcinoma within the dermis and subcutis in the background of a benign trichoblastoma or trichoepithelioma 1,2,8–11 . The malignant component typically shows large expansile nodular growth with invasion of the dermis and subcutis.…”

Section: Discussionmentioning

confidence: 99%

“…Trichoblastic carcinomas are rare and poorly documented neoplasms presenting in adulthood without a sex bias. They are sporadic but may also arise in patients with Brooke–Spiegler syndrome 1,2 . Histopathologically, the tumours are characterised by malignant transformation of the epithelial component of benign hair follicle tumours, such as trichoblastoma or trichoepithelioma, and they are subdivided into morphologically low‐grade and high‐grade malignant neoplasms 3 .…”

Section: Introductionmentioning

confidence: 99%

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Morphologically high‐grade trichoblastic carcinoma: a clinicopathological study with long‐term clinical follow‐up

et al. 2021

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Aims Trichoblastic carcinomas (malignant trichoblastomas) are rare and poorly documented neoplasms characterised by malignant transformation of a pre‐existing benign trichoblastoma, and are subdivided histologically into low‐grade and high‐grade tumours. Whereas morphologically low‐grade trichoblastic carcinomas show indolent behaviour, morphologically high‐grade trichoblastic carcinomas have been associated with a poor prognosis, but little is known about their true biological potential. The aim of this study was to better define the clinicopathological features and outcomes of morphologically high‐grade trichoblastic carcinomas. Methods and results Four high‐grade trichoblastic carcinomas were retrieved from departmental files, and the clinical and histopathological features and follow‐up were recorded. The tumours presented as nodules on the scalps, necks and shoulders of adults (median age, 40 years; range, 30–55 years) with a female predominance of 3:1. Three patients had a longstanding history with recent change. Histologically, three tumours were characterised by an expansile cellular nodule composed of sheets of pleomorphic epithelioid cells with brisk mitotic activity and necrosis arising in a background of a benign trichoblastoma. One tumour showed a more gradual transition from a benign trichoepithelioma to an undifferentiated carcinoma with infiltrative growth and perineural infiltration. All patients were alive with no evidence of recurrence or metastasis following complete excision after a median follow‐up of 96 months (range, 30–180 months). Conclusions The correct diagnosis of high‐grade trichoblastic carcinoma relies on adequate sampling and recognition of the benign trichoblastic precursor lesion, i.e. trichoblastoma or trichoepithelioma. Despite the concerning histological features of the malignant component, the tumours appear to be less aggressive than previously thought.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Morphologically high‐grade trichoblastic carcinoma: a clinicopathological study with long‐term clinical follow‐up

et al. 2021

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“…Trichoblastic carcinoma (TBC), or malignant trichoblastoma, is a rare malignant adnexal neoplasm. TBC is conventionally thought to develop within pre-existing trichoblastoma [ 1 ] but can also occur within trichoepithelioma in conjunction with genodermatoses such as multiple familial trichoepitheliomas or Brooke-Spiegler syndrome [ 2 - 4 ]. In contrast to prior views, a recent retrospective review reported that most cases (87%) seemed to have occurred spontaneously, though the authors noted that pre-existing benign lesions might have gone undetected before the diagnosis of TBC [ 5 ].…”

Section: Introductionmentioning

confidence: 99%

Trichoblastic Carcinoma in the Glabella Treated With Mohs Micrographic Surgery

Park,

Crasto,

Kubicki

et al. 2024

Cureus

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Trichoblastic carcinoma (TBC) is a rare adnexal neoplasm of follicular germ cell differentiation with the potential for local invasion and metastasis. Histologic features of trichoblastic carcinoma have significant overlap with trichoblastoma and basal cell carcinoma (BCC), making diagnosis difficult in some cases. Treatment strategies are not well defined and include surgical excision for localized tumors and systemic therapies for metastatic disease. We present a case of trichoblastic carcinoma clinically resembling a benign cyst that was ultimately treated with Mohs micrographic surgery (MMS).

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