| Summary: | Background: Acquired smooth muscle hamartoma (ASMH) is a rare benign lesion characterized clinically by hyperpigmented plaques with hypertrichosis and some follicular papules. The main histologic finding is the presence of disorganized smooth muscle bundles in the dermis. Only 25 cases of ASMH have been reported in the literature. Clinical case: We present the case of an 18-year-old male who reported a pigmented area and increased hair growth on the left hemifacial with one year of evolution. Clinically, a plaque was observed in the preauricular region and on the left cheek with a linear Blaschkoid path, consisting of hyperpigmentation, hypertrichosis, and some papular lesions, with negative pseudo-Darier sign. Histological analysis showed an increase in the number of smooth muscle bundles in the middle and deep dermis surrounding abundant sebaceous glands and numerous hair follicles in different stages of evolution. Conclusions: The sebaceous component in this lesion was prominent. Therefore, we considered this lesion part of a spectrum where the acquired smooth muscle hamartoma and folliculosebaceous cystic hamartoma are found at the extremes. This case would fall in the middle of the range, as it combines both histological features.
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