Kaiser Adeel, Spence Robert J, Parwani Anil, Tihan Tarik, Barrett Terry L
Department of Pathology, Johns Hopkins University School of Medicine, Baltimore, Maryland 21287, USA.
J Cutan Med Surg. 2003 Sep-Oct;7(5):395-8. doi: 10.1007/s10227-002-0147-9. Epub 2003 Sep 24.
Basaloid follicular hamartomas (BFH) are rare, benign, adnexal lesions with diverse clinical presentations. Previous studies documented BFHs with fibroepithelioma of Pinkus-like proliferations, or proliferations that resemble trichoepitheliomas.
We report on a patient with linear, unilateral BFH and extensive trichoblastomatous proliferations involving the right arm, torso, and leg. An 18-year-old female presented with multiple, hyperkeratotic, linear nodules and plaques limited to her right side from the shoulder to the leg. The lesions had existed since birth and gradually increased over time.
The lesions contained hyperpigmented, exophytic nodules with acanthosis, pseudoepitheliomatous hyperplasia, focally associated with hyperkeratosis, and squamous eddies. Some areas contained trichoepithelioma-like proliferations, or large nodules of basaloid cells with numerous cystic spaces, marked hyperpigmentation, and melanophages. The diagnosis was linear, unilateral BFH with an unusual trichoblastomatous component.
While trichoblastomatous proliferations could occur in a BFH, to our knowledge this finding has not been reported.
基底样毛囊错构瘤（BFH）是一种罕见的良性附属器病变，临床表现多样。既往研究记录了伴有平卡斯样纤维上皮瘤样增生或类似毛发上皮瘤增生的BFH。
我们报告1例患有线性单侧BFH且右臂、躯干和腿部出现广泛毛母细胞瘤样增生的患者。一名18岁女性，其右侧从肩部到腿部出现多个角化过度的线性结节和斑块。这些病变自出生就存在，且随时间逐渐增多。
病变包含色素沉着过度的外生性结节，伴有棘层肥厚、假上皮瘤样增生，局部伴有角化过度和鳞状涡。部分区域可见毛发上皮瘤样增生，或有大量囊腔、明显色素沉着过度及噬黑素细胞的基底样细胞大结节。诊断为伴有不寻常毛母细胞瘤成分的线性单侧BFH。
虽然毛母细胞瘤样增生可发生于BFH，但据我们所知，这一发现尚未见报道。
