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平滑肌错构瘤和横纹肌错构瘤:两种罕见实体的临床病理特征及文献综述

Smooth muscle hamartoma and striated muscle hamartoma: Clinicopathologic characterization of two rare entities and literature review.

作者信息

González Iván A, Dehner Louis P

机构信息

The Lauren V. Ackerman Laboratory of Surgical Pathology, Department of Pathology and Immunology, St. Louis Children's Hospital, Washington University School of Medicine, St. Louis, Missouri, USA.

The Lauren V. Ackerman Laboratory of Surgical Pathology, Department of Pediatrics, St. Louis Children's Hospital, Washington University School of Medicine, St. Louis, Missouri, USA.

出版信息

J Cutan Pathol. 2021 Feb;48(2):237-246. doi: 10.1111/cup.13850. Epub 2020 Nov 8.

Abstract

Smooth muscle hamartoma (SMH) and striated muscle hamartoma (STH) are anomalous proliferations of smooth muscle or striated muscle, respectively, in anatomic sites where these tissues are normally present. To date, only limited cases have been reported describing these lesions. In this study, we sought to characterize the clinicopathologic features of both SMH and STH. A total of 27 cases of SMH and 12 cases of STH from 1990 to 2020 were identified. SMH cases had a slight male predominance (63%) and a mean age of presentation of 20 years (range: 4 months-91 years), with a mean size of 9.3 mm (±13.3). In contrast, STH were equally distributed in gender, with a mean age of presentation of 40 years (range: 3 months-66 years) and a mean size of 5.7 mm (±3.6). SMH were more commonly located in the torso and extremities (70%) and STH in the head and neck area (92%). One case of SMH recurred after 1.1 years and in the initial diagnosis the lesion was present at the tissue edge. None of the cases of STH had a recurrence. We present the largest cohort of SMH and STH, and report the first case of a recurrent SMH, suggesting the importance of obtaining a clean margin for these lesions.

摘要

平滑肌瘤错构瘤(SMH)和横纹肌瘤错构瘤(STH)分别是平滑肌或横纹肌在这些组织正常存在的解剖部位的异常增殖。迄今为止,仅有有限的病例报道描述了这些病变。在本研究中,我们试图描述SMH和STH的临床病理特征。共确定了1990年至2020年的27例SMH和12例STH。SMH病例男性略占优势(63%),平均发病年龄为20岁(范围:4个月至91岁),平均大小为9.3mm(±13.3)。相比之下,STH在性别上分布均匀,平均发病年龄为40岁(范围:3个月至66岁),平均大小为5.7mm(±3.6)。SMH更常见于躯干和四肢(70%),而STH多见于头颈部区域(92%)。1例SMH在1.1年后复发,初次诊断时病变位于组织边缘。STH病例均无复发。我们展示了最大规模的SMH和STH队列,并报告了首例复发性SMH,提示这些病变切缘干净的重要性。

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