Lin Yi-Ting, Chen Chien-Ming, Yang Chih-Hsun, Chuang Yea-Huey
Department of Dermatology, Chang Gung Memorial Hospital at Taipei, Chang Gung University College of Medicine, Taoyuan, Taiwan.
Chang Gung Med J. 2012 Mar-Apr;35(2):167-77. doi: 10.4103/2319-4170.106154.
Eccrine angiomatous hamartoma (EAH) comprises a rare nevoid proliferation of normal eccrine glands and small blood vessels and occasionally other elements in the middle and deep dermis with variable clinical manifestations. Case series have rarely been published except for case reports and literature reviews. The aims of this article were to investigate the clinical and pathologic features of patients with EAH in Taiwan and to compare our results with the results of previous studies.
A retrospective review of medical records and histopathological findings was performed on patients diagnosed with EAH in a medical center in Taiwan between 1994 and 2010.
Fifteen patients with pathologically diagnosed EAH were collected. The mean age at the time of diagnosis was 38.6 years (range, birth to 67 years). The male to female ratio was 3 to 2. In most cases, EAH arose as a single lesion on a lower extremity. The symptoms and signs most commonly associated with EAH were pain (60%), hypertrichosis (13.3%), itching (13.3%) and hyperhidrosis (6.7%). Additional pathological findings included hemangioma (13.3%), verrucous hemangioma (6.7%), arteriovenous malformation (6.7%), and angiokeratoma (6.7%). None of the patients experienced spontaneous regression of the lesions before excision. Excisions were done in one patient under general anesthesia, and ten patients with local anesthesia. Four patients were kept under observation. Tumor recurrences were noted in two out of the eleven patients whose lesions were excised.
Compared with cases in the literature, we found additional histopathological findings and an increased tumor recurrence risk in our cohort. EAH remains a benign and uncommon hamartomatous condition. Further multi-center, retrospective studies with larger case numbers are needed to better characterize the disease presentation in Asian populations.
小汗腺血管瘤样错构瘤(EAH)是一种罕见的、由正常小汗腺和小血管以及偶尔真皮中层和深层的其他成分构成的痣样增生,临床表现多样。除了病例报告和文献综述外,病例系列报道很少。本文旨在研究台湾地区EAH患者的临床和病理特征,并将我们的结果与以往研究结果进行比较。
对1994年至2010年期间在台湾某医疗中心诊断为EAH的患者的病历和组织病理学检查结果进行回顾性分析。
收集了15例经病理诊断为EAH的患者。诊断时的平均年龄为38.6岁(范围为出生至67岁)。男女比例为3:2。在大多数情况下,EAH表现为下肢的单个病变。与EAH最常相关的症状和体征是疼痛(60%)、多毛症(13.3%)、瘙痒(13.3%)和多汗症(6.7%)。其他病理发现包括血管瘤(13.3%)、疣状血管瘤(6.7%)、动静脉畸形(6.7%)和血管角皮瘤(6.7%)。所有患者在切除前病变均未自发消退。1例患者在全身麻醉下进行切除,10例患者在局部麻醉下进行切除。4例患者进行观察。在11例接受病变切除的患者中,有2例出现肿瘤复发。
与文献中的病例相比,我们发现我们的队列中有更多的组织病理学发现和更高的肿瘤复发风险。EAH仍然是一种良性且罕见的错构瘤性疾病。需要进一步开展多中心、大样本量的回顾性研究,以更好地描述亚洲人群中的疾病表现。