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一例罕见的伴有嗜酸性粒细胞增多的血管淋巴样增生症及一种新的有效治疗方法

A Rare Case of Angiolymphoid Hyperplasia With Eosinophilia With a New Effective Treatment.

作者信息

Hasan Unaiza, Ahmed Najia, Malik Tariq, Shah Syed Arbab, Subhan Uroosa

机构信息

Dermatology, Pakistan Navy Station (PNS) Shifa Hospital, Karachi, PAK.

Dermatology, Bahria University of Health Sciences, Pakistan Navy Station (PNS) Shifa Hospital, Karachi, PAK.

出版信息

Cureus. 2023 Jun 5;15(6):e39966. doi: 10.7759/cureus.39966. eCollection 2023 Jun.

DOI:10.7759/cureus.39966
PMID:37415992
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10320733/
Abstract

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a benign locally proliferating lesion of unknown etiology, composed of vascular channels lined by endothelial cells, surrounded by lymphocytes and eosinophils. It presents clinically as a cluster of skin to violaceous-colored nodules on the head and neck, particularly in and around the ear. We present the case of a 50-year-old, Pakistani woman with unilateral multiple nodular lesions for eight years in the left ear concha and postauricular area causing complete obliteration of the external auditory meatus with conductive hearing loss of the left ear for seven years. Biopsy showed lymphoid follicles and dilated blood vessels with mixed infiltrate predominantly eosinophils corresponding to the diagnosis of angiolymphoid hyperplasia with eosinophilia. Surgical excision was not feasible, and there was no response to topical steroids. The patient was started on beta blockers. After three months, postauricular lesions completely resolved, and the size of the rest of the nodules decreased markedly; then hearing loss also recovered. Our objective in this study is to emphasize the importance of considering beta blockers for the treatment of ALHE.

摘要

嗜酸性粒细胞增多性血管淋巴样增生(ALHE)是一种病因不明的良性局部增生性病变,由内皮细胞衬里的血管通道组成,周围有淋巴细胞和嗜酸性粒细胞。临床上表现为头颈部出现一簇皮肤至紫蓝色结节,尤其是耳部及其周围。我们报告一例50岁的巴基斯坦女性病例,其左耳甲腔和耳后区域出现单侧多发性结节性病变8年,导致外耳道完全闭塞,左耳传导性听力损失7年。活检显示淋巴滤泡和扩张的血管,伴有以嗜酸性粒细胞为主的混合浸润,符合嗜酸性粒细胞增多性血管淋巴样增生的诊断。手术切除不可行,局部使用类固醇也无反应。患者开始使用β受体阻滞剂治疗。三个月后,耳后病变完全消退,其余结节大小明显减小;随后听力损失也恢复了。我们在本研究中的目的是强调考虑使用β受体阻滞剂治疗ALHE的重要性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2a82/10320733/a77c014a694b/cureus-0015-00000039966-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2a82/10320733/ec0adf836254/cureus-0015-00000039966-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2a82/10320733/baa816643e59/cureus-0015-00000039966-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2a82/10320733/a77c014a694b/cureus-0015-00000039966-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2a82/10320733/ec0adf836254/cureus-0015-00000039966-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2a82/10320733/baa816643e59/cureus-0015-00000039966-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2a82/10320733/a77c014a694b/cureus-0015-00000039966-i03.jpg

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Successful treatment of angiolymphoid hyperplasia with eosinophilia with oral propranolol in two cases.
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