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小儿生殖器硬化性苔藓中的血管病变

Vascular lesions in genital lichen sclerosus in pediatric patients.

作者信息

Friedland Rivka, Ben-Amitai Dan, Didkovsky Elena, Feinmesser Meora, Zvulunov Alex

机构信息

Pediatric Dermatology Unit, Schneider Children's Medical Center of Israel, Petach Tikva, Israel.

Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel.

出版信息

Pediatr Dermatol. 2020 Sep;37(5):849-852. doi: 10.1111/pde.14277. Epub 2020 Jul 8.

DOI:10.1111/pde.14277
PMID:32638397
Abstract

BACKGROUND/OBJECTIVES: Lichen sclerosus is a rare, pruritic, mucocutaneous disease affecting mostly the anogenital area. Reports have occasionally associated lichen sclerosus with overlapping vascular lesions. This study explores this association in children.

METHODS

A retrospective study was conducted in the dermatology unit of a pediatric tertiary care medical center. Electronic medical records were searched for patients diagnosed with lichen sclerosus from 2006 to 2019. Review of the cases was performed to identify overlapping vascular lesions and review the clinical course of overlap cases.

RESULTS

Of 74 children diagnosed with lichen sclerosus during the study period, five (6.75%) had overlapping vascular lesions and genital lichen sclerosus. Four patients presented with reticular telangiectatic macules and patches (n = 4, 5.4%) that appeared at or shortly after disease onset; resolution occurred a few months after treatment initiation. The fifth patient presented with telangiectases that appeared more than 2 years after the onset of the first symptoms of lichen sclerosus (n = 1, 1.3%).

CONCLUSION

Vascular lesions in children with genital lichen sclerosus are common and have variable clinical manifestations. Early appearance of reticular macules, patches, and papules is a variant of the disease and is followed by prompt resolution of these lesions. Pathogenesis is attributed to structural changes and repositioning of the papillary vascular plexus. These changes may be alarming to parents and therefore must be recognized by physicians to prevent unnecessary concern and investigations.

摘要

背景/目的:硬化性苔藓是一种罕见的、瘙痒性的黏膜皮肤疾病,主要累及肛门生殖器区域。有报告偶尔将硬化性苔藓与重叠性血管病变相关联。本研究探讨儿童中的这种关联。

方法

在一家儿科三级医疗中心的皮肤科进行了一项回顾性研究。检索2006年至2019年诊断为硬化性苔藓的患者的电子病历。对病例进行回顾以确定重叠性血管病变,并回顾重叠病例的临床病程。

结果

在研究期间诊断为硬化性苔藓的74名儿童中,5名(6.75%)有重叠性血管病变和生殖器硬化性苔藓。4名患者出现网状毛细血管扩张性斑疹和斑片(n = 4,5.4%),在疾病发作时或发作后不久出现;在开始治疗后几个月消退。第5名患者出现的毛细血管扩张在硬化性苔藓首次症状发作2年多后出现(n = 1,1.3%)。

结论

生殖器硬化性苔藓患儿的血管病变很常见,且有多种临床表现。网状斑疹、斑片和丘疹的早期出现是该疾病的一种变体,随后这些病变会迅速消退。发病机制归因于乳头血管丛的结构变化和重新定位。这些变化可能会使家长感到担忧,因此医生必须认识到这一点,以避免不必要的担忧和检查。

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Vascular lesions in genital lichen sclerosus in pediatric patients.小儿生殖器硬化性苔藓中的血管病变
Pediatr Dermatol. 2020 Sep;37(5):849-852. doi: 10.1111/pde.14277. Epub 2020 Jul 8.
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引用本文的文献

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Comparison of lichen sclerosus in boys and girls: A systematic literature review of epidemiology, symptoms, genetic background, risk factors, treatment, and prognosis.比较男童和女童的硬化性苔藓:流行病学、症状、遗传背景、危险因素、治疗和预后的系统文献回顾。
Pediatr Dermatol. 2022 May;39(3):400-408. doi: 10.1111/pde.14967. Epub 2022 Mar 1.
2
Pediatric Vulvar Lichen Sclerosus-A Review of the Literature.儿童外阴硬化性苔藓——文献综述。
Int J Environ Res Public Health. 2021 Jul 4;18(13):7153. doi: 10.3390/ijerph18137153.