Infantile Hemangioma with Minimal or Arrested Growth: Different Clinical Presentations in a Retrospective Case Series

Jinia El-Feghaly, Molly Marous, Heidi Bai, M. Cordisco
{"title":"Infantile Hemangioma with Minimal or Arrested Growth: Different Clinical Presentations in a Retrospective Case Series","authors":"Jinia El-Feghaly, Molly Marous, Heidi Bai, M. Cordisco","doi":"10.1097/jova.0000000000000086","DOIUrl":null,"url":null,"abstract":"\n \n Infantile hemangioma with minimal or arrested growth (IHMAG) has been established as a subtype of infantile hemangioma (IH) due to positive glucose transporter-1 staining; however, it is clinically differentiated by a proliferative component of <25% of its total surface area. IHMAG can be misdiagnosed as a capillary malformation, an IH precursor, a noninvoluting congenital hemangioma, or an arteriovenous malformation among others. In this case series, we aim to further illustrate the various clinical characteristics of IHMAG and the features that distinguish this unique entity from the other vascular entities. It is important to recognize the differentiating characteristics to avoid unnecessary evaluation and provide patients with the appropriate management plan.\n \n \n \n A retrospective chart review approved by the University of Rochester Medical Center Institutional Review Board was performed from January 2014 to December 2020, with the search terms: “infantile hemangioma with arrested or minimal growth,” “IHMAG” and “abortive hemangioma.” In total, 29 IHMAGs were identified in 27 patients. Charts were reviewed for demographic and clinical characteristics as well as workup, response to treatment, and outcomes.\n \n \n \n In total, we identified 29 IHMAGs in 27 patients. Female to male ratio was 2:1. Average gestational age was 40 weeks. Lesions were present at birth in 22/27 patients. Out of the 29 IHMAGs, 18 were focal (62%) and 11 were segmental (38%). In most patients, skin examination revealed fine telangiectatic patches with focal areas of bright red papules. Out of 29 IHMAGs, 20 involved the lower body and 18 were focal, whereas 11 were segmental. PHACES syndrome (posterior fossa anomalies, infantile hemangioma, arterial anomalies, cardiac anomalies, eye anomalies, and midline skin defects) was identified in 2 of 2 patients with facial segmental IHMAGs. LUMBAR syndrome (lower body infantile hemangiomas and other skin defects; urogenital anomalies and ulceration; myelopathy; bony deformities; anorectal malformations and arterial anomalies; and rectal anomalies) was ruled out in 2 of 2 patients with extensive IHMAGs involving the sacral area. Semicircular lipoatrophy was seen in 1 patient with segmental circumferential IHMAG of the leg. Ultrasonography was the most used modality. Out of 27 infants, 18 were treated with topical timolol, 4 were treated with oral propranolol, and 2 patients were treated with a combination of both.\n \n \n \n Trends of the epidemiologic, clinical, and prognostic data of our 27 cases are in concordance with prior reports on IHMAG, further consolidating our understanding of this peculiar entity. IHMAG typically presents as a telangiectatic pink to violaceous patch present at birth in term newborns predominantly involving the lower half of the body. It is important to keep a high index of suspicion for the other vascular anomalies in the differential diagnosis. A segmental IHMAG should prompt providers to screen for associated syndromes such as PHACES and LUMBAR. Additional studies are warranted to better elucidate the pathogenesis of IHMAG and its associated anomalies. Level of evidence: Original report (case series).\n","PeriodicalId":74008,"journal":{"name":"Journal of vascular anomalies","volume":null,"pages":null},"PeriodicalIF":0.0000,"publicationDate":"2024-04-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Journal of vascular anomalies","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1097/jova.0000000000000086","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"","JCRName":"","Score":null,"Total":0}
引用次数: 0

Abstract

Infantile hemangioma with minimal or arrested growth (IHMAG) has been established as a subtype of infantile hemangioma (IH) due to positive glucose transporter-1 staining; however, it is clinically differentiated by a proliferative component of <25% of its total surface area. IHMAG can be misdiagnosed as a capillary malformation, an IH precursor, a noninvoluting congenital hemangioma, or an arteriovenous malformation among others. In this case series, we aim to further illustrate the various clinical characteristics of IHMAG and the features that distinguish this unique entity from the other vascular entities. It is important to recognize the differentiating characteristics to avoid unnecessary evaluation and provide patients with the appropriate management plan. A retrospective chart review approved by the University of Rochester Medical Center Institutional Review Board was performed from January 2014 to December 2020, with the search terms: “infantile hemangioma with arrested or minimal growth,” “IHMAG” and “abortive hemangioma.” In total, 29 IHMAGs were identified in 27 patients. Charts were reviewed for demographic and clinical characteristics as well as workup, response to treatment, and outcomes. In total, we identified 29 IHMAGs in 27 patients. Female to male ratio was 2:1. Average gestational age was 40 weeks. Lesions were present at birth in 22/27 patients. Out of the 29 IHMAGs, 18 were focal (62%) and 11 were segmental (38%). In most patients, skin examination revealed fine telangiectatic patches with focal areas of bright red papules. Out of 29 IHMAGs, 20 involved the lower body and 18 were focal, whereas 11 were segmental. PHACES syndrome (posterior fossa anomalies, infantile hemangioma, arterial anomalies, cardiac anomalies, eye anomalies, and midline skin defects) was identified in 2 of 2 patients with facial segmental IHMAGs. LUMBAR syndrome (lower body infantile hemangiomas and other skin defects; urogenital anomalies and ulceration; myelopathy; bony deformities; anorectal malformations and arterial anomalies; and rectal anomalies) was ruled out in 2 of 2 patients with extensive IHMAGs involving the sacral area. Semicircular lipoatrophy was seen in 1 patient with segmental circumferential IHMAG of the leg. Ultrasonography was the most used modality. Out of 27 infants, 18 were treated with topical timolol, 4 were treated with oral propranolol, and 2 patients were treated with a combination of both. Trends of the epidemiologic, clinical, and prognostic data of our 27 cases are in concordance with prior reports on IHMAG, further consolidating our understanding of this peculiar entity. IHMAG typically presents as a telangiectatic pink to violaceous patch present at birth in term newborns predominantly involving the lower half of the body. It is important to keep a high index of suspicion for the other vascular anomalies in the differential diagnosis. A segmental IHMAG should prompt providers to screen for associated syndromes such as PHACES and LUMBAR. Additional studies are warranted to better elucidate the pathogenesis of IHMAG and its associated anomalies. Level of evidence: Original report (case series).
查看原文
分享 分享
微信好友 朋友圈 QQ好友 复制链接
本刊更多论文
生长缓慢或停止的婴儿血管瘤:回顾性病例系列中的不同临床表现
由于葡萄糖转运体-1 染色阳性,具有极小或生长停滞的婴儿血管瘤(IHMAG)已被确定为婴儿血管瘤(IH)的一种亚型;然而,它在临床上的区别在于其增殖成分占总表面积的比例小于 25%。IHMAG可能被误诊为毛细血管畸形、IH前兆、非膨胀性先天性血管瘤或动静脉畸形等。在本病例系列中,我们旨在进一步说明 IHMAG 的各种临床特征以及将这种独特实体与其他血管实体区分开来的特征。认识这些鉴别特征对于避免不必要的评估和为患者提供适当的治疗方案非常重要。 经罗切斯特大学医学中心机构审查委员会批准,我们在 2014 年 1 月至 2020 年 12 月期间进行了一项回顾性病历审查,搜索关键词为:"婴儿血管瘤":"生长停止或极少的婴儿血管瘤"、"IHMAG "和 "中止性血管瘤"。总共在 27 名患者中发现了 29 个 IHMAG。我们对病历进行了审查,以了解人口统计学和临床特征以及检查、治疗反应和结果。 我们总共在 27 名患者中发现了 29 个 IHMAG。男女比例为 2:1。平均孕周为 40 周。22/27例患者出生时即有病变。在 29 例 IHMAG 中,18 例为局灶性(62%),11 例为节段性(38%)。大多数患者的皮肤检查显示有细小的毛细血管扩张斑块,病灶部位有鲜红色丘疹。在 29 例 IHMAG 中,20 例累及下半身,18 例为局灶性,11 例为节段性。在 2 名面部节段性 IHMAG 患者中,有 2 人被发现患有 PHACES 综合征(后窝异常、婴儿血管瘤、动脉异常、心脏异常、眼部异常和中线皮肤缺损)。在 2 例骶骨部位有广泛 IHMAGs 的患者中,有 2 例排除了 LUMBAR 综合征(下半身婴儿血管瘤和其他皮肤缺损;泌尿生殖器畸形和溃疡;脊髓病变;骨骼畸形;肛门直肠畸形和动脉畸形;直肠畸形)。一名腿部节段性环状 IHMAG 患者出现半圆形脂肪萎缩。超声检查是最常用的方法。在 27 名婴儿中,18 人接受了局部噻吗洛尔治疗,4 人接受了口服普萘洛尔治疗,2 名患者接受了两者的联合治疗。 27 例病例的流行病学、临床和预后数据趋势与之前有关 IHMAG 的报道一致,进一步巩固了我们对这一特殊病例的认识。IHMAG 通常表现为足月儿出生时出现的毛细血管扩张性粉红色至暴发性斑块,主要累及下半身。在鉴别诊断时,必须高度怀疑其他血管异常。节段性 IHMAG 应促使医疗机构筛查相关综合征,如 PHACES 和 LUMBAR。为了更好地阐明 IHMAG 及其相关异常的发病机制,有必要进行更多的研究。证据级别:原始报告(病例系列)。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
求助全文
约1分钟内获得全文 去求助
来源期刊
自引率
0.00%
发文量
0
期刊最新文献
Identifying Genetic Mutations in Vascular Anomalies Using a Sequencing Panel for Childhood Cancers: A Pilot Study Diagnostic Pitfalls: Soft Tissue Lymphoma: Superficial Soft Tissue Lymphoma Mimicking a Venous Malformation Multidisciplinary Fusion: A recurrent expansive prevertebral vascular anomaly with EWSR1::NFATC2 fusion The Lived Experience of Patients with Vascular Malformations: A Qualitative Meta-synthesis The Economic Consequences of Sirolimus Treatment in Patients With Vascular Malformations
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
现在去查看 取消
×
提示
确定
0
微信
客服QQ
Book学术公众号 扫码关注我们
反馈
×
意见反馈
请填写您的意见或建议
请填写您的手机或邮箱
已复制链接
已复制链接
快去分享给好友吧!
我知道了
×
扫码分享
扫码分享
Book学术官方微信
Book学术文献互助
Book学术文献互助群
群 号:481959085
Book学术
文献互助 智能选刊 最新文献 互助须知 联系我们:info@booksci.cn
Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。
Copyright © 2023 Book学术 All rights reserved.
ghs 京公网安备 11010802042870号 京ICP备2023020795号-1