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处于消退期的深部婴儿血管瘤,术前诊断困难。

Deep Infantile Hemangioma in the Involuting Phase That Was Difficult to Diagnose before Surgery.

作者信息

Sakai Yuki, Tsuge Itaru, Kataoka Masako, Takeuchi Yasuhide, Katayama Yasuhiro, Yamanaka Hiroki, Katsube Motoki, Sowa Yoshihiro, Sakamoto Michiharu, Morimoto Naoki

机构信息

From the Department of Plastic and Reconstructive Surgery, Graduate School of Medicine, Kyoto University, Kyoto, Japan.

Department of Diagnostic Imaging and Nuclear Medicine, Graduate School of Medicine, Kyoto University, Kyoto, Japan.

出版信息

Plast Reconstr Surg Glob Open. 2023 May 10;11(5):e4975. doi: 10.1097/GOX.0000000000004975. eCollection 2023 May.

DOI:10.1097/GOX.0000000000004975
PMID:37180982
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10171777/
Abstract

Infantile hemangioma (IH) is a common pediatric vascular tumor and is easily diagnosed in most cases based on the clinical course and appearance, but deep IHs are difficult to diagnose based on external appearance alone. Clinical and imaging findings are therefore important clues to the diagnosis of soft tissue tumors; however, a definitive diagnosis is decided based on the pathological examination of biopsy or resection specimens. A 1-year-old girl with a subcutaneous mass on her glabella was referred to our hospital. At 3 months of age, her mother noticed a tumor that swelled when she cried. It gradually enlarged, and ultrasonography and magnetic resonance imaging were performed at 12 months of age. Doppler ultrasonography showed a hypo-vascular mass. Magnetic resonance imaging revealed a subcutaneous mass with low-intensity on T1-weighted image and slightly high-intensity on T2-weighted image, with tiny flow voids. Computed tomography showed no frontal bone defect. The soft tissue tumor could not be diagnosed based on these imaging findings; thus, we decided to perform total resection under general anesthesia. A histopathological examination showed a highly cellular tumor with capillaries with opened small vascular channels and glucose transporter 1 positivity. Thus, it was diagnosed as deep IH transitioning from the proliferative phase to the involuting phase. Deep IHs are difficult to diagnose because characteristic imaging findings disappear during the involuting phase. We emphasize the importance of performing Doppler ultrasonography in the early phase (eg, at 6 months of age) for soft tissue tumors of infancy.

摘要

婴儿血管瘤(IH)是一种常见的儿科血管肿瘤,大多数情况下根据临床病程和外观很容易诊断,但深部IH仅根据外观很难诊断。因此,临床和影像学表现是软组织肿瘤诊断的重要线索;然而,最终诊断是基于活检或切除标本的病理检查来确定的。一名1岁女童因眉间皮下肿块被转诊至我院。3个月大时,她的母亲注意到一个肿块,孩子哭闹时会肿胀。肿块逐渐增大,12个月大时进行了超声检查和磁共振成像。多普勒超声显示为低血流肿块。磁共振成像显示皮下肿块在T1加权像上呈低信号,在T2加权像上呈稍高信号,有微小的流空信号。计算机断层扫描显示额骨无缺损。根据这些影像学表现无法诊断该软组织肿瘤;因此,我们决定在全身麻醉下进行完整切除。组织病理学检查显示为高细胞性肿瘤,有开放的小血管通道的毛细血管,葡萄糖转运蛋白1呈阳性。因此,诊断为深部IH,正从增殖期过渡到消退期。深部IH难以诊断,因为在消退期特征性的影像学表现会消失。我们强调在婴儿期软组织肿瘤的早期阶段(如6个月大时)进行多普勒超声检查的重要性。

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本文引用的文献

1
Infantile hemangioma. Part 1: Epidemiology, pathogenesis, clinical presentation and assessment.婴儿血管瘤。第 1 部分:流行病学、发病机制、临床表现和评估。
J Am Acad Dermatol. 2021 Dec;85(6):1379-1392. doi: 10.1016/j.jaad.2021.08.019. Epub 2021 Aug 19.
2
Differential diagnosis difficulties related to infantile hemangioma - case report and literature review.与婴儿血管瘤相关的鉴别诊断困难——病例报告及文献综述
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Distribution analysis of infantile hemangioma or capillary malformation on the head and face in Japanese patients.
日本患者头面部婴幼儿血管瘤或毛细血管畸形的分布分析。
J Dermatol. 2019 Oct;46(10):849-852. doi: 10.1111/1346-8138.15051. Epub 2019 Aug 16.
4
Location of infantile hemangioma is a predictor of volumetric sequelae after involution.婴儿血管瘤的位置是消退后体积后遗症的预测因素。
J Dermatol. 2019 May;46(5):371-375. doi: 10.1111/1346-8138.14847. Epub 2019 Mar 19.
5
Soft-tissue vascular malformations and tumors. Part 1: classification, role of imaging and high-flow lesions.软组织血管畸形与肿瘤。第1部分:分类、影像学作用及高流量病变
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Clinical and sonographic features of pediatric soft-tissue vascular anomalies part 1: classification, sonographic approach and vascular tumors.小儿软组织血管异常的临床和超声特征 第1部分:分类、超声检查方法及血管肿瘤
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Small, superficial, indeterminate soft-tissue lesions as suspected sarcomas: is primary excision biopsy suitable?疑似肉瘤的小而表浅的不确定软组织病变:初次切除活检是否合适?
Skeletal Radiol. 2017 Jul;46(7):919-924. doi: 10.1007/s00256-017-2635-4. Epub 2017 Mar 30.
8
Propranolol versus corticosteroids: what should be the treatment of choice in infantile hemangiomas?普萘洛尔与皮质类固醇:婴儿血管瘤的首选治疗方法应该是什么?
Ann Plast Surg. 2015 Feb;74(2):237-41. doi: 10.1097/SAP.0b013e318299cd4e.
9
Vasculogenesis in infantile hemangioma.婴儿血管瘤中的血管生成
Angiogenesis. 2009;12(2):197-207. doi: 10.1007/s10456-009-9148-2. Epub 2009 May 10.