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伴延迟性肥大的非消退性先天性血管瘤:病例系列

Non-involuting congenital hemangioma with delayed hypertrophy: A case series.

作者信息

Konanur Anisha, Jimenez Joaquin E, Kochin Megan, McCormick Andrew, Salgado Claudia, Yilmaz Sabri, Bykowski Michael, Padia Reema

机构信息

University of Pittsburgh School of Medicine, 3550 Terrace Street, Pittsburgh, PA, 15261, United States.

Vascular Anomalies Center, UPMC Children's Hospital of Pittsburgh, 4401 Penn Ave, Pittsburgh, PA, 15224, United States.

出版信息

Int J Pediatr Otorhinolaryngol. 2022 Jun;157:111102. doi: 10.1016/j.ijporl.2022.111102. Epub 2022 Mar 12.

Abstract

BACKGROUND

Noninvoluting congenital hemangiomas (NICH) are rare and poorly understood vascular tumors that are present at birth, characterized by lack of growth after birth and lack of involution. We report uncharacteristic cases of NICH hypertrophy occurring later in life.

METHODS

This is a case series describing the clinical presentation, management, and histologic characteristics of two cases of NICH hypertrophy.

RESULTS

Two patients with a NICH of the scalp experienced lesion hypertrophy in teenage or early adult life. Case 1 is a 14-year-old female who presented with a flat left parietal scalp lesion that at first grew slowly with the patient; however, over the span of months grew substantially resulting in an exophytic lesion. The patient had the lesion surgically excised. Case 2 is a 26-year-old female with NICH of left occipital scalp and posterior neck who noted new nodules on the inferior border of the lesion. MRA/MRI showed extension into the occipital calvarium, level V of the neck, and paraspinal musculature. The patient elected to observe given the extent of the lesion and her minimal symptoms.

CONCLUSION

Although postnatal growth of NICH have been described, cases usually occur during the pre-adolescent period where growth is usually proportional to overall growth of the patient. This study describes two cases of rapid onset NICH hypertrophy occurring later in life. Knowledge of the potential for delayed hypertrophy may lead families to seek earlier intervention or opt for more definitive interventions. Additionally, recognition of these variable distinctions will contribute to a better understanding of CH and its various subtypes.

摘要

背景

非消退性先天性血管瘤(NICH)是一种罕见且了解甚少的血管肿瘤,出生时即存在,其特征为出生后无生长且无消退。我们报告了在生命后期发生的非典型NICH肥大病例。

方法

这是一个病例系列,描述了两例NICH肥大的临床表现、治疗及组织学特征。

结果

两名头皮患有NICH的患者在青少年期或成年早期出现病变肥大。病例1是一名14岁女性,其左顶叶头皮有一扁平病变,起初随患者生长缓慢;然而,在数月内显著增大,形成一个外生性病变。该患者接受了手术切除。病例2是一名26岁女性,左枕部头皮和后颈部患有NICH,她注意到病变下缘出现新结节。磁共振血管造影/磁共振成像显示病变延伸至枕骨、颈部V级及椎旁肌肉组织。鉴于病变范围及患者症状轻微,该患者选择观察。

结论

尽管已有NICH出生后生长的描述,但病例通常发生在青春期前,此时生长通常与患者的整体生长成比例。本研究描述了两例在生命后期快速发生的NICH肥大病例。了解延迟肥大的可能性可能会促使家庭寻求更早的干预或选择更确切的干预措施。此外,认识到这些不同的特征将有助于更好地理解先天性血管瘤及其各种亚型。

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