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儿童头颈部脂肪母细胞瘤:病例报告及系统回顾。

Head and neck lipoblastoma in children: A case report and systematic review.

机构信息

Children's Hospital of Bydgoszcz, Department of Otolaryngology, Audiology and Phoniatrics, Pediatric Cochlear Implant Program, Bydgoszcz, Poland.

Children's Hospital of Bydgoszcz, Department of Otolaryngology, Audiology and Phoniatrics, Pediatric Cochlear Implant Program, Bydgoszcz, Poland; University Clinical Center, Gdańsk, Poland.

出版信息

Int J Pediatr Otorhinolaryngol. 2023 Oct;173:111699. doi: 10.1016/j.ijporl.2023.111699. Epub 2023 Aug 16.

DOI:10.1016/j.ijporl.2023.111699
PMID:37597314
Abstract

BACKGROUND

Lipoblastoma is a rare and benign tumor arising from embryonal fat cells. It develops primarily in infancy and early childhood. We present a case study of a 10-month-old child treated for an extensive tumor of the infratemporal fossa and parapharyngeal space. The systematic review of 60 reports in 80 children with lipoblastoma of the head and neck is also reported.

MATERIAL AND METHODS

Systematic review of all articles included lipoblastomas of the head and neck area in children published from 1964 to 2022 in the PubMed database was conducted. Clinical presentation of extensive lipoblastoma of the head and neck in a child.

RESULTS

On the basis of all inclusion criteria 83 articles were identified concerning pediatric lipoblastoma. There were 36 F (45%) and 39 M (48%), in 6 patients, (7%) gender was not specified. Ages ranged from 0mth (6h) to 15 yo (180mth). The tumor was located in the head in 22 (27%) cases, while the neck area accounted for 53 (65%) cases. General description without precise location was shown in 6 (7%) cases. All patients underwent complete surgical excision. Post surgical recurrence was noted in 6% clinical cases in the analyzed articles.

CONCLUSIONS

Lipoblastoma is characterized by a rapidly growing predominantly adipose mass. The treatment of choice is complete surgical excision. In selected cases when the pathology is hard to reach, as a consequence of the extensive penetration of the mass, we recommend performing the procedure with an interdisciplinary team. Endoscopy assistance and microdebrider significantly facilitated the removal of pathology in the described advanced case of lipoblastoma. This is the only case reported in the pediatric literature of a head and neck lipoblastoma, where due to extensive involvement and location of the disease the cranial nerves V2 and inferior alveolar branch of V3 could not be spared. Long-term follow-up even to 10 years is required because there is a reported tendency for these tumors to recur.

摘要

背景

脂肪母细胞瘤是一种罕见的良性肿瘤,起源于胚胎脂肪细胞。它主要在婴儿期和幼儿期发展。我们报告了一例 10 个月大的儿童,该儿童患有广泛的颞下窝和咽旁间隙肿瘤。还报告了对 80 例头颈部脂肪母细胞瘤儿童的 60 份报告的系统评价。

材料和方法

对 1964 年至 2022 年期间在 PubMed 数据库中发表的所有包含头颈部儿童脂肪母细胞瘤的文章进行了系统评价。儿童头颈部广泛脂肪母细胞瘤的临床表现。

结果

根据所有纳入标准,确定了 83 篇关于儿科脂肪母细胞瘤的文章。在 6 例患者中,有 36 例女性(45%)和 39 例男性(48%),6 例(7%)性别未指定。年龄从 6 小时到 15 岁(180 个月)不等。肿瘤位于头部的有 22 例(27%),位于颈部的有 53 例(65%)。无精确位置的一般描述显示在 6 例(7%)病例中。所有患者均行完全手术切除。在分析的文章中,6%的临床病例出现术后复发。

结论

脂肪母细胞瘤的特征是快速生长的主要脂肪肿块。治疗选择是完全手术切除。在某些情况下,由于肿瘤广泛浸润,难以到达病变部位,我们建议由多学科团队进行手术。在描述的高级脂肪母细胞瘤病例中,内窥镜辅助和微动力系统显著有助于切除病变。这是唯一一例在儿科文献中报道的头颈部脂肪母细胞瘤病例,由于疾病的广泛受累和位置,无法保留颅神经 V2 和 V3 下牙槽分支。需要进行长达 10 年的长期随访,因为这些肿瘤有复发的趋势。

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