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新生儿肺泡淋巴管瘤:一例免疫特征病例报告

Alveolar Lymphangioma in Neonate: A Case Report With Immune Profile.

作者信息

Nizar Muhammad Aiman Bin Mohd, Seo Benedict, Hussaini Haizal M, Young Brendan, Rich Alison M

机构信息

Department of Oral Diagnostic and Surgical Sciences, Faculty of Dentistry, University of Otago, Dunedin, New Zealand.

Department of Craniofacial Diagnostic and Biosciences, Faculty of Dentistry, Universiti Kebangsaan Malaysia, Kuala Lumpur, Malaysia.

出版信息

Int J Paediatr Dent. 2025 Sep;35(5):932-935. doi: 10.1111/ipd.13309. Epub 2025 Mar 14.

DOI:10.1111/ipd.13309
PMID:40087399
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12332113/
Abstract

BACKGROUND

Alveolar lymphangioma is a rare condition that commonly develops on the alveolar ridge of Black male neonates. It typically presents as a bluish, dome-shaped swelling smaller than 1 cm on the posterior alveolar ridge of the mandible or/and maxilla, that allows it to be diagnosed clinically. Most previously reported cases of alveolar lymphangioma report spontaneous resolution, and biopsy is seldom performed unless the lesion becomes symptomatic or interferes with feeding.

CASE REPORT

We report a case of oral alveolar lymphangioma on the anterior maxillary alveolar ridge of an 11-day-old infant of non-black descent. The lesion presented as a pedunculated, reddish, lobular mass in the anterior maxilla. Given its atypical presentation and the potential for feeding difficulties, an excisional biopsy was performed. The diagnosis was confirmed histologically through biopsy and supported by immunohistochemical staining.

CONCLUSION

This case expands the understanding of alveolar lymphangioma by reporting it in a non-Black neonate with an unusual location and appearance, emphasising the need for excisional biopsy to rule out other potential oral lesions, especially neoplasms.

摘要

背景

肺泡淋巴管瘤是一种罕见病症,常见于黑人男性新生儿的牙槽嵴。它通常表现为在下颌骨或/和上颌骨后牙槽嵴上小于1厘米的蓝色圆顶状肿胀,这使其能够在临床上得到诊断。之前报道的大多数肺泡淋巴管瘤病例报告显示其可自行消退,除非病变出现症状或影响进食,否则很少进行活检。

病例报告

我们报告一例非黑人血统11天大婴儿上颌前牙槽嵴的口腔肺泡淋巴管瘤病例。该病变表现为上颌前部带蒂的红色小叶状肿物。鉴于其不典型表现及存在喂养困难的可能性,遂进行了切除活检。通过活检在组织学上确诊,并得到免疫组化染色支持。

结论

本病例通过报道非黑人新生儿中具有不寻常位置和外观的肺泡淋巴管瘤,扩展了对该疾病的认识,强调了切除活检以排除其他潜在口腔病变尤其是肿瘤的必要性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/53b1/12332113/a9d9e38e9ede/IPD-35-932-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/53b1/12332113/f7501ffb19a3/IPD-35-932-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/53b1/12332113/a9d9e38e9ede/IPD-35-932-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/53b1/12332113/f7501ffb19a3/IPD-35-932-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/53b1/12332113/a9d9e38e9ede/IPD-35-932-g001.jpg

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本文引用的文献

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Neonatal alveolar lymphangioma: A rare benign oral disease case report and literature review.新生儿肺泡状淋巴管瘤:一种罕见的良性口腔疾病病例报告及文献复习。
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ISSVA Classification of Vascular Anomalies and Molecular Biology.ISSVA 血管异常分类与分子生物学。
Int J Mol Sci. 2022 Feb 21;23(4):2358. doi: 10.3390/ijms23042358.
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Oral lymphatic malformations: A multicenter study of 208 cases and literature review.口腔淋巴管畸形:208 例多中心研究及文献复习。
Head Neck. 2021 Nov;43(11):3562-3571. doi: 10.1002/hed.26854. Epub 2021 Sep 13.
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Multiple lymphangiomas of the alveolar ridge in a neonate: case study.新生儿牙槽嵴多发性淋巴管瘤:病例报告
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