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多灶性淋巴管内皮瘤病合并血小板减少症(MLT)出血婴儿的安全内镜治疗

Safe Endoscopic Treatment for a Bleeding Infant With Multifocal Lymphangioendotheliomatosis and Thrombocytopenia (MLT).

作者信息

Steg Saban Or, Pope Elena, Chavhan Govind B, Carcao Manuel, Talarico Susanna, Ling Simon C

机构信息

Division of Gastroenterology, Hepatology and Nutrition, The Hospital for Sick Children, Toronto, ON.

Division of Pediatric Dermatology, The Hospital for Sick Children, Toronto, ON.

出版信息

ACG Case Rep J. 2025 Jun 27;12(7):e01755. doi: 10.14309/crj.0000000000001755. eCollection 2025 Jul.

DOI:10.14309/crj.0000000000001755
PMID:40585778
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12204144/
Abstract

We present the case of a 3-week-old girl with multiple cutaneous vascular lesions, melena, hematemesis, severe anemia (hemoglobin 4.4 g/dL), and thrombocytopenia (72 × 10/µL), with clinical features consistent with multifocal lymphangioendotheliomatosis with thrombocytopenia (MLT). Upper gastrointestinal endoscopy revealed numerous actively bleeding angiomatous lesions, measuring 1-3 mm in diameter. Homeostasis was successfully achieved using argon plasma coagulation (APC). To our knowledge, this is among the few reports describing the effective use of APC in managing life-threatening gastric hemorrhage in an infant with MLT. While gastrointestinal perforation has been previously reported as a complication of endoscopic therapy in this context, our findings suggest that APC may be a viable option for refractory bleeding, provided that surgical support is immediately accessible.

摘要

我们报告了一名3周大的女婴病例,她有多处皮肤血管病变、黑便、呕血、严重贫血(血红蛋白4.4 g/dL)和血小板减少(72×10/µL),临床特征符合伴血小板减少的多灶性淋巴管内皮瘤病(MLT)。上消化道内镜检查发现大量直径为1 - 3毫米的活动性出血性血管瘤病变。使用氩离子凝固术(APC)成功实现了止血。据我们所知,这是少数描述APC有效用于治疗MLT婴儿危及生命的胃出血的报告之一。虽然在此背景下内镜治疗的并发症此前曾报道有胃肠道穿孔,但我们的研究结果表明,只要能立即获得手术支持,APC可能是治疗难治性出血的可行选择。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8dc3/12204144/638f5bf49867/ac9-12-e01755-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8dc3/12204144/c76cb4b7035b/ac9-12-e01755-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8dc3/12204144/1079e791d5ed/ac9-12-e01755-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8dc3/12204144/ea81a84aa8c4/ac9-12-e01755-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8dc3/12204144/638f5bf49867/ac9-12-e01755-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8dc3/12204144/c76cb4b7035b/ac9-12-e01755-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8dc3/12204144/1079e791d5ed/ac9-12-e01755-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8dc3/12204144/ea81a84aa8c4/ac9-12-e01755-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8dc3/12204144/638f5bf49867/ac9-12-e01755-g004.jpg

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本文引用的文献

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A Case of Multifocal Lymphangioendotheliomatosis With Thrombocytopenia and Changes in Coagulopathy.多灶性淋巴管内皮细胞瘤伴血小板减少和凝血功能变化 1 例
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Successful Treatment With Everolimus for Multifocal Lymphangioendotheliomatosis With Thrombocytopenia in an Infant.
依维莫司成功治疗一名患有血小板减少症的婴儿多灶性淋巴管内皮瘤病
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Clinical variability in multifocal lymphangioendotheliomatosis with thrombocytopenia: a review of the literature.多灶性淋巴管内皮瘤病伴血小板减少症的临床变异性:文献复习。
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Multifocal Lymphangioendotheliomatosis with Thrombocytopenia: Presentation of Two Cases Treated with Sirolimus.多灶性淋巴管内皮瘤病伴血小板减少症:两例西罗莫司治疗病例报告
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Endoscopic management of gastrointestinal bleeding from multifocal lymphangioendotheliomatosis with thrombocytopenia: limited efficacy and complications.内镜治疗伴血小板减少的多灶性淋巴管内皮瘤病所致胃肠道出血:疗效有限且有并发症
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