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一例患有溃疡性骶尾部血管瘤和脊柱裂的婴儿的腰椎综合征病例报告。

A Case Report on LUMBAR Syndrome in an Infant With Ulcerated Sacral Hemangioma and Spinal Dysraphism.

作者信息

Soares João, Craveiro Costa Ricardo, Cardoso Rúben, Pinto Joana, Carvalho Sílvia, Castendo Ramos Leonor

机构信息

Dermatology, Hospital Pediátrico, Unidade Local de Saúde de Coimbra, Coimbra, PRT.

Pediatrics, Hospital Pediátrico, Unidade Local de Saúde de Coimbra, Coimbra, PRT.

出版信息

Cureus. 2025 Mar 5;17(3):e80097. doi: 10.7759/cureus.80097. eCollection 2025 Mar.

DOI:10.7759/cureus.80097
PMID:40190860
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11970802/
Abstract

Infantile hemangiomas are the most common soft tissue tumors in infancy, most following a benign and predictable course. However, some hemangiomas, particularly those in the lower body, can indicate underlying syndromic anomalies, as seen in lower body hemangiomas and other cutaneous defects, urogenital anomalies, ulceration, myelopathy, bony deformities, anorectal malformations, arterial anomalies, and renal anomalies (LUMBAR) syndrome. This report presents the case of a term infant with a large superficial ulcerated sacral hemangioma and associated spinal dysraphism, including tethered cord, partial sacral agenesis, and intraspinal lipoma. The early multidisciplinary evaluation confirmed the diagnosis of LUMBAR syndrome, and the patient underwent surgical management of cutaneous discontinuities and initiated oral propranolol. Propranolol was effective in resolving the ulcerated component of the hemangioma. This case highlights the importance of recognizing lower body hemangiomas as markers for potential underlying anomalies, emphasizing the role of early diagnosis, comprehensive imaging, and multidisciplinary care in optimizing outcomes for this rare but complex syndrome.

摘要

婴儿血管瘤是婴儿期最常见的软组织肿瘤,大多数病程呈良性且可预测。然而,一些血管瘤,尤其是下半身的血管瘤,可能提示潜在的综合征性异常,如下半身血管瘤合并其他皮肤缺损、泌尿生殖系统异常、溃疡、脊髓病、骨骼畸形、肛门直肠畸形、动脉异常和肾脏异常(LUMBAR)综合征。本报告介绍了一名足月儿,患有巨大浅表溃疡性骶部血管瘤及相关脊柱发育异常,包括脊髓拴系、部分骶骨发育不全和椎管内脂肪瘤。早期多学科评估确诊为LUMBAR综合征,患者接受了皮肤缺损的手术治疗,并开始口服普萘洛尔。普萘洛尔有效解决了血管瘤的溃疡部分。该病例强调了将下半身血管瘤识别为潜在潜在异常标志物的重要性,强调了早期诊断、全面成像和多学科护理在优化这种罕见但复杂综合征治疗结果中的作用。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5941/11970802/17271c0bc18d/cureus-0017-00000080097-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5941/11970802/a19294b593a7/cureus-0017-00000080097-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5941/11970802/97dffb370a38/cureus-0017-00000080097-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5941/11970802/17271c0bc18d/cureus-0017-00000080097-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5941/11970802/a19294b593a7/cureus-0017-00000080097-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5941/11970802/97dffb370a38/cureus-0017-00000080097-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5941/11970802/17271c0bc18d/cureus-0017-00000080097-i03.jpg

相似文献

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A Case Report on LUMBAR Syndrome in an Infant With Ulcerated Sacral Hemangioma and Spinal Dysraphism.一例患有溃疡性骶尾部血管瘤和脊柱裂的婴儿的腰椎综合征病例报告。
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[PELVIS or LUMBAR syndrome: the same entity. Two case reports].[骨盆或腰椎综合征:同一实体。两例病例报告]
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本文引用的文献

1
Delphi Consensus on Diagnostic Criteria for LUMBAR Syndrome.德尔福腰椎综合征诊断标准共识。
J Pediatr. 2024 Sep;272:114101. doi: 10.1016/j.jpeds.2024.114101. Epub 2024 May 15.
2
Clinical overlap of PHACE and LUMBAR syndromes.PHACE综合征和腰段综合征的临床重叠。
Pediatr Dermatol. 2022 Sep;39(5):752-756. doi: 10.1111/pde.15031. Epub 2022 Jul 27.
3
Recurrence rate of infantile hemangioma after oral propranolol therapy.婴幼儿血管瘤口服普萘洛尔治疗后的复发率。
Eur J Pediatr. 2021 Feb;180(2):585-590. doi: 10.1007/s00431-020-03872-5. Epub 2020 Nov 13.
4
Hamartomas and midline anomalies in association with infantile hemangiomas, PHACE, and LUMBAR syndromes.错构瘤及中线异常与婴儿血管瘤、PHACE综合征和LUMBAR综合征相关。
Pediatr Dermatol. 2020 Jan;37(1):78-85. doi: 10.1111/pde.14006. Epub 2019 Oct 20.
5
Propranolol and infantile hemangiomas four years later: a systematic review.四年后的普萘洛尔与婴儿血管瘤:一项系统综述
Pediatr Dermatol. 2013 Mar-Apr;30(2):182-91. doi: 10.1111/pde.12089. Epub 2013 Feb 14.
6
LUMBAR: association between cutaneous infantile hemangiomas of the lower body and regional congenital anomalies.腰部:下半身皮肤婴幼儿血管瘤与局部先天性畸形的关系。
J Pediatr. 2010 Nov;157(5):795-801.e1-7. doi: 10.1016/j.jpeds.2010.05.027. Epub 2010 Jul 2.
7
SACRAL syndrome: spinal dysraphism, anogenital, cutaneous, renal and urologic anomalies, associated with an angioma of lumbosacral localization.骶骨综合征:脊髓发育异常、肛门生殖器、皮肤、肾脏和泌尿系统异常,与腰骶部血管瘤相关。
Dermatology. 2007;214(1):40-5. doi: 10.1159/000096911.
8
PELVIS Syndrome.骨盆综合征
Arch Dermatol. 2006 Jul;142(7):884-8. doi: 10.1001/archderm.142.7.884.
9
Hemangiomas in children.儿童血管瘤
N Engl J Med. 1999 Jul 15;341(3):173-81. doi: 10.1056/NEJM199907153410307.