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伴有胡勒氏病的非典型蒙古斑:一例报告

Atypical Mongolian Spots With Hurler's Disease: A Case Report.

作者信息

Athanti Saranya, Mouzam Sadaf, Sohail Ahmed Mohammad

机构信息

Pediatrics, Employee State Insurance Corporation Medical College and Hospital, Sanathnagar, Hyderabad, IND.

出版信息

Cureus. 2024 Apr 17;16(4):e58501. doi: 10.7759/cureus.58501. eCollection 2024 Apr.

Abstract

Mongolian spots are bluish-grey, irregular, hyperpigmented macules present at birth or that appear in the first few weeks of life. They are classified as atypical if they occur in unusual locations without spontaneous disappearance after infancy; or if new lesions continue to appear beyond early infancy. Although they are generally considered benign, recent studies have shown that atypical Mongolian spots may be associated with inborn errors of metabolism, such as lysosomal storage disorders and neurocristopathies. An 11-month-old male presented with multiple aberrant Mongolian spots on the abdomen, back, buttocks, arms, and legs, with the largest patch measuring 10x10 cm. Additionally, the child exhibited coarse facial features, a high-arched palate, low-set ears, and a depressed nasal bridge. Systemic examination revealed hepatosplenomegaly, fundus examination showed a hazy cornea, and the urine glycosaminoglycan test was positive, prompting us to conduct further research prioritising lysosomal storage disorders. The mucopolysaccharidosis (MPS) spot test was positive, and electrophoresis for MPS revealed bands for chondroitin sulfate and dermatan sulfate, confirming the diagnosis of MPS. Enzyme assay revealed no alpha-iduronidase activity and normal beta-galactosidase activity, thus confirming Hurler's disease. This case report highlights the importance of considering atypical Mongolian spots as a potential indicator of underlying storage disorders, enabling early intervention.

摘要

蒙古斑是出生时即存在或在出生后几周内出现的蓝灰色、不规则、色素沉着过度的斑疹。如果蒙古斑出现在不寻常的部位且婴儿期后不会自行消失,或者如果在婴儿早期过后仍有新的皮损持续出现,则被归类为非典型蒙古斑。尽管它们通常被认为是良性的,但最近的研究表明,非典型蒙古斑可能与先天性代谢缺陷有关,如溶酶体贮积症和神经嵴病。一名11个月大的男性患儿腹部、背部、臀部、手臂和腿部出现多处异常蒙古斑,最大的斑块尺寸为10×10厘米。此外,该患儿面部特征粗糙,腭弓高,耳朵低位,鼻梁凹陷。全身检查发现肝脾肿大,眼底检查显示角膜模糊,尿糖胺聚糖试验呈阳性,促使我们进一步研究,优先考虑溶酶体贮积症。黏多糖贮积症(MPS)斑点试验呈阳性,MPS电泳显示硫酸软骨素和硫酸皮肤素条带,确诊为MPS。酶分析显示无α-艾杜糖醛酸酶活性,β-半乳糖苷酶活性正常,从而确诊为Hurler病。本病例报告强调了将非典型蒙古斑视为潜在的潜在贮积症指标的重要性,以便能够早期干预。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/4f19/11101911/62bfe6a2caf9/cureus-0016-00000058501-i01.jpg

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