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舍格伦-拉尔松综合征:一种伴有发育迟缓的罕见表现。

Sjögren-Larsson Syndrome: A Rare Presentation With Developmental Delay.

作者信息

J Srilakshmi K, Waheed Muhammad Daniyal, Batool Saima, Holder Shaniah S, Rodriguez Reyes Yadelys, Guntha Manisha

机构信息

Pediatrics, Dr. B. R. Ambedkar Medical College and Hospital, Bengaluru, IND.

Internal Medicine, Foundation University Medical College, Islamabad, PAK.

出版信息

Cureus. 2023 Feb 18;15(2):e35159. doi: 10.7759/cureus.35159. eCollection 2023 Feb.

DOI:10.7759/cureus.35159
PMID:36950004
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10028308/
Abstract

Sjögren-Larsson syndrome (SLS) is a rare, inherited disorder passed down through families in an autosomal recessive pattern. Its main characteristics are spastic diplegic paralysis, congenital ichthyotic hyperkeratosis, and mild-to-moderate mental retardation. Lack of activity of microsomal fatty aldehyde dehydrogenase (FALDH) or its complete absence is the primary cause of this syndrome, leading to the build-up of fatty aldehydes and fatty alcohols in the body, particularly in the skin. In order to provide the best care for patients, educating them about the management of dry skin and offering genetic counseling are essential. We hereby present a case of an eight-year-old patient with spastic diplegia, congenital ichthyosis, and intellectual disability diagnosed with SLS.

摘要

舍格伦-拉松综合征(SLS)是一种罕见的遗传性疾病,以常染色体隐性模式在家族中遗传。其主要特征是痉挛性双侧瘫痪、先天性鱼鳞病样角化过度以及轻度至中度智力障碍。微粒体脂肪醛脱氢酶(FALDH)活性缺乏或完全缺失是该综合征的主要病因,导致体内尤其是皮肤中脂肪醛和脂肪醇的蓄积。为了为患者提供最佳护理,对他们进行干性皮肤管理教育并提供遗传咨询至关重要。我们在此呈现一例8岁患有痉挛性双侧瘫痪、先天性鱼鳞病和智力残疾并被诊断为舍格伦-拉松综合征的患者。

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本文引用的文献

1
Sjogren-Larsson Syndrome: Mechanisms and Management.舍格伦-拉松综合征:发病机制与治疗
Appl Clin Genet. 2020 Jan 7;13:13-24. doi: 10.2147/TACG.S193969. eCollection 2020.
2
A rare case of Sjogren-Larsson syndrome with recurrent pneumonia and asthma.一例罕见的伴有复发性肺炎和哮喘的舍格伦-拉尔松综合征病例。
Korean J Pediatr. 2016 Jun;59(6):276-9. doi: 10.3345/kjp.2016.59.6.276. Epub 2016 Jun 30.
3
Zileuton for Pruritus in Sjögren-Larsson Syndrome: A Randomized Double-blind Placebo-controlled Crossover Trial.齐留通治疗舍格伦-拉松综合征瘙痒症:一项随机双盲安慰剂对照交叉试验
Acta Derm Venereol. 2016 Feb;96(2):255-6. doi: 10.2340/00015555-2195.
4
Fatty aldehyde and fatty alcohol metabolism: review and importance for epidermal structure and function.脂肪醛和脂肪醇代谢:综述及其对表皮结构和功能的重要性
Biochim Biophys Acta. 2014 Mar;1841(3):377-89. doi: 10.1016/j.bbalip.2013.09.001. Epub 2013 Sep 12.
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Sjogren-Larsson syndrome: A case report of a rare disease.舍格伦-拉松综合征:一例罕见疾病的病例报告。
Indian Dermatol Online J. 2011 Jan;2(1):31-3. doi: 10.4103/2229-5178.79866.
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Ichthyosis in Sjögren-Larsson syndrome reflects defective barrier function due to abnormal lamellar body structure and secretion.干燥综合征-鱼鳞病综合征中的鱼鳞癣反映了由于板层小体结构和分泌异常导致的屏障功能缺陷。
Arch Dermatol Res. 2010 Aug;302(6):443-51. doi: 10.1007/s00403-009-1022-y. Epub 2010 Jan 5.
7
Sjögren-Larsson syndrome: molecular genetics and biochemical pathogenesis of fatty aldehyde dehydrogenase deficiency.舍格伦-拉松综合征:脂肪醛脱氢酶缺乏症的分子遗传学与生化发病机制
Mol Genet Metab. 2007 Jan;90(1):1-9. doi: 10.1016/j.ymgme.2006.08.006. Epub 2006 Sep 22.
8
MR imaging and proton MR spectroscopic studies in Sjögren-Larsson syndrome: characterization of the leukoencephalopathy.干燥综合征-拉松综合征的磁共振成像和质子磁共振波谱研究:脑白质病变的特征
AJNR Am J Neuroradiol. 2004 Apr;25(4):649-57.
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Indian J Pediatr. 2002 Feb;69(2):193-4. doi: 10.1007/BF02859387.
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Update on Sjögren-Larsson syndrome.干燥综合征-拉松综合征最新进展。
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