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严重型马罗-拉米综合征双胞胎接受酶替代治疗后的临床演变

Clinical Evolution After Enzyme Replacement Therapy in Twins with the Severe Form of Maroteaux-Lamy Syndrome.

作者信息

Pineda M, O'Callaghan M, Fernandez Lopez A, Coll M J, Ullot R, Garcia-Fructuoso G

机构信息

Fundación y Servicio de Neuropediatría, Hospital Universitario Sant Joan de Déu, CIBERER, Barcelona, Spain.

Servicio de pediatría, Hospital Sant Joan de Déu, Universidad de Barcelona, Barcelona, Spain.

出版信息

JIMD Rep. 2016;30:7-14. doi: 10.1007/8904_2016_530. Epub 2016 Feb 27.

Abstract

Mucopolysaccharidosis type VI (MPS VI) is a progressive, autosomal, recessive lysosomal disorder. This disorder, due to a deficiency in N-acetylgalactosamine-4-sulfatase (ASB), results in an accumulation of glycosaminoglycan (GAG), causing multiple organ failures. In this study, monochorionic biamniotic twins with the severe form of MPS VI underwent enzyme replacement therapy (ERT) with weekly infusions of recombinant human ASB (galsulfase) at 1 mg/kg. After 9 years of ERT, a comprehensive clinical examination was performed. Several types of biochemical, immunological, and genetic investigations were also conducted. Both twins showed the typical symptoms and signs of MPS VI at baseline, including short stature, progressive dysmorphic facial features, and dysostosis multiplex. Twin 2 presented stronger multisystemic involvement, with marked musculoskeletal, neurological, and odontological components. She also developed an ischemic spinal cord lesion after surgery, which is the first case described in the literature in Maroteaux-Lamy syndrome. However, the extent of disease was found to be equally stabilized in the two sisters, concretely the cardiac and respiratory functions and body length. The early diagnosis and treatment of MPS VI are critical for an optimal clinical outcome, and further evidence for the new treatment strategies is needed.

摘要

VI型黏多糖贮积症(MPS VI)是一种进行性常染色体隐性溶酶体疾病。由于N - 乙酰半乳糖胺 - 4 - 硫酸酯酶(ASB)缺乏,该疾病导致糖胺聚糖(GAG)蓄积,进而引发多器官功能衰竭。在本研究中,患有严重形式MPS VI的单绒毛膜双羊膜囊双胞胎接受了酶替代疗法(ERT),每周静脉输注1mg/kg重组人ASB(galsulfase)。ERT治疗9年后,进行了全面的临床检查。还开展了多种类型的生化、免疫和基因研究。在基线时,双胞胎均表现出MPS VI的典型症状和体征,包括身材矮小、进行性面部畸形特征和多发性骨发育异常。双胞胎2表现出更强的多系统受累,伴有明显的肌肉骨骼、神经和牙科学方面的症状。她在手术后还出现了缺血性脊髓病变,这是文献中首次报道的Maroteaux - Lamy综合征病例。然而,发现两姐妹的疾病程度同样得到了稳定,具体体现在心脏和呼吸功能以及身长方面。MPS VI的早期诊断和治疗对于获得最佳临床结果至关重要,并且需要新治疗策略的进一步证据。

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Enzyme replacement therapy attenuates disease progression in two Japanese siblings with mucopolysaccharidosis type VI.
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