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[在一例存在全身性过氧化物酶体缺陷的病例中,是泽尔韦格综合征、新生儿肾上腺脑白质营养不良还是婴儿型雷夫叙姆病?]

[Zellweger syndrome, neonatal adrenoleukodystrophy or infantile Refsum's disease in a case with generalized peroxisome defect?].

作者信息

Schmitt K, Molzer B, Stöckler S, Tulzer G, Tulzer W

机构信息

Landes-Kinderklinik, Linz.

出版信息

Wien Klin Wochenschr. 1993;105(11):320-2.

PMID:7687405
Abstract

An eleven month-old boy presented clinically with craniofacial dysmorphia, severe psychomotor retardation, neurological deterioration, no response to visual and acoustic stimuli, failure to thrive, hepatomegaly and adrenal insufficiency. Specific biochemical markers for a peroxisomal deficiency disorder (Zellweger's syndrome, neonatal adrenoleukodystrophy, infantile Refsum's disease) revealed pathological results for very long chain fatty acids, phytanic acid, pristanic acid, plasmalogen biosynthesis and catalase, thus confirming the clinical diagnosis. Comparison of clinical and biochemical findings in the patient with the characteristics of the three peroxisomal deficiency disorders showed overlapping with each of these disorders, which corresponds to the current view that these three peroxisomal disorders differ only with respect to onset and severity of the clinical manifestations, but not with regard to the biochemical defects.

摘要

一名11个月大的男童临床表现为颅面畸形、严重精神运动发育迟缓、神经功能恶化、对视觉和听觉刺激无反应、生长发育不良、肝肿大和肾上腺功能不全。针对过氧化物酶体缺乏症(泽尔韦格综合征、新生儿肾上腺脑白质营养不良、婴儿型雷夫叙姆病)的特定生化标志物显示,极长链脂肪酸、植烷酸、降植烷酸、缩醛磷脂生物合成和过氧化氢酶的检测结果呈病理性,从而证实了临床诊断。将该患者的临床和生化检查结果与三种过氧化物酶体缺乏症的特征进行比较,发现与每种疾病均有重叠,这与目前的观点相符,即这三种过氧化物酶体疾病仅在临床表现的起病和严重程度方面存在差异,而在生化缺陷方面并无不同。

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