IIIB型迟发性粘多糖病患者的精神分裂症样表现和神经功能突然下降。

IF 1.5 4区 医学 Q4 GENETICS & HEREDITY
Yorran Hardman Araújo Montenegro, Guilherme Baldo, Roberto Giugliani, Fabiano de Oliveira Poswar, Ruy Pires de Oliveira Sobrinho, Carlos Eduardo Steiner
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引用次数: 2

摘要

由于先天性代谢异常的发生率低和一些非典型表现,经常被误诊或诊断不足,这阻碍了这些患者的正确治疗。为了说明这一点,我们在这里介绍一位患者,与她的同学相比,她在上学初期就有学习障碍,尤其是在写作方面。她在一所普通学校完成了基础教育,然后转到一所为有特殊需要的学生开设的中学。18岁 几岁时,她出现了第一次精神病突发:她尖叫了一个月,没有睡觉。行为问题随后变得明显,尤其是多动、破坏性和混乱的行为、焦虑、自我攻击性和异性攻击性。诊断为精神分裂样障碍。临床遗传评估显示面部粗糙、舌大、毛发粗厚、轻度肝肿大,并提出粘多糖病III的假说。实验室测试表明,尿液中的糖胺聚糖含量很高,NAGLU活性几乎检测不到,证实了这一诊断。NAGLU基因测序显示c.1318G>c(p.Gly440Arg)和c.1834A>G(p.Ser612Gly)突变。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Schizophreniform presentation and abrupt neurologic decline in a patient with late-onset mucopolysaccharidosis type IIIB.

Due to their low frequency and some atypical presentations, inborn errors of metabolism are frequently misdiagnosed or underdiagnosed, which hinders the correct management of these patients. To illustrate that, here we present a patient that, at early school age, had learning disabilities compared to her classmates, especially for writing. She completed basic education in a regular school and was transferred to a secondary school for students with special needs. At 18 years of age, she presented a first psychiatric abrupt outbreak: she spent a month screaming and without sleeping. Behavioral problems then became apparent, especially hyperactivity, destructive and chaotic behavior, anxiety, and auto-aggressivity and hetero-aggressivity. A diagnosis of schizophreniform disorder was established. Clinical genetic evaluation revealed coarse face, macroglossia, coarse thick hair, and mild hepatomegaly, and the hypothesis of mucopolysaccharidosis-III was raised. Laboratory tests indicated high levels of urinary glycosaminoglycans and almost undetectable NAGLU activity, confirming the diagnosis. Sequencing of the NAGLU gene revealed the c.1318G>C (p.Gly440Arg) and c.1834A>G (p.Ser612Gly) mutations.

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来源期刊
Psychiatric Genetics
Psychiatric Genetics 医学-神经科学
CiteScore
2.30
自引率
0.00%
发文量
39
审稿时长
3 months
期刊介绍: ​​​​​​The journal aims to publish papers which bring together clinical observations, psychological and behavioural abnormalities and genetic data. All papers are fully refereed. Psychiatric Genetics is also a forum for reporting new approaches to genetic research in psychiatry and neurology utilizing novel techniques or methodologies. Psychiatric Genetics publishes original Research Reports dealing with inherited factors involved in psychiatric and neurological disorders. This encompasses gene localization and chromosome markers, changes in neuronal gene expression related to psychiatric disease, linkage genetics analyses, family, twin and adoption studies, and genetically based animal models of neuropsychiatric disease. The journal covers areas such as molecular neurobiology and molecular genetics relevant to mental illness. Reviews of the literature and Commentaries in areas of current interest will be considered for publication. Reviews and Commentaries in areas outside psychiatric genetics, but of interest and importance to Psychiatric Genetics, will also be considered. Psychiatric Genetics also publishes Book Reviews, Brief Reports and Conference Reports.
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