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开始使用苯海索后出现精神病性特征的伍德豪斯-萨卡蒂综合征:一例报告

Woodhouse-Sakati Syndrome Presenting With Psychotic Features After Starting Trihexyphenidyl: A Case Report.

作者信息

Aljaffer Mohammed A, Almadani Ahmad H, AlMutlaq Mohammad, Alhammad Abdulaziz, Alyahya Ahmed S

机构信息

Department of Psychiatry, College of Medicine, King Saud University, Riyadh, SAU.

College of Medicine, King Saud University, Riyadh, SAU.

出版信息

Cureus. 2022 Aug 1;14(8):e27576. doi: 10.7759/cureus.27576. eCollection 2022 Aug.

DOI:10.7759/cureus.27576
PMID:36059322
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC9433055/
Abstract

Woodhouse-Sakati syndrome is a rare, autosomal recessive, multisystemic disorder first identified as a constellation of hypogonadism, mental retardation, diabetes, alopecia, deafness, and electrocardiogram abnormalities.  We report a case of a 33-year-old woman who was born to consanguineous parents. She is suffering from hypergonadotropic hypogonadism, extrapyramidal symptoms, hypothyroidism, alopecia, and sensorineural hearing loss. Her MRI showed iron depositions in globus pallidus bilaterally. She underwent genetic testing and was diagnosed with Woodhouse-Sakati syndrome. She was started on trihexyphenidyl to treat her extrapyramidal symptoms. A few months later, she started to have psychotic symptoms in the form of auditory hallucinations and delusions of persecution.  Although she exhibited psychotic symptoms after starting trihexyphenidyl, it is less likely to be causing her symptoms since the symptoms started a few months after taking the medication and she was not on high doses. Thus, it is more likely to be a part of Woodhouse-Sakati syndrome.

摘要

伍德豪斯-萨卡蒂综合征是一种罕见的常染色体隐性多系统疾病,最初被确定为性腺功能减退、智力迟钝、糖尿病、脱发、耳聋和心电图异常的组合。我们报告一例33岁女性病例,其父母为近亲结婚。她患有高促性腺激素性性腺功能减退、锥体外系症状、甲状腺功能减退、脱发和感音神经性听力损失。她的MRI显示双侧苍白球有铁沉积。她接受了基因检测,被诊断为伍德豪斯-萨卡蒂综合征。她开始服用苯海索来治疗锥体外系症状。几个月后,她开始出现以幻听和被害妄想形式的精神症状。虽然她在开始服用苯海索后出现了精神症状,但由于症状在服药几个月后才开始,且服用剂量不高,所以不太可能是药物导致的症状。因此,更有可能是伍德豪斯-萨卡蒂综合征的一部分。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5ecd/9433055/52fcbc376af8/cureus-0014-00000027576-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5ecd/9433055/52fcbc376af8/cureus-0014-00000027576-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5ecd/9433055/52fcbc376af8/cureus-0014-00000027576-i01.jpg

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Patterns of neurological manifestations in Woodhouse-Sakati Syndrome.伍德豪斯-萨卡蒂综合征的神经表现模式。
Parkinsonism Relat Disord. 2019 Dec;69:99-103. doi: 10.1016/j.parkreldis.2019.10.007. Epub 2019 Oct 13.
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