关键词: Case report Corpus callosum agenesis Hypothermia Iatrogenic Shapiro’s syndrome Spontaneous hypothermia

Mesh : Female Humans Middle Aged Hypothermia / complications Agenesis of Corpus Callosum Hyperhidrosis / complications Corpus Callosum / pathology Magnetic Resonance Imaging Iatrogenic Disease

来  源:   DOI:10.1007/s10072-023-07263-0

Abstract:
OBJECTIVE: Shapiro\'s syndrome (SS) is a rare condition characterized by spontaneous periodic hypothermia. The underlying pathophysiological mechanisms and etiology of this syndrome remain controversial, and fewer than 100 cases have been reported to date. The objective of this case report is to present a unique iatrogenic case of SS and contribute additional insights into the underlying etiology of this rare disorder.
METHODS: We conducted an analysis of existing medical literature and described a clinical case of SS secondary to a neurosurgical procedure.
RESULTS: To our knowledge, we present the first iatrogenic case of SS in a 53-year-old woman who underwent a partial right parieto-occipital lobectomy in 2003 as a treatment for refractory epilepsy. Several years after the surgical procedure, she began experiencing recurrent episodes of hypothermia. Brain magnetic resonance imaging (MRI) revealed the absence of the splenium of the corpus callosum (CC) and pituitary hyperplasia. After ruling out other potential causes of hypothermia, a diagnosis of SS was made.
CONCLUSIONS: The most plausible mechanism to explain the recurrent hypothermia associated with SS in our patient is a probable disruption of the pathways involved in thermoregulation through the CC as a consequence of the surgical procedure. This case report provides further insights into the etiology of this rare disorder.
摘要:
目的:Shapiro综合征(SS)是一种罕见的疾病,其特征是自发性周期性低温。该综合征的潜在病理生理机制和病因仍存在争议,迄今为止,报告的病例不到100例。本病例报告的目的是提出一个独特的SS医源性病例,并为这种罕见疾病的潜在病因提供更多见解。
方法:我们对现有医学文献进行了分析,并描述了一例神经外科手术继发SS的临床病例。
结果:据我们所知,我们介绍了首例医源性SS的病例,该病例于2003年在一名53岁女性中接受了部分右顶枕叶切除术治疗难治性癫痫.手术后几年,她开始反复出现体温过低.脑磁共振成像(MRI)显示没有call体(CC)和垂体增生。在排除了体温过低的其他潜在原因后,诊断为SS。
结论:解释我们患者与SS相关的复发性低体温的最合理的机制是,外科手术可能导致通过CC参与体温调节的通路中断。此病例报告提供了对这种罕见疾病病因的进一步见解。
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