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Updated: Jun 27, 2026
05:00
Short-Duration Hypothermia Induction in Rats using Models for Studies examining Clinical Relevance and Mechanisms
Published on: March 3, 2021
Spontaneous periodic hypothermia and hyperhidrosis.
Nihal Olgac Dundar1, Adil Boz, Ozgur Duman
1Department of Child Neurology, Faculty of Medicine, Akdeniz University, Antalya, Turkey.
Pediatric Neurology
|November 26, 2008
Summary
Shapiro syndrome, a rare neurological disorder, can occur without corpus callosum agenesis. This case highlights increased brain perfusion in specific areas during hypothermic episodes, suggesting a unique presentation of this condition.
Area of Science:
- Neurology
- Pediatrics
- Medical Imaging
Background:
- Shapiro syndrome is a rare neurological disorder characterized by recurrent episodes of hypothermia.
- Corpus callosum agenesis is sometimes associated with Shapiro syndrome, but its absence does not rule out the diagnosis.
Observation:
- A 4-year-old girl presented with daily episodes of sweating, cooling, and drowsiness lasting approximately one hour.
- Standard laboratory tests, Holter monitoring, echocardiography, and electroencephalography during an attack were unremarkable.
- Cranial magnetic resonance imaging confirmed an intact corpus callosum.
Findings:
- Technetium 99m-labeled hexamethylpropylene amine oxime brain single-photon emission computed tomography revealed increased perfusion in the right thalamus, basal ganglia, and inferior frontal regions during a hypothermic episode.
- Oxcarbazepine partially reduced attack frequency, while carbamazepine showed a better response.
Implications:
- This case expands the phenotypic spectrum of Shapiro syndrome, demonstrating its occurrence without corpus callosum agenesis.
- Abnormal brain perfusion patterns identified via SPECT imaging may serve as a diagnostic indicator.
- The differential response to antiepileptic drugs, particularly carbamazepine, warrants further investigation for therapeutic strategies in Shapiro syndrome.
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