Evidence map›Paper›PMID 41426901›Full record

ArticleCureus2025

Shapiro Syndrome: A Case Report.

Manya Motsara

Abstract readCase Reports
In one paragraph

Article in Cureus, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

1 citing paper in PubMed.

  1. Adult-onset spontaneous periodic hypothermia with hyperhidrosis: three cases of a Shapiro syndrome variant.Clinical autonomic research : official journal of the Clinical Autonomic Research Society · 2026
    Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

1 author.

Manya MotsaraGeneral Medicine, King's College Hospital NHS Foundation Trust, Kent, GBR.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Shapiro syndrome is a rare dysautonomic disorder defined by the triad of spontaneous recurrent hypothermia, hyperhidrosis, and agenesis of the corpus callosum. Recognition in older adults is uncommon and often delayed, as episodes can mimic infection or endocrine disease. We report an 81-year-old woman with established Shapiro syndrome who presented with recurrent admissions for profound hypothermia (31.3-33.6 °C) accompanied by delirium and sinus bradycardia. Previous MRI demonstrated complete agenesis of the corpus callosum with colpocephaly and hippocampal hypoplasia. Episodes were managed with active rewarming, hydration, and adherence to a neurology plan emphasizing conservative temperature control and avoidance of antipyretics during hypothermia. For prophylaxis, she received clonidine 25 mg (three tablets in the morning, two at midday, and three at night) and lamotrigine 50 mg twice daily for background epilepsy. With this protocol, episode severity and duration decreased. This case highlights classic Shapiro syndrome persisting into advanced age. In elderly patients with unexplained recurrent hypothermia and sweating, especially with documented agenesis of the corpus callosum, early recognition and a structured conservative plan can reduce hospitalizations and iatrogenic interventions.

Indexed as

agenesis of the corpus callosumelderly femaleepisodic hypothermiageneral internal medicinegeneralized hyperhidrosisneurologyrare disease or conditionshapiro syndrome

Identifiers

PMID41426901
PMCPMC12717870

What OpenQuestion holds

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.