ArticleCase reports in hematology2013
Cyclic bicytopenia in a patient with shapiro syndrome.
Article in Case reports in hematology, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.
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Who cites it
6 citing papers in PubMed, 10 citations in OpenAlex.
- Acquired spontaneous periodic hypothermia and pancytopenia secondary to hypothalamic compression from a large skull base meningioma.Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology · 2026Article
- Iatrogenic Shapiro syndrome: a case report.Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology · 2024Article
- A new perspective of hypothalamic disease: Shapiro's syndrome.Frontiers in neurology · 2022Review
- [Prolonged severe hypothermia of 27.4 °C and thrombocytopenia].Zeitschrift fur Gerontologie und Geriatrie · 2021Article
- Episodic headache with spontaneous hypothermia reveal Shapiro's syndrome variant with effectiveness of clonidine therapy.The journal of headache and pain · 2021Article
- Chronic Pancytopenia due to Centrally Mediated Hypothermia in Two Children with Severe Neurological Impairment.Children (Basel, Switzerland) · 2020Article
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Authors and funding
5 authors at 2 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Shapiro syndrome and periodic hypothermia have been reported approximately fifty times in the literature. Shapiro syndrome is defined as the constellation of periodic hypothermia and hyperhidrosis along with agenesis of the corpus callosum by Shapiro et al. in 1969. Periodic hypothermia is a more broad diagnosis with a number of proposed mechanisms; it occurs in patients without structural brain abnormalities. Hematologic abnormalities beyond iron-deficiency anemia have not been documented in any of the reported cases of Shapiro syndrome or periodic hypothermia. Though accidental and therapeutic hypothermia have been associated with thrombocytopenia, this is, to our knowledge, the first reported case of periodic intrinsic hypothermia causing bicytopenia. In this report, we present the case of a patient with Shapiro syndrome who experienced cyclic bicytopenia mirroring hypothermic episodes. We address the differential diagnosis of bicytopenia, review the mechanisms proposed for cytopenias related to hypothermia, and propose possible mechanisms for the finding in this case.
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