Evidence map›Paper›PMID 34847858›Full record

ArticleBMC neurology2021

Arterial tortuosity syndrome causing recurrent transient ischemic attacks in young adult: a case report.

Stefano Cotti Piccinelli, Enrico Premi, Sonia Bonacina, Nicola Gilberti, Veronica Vergani, Massimo Gamba, Raffaella Spezi, Ilenia Delrio, Michaël Bernier, Angelo Costa and 5 more

Open access · goldAbstract readCase Reports
In one paragraph

Article in BMC neurology, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
0.2field-weighted citation impact, top 37% of its field
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

2 citing papers in PubMed, 2 citations in OpenAlex.

  1. Article
  2. 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

15 authors at 3 institutions in 2 countries.

Stefano Cotti PiccinelliNeurology Unit, Department of Clinical and Experimental Sciences, University of Brescia, Pz.le Spedali Civili 1, 25100, Brescia, Italy. stefanocottipiccinelli@gmail.com.
Enrico PremiStroke Unit, Unità Operativa Neurologia Vascolare, ASST "Spedali Civili", Spedali Civili, Brescia, Italy.
Sonia BonacinaNeurology Unit, Department of Clinical and Experimental Sciences, University of Brescia, Pz.le Spedali Civili 1, 25100, Brescia, Italy.
Nicola GilbertiStroke Unit, Unità Operativa Neurologia Vascolare, ASST "Spedali Civili", Spedali Civili, Brescia, Italy.
Veronica VerganiStroke Unit, Unità Operativa Neurologia Vascolare, ASST "Spedali Civili", Spedali Civili, Brescia, Italy.
Massimo GambaStroke Unit, Unità Operativa Neurologia Vascolare, ASST "Spedali Civili", Spedali Civili, Brescia, Italy.
Raffaella SpeziStroke Unit, Unità Operativa Neurologia Vascolare, ASST "Spedali Civili", Spedali Civili, Brescia, Italy.
Ilenia DelrioStroke Unit, Unità Operativa Neurologia Vascolare, ASST "Spedali Civili", Spedali Civili, Brescia, Italy.
Michaël BernierMartinos Center for Biomedical Imaging, Harvard Medical School, Massachusetts General Hospital, Charlestown, MA, USA.
Angelo CostaStroke Unit, Unità Operativa Neurologia Vascolare, ASST "Spedali Civili", Spedali Civili, Brescia, Italy.
Marco RavanelliRadiology Unit, Department of Medical-Surgical Specialties, Radiological Sciences and Public Health, University of Brescia, Brescia, Italy.
Marina ColombiDivision of Biology and Genetics, Department of Molecular and Translational Medicine, University of Brescia, Brescia, Italy.
Roberto GasparottiNeuroradiology Unit, Department of Medical-Surgical Specialties, Radiological Sciences and Public Health, University of Brescia, Brescia, Italy.
Alessandro PadovaniNeurology Unit, Department of Clinical and Experimental Sciences, University of Brescia, Pz.le Spedali Civili 1, 25100, Brescia, Italy.
Mauro MagoniStroke Unit, Unità Operativa Neurologia Vascolare, ASST "Spedali Civili", Spedali Civili, Brescia, Italy.
Azienda Socio Sanitaria Territoriale degli Spedali Civili di Brescia · ITUniversity of Brescia · ITMassachusetts General Hospital · US

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundArterial Tortuosity Syndrome (ATS) is a rare autosomal recessive disorder characterized by elongated and tortuous arteries. Although ATS showed a significant clinical and pathophysiological overlap with other syndromes involving connective tissues, only few cases of cerebrovascular events related to this syndrome have been described so far. CASE PRESENTATION: We report the case of a 33-years-old male diagnosed with ATS since childhood, that experienced three sudden episodes of expressive aphasia and right hemiparesis with spontaneous resolution. He was treated with recombinant tissue plasminogen activator (r-TPA) at a dosage of 0.9 mg/kg with a complete recovery. Brain Magnetic Resonance Imaging (MRI) showed the absence of acute ischemic lesions and the patient was diagnosed with recurrent transient ischemic attacks (TIA). Intracranial and supra-aortic trunks Magnetic Resonance Angiography (MRA) and Angio-CT scan of the thoracic and abdominal aorta showed marked vessel tortuosity without stenosis. To our knowledge, this is the first reported case of an ATS patient with TIA in young age that was treated with intravenous thrombolysis with recombinant plasminogen activator.

conclusionOur report strengthens the relationship between ATS and juvenile cerebrovascular events, suggesting that an extensive study of body vessels in order to detect potential stenoses or occlusions in these cases is needed. The greater predisposition to cerebrovascular events in ATS could benefit from a more aggressive primary and secondary prevention therapy.

Indexed as

Ischemic Attack, TransientSkin Diseases, GeneticAdultArteriesHumansJoint InstabilityMaleTissue Plasminogen ActivatorVascular MalformationsTissue Plasminogen ActivatorArterial tortuosity syndromeThrombolysisTransient ischemic attack

Identifiers

PMID34847858
PMCPMC8630900
OpenAlexW3214882867

What OpenQuestion holds

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LicenceCC BY
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Registered trials

None linked

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.