Evidence map›Paper›PMID 25374739›Full record

ArticleCase reports in immunology2013

A precocious cerebellar ataxia and frequent Fever episodes in a 16-month-old infant revealing ataxia-telangiectasia syndrome.

Luigi Nespoli, Annapia Verri, Silvia Tajè, Francesco Paolo Pellegrini, Maddalena Marinoni

Open access · goldAbstract read
In one paragraph

Article in Case reports in immunology, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed, 1 pooled it
0.1field-weighted citation impact, top 43% 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, 1 synthesis or guideline pooled it, 3 citations in OpenAlex.

  1. Pooled it
  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

5 authors at 2 institutions in 1 country.

Luigi NespoliPediatrics Unit, Department of Clinical and Experimental Medicine, University of Insubria, 21100 Varese, Italy.
Annapia VerriNeurological Institute C. Mondino Foundation IRCCS, 27100 Pavia, Italy.ORCID 0000-0002-1211-4799
Silvia TajèPediatrics Unit, Department of Clinical and Experimental Medicine, University of Insubria, 21100 Varese, Italy.
Francesco Paolo PellegriniPediatrics Unit, Department of Clinical and Experimental Medicine, University of Insubria, 21100 Varese, Italy.ORCID 0000-0001-6594-6727
Maddalena MarinoniPediatrics Unit, Department of Clinical and Experimental Medicine, University of Insubria, 21100 Varese, Italy.
University of Insubria · ITFondazione Istituto Neurologico Nazionale Casimiro Mondino · IT

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Ataxia-telangiectasia (AT) is the most frequent progressive cerebellar ataxia in infancy and childhood. Immunodeficiency which includes both cellular and humoral arms has variable severity. Since the clinical presentation is extremely variable, a high clinical suspicion will allow an early diagnosis. Serum alpha-fetoprotein is elevated in 80-85% of patients and therefore could be used as a screening tool. Here, we present a case of a 5-year-old female infant who was admitted to our department at the age of 16 months because of gait disorders and febrile episodes that had begun at 5 months after the cessation of breastfeeding. Serum alfa-fetoprotein level was elevated. Other investigations showed leukocytopenia with lymphopenia, reduced IgG2 and IgA levels, and low titers of specific postimmunization antibodies against tetanus toxoid and Haemophilus B polysaccharide. Peripheral lymphocytes subsets showed reduction of T cells with a marked predominance of T cells with a memory phenotype and a corresponding reduction of naïve T cells; NK cells were very increased (41%) with normal activity. The characterization of the ATM gene mutations revealed 2 specific mutations (c.5692C > T/c.7630-2A > C) compatible with AT diagnosis. It was concluded that AT syndrome should be considered in children with precocious signs of cerebellar ataxia and recurrent fever episodes.

Identifiers

PMID25374739
PMCPMC4207493
OpenAlexW2050172520

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.