Evidence map›Paper›PMID 29680425›Full record

ArticleSleep medicine2018

Prevalence and characteristics of sleep-disordered breathing in familial dysautonomia.

Kanwaljit Singh, Jose-Alberto Palma, Horacio Kaufmann, Nataliya Tkachenko, Lucy Norcliffe-Kaufmann, Christy Spalink, Mikhail Kazachkov, Sanjeev V Kothare

Open access · greenAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Sleep medicine, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 5 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
5citing papers in PubMed, 1 pooled it
1.0field-weighted citation impact, top 30% 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

5 citing papers in PubMed, 1 synthesis or guideline pooled it, 14 citations in OpenAlex.

  1. Pooled it
  2. Review
  3. Review
  4. Improvement of daytime hypercapnia with nocturnal non-invasive ventilation in familial dysautonomia.Clinical autonomic research : official journal of the Clinical Autonomic Research Society · 2019
    Article
  5. Orthostatic hypotension: does the heart rate matter? And other updates on recent autonomic research.Clinical autonomic research : official journal of the Clinical Autonomic Research Society · 2018
    Article
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

8 authors at 3 institutions in 1 country.

Kanwaljit SinghUniversity of Massachusetts Medical School, Worcester, MA, USA.
Jose-Alberto PalmaDepartment of Neurology, Dysautonomia Center, New York University School of Medicine, New York, NY, USA.
Horacio KaufmannDepartment of Neurology, Dysautonomia Center, New York University School of Medicine, New York, NY, USA.
Nataliya TkachenkoDepartment of Pediatrics, New York University Langone Medical Center, New York, NY, USA.
Lucy Norcliffe-KaufmannDepartment of Neurology, Dysautonomia Center, New York University School of Medicine, New York, NY, USA.
Christy SpalinkDepartment of Neurology, Dysautonomia Center, New York University School of Medicine, New York, NY, USA.
Mikhail KazachkovDepartment of Pediatrics, New York University Langone Medical Center, New York, NY, USA.
Sanjeev V KothareDepartment of Neurology, Dysautonomia Center, New York University School of Medicine, New York, NY, USA. Electronic address: sanjeevkothare@hotmail.com.
Dysautonomia Foundation · USNYU Langone Health · USUniversity of Massachusetts Chan Medical School · US

Funding

Training CoreU54NS065736 · NINDS · VANDERBILT UNIVERSITY MEDICAL CENTER · PI FREEMAN, ROY · 2009 to 2018
$11.6M
NINDS NIH HHS U54 NS065736
6 · The paper itself

Abstract

objectiveFamilial dysautonomia (FD) is an autosomal recessive disorder characterized by impaired development of sensory and afferent autonomic nerves. Untreated sleep-disordered breathing (SDB) has been reported to increase the risk of sudden unexpected death in FD. We aimed to describe the prevalence and characteristics of SDB in FD. PATIENTS/

methodsSeventy-five patients with FD (20 adults and 55 children) underwent in-lab polysomnography, including peripheral capillary oxygen saturation (SpO

resultsOverall, 85% of adults and 91% of pediatric patients had some degree of SDB. Obstructive sleep apneas were more severe in adults (8.5 events/h in adults vs. 3.5 events/h in children, p = 0.04), whereas central apneas were more severe (10.8 vs. 2.8 events/h, p = 0.04) and frequent (61.8% vs. 45%, p = 0.017) in children. Overall, a higher apnea-hypopnea index was associated with increased severity of hypoxia and hypoventilation, although in a significant fraction of patients (67% and 46%), hypoxemia and hypoventilation occurred independent of apneas.

conclusionMost adult and pediatric patients with FD suffer from some degree of SDB. There was a differential effect of age in the pattern of SDB observed. In some FD patients, hypoventilation and hypoxia occurred independently of apneas. Therefore, we recommend including EtCO

Indexed as

AdolescentAdultAge FactorsAutonomic Nervous SystemDysautonomia, FamilialFemaleHumansHypoxiaMaleOxygen ConsumptionPolysomnographyPrevalenceProspective StudiesSeverity of Illness IndexSleep Apnea SyndromesUnited StatesEtCO(2)Familial dysautonomiaPolysomnographySleep-disordered breathingSpO(2)

Identifiers

PMID29680425
PMCPMC5918267
OpenAlexW2793571327

What OpenQuestion holds

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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.