Evidence map›Paper›PMID 35314496›Full record

ArticleNeurology2022

Development and Validation of the Pediatric Hypersomnolence Survey.

Kiran Maski, Jennifer Worhach, Erin Steinhart, Madeline Boduch, Anne Marie Morse, Michael Strunc, Thomas Scammell, Judith Owens, Lindsay Jesteadt, Claire Crisp and 2 more

Open access · greenAbstract read
In one paragraph

Article in Neurology, 2022. 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
1.2field-weighted citation impact, top 24% 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, 11 citations in OpenAlex.

  1. Article
  2. Recommended protocols for the Multiple Sleep Latency Test and Maintenance of Wakefulness Test in children: guidance from the American Academy of Sleep Medicine.Journal of clinical sleep medicine : JCSM : official publication of the American Academy of Sleep Medicine · 2024
    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

12 authors at 1 institution in 2 countries.

Kiran MaskiFrom the Department of Neurology (K.M., J.W., M.B., T.S., J.O.), Boston Children's Hospital; Massachusetts General Hospital (E.S.), Boston; Department of Neurology (A.M.), Geisinger Medical Center, Danville, PA; Department of Neurology (M.S.), Children's Hospital of the King's Daughter, Norfolk, VA; Department of Neurology (T.S.), Beth Israel Deaconess Medical Center, Boston; 6. Wake Up Narcolepsy, Inc (L.J., C.C.), Worcester; and ICCTR Biostatistics and Research Design Center (D.W., G.S.), Boston Children's Hospital, MA.ORCID 0000-0003-4073-4036
Jennifer WorhachFrom the Department of Neurology (K.M., J.W., M.B., T.S., J.O.), Boston Children's Hospital; Massachusetts General Hospital (E.S.), Boston; Department of Neurology (A.M.), Geisinger Medical Center, Danville, PA; Department of Neurology (M.S.), Children's Hospital of the King's Daughter, Norfolk, VA; Department of Neurology (T.S.), Beth Israel Deaconess Medical Center, Boston; 6. Wake Up Narcolepsy, Inc (L.J., C.C.), Worcester; and ICCTR Biostatistics and Research Design Center (D.W., G.S.), Boston Children's Hospital, MA.ORCID 0000-0002-5254-2894
Erin SteinhartFrom the Department of Neurology (K.M., J.W., M.B., T.S., J.O.), Boston Children's Hospital; Massachusetts General Hospital (E.S.), Boston; Department of Neurology (A.M.), Geisinger Medical Center, Danville, PA; Department of Neurology (M.S.), Children's Hospital of the King's Daughter, Norfolk, VA; Department of Neurology (T.S.), Beth Israel Deaconess Medical Center, Boston; 6. Wake Up Narcolepsy, Inc (L.J., C.C.), Worcester; and ICCTR Biostatistics and Research Design Center (D.W., G.S.), Boston Children's Hospital, MA.
Madeline BoduchFrom the Department of Neurology (K.M., J.W., M.B., T.S., J.O.), Boston Children's Hospital; Massachusetts General Hospital (E.S.), Boston; Department of Neurology (A.M.), Geisinger Medical Center, Danville, PA; Department of Neurology (M.S.), Children's Hospital of the King's Daughter, Norfolk, VA; Department of Neurology (T.S.), Beth Israel Deaconess Medical Center, Boston; 6. Wake Up Narcolepsy, Inc (L.J., C.C.), Worcester; and ICCTR Biostatistics and Research Design Center (D.W., G.S.), Boston Children's Hospital, MA.
Anne Marie MorseFrom the Department of Neurology (K.M., J.W., M.B., T.S., J.O.), Boston Children's Hospital; Massachusetts General Hospital (E.S.), Boston; Department of Neurology (A.M.), Geisinger Medical Center, Danville, PA; Department of Neurology (M.S.), Children's Hospital of the King's Daughter, Norfolk, VA; Department of Neurology (T.S.), Beth Israel Deaconess Medical Center, Boston; 6. Wake Up Narcolepsy, Inc (L.J., C.C.), Worcester; and ICCTR Biostatistics and Research Design Center (D.W., G.S.), Boston Children's Hospital, MA.ORCID 0000-0003-4494-0195
Michael StruncFrom the Department of Neurology (K.M., J.W., M.B., T.S., J.O.), Boston Children's Hospital; Massachusetts General Hospital (E.S.), Boston; Department of Neurology (A.M.), Geisinger Medical Center, Danville, PA; Department of Neurology (M.S.), Children's Hospital of the King's Daughter, Norfolk, VA; Department of Neurology (T.S.), Beth Israel Deaconess Medical Center, Boston; 6. Wake Up Narcolepsy, Inc (L.J., C.C.), Worcester; and ICCTR Biostatistics and Research Design Center (D.W., G.S.), Boston Children's Hospital, MA.
Thomas ScammellFrom the Department of Neurology (K.M., J.W., M.B., T.S., J.O.), Boston Children's Hospital; Massachusetts General Hospital (E.S.), Boston; Department of Neurology (A.M.), Geisinger Medical Center, Danville, PA; Department of Neurology (M.S.), Children's Hospital of the King's Daughter, Norfolk, VA; Department of Neurology (T.S.), Beth Israel Deaconess Medical Center, Boston; 6. Wake Up Narcolepsy, Inc (L.J., C.C.), Worcester; and ICCTR Biostatistics and Research Design Center (D.W., G.S.), Boston Children's Hospital, MA.
Judith OwensFrom the Department of Neurology (K.M., J.W., M.B., T.S., J.O.), Boston Children's Hospital; Massachusetts General Hospital (E.S.), Boston; Department of Neurology (A.M.), Geisinger Medical Center, Danville, PA; Department of Neurology (M.S.), Children's Hospital of the King's Daughter, Norfolk, VA; Department of Neurology (T.S.), Beth Israel Deaconess Medical Center, Boston; 6. Wake Up Narcolepsy, Inc (L.J., C.C.), Worcester; and ICCTR Biostatistics and Research Design Center (D.W., G.S.), Boston Children's Hospital, MA.
Lindsay JesteadtFrom the Department of Neurology (K.M., J.W., M.B., T.S., J.O.), Boston Children's Hospital; Massachusetts General Hospital (E.S.), Boston; Department of Neurology (A.M.), Geisinger Medical Center, Danville, PA; Department of Neurology (M.S.), Children's Hospital of the King's Daughter, Norfolk, VA; Department of Neurology (T.S.), Beth Israel Deaconess Medical Center, Boston; 6. Wake Up Narcolepsy, Inc (L.J., C.C.), Worcester; and ICCTR Biostatistics and Research Design Center (D.W., G.S.), Boston Children's Hospital, MA.
Claire CrispFrom the Department of Neurology (K.M., J.W., M.B., T.S., J.O.), Boston Children's Hospital; Massachusetts General Hospital (E.S.), Boston; Department of Neurology (A.M.), Geisinger Medical Center, Danville, PA; Department of Neurology (M.S.), Children's Hospital of the King's Daughter, Norfolk, VA; Department of Neurology (T.S.), Beth Israel Deaconess Medical Center, Boston; 6. Wake Up Narcolepsy, Inc (L.J., C.C.), Worcester; and ICCTR Biostatistics and Research Design Center (D.W., G.S.), Boston Children's Hospital, MA.
David WilliamsFrom the Department of Neurology (K.M., J.W., M.B., T.S., J.O.), Boston Children's Hospital; Massachusetts General Hospital (E.S.), Boston; Department of Neurology (A.M.), Geisinger Medical Center, Danville, PA; Department of Neurology (M.S.), Children's Hospital of the King's Daughter, Norfolk, VA; Department of Neurology (T.S.), Beth Israel Deaconess Medical Center, Boston; 6. Wake Up Narcolepsy, Inc (L.J., C.C.), Worcester; and ICCTR Biostatistics and Research Design Center (D.W., G.S.), Boston Children's Hospital, MA.
Georgios SideridisFrom the Department of Neurology (K.M., J.W., M.B., T.S., J.O.), Boston Children's Hospital; Massachusetts General Hospital (E.S.), Boston; Department of Neurology (A.M.), Geisinger Medical Center, Danville, PA; Department of Neurology (M.S.), Children's Hospital of the King's Daughter, Norfolk, VA; Department of Neurology (T.S.), Beth Israel Deaconess Medical Center, Boston; 6. Wake Up Narcolepsy, Inc (L.J., C.C.), Worcester; and ICCTR Biostatistics and Research Design Center (D.W., G.S.), Boston Children's Hospital, MA.
Boston Children's Hospital · US

Funding

Disrupted Nighttime Sleep (DNS) in Pediatric NarcolepsyK23NS104267 · NINDS · BOSTON CHILDREN'S HOSPITAL · PI MASKI, KIRAN PRASAD · 2019 to 2022
$754k
NINDS NIH HHS K23 NS104267
6 · The paper itself

Abstract

BACKGROUND AND

objectivesNarcolepsy and idiopathic hypersomnia usually begin in early adolescence, but diagnostic delays ranging from 5 to 10 years are common, affecting disease burden. To improve early identification of these treatable conditions, we developed and validated the Pediatric Hypersomnolence Survey (PHS).

methodsContent was developed through literature review, patient focus groups, interviews with experts in the field, and field testing. We then validated the 14-item self-reported survey across 3 hospitals and web recruitment from patient groups. In the validation phase, we recruited a total of 331 participants (patients with narcolepsy type 1 [n = 64], narcolepsy type 2 [n = 34], idiopathic hypersomnia [n = 36], and other sleep disorders [n = 97] and healthy controls [n = 100], ages 8-18 years) to complete the survey. We assessed a range of psychometric properties, including discriminant diagnostic validity for CNS disorders of hypersomnolence using receiver operating characteristic curve analysis and reliability across a 1-week period.

resultsConfirmatory factor analysis indicated a 4-domain solution with good reliability expressed by satisfactory omega values. Across groups, the PHS total score showed appropriate positive correlations with other validated surveys of sleepiness ( DISCUSSION: The PHS is a valid and reliable tool for clinicians to identify pediatric patients with narcolepsy and idiopathic hypersomnia. Implemented in clinical practice, the PHS will potentially decrease diagnostic delays and time to treatment, ultimately reducing disease burden for these debilitating conditions. CLASSIFICATION OF EVIDENCE: This study provides Class III evidence that the PHS accurately identifies patients with central disorders of hypersomnolence.

Indexed as

Disorders of Excessive SomnolenceIdiopathic HypersomniaNarcolepsyAdolescentChildHumansReproducibility of ResultsSleep Latency

Identifiers

PMID35314496
PMCPMC9141629
OpenAlexW4220812303

What OpenQuestion holds

Textmetadata
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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.