Evidence map›Paper›PMID 33519922›Full record

ReviewFrontiers in genetics2020

The Skeleton of Lateral Meningocele Syndrome.

Ernesto Canalis

Open access · goldAbstract readReview
In one paragraph

Review in Frontiers in genetics, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 8 papers.

0numbers the graph read from it
0cells of the map it votes in
8citing papers in PubMed
1.2field-weighted citation impact, top 20% 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

8 citing papers in PubMed, 11 citations in OpenAlex.

  1. Article
  2. Article
  3. Role of Notch signaling in corticalization.Journal of endocrinological investigation · 2026
    Review
  4. Article
  5. Article
  6. Article
  7. Article
  8. 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

1 author at 1 institution in 1 country.

Ernesto CanalisDepartment of Orthopaedic Surgery and Medicine, UConn Musculoskeletal Institute, UConn Health, Farmington, CT, United States.
UConn Health · US

Funding

The Skeleton of Lateral Meningocele SyndromeR01AR072987 · NIAMS · UNIVERSITY OF CONNECTICUT SCH OF MED/DNT · PI CANALIS, ERNESTO · 2018 to 2023
$2.2M
Targeting the Notch3 Mutation to Cure Lehman SyndromeR61AR076747 · NIAMS · UNIVERSITY OF CONNECTICUT SCH OF MED/DNT · PI CANALIS, ERNESTO · 2019 to 2020
$799k
Targeting the Notch3 Mutation to Cure Lehman SyndromeR33AR076747 · NIAMS · UNIVERSITY OF CONNECTICUT SCH OF MED/DNT · PI CANALIS, ERNESTO · 2022 to 2022
$411k
NIAMS NIH HHS R01 AR072987NIAMS NIH HHS R33 AR076747NIAMS NIH HHS R61 AR076747
6 · The paper itself

Abstract

Notch (Notch1 through 4) are transmembrane receptors that determine cell differentiation and function, and are activated following interactions with ligands of the Jagged and Delta-like families. Notch has been established as a signaling pathway that plays a critical role in the differentiation and function of cells of the osteoblast and osteoclast lineages as well as in skeletal development and bone remodeling. Pathogenic variants of Notch receptors and their ligands are associated with a variety of genetic disorders presenting with significant craniofacial and skeletal manifestations. Lateral Meningocele Syndrome (LMS) is a rare genetic disorder characterized by neurological manifestations, meningoceles, skeletal developmental abnormalities and bone loss. LMS is associated with NOTCH3 gain-of-function pathogenic variants. Experimental mouse models of LMS revealed that the bone loss is secondary to increased osteoclastogenesis due to enhanced expression of receptor activator of nuclear factor kappa B ligand by cells of the osteoblast lineage. There are no effective therapies for LMS. Antisense oligonucleotides targeting

Indexed as

genetic disorderslateral meningocele syndromeLehman syndromeNotchosteoblastosteoclastosteoporosis

Identifiers

PMID33519922
PMCPMC7841456
OpenAlexW3119097972

What OpenQuestion holds

Textmetadata
LicenceCC BY
Read underepoch 390

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.