Evidence map›Paper›PMID 27491211›Full record

ReviewPediatric endocrinology reviews : PER2016

Combination Therapies for Lysosomal Storage Diseases: A Complex Answer to a Simple Problem.

Shannon L Macauley

Open access · greenAbstract readReview
In one paragraph

Review in Pediatric endocrinology reviews : PER, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 9 papers.

0numbers the graph read from it
0cells of the map it votes in
9citing papers in PubMed
1.4field-weighted citation impact, top 18% 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

9 citing papers in PubMed, 16 citations in OpenAlex.

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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.

Shannon L Macauley

Funding

Effects of Hyperglycemia on Neuronal Activity, Cerebral Metabolism, and Amyloid-beta LevelsK01AG050719 · NIA · WASHINGTON UNIVERSITY · PI MACAULEY-RAMBACH, SHANNON L · 2016 to 2020
$609k
Effects of Altered Glucose Utilization on AB Levels and Functional ConnectivityF32NS080320 · NINDS · WASHINGTON UNIVERSITY · PI MACAULEY-RAMBACH, SHANNON L · 2013 to 2015
$126k
NIA NIH HHS K01 AG050719NINDS NIH HHS F32 NS080320
6 · The paper itself

Abstract

Abstract Lysosomal storage diseases (LSDs) are a group of 40-50 rare monogenic disorders that result in disrupted lysosomal function and subsequent lysosomal pathology. Depending on the protein or enzyme deficiency associated with each disease, LSDs affect an array of organ systems and elicit a complex set of secondary disease mechanisms that make many of these disorders difficult to fully treat. The etiology of most LSDs is known and the innate biology of lysosomal enzymes favors therapeutic intervention, yet most attempts at treating LSDs with enzyme replacement strategies fall short of being curative. Even with the advent of more sophisticated approaches, like substrate reduction therapy, pharmacologic chaperones, gene therapy or stem cell therapy, comprehensive treatments for LSDs have yet to be achieved. Given the limitations with individual therapies, recent research has focused on using a combination approach to treat LSDs. By coupling protein-, cell-, and gene- based therapies with small molecule drugs, researchers have found greater success in eradicating the clinical features of disease. This review seeks to discuss the positive and negatives of singular therapies used to treat LSDs, and discuss how, in combination, studies have demonstrated a more holistic benefit on pathological and functional parameters. By optimizing routes of delivery, therapeutic timing, and targeting secondary disease mechanisms, combination therapy represents the future for LSD treatment.

Indexed as

Bone Marrow TransplantationCombined Modality TherapyEnzyme Replacement TherapyGenetic TherapyHumansLysosomal Storage Diseases

Identifiers

PMID27491211
PMCPMC5374980
OpenAlexW2519937269

What OpenQuestion holds

Textmetadata
LicenceTDM
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.