Evidence map›Paper›PMID 36941958›Full record

ArticleMolecular genetics and metabolism reports2023

Use of dexamethasone in acute rhabdomyolysis in

Mehdi Yeganeh, Kaitlin March, Catherine Jones, Gloria Ho, Kathryn A Selby, Jean-Pierre Chanoine, Sylvia Stockler, Ramona Salvarinova, Gabriella Horvath, Catherine Brunel-Guitton

Open access · goldAbstract readCase Reports
PubMed Full text at PMC Publisher
In one paragraph

Article in Molecular genetics and metabolism reports, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing papers in PubMed
0.6field-weighted citation impact, top 32% 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

0 citing papers in PubMed, 2 citations in OpenAlex.

No citing paper in PubMed yet.

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

10 authors at 2 institutions in 1 country.

Mehdi YeganehDivision of Biochemical Genetics, Department of Pediatrics, University of British Columbia, BC Children's Hospital, Vancouver V6H 3N1, BC, Canada.
Kaitlin MarchDivision of Biochemical Genetics, Department of Pediatrics, University of British Columbia, BC Children's Hospital, Vancouver V6H 3N1, BC, Canada.
Catherine JonesDivision of Biochemical Genetics, Department of Pediatrics, University of British Columbia, BC Children's Hospital, Vancouver V6H 3N1, BC, Canada.
Gloria HoDivision of Biochemical Genetics, Department of Pediatrics, University of British Columbia, BC Children's Hospital, Vancouver V6H 3N1, BC, Canada.
Kathryn A SelbyDivision of Neurology, Department of Pediatrics, University of British Columbia, BC Children's Hospital, Vancouver, V6H 3N1, BC, Canada.
Jean-Pierre ChanoineDivision of Endocrinology, Department of Pediatrics, University of British Columbia, BC Children's Hospital, Vancouver, V6H 3N1, BC, Canada.
Sylvia StocklerDivision of Biochemical Genetics, Department of Pediatrics, University of British Columbia, BC Children's Hospital, Vancouver V6H 3N1, BC, Canada.
Ramona SalvarinovaDivision of Biochemical Genetics, Department of Pediatrics, University of British Columbia, BC Children's Hospital, Vancouver V6H 3N1, BC, Canada.
Gabriella HorvathDivision of Biochemical Genetics, Department of Pediatrics, University of British Columbia, BC Children's Hospital, Vancouver V6H 3N1, BC, Canada.
Catherine Brunel-GuittonDivision of Biochemical Genetics, Department of Pediatrics, University of British Columbia, BC Children's Hospital, Vancouver V6H 3N1, BC, Canada.
University of British Columbia · CABC Children's Hospital · CA

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Introduction: LPIN1 deficiency is an autosomal recessive form of early childhood recurrent severe rhabdomyolysis. Although not completely lucid yet, LPIN1 has been shown to modulate endosomal-related pro-inflammatory responses via peroxisome proliferator-activated receptor α (PPARα) and PPARγ coactivator 1α (PGC-1α). Treatment with anti-inflammatory agents such as dexamethasone has been proposed to improve the outcome. Case: We report a male toddler with recurrent episodes of complicated rhabdomyolysis, requiring prolonged intensive care unit admissions. Whole exome sequencing revealed a common homozygous 1.7 kb intragenic deletion in Results: Dexamethasone administration shortened the course of active rhabdomyolysis, intensive care admission and rehabilitation. It also prevented rhabdomyolysis-related complications such as kidney injury and compartment syndrome. Conclusion: Our patient showed a favorable response to parenteral dexamethasone, in addition to hyperhydration with IV fluids, sufficient calorie intake, and restricted dietary fat. The improvement with corticosteroids suggests an uncontrolled inflammatory response as the pathophysiology of LPIN1 deficiency.

Indexed as

DexamethasoneHereditary rhabdomyolysisLPIN1

Identifiers

PMID36941958
PMCPMC10024045
OpenAlexW4323927952

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-ND
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.