Evidence map›Paper›PMID 35756860›Full record

ArticleMolecular genetics and metabolism reports2022

Maple syrup urine disease decompensation misdiagnosed as a psychotic event.

Tomoyasu Higashimoto, Matthew T Whitehead, Erin MacLeod, Danielle Starin, Debra S Regier

Open access · goldAbstract readCase Reports
In one paragraph

Article in Molecular genetics and metabolism reports, 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
0.5field-weighted citation impact, top 36% 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, 5 citations in OpenAlex.

  1. Review
  2. Review
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

5 authors at 3 institutions in 1 country.

Tomoyasu HigashimotoNational Human Genome Research Institute, National Institutes of Health, Bethesda, MD, United States of America.
Matthew T WhiteheadDivision of Radiology, Children's National Hospital, Washington, DC, United States of America.
Erin MacLeodChildren's National Rare Disease Institute, Washington, DC, United States of America.
Danielle StarinChildren's National Rare Disease Institute, Washington, DC, United States of America.
Debra S RegierChildren's National Rare Disease Institute, Washington, DC, United States of America.
Children's National · USNational Institutes of Health · USUniversity of Pennsylvania · US

Funding

Postdoctoral Medical Genetics Training GrantT32GM007471 · NIGMS · JOHNS HOPKINS UNIVERSITY · PI TAO WANG · 1985 to 2026
$8.0M
NIGMS NIH HHS T32 GM007471
6 · The paper itself

Abstract

Maple syrup urine disease (MSUD) is an autosomal recessive metabolic disease resulting in impaired or absent breakdown of branched-chain amino acids (BCAA) valine, isoleucine, and leucine. Classic MSUD often presents in post-natal periods, at times before newborn screening results, and is treated with a protein restricted diet supplemented with medical food and close follow up to prevent toxic buildup of blood leucine. Acute episodes of decompensation are prevented by early recognition and treatment. Acute episodes of metabolic decompensation in patients with MSUD are medical emergencies that require immediate treatments as cerebral edema may lead to brain-stem compression resulting in death. As the early outcomes improve for MSUD patients, the long-term sequelae of chronic hyperleucemia are being elucidated and include cognitive impairment, mental health disorders, and movement disorders. In this report we present an adult patient with MSUD with attention deficit, hyperactivity type (ADHD) and depression due to prolonged exposure to elevated leucine managed with community support services who presented to the emergency department with new onset of acute hallucinations. He was held in the emergency department awaiting involuntary commitment to a psychiatric facility and underwent psychiatric treatments for suspected new onset hallucinations without improvement. Upon notification of metabolic specialists and initiation of appropriate therapy of MSUD, his leucine level normalized rapidly with resolution of his acute psychosis. This case describes the acute presentation of psychosis in the setting of long-term toxicity of leucine. This case also highlights the importance of transition of care, education and planning in patients with inborn errors of metabolism.

Indexed as

ADHD, attention deficit hyperactivity disorderBCAA, branch chain amino acidsBranched-chain amino acidsBranched-chain α-ketoacid dehydrogenase enzyme complexCT, computed tomographyIEM, inborn errors of metabolismMaple syrup urine diseaseMSUD, maple syrup urine diseaseTransition of care

Identifiers

PMID35756860
PMCPMC9218201
OpenAlexW4283067279

What OpenQuestion holds

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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.