Evidence map›Paper›PMID 37808418›Full record

ArticleRadiology case reports2023

Guanidinoacetate N-methyltransferase deficiency: Case report and brief review of the literature.

Joshua L Libell, Dhairya A Lakhani, Aneri B Balar, Musharaf Khan, Jeffrey S Carpenter, Joe T Joseph

Abstract readCase Reports
In one paragraph

Article in Radiology case reports, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 5 papers.

0numbers the graph read from it
0cells of the map it votes in
5citing papers in PubMed
–field-weighted citation impact
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

5 citing papers in PubMed.

  1. Article
  2. Article
  3. Transcriptomic Profiling of Zebrafish Mutant forInternational journal of molecular sciences · 2025
    Article
  4. Article
  5. [Two cases of creatine deficiency syndrome caused byZhongguo dang dai er ke za zhi = Chinese journal of contemporary pediatrics · 2025
    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

6 authors.

Joshua L LibellSchool of Medicine, West Virginia University, Morgantown, WV, 26506, USA.
Dhairya A LakhaniDepartment of Radiology, West Virginia University, 1 Medical Center Dr, Morgantown, WV 26506, USA.
Aneri B BalarDepartment of Radiology, West Virginia University, 1 Medical Center Dr, Morgantown, WV 26506, USA.
Musharaf KhanDepartment of Radiology, West Virginia University, 1 Medical Center Dr, Morgantown, WV 26506, USA.
Jeffrey S CarpenterDepartment of Radiology, West Virginia University, 1 Medical Center Dr, Morgantown, WV 26506, USA.
Joe T JosephDepartment of Radiology, West Virginia University, 1 Medical Center Dr, Morgantown, WV 26506, USA.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Guanidinoacetate N-methyltransferase (GAMT) deficiency is a rare autosomal recessive disorder characterized by a decrease in creatine synthesis, resulting in cerebral creatine deficiency syndrome (CCDS). GAMT deficiency is caused by mutations in the GAMT gene located on chromosome 19, which impairs the conversion of guanidinoacetic acid (GAA) to creatine. The resulting accumulation of the toxic metabolite GAA and the lack of creatine lead to various symptoms, including global developmental delays, behavioral issues, and epilepsy. The gold standard for diagnosis of GAMT deficiency is genetic testing. Treatment options for GAMT deficiency include creatine supplementation, ornithine supplementation, arginine restriction, and sodium benzoate supplementation. These treatment options have been shown to improve movement disorders and epileptic symptoms, but their impact on intellectual and speech development is limited. Early intervention has shown promising results in normalizing neurological development in a minor subgroup of patients. Therefore, there is a growing need for newborn screening techniques to detect GAMT deficiency early and prevent permanent neurological delays. Here we report a case of GAMT deficiency with emphasis on imaging presentation. Our case showed reduced brain parenchyma creatine stores on MR Spectroscopy, which may provide an avenue to aid in early diagnosis.

Indexed as

Cerebral creatine deficiency syndromeCreatineGenetic testingGlobal developmental delayGuanidinoacetate N-MethyltransferaseMagnetic resonance spectroscopy

Identifiers

PMID37808418
PMCPMC10550807

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Registered trials

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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.