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HYPERORNITHINEMIA, HYPERAMMONEMIA, HOMOCITRULLINEMIA (HHH) SYNDROME PRESENTING WITH SEVERE LIVER FAILURE

2006· article· en· W4391606017 on OpenAlexaff
Muath Al Turaiki, Hein Huyn, Alicia Chan, Halliday Idikio

Bibliographic record

VenueJournal of Pediatric Gastroenterology and Nutrition · 2006
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicMetabolism and Genetic Disorders
Canadian institutionsUniversity of Alberta Hospital
Fundersnot available
KeywordsHyperammonemiaMedicineLiver failurePediatricsSurgeryInternal medicine

Abstract

fetched live from OpenAlex

Hyperornithinemia, Hyperammonemia, Homocitrullinemia (HHH) syndrome is a rare autosomal recessive disorder of amino acid metabolism, which was first described by Shih et al in 1969. It has now been described in more than 50 patients. Patients present clinically with vomiting, lethargy, impaired growth and delayed developmental milestones. The characteristic neurological picture may include spastic gait, pyramidal tract signs, seizures and ataxia. As it is a urea cycle defect, patients will have elevated serum ammonia. The amino acid ornithine is elevated in the plasma and homocitrulline is elevated in the urine. The gene (ORNT1) for HHH syndrome has been identified and maps to 13q14. Different mutations in this gene have been reported. Liver involvement such as mild coagulopathy, hepatitis picture and acute liver failure responding to a protein restricted diet has been reported in HHH syndrome. Histopathology of the liver may show the typical picture of vacuolated hepatocytes filled with glycogen. Bizarre shapes and structures of the mitochondria are the usual findings by electron microscopy. We report a child who presented with severe coagulopathy and elevated liver transaminases. She was transferred to our centre for a possible liver transplant. A diagnosis of HHH syndrome was confirmed and the child was treated for her HHH syndrome. Complete recovery of the liver function was achieved. Once a protein restricted diet with adequate calories was started along with citrulline supplementation, the liver function including the elevated liver enzymes and coagulopathy dramatically returned to normal. To our knowledge, there are no reported cases in the literature of a patient who has HHH syndrome presenting with such severe liver failure. It is important to consider the diagnosis of HHH syndrome in patients presenting with liver failure and to treat them aggressively prior to considering liver transplantation.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

How this classification was reachedexpand

Full frame machine prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.001
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Case report · Consensus signal: Case report
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.002
Threshold uncertainty score0.006

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0010.001
Science and technology studies0.0010.001
Scholarly communication0.0000.001
Open science0.0000.001
Research integrity0.0010.001
Insufficient payload (model declined to judge)0.0020.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.003
GPT teacher head0.183
Teacher spread0.180 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it

Classification

machine, unvalidated

Machine predicted; a candidate call from one source (direct Gemma or distilled Codex), not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designCase report
Domainnot available
GenreEmpirical

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

Quick stats

Citations0
Published2006
Admission routes1
Has abstractyes

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