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Record W4386202650 · doi:10.15690/vsp.v22i4.2613

Idursulfase Beta (Hunterase) Therapeutic Experience in a Patient with Mucopolysaccharidosis Type II

2023· article· en· W4386202650 on OpenAlexaff
Nato D. Vashakmadze, Nataliya V. Zhurkova, Ekaterina Zakharova, Ludmila K. Mikhaylova, Marina А. Babaykina

Bibliographic record

VenueВопросы современной педиатрии · 2023
Typearticle
Languageen
FieldMedicine
TopicLysosomal Storage Disorders Research
Canadian institutionsChildren’s Health Research Institute
Fundersnot available
KeywordsHunter syndromeEnzyme replacement therapyMucopolysaccharidosis type IIHeparan sulfateMucopolysaccharidosisDiseaseMedicineLysosomal storage diseasePathologicalInternal medicinePediatricsHeparin

Abstract

fetched live from OpenAlex

Background . Mucopolysaccharidosis type II (MPS II, Hunter syndrome) is an X-linked recessive disease caused by lysosomal enzyme iduronate-2-sulfatase deficiency resulting in progressive glycosaminoglycans (GAG) accumulation in tissues (dermatan sulfate and heparan sulfate). GAG accumulation in cells leads to the development of progressive pathological disorders, malfunction of various organs and systems, early disability, and decrease in life expectancy. Enzyme replacement therapy (ERT) reduces the rate of life-threatening conditions development in patient. ERT would be more effective if it is prescribed early, especially at preclinical stage. This is the time when there are no severe irreversible changes in the cell, thus, pathogenetic therapy will decrease GAG accumulation in lysosomes, slow down the pathological process, and improve patient's condition. Clinical case description . Male patient diagnosed with MPS II was administrated with ERT idursulfase beta at the age of 6 months. It led to milder disease course compared to proband uncle who had similar mutation in the IDS gene, severe disease phenotype, and later initiation of ERT (at the age of 2.5 years). Conclusion . Early ERT initiation in patients with MPS II significantly slows down development of severe and life-threatening complications, increases the duration and improves the quality of life.

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 distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesMeta-epidemiology (narrow), Insufficient payload (model declined to judge)
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.215
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0010.003
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.001
Insufficient payload (model declined to judge)0.0010.001

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 head0.048
GPT teacher head0.336
Teacher spread0.288 · 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 teacher head, not a consensus.

Study designObservational
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
Published2023
Admission routes1
Has abstractyes

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