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Safety and Efficacy of Genistein in Sanfilippo Syndrome - A SystematicReview

2023· review· en· W4324147306 on OpenAlexaboutno aff
Haya Majid, Md Masoom, Mohd Ashif Khan, Aakriti Garg

Bibliographic record

VenueCurrent Drug Therapy · 2023
Typereview
Languageen
FieldMedicine
TopicLysosomal Storage Disorders Research
Canadian institutionsnot available
Fundersnot available
KeywordsMedicineMucopolysaccharidosisCochrane LibraryGenisteinHunter syndromeInternal medicinePediatricsIntensive care medicineMeta-analysis

Abstract

fetched live from OpenAlex

Background: Sanfilippo syndrome, commonly known as mucopolysaccharidosis type III (MPS III), is a rare autosomal recessive lysosomal storage disease that primarily affects the brain and spinal cord. It is caused by a deficiency of enzymes involved in the catabolism of glycosaminoglycan (GAG) and heparin sulfate (HS). Although Genistein has been presented as a potential therapeutic, its safety and efficacy for the treatment of Sanfilippo syndrome are not well established. Objective: This systematic review aims to evaluate the safety and efficacy of Genistein with a primary endpoint of providing an inference whether the medication is producing any improvements when administered in patients suffering from Sanfilippo syndrome. Methods: An intensive computerized literature search was performed according to PRISMA guidelines in major databases such as PubMed, ScienceDirect, Embase, Science Citation Index Expanded, The Cochrane Library, and Web of Science until 30 April 2022. Studies evaluating the efficacy and safety of genistein in patients with Sanfilippo Syndrome were included. The quality of the included studies was assessed using the New Castle Ottawa Scale. Results: A total of 558 studies were identified in the initial search. After removing duplicates, 25 studies were screened based on title and abstract, of which 06 studies were included in this systematic review. Among them, 02 studies were of high quality and 04 studies were of moderate quality. After administration of genistein, a decrease in urinary GAG levels and HS levels was observed. Conclusion: Based on the limited evidence present in the literature, this systematic review suggests that genistein can be safely used to treat Sanfilippo syndrome as it lowers GAG levels. However, data on the long-term benefit profile of genistein and high-quality evidence are still needed as a conclusive result could not be withdrawn.

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)0.001
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesMeta-epidemiology (narrow)
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Not applicable · Consensus signal: none
GenreCandidate signal: Review · Consensus signal: Review
Teacher disagreement score0.782
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.000
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0040.001
Bibliometrics0.0010.001
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.001
Insufficient payload (model declined to judge)0.0000.000

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 head0.133
GPT teacher head0.427
Teacher spread0.293 · 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 designNot applicable
Domainnot available
GenreReview

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

Citations2
Published2023
Admission routes1
Has abstractyes

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