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Record W4415588054 · doi:10.1186/s13023-025-03980-1

French national diagnosis and care protocol (Protocole National De Diagnostic et de Soins; PNDS): Gaucher disease

2025· review· en· W4415588054 on OpenAlexaff
Fabrice Camou, J. Serratrice, Magali Pettazzoni, Yann Nadjar, Anaïs Brassier, Soumeya Bekri, B. Cador-Rousseau, Louis Dagneaux, Florence Dalbiès, Roseline Froissart, Delphine Genevaz, Anne‐Sophie Guémann, Bénédicte Hivert, V. Leguy‐Seguin, Catherine Marcel, A. Masseau, Martin Michaud, Yves‐Marie Pers, Jérôme Stirnemann, Sabrina Vergnaud, Yann Nguyen, Catherine Caillaud, Samia Pichard, Marc Berger, Nadia Belmatoug

Bibliographic record

VenueOrphanet Journal of Rare Diseases · 2025
Typereview
Languageen
FieldMedicine
TopicLysosomal Storage Disorders Research
Canadian institutionsHotel Dieu Hospital
Fundersnot available
KeywordsEnzyme replacement therapyGlucocerebrosidaseAsymptomaticDiseaseMagnetic resonance imagingEncephalopathyMultidisciplinary approachGenetic testingCytopenia

Abstract

fetched live from OpenAlex

Gaucher disease (GD) is a rare autosomal recessive lysosomal disorder caused by glucocerebrosidase deficiency, with a prevalence in France of around 1/130,000 people. The clinical picture of GD is very heterogeneous, ranging from lifelong asymptomatic forms to severe forms with onset during childhood, such as GD type 2 (< 1% of cases). GD type 1, the most common form (95% of cases), manifests with varying degrees of organomegaly, cytopenia and bone manifestations. Progressive encephalopathy of varying severity is also observed in GD type 3. Symptoms may result in acute and/or chronic pain and asthenia, and lead to disability. The aim of the French National Diagnosis and Care Protocol (Protocole National de Diagnostic et de Soins; PNDS) is to provide health care professionals with guidance for the optimal management and care of patients with GD. GD diagnosis is usually based on laboratory analyses revealing low or absent glucocerebrosidase activity, and can be confirmed by identification of glucocerebrosidase (GBA1) gene pathogenic variants. Additional assessments should include biological analyses (hemogram test, serum protein electrophoresis, and measurement of GD biomarkers), imaging examinations (X-rays, abdominal and bone magnetic resonance imaging, bone densitometry, echocardiography), and electrocardiogram.Patient management in France is multidisciplinary and should be coordinated by a GD specialist, in conjunction with the Committee for the Evaluation and Treatment of Gaucher Disease, the Reference Center for Lysosomal Diseases or a reference/competence center for inherited metabolic diseases. The indication for treatment is not systematic and is based on the presence of clinical, biological, and imaging criteria. Current treatments such as intravenous enzyme replacement therapy or oral substrate reduction therapy, generally lead to significant improvements in disease characteristics within one to five years and early initiation can prevent complications. Follow-up should include a clinical examination, biological analyses to monitor disease biomarkers twice a year then yearly for stable patients, and imaging evaluations initially every year and then every 3 to 4 years for patients with stable disease in whom therapeutic objectives have been achieved. Intercurrent pathologies can be managed by the attending physician in collaboration with a GD specialist.

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)0.029
metaresearch head score (Gemma)0.049
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Not applicable · Consensus signal: Not applicable
GenreCandidate signal: Protocol · Consensus signal: Protocol
Teacher disagreement score0.072
Threshold uncertainty score0.153

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0290.049
Meta-epidemiology (narrow)0.0010.001
Meta-epidemiology (broad)0.0020.003
Bibliometrics0.0030.002
Science and technology studies0.0020.001
Scholarly communication0.0020.001
Open science0.0020.002
Research integrity0.0040.002
Insufficient payload (model declined to judge)0.0310.007

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 head0.038
GPT teacher head0.408
Teacher spread0.370 · 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 designNot applicable
Domainnot available
GenreProtocol

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

Citations1
Published2025
Admission routes1
Has abstractyes

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