Evidence map›Paper›PMID 42036217›Full record

ReviewJournal of inherited metabolic disease2026

2025 Consensus Clinical Management Guidelines for Niemann-Pick Disease Type C.

Tarekegn Hiwot, Forbes D Porter, Tatiana Bremova-Ertl, Uma Ramaswami, Caroline Hastings, Bénédicte Héron, Justin Hopkin, Joella Melville, Hernan Amartino, Mireia Del Toro and 24 more

Abstract readReview
In one paragraph

Review in Journal of inherited metabolic disease, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing 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

1 citing paper in PubMed.

  1. 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

34 authors.

Tarekegn HiwotInstitute of Metabolism and System Research, University of Birmingham, Birmingham, UK.ORCID https://orcid.org/0000-0002-3629-2338
Forbes D PorterEunice Kennedy Shriver National Institute of Child Health and Human Development, National Institutes of Health, Bethesda, Maryland, USA.ORCID https://orcid.org/0000-0001-9397-0046
Tatiana Bremova-ErtlDepartment of Neurology, University Hospital Inselspital, Bern, Switzerland.
Uma RamaswamiRoyal Free London NHS Foundation Trust, London, UK.ORCID https://orcid.org/0000-0002-4703-7447
Caroline HastingsUCSF Benioff Children's Hospital Oakland, Oakland, California, USA.ORCID https://orcid.org/0000-0003-3149-0762
Bénédicte HéronDepartment of Pediatric Neurology, Reference Center of Lysosomal Diseases, Armand Trousseau Hospital, Paris, France.
Justin HopkinUniversity of Rochester School of Medicine and Dentistry, Rochester, New York, USA.
Joella Melville67health, London, UK.
Hernan AmartinoServicio de Neurología Infantil, Hospital Universitario Austral and Instituto Neurogenia, Buenos Aires, Argentina.
Mireia Del ToroPediatric Neurology Department, Unit of Hereditary Metabolic Disorders, Hospital Universitari Vall d'Hebron, Barcelona, Spain.
Federica DeodatoDivision of Metabolic Diseases and Hepatology, Ospedale Pediatrico Bambino Gesù, IRCCS, Rome, Italy.ORCID https://orcid.org/0000-0002-1914-6048
Fatih EzgüDepartment of Pediatrics, Faculty of Medicine, Gazi University, Ankara, Turkey.
Paul GissenNational Institute for Health Research, Great Ormond Street Hospital, Biomedical Research Centre, University College London, London, UK.
James B GibsonMetabolic Genetics, Dell Children's Medical Group, Austin, Texas, USA.
Can FiciciogluThe Children's Hospital of Philadelphia, Division of Genetics/Metabolism, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, Pennsylvania, USA.
Roberto GiuglianiUniversidade Federal do Rio Grande do Sul, Hospital de Clinicas de Porto Alegre, IMASP, Dasa Genomica, Casa dos Raros, Porto Alegre, Brazil.
Orna Staretz-ChachamCenter for Rare Diseases, Soroka Medical Center, BenGurion University, Beer Sheva, Israel.
Frances PlattDepartment of Pharmacology, University of Oxford, Oxford, UK.
Nathalie GuffonReference Center for Inborn Errors of Metabolism, Hôpital Femme Mère Enfant, Hospices Civils de Lyon, Bron, France.
Kristina JulichThe University of Texas at Austin Dell Medical School, Austin, Texas, USA.ORCID https://orcid.org/0000-0001-5180-7312
Nikola KresojevićNeurology Clinic, University Clinical Centre of Serbia, Faculty of Medicine, University of Belgrade, Belgrade, Serbia.
Anna LehmanFaculty of Medicine, Department of Medical Genetics, University of British Columbia, Vancouver, Canada.
Yann NadjarDépartement de Neurologie/UF Neuro-Métabolisme, Centre de Référence des Maladies Métaboliques et Lysosomales Neurologiques (CRML-Neuro), Hôpital Pitié-Salpêtrière, Paris, France.ORCID https://orcid.org/0000-0003-1466-6957
Susanne A SchneiderINSPIRE-PNRM+, Neuroimaging Center (NIC), University Medical Center of the Johannes Gutenberg University Mainz, Mainz, Germany.
Simon JonesGenomic Medicine, Manchester University National Health Service Foundation Trust, Manchester, UK.
Eugen MengelClinical Science for LSD, SphinCS, Hochheim, Germany.
Michel TchanWestmead Hospital, Westmead, New South Wales, Australia.
Mark WalterfangNeuropsychiatry, Royal Melbourne Hospital, Parkville, Australia.
Ozlem Goker-AlpanLysosomal and Rare Diseases Research and Treatment Center (LDRTC), Fairfax, Virginia, USA.ORCID https://orcid.org/0000-0003-4758-6793
Charlotte DawsonUniversity Hospitals Birmingham NHS Foundation Trust, Birmingham, UK.
Sandra CowieInternational Niemann-Pick Disease Alliance, Washington, Tyne and Wear, UK.
Toni MathiesonNiemann-Pick UK, Washington, Tyne and Wear, UK.ORCID https://orcid.org/0009-0001-4681-665X
Elizabeth Berry-KravisDepartment of Pediatrics, Neurological Sciences, and Anatomy/Cell Biology, Rush University Medical Center, Chicago, Illinois, USA.
Marc C PattersonDepartment of Neurology, Mayo Clinic, Rochester, Minnesota, USA.

Funding

International Niemann-Pick Disease Alliance
6 · The paper itself

Abstract

In 2018, the International Niemann-Pick Disease Alliance (INPDA) and the International Niemann-Pick Disease Registry (INPDR) developed and published comprehensive clinical management guidelines to support inclusive and standardized care pathways in Niemann-Pick disease type C (NPC)-an ultra-rare, autosomal recessive, neurovisceral lysosomal disorder. Since then, advances in diagnostics, care, and the approval of two novel disease-modifying agents have underscored the need to revise these guidelines to ensure safe, consistent, and high-quality care for those affected by NPC. In response, the INPDA and INPDR convened a multidisciplinary Guidelines Development Group (GDG) comprising individuals with NPC expertise from 14 countries across five continents, representing a broad range of specialties, as well as patients and families involved in NPC care. Informed by a comprehensive literature review and two meetings, the GDG systematically reviewed, revised, and updated the 2018 guideline statements, re-evaluating the level of evidence, strength of recommendations, and expert agreement for each. The resulting 2025 consensus clinical management guidelines constitute a timely, up-to-date, and internationally applicable resource for the diagnosis, treatment, and holistic management of individuals with NPC. These guidelines serve as a critical resource for specialist centers, hospital-based medical teams, staff involved in NPC patient care, family physicians and other primary caregivers, and, importantly, patients and their families.

Indexed as

Niemann-Pick Disease, Type CConsensusDisease ManagementHumansPractice Guidelines as TopicdiagnosisguidelinesmanagementNiemann‐Pick disease type CNPCtreatment

Identifiers

PMID42036217
PMCPMC13110917

What Socratic holds

Textmetadata
LicenceCC BY
Read underepoch 390

Registered trials

None linked

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 Socratic graph.