Evidence map›Paper›PMID 40167220›Full record

ArticleAmerican journal of medical genetics. Part C, Seminars in medical genetics2025

Repurposing With Purpose: Treatment of Bachmann-Bupp Syndrome With Eflornithine and Implications for Other Polyaminopathies.

Caleb P Bupp, Elizabeth A VanSickle, Julianne Michael, Chad R Schultz, Kelly Nguyen, Melissa Hoefer, Surender Rajasekaran, André S Bachmann

Abstract read
In one paragraph

Article in American journal of medical genetics. Part C, Seminars in medical genetics, 2025. 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. Article
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

8 authors.

Caleb P BuppCorewell Health and Helen Devos Children's Hospital, Grand Rapids, Michigan, USA.ORCID 0000-0003-4255-0396
Elizabeth A VanSickleCorewell Health and Helen Devos Children's Hospital, Grand Rapids, Michigan, USA.
Julianne MichaelCorewell Health and Helen Devos Children's Hospital, Grand Rapids, Michigan, USA.
Chad R SchultzInternational Center for Polyamine Disorders, Grand Rapids, Michigan, USA.
Kelly NguyenCorewell Health Research Institute, Corewell Health, Grand Rapids, Michigan, USA.
Melissa HoeferCorewell Health Investigative Drug Services Pharmacy, Grand Rapids, Michigan, USA.
Surender RajasekaranCorewell Health and Helen Devos Children's Hospital, Grand Rapids, Michigan, USA.
André S BachmannInternational Center for Polyamine Disorders, Grand Rapids, Michigan, USA.

Funding

Leveraging modulation of polyamine metabolism for therapeutic advantage in genetic disordersR01HD110500 · NICHD · MICHIGAN STATE UNIVERSITY · PI ANDRE S BACHMANN, Caleb Bupp · 2023 to 2026
$2.7M
Acrisure Center for Innovation in Children's HealthCorewell Health Michigan State University AllianceHelen DeVos Children's Hospital FoundationNICHD NIH HHS R01 HD110500NIH HHS
6 · The paper itself

Abstract

Rare diseases impact approximately 1 in 10 people worldwide, and yet, less than 5% of all rare diseases currently have an approved treatment option available. This is due to many challenges unique to rare diseases, including small, diverse patient populations, the cost of drug development that is not proportionate to the number of patients who could potentially benefit from treatment, and difficulty with clinical trial design to validate new therapeutics. As a result, drug repurposing has become an increasingly promising option for finding treatment options for rare diseases. First described in 2018, Bachmann-Bupp Syndrome (BABS) is a rare neurodevelopmental disorder that is caused by gain-of-function variants in the ornithine decarboxylase (ODC1) gene and is characterized by developmental delay, hypotonia, and alopecia. Through collaboration and the use of a unique drug repurposing strategy, the first patient identified with BABS was treated with the repurposed drug eflornithine, also known as α-difluoromethylornithine (DFMO), in just 16 months. Currently, five additional patients with BABS are being treated with DFMO. This model of drug repurposing of an FDA-approved drug for use in another indication can serve as an example of what is possible in the scope of other rare diseases, specifically in other polyaminopathies.

Indexed as

AlopeciaDrug RepositioningEflornithineHumansOrnithine DecarboxylaseRare DiseasesEflornithineOrnithine DecarboxylaseBachmann–Bupp syndromeDFMOdrug repurposinggain‐of‐function variantsODC1rare disease treatment

Identifiers

PMID40167220
PMCPMC12353620

What OpenQuestion holds

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Registered trials

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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 OpenQuestion graph.