Evidence map›Paper›PMID 41695072›Full record

ArticleJPGN reports2026

Maralixibat for the treatment of severe xanthomas in two children with Alagille syndrome: Case reports.

Geetanjali Bora, Ruben E Quiros-Tejeira, Bernadette Vitola

Abstract read
In one paragraph

Article in JPGN reports, 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. 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

3 authors.

Geetanjali BoraDivision of Pediatric Gastroenterology Hepatology and Nutrition, Medical College of Wisconsin Milwaukee Wisconsin USA.
Ruben E Quiros-TejeiraDepartment of Pediatrics, Children's Nebraska University of Nebraska Medical Center Omaha Nebraska USA.
Bernadette VitolaDivision of Pediatric Gastroenterology Hepatology and Nutrition, Medical College of Wisconsin Milwaukee Wisconsin USA.ORCID https://orcid.org/0000-0002-6438-2648

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Alagille syndrome (ALGS) is a rare, autosomal dominant disorder which presents with a broad range of clinical manifestations, including cholestatic pruritus. A unique manifestation of ALGS is the presence of xanthomas in 24%-42% of patients, which can lead to liver transplantation. Maralixibat, an ileal bile acid transporter (IBAT) inhibitor, has demonstrated improvements in both cholestatic pruritus and xanthomas in clinical trials. We report here on the use of maralixibat in two patients with ALGS and unusual manifestations of xanthomatosis, including one patient with airway xanthomas and a second patient with severe, diffuse xanthomas. In both cases, almost complete resolution of severe, debilitating xanthomas and clinically meaningful improvements in pruritus and serum bile acid levels were observed after up to 1 year of treatment with maralixibat. These cases support the utilization of maralixibat for the management of ALGS beyond cholestatic pruritus.

Indexed as

cholestasisileal bile acid transporter inhibitorpruritus

Identifiers

PMID41695072
PMCPMC12894057

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.