Evidence map›Paper›PMID 39697951›Full record

ArticleCureus2024

A Novel Compound Heterozygous Mutation in Progressive Familial Intrahepatic Cholestasis (PFIC) 4: A Rare Case Report With Literature Review.

Deepika Pandey, Sanjay Chandnani, Harsh Gandhi, Mavuri Vishal, Pravin M Rathi

Abstract readCase Reports
In one paragraph

Article in Cureus, 2024. 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

5 authors.

Deepika PandeyDepartment of Gastroenterology, Topiwala National Medical College & BYL Nair Charitable Hospital, Mumbai, IND.
Sanjay ChandnaniDepartment of Gastroenterology, Topiwala National Medical College & BYL Nair Charitable Hospital, Mumbai, IND.
Harsh GandhiDepartment of Gastroenterology, Topiwala National Medical College & BYL Nair Charitable Hospital, Mumbai, IND.
Mavuri VishalDepartment of Gastroenterology, Topiwala National Medical College & BYL Nair Charitable Hospital, Mumbai, IND.
Pravin M RathiDepartment of Gastroenterology, Topiwala National Medical College & BYL Nair Charitable Hospital, Mumbai, IND.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

A 12-year-old female, resident of western India, presented with a history of pruritus associated with jaundice for two months. On presentation, she had icterus with mild palpable hepatomegaly. Investigations revealed direct hyperbilirubinemia and elevated transaminases, while gamma-glutamyl transferase levels were normal. Serology for anti-hepatitis A, E, B, and C were negative. Autoimmune markers such as antinuclear antibody, smooth muscle antibody, and anti-liver kidney microsomal antibody were negative. Serum IgG levels were within the normal range. A normal magnetic resonance cholangiopancreatography ruled out any ductal abnormalities. A liver biopsy was also conducted but proved to be inconclusive. Despite extensive workup, the diagnosis remained unclear. However, genetic testing through whole exome sequencing identified a novel compound heterozygous variation, a novel in exon 5 and exon 4 of the Tight-Junction Protein 2 gene, and confirmed the diagnosis of cholestatic liver disease as progressive familial intrahepatic cholestasis type 4. This case highlights the importance of genetic testing for diagnosing cholestatic liver diseases, especially when conventional tests do not provide a clear diagnosis. Whole exome sequencing revealed a novel mutation in the TJP2 gene, ultimately confirming the diagnosis of PFIC4.

Indexed as

ana: antinuclear antibodyanti-lkm: anti-liver kidney microsomal antibodybric: benign recurrent intrahepatic cholestasishcc: hepatocellular carcinomansg: next-generation sequencingpfic: progressive familial intrahepatic cholestasissgot: serum glutamic oxaloacetic transaminasesgpt: serum glutamic pyruvate transaminasesma: smooth muscle antibodytjp2: tight-junction protein 2

Identifiers

PMID39697951
PMCPMC11654999

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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.