Evidence map›Paper›PMID 39376842›Full record

ArticleCureus2024

A Rare Case of Untreated Wilson's Disease in a Teen With Lethal Exit: Morphological Findings From an Autopsy Study.

Anastasia I Bekyarova, Ina Kobakova, Snejana Spasova

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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

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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

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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.

Anastasia I BekyarovaDepartment of General and Clinical Pathology, Forensic Medicine and Deontology, Medical University of Varna, Varna, BGR.
Ina KobakovaDepartment of General and Clinical Pathology, Forensic Medicine and Deontology, Dr. Marko Markov Specialized Hospital for Treatment of Oncological Diseases, Varna, BGR.
Snejana SpasovaDepartment of General and Clinical Pathology, Forensic Medicine and Deontology, Dr. Marko Markov Specialized Hospital for Treatment of Oncological Diseases, Varna, BGR.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Wilson's disease (WD) is an autosomal recessive genetic disorder caused by more than 50 different mutations in the APT7B gene. A defect in the gene product results in copper accumulation mainly in the liver, basal ganglia in the brain, cornea, kidneys, and heart, leading to dysfunction and eventually organ failure. We present a case of a 15-year-old male with a minority background who did not receive any form of treatment and ultimately succumbed to the disease. He was previously hospitalized due to suspected autoimmune-mediated acute liver failure (ALF) with positive antinuclear autoantibodies. Abdominal ultrasound revealed uneven contours and diffusely abnormal structure of the liver, interpreted as liver cirrhosis (LC), and splenomegaly. In view of WD as a potential differential diagnosis, a genetic consultation recommended the performance of genetic testing. The patient received symptomatic and corticosteroid therapy and was discharged from the hospital with improved general status. Three days later, the teen experienced deterioration and was readmitted to the hospital in a critical state. Reanimation measures had a temporary effect and ultimately exitus letalis was registered. The autopsy study revealed mixed micronodular and macronodular LC, chronic steatohepatitis, hepatosplenomegaly, ascites, icterus, gynecomastia, telangiectasias, subcutaneous hemorrhages, absence of male pattern body hair, hypogonadism, and chronic calculous cholecystitis as a result of untreated WD. Complications of the main disease appeared to be hepatorenal syndrome, severe bilateral purulent-hemorrhagic pneumonia probably with mixed etiology, acute cardiac failure with congestive changes in all internal organs, pleural and pericardial effusions, pulmonary edema, and cerebral edema with tonsillar herniation. The ATP7B gene sequencing supported the clinical diagnosis and the autopsy suspicion of WD, showing that the boy was homozygous for an H1069Q mutation.

Indexed as

autopsyminority backgroundmorphologyuntreated wilson's diseasewilson's disease in children

Identifiers

PMID39376842
PMCPMC11457893

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