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Case Report
Biliary adenofibroma in a setting of primary biliary cholangitis and small duct destruction
1 Department of Pathology, The University of Texas Health Sciences Center at San Antonio, 7703 Floyd Curl Dr, San Antonio, TX 78229, USA
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Carlos Bouchot
MD, Department of Pathology, The University of Texas Health Sciences Center at San Antonio, 7703 Floyd Curl Dr, San Antonio, TX 78229,
USA
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Article ID: 100100Z11YK2026
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Trinidad C, Bouchot C, Sharkey F, Nazarullah A. Biliary adenofibroma in a setting of primary biliary cholangitis and small duct destruction. J Case Rep Images Pathol 2026;12(1):16–21.ABSTRACT
Biliary adenofibroma (BAF) is a very rare benign fibroepithelial neoplasm of the liver with the potential for malignant transformation. Most published cases present with either nonspecific clinical symptoms or are asymptomatic, typically with unremarkable laboratory findings. To the best of our knowledge, this is the first reported case of BAF in the setting of Primary Biliary Cholangitis (PBC). We present a finding of BAF in a 56-year-old woman with end-stage liver disease due to PBC. The explant liver showed a 2 cm area of distinct yellow-green nodularity in segments 7–8, with microscopic features diagnostic of BAF. We provide a comprehensive literature review of BAF, including characteristic histopathological findings of both benign and malignant forms, a review of pathogenesis, and a brief description of molecular and/or genetic alterations. These findings expand the clinicopathologic spectrum of BAF and underscore its occurrence in the setting of chronic cholestatic liver disease, with implications for recognition and assessment of malignant potential.
Keywords: Adenofibroma, Bile duct, Biliary, Liver, Neoplasm, Primary biliary cholangitis
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Author Contributions
Camille Trinidad - Conception of the work, Design of the work, Acquisition of data, Analysis of data, Drafting the work, Revising the work critically for important intellectual content, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.
Carlos Bouchot - Drafting the work, Revising the work critically for important intellectual content, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.
Francis Sharkey - Conception of the work, Design of the work, Acquisition of data, Revising the work critically for important intellectual content, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.
Alia Nazarullah - Conception of the work, Design of the work, Acquisition of data, Analysis of data, Drafting the work, Revising the work critically for important intellectual content, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.
Guarantor of SubmissionThe corresponding author is the guarantor of submission.
Source of SupportNone
Consent StatementWritten informed consent was obtained from the patient for publication of this article.
Data AvailabilityAll relevant data are within the paper and its Supporting Information files.
Conflict of InterestAuthors declare no conflict of interest.
Copyright© 2026 Camille Trinidad et al. This article is distributed under the terms of Creative Commons Attribution License which permits unrestricted use, distribution and reproduction in any medium provided the original author(s) and original publisher are properly credited. Please see the copyright policy on the journal website for more information.
