Biliary duct hamartomas are benign intrahepatic bile duct lesions. Despite being primarily incidental findings on imaging, these lesions can provide a diagnostic conundrum due to their shared characteristics with malignant tumors. The goal of this systematic review is to offer a thorough clinical profile of biliary duct hamartomas. There were 139 cases of biliary duct hamartomas identified in a structured systematic review of the literature. Patient demographics, clinical presentation, significant laboratory and imaging data, diagnostic modalities, treatment choices, and outcomes were all studied and reported. Biliary duct hamartomas present with mild symptoms and laboratory abnormalities, and while being visible on imaging, the results are non-specific and may require biopsy in case of red flag signs such as weight loss and a progressive increase in the size of the lesion. Furthermore, there are currently no published guidelines for the treatment of biliary duct hamartomas, and many people have had surgery despite the clinically benign nature of these abnormalities. As per the findings of the study, individuals who exhibit signs of malignancy should be investigated further. Eyeballing for red flag symptoms, followed by a specialized imaging scan and invasive treatment, is the three-step approach to biliary duct hamartomas. Since our recommendations include a shift in strategy and do not contradict existing rules, there are likely to be few roadblocks to improvement; the key barriers being technological equipment and image quality. In this study, we intended to pave the way for future research in the field. In our opinion, the next decade will bring a better understanding of the characteristics of biliary hamartomas, disease symptoms, and better recognition of any suspicious features. These indications will aid in reducing the number of unneeded surgical or invasive operations. Finally, the findings of these future studies will allow the medical community to improve and provide the best care possible.

Bile duct hamartomas are typically small benign liver lesions that can radiologically mimic metastases on ultrasound and computed tomography, as well as macroscopically. We present a rare and interesting case and review the relevant literature. A 49-year-old woman underwent ultrasound investigation for right upper quadrant pain, which revealed diffuse liver lesions. In the setting of her previous vulval cancer, it was suspected that she had hepatic metastases. This was strongly reinforced with computed tomography and elevated CA 19-9 levels. A liver biopsy revealed diffuse and multifocal bile duct hamartomas and positron emission tomography was negative for metastases or features of cancer recurrence. A diagnosis of diffuse liver hamartomatosis was made. In view of the continuing clinical and laboratory picture, she required regular follow-up. The collective features of this case are unique, as the isolated characteristics of particular interest have not been previously described in the context of a single case. Bile duct hamartomas should be included in the differential diagnosis of multiple liver lesions. CA 19-9 is not a reliable marker for differential diagnosis of this entity.

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