Multiple biliary micro-hamartomas diagnosed in an unsuspecting elderly patient

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BACKGROUND

The discovery of multiple biliary hamartomas, as described in this case, is relatively rare and often an incidental finding during imaging studies. Understanding and accurately diagnosing these lesions are important, as they can mimic other hepatic lesions, such as cysts or tumors, leading to potentially unnecessary invasive procedures or treatments [1-4].

 

DESCRIPTION OF THE CASE

Anamnesis:

The patient, an 82-year-old male, presented to the hospital with the primary complaint of abdominal pain. This symptom had been ongoing for weeks and was the primary reason for seeking medical attention. The patient hasn’t a personal/family history significant for liver disease. A thorough medical evaluation was conducted, including physical examination and blood tests. However, these initial assessments did not yield conclusive results or provide a clear diagnosis.

 

Diagnostic assessment.

The patient underwent contrast-enhanced CT and subsequently an MRI [5-6]

The CT revealed the accidental finding of millimetric multiple disorganized hypodense lesions are appreciable, both subcapsular and intraparenchymal, in both hepatic lobes.

After the administration of the contrast medium, they do not show enhancement (Fig.1).

 

Figure 1. Axial CT image: hypodense lesions, without significant contrast enhancement in the arterial and venous phases.

 

The subsequent MRI examination identified the same lesions with different characteristics in various sequences. In T2-weighted MRI images, the areas appear homogeneously and intensely hyperintense (Fig.2).

 

Figure 2. Coronal and axial MRI images with HASTE T2 sequence: tiny multiple hyperintense lesions throughout the hepatic area.

 

In T1-weighted images, all the lesions are homogeneously hypointense.

In diffusion-weighted imaging (DWI), the lesions persist as hyperintense at a low B-value (50) but disappear at a high B-value (800) (Fig.3).

 

Figure 3. Axial MRI image with DWI sequence: hyperintense lesions at low b-value (50) on the right, disappearing at high b-values on the left (800).

 

In T2-weighted MRI-cholangiography sequences, the liver presents a 'florid tree' or 'starry sky' appearance due to the presence of multiple minute hyperintense lesions; however, continuity and communication with the biliary ducts are usually not recognized [7] (Fig.4).

 

Figure 4. MRI image with T2 MRI-cholangiography sequence: multiple small hyperintense lesions, the liver has a 'starry sky' appearance.

The dynamic study after contrast administration typically does not show enhancement in the arterial and venous phases (Fig.5).

 

Figure 5. Axial MRI image with T1 sequence: hypointense lesions, without significant contrast enhancement in the arterial and venous phases.

 

The differential diagnosis.

Radiographic findings can be non-specific and may be hard to differentiate from other lesions.

The differential diagnosis is made with polycystic liver disease, multiple simple hepatic cysts, metastases, micro-abscesses, and Caroli's disease [8]. In the latter, a more heterogeneous appearance of the lesions and communication with the bile ducts is observed.

 

Interventions.

In the case under consideration, the lesions were not directly related to symptoms or complications, and therefore, no surgical or pharmacological treatment was necessary.

If Von Meyenburg complexes are causing symptoms or complications, treatment options may include surgical removal of the affected liver tissue or drainage of large cysts [9-11].

It is essential for individuals diagnosed with these complexes to consult with a hepatologist or liver specialist to determine the appropriate management approach based on their specific case.

 

Follow-up and outcomes.

The management of the patient's condition was grounded in vigilant observation and scheduled monitoring, allowing for the timely assessment of any potential alterations. However, there were no significant changes that led to alterations in the diagnostic choices made.

 

DISCUSSION

The Multiple biliary hamartomas, also known as Von Meyenburg complexes, are rare benign liver lesions characterized by small disorganized cystic structures affecting both hepatic lobes with some predilection for subcapsular regions [12-13]. They are multiple small and round or irregular lesions and are usually 5-30 mm in size when detected by imaging.

These structures are composed of abnormal bile ducts and are typically discovered incidentally during medical imaging studies such as ultrasounds, CT scans, or MRI scans, which are often performed for other reasons. Although they may communicate with the biliary tree, they generally do not. They are thought to arise from embryonic bile duct remnants that have failed to involute.

Von Meyenburg complexes are considered noncancerous and are usually asymptomatic.

They are generally not associated with liver dysfunction or clinical symptoms. Laboratory data are generally nonspecific and within normal limits. Most individuals with these lesions do not require treatment. In some cases, Von Meyenburg complexes can be associated with various liver conditions, including polycystic liver disease, Caroli disease, or congenital hepatic fibrosis [14].

When these conditions are present, they can lead to more significant liver-related problems and may require medical management.

CONCLUSION

Multiple biliary hamartomas represent a fascinating yet rare medical condition, often detected incidentally. This article underscores the importance of accurate diagnosis, highlighting the challenges in distinguishing them from other hepatic lesions. Management based on observation and periodic monitoring can be a prudent approach when the lesions are asymptomatic. However, it is crucial to enhance clinical awareness of this condition to avoid misdiagnosis and unnecessary invasive interventions. Future studies may further our understanding of this condition and its clinical implications.

About the authors

Marina Balbino

Department of Clinical and Experimental Medicine, Foggia University School of Medicine

Email: marinabalbino93@gmail.com
Italy, University School of Medicine Viale L. Pinto 1, 71121 Foggia, Italy.

Manuela Montatore

Department of Clinical and Experimental Medicine, Foggia University School of Medicine

Email: manuela.montatore@unifg.it
Italy, University School of Medicine Viale L. Pinto 1, 71121 Foggia, Italy.

Giacomo Fascia

Department of Clinical and Experimental Medicine, Foggia University School of Medicine

Email: giacomo.fascia@unifg.it
Italy, University School of Medicine Viale L. Pinto 1, 71121 Foggia, Italy

Ruggiero Tupputi

Radiology Unit, “Dimiccoli” Hospital of Barletta

Email: rutudott@gmail.com
Italy, Viale Ippocrate 15, 70051, Barletta (BT), Italy.

Federica Masino

Department of Clinical and Experimental Medicine, Foggia University School of Medicine

Email: federicamasino@gmail.com
Italy, University School of Medicine Viale L. Pinto 1, 71121 Foggia, Italy

Gianmichele Muscatella

Department of Clinical and Experimental Medicine, Foggia University School of Medicine

Email: muscatella94@gmail.com
Italy, University School of Medicine Viale L. Pinto 1, 71121 Foggia, Italy.

Domenico Mannatrizio

Department of Clinical and Experimental Medicine, Foggia University School of Medicine

Email: dr.mannatrizio@gmail.com
Italy, University School of Medicine Viale L. Pinto 1, 71121 Foggia, Italy

Giuseppe Guglielmi

University of Foggia

Author for correspondence.
Email: giuseppe.guglielmi@unifg.it
ORCID iD: 0000-0002-4325-8330

Medical Doctor, Full Professor of Radiology.

Department of Clinical and Experimental Medicine.

Italy

References

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