Unexpected Diagnosis in Females with Abnormal Liver Ultrasound

A 69-year-old Nicaraguan woman presented to our liver unit with worsening pain in the right hypochondrium and fever. Her medical history included arterial hypertension, bilateral mastectomy for cancer and breast implant 33 years ago. Physical examination excluded peritoneal irritation. Blood tests revealed thrombocytopenia (78,000/mm3) with normal hemoglobin and white-cell count. Liver function tests were within normal limits. Due to suspicion of underlying chronic liver disease, further tests were requested including HBV-HCV-HIV profiles, all of which were negative; and immunoglobulins G, M and A, all normal. Liver ultrasound was limited due to a hyperechoic beam observed in both hepatic and splenic parenchyma, 2 cm thick, of unknown significance. Abdominal-MRI showed an enlarged liver volume, thickening of periportal spaces and splenomegaly, compatible with chronic liver disease. Additionally, there was evidence of numerous millimetric biliary cists. A liver biopsy was subsequently performed (Fig. 1A,B). 1) Lipopeliosis 2) Immunological diseases (primary biliary cholangitis) 3) Metabolic dysfunction-associated steatohepatitis (MASH) 4) Foreign body reaction Histopathologic features, clinical history and imaging findings were consistent with a diagnosis of hepatic silicone granulomas. Interestingly, the patient had a breast rupture 17 years before and received new prosthetic implants 1 year later. Despite being previously considered an inert material, silicone may infiltrate tissues and organs either locally or at distance. The spread of silicone can occur through both its migratory ability and via the reticuloendothelial system, with consequent dissemination along vessels, perineural areas and tissue planes.1Hudacko R. Anand K. Gordon R. et al.Hepatic silicone granulomas secondary to ruptured breast implants: a report of two cases.Case Rep Hepatol. 2019 Nov 3; 20197348168Google Scholar Besides, when prothesis rupture occurs, direct leakage/diffusion of gel from the prosthesis may trigger a late complication that manifests as a granulomatous reaction known as "siliconoma". Siliconomas can cause pain and typically result in the formation of nodules in lymph nodes, the liver, spleen and nervous system, resembling metastases at radiological imaging in cases of pre-existing cancer. Biopsy samples may show histiocytes stuffed with vacuoles and droplets of silicone in both the tissue and blood, resulting in a "Swiss-cheese-like" appearance due to silicone vacuoles in non-necrotic granuloma.1Hudacko R. Anand K. Gordon R. et al.Hepatic silicone granulomas secondary to ruptured breast implants: a report of two cases.Case Rep Hepatol. 2019 Nov 3; 20197348168Google Scholar,2Kappel RM. Gel Bleed and Rupture of Silicone Breast Implants Investigated by Light-, Electron Microscopy and Energy Dispersive X-ray Analysis of Internal Organs and Nervous Tissue. [cited 2023 Sep 12]; Available from: https://clinmedjournals.org/articles/cmrcr/clinical-medical-reviews-and-case-reports-cmrcr-3-087.php?jid=cmrcr.Google Scholar This chronic inflammatory reaction may lead to periportal fibrosis in the liver parenchyma. Liver siliconomas are a rare condition; literature describing this histologic finding is scarce. Only anecdotal cases have been reported.3Agilinko J. Raj D. Wong K.V. et al.Hepatobiliary complications from ruptured silicone breast implants - a comprehensive literature review.Ger Med Sci GMS E-J. 2021; 19 (Doc05)Google Scholar We herein describe one case of hepatic granulomas observed in a patient with silicone prostheses. At histomorphologic examination, the portal tracts were expanded due to the accumulation of whitish droplets (Fig. 1A); at higher magnification, the droplets were identified as macrophages engulfed with foreign material, resulting in a "Swiss-cheese-like" appearance on liver biopsy (Fig. 1B). The foreign material stained negative for periodic acid Schiff-diastase (Fig. 2A), Congo Red (Fig. 2B) and Alcian blue (Fig. 2C). Certainly, the consideration of differential diagnoses is imperative and includes diseases characterized by fat accumulation or granulomas. Both metabolic dysfunction-associated steatohepatitis and lipopeliosis exhibit fatty accumulation within the lobule rather than the portal tract, with fat accumulating either within the adipocytes or in the space of Disse, respectively. Conversely, hepatic granulomas are observed in up to 4% of liver biopsies. They may arise as a localized response following exposure to microorganisms, foreign bodies, or drugs; or they may indicate immune dysfunction. The presence of central necrosis favors a diagnosis of infection, while involvement of the biliary tract aligns more closely with primary biliary cholangitis. In the case reported herein, these two diagnostic clues were absent, along with typical biochemical or clinical features, thereby ruling out these diagnoses. Furthermore, the presence of foreign material prompted consideration of this diagnosis, which was subsequently confirmed by careful review of the clinical history. It is noteworthy that radiological findings in our patient are not exclusive; despite silicone infiltration of the liver being responsible for the previously mentioned hyperechoic beam, which hindered ultrasound visualization, the material may also aggregate as nodules. This behavior was observed in a second case involving a 58-year-old woman who underwent gluteal silicone injection in 2000. She presented to our liver unit in April 2023 for worsening abdominal pain. A contrast-enhanced abdominal CT scan revealed two hepatic lesions in segments 4-5 (26 mm) and 6-7 (10 mm), with wash-in pattern in delayed phases, as well as several scattered nodular masses in the deep adipose tissue of the lower back and gluteal regions. Biopsy of one of the nodules was then performed; histological analysis revealed the presence of amorphous material filling portal tracts and phagocytosed by macrophages (Fig. 3). The morphological characteristics and staining results were consistent with silicone granulomas. Thus, nodules in the adipose tissue were attributed to silicone spreading along tissue planes. There is no specific treatment for silicone granulomas except for pain management. Our patient was treated with analgesics, resulting in rapid improvement. Besides, due to evidence of advanced liver disease with signs of portal hypertension she was referred to our unit for hepatocellular carcinoma surveillance. In conclusion, this report describes a case of silicone infiltration within the liver in a patient with silicone implants placed several decades prior. The rarity of this condition may be explained by the absence of symptoms and typical incidental radiological findings not necessarily manifesting as liver nodules. A liver biopsy is essential for definitive diagnosis. The authors did not receive any financial support in order to complete the study or write the manuscript. No author reported a conflict of interest with respect to this manuscript. Please refer to the accompanying ICMJE disclosure forms for further details. The authors confirm contribution to the paper as follows: study conception and design: A. Aghemo, F. Colapietro; data collection: F. Colapietro; image data: R. Akpinar, L. Di Tommaso; draft manuscript preparation: all authors. All authors reviewed the results and approved the final version of the manuscript. The following are the supplementary data to this article: Download .pdf (.31 MB) Help with pdf files Multimedia component 1