Inflammatory angiomyolipoma of the liver: a rare hepatic tumor

Liu, Yang; Wang, Jian; Lin, Xu‐Yong; Xu, Hong-Tao; Qiu, Xueshan; Wang, Enhua

doi:10.1186/1746-1596-7-122

Cited by 15 publications

(5 citation statements)

References 23 publications

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“…An association between hepatic angiomyolipoma and IgG4-related disease (IgG4-RD) was suggested in the previous report [ 14 ]. In this case, foci of inflammatory cells were found not only in the tumor area but also in the pancreatic parenchyma at some distance from the tumor.…”

Section: Discussionmentioning

confidence: 99%

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A case of pancreatic PEComa with prominent inflammatory cell infiltration: the inflammatory subtype is a distinct histologic group of PEComa

Tsukita,

Koyama,

Ishinari

et al. 2024

Diagn Pathol

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Background PEComa is a mesenchymal tumor that can occur in various organs including the uterus and soft tissues. PEComas are composed of perivascular epithelioid cells, and angiomyolipoma (AML), clear cell sugar tumor (CCST), and lymphangiomyomatosis (LAM) are considered lesions of the same lineage as tumors of the PEComa family. Histologically, a common PEComa shows solid or sheet-like proliferation of epithelioid cells. This is accompanied by an increase in the number of dilated blood vessels. Here, we report a case of pancreatic PEComa with marked inflammatory cell infiltration. Case presentation A 74-year-old male patient underwent an appendectomy for acute appendicitis. Postoperative computed tomography and magnetic resonance imaging revealed a 30 × 25 mm non-contrast-enhanced circular lesion in the tail of the pancreas. The imaging findings were consistent with a malignant tumor, and distal pancreatectomy was performed. Histologically, most area of the lesion was infiltrated with inflammatory cells. A few epithelioid cells with large, round nuclei, distinct nucleoli, and eosinophilic granular cytoplasm were observed. Spindle-shaped tumor cells were observed. Delicate and dilated blood vessels were observed around the tumor cells. Immunohistochemically, the atypical cells were positive for αSMA, Melan A, HMB-45, and TFE3. The cytological characteristics of the tumor cells and the results of immunohistochemical staining led to a diagnosis of pancreatic PEComa. Conclusions A histological variant known as the inflammatory subtype has been defined for hepatic AML. A small number of tumor cells present with marked inflammatory cell infiltration, accounting for more than half of the lesions, and an inflammatory myofibroblastic tumor-like appearance. To our knowledge, this is the first report of pancreatic PEComa with severe inflammation. PEComa is also a generic term for tumors derived from perivascular epithelioid cells, such as AML, CCST, and LAM. Thus, this case is considered an inflammatory subtype of PEComa. It has a distinctive morphology that is not typical of PEComa. This histological phenotype should be widely recognized.

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Section: Discussionmentioning

confidence: 99%

“…Delicate dilated vessels were present, similar to those observed in typical AML. Cavernous-like sinusoidal vessels were also observed [ 14 ]. The presence of mature adipocytes varied according to previous reports [ 15 , 16 ].…”

Section: Discussionmentioning

confidence: 99%

A case of pancreatic PEComa with prominent inflammatory cell infiltration: the inflammatory subtype is a distinct histologic group of PEComa

Tsukita,

Koyama,

Ishinari

et al. 2024

Diagn Pathol

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“…В нашем случае трудности диагностики были обусловлены наличием изолированного поражения печени без сочетанного поражения почек, которое в мировой литературе описано крайне мало [11]. Также затрудняло дифференциально диагностический поиск наличие миоматозного варианта опухоли.…”

Section: Discussionunclassified

“…В мире было зарегистрировано всего несколько случаев развития ангиомиолипомы в печени без одновременного вовлечения почек [11]. Наличие миоматозного (монофазного) варианта опухоли затрудняет дифференциальную диагностику ангиомиолипомы, так как в подобных случаях опухоль схожа с ГЦК, а также другими гиперваскулярными новообразованиями печени, для которых характерно быстрое вымывание (wash-out) контрастного препарата [1,4,12,16].…”

Section: Introductionunclassified

The Difficulties of Diagnosis Giant Angiomyolipoma of the Liver: Clinical Case and Literature Review

Arablinskii,

Myasnyankina,

Sedova

et al. 2024

jour

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Purpose: To report a clinical case demonstrating difficulties of diagnosis and successful surgical treatment of myomatous type of angiomyolipoma of the liver without simultaneous damage to the kidneys.Material and methods: A female patient, 49 years old, was admitted to the hospital in August 2023, had no specific complaints, an abdominal ultrasound revealed extraperitoneal extraorgan mass in the epigastrium and mesogastrium and masses in the right lobe of liver, most likely hemangioma. The contrast-enhanced MSCT was performed on admission. Patient was rehospitalized for a surgery in September. During the operation the tumor was removed and sent for histological examination, a further immunohistochemical study was also carried out.Results: According to the results of MSCT in S3, S4, S5 of the liver oval-shaped mass (198×104×177 mm) with heterogeneous density and solid, cystic and fat features was found. Three hypovascular hypodense inclusions sizes up to 11 mm was also identified in S7/8. There was no pathologic formation in the kidneys. The liver tumor was surgically removed, the resected material was directed to a scheduled histological examination. To confirm the diagnosis an IHC analysis was also done and it revealed the expression of melanocytic (HBM-45) and smooth muscle (SMA) markers.Discussion: In this case we encountered very sparsely described in the world literature localization of angiomyolipoma — isolated hepatic one (without kidney damage). Like other authors we encountered the similarity of muomatous tumor type to the HCC and adenoma of the liver due to the characteristic features of visualization: hypervascularity, early contrast enhancement into an arterial phase and reduced density relative to the preserved parenchyma of the liver into a delayed phase of contrast.Conclusion: This case demonstrates the difficulty of diagnosing the myomatous type of AML simulating the HCC. As one of the most informative methods of diagnosis, we single out MSCT, which allowed us to think about the possibility of a benign nature of the tumor. In our case the main features excluding HCC were the absence of rapid “washout” of contrast drug, the presence of lipomatous areas and no accumulation of contrast agent by the capsule.

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“…Hepatic tumor has different cell types, such as hepatocellular carcinoma, cholangiocarcinoma, bile duct cystadenocarcinoma, combined hepatocellular and cholangiocarcinoma, hepatoblastoma, undifferentiated carcinoma, hepatic angiomyolipoma, while different biomarkers have been developed and investigated for diagnosis, tumor progression, and prognosis of them [23-26]. It is also well recognized that biomarkers which may play important roles in physiologic and pathologic processes are important for diagnosis, prognosis, and prediction of HCC.…”

Section: Discussionmentioning

confidence: 99%

Serum peptidome patterns of hepatocellular carcinoma based on magnetic bead separation and mass spectrometry analysis

et al. 2013

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BackgroundHepatocellular carcinoma (HCC) is one of the most common cancers in the world,and the identification of biomarkers for the early detection is a relevant target. The purpose of the study is to discover specific low molecular weight (LMW) serum peptidome biomarkers and establish a diagnostic pattern for HCC.MethodsWe undertook this pilot study using a combined application of magnetic beads with Matrix-assisted laser desorption/ionization time-of-flight mass spectrometry (MALDI-TOF MS) technique and ClinPro Tools v2.2 to detect 32 patients with HCC, 16 patients with chronic hepatitis (CH), 16 patients with liver cirrhosis (LC) and 16 healthy volunteers.ResultsThe results showed 49, 33 and 37 differential peptide peaks respectively appeared in HCC, LC and CH groups. A Supervised Neural Network (SNN) algorithm was used to set up the classification model. Eleven of the identified peaks at m/z 5247.62, 7637.05, 1450.87, 4054.21, 1073.37, 3883.64, 5064.37, 4644.96, 5805.51, 1866.47 and 6579.6 were used to construct the peptides patterns. According to the model, we could clearly distinguish between HCC patients and healthy controls as well as between LC or CH patients and healthy controls.ConclusionsThe study demonstrated that a combined application of magnetic beads with MALDI-TOF MB technique was suitable for identification of potential serum biomarkers for HCC and it is a promising way to establish a diagnostic pattern.Virtual slidesThe virtual slide(s) for this article can be found here: http://www.diagnosticpathology.diagnomx.eu/vs/1503629821958720.

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Inflammatory angiomyolipoma of the liver: a rare hepatic tumor

Cited by 15 publications

References 23 publications

A case of pancreatic PEComa with prominent inflammatory cell infiltration: the inflammatory subtype is a distinct histologic group of PEComa

A case of pancreatic PEComa with prominent inflammatory cell infiltration: the inflammatory subtype is a distinct histologic group of PEComa

The Difficulties of Diagnosis Giant Angiomyolipoma of the Liver: Clinical Case and Literature Review

Serum peptidome patterns of hepatocellular carcinoma based on magnetic bead separation and mass spectrometry analysis

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