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Fat-poor renal angiomyolipoma with prominent cystic degeneration:A case report and review of the literature

World Journal of Clinical Cases 2023年第2期11卷 417-425页

作者： Shi-Qi Lu Wei Lv You-Jun Liu Huan Deng Medical College Nanchang UniversityNanchang 330006Jiangxi ProvinceChina Department of Gastroenterology West China HospitalSichuan UniversityChengdu 610044Sichuan ProvinceChina Department of Radiology The Fourth Affiliated Hospital of Nanchang UniversityNanchang 330003Jiangxi ProvinceChina Department of Pathology The Fourth Affiliated Hospital of Nanchang UniversityNanchang 330003Jiangxi ProvinceChina

BACKGROUND angiomyolipoma(AML),the most common benign tumor of the kidney,is usually composed of dysmorphic blood vessels,smooth muscle,and mature adipose tissue.To our knowledge,AML with cystic degeneration has rarely been documented.Cystic degeneration,hemorrhage,and a lack of fat bring great challenges to the diagnosis.CASE SUMMARY A 60-year-old man with hypertension presented with a 5-year history of cystic mass in his left kidney.He fell 2 mo ago.A preoperative computed tomography(CT)scan showed a mixed-density cystic lesion without macroscopic fat density,the size of which had increased compared with before,probably due to hemorrhage caused by a trauma.Radical nephrectomy was performed.Histopathological studies revealed that the lesion mainly consisted of tortuous,ectatic,and thick-walled blood vessels,mature adipose tissue,and smooth muscle-like spindle cells arranged around the abnormal blood vessels.The tumor cells exhibited positivity for human melanoma black-45,Melan-A,smooth muscle actin,calponin,S-100,and neuron-specific enolase,rather than estrogen receptor,progesterone receptor,CD68,and cytokeratin.The Ki-67 labeling index was less than 5%.The final diagnosis was a fat-poor renal AML(RAML)with prominent cystic degeneration.CONCLUSION When confronting a large renal cystic mass,RAML should be included in the differential diagnosis.

关键词： Kidney angiomyolipoma Cystic degeneration Pathogenesis Diagnosis Case report

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angiomyolipoma arising in the gluteal region

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Chinese Medical Journal 2007年第14期120卷 1284-1286页

作者： Emmanouil Pikoulis Constantine Bramis Othon Michail George Liapis Evangelos Felekouras Vassiliki Kyriakou John Griniatsos First Department of Surgery Laikon Hospital University of Athens Medical SchoolAthens 115-27Greece Department of Pathology Laikon Hospital University of Athens Medical SchoolAthens 115-27Greece

angiomyolipoma （AML） is a tumour of uncertain .histogenesis originally believed to be a hamartomatous lesion, but recently recognized as a usually benign clonal mesenchymal neoplasm. Along with lymphagiomyomatosis （LAM）, clear cell ＂sugar＂ turnout （CCST） and clear cell myelomelanocytic tumour （CCMMT）, AML was classified in the so called perivascular epithelioid cell （PEComa） neoplasm family.

关键词： angiomyolipoma buttocks

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Two case reports and literature review for hepatic epithelioid angiomyolipoma: Pitfall of misdiagnosis

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World Journal of Clinical Cases 2019年第8期7卷 972-983页

作者： Jia-Xi Mao Fei Teng Cong Liu Hang Yuan Ke-Yan Sun You Zou Jia-Yong Dong Jun-Song Ji Jun-Feng Dong Hong Fu Guo-Shan Ding Wen-Yuan Guo Department of Liver Surgery and Organ Transplantation Changzheng Hospital Naval Medical University

BACKGROUND Hepatic epithelioid angiomyolipoma(HEAML) is a rare liver disease and is easily misdiagnosed. Enhanced recognition of HEAML is beneficial to the differential diagnosis of rare liver diseases.CASE SUMMARY We presented two cases of HEAML in Changzheng Hospital, Naval Medical University, and then collected and analyzed all reports about HEAML recorded in PubMed, MEDLINE, China Science Periodical Database, and VIP database from January 2000 to March 2018. A total of 409 cases of HEAML in 97 reports were collected, with a ratio of men to women of 1:4.84 and an age range from 12 years to 80 years(median 44 years). Among the patients with clinical symptoms mentioned, 61.93%(205/331) were asymptomatic, 34.74%(115/331) showed upper or right upper quadrant abdomen discomfort, while a few of them showed abdominal mass, gastrointestinal symptoms, low fever, or weight loss. The misdiagnosis rate of HEAML was as high as 40.34%(165/409) due to its nonspecific imaging findings. Most of the tumors were solitary and round in morphology, with clear boundaries. Ultrasound scan indicated low echo with internal nonuniformity and rich blood supply in most cases. Computer tomography/magnetic resonance imaging enhanced scan showed varied characteristics. The ratio of fast wash-in and fast wash-out, fast wash-in and slow wash-out, and delayed enhancement was roughly 4:5:1. A definite diagnosis of HEAML depended on the pathological findings of the epithelioid cells in lesions and the expression of human melanoma black 45, smooth muscle actin,melanoma antigen, and actin by immunohistochemical staining. HEAML had a relatively low malignant rate of 3.91%. However, surgical resection was the main treatment for HEAML, due to the difficulty diagnosing before operation.CONCLUSION HEAML is a rare and easily misdiagnosed disease, and it should be diagnosed carefully, taking into account clinical course, imaging, pathological,and immunohistochemical findings.

关键词： Hepatic epithelioid angiomyolipoma Imaging Pathology Misdiagnosis Potentially malignant Case report

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Malignancy of renal angiomyolipoma from tuberous sclerosis complex with TSC2 mutation

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Chinese Medical Journal 2019年第1期132卷 103-105页

作者： Yu-Jing Huang Zong-Pei Jiang Yu-Ping Chen Jin-Quan Wu Jia-Li Huang Yan-Qiang Chen Meng-Jun Liang Department of Nephrology The First People's Hospital of ZhaoqingZhaoqingGuangdong 526000China Department of Nephrolegy The Six Affiliated HospitalSun Yat-sen UniversityGuangzhouGuangdong 510655China

To the Editor:Tuberous sclerosis complex (TSC),with the birth incidence of 1:6000,[1] is an autosomal dominant inherited,multi-system disorder characterized by cellular hyperplasia and tissue dysplasia,among which,renal angiomyolipoma (AML) is one common comorbidity.However,malignancy of renal AML is rare.Herein,we shared a case of malignancy of renal AML from TSC in a young man.

关键词： Malignancy renal angiomyolipoma tuberous sclerosis complex TSC2 mutation

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Diagnostic value of ultrasound in the spontaneous rupture of renal angiomyolipoma during pregnancy:A case report

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World Journal of Clinical Cases 2020年第17期8卷 3875-3880页

作者： Tong Zhang Shuai Xue Zheng-Min Wang Xiu-Mei Duan Dong-Xuan Wang Department of Ultrasound The First Hospital of Jilin UniversityChangchun 130000Jilin ProvinceChina Department of Thyroid Surgery The First Hospital of Jilin UniversityChangchun 130000Jilin ProvinceChina Department of Pathology The First Hospital of Jilin UniversityChangchun 130000Jilin ProvinceChina

BACKGROUND Spontaneous rupture and hemorrhage of renal angiomyolipoma(RAML)is a lifethreatening clinical emergency.When it occurs during pregnancy,it is compared to a"bomb explosion,"which makes the diagnosis and treatment more challenging.An ultrasound examination is a quick and safe examination with the benefit of no radiation exposure,which is always preferred for pregnant women.Currently,cases of spontaneous rupture and hemorrhage of RAML during pregnancy are rare,as is the diagnostic value and characteristics of ultrasound.The lack of understanding of the condition among ultrasound doctors makes it prone to misdiagnosis.In this study,we present the case of a pregnant woman who was preliminarily diagnosed with spontaneous rupture and hemorrhage of the left RAML using ultrasound and discuss the ultrasound characteristics.CASE SUMMARY A 38-year-old woman in her 19 th wk of pregnancy(G2 P1)was referred to our clinic for a sudden,persistent pain on the left side of the waist.She had not undergone any previous related abdominal examination.Ultrasound of the urinary system revealed a giant nonhomogenous lump in the left kidney area.The diagnosis was considered spontaneous rupture and hemorrhage of the left RAML in pregnancy via ultrasound.Her left-side waist pain continued to be intense.Subsequently,she underwent computed tomography,which led to the same diagnosis.Based on many factors,the patient underwent left nephrectomy after the induction of labor.The pathological result was the rupture and hemorrhage of a vascular leiomyoma lipoma.CONCLUSION Ultrasound examination plays an important role in the diagnosis of the spontaneous rupture and hemorrhage of RAML during pregnancy.

关键词： angiomyolipoma Kidney Rupture Pregnancy Ultrasonography Case report

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Clinical characteristics and outcomes of patients with hepatic angiomyolipoma:A literature review

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World Journal of Gastroenterology 2021年第19期27卷 2299-2311页

作者： Paul Calame Gaelle Tyrode Delphine Weil Verhoeven Sophie Felix Anne Julia Klompenhouwer Vincent Di Martino Eric Delabrousse Thierry Thevenot Department of Radiology Jean Minjoz University HospitalBesançon 25030France Department of Hepatology Jean Minjoz University HospitalBesançon 25030France Department of Pathology Jean Minjoz University HospitalBesançon 25000France Department of Surgery Erasmus University Medical CenterRotterdam PO Box 2040The Netherlands

First reported in 1976,hepatic angiomyolipoma(HAML)is a rare mesenchymal liver tumor occurring mostly in middle-aged women.Diagnosis of the liver mass is often incidental on abdominal imaging due to the frequent absence of specific symptoms.Nearly 10%of HAMLs are associated with tuberous sclerosis complex.HAML contains variable proportions of blood vessels,smooth muscle cells and adipose tissue,which renders radiological diagnosis hazardous.Cells express positivity for HMB-45 and actin,thus these tumors are integrated into the group of perivascular epithelioid cell tumors.Typically,a HAML appears on magnetic resonance imaging(or computed tomography scan)as a hypervascular solid tumor with fatty areas and with washout,and can easily be misdiagnosed as other liver tumors,particularly hepatocellular carcinoma.The therapeutic strategy is not clearly defined,but surgical resection is indicated for symptomatic patients,for tumors showing an aggressive pattern(i.e.,changes in size on imaging or high proliferation activity and atypical epithelioid pattern on liver biopsy),for large(>5 cm)biopsy-proven HAML,and if doubts remain on imaging or histology.Conservative management may be justified in other conditions,since most cases follow a benign clinical course.In summary,the correct diagnosis of HAML is challenging on imaging and relies mainly on pathological findings.

关键词： angiomyolipoma Liver Tuberous sclerosis complex Imaging Pathology Potentially malignant

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Clinical features of hepatic angiomyolipoma

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Hepatobiliary & Pancreatic Diseases International 2008年第3期7卷 284-287页

作者： Zhou, Yan-Ming Li, Bin Xu, Feng Wang, Bin Li, Dian-Qi Zhang, Xiao-Feng Liu, Peng Yang, Jia-Mei Second Mil Med Univ Dept Special Treatment Eastern Hepatobiliary Surg Hosp Shanghai 200438 Peoples R China Fujian Med Univ Dept Hepatobiliary Pancreatovasc Surg Xiamen Hosp 1 Xiamen 361003 Peoples R China Second Mil Med Univ Dept Pathol Eastern Hepatobiliary Surg Hosp Shanghai 200438 Peoples R China

BACKGROUND: Hepatic angiomyolipoma (HAML) is a rare hepatic mesenchymal tumor. This study was designed to explore its clinical features. METHODS: Clinical data from 26 patients who had been pathologically confirmed with HAML and had received surgical resection at our hospital were analyzed retrospectively. RESULTS: HAML was seen more frequently in females (18/26) in this series, and most of the patients presented no significant symptoms except for one who had a spontaneous rupture hemorrhage. Serum alpha-fetoprotein (AFP), carbohydrate antigen 19-9 (CA19-9) and carcinoembryonic antigen (CEA) were negative in all patients. Imaging presentations were diverse. Pre-operative diagnosis was made in only 3 patients. Pathological study showed that the tumor was composed of adipose tissue, smooth muscle and blood vessels in different proportions. One patient showed hepatic vessel invasion. HMB-45 immunohistochemical staining was positive in all tumors. All patients underwent surgical resection without significant complications. Except for one patient who died 14 months after operation because of recurrent disease, no tumor recurrence was observed in the remaining 25 patients during a 2-3 years follow-up. CONCLUSIONS: Pre-operative diagnosis of HAML is difficult. There are potential risks of spontaneous rupture and malignant transformation. Surgical resection is the treatment of choice for HAML.

关键词： angiomyolipoma hemorrhage liver neoplasms

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Giant exophytic renal angiomyolipoma masquerading as a retroperitoneal liposarcoma: A case report and review of literature

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World Journal of Clinical Oncology 2018年第7期9卷 162-166页

作者： Gopal Sharma Ayush Jain Prerit Sharma Sonal Sharma Vinita Rathi Pankaj Kumar Garg Department of Surgery University College of Medical Sciences and Guru Teg Bahadur Hospital University of Delhi Department of Pathology University College of Medical Sciences and Guru Teg Bahadur Hospital University of Delhi Department of Radiology University College of Medical Sciences and Guru Teg Bahadur Hospital University of Delhi

A 42-years-old lady, presented with a large retroperitoneal mass which was preoperatively diagnosed as a retroperitoneal liposarcoma following an image guided core biopsy. She underwent a margin-negative resection of the retroperitoneal mass(multi visceral resection-enbloc excision of the retroperitoneal mass with a left nephrectomy and a segmental descending colectomy). The final histopathological examination of the resected specimen confirmed an exophytic renal angiomyolipoma(AML) which was extending into the retroperitoneum. AML is a rare benign tumor arising most commonly from the kidney. It can sometimes present as a diagnostic challenge as it mimics a retroperitoneal liposarcoma or a fat-containing renal cell carcinomas closely. We present this case to share our experience of managing a case of giant exophytic AML which resembled retroperitoneal liposarcoma closely and resulted into an aggressive surgery.

关键词： angiomyolipoma Retroperitoneum Liposarcoma Diagnosis Biopsy

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Occult Renal Cell Carcinoma of Eosinophilic Morphology Detected within Renal angiomyolipoma Mass in a Patient with Tuberous Sclerosis Complex

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Chinese Medical Journal 2017年第12期130卷 1505-1506页

作者： Chun-NianWang Su-Ying Wang Min-Jiang Lin Hong-Wei Wang Department of Pathology Ningbo Clinical and Pathological Diagnosis Center Ningbo Zhejiang 315000 China Department of Radiology Ningbo City Medical Treatment Center Lihuili Hospital Ningbo Zhejiang 315000 China

The kidney is affected in about 80-85% of tuberous sclerosis complex （TSC） patients. Renal manifestations in TSC patients include an increased incidence of epithelial cysts and tumors, such as multiple renal angiomyolipomas （AMLs）, renal cell carcinoma （RCC）, and oncocytoma. The coexistence of RCC and renal AML within same tumor masses, namely collision tumor, is very rare, and about six cases have been reported. Here, we present a case of a young male with TSC and multiple AMLs, containing RCC with eosinophilic morphology.

关键词： angiomyolipoma Collision Tumor Renal Cell Carcinoma Tuberous Sclerosis Complex

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Juxta-caval hepatic angiomyolipoma masquerading as hepatocellular carcinoma

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Hepatobiliary & Pancreatic Diseases International 2003年第4期2卷 617-621页

作者： Alan M Hawxby Michael Torbenson Elliot Fishman Leo Lawler Luis M Arrazola Andrew S Klein the Departments of Surgery The Johns Hopkins Hospital Baltimore MD 21287 USA Pathology The Johns Hopkins Hospital Baltimore MD 21287 USA Radiology The Johns Hopkins Hospital Baltimore MD 21287 USA the Departments of Surgery The Johns Hopkins Hospital Baltimore MD 21287 USA

Hepatic angiomyolipoma is an uncommon primary benign epithelial liver neoplasm. Most patients with angiomyolipomas are middle-aged women who commonly present with epigastric fullness or pain. angiomyolipomas are tumors consisting of three tissue types: blood vessels, smooth muscle, and fat. Hepatic angiomyolipomas may be difficult to differentiate from other liver neoplasms by noninvasive imaging. We report a 58-year-old asymptomatic woman with a mass in the right lobe of the liver, found incidentally on routine abdominal sonography. Preoperative radiographic evaluation revealed a 6.5-cm hypervascular lesion abutting the inferior vena cava. Preoperative histologic study demonstrated an epithelial neoplasm suspicious for hepatocellular carcinoma. Metastatic workup was negative. At resection, the tumor was found to be an angiomyolipoma composed of lipoid, vascular and smooth muscle cells. Further staining was positive for HMB-45. Resection margins were negative. The woman had an uneventful recovery and was discharged on postoperative day seven. She currently remains well several months after her right hepatectomy.

关键词： angiomyolipoma hepatectomy radiographic evaluation multiplanar imaging

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