Angiomyolipoma of the Adrenal Gland: A Report of Two Cases and Review of the Literature

Ghimire, Obin; Li, Wenzheng; Liu, Huaping; Liu, Wenguang; Pei, Yigang; Hou, Jing

doi:10.12659/ajcr.903908

Cited by 11 publications

(9 citation statements)

References 22 publications

(29 reference statements)

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“…Angiomyolipoma is a mesenchymal benign tumor involving the smooth muscle tissue, mature adipocytes, as well as large and small blood vessels, the majority of which are present in the kidney [1]. Although angiomyolipoma that develops outside the kidney is rare, it has been reported to occur in various parts, such as liver, uterus, adrenal gland and retroperitoneum, among others [1][2][3]. However, we came across only one case report on angiomyolipoma originating from the spleen when searching in PubMed with the keywords "spleen" and "angiomyolipoma" [4].…”

Section: Discussionmentioning

confidence: 99%

Surgical resection of extremely rare primary giant splenic angiomyolipoma: a case report

et al. 2021

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Background Angiomyolipoma is a benign mesenchymal tumor that develops commonly in the kidney and rarely in other organs. The involvement of the spleen in angiomyolipoma is extremely rare, and only one such case has been reported in the English literature. Case presentation A 27-year-old man presented with adenoid hyperplasia and bilateral palatal tonsillar hyperplasia. During the treatment for adenoid hyperplasia, a 15-cm tumor was detected in the spleen using abdominal ultrasonography and enhanced computed tomography. Partial resection of the spleen was successfully performed. A giant tumor of approximately 13 cm with a smooth surface was observed in the upper left quadrant of the abdomen. The tumor was confirmed to be continuous with the upper spleen, and there was no invasion of the other organs. The postoperative course was good, and the patient was discharged on the 7th postoperative day. The excised specimen was a smooth, extremely soft tumor measuring 123 × 120 × 82 mm. The cleaved surface of the tumor was reddish brown, and a distressing yellow color was observed. Pathological examination revealed a proliferation of mature adipocytes and an increase in the number of blood vessels of various sizes. Furthermore, spindle-shaped cell proliferation foci were visible between the adipocytes and the surrounding blood vessels. Profuse leakage of erythrocytes from the blood vessels, hemosiderin deposition, and small round cell infiltration were also noted. Immunostaining disclosed that the spindle-shaped cells were weakly positive for smooth muscle antibody and were identified as smooth muscle cells. The adipocytes and spindle cells were negative for HMB 45, Melan A, MDM, and CDK4. However, some parts of the cells were positive for estrogen and progesterone receptors. Besides, vascular endothelial cells were positive for CD31 and CD34 and negative for CD8. Based on these findings, the patient was diagnosed to have primary angiomyolipoma of the spleen. Conclusions We have reported the surgical treatment for an extremely rare case of giant splenic angiomyolipoma in a young man. Globally, this is the second report on this condition. We believe that partial splenic resection is a feasible option for the management of giant tumors.

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Section: Discussionmentioning

confidence: 99%

Surgical resection of extremely rare primary giant splenic angiomyolipoma: a case report

et al. 2021

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“…e average age at diagnosis for AMLs in general is around 40 years old, and the most common initial presentation is focal pain [6,10,11]. Case reports of adrenal involvement from AMLs presenting with retroperitoneal hemorrhage after spontaneous rupture have also been described [12]. e diagnosis of EAMLs can be challenging.…”

Section: Discussionmentioning

confidence: 99%

A Primary Adrenal Epithelioid Angiomyolipoma (PEComa) in a Patient with Tuberous Sclerosis Complex: Report of a Case and Review of the Literature

Luna

Mosquera

Comba

et al. 2020

Case Reports in Medicine

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Epithelioid angiomyolipomas (EAMLs) are mesenchymal tumors that are part of the family of the perivascular epithelioid cell neoplasms (PEComas). These tumors portray a potential aggressive behavior with metastatic lesions found in around 30% of reported cases. EAMLs might present sporadically or in association with the tuberous sclerosis complex (TSC). They typically involve the kidneys, liver, and lungs. It is extremely rare for these tumors to arise from other organs. The present report describes an unusual case of an adult patient with a history of TSC who developed EAML of the adrenal gland. Moreover, he presented with metastatic disease to the liver, a feature rarely described. The diagnosis of EAMLs can be challenging as they are hard to distinguish from other adrenal or renal tumors without a thorough histopathologic and immunohistochemical evaluation. Due to the potential aggressive behavior of these malignancies, timely diagnosis is extremely important and has significant therapeutic and prognostic implications.

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“…Isolated neoplasms are usually asymptomatic. 20 to 50% have been reported to be associated with tuberous sclerosis in various literatures [1][2][3] . Though commonly benign, metastasis have been reported.…”

Section: Discussionmentioning

confidence: 99%

HMB-45 Negative Adrenal Angiomyolipoma With A Synchronous Adrenal Adenoma – An Unusual Association

Nair

Raju

Augustine

et al. 2020

Ann of Pathol and Lab Med

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Background: Angiomyolipoma (AML) is an uncommon mesenchymal tumour usually found in the kidney. The most common extrarenal site is usually the liver. We report a case of adrenal AML with a predominant vascular component co-existing with an adrenal adenoma. Case report: A fifty-year-old male presented with two months history of dysuria. Computed tomography showed a heterogeneous lesion involving the right adrenal gland abutting the right kidney and adjacent liver capsule. Urine noradrenaline levels were elevated. Adrenalectomy specimen showed an encapsulated mass measuring 8 x 5 x3.5 cm with a lobulated, grey to tan brown cut surface with areas of haemorrhage and tiny cystic spaces. Another yellowish nodule measuring 1.3 x 1.2 x 1 cm was also noted at one pole. Microscopy showed a neoplasm composed predominantly thick-walled vascular channels admixed with foci of smooth muscle bundles and adipocytes. Yellowish nodule showed features of adrenal adenoma. HMB-45 was negative and CD34 positivity highlighted the thick and thin walled vessels. Conclusion: We present the very first case of HMB-45 negative adrenal AML with a predominant vascular component and a synchronous adrenal adenoma.

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Angiomyolipoma of the Adrenal Gland: A Report of Two Cases and Review of the Literature

Cited by 11 publications

References 22 publications

Surgical resection of extremely rare primary giant splenic angiomyolipoma: a case report

Surgical resection of extremely rare primary giant splenic angiomyolipoma: a case report

A Primary Adrenal Epithelioid Angiomyolipoma (PEComa) in a Patient with Tuberous Sclerosis Complex: Report of a Case and Review of the Literature

HMB-45 Negative Adrenal Angiomyolipoma With A Synchronous Adrenal Adenoma – An Unusual Association

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