[Angiomyolipoma of the kidney: a case report and a statistical study of 429 cases in Japan].
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A 40-year-old housewife was found to have a renal mass on her left side through an ultrasonogram at the hospital during a medical check up. The patient had a dull pain in her left flank intermittently for three years. She had no personal or family history of stigmas of tuberous sclerosis. The mass showed a high echogenicity on the ultrasonogram and a low density (-84HU) on the CT scan. Preoperative diagnosis was reported as renal angiomyolipoma and an enucleation of the tumor was performed with CUSA. A statistical study was done on data taken from 429 cases of renal angiomyolipoma in the Japanese literature, including our case. The male to female ratio was 1 to 2.9. Thirty three percent of the cases were associated with tuberous sclerosis. The ratio of bilateral cases to unilateral ones was 1:4. The main clinical signs were abdominal pain, abdominal mass and gross hematuria. Nephrectomy was done in 71.0% of the cases, partial nephrectomy and enucleation in 13.7% and embolization in 2.2%. Through the composition of the CT scan and the ultrasonogram, preoperative diagnosis has become possible in many cases, so recently the ratio of nephrectomy has been decreasing. We think partial nephrectomy and enucleation compose the most effective therapy for renal angiomyolipoma.Keywords:
Angiomyolipoma
Ultrasonogram
Abdominal mass
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Retrospective analysis of clinical and histomorphological features and follow up of eleven cases of renal angiomyolipoma (AML) encountered during the past twelve years at our centre was performed. These included seven female and four male patients, nine of them were symptomatic and two were incidentally detected on ultrasonography for other reasons. Eight patients had solitary and three had bilateral lesions on radiological investigations. None of the three patients with bilateral AML had associated tuberous sclerosis, however, a patient with tuberous sclerosis, showing solitary lesion on radiological investigation, was detected to have multiple lesions on thorough pathological examination of the nephrectomy specimen. The surgical treatment comprised of nephrectomy in six cases, enucleation in four cases and trueut biopsy in one case. Enucleation of the largest lesion was done in all three patients of bilateral AML. The mean follow up was of three years. Two patients with bilateral AML had radiological progression of size of other lesions, whereas one died due to excessive bleeding in the postoperative period. None of the patients had any evidence of recurrence or metastasis. Bilateral AML is not always associated with tuberous sclerosis, however, in patients with tuberous sclerous thorough sampling of nephrectomy specimen is recommended.
Angiomyolipoma
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The authors present their own experience as well as data from the literature concerning the diagnosis and treatment of renal angiomyolipomas. In a group of 320 patients hospitalised at the Urological Clinic in 1990-1994 these tumours were diagnosed before surgery and confirmed by histological examination in 7 patients (2.2%). The group comprised three men and four women, mean age 51.7 years. Only one female patient had classical manifestations of tuberous sclerosis, the remaining patients did not have manifestations of tuberous sclerosis. Two patients were admitted with symptoms of haemorrhage into the retroperitoneum, three suffered from pain on pressure in the side and in two patients the tumour was revealed incidentally during US examination. Four patients were subjected to nephrectomy, in two enucleation of the tumour was performed and in one female patient with classical manifestations of tuberous sclerosis with a bilateral tumour conservative treatment was applied.
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Wünderlich syndrome (WS) is a spontaneous retroperitoneal hemorrhage confined to the subcapsular or perinephric space without a history of trauma. Since it is a rare condition with a significant mortality rate if not treated timely, it is essential to identify its risk factors and early clinical manifestations for a favorable outcome. Various conditions are associated, but the most common causes are benign and malignant renal neoplasms. We present a 26-year-old female with a history of tonic-clonic seizures who presented to the ED with intense abdominal pain located on the right flank with a palpable mass. Management included IV fluids and blood transfusion. She underwent a right total nephrectomy. She was later diagnosed with tuberous sclerosis. A 44-year-old female with a three-year history of right costovertebral pain and recurrent urinary tract infections that presented to the ED with acute right flank pain was diagnosed with WS secondary to an angiomyolipoma and underwent right total nephrectomy. WS is a very rare pathology that represents a diagnostic challenge for the physician. The treatment will depend on the hemodynamic condition of the patient. Active follow-up should be reserved for those who have small tumors, are asymptomatic, and have hemodynamic stability. Surgical or radiology intervention is reserved for those who are hemodynamically unstable or who have a suspicion of renal cell carcinoma.
Angiomyolipoma
Flank pain
Retroperitoneal space
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Tuberous sclerosis complex (TSC) is an autosomal-dominant neurocutaneous disorder involving the skin, brain, kidney, heart and other organs. Renal manifestations are mainly angiomyolipomas, renal cysts and cancer. We report three female patients, mean aged of 31 years (range: 22-37), with a family history of TSC in one case. Cutaneous and nervous symptoms were found in all patients. Clinical complaints were mainly abdominal pain (3 cases), hematuria (2 cases), right flank palpable mass (1 case). Angiomyolipomas (AML) were diagnosed by ultrasonography in all patients, associated to renal cysts in one case. A patient underwent total right nephrectomy without any event during the follow-up. In a second patient, a selective arterial embolization of AML was indicated. Total left nephrectomy was performed due to the persistence of hematuria and the increased flank mass. Pathological examination of the kidney revealed a clear cell carcinoma. The third patient with small AML, associated with renal cysts, required careful monitoring. Renal manifestations in TSC are frequent and serious, they are the second leading cause of death after nervous lesions. Clinical and morphological aspects are variable and different therapeutic indications must be discussed.
Abdominal mass
Angiomyolipoma
Flank pain
Abdominal ultrasonography
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A 40-year-old housewife was found to have a renal mass on her left side through an ultrasonogram at the hospital during a medical check up. The patient had a dull pain in her left flank intermittently for three years. She had no personal or family history of stigmas of tuberous sclerosis. The mass showed a high echogenicity on the ultrasonogram and a low density (-84HU) on the CT scan. Preoperative diagnosis was reported as renal angiomyolipoma and an enucleation of the tumor was performed with CUSA. A statistical study was done on data taken from 429 cases of renal angiomyolipoma in the Japanese literature, including our case. The male to female ratio was 1 to 2.9. Thirty three percent of the cases were associated with tuberous sclerosis. The ratio of bilateral cases to unilateral ones was 1:4. The main clinical signs were abdominal pain, abdominal mass and gross hematuria. Nephrectomy was done in 71.0% of the cases, partial nephrectomy and enucleation in 13.7% and embolization in 2.2%. Through the composition of the CT scan and the ultrasonogram, preoperative diagnosis has become possible in many cases, so recently the ratio of nephrectomy has been decreasing. We think partial nephrectomy and enucleation compose the most effective therapy for renal angiomyolipoma.
Angiomyolipoma
Ultrasonogram
Abdominal mass
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A 49-year-old woman with tuberous sclerosis visited our hospital complaining of left abdominal pain. She had been diagnosed with bilateral renal angiomyolipoma (AML) when she was 32 years old and had been on maintenance hemodialysis since the age of 44. Magnetic resonance imaging showed bilateral giant renal AML and hemorrhage in the AML of the left kidney. Transthoracolumbar left nephrectomy was performed in March 2008. The resected specimen weighed 3.2 kg. The histological diagnosis was consistent with AML. In addition, the Japanese literature on giant renal AML was reviewed and discussed.
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Abdominal mass
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A 37-year-old man with no symptoms or family history of tuberous sclerosis complex presented to our hospital with abdominal pain in 2013. Abdominal computed tomography (CT) revealed a rupture in the right renal angiomyolipoma (AML) having a maximum diameter of 7 cm. He had undergone a transarterial embolization. Follow-up CT showed an increase in the size of the right tumor to 11 cm, and therefore, right nephrectomy was performed in 2016. The diagnosis of epithelioid AML (EAML) was confirmed. In 2019, he was diagnosed with a solitary tumor near right-sided transverse colon, which was resected and showed recurrence of EAML. He was disease-free 6 months after surgery. EAML has malignant potential, with 30-50% of reported EAML cases resulting in local recurrence or distant metastasis. Previous recurrence or metastasis may occur 0.25-12 years postoperatively. Furthermore, multiple and unresectable recurrences or metastases, arising early in the postoperative period may lead to a poor outcome. Therefore, close and long-term follow-up is required.
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Transverse colon
Abdominal cavity
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Angiomyolipoma
Renal vein
Renal capsule
Benign tumor
Nephrology
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Angiomyolipoma
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A case of bilateral renal angiomyolipoma is reported. A 21-year-old female visited our hospital with the complaint of severe left flank pain and macroscopic hematuria Bilateral renal mass was suspected from the intravenous pyelography. CT scan revealed bilateral renal angiomyolipoma accompanied by retroperitoneal bleeding. There were no symptoms or signs of tuberous sclerosis. Left partial nephrectomy and right tumor enucleation were performed uneventfully with no recurrence during 4.5 years of follow-up. Several case reports of renal angiomyolipoma have been documented; we sum up these cases with special consideration as to treatment. In the majority of the cases, nephrectomy has been performed after retroperitoneal bleeding. We emphasize that in some cases it is favorable to carry out renal-tissue-conserving operation before the occurrence of the bleeding, because the angiomyolipoma tends to rupture easily.
Angiomyolipoma
Intravenous pyelography
Flank pain
Benign tumor
Hamartoma
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