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1. Bissler JJ, McCormack FX, Young LR, Elwing JM, Chuck G, Leonard JM, Schmithorst VJ, Laor T, Brody AS, Bean J, Salisbury S, Franz DN: Sirolimus for angiomyolipoma in tuberous sclerosis complex or lymphangioleiomyomatosis. N Engl J Med; 2008 Jan 10;358(2):140-51
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  • [Title] Sirolimus for angiomyolipoma in tuberous sclerosis complex or lymphangioleiomyomatosis.
  • The drug sirolimus suppresses mTOR signaling.
  • METHODS: We conducted a 24-month, nonrandomized, open-label trial to determine whether sirolimus reduces the angiomyolipoma volume in patients with the tuberous sclerosis complex or sporadic lymphangioleiomyomatosis.
  • Serial magnetic resonance imaging of angiomyolipomas and brain lesions, computed tomography of lung cysts, and pulmonary-function tests were performed.
  • The mean (+/-SD) angiomyolipoma volume at 12 months was 53.2+/-26.6% of the baseline value (P<0.001) and at 24 months was 85.9+/-28.5% of the baseline value (P=0.005).
  • At 24 months, five patients had a persistent reduction in the angiomyolipoma volume of 30% or more.
  • During the period of sirolimus therapy, among patients with lymphangioleiomyomatosis, the mean forced expiratory volume in 1 second (FEV1) increased by 118+/-330 ml (P=0.06), the forced vital capacity (FVC) increased by 390+/-570 ml (P<0.001), and the residual volume decreased by 439+/-493 ml (P=0.02), as compared with baseline values.
  • CONCLUSIONS: Angiomyolipomas regressed somewhat during sirolimus therapy but tended to increase in volume after the therapy was stopped.
  • Some patients with lymphangioleiomyomatosis had improvement in spirometric measurements and gas trapping that persisted after treatment.
  • Suppression of mTOR signaling might constitute an ameliorative treatment in patients with the tuberous sclerosis complex or sporadic lymphangioleiomyomatosis. (ClinicalTrials.gov number, NCT00457808. )
  • [MeSH-major] Angiomyolipoma / drug therapy. Immunosuppressive Agents / therapeutic use. Lung Neoplasms / complications. Lymphangioleiomyomatosis / complications. Sirolimus / therapeutic use. Tuberous Sclerosis / complications
  • [MeSH-minor] Adult. Brain / pathology. Female. Humans. Kidney Diseases / pathology. Liver Diseases / pathology. Lung / diagnostic imaging. Lung / physiopathology. Magnetic Resonance Imaging. Male. Middle Aged. Protein Kinase Inhibitors / therapeutic use. Protein Kinases / metabolism. Radiography. Respiratory Function Tests. TOR Serine-Threonine Kinases


2. Yokoo H, Isoda K, Nakazato Y, Nakayama Y, Suzuki Y, Nakamura T, Shinkai H, Aiba M: Retroperitoneal epithelioid angiomyolipoma leading to fatal outcome. Pathol Int; 2000 Aug;50(8):649-54
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  • [Title] Retroperitoneal epithelioid angiomyolipoma leading to fatal outcome.
  • Epithelioid angiomyolipoma (AML) is a newly established variant of AML, characterized by monomorphous epithelioid cells that show HMB-45 immunopositivity, and it often displays aggressive behavior.
  • To date, they have mostly appeared in the kidneys; however, the present autopsy case of a 43-year-old female without the stigmata of tuberous sclerosis complex had a huge retroperitoneal mass, accompanied by involvement of the regional lymph nodes.
  • In spite of curative surgery and repeated radio- and chemotherapy, the tumor continued to grow and brought about the patient's death 4 years after the initial symptoms.
  • [MeSH-major] Angiomyolipoma / pathology. Epithelioid Cells / pathology. Retroperitoneal Neoplasms / pathology
  • [MeSH-minor] Adult. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Biomarkers, Tumor / analysis. Carboplatin / therapeutic use. Cyclophosphamide / therapeutic use. Dacarbazine / therapeutic use. Fatal Outcome. Female. Humans. Immunohistochemistry. Neoplasm Proteins / analysis. Neoplasm Recurrence, Local / chemistry. Neoplasm Recurrence, Local / pathology. Neoplasm Recurrence, Local / surgery. Neoplasm Recurrence, Local / therapy. Radiotherapy, Adjuvant. Tomography, X-Ray Computed

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  • (PMID = 10972864.001).
  • [ISSN] 1320-5463
  • [Journal-full-title] Pathology international
  • [ISO-abbreviation] Pathol. Int.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] AUSTRALIA
  • [Chemical-registry-number] 0 / Biomarkers, Tumor; 0 / Neoplasm Proteins; 7GR28W0FJI / Dacarbazine; 8N3DW7272P / Cyclophosphamide; BG3F62OND5 / Carboplatin
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3. Micozkadioglu H, Koc Z, Ozelsancak R, Yildiz I: Rapamycin therapy for renal, brain, and skin lesions in a tuberous sclerosis patient. Ren Fail; 2010;32(10):1233-6
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  • [Title] Rapamycin therapy for renal, brain, and skin lesions in a tuberous sclerosis patient.
  • Tuberous sclerosis complex (TSC) is an inherited multisystem disorder; it may involve kidney, brain, skin, lungs, and liver.
  • Radiologic examination revealed additional brain and renal lesions consisting of tumors, cysts, and angiomyolipomas.
  • Treatment with rapamycin disclosed improvement in skin lesions.
  • During the ninth month of treatment, magnetic resonance imaging was repeated for renal and brain lesions.
  • Imaging results showed reduction in tumor and angiomyolipoma volumes.
  • Oral rapamycin therapy can improve renal, brain, and skin lesions in TSC disease.
  • Therefore, it may be an alternative therapy for TSC patients.
  • [MeSH-major] Brain Neoplasms / drug therapy. Immunosuppressive Agents / therapeutic use. Kidney Neoplasms / drug therapy. Sirolimus / therapeutic use. Skin Neoplasms / drug therapy. Tuberous Sclerosis / complications
  • [MeSH-minor] Adult. Angiofibroma / drug therapy. Angiofibroma / etiology. Facial Neoplasms / drug therapy. Female. Humans. Magnetic Resonance Imaging


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4. Okoń K: Pathology of renal tumors in adults. Molecular biology, histopathological diagnosis and prognosis. Pol J Pathol; 2008;59(3):129-76
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  • [Title] Pathology of renal tumors in adults. Molecular biology, histopathological diagnosis and prognosis.
  • Malignant renal tumors constitute 3% of human cancers, although their frequency differs greatly in various areas.
  • Since the fifties, the incidence of renal cancers has been increasing, but at the some time the prognosis has been improving.
  • In particular, in the last years, several new treatment modalities have been introduced, relying on the understanding of renal cancer biology.
  • The identified etiological factors include smoking, increased body mass, dietary factors and chronic renal disease.
  • There are several renal tumor types differing in morphology, molecular genetics and biology.
  • At least two types of papillary carcinomas exist, which have different morphology and prognosis.
  • Differential diagnosis of these tumors is particularly difficult and may require extensive immunohistochemical and molecular studies.
  • Some renal tumors have been described or recognized only relatively recently; these newer entities include multilocular cystic clear cell carcinoma, spindle cell papillary mucinous carcinoma, tubulocystic carcinoma, renal epithelial and stromal tumor, epithelioid and oncocytic angiomyolipoma.
  • The improved prognosis in renal cancer depends on earlier detection, but also on refinement of therapeutic methods.
  • Renal carcinoma is notorious for its low sensitivity to chemotherapy and radiotherapy.
  • For several years, immunological treatment with IL-2 and INF-alpha was the only adjuvant therapy method.
  • However, recently several new drugs have been introduced; they act on tyrosine-kinase receptors, VEGF, c-Met or mTOR pathway.
  • With this progress, perfect understanding of renal tumor biology and exact histological diagnosis have become of prime practical importance.
  • [MeSH-major] Kidney Neoplasms


5. Marazuela M, Domínguez-Gadea L, Larrañaga E, Rodríguez-Ramos R, López-Gallardo G, Rodríguez-Eyre JL, Gómez-Pan A: [The use of suprarenal scintigraphy in the differential diagnosis of suprarenal incidentaloma]. Rev Clin Esp; 2005 Jul;205(7):316-21
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  • [Title] [The use of suprarenal scintigraphy in the differential diagnosis of suprarenal incidentaloma].
  • OBJECTIVES: To assess the usefulness of adrenal scintigraphy for clinical evaluation of adrenal incidentalomas, and its relation with pathological diagnosis and follow-up.
  • The lesions were discovered with abdominal computerized tomography in the study of a primary tumor (22%) or in the evaluation of benign pathology (78%).
  • RESULTS: Of 46 adrenal lesions, seven didn't show uptake (three metastases, one cyst, one adrenal carcinoma, one pheochromocytoma, and one angiomyolipoma), 34 showed excessive uptake (29 nonfunctional adrenal nodules and 5 hyperfunctional adrenal nodules), and five had normal uptake (nonfunctional adrenal nodules).
  • Adrenal scintigraphy was compatible in all cases with cytological study or the response to chemotherapy.
  • CONCLUSIONS: Detection of a lesion with no uptake in adrenal scintigraphy forces to carry out complementary explorations in order to rule out malignant pathology.

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  • (PMID = 16029757.001).
  • [ISSN] 0014-2565
  • [Journal-full-title] Revista clínica española
  • [ISO-abbreviation] Rev Clin Esp
  • [Language] spa
  • [Publication-type] English Abstract; Journal Article
  • [Publication-country] Spain
  • [Chemical-registry-number] WI4X0X7BPJ / Hydrocortisone
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6. Bissler JJ, Racadio J, Donnelly LF, Johnson ND: Reduction of postembolization syndrome after ablation of renal angiomyolipoma. Am J Kidney Dis; 2002 May;39(5):966-71
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  • [Title] Reduction of postembolization syndrome after ablation of renal angiomyolipoma.
  • Approximately 75% of patients with tuberous sclerosis complex develop renal angiomyolipomas.
  • These hamartomatous lesions distort and damage renal parenchyma and can lead to hemorrhage.
  • To reduce the risk of hemorrhage, transarterial embolization is used to necrose the angiomyolipoma while sparing normal renal tissue.
  • Although an effective renal-sparing procedure, embolization most often is associated with an inflammatory response that causes significant fever and pain that can last for several days despite the use of acetaminophen.
  • Reported cases show that 49 of 55 patients who underwent embolization developed this syndrome.
  • The use of such nonsteroidal anti-inflammatory drugs as aspirin is contraindicated because of their adverse effects on platelet function.
  • Nine patients underwent this pharmacological intervention, and one patient abstained.
  • Only two patients treated with steroids developed fever, which was assuaged with acetaminophen, and no patient reported pain.
  • [MeSH-major] Angiomyolipoma / therapy. Catheter Ablation / adverse effects. Embolization, Therapeutic / adverse effects. Kidney Neoplasms / therapy
  • [MeSH-minor] Acetaminophen / administration & dosage. Acetaminophen / therapeutic use. Adult. Analgesics, Non-Narcotic / administration & dosage. Analgesics, Non-Narcotic / therapeutic use. Angiography / methods. Drug Administration Schedule. Female. Humans. Inflammation / prevention & control. Injections, Intravenous. Kidney Diseases / therapy. Male. Syndrome. Tuberous Sclerosis / therapy

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  • [Copyright] Copyright 2002 by the National Kidney Foundation, Inc.
  • (PMID = 11979340.001).
  • [ISSN] 1523-6838
  • [Journal-full-title] American journal of kidney diseases : the official journal of the National Kidney Foundation
  • [ISO-abbreviation] Am. J. Kidney Dis.
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't; Review
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Analgesics, Non-Narcotic; 362O9ITL9D / Acetaminophen
  • [Number-of-references] 40
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7. Miniati D, Gay AN, Parks KV, Naik-Mathuria BJ, Hicks J, Nuchtern JG, Cass DL, Olutoye OO: Imaging accuracy and incidence of Wilms' and non-Wilms' renal tumors in children. J Pediatr Surg; 2008 Jul;43(7):1301-7
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  • [Title] Imaging accuracy and incidence of Wilms' and non-Wilms' renal tumors in children.
  • PURPOSE: The purpose of this study is to determine the actual incidence, age distribution, and preoperative imaging accuracy of non-Wilms' tumors (nWT) in children with renal masses.
  • METHODS: Pathologic reports from all tumor nephrectomies or open renal biopsies performed at a single institution from September 1999 to June 2005 were analyzed.
  • The nWT group included congenital mesoblastic nephroma (5), clear cell sarcoma (4), neuroblastoma (4), renal cell carcinoma (4), lymphoma (2), angiomyolipoma (2), teratoma (1), hemangioma (1), and renal epithelial tumor (1).
  • Sensitivity, specificity, positive predictive value, and negative predictive value for computed tomography (CT) determining a diagnosis of WT were 0.92, 0.55, 0.84, and 0.73, respectively.
  • The CT reports explicitly stated a potential diagnosis in 89% of cases, with a diagnostic accuracy of 82%.
  • CONCLUSIONS: Non-Wilms' tumors may represent a significant proportion of renal tumors in children, especially in children aged less than 6 months or greater than 12 years.
  • These data have significant implications for parental counseling, surgical plan, and the choice of neoadjuvant chemotherapy and argue in favor of obtaining a tissue diagnosis before instituting therapy.
  • [MeSH-major] Kidney / pathology. Kidney Neoplasms / diagnosis. Wilms Tumor / diagnosis

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  • (PMID = 18639686.001).
  • [ISSN] 1531-5037
  • [Journal-full-title] Journal of pediatric surgery
  • [ISO-abbreviation] J. Pediatr. Surg.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] United States
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8. Anisya-Vasanth AV, Satishchandra P, Nagaraja D, Swamy HS, Jayakumar PN: Spectrum of epilepsy in tuberous sclerosis. Neurol India; 2004 Jun;52(2):210-2
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  • Tuberous sclerosis (TS) is an autosomal dominant disease that affects the brain, skin, eye, heart and kidney.
  • There are relatively few Indian studies on this disorder.
  • Patients often had more than one seizure type.
  • One patient each had retinal phakoma and renal angiomyolipoma.
  • Most patients were on combinations of anti-convulsants and response to therapy was usually not very satisfactory.
  • [MeSH-minor] Adolescent. Anticonvulsants / therapeutic use. Child. Child, Preschool. Drug Therapy, Combination. Female. Humans. Infant. Male. Mental Disorders / etiology. Skin Diseases / etiology. Vigabatrin / therapeutic use


9. Wen MC, Jan YJ, Li MC, Wang J, Lin A: Monotypic epithelioid angiomyolipoma of the liver with TFE3 expression. Pathology; 2010 Apr;42(3):300-2
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Monotypic epithelioid angiomyolipoma of the liver with TFE3 expression.
  • [MeSH-major] Angiomyolipoma / metabolism. Angiomyolipoma / pathology. Basic Helix-Loop-Helix Leucine Zipper Transcription Factors / biosynthesis. Liver Neoplasms / metabolism. Liver Neoplasms / pathology
  • [MeSH-minor] Adult. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Biomarkers, Tumor / analysis. Cyclophosphamide / therapeutic use. Doxorubicin / therapeutic use. Humans. Immunohistochemistry. Lymphoma, Large B-Cell, Diffuse / drug therapy. Male. Neoplasms, Second Primary / metabolism. Neoplasms, Second Primary / pathology. Prednisone / therapeutic use. Vincristine / therapeutic use

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  • (PMID = 20350230.001).
  • [ISSN] 1465-3931
  • [Journal-full-title] Pathology
  • [ISO-abbreviation] Pathology
  • [Language] eng
  • [Publication-type] Case Reports; Letter
  • [Publication-country] England
  • [Chemical-registry-number] 0 / Basic Helix-Loop-Helix Leucine Zipper Transcription Factors; 0 / Biomarkers, Tumor; 0 / TFE3 protein, human; 5J49Q6B70F / Vincristine; 80168379AG / Doxorubicin; 8N3DW7272P / Cyclophosphamide; VB0R961HZT / Prednisone; CHOP protocol
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10. Zhang J, Kang SK, Wang L, Touijer A, Hricak H: Distribution of renal tumor growth rates determined by using serial volumetric CT measurements. Radiology; 2009 Jan;250(1):137-44
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  • [Title] Distribution of renal tumor growth rates determined by using serial volumetric CT measurements.
  • PURPOSE: To retrospectively determine the distribution of growth rates across different sizes and subtypes of renal cortical tumors by assessing tumor volume and maximum tumor diameter at serial volumetric computed tomographic (CT) examinations.
  • Fifty-three of 2304 patients (34 men, 19 women; mean age, 67 years +/- 10 [standard deviation; range, 39-88 years) who underwent nephrectomy from 1989 to 2006 did not receive preoperative chemotherapy or radiation therapy and underwent at least two preoperative contrast material-enhanced CT examinations (at least 3 months apart) with identical section thickness that was no more than one-fifth of longitudinal tumor diameter.
  • Reciprocal of doubling time (DT) (RDT) was calculated.
  • RESULTS: Thirty-two clear cell carcinomas, 10 papillary carcinomas, six chromophobe carcinomas, four oncocytomas, and one angiomyolipoma were analyzed.
  • DT ranged from -78476.54 to 18057.43 days (mean, -1230.73 days; median, 590.51 days).
  • Small renal tumors (<or=3.5 cm) were similar to larger tumors in subtype and growth rate.
  • Age at diagnosis correlated negatively with renal tumor growth rate (P = .03).
  • CONCLUSION: Growth rates in renal tumors of different sizes, subtypes, and grades represent a wide range and overlap substantially.
  • Small renal tumors appear to be similar to larger ones in nature.
  • [MeSH-major] Cone-Beam Computed Tomography / methods. Kidney Cortex / radiography. Kidney Neoplasms / radiography
  • [MeSH-minor] Adenoma, Oxyphilic / pathology. Adenoma, Oxyphilic / radiography. Adenoma, Oxyphilic / surgery. Adult. Aged. Aged, 80 and over. Angiomyolipoma / pathology. Angiomyolipoma / radiography. Angiomyolipoma / surgery. Carcinoma, Papillary / pathology. Carcinoma, Papillary / radiography. Carcinoma, Papillary / surgery. Carcinoma, Renal Cell / pathology. Carcinoma, Renal Cell / radiography. Carcinoma, Renal Cell / surgery. Disease Progression. Female. Humans. Male. Mathematical Computing. Middle Aged. Neoplasm Staging. Prognosis. Retrospective Studies. Tumor Burden

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  • [Copyright] (c) RSNA, 2009.
  • [CommentIn] Radiology. 2013 Dec;269(3):949-50 [24261507.001]
  • [CommentIn] Radiology. 2009 Jul;252(1):314; author reply 314-5 [19561267.001]
  • [ErratumIn] Radiology. 2013 Dec;269(3):950
  • [ErratumIn] Radiology. 2009 Jul;252(1):318
  • (PMID = 19092093.001).
  • [ISSN] 1527-1315
  • [Journal-full-title] Radiology
  • [ISO-abbreviation] Radiology
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] United States
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11. Rigby H, Yu W, Schmidt MH, Fernandez CV: Lack of response of a metastatic renal perivascular epithelial cell tumor (PEComa) to successive courses of DTIC based-therapy and imatinib mesylate. Pediatr Blood Cancer; 2005 Aug;45(2):202-6
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  • [Title] Lack of response of a metastatic renal perivascular epithelial cell tumor (PEComa) to successive courses of DTIC based-therapy and imatinib mesylate.
  • Subsequent investigations led to the diagnosis of metastatic perivascular epithelioid cell tumor (PEComa) arising from the left kidney.
  • Effective treatment for this rare tumor is not yet known.
  • The tumor did not respond to an initial treatment of two cycles of a dacarbazine (DTIC) based regimen.
  • She was placed on a trial of imatinib mesylate based on tumor expression of c-KIT, a tyrosine kinase targeted by this drug.
  • Lack of response and adverse effects of the drug required discontinuation of therapy.
  • [MeSH-major] Angiomyolipoma / drug therapy. Antineoplastic Agents / therapeutic use. Epithelioid Cells / pathology. Kidney Neoplasms / drug therapy. Piperazines / therapeutic use. Pyrimidines / therapeutic use
  • [MeSH-minor] Benzamides. Child. Dacarbazine / therapeutic use. Fatal Outcome. Female. Humans. Imatinib Mesylate. Neoplasm Metastasis. Treatment Failure

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  • (PMID = 15704192.001).
  • [ISSN] 1545-5009
  • [Journal-full-title] Pediatric blood & cancer
  • [ISO-abbreviation] Pediatr Blood Cancer
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / Benzamides; 0 / Piperazines; 0 / Pyrimidines; 7GR28W0FJI / Dacarbazine; 8A1O1M485B / Imatinib Mesylate
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12. Benden C, Rea F, Behr J, Corris PA, Reynaud-Gaubert M, Stern M, Speich R, Boehler A: Lung transplantation for lymphangioleiomyomatosis: the European experience. J Heart Lung Transplant; 2009 Jan;28(1):1-7
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  • BACKGROUND: Lung transplantation has been accepted widely as therapy for end-stage pulmonary lymphangioleiomyomatosis (LAM); however, single-center and national experience is limited due to the rarity of LAM.
  • LAM-related complications included renal angiomyolipoma and pneumothorax in the native lung.
  • [MeSH-minor] Adult. Cause of Death. Drug Therapy, Combination. Europe. Exercise Test. Female. Forced Expiratory Volume. Humans. Immunosuppressive Agents / therapeutic use. Intraoperative Complications / classification. Intraoperative Complications / epidemiology. Lung Diseases / epidemiology. Respiratory Function Tests. Surveys and Questionnaires. Survival Analysis. Tissue Adhesions / epidemiology. Treatment Outcome

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  • (PMID = 19134523.001).
  • [ISSN] 1557-3117
  • [Journal-full-title] The Journal of heart and lung transplantation : the official publication of the International Society for Heart Transplantation
  • [ISO-abbreviation] J. Heart Lung Transplant.
  • [Language] eng
  • [Publication-type] Journal Article; Multicenter Study
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Immunosuppressive Agents
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13. Bissler JJ, Kingswood JC: Renal angiomyolipomata. Kidney Int; 2004 Sep;66(3):924-34
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  • [Title] Renal angiomyolipomata.
  • Renal angiomyolipomata can exist as an imaging curiosity or represent a life-threatening condition.
  • There are likely over 10 million people world-wide who have a renal angiomyolipomata and approximately one tenth of these people also have tuberous sclerosis complex.
  • The optimum treatment of angiomyolipomata is focused on sparing renal tissue and has included no intervention, both total and partial nephrectomy, and embolization.
  • As basic science investigation into the biology of angiomyolipomata progresses, there is even hope for successful drug therapy.
  • Because these renal lesions can be associated with other organ system dysfunction, a number of medical specialists become involved in the care of affected patients.
  • The purpose of this article is to update the nephrologist on the molecular understanding of renal angiomyolipomata and for the possibilities of pharmacologic therapy in the future.
  • [MeSH-major] Angiomyolipoma / pathology. Kidney / pathology. Kidney Neoplasms / pathology

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  • (PMID = 15327383.001).
  • [ISSN] 0085-2538
  • [Journal-full-title] Kidney international
  • [ISO-abbreviation] Kidney Int.
  • [Language] eng
  • [Grant] United States / NCI NIH HHS / CA / CA103486; United States / NIDDK NIH HHS / DK / DK61458
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't; Research Support, U.S. Gov't, P.H.S.; Review
  • [Publication-country] United States
  • [Number-of-references] 113
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14. Higa F, Uchihara T, Haranaga S, Yara S, Tateyama M, Oshiro Y, Shiraishi M, Kumasaka T, Seyama K, Fujita J: Malignant epithelioid angiomyolipoma in the kidney and liver of a patient with pulmonary lymphangioleiomyomatosis: lack of response to sirolimus. Intern Med; 2009;48(20):1821-5
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  • [Title] Malignant epithelioid angiomyolipoma in the kidney and liver of a patient with pulmonary lymphangioleiomyomatosis: lack of response to sirolimus.
  • A 26-year-old woman with lymphangioleiomyomatosis (LAM) was hospitalized for the surgical excision of a giant abdominal tumor of right kidney origin.
  • The pathological diagnosis of the tumor was conventional angiomyolipoma (AML).
  • However, the drug did not inhibit the rapid growth of the tumor at all.
  • This finding suggests that sirolimus might not be effective against epithelioid AML, and in such cases, complete surgical resection should be the treatment of choice.
  • [MeSH-major] Angiomyolipoma / diagnosis. Kidney Neoplasms / diagnosis. Liver Neoplasms / diagnosis. Lung Neoplasms / diagnosis. Lymphangioleiomyomatosis / diagnosis. Sirolimus / therapeutic use
  • [MeSH-minor] Adult. Fatal Outcome. Female. Humans. Neoplasm Recurrence, Local / complications. Neoplasm Recurrence, Local / diagnosis. Neoplasm Recurrence, Local / drug therapy


15. Paul E, Thiele E: Efficacy of sirolimus in treating tuberous sclerosis and lymphangioleiomyomatosis. N Engl J Med; 2008 Jan 10;358(2):190-2
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [MeSH-major] Immunosuppressive Agents / therapeutic use. Lung Neoplasms / drug therapy. Lymphangioleiomyomatosis / drug therapy. Sirolimus / therapeutic use. Tuberous Sclerosis / drug therapy
  • [MeSH-minor] Angiomyolipoma / drug therapy. Humans. Kidney Diseases / drug therapy. Protein Kinase Inhibitors / therapeutic use. Protein Kinases / metabolism. Respiratory Function Tests. TOR Serine-Threonine Kinases. Tumor Suppressor Proteins / genetics. Tumor Suppressor Proteins / metabolism

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  • [CommentOn] N Engl J Med. 2008 Jan 10;358(2):140-51 [18184959.001]
  • (PMID = 18184966.001).
  • [ISSN] 1533-4406
  • [Journal-full-title] The New England journal of medicine
  • [ISO-abbreviation] N. Engl. J. Med.
  • [Language] eng
  • [Publication-type] Comment; Editorial
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Immunosuppressive Agents; 0 / Protein Kinase Inhibitors; 0 / Tumor Suppressor Proteins; 0 / tuberous sclerosis complex 1 protein; 4JG2LF96VF / tuberous sclerosis complex 2 protein; EC 2.7.- / Protein Kinases; EC 2.7.1.1 / MTOR protein, human; EC 2.7.1.1 / TOR Serine-Threonine Kinases; W36ZG6FT64 / Sirolimus
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16. Yu W, Fraser RB, Gaskin DA, Fernandez CV, Wright JR Jr: C-Kit-positive metastatic malignant pigmented clear-cell epithelioid tumor arising from the kidney in a child without tuberous sclerosis. Ann Diagn Pathol; 2005 Dec;9(6):330-4
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] C-Kit-positive metastatic malignant pigmented clear-cell epithelioid tumor arising from the kidney in a child without tuberous sclerosis.
  • We report the first pediatric case of malignant pigmented epithelioid clear-cell tumor arising from kidney; the lesion occurred in a 12-year-old girl without tuberous sclerosis.
  • The tumor was widely metastatic to the retroperitoneum and chest, and the patient died of the disease 9 months after diagnosis, despite active chemotherapy.
  • Pigmented epithelioid clear-cell tumor of the kidney is a rare variant of epithelioid angiomyolipoma and a member of the family of perivascular epithelioid cell tumors (PEComas).
  • The tumor demonstrated overlapping features between clear-cell sugar tumor and epithelioid variant of angiomyolipoma.
  • [MeSH-major] Angiomyolipoma / metabolism. Kidney Neoplasms / metabolism. Proto-Oncogene Proteins c-kit / metabolism

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  • (PMID = 16308163.001).
  • [ISSN] 1092-9134
  • [Journal-full-title] Annals of diagnostic pathology
  • [ISO-abbreviation] Ann Diagn Pathol
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] EC 2.7.10.1 / Proto-Oncogene Proteins c-kit
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17. Zderic SA: Renal and adrenal tumors in children. Urol Clin North Am; 2004 Aug;31(3):607-17, xi
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Renal and adrenal tumors in children.
  • Survival rates for children with kidney tumors approach 90% for even the most advanced stages of disease, but the surgical management of large lesions remains challenging.
  • With the development of additional chemotherapeutic regimens and the use of radiation therapy, survival rates have improved dramatically.
  • The National Wilms' Tumor Study has conducted four long-term studies addressing how adjunctive therapy may be tailored optimally to maximize survival and minimize the exposure to chemotherapy and radiation therapy.
  • [MeSH-major] Adrenal Gland Neoplasms / therapy. Kidney Neoplasms / therapy
  • [MeSH-minor] Adrenocortical Carcinoma / diagnosis. Angiomyolipoma / diagnosis. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Child. Clinical Trials as Topic. Combined Modality Therapy. Denys-Drash Syndrome / diagnosis. Humans. Neoplasm Staging. Nephrectomy. Pheochromocytoma / diagnosis. Pheochromocytoma / therapy. Tomography, X-Ray Computed. Wilms Tumor / drug therapy. Wilms Tumor / pathology. Wilms Tumor / radiography. Wilms Tumor / surgery

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  • (PMID = 15313069.001).
  • [ISSN] 0094-0143
  • [Journal-full-title] The Urologic clinics of North America
  • [ISO-abbreviation] Urol. Clin. North Am.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] United States
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18. Biecker E, Fischer HP, Strunk H, Sauerbruch T: Benign hepatic tumours. Z Gastroenterol; 2003 Feb;41(2):191-200
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  • Due to advances in imaging procedures like MRI, Cf-scan and ultrasound as well as progress in immunohistochemistry, the appropriate diagnosis is made ina high percentage of patients without laparotomy and resection.
  • Most important in clinical practice is the differential diagnosis of focal nodular hyperplasia and hepatocellular adenoma because of the risk of rupture and bleeding in the latter.
  • Cavernous haemangioma, the most common benign hepatic tumour, rarely needs treatment.
  • The diagnosis of nodular regenerative hyperplasia is often missed and patients present with secondary complications and signs of portal hypertension that necessitate treatment.
  • The main problem in angiomyolipoma is to distinguish it from malignant processes which do require treatment.
  • Therapeutic options are drug therapy or surgical resection.
  • [MeSH-major] Liver Diseases / diagnosis. Liver Neoplasms / diagnosis
  • [MeSH-minor] Adenoma, Liver Cell / diagnosis. Adenoma, Liver Cell / pathology. Adult. Aged. Diagnostic Imaging. Focal Nodular Hyperplasia / diagnosis. Focal Nodular Hyperplasia / pathology. Granuloma, Plasma Cell / diagnosis. Granuloma, Plasma Cell / pathology. Hemangioendothelioma / diagnosis. Hemangioendothelioma / pathology. Hemangioma, Cavernous / diagnosis. Hemangioma, Cavernous / pathology. Humans. Infant. Liver / pathology. Liver Regeneration / physiology. Middle Aged

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  • (PMID = 12650132.001).
  • [ISSN] 0044-2771
  • [Journal-full-title] Zeitschrift für Gastroenterologie
  • [ISO-abbreviation] Z Gastroenterol
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] Germany
  • [Number-of-references] 138
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