Effect of Everolimus Treatment for Regrown Renal Angiomyolipoma Associated with Tuberous Sclerosis Complex After Transcatheter Arterial Embolization

Overview

Journal Int J Clin Oncol

Specialty Oncology

Date 2018 Aug 3

PMID 30069798

Citations 4

Authors

Takashi Hatano

Taishi Matsu-Ura

Kei-Ichiro Mori

Hiroyuki Inaba

Katsuhisa Endo

Mayumi Tamari

Shin Egawa

Affiliations

Soon will be listed here.

Abstract

Background: The aim of this study was to evaluate the effects and the utility of second-line everolimus treatment for regrown renal angiomyolipoma (AML) with tuberous sclerosis complex (TSC) after transcatheter arterial embolization (TAE).

Methods: We investigated a total of 14 patients who underwent second-line everolimus treatment for TSC-AML that regrew after TAE, and assessed their effects and adverse events. Everolimus treatment was performed for AML with a maximum diameter of 4 cm. To determine the reduction ratio of AML, the volume of AML was measured using multislice helical computed tomography. Adverse events were evaluated according to CTCAE v4.0-JCOG. We further compared the treatment effect and adverse events with those in patients receiving first-line everolimus treatment.

Results: The AML volume decreased in all patients, with a ≥ 50% volume decrease in 57% (8 of 14) of the cases, and the mean reduction rate was 53%. We observed no significant difference in the mean reduction rate of AML between second-line everolimus treatment for regrown TSC-AML after TAE and first-line everolimus treatment for TSC-AML. The adverse events were mild and consistent with those reported in our previous study.

Conclusion: Although further studies are needed, everolimus appears to be effective as second-line treatment for TSC-AML that regrew after TAE and a beneficial treatment option for TSC-AML.

Citing Articles

Management of Renal Angiomyolipomas in Tuberous Sclerosis: A Case Series.

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Advances in Molecular Mechanisms of Kidney Disease: Integrating Renal Tumorigenesis of Hereditary Cancer Syndrome.

Cicchetti R, Basconi M, Litterio G, Mascitti M, Tamborino F, Orsini A Int J Mol Sci. 2024; 25(16).

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Clinical practice recommendations for kidney involvement in tuberous sclerosis complex: a consensus statement by the ERKNet Working Group for Autosomal Dominant Structural Kidney Disorders and the ERA Genes & Kidney Working Group.

Mekahli D, Muller R, Marlais M, Wlodkowski T, Haeberle S, de Argumedo M Nat Rev Nephrol. 2024; 20(6):402-420.

PMID: 38443710 DOI: 10.1038/s41581-024-00818-0.

Sporadic Angiomyolipomas Growth Kinetics While on Everolimus: Results of a Phase II Trial.

Geynisman D, Kadow B, Shuch B, Boorjian S, Matin S, Rampersaud E J Urol. 2020; 204(3):531-537.

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References

Krueger D, Northrup H . Tuberous sclerosis complex surveillance and management: recommendations of the 2012 International Tuberous Sclerosis Complex Consensus Conference. Pediatr Neurol. 2013; 49(4):255-65. PMC: 4058297. DOI: 10.1016/j.pediatrneurol.2013.08.002. View

Hatano T, Chikaraishi K, Inaba H, Endo K, Egawa S . Outcomes of everolimus treatment for renal angiomyolipoma associated with tuberous sclerosis complex: A single institution experience in Japan. Int J Urol. 2016; 23(10):833-838. DOI: 10.1111/iju.13168. View

. Identification and characterization of the tuberous sclerosis gene on chromosome 16. Cell. 1993; 75(7):1305-15. DOI: 10.1016/0092-8674(93)90618-z. View

Krummel T, Garnon J, Lang H, Gangi A, Hannedouche T . Percutaneous cryoablation for tuberous sclerosis-associated renal angiomyolipoma with neoadjuvant mTOR inhibition. BMC Urol. 2014; 14:77. PMC: 4177726. DOI: 10.1186/1471-2490-14-77. View

Yamakado K, Tanaka N, Nakagawa T, Kobayashi S, Yanagawa M, Takeda K . Renal angiomyolipoma: relationships between tumor size, aneurysm formation, and rupture. Radiology. 2002; 225(1):78-82. DOI: 10.1148/radiol.2251011477. View

Bissler J, Kingswood J, Radzikowska E, Zonnenberg B, Frost M, Belousova E . Everolimus for renal angiomyolipoma in patients with tuberous sclerosis complex or sporadic lymphangioleiomyomatosis: extension of a randomized controlled trial. Nephrol Dial Transplant. 2015; 31(1):111-9. DOI: 10.1093/ndt/gfv249. View

Sheth R, Feldman A, Paul E, Thiele E, Walker T . Angiographic and volumetric effects of mammalian target of rapamycin inhibitors on angiomyolipomas in tuberous sclerosis. World J Radiol. 2016; 8(3):308-15. PMC: 4807340. DOI: 10.4329/wjr.v8.i3.308. View

Neumann H, Schwarzkopf G, Henske E . Renal angiomyolipomas, cysts, and cancer in tuberous sclerosis complex. Semin Pediatr Neurol. 1999; 5(4):269-75. DOI: 10.1016/s1071-9091(98)80005-3. View

Rakowski S, Winterkorn E, Paul E, Steele D, Halpern E, Thiele E . Renal manifestations of tuberous sclerosis complex: Incidence, prognosis, and predictive factors. Kidney Int. 2006; 70(10):1777-82. DOI: 10.1038/sj.ki.5001853. View

10.

Kumar S, Jayant K, Singh S, Agrawal S . A Case Series & Review of Literature of Angiomyolipoma with Medical & Surgical Perspective. J Clin Diagn Res. 2015; 9(9):PD05-7. PMC: 4606276. DOI: 10.7860/JCDR/2015/13700.6430. View

11.

Wataya-Kaneda M, Tanaka M, Hamasaki T, Katayama I . Trends in the prevalence of tuberous sclerosis complex manifestations: an epidemiological study of 166 Japanese patients. PLoS One. 2013; 8(5):e63910. PMC: 3656843. DOI: 10.1371/journal.pone.0063910. View

12.

Sonis S . Mucositis: The impact, biology and therapeutic opportunities of oral mucositis. Oral Oncol. 2009; 45(12):1015-20. DOI: 10.1016/j.oraloncology.2009.08.006. View

13.

Hatano T, Inaba H, Endo K, Egawa S . Intermittent everolimus administration for renal angiomyolipoma associated with tuberous sclerosis complex. Int J Urol. 2017; 24(11):780-785. DOI: 10.1111/iju.13428. View

14.

Rouviere O, Nivet H, Grenier N, Zini L, Lechevallier E . Kidney damage due to tuberous sclerosis complex: management recommendations. Diagn Interv Imaging. 2013; 94(3):225-37. DOI: 10.1016/j.diii.2013.01.003. View

15.

Park B, Kim C, Park S, Shen S . Percutaneous radiofrequency ablation of renal cell carcinomas in patients with von Hippel Lindau disease: indications, techniques, complications, and outcomes. Acta Radiol. 2013; 54(4):418-27. DOI: 10.1177/0284185113475441. View

16.

Osborne J, Fryer A, Webb D . Epidemiology of tuberous sclerosis. Ann N Y Acad Sci. 1991; 615:125-7. DOI: 10.1111/j.1749-6632.1991.tb37754.x. View

17.

Castagnetti M, Vezzu B, Laverda A, Zampieri S, Rigamonti W . Urological counseling and followup in pediatric tuberous sclerosis complex. J Urol. 2007; 178(5):2155-9. DOI: 10.1016/j.juro.2007.07.058. View

18.

Curatolo P, Bombardieri R, Jozwiak S . Tuberous sclerosis. Lancet. 2008; 372(9639):657-68. DOI: 10.1016/S0140-6736(08)61279-9. View

19.

Ewalt D, Diamond N, Rees C, Sparagana S, Delgado M, Batchelor L . Long-term outcome of transcatheter embolization of renal angiomyolipomas due to tuberous sclerosis complex. J Urol. 2005; 174(5):1764-6. DOI: 10.1097/01.ju.0000177497.31986.64. View

20.

Hatano T, Atsuta M, Inaba H, Endo K, Egawa S . Effect of everolimus treatment for renal angiomyolipoma associated with tuberous sclerosis complex: an evaluation based on tumor density. Int J Clin Oncol. 2017; 23(3):547-552. DOI: 10.1007/s10147-017-1224-9. View