{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,19]],"date-time":"2026-03-19T21:08:20Z","timestamp":1773954500621,"version":"3.50.1"},"reference-count":27,"publisher":"Wiley","issue":"7","license":[{"start":{"date-parts":[[2025,7,8]],"date-time":"2025-07-08T00:00:00Z","timestamp":1751932800000},"content-version":"vor","delay-in-days":7,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"},{"start":{"date-parts":[[2025,7,1]],"date-time":"2025-07-01T00:00:00Z","timestamp":1751328000000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Nephrology"],"published-print":{"date-parts":[[2025,7]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>\n                    Renal angiomyolipomas (AMLs) are a common renal manifestation in tuberous sclerosis complex (TSC), occurring in 50%\u201385% of cases. Even in asymptomatic individuals, treatment is often initiated based on AML size, with mTOR inhibitors being the primary medical therapy. AMLs tend to enlarge with age, and treatment during school age is rare. Secondary AML shrinkage in paediatric TSC patients has been reported who received everolimus for subependymal giant cell astrocytomas (SEGA) or epilepsy, but studies on its direct use for AMLs in paediatric cases remain limited. A 9\u2010year\u2010old girl born to a mother with TSC was diagnosed at birth due to cardiac rhabdomyomas, cortical tubers, and Shagreen patches. At the age of 4, ultrasound detected multiple AMLs in both kidneys, with the largest lesions measuring 2.3\u2009cm in the right kidney and 1.6\u2009cm in the left. By the age of 7, MRI showed further growth to 4.8\u2009cm. Despite the absence of symptoms, the risk of haemorrhage led to treatment initiation. Everolimus was started at 1.0\u2009mg\/day (1.3\u2009mg\/m\n                    <jats:sup>2<\/jats:sup>\n                    \/day), significantly lower than the standard 4.5\u2009mg\/m\n                    <jats:sup>2<\/jats:sup>\n                    \/day. Trough levels remained below the target range, yet AMLs showed a shrinking tendency, with MRI at age 9 revealing a reduction in the largest AML to 2.4\u2009cm. Stomatitis occurred intermittently, but no serious adverse effects were observed. This case suggests that low\u2010dose everolimus can effectively reduce AML size while minimising adverse effects, highlighting its potential as a treatment option for AMLs in paediatric TSC patients.\n                  <\/jats:p>","DOI":"10.1111\/nep.70069","type":"journal-article","created":{"date-parts":[[2025,7,9]],"date-time":"2025-07-09T15:25:01Z","timestamp":1752074701000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Successful Treatment of Growing Renal Angiomyolipomas With Low\u2010Dose Everolimus in a School\u2010Aged Child With Tuberous Sclerosis Complex"],"prefix":"10.1111","volume":"30","author":[{"given":"Satoshi","family":"Senoo","sequence":"first","affiliation":[{"name":"Department of Pediatrics The University of Tokyo Faculty of Medicine  Bunkyo\u2010ku Tokyo Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6674-1221","authenticated-orcid":false,"given":"Shoichiro","family":"Kanda","sequence":"additional","affiliation":[{"name":"Department of Pediatrics The University of Tokyo Faculty of Medicine  Bunkyo\u2010ku Tokyo Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Keiichi","family":"Takizawa","sequence":"additional","affiliation":[{"name":"Department of Pediatrics The University of Tokyo Faculty of Medicine  Bunkyo\u2010ku Tokyo Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hiroshi","family":"Terashima","sequence":"additional","affiliation":[{"name":"Department of Pediatrics The University of Tokyo Faculty of Medicine  Bunkyo\u2010ku Tokyo Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yuko","family":"Kajiho","sequence":"additional","affiliation":[{"name":"Department of Pediatrics The University of Tokyo Faculty of Medicine  Bunkyo\u2010ku Tokyo Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yutaka","family":"Harita","sequence":"additional","affiliation":[{"name":"Department of Pediatrics The University of Tokyo Faculty of Medicine  Bunkyo\u2010ku Tokyo Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2025,7,8]]},"reference":[{"key":"e_1_2_11_2_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMra055323"},{"key":"e_1_2_11_3_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1464-410X.2012.11618.x"},{"key":"e_1_2_11_4_1","doi-asserted-by":"publisher","DOI":"10.1093\/ndt\/gfy063"},{"key":"e_1_2_11_5_1","doi-asserted-by":"publisher","DOI":"10.3389\/fped.2021.767394"},{"key":"e_1_2_11_6_1","doi-asserted-by":"publisher","DOI":"10.3389\/fneur.2020.00972"},{"key":"e_1_2_11_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.pediatrneurol.2021.07.011"},{"key":"e_1_2_11_8_1","doi-asserted-by":"publisher","DOI":"10.1038\/sj.ki.5001853"},{"key":"e_1_2_11_9_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.33.6.480"},{"key":"e_1_2_11_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-5347(01)63072-6"},{"key":"e_1_2_11_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-5347(17)35895-0"},{"key":"e_1_2_11_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1071-9091(98)80005-3"},{"key":"e_1_2_11_13_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1523-1755.2004.00838.x"},{"key":"e_1_2_11_14_1","doi-asserted-by":"publisher","DOI":"10.1111\/iju.15213"},{"key":"e_1_2_11_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.juro.2016.09.082"},{"key":"e_1_2_11_16_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0063910"},{"key":"e_1_2_11_17_1","doi-asserted-by":"publisher","DOI":"10.1038\/ejhg.2015.47"},{"key":"e_1_2_11_18_1","doi-asserted-by":"publisher","DOI":"10.1097\/GIM.0b013e31803068c7"},{"key":"e_1_2_11_19_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00467-006-0133-3"},{"key":"e_1_2_11_20_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00467-017-3806-1"},{"key":"e_1_2_11_21_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa1001671"},{"key":"e_1_2_11_22_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.urology.2019.12.041"},{"key":"e_1_2_11_23_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10147-020-01792-w"},{"key":"e_1_2_11_24_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.2012-3607"},{"key":"e_1_2_11_25_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(12)61134-9"},{"key":"e_1_2_11_26_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(12)61767-X"},{"key":"e_1_2_11_27_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(16)31419-2"},{"key":"e_1_2_11_28_1","doi-asserted-by":"publisher","DOI":"10.3389\/fneur.2021.581102"}],"container-title":["Nephrology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/nep.70069","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/nep.70069","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/nep.70069","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,3,10]],"date-time":"2026-03-10T09:10:52Z","timestamp":1773133852000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/nep.70069"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,7]]},"references-count":27,"journal-issue":{"issue":"7","published-print":{"date-parts":[[2025,7]]}},"alternative-id":["10.1111\/nep.70069"],"URL":"https:\/\/doi.org\/10.1111\/nep.70069","archive":["Portico"],"relation":{},"ISSN":["1320-5358","1440-1797"],"issn-type":[{"value":"1320-5358","type":"print"},{"value":"1440-1797","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,7]]},"assertion":[{"value":"2025-02-07","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-06-12","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-07-08","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e70069"}}