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肾上腺皮质癌年轻患者中高雄激素血症诱导的卓越运动能力:10年缓解期

Hyperandrogenism-induced Athletic Excellence in a Young Patient with Adrenocortical Carcinoma: 10 Years of Remission.

作者信息

Kono Takashi, Kono Satomi, Nakamura Yasuhiro, Fujii Yoichi, Sasano Hironobu, Tanaka Tomoaki

机构信息

Department of Molecular Diagnosis, Chiba University Graduate School of Medicine, Chiba 260-8670, Japan.

Research Institute of Disaster Medicine, Chiba University, Chiba 260-8670, Japan.

出版信息

JCEM Case Rep. 2025 Jul 31;3(9):luaf170. doi: 10.1210/jcemcr/luaf170. eCollection 2025 Sep.

DOI:10.1210/jcemcr/luaf170
PMID:40746503
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12311429/
Abstract

A 17-year-old female softball player presented with progressive virilization and markedly enhanced athletic performance. Laboratory evaluation revealed elevated serum testosterone with autonomous adrenal secretion. Imaging analysis demonstrated an 8-cm right adrenal mass. Adrenalectomy was performed, and histopathological examination confirmed stage II adrenocortical carcinoma (ACC) with a Ki-67 labeling index of 14%. Postoperatively, she received adjuvant mitotane therapy. At 34 months, a solitary hepatic metastasis was successfully treated with radiofrequency ablation and combination chemotherapy. She has remained disease-free for more than 8 years following initial surgery. This case demonstrates the clinical impact of pathological hyperandrogenism on athletic performance and highlights the potential for long-term remission in young patients with aggressive ACC managed through multimodal therapy.

摘要

一名17岁的女性垒球运动员出现进行性男性化和运动表现显著增强。实验室检查显示血清睾酮升高,肾上腺自主分泌。影像学分析显示右侧肾上腺有一个8厘米的肿块。进行了肾上腺切除术,组织病理学检查证实为II期肾上腺皮质癌(ACC),Ki-67标记指数为14%。术后,她接受了米托坦辅助治疗。34个月时,通过射频消融和联合化疗成功治疗了孤立性肝转移。初次手术后,她已无病生存超过8年。该病例展示了病理性高雄激素血症对运动表现的临床影响,并突出了通过多模式治疗管理的侵袭性ACC年轻患者实现长期缓解的潜力。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a594/12311429/a8f768067f8a/luaf170f5.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a594/12311429/35a68168e151/luaf170f1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a594/12311429/8511a8c4ce71/luaf170f2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a594/12311429/b3fb35736697/luaf170f3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a594/12311429/6f0627a09501/luaf170f4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a594/12311429/a8f768067f8a/luaf170f5.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a594/12311429/35a68168e151/luaf170f1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a594/12311429/8511a8c4ce71/luaf170f2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a594/12311429/b3fb35736697/luaf170f3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a594/12311429/6f0627a09501/luaf170f4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a594/12311429/a8f768067f8a/luaf170f5.jpg

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本文引用的文献

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Giant androgen-producing adrenocortical carcinoma causing male characteristics.产生雄激素的巨大肾上腺皮质癌导致男性化特征。
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BCOR gene alterations in hematologic diseases.BCOR 基因改变与血液系统疾病。
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Can the Neuromuscular Performance of Young Athletes Be Influenced by Hormone Levels and Different Stages of Puberty?年轻运动员的神经肌肉表现是否会受到激素水平和青春期不同阶段的影响?
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Adjuvant Therapy in Adrenocortical Carcinoma: Reflections and Future Directions.肾上腺皮质癌的辅助治疗:思考与未来方向
Cancers (Basel). 2020 Feb 22;12(2):508. doi: 10.3390/cancers12020508.
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Adrenocortical carcinoma - towards genomics guided clinical care.肾上腺皮质癌——迈向基于基因组学的临床治疗。
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European Society of Endocrinology Clinical Practice Guidelines on the management of adrenocortical carcinoma in adults, in collaboration with the European Network for the Study of Adrenal Tumors.欧洲内分泌学会成人肾上腺皮质癌管理临床实践指南,与欧洲肾上腺肿瘤研究网络合作。
Eur J Endocrinol. 2018 Oct 1;179(4):G1-G46. doi: 10.1530/EJE-18-0608.
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Circulating Testosterone as the Hormonal Basis of Sex Differences in Athletic Performance.循环睾酮作为运动表现性别差异的激素基础。
Endocr Rev. 2018 Oct 1;39(5):803-829. doi: 10.1210/er.2018-00020.
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The Characteristics and Trends in Adrenocortical Carcinoma: A United States Population Based Study.肾上腺皮质癌的特征与趋势:一项基于美国人群的研究
J Clin Med Res. 2018 Aug;10(8):636-640. doi: 10.14740/jocmr3503w. Epub 2018 Jun 27.
9
Effects of mitotane on the hypothalamic-pituitary-adrenal axis in patients with adrenocortical carcinoma.米托坦对肾上腺皮质癌患者下丘脑-垂体-肾上腺轴的影响。
Eur J Endocrinol. 2017 Oct;177(4):361-367. doi: 10.1530/EJE-17-0452. Epub 2017 Aug 5.
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Comprehensive Pan-Genomic Characterization of Adrenocortical Carcinoma.肾上腺皮质癌的全基因组综合特征分析
Cancer Cell. 2016 May 9;29(5):723-736. doi: 10.1016/j.ccell.2016.04.002.