Takenaka Shun, Hattanda Fumihiko, Kawamura Takuro, Watanabe-Kusunoki Kanako, Nakazawa Daigo, Kameda Hiraku, Amengual Olga, Atsumi Tatsuya, Nishio Saori
Department of Rheumatology, Endocrinology and Nephrology, Faculty of Medicine, Graduate School of Medicine, Hokkaido University Hospital, Kita 14, Nishi 5, Kita-ku, Sapporo, Hokkaido, 060-8648, Japan.
Department of Hemodialysis and Apheresis, Hokkaido University Hospital, Kita14, Nishi5, Kita-Ku, Sapporo, Hokkaido, 060-8648, Japan.
CEN Case Rep. 2025 Jun 27. doi: 10.1007/s13730-025-01013-1.
A 35-year-old female was diagnosed with autosomal dominant polycystic kidney disease (ADPKD) in 2002 and referred to the Nephrology department of our hospital for follow-up and management. Treatment with tolvaptan was started in 2016, which failed to reduce her total kidney volume (TKV) and slow the decline in kidney function. In 2018, she experienced bitemporal hemianopia. Examination revealed enlarged fingers, characteristic facial features, and elevated serum insulin-like growth factor 1 (IGF-1) and growth hormone (GH) levels. A pituitary tumor was identified through the brain, and she was subsequently diagnosed with acromegaly. Lanreotide treatment was initiated, effectively reducing IGF-1 and GH serum levels and TKV within six months. After transsphenoidal surgery, lanreotide was discontinued. However, her oral glucose tolerance test after surgery did not show remission (GH nadir > 0.4 ng/mL), and serum GH levels gradually increased. Furthermore, TKV increased and kidney function decreased rapidly. Lanreotide treatment was restarted, resulting in further TKV reduction, suppression of liver volume enlargement and slowing kidney function decline. Compared to tolvaptan, the evidence for the effectiveness of somatostatin analogs in reducing TKV in ADPKD is not sufficiently established. Our case demonstrates that lanreotide effectively suppressed the progression of TKV, liver volume and kidney dysfunction with correlation to IGF-1 or GH in a patient with ADPKD. Our experience indicates that elevated levels of IGF-1 or GH may contribute to increased kidney and liver volume. Herein, we report the first case of tolvaptan-resistant ADPKD with concomitant acromegaly successfully managed with lanreotide.
一名35岁女性于2002年被诊断为常染色体显性多囊肾病(ADPKD),并转诊至我院肾内科进行随访和治疗。2016年开始使用托伐普坦治疗,但未能减少其总肾体积(TKV),也未能减缓肾功能下降。2018年,她出现双侧颞侧偏盲。检查发现手指粗大、具有特征性面容,血清胰岛素样生长因子1(IGF-1)和生长激素(GH)水平升高。通过脑部检查发现垂体瘤,随后她被诊断为肢端肥大症。开始使用兰瑞肽治疗,在6个月内有效降低了IGF-1和GH血清水平以及TKV。经蝶窦手术后,停用了兰瑞肽。然而,术后她的口服葡萄糖耐量试验未显示缓解(GH最低点>0.4 ng/mL),血清GH水平逐渐升高。此外,TKV增加,肾功能迅速下降。重新开始使用兰瑞肽治疗,导致TKV进一步降低,抑制了肝脏体积增大并减缓了肾功能下降。与托伐普坦相比,生长抑素类似物在降低ADPKD患者TKV方面的有效性证据尚不充分。我们的病例表明,兰瑞肽在一名ADPKD患者中有效抑制了TKV、肝脏体积和肾功能障碍的进展,且与IGF-1或GH相关。我们的经验表明,IGF-1或GH水平升高可能导致肾脏和肝脏体积增加。在此,我们报告首例对托伐普坦耐药的ADPKD合并肢端肥大症患者成功使用兰瑞肽治疗的病例。