Department of Internal Medicine, Chonnam National University Medical School, Gwangju, South Korea.
Front Endocrinol (Lausanne). 2020 Jul 14;11:451. doi: 10.3389/fendo.2020.00451. eCollection 2020.
Pituitary carcinomas are rare diseases defined as pituitary tumors with metastases. In this report, we describe a case of malignant prolactinoma with liver metastases masquerading as metastatic gastrointestinal stromal tumor (GIST). A 54-years-old woman received dopamine agonists for macroprolactinoma for 2 years, followed by transsphenoidal surgery due to a poor response to medical therapy. Despite the continuation of dopamine agonist after surgery, serum prolactin level progressively increased to above 8,000 ng/ml in 5 years. There was no evidence of disease recurrence on sella magnetic resonance imaging (MRI). She stopped medical therapy. Meanwhile, she was diagnosed with GIST accompanied by liver and peritoneal metastases. After a 2-months treatment with imatinib, she suddenly presented with headache and visual impairment. Sella MRI showed a 3.3-cm sized pituitary mass, and serum prolactin levels were still high. For the recurred mass, she underwent a second surgery followed by radiation therapy. During the imatinib treatment for GIST, main mass and peritoneal metastases were dramatically decreased, but liver metastases were markedly aggravated. Liver masses were eventually confirmed as metastases from prolactin-producing pituitary carcinoma and not from GIST by percutaneous biopsy. Unfortunately, she died 6 months after the second surgery due to acute renal failure and sepsis. This case suggests that highly sustained serum prolactin levels during the dopamine agonist may indicate prolactin-producing pituitary carcinomas with hidden metastases.
垂体癌是一种罕见的疾病,定义为发生转移的垂体肿瘤。在本报告中,我们描述了一例以肝转移为表现的恶性催乳素瘤,误诊为转移性胃肠道间质瘤(GIST)。一名 54 岁女性因患有大催乳素瘤接受了多巴胺激动剂治疗 2 年,由于对药物治疗反应不佳,随后接受了经蝶窦手术。尽管手术后继续使用多巴胺激动剂,但 5 年内血清催乳素水平逐渐升高至 8000ng/ml 以上。鞍区磁共振成像(MRI)未见疾病复发迹象。她停止了药物治疗。同时,她被诊断为伴有肝和腹膜转移的 GIST。在接受伊马替尼治疗 2 个月后,她突然出现头痛和视力障碍。鞍区 MRI 显示 3.3cm 大小的垂体肿块,血清催乳素水平仍较高。对于复发性肿块,她接受了第二次手术和放射治疗。在 GIST 接受伊马替尼治疗期间,主要肿块和腹膜转移明显减少,但肝转移明显加重。经皮肝穿刺活检证实肝转移瘤来源于分泌催乳素的垂体癌,而非 GIST。不幸的是,她在第二次手术后 6 个月因急性肾衰竭和败血症死亡。本病例提示,多巴胺激动剂治疗期间持续高血清催乳素水平可能提示存在隐匿性转移的催乳素分泌性垂体癌。