Griffin Tomás P, Bogdanet Delia, Navin Patrick, Callagy Grace, O'Shea Paula M, Bell Marcia
Centre for Diabetes, Endocrinology and Metabolism, Galway University Hospitals, Galway, Ireland.
Department of Radiology, Galway University Hospitals, Galway, Ireland.
Ir J Med Sci. 2018 Nov;187(4):993-998. doi: 10.1007/s11845-018-1756-7. Epub 2018 Feb 19.
A 51-year-old male presented 25 years ago with excessive sweating and haematuria. Blood pressure was labile. CT abdomen showed a large right-sided adrenal mass. Two 24-h urine collections showed elevated urinary catecholamines. Right adrenal resection was performed; a phaeochromocytoma (PC) was confirmed histologically. Two decades later, the patient represented with excessive sweating and measured variable blood pressure readings. Measurement of plasma metanephrines (PMets) showed elevated normetanephrine (NMN) [50,250 (R.I. 0-1180) pmol/L] and metanephrine (MN) [1030 (R.I. 0-510) pmol/L] values. CT abdomen showed a 100 × 90 × 63 mm enhancing mass in the right retroperitoneum. Curative resection was undertaken confirming recurrent PC. Follow-up post-resection, plasma NMN was discordant, 1314 pmol/L (above decision threshold) at 30 min and 911 pmol/L (below decision threshold) at 40 min. Acute clinical awareness of persistent disease mandated the performance of a metaiodobenzylguanidine (MIBG) scan and CT abdomen. These confirmed residual disease in the upper right side of the retroperitoneum. Persistent disease following redo surgery could have been missed if only seated-sampling upper reference limits were applied to PMets collected at 40 min. Our experience with this patient triggered a review of our PMets sampling strategy. There was no statistically significant difference in PMets sampled at 30 and at 40 min seated-rest. Optimum diagnostic test accuracy was achieved using a supine-sampling strategy at a single time point (30 min). Our case highlights the importance of maintaining a high index of clinical suspicion for residual/recurrent disease in the face of inconclusive biochemistry, followed by appropriate targeted radiology using MIBG or PET-CT in patients with PPGL.
一名51岁男性于25年前出现多汗和血尿。血压不稳定。腹部CT显示右侧肾上腺有一个大肿块。两次24小时尿样收集显示尿儿茶酚胺升高。进行了右侧肾上腺切除术;组织学证实为嗜铬细胞瘤(PC)。二十年后,患者再次出现多汗,血压测量值波动。血浆间甲肾上腺素(PMets)测量显示去甲间甲肾上腺素(NMN)升高[50,250(参考区间0 - 1180)pmol/L],间甲肾上腺素(MN)[1030(参考区间0 - 510)pmol/L]升高。腹部CT显示右后腹膜有一个100×90×63mm的强化肿块。进行了根治性切除,证实为复发性PC。切除术后随访,血浆NMN不一致,30分钟时为1314pmol/L(高于判定阈值),40分钟时为911pmol/L(低于判定阈值)。对持续性疾病的敏锐临床意识促使进行了间碘苄胍(MIBG)扫描和腹部CT。这些检查证实后腹膜右上侧有残留疾病。如果仅将坐位采样的上限参考值应用于40分钟时采集的PMets,再次手术后的持续性疾病可能会被漏诊。我们对该患者的经验促使我们对PMets采样策略进行了回顾。坐位休息30分钟和40分钟时采集的PMets无统计学显著差异。在单个时间点(30分钟)采用仰卧位采样策略可实现最佳诊断测试准确性。我们 的病例强调了在生化检查结果不明确的情况下,对PPGL患者残留/复发性疾病保持高度临床怀疑指数的重要性,随后进行适当的靶向放射学检查,如使用MIBG或PET - CT。