库欣病合并肺栓塞一例并文献复习_Chinese Journal of Respiratory and Critical Care Medicine

Authors：

郝娜娜 ^1,3 ,  谭惠文 ^1,2 , 王椿 ^1,2 , 李建薇 ^1,2 , 余叶蓉 ^1,2

1. ;
2. ;
3. ;

Corresponding author：

谭惠文, Email: huiwent2016@scu.edu.cn

Keywords：

库欣病; 库欣综合征; 肺栓塞; 静脉血栓栓塞症

DOI：

10.7507/1671-6205.202210004

Video：

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目的总结分析库欣综合征（Cushing syndrome，CS）合并静脉血栓栓塞症（venous thromboembolic events，VTE）的发病机制、临床风险评估及治疗，提高临床医师对此并发症的认识。方法报道1例库欣病（Cushing disease，CD）合并肺栓塞患者的临床诊治经过，并复习国内外相关文献。结果患者31岁，女性，确诊CD，行鞍区占位切除术，术后第6天肺动脉血管三维重建增强计算机断层扫描（computed tomography angiography，CTA）提示肺栓塞，先后予以低分子肝素及利伐沙班抗凝治疗，3个月后复查肺动脉CTA未见异常。以“库欣病、库欣综合征”与“肺栓塞、静脉血栓栓塞症”为关键词检索万方医学数据库、中国知网医学数据库和维普网数据库，国内仅3篇相关文献。检索pubmed数据库，共检索到相关文献73篇其中28篇个案报道，10篇综述，35篇临床研究文章。CS发生VTE风险比普通人群高10余倍，发病机制及临床风险评估复杂，预防抗凝时间及方案尚未达成共识。结论 CS发生VTE的风险显著升高，起病急，病死率高。临床上需警惕、避免、甄别CS相关并发症，做到早诊断、早治疗。

Citation： 郝娜娜, 谭惠文, 王椿, 李建薇, 余叶蓉. 库欣病合并肺栓塞一例并文献复习. Chinese Journal of Respiratory and Critical Care Medicine, 2023, 22(8): 587-590. doi: 10.7507/1671-6205.202210004 Copy

1.	Dekkers OM, Horváthpuhó E, Jørgensen JO, et al. Multisystem morbidity and mortality in Cushing's syndrome: a cohort study. J Clin Endocrinol Metab, 2013, 98(6): 2277-2284.
2.	Wagner J, Langlois F, Lim DST, et al. Hypercoagulability and risk of venous thromboembolic events in endogenous Cushing's syndrome: a systematic meta-analysis. Front Endocrinol (Lausanne), 2019, 9: 805.
3.	Broder MS, Neary MP, Chang E, et al. Incidence of Cushing's syndrome and Cushing's disease in commercially-insured patients <65 years old in the United States. Pituitary, 2015, 18(3): 283-289.
4.	Amodru V, Ferriere A, Tabarin A, et al. Cushing's syndrome in the elderly: data from the European Registry on Cushing's syndrome. Eur J Endocrinol, 2023, 188(4): 395-406.
5.	吴遐, 高路, 幸兵. 库欣综合征所致高凝状态的研究进展. 基础医学与临床, 2016, 36(1): 121-124.
6.	赵明伟, 周志安, 边树伟. 以肺栓塞入院的库欣综合征患者1例. 承德医学院学报, 2022, 39(6): 521-522.
7.	孙旭, 卢琳, 冯铭, 等. 库欣综合征伴围手术期肺栓塞临床特点分析. 中国现代神经疾病杂志, 2021, 21(3): 177-184.
8.	Huckhagel T, Atlihan G, Langer F, et al. Prevalence of perioperative asymptomatic venous thromboses of the lower extremity in 30 consecutive patients undergoing transsphenoidal surgery for Cushing's disease. Sci Rep, 2023, 13((1): 3217.
9.	Feelders RA, Nieman LK. Hypercoagulability in Cushing's syndrome: incidence, pathogenesis and need for thromboprophylaxis protocols. Pituitary, 2022, 25(5): 746-749.
10.	Birdwell L, Lodish M, Tirosh A, et al. Coagulation profile dynamics in pediatric patients with Cushing syndrome: a prospective, observational comparative study. J Pediatr, 2016, 177: 227-231.
11.	Erem C, Nuhoglu I, Yilmaz M, et al. Blood coagulation and fibrinolysis in patients with Cushing's syndrome: increased plasminogen activator inhibitor-1, decreased tissue factor pathway inhibitor, and unchanged thrombin-activatable fibrinolysis inhibitor levels. J Endocrinol Invest, 2009, 32(2): 169-174.
12.	Coelho MC, Santos CV, Vieira Neto L, et al. Adverse effects of glucocorticoids: coagulopathy. Eur J Endocrinol, 2015, 173(4): M11-M21.
13.	van der Pas R, de Bruin C, Leebeek FW, et al. The hypercoagulable state in Cushing's disease is associated with increased levels of procoagulant factors and impaired fibrinolysis, but is not reversible after short-term biochemical remission induced by medical therapy. J Clin Endocrinol Metab, 2012, 97(4): 1303-1310.
14.	Manetti L, Bogazzi F, Giovannetti C, et al. Changes in coagulation indexes and occurrence of venous thromboembolism in patients with Cushing's syndrome: results from a prospective study before and after surgery. Eur J Endocrinol, 2010, 163(5): 783-791.
15.	Zilio M, Mazzai L, Sartori MT, et al. A venous thromboembolism risk assessment model for patients with Cushing's syndrome. Endocrine, 2016, 52(2): 322-332.
16.	Waqar M, Chadwick A, Kersey J, et al. Venous thromboembolism chemical prophylaxis after endoscopic trans-sphenoidal pituitary surgery. Pituitary, 2022, 25(2): 267-274.
17.	Boscaro M, Sonino N, Scarda A, et al. Anticoagulant prophylaxis markedly reduces thromboembolic complications in Cushing's syndrome. J Clin Endocrinol Metab, 2002, 87(8): 3662-3666.
18.	Barbot M, Daidone V, Zilio M, et al. Perioperative thromboprophylaxis in Cushing's disease: What we did and what we are doing? Pituitary, 2015, 18(4): 487-493.
19.	Babic B, De Roulet A, Volpe A, et al. Is VTE prophylaxis necessary on discharge for patients undergoing adrenalectomy for Cushing syndrome? J Endocr Soc, 2018, 3(2): 304-313.
20.	Suarez MG, Stack M, Hinojosa-Amaya JM, et al. Hypercoagulability in Cushing syndrome, prevalence of thrombotic events: a large, single-center, retrospective study. J Endocr Soc, 2019, 4(2): bvz033.
21.	Ferrante E, Serban AL, Clerici M, et al. Evaluation of procoagulant imbalance in Cushing's syndrome after short- and long-term remission of disease. J Endocrinol Invest, 2022, 45(1): 9-16.
22.	Stuijver DJ, van Zaane B, Feelders RA, et al. Incidence of venous thromboembolism in patients with Cushing's syndrome: a multicenter cohort study. J Clin Endocrinol Metab, 2011, 96(11): 3525-3532.
23.	McLeod MK. Complications following adrenal surgery. J Natl Med Assoc, 1991, 83(2): 161-164.
24.	Fukuoka H, Takeuchi T, Matsumoto R, et al. D-dimer as a significant marker of deep vein thrombosis in patients with subclinical or overt Cushing's syndrome. Endocr J, 2014, 61(10): 1003-1010.
25.	van Haalen FM, Kaya M, Pelsma ICM, et al. Current clinical practice for thromboprophylaxis management in patients with Cushing's syndrome across reference centers of the European Reference Network on Rare Endocrine Conditions (Endo-ERN). Orphanet J Rare Dis, 2022, 17(1): 178.
26.	中华医学会呼吸病学分会肺栓塞与肺血管病学组, 中国医师协会呼吸医师分会肺栓塞与肺血管病工作委员会, 全国肺栓塞与肺血管病防治协作组. 肺血栓栓塞症诊治与预防指南. 中华医学杂志, 2018, 98(14): 1060-1087.

1. Dekkers OM, Horváthpuhó E, Jørgensen JO, et al. Multisystem morbidity and mortality in Cushing's syndrome: a cohort study. J Clin Endocrinol Metab, 2013, 98(6): 2277-2284.
2. Wagner J, Langlois F, Lim DST, et al. Hypercoagulability and risk of venous thromboembolic events in endogenous Cushing's syndrome: a systematic meta-analysis. Front Endocrinol (Lausanne), 2019, 9: 805.
3. Broder MS, Neary MP, Chang E, et al. Incidence of Cushing's syndrome and Cushing's disease in commercially-insured patients <65 years old in the United States. Pituitary, 2015, 18(3): 283-289.
4. Amodru V, Ferriere A, Tabarin A, et al. Cushing's syndrome in the elderly: data from the European Registry on Cushing's syndrome. Eur J Endocrinol, 2023, 188(4): 395-406.
5. 吴遐, 高路, 幸兵. 库欣综合征所致高凝状态的研究进展. 基础医学与临床, 2016, 36(1): 121-124.
6. 赵明伟, 周志安, 边树伟. 以肺栓塞入院的库欣综合征患者1例. 承德医学院学报, 2022, 39(6): 521-522.
7. 孙旭, 卢琳, 冯铭, 等. 库欣综合征伴围手术期肺栓塞临床特点分析. 中国现代神经疾病杂志, 2021, 21(3): 177-184.
8. Huckhagel T, Atlihan G, Langer F, et al. Prevalence of perioperative asymptomatic venous thromboses of the lower extremity in 30 consecutive patients undergoing transsphenoidal surgery for Cushing's disease. Sci Rep, 2023, 13((1): 3217.
9. Feelders RA, Nieman LK. Hypercoagulability in Cushing's syndrome: incidence, pathogenesis and need for thromboprophylaxis protocols. Pituitary, 2022, 25(5): 746-749.
10. Birdwell L, Lodish M, Tirosh A, et al. Coagulation profile dynamics in pediatric patients with Cushing syndrome: a prospective, observational comparative study. J Pediatr, 2016, 177: 227-231.
11. Erem C, Nuhoglu I, Yilmaz M, et al. Blood coagulation and fibrinolysis in patients with Cushing's syndrome: increased plasminogen activator inhibitor-1, decreased tissue factor pathway inhibitor, and unchanged thrombin-activatable fibrinolysis inhibitor levels. J Endocrinol Invest, 2009, 32(2): 169-174.
12. Coelho MC, Santos CV, Vieira Neto L, et al. Adverse effects of glucocorticoids: coagulopathy. Eur J Endocrinol, 2015, 173(4): M11-M21.
13. van der Pas R, de Bruin C, Leebeek FW, et al. The hypercoagulable state in Cushing's disease is associated with increased levels of procoagulant factors and impaired fibrinolysis, but is not reversible after short-term biochemical remission induced by medical therapy. J Clin Endocrinol Metab, 2012, 97(4): 1303-1310.
14. Manetti L, Bogazzi F, Giovannetti C, et al. Changes in coagulation indexes and occurrence of venous thromboembolism in patients with Cushing's syndrome: results from a prospective study before and after surgery. Eur J Endocrinol, 2010, 163(5): 783-791.
15. Zilio M, Mazzai L, Sartori MT, et al. A venous thromboembolism risk assessment model for patients with Cushing's syndrome. Endocrine, 2016, 52(2): 322-332.
16. Waqar M, Chadwick A, Kersey J, et al. Venous thromboembolism chemical prophylaxis after endoscopic trans-sphenoidal pituitary surgery. Pituitary, 2022, 25(2): 267-274.
17. Boscaro M, Sonino N, Scarda A, et al. Anticoagulant prophylaxis markedly reduces thromboembolic complications in Cushing's syndrome. J Clin Endocrinol Metab, 2002, 87(8): 3662-3666.
18. Barbot M, Daidone V, Zilio M, et al. Perioperative thromboprophylaxis in Cushing's disease: What we did and what we are doing? Pituitary, 2015, 18(4): 487-493.
19. Babic B, De Roulet A, Volpe A, et al. Is VTE prophylaxis necessary on discharge for patients undergoing adrenalectomy for Cushing syndrome? J Endocr Soc, 2018, 3(2): 304-313.
20. Suarez MG, Stack M, Hinojosa-Amaya JM, et al. Hypercoagulability in Cushing syndrome, prevalence of thrombotic events: a large, single-center, retrospective study. J Endocr Soc, 2019, 4(2): bvz033.
21. Ferrante E, Serban AL, Clerici M, et al. Evaluation of procoagulant imbalance in Cushing's syndrome after short- and long-term remission of disease. J Endocrinol Invest, 2022, 45(1): 9-16.
22. Stuijver DJ, van Zaane B, Feelders RA, et al. Incidence of venous thromboembolism in patients with Cushing's syndrome: a multicenter cohort study. J Clin Endocrinol Metab, 2011, 96(11): 3525-3532.
23. McLeod MK. Complications following adrenal surgery. J Natl Med Assoc, 1991, 83(2): 161-164.
24. Fukuoka H, Takeuchi T, Matsumoto R, et al. D-dimer as a significant marker of deep vein thrombosis in patients with subclinical or overt Cushing's syndrome. Endocr J, 2014, 61(10): 1003-1010.
25. van Haalen FM, Kaya M, Pelsma ICM, et al. Current clinical practice for thromboprophylaxis management in patients with Cushing's syndrome across reference centers of the European Reference Network on Rare Endocrine Conditions (Endo-ERN). Orphanet J Rare Dis, 2022, 17(1): 178.
26. 中华医学会呼吸病学分会肺栓塞与肺血管病学组, 中国医师协会呼吸医师分会肺栓塞与肺血管病工作委员会, 全国肺栓塞与肺血管病防治协作组. 肺血栓栓塞症诊治与预防指南. 中华医学杂志, 2018, 98(14): 1060-1087.

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