庫欣病合并肺栓塞一例并文獻復習_Chinese Journal of Respiratory and Critical Care Medicine

Authors：

郝娜娜 ^1,3 ,  譚惠文 ^1,2 , 王椿 ^1,2 , 李建薇 ^1,2 , 余葉蓉 ^1,2

1. ;
2. ;
3. ;

Corresponding?author：

譚惠文, Email: huiwent2016@scu.edu.cn

Keywords：

庫欣病; 庫欣綜合征; 肺栓塞; 靜脈血栓栓塞癥

DOI：

10.7507/1671-6205.202210004

Video：

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目的總結分析庫欣綜合征（Cushing syndrome，CS）合并靜脈血栓栓塞癥（venous thromboembolic events，VTE）的發病機制、臨床風險評估及治療，提高臨床醫師對此并發癥的認識。方法報道1例庫欣病（Cushing disease，CD）合并肺栓塞患者的臨床診治經過，并復習國內外相關文獻。結果患者31歲，女性，確診CD，行鞍區占位切除術，術后第6天肺動脈血管三維重建增強計算機斷層掃描（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療，3個月后復查肺動脈CTA未見異常。以“庫欣病、庫欣綜合征”與“肺栓塞、靜脈血栓栓塞癥”為關鍵詞檢索萬方醫學數據庫、中國知網醫學數據庫和維普網數據庫，國內僅3篇相關文獻。檢索pubmed數據庫，共檢索到相關文獻73篇其中28篇個案報道，10篇綜述，35篇臨床研究文章。CS發生VTE風險比普通人群高10余倍，發病機制及臨床風險評估復雜，預防抗凝時間及方案尚未達成共識。結論 CS發生VTE的風險顯著升高，起病急，病死率高。臨床上需警惕、避免、甄別CS相關并發癥，做到早診斷、早治療。

Citation： 郝娜娜, 譚惠文, 王椿, 李建薇, 余葉蓉. 庫欣病合并肺栓塞一例并文獻復習. 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, J?rgensen 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.	吳遐, 高路, 幸兵. 庫欣綜合征所致高凝狀態的研究進展. 基礎醫學與臨床, 2016, 36(1): 121-124.
6.	趙明偉, 周志安, 邊樹偉. 以肺栓塞入院的庫欣綜合征患者1例. 承德醫學院學報, 2022, 39(6): 521-522.
7.	孫旭, 盧琳, 馮銘, 等. 庫欣綜合征伴圍手術期肺栓塞臨床特點分析. 中國現代神經疾病雜志, 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.	中華醫學會呼吸病學分會肺栓塞與肺血管病學組, 中國醫師協會呼吸醫師分會肺栓塞與肺血管病工作委員會, 全國肺栓塞與肺血管病防治協作組. 肺血栓栓塞癥診治與預防指南. 中華醫學雜志, 2018, 98(14): 1060-1087.

1. Dekkers OM, Horváthpuhó E, J?rgensen 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. 吳遐, 高路, 幸兵. 庫欣綜合征所致高凝狀態的研究進展. 基礎醫學與臨床, 2016, 36(1): 121-124.
6. 趙明偉, 周志安, 邊樹偉. 以肺栓塞入院的庫欣綜合征患者1例. 承德醫學院學報, 2022, 39(6): 521-522.
7. 孫旭, 盧琳, 馮銘, 等. 庫欣綜合征伴圍手術期肺栓塞臨床特點分析. 中國現代神經疾病雜志, 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. 中華醫學會呼吸病學分會肺栓塞與肺血管病學組, 中國醫師協會呼吸醫師分會肺栓塞與肺血管病工作委員會, 全國肺栓塞與肺血管病防治協作組. 肺血栓栓塞癥診治與預防指南. 中華醫學雜志, 2018, 98(14): 1060-1087.

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