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Frontiers of Medicine

ISSN 2095-0217

ISSN 2095-0225(Online)

CN 11-5983/R

Postal Subscription Code 80-967

2018 Impact Factor: 1.847

Front. Med.    2015, Vol. 9 Issue (3) : 380-383     DOI: 10.1007/s11684-015-0407-x
Cushing’s syndrome during pregnancy caused by adrenal cortical adenoma: a case report and literature review
Wei Wang(),Fengyi Yuan,Dan Xu
Department of Endocrinology and Metabolism, Shenzhen People’s Hospital, Shenzhen 518020, China
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Cushing’s syndrome (CS) during pregnancy is a rare condition with significant maternal and fetal complications. A case of CS during the third trimester of pregnancy secondary to adrenocortical adenoma was reported. Literature review revealed the disadvantages of different treatments in this period. Besides the conservative treatment, surgery is recommended for CS during the third trimester of pregnancy secondary to adrenal adenoma, if an experienced surgeon is available.

Keywords Cushing’s syndrome      pregnancy      adrenocortical adenoma     
Corresponding Authors: Wei Wang   
Just Accepted Date: 10 July 2015   Online First Date: 21 August 2015    Issue Date: 26 August 2015
URL:     OR
Results Normal
8 AM serum cortisol (μg/dl) 31.35 8.7-22.4
4 AM serum cortisol (μg/dl)0 AM serum cortisol (μg/dl)24 h urine cortisol (μg)8 AM serum ACTH (pg/ml)Low-dose dexamethasone midnight suppression testHigh-dose dexamethasone suppression test 24.7625.592131.38<18.72%8.16% 39-3487.2-63.3>50%
Tab.1  Results of serum and urine chemical tests of patient
Group A(35 pregnancies) Group B(9 pregnancies)
Spontaneous abortion 3 (8.6%) 0 (0%)
Stillbirth 4 (11.4) 0 (0%)
Neonatal death 3 (8.6%) 0 (0%)
Preterm birth 19 (54.3%) 3 (33.3%)
Term birth 9 (25.7%) 6 (66.6%)
IUGR 4 (11.4) 0 (0%)
Tab.2  Perinatal outcomes of Groups A and B
Group A(35 pregnancies) Group B(9 pregnancies)
Hypertension 33 (94.3%) 5 (55.6%)
Pre-eclampsia 6 (17.1) 0 (0%)
Diabetes mellitus 15 (42.9%) 1 (11.1%)
Pulmonary edema 6 (17.1%) 0 (0%)
Wound complication 3 (8.6%) 0 (0%)
Maternal death 0 0 (0%)
Tab.3  Maternal outcomes of Groups A and B
1 Lekarev O, New MI. Adrenal disease in pregnancy. Best Pract Res Clin Endocrinol Metab 2011; 25(6): 959–973
doi: 10.1016/j.beem.2011.08.004 pmid: 22115169
2 Pickard J, Jochen AL, Sadur CN, Hofeldt FD. Cushing’s syndrome in pregnancy. Obstet Gynecol Surv 1990; 45(2): 87–93
doi: 10.1097/00006254-199002000-00002 pmid: 2405312
3 Kamiya Y, Okada M, Yoneyama A, Jin-no Y, Hibino T, Watanabe O, Kajiura S, Suzuki Y, Iwata H, Kobayashi S. Surgical successful treatment of Cushing’s syndrome in a pregnant patient complicated with severe cardiac involvement. Endocr J 1998; 45(4): 499–504
doi: 10.1507/endocrj.45.499 pmid: 9881899
4 Nakashima YM, Itonaga Y, Inoue H, Takahashi S. Pulmonary edema after laparoscopic adrenalectomy in a pregnant patient with Cushing’s syndrome. J Anesth 2000; 14(3): 157–159
doi: 10.1007/s005400070026 pmid: 14564585
5 Aishima M, Tanaka M, Haraoka M, Naito S. Retroperitoneal laparoscopic adrenalectomy in a pregnant woman with Cushing’s syndrome. J Urol 2000; 164(3 Pt 1): 770–771
pmid: 10953144
6 Shaw JA, Pearson DW, Krukowski ZH, Fisher PM, Bevan JS. Cushing’s syndrome during pregnancy: curative adrenalectomy at 31 weeks gestation. Eur J Obstet Gynecol Reprod Biol 2002; 105(2): 189–191
doi: 10.1016/S0301-2115(02)00148-3 pmid: 12381486
7 Tejura H, Weiner J, Gibby O, O’Leary AJ, Avasarala S. Cushing’s syndrome in pregnancy. J Obstet Gynaecol 2005; 25(7): 713–714
doi: 10.1080/01443610500303648 pmid: 16263550
8 Sammour RN, Saiegh L, Matter I, Gonen R, Shechner C, Cohen M, Ohel G, Dickstein G. Adrenalectomy for adrenocortical adenoma causing Cushing’s syndrome in pregnancy: a case report and review of literature. Eur J Obstet Gynecol Reprod Biol 2012; 165(1): 1–7
doi: 10.1016/j.ejogrb.2012.05.030 pmid: 22698457
9 Chang I, Cha HH, Kim JH, Choi SJ, Oh SY, Roh CR. Cushing syndrome in pregnancy secondary to adrenal adenoma. Obstet Gynecol Sci 2013; 56(6): 400–403
doi: 10.5468/ogs.2013.56.6.400 pmid: 24396819
10 Lo KW, Lau TK. Cushing’s syndrome in pregnancy secondary to adrenal adenoma. A case report and literature review. Gynecol Obstet Invest 1998; 45(3): 209–212
doi: 10.1159/000009958 pmid: 9565149
11 Handler J. Cushing’s syndrome with uncontrolled hypertension, occasional hypokalemia, and two pregnancies. J Clin Hypertens (Greenwich)2010; 12(7): 516–521
pmid: 20629814
12 Holgado-Galicia MV, Magno JD, Acelajado-Valdenor C, Isip-Tan IT, Lim-Abrahan MA. Cushing’s syndrome in pregnancy. BMJ Case Rep 2011; 2011: bcr0120113720
doi: 10.1136/bcr.01.2011.3720 pmid: 22696666
13 Choi WJ, Jung TS, Paik WY. Cushing’s syndrome in pregnancy with a severe maternal complication: a case report. J Obstet Gynaecol Res 2011; 37(2): 163–167
doi: 10.1111/j.1447-0756.2010.01339.x pmid: 21159041
14 Borna S, Akbari S, Eftekhar T, Mostaan F. Cushing’s syndrome during pregnancy secondary to adrenal adenoma. Acta Med Iran 2012; 50(1): 76–78
pmid: 22267384
15 Kita M, Sakalidou M, Saratzis A, Ioannis S, Avramidis A. Cushing’s syndrome in pregnancy: report of a case and review of the literature. Hormones (Athens)2007; 6(3): 242–246
pmid: 17724009
16 Al Banna R, Husain A, Al Aali J, Ebrahim K, Mohammed. Reversible Cushing dilated cardiomyopathy mimicking peripartum cardiomyopathy with successful subsequent pregnancy. BMJ Case Rep 2011; 2011: bcr0920114825
doi: 10.1136/bcr.09.2011.4825 pmid: 22674115
17 Kim HG, Lee KH, Je GH, Han MS. A case of Cushing’s syndrome in pregnancy secondary to an adrenal cortical adenoma. J Korean Med Sci 2003; 18(3): 444–446
doi: 10.3346/jkms.2003.18.3.444 pmid: 12808338
18 Hunt AB, McCONAHEY WM. Pregnancy associated with diseases of the adrenal glands. Am J Obstet Gynecol 1953; 66(5): 970–987
pmid: 13104499
19 Lindsay JR, Jonklaas J, Oldfield EH, Nieman LK. Cushing’s syndrome during pregnancy: personal experience and review of the literature. J Clin Endocrinol Metab 2005; 90(5): 3077–3083
doi: 10.1210/jc.2004-2361 pmid: 15705919
20 Vilar L, Freitas MC, Lima LH, Lyra R, Kater CE. Cushing’s syndrome in pregnancy: an overview. Arq Bras Endocrinol Metabol 2007; 51(8): 1293–1302
doi: 10.1590/S0004-27302007000800015 pmid: 18209867
21 Cohade C, Broussaud S, Louiset E, Bennet A, Huyghe E, Caron P. Ectopic Cushing’s syndrome due to a pheochromocytoma: a new case in the post-partum and review of literature. Gynecol Endocrinol 2009; 25(9): 624–627
doi: 10.1080/09513590903015411 pmid: 19591016
22 Rask E, Schvarcz E, Hellman P, Hennings J, Karlsson FA, Rao CV. Adrenocorticotropin-independent Cushing’s syndrome in pregnancy related to overexpression of adrenal luteinizing hormone/human chorionic gonadotropin receptors. J Endocrinol Invest 2009; 32(4): 313–316
doi: 10.1007/BF03345718 pmid: 19636197
23 Terhune KP, Jagasia S, Blevins LS Jr, Phay JE. Diagnostic and therapeutic dilemmas of hypercortisolemia during pregnancy: a case report. Am Surg 2009; 75(3): 232–234
pmid: 19350859
24 Casonato A, Daidone V, Pontara E, Albiger N, Cattini MG, Scaroni C. Hypercortisolism and pregnancy upregulate von Willebrand factor through different mechanisms: report on a pregnant patient with Cushing’s syndrome. Blood Coagul Fibrinolysis 2010; 21(5): 476–479
doi: 10.1097/MBC.0b013e328338dbba pmid: 20614575
25 Pollack RP, Brett EM. Adrenocorticotropic hormone-independent Cushing syndrome manifesting during pregnancy. Endocr Pract 2010; 16(2): 260–263
doi: 10.4158/EP09232.CR pmid: 20061276
26 Asicioglu E, Gonenli G, Kahveci A, Yildizeli B, Deyneli O, Yavuz D, Yuksel M, Akalin S. Thymic neuroendocrine carcinoma presenting as cushing’s syndrome: treatment with octreotide combined with surgery and radiotherapy. Onkologie 2011; 34(1-2): 46–49
doi: 10.1159/000323348 pmid: 21346385
27 Bednarek-Tupikowska G, Kubicka E, Sicińska-Werner T, Kazimierczak A, Winowski J, Tupikowska M, Janczyszyn M, Bolanowski M. A case of Cushing’s syndrome in pregnancy. Endokrynol Pol 2011; 62(2): 181–185
pmid: 21528482
28 Boronat M, Marrero D, López-Plasencia Y, Barber M, Schamann Y, Nóvoa FJ. Successful outcome of pregnancy in a patient with Cushing’s disease under treatment with ketoconazole during the first trimester of gestation. Gynecol Endocrinol 2011; 27(9): 675–677
doi: 10.3109/09513590.2010.521268 pmid: 20923280
29 Toutounchi S, Makowska A, Krajewska E, Fiszer P, Cie?la W, Pogorzelski R, Bartnik A, Wielgo? M, Bar-Andziak E, Skórski M. Laparoscopic treatment of Cushing’s syndrome in a woman in late pregnancy- a case presentation. Wideochir Inne Tech Malo Inwazyjne 2011; 6(4): 261–263
doi: 10.5114/wiitm.2011.26263 pmid: 23255991
30 Achong N, D’Emden M, Fagermo N, Mortimer R. Pregnancy-induced Cushing’s syndrome in recurrent pregnancies: case report and literature review. Aust N Z J Obstet Gynaecol 2012; 52(1): 96–100
doi: 10.1111/j.1479-828X.2011.01388.x pmid: 22188308
31 Gopal RA, Acharya SV, Bandgar TR, Menon PS, Shah NS. Cushing disease with pregnancy. Gynecol Endocrinol 2012; 28(7): 533–535
doi: 10.3109/09513590.2011.632789 pmid: 22551350
32 Homer L, Viatge M, Gayet FX, Laurent Y, Kerlan V. Cushing syndrome and pregnancy: a propos of a malignant adrenocortical carcinoma. Gynecol Obstet Fertil 2012; 40(3): e1–e4
doi: 10.1016/j.gyobfe.2011.07.053 pmid: 22342109
33 Kimura Y, Okuyama K, Mitsui K, Tamaki F, Nonaka A, Matsukawa T. Anesthetic management for laparoscopic adrenalectomy in a pregnant patient with Cushing’s syndrome. Masui 2012; 61(6): 605–609
pmid: 22746024
34 Garla V, Kheetan R, Saleem T. Cushing’s syndrome in pregnancy: a diagnostic conundrum. W V Med J 2013; 109(6): 34–36
pmid: 24371863
35 Lim WH, Torpy DJ, Jeffries WS. The medical management of Cushing’s syndrome during pregnancy. Eur J Obstet Gynecol Reprod Biol 2013; 168(1): 1–6
doi: 10.1016/j.ejogrb.2012.12.015 pmid: 23305861
36 Xu JJ, Wang YJ, Shan LZ, Gu W, Zeng WH. Recurrent ACTH-independent Cushing’s syndrome in multiple pregnancies. Gynecol Endocrinol 2013; 29(4): 309–310
doi: 10.3109/09513590.2012.743014 pmid: 23323736
37 Williams RH, Melmed S. Williams Textbook of Endocrinology. Philadelphia: Elsevier/Saunders, 2011: online resource (1920 p.)
38 Lindsay JR, Nieman LK. Adrenal disorders in pregnancy. Endocrinol Metab Clin North Am 2006; 35(1): 1–20, v
doi: 10.1016/j.ecl.2005.09.010 pmid: 16310640
39 Lindsay JR, Nieman LK. The hypothalamic-pituitary-adrenal axis in pregnancy: challenges in disease detection and treatment. Endocr Rev 2005; 26(6): 775–799
doi: 10.1210/er.2004-0025 pmid: 15827110
40 Polli N, Pecori Giraldi F, Cavagnini F. Cushing’s syndrome in pregnancy. J Endocrinol Invest 2003; 26(10): 1045–1050
doi: 10.1007/BF03348206 pmid: 14759081
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