Abstract
Introduction
During normal pregnancy there are significant changes in hypothalamic-pituitary-adrenal axis, with increased levels of plasma cortisol and adrenocorticotropic hormone which sometimes reach values observed in patients with Cushing’s syndrome. Cushing’s syndrome (CS) is rarely encountered during pregnancy, but is associated with serious maternal and fetal complications.
Case presentation
A 31-year-old female was admitted to our institution four weeks after delivery. Physical examination revealed moon face, purple striae throughout the abdomen, bruising over the legs, a dorsocervical fat pad and hirsutism. She delivered a eutrophic preterm newborn at 34 weeks gestation, without any maternal or fetal complications during delivery. Imaging showed a mass in the right suprarenal gland with a normal pituitary. After four weeks the patient underwent a right adrenalectomy. The mass was eventually identified as an adrenocortical adenoma.
Conclusion
In our case the diagnosis of CS was established only after pregnancy, which enabled the development of numerous adverse consequences secondary to increased plasma cortisol. If CS is recognized during pregnancy, treatment and its timing could be carefully chosen according to the patient’s individual characteristics.
Similar content being viewed by others
References
Steffensen C., Bak A.M., Rubeck K.Z., Jorgensen J.O., Epidemiology of Cushing’s syndrome, Neuroendocrinology, 2010, 92Suppl 1:1–5
Alves M., Neves C., Medina J.L., Laboratorial diagnosis of Cushing’s syndrome, Acta Med Port., 2010, 23(1),63–67
Aron D.C., Cushing’s syndrome: why is diagnosis so difficult? Rev. Endocr. Metab. Disord. 2010, 11(2):105–116
Vilar. L, Freitas Mda. C., Faria M., Montenegro R., Casulari L.A., Naves L., Bruno O.D., Pitfalls in the diagnosis of Cushing’s syndrome, Arq. Bras. Endocrino.l Metabol., 2007, 51(8),1207–1216
Santos S., Santos E., Gaztambide S., Salvador J., Diagnosis and differential diagnosis of Cushing’s syndrome, Endocrinol. Nutr., 2009, 56(2),71–84
Bolland M.J., Holdaway I.M., Berkeley J.E., Lim S., Dransfield W.J., Conaglen J.V., et al., Mortality and Morbidity in Cushing’s syndrome in New Zeland, Clin. Endocrinol. (Oxf.), 2011, May 24.doi 10.1111/j.1365-2265.,2011,014124
Chanson P., Salenave S., Metabolic syndrome in Cushing’s syndrome. Neuroendocrinology., 2010, 92Suppl 1:96–101
Szappanos A., Toke J., Lippai D., Patocs A., Szucs N., Futo L., et al., Bone turnover in patients with endogenous Cushing’s syndrome before and after succesful treatment., Osteoporos. Int., 2010, 21(4):637–645
Bednarek-Tupikowska G., Kubicka E., Hypercortisolism in pregnancy, Pol. Merkur Lekarski. 2006, 20(116),232–235
Peris P., Guanabens N., Martinez de Osaba M.J., Monegal A., Alvarez L., Pons F., et al., Clinical characteristics and etiologic factors of premenopausal osteoporosis in a group of Spanish women, Semin. Arthritis. Rheum. 2002, 32(1),64–70
Randazzo M.E., Grossrubatscher E., Dalino Ciaramella P., Vanzulli A., Lili P., Spontaneous recovery of bone mass after cure of endogenous hypercortisolism, Pituitary, 2011 Apr 8. DOI: 10.1007/ s11102-011-0306-3
Takeshita M., Mizuno Y., Effect of glucose and bone metabolism in endocrinal disorder, Clin. Calcium., 2009, 19(9),1282–1290
Smets P., Meyer E., Maddens B., Daminet S., Cushing’s syndrome, glucocorticoids and the kidney, Gen. Comp. Endocrinol., 2010, 169(1),1–10.
Bruno O.D., Juarez-Allen L., Rossi M.A., Longobardi V., In what clinical settings should Cushing’s syndrome be suspected?, Medicina (B. Aires). 2009, 69(6),674–680
Kalantaridou S.N., Makrigiannakis A., Zoumakis E., Chrousos G.P., Stress and female reproductive system, J. Reprod. Immunol., 2004, 62(1–2),61–68
Sonino N., Fallo F., Fava G.A., Psychosomatic aspects of Cushing’s syndrome, Rev. Endocr. Metab. disord., 2010, 11(2),95–104
Wolkowitz O.M., Burke H., Epel E.S., Reus V.I., Glucocorticoids. Mood, memory, and mechanisms, Ann. N Y Acad. Sci., 2009, 1179,19–40
Manetti L., Bogazzi F., Giovannetti C., Raffaelli V., Genovesi M., Pellegrini G., et al., Changes in coagulation indexes and occurence of venous thromboembolism in patients with Cushing’s syndrome: result from a prospective study before and after surgery, Eur. J. Endocrinol., 2010, 163(5),783–1891
Van Zaane B., Nur E., Sqizzato A., Dekkers O.M., Twickler M.T., Fliers E., et al., Hypercoagulable state in Cushing’s syndrome: a systematic review, J. Clin. Endocrinol. Metab., 2009, 94(8),2743–2750
Erem C., Nuhoglu I., Yilmaz M., Kocak M., Demirel A., Ucuncu O., et al., Blood coagulation and fibrinolysis in patient 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),169–1674
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
Bertherat J., Bilaud L., Guilhaume B., Cushing’s syndrome and adrenal insuffiency in pregnancy, Ann. Endocrinol. (Paris), 2002, Oct,63(5),452–456
Delibasi T., Ustun I., Aydin Y., Berker D., Erol H.K., Gul K., et al., Early severe pre-eclamptic findings in a patient with Cushing’s syndrome. Gynecol. Endocrinol., 2006, 22(12),710–712
Lindsay J.R., Nieman L.K., The hypotalamic-pituitary-adrenal axis in pregnancy: challenges in disease detection and treatment, Endoc. Rev., 2005, 26(6),775–799
Vilar L., Freitas Mda C., Lima L.H., Lyra R., Kater C.E., Cushing’s syndrome in pregnancy: an overview, Arq. Bras. Endocrinol. Metabol., 2007, 51(8),1293–1302
Bednarek-Tupikowska G., Kubicka E., Hypercortisolism in pregnancy, Pol. Merkur Lekarski, 2006, 20(116),232–235
Pollack R.P., Brett E.M., Adrenocorticotropic hormone-independent Cushing’s syndrome manifesting during pregnancy, Endocr. Pract., 2010, 16(2),260–263
Terhune K.P., Jagasia S., Blevins L.S.Jr., Phay J.E., Diagnostic and therapeutic dilemmas of hypercortisolemia during pregnancy: a case report. Am. surg., 2009, 75(3),232–234
Lo C.Y., Lo C.M., Lam K.Y., Cushing’s syndrome secondary to adrenal adenoma during pregnancy, Surg Endosc., 2002,16(1),219–220
Kim Hg, Lee K.H., Je G.H., Han M.S., A case of Cushing’s syndrome in pregnancy secondary to an adrenal cortical adenoma, J. Korean Med. Sci., 2003, 18(3),444–446
Choi WJ, Jung TS, Paik WY. Cushing’s syndrome in pregnancy with a severe maternal complication: a case report. J Obstet Gynaecol Res. 2011,Feb;37(2):163–167
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 Jun 13. [Epub ahead of print] PubMed PMID: 22698457
Holgado-Galicia MV, Magno JD, Acelajado-Valdenor C, Isip-Tan IT, Lim-Abrahan MA. Cushing’s syndrome in pregnancy. BMJ Case Rep. 2011 Apr 26;2011. pii: bcr0120113720. doi: 10.1136/bcr.01.2011.3720. PubMed PMID: 22696666
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 Mar;40(3):e1–4
Kotteas E, Ioachim E, Pavlidis N. A pregnant patient with adrenocortical carcinoma: case report. Onkologie. 2012;35(9):517–951
Lubin V, Gautier JF, Antoine JM, Beressi JP, Vexiau P. Cushing’s syndrome during pregnancy. Presse Med. 2002; 31(36):1706–1713
Author information
Authors and Affiliations
Corresponding author
About this article
Cite this article
Andjelkovic, M.V., Mladenovic, V.S., Djukic, A.L. et al. Cushing’s syndrome diagnosed after delivery: a case report. cent.eur.j.med 8, 674–678 (2013). https://doi.org/10.2478/s11536-012-0128-3
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.2478/s11536-012-0128-3