Adrenocortical oncocytoma presenting as Cushing´s syndrome in pregnancy with spontaneous postpartum uterine rupture
Authors:
M. Kotoulová 1; I. Mikysková 1; J. Dušková 2; J. Vláčil 1; M. Dvořák 1; M. Halaška 1
Authors‘ workplace:
Gynekologicko-porodnická klinika 1. LF UK a Nemocnice Na Bulovce, Praha, přednosta prof. MUDr. M. Halaška, DrSc.
1; Ústav patologie 1. LF UK a VFN, Praha, přednosta doc. MUDr. P. Dundr, Ph. D.
2
Published in:
Ceska Gynekol 2016; 81(3): 228-232
Overview
Objective:
The description of rare case of the Cushing’s syndrome in pregnancy resulting in the spontaneous rupture of uterus.
Designs:
A case report.
Setting:
Department of Gynecology and Obstetrics, 1st Medical Faculty of Charles University and Hospital Na Bulovce.
Case report:
The authors report the case of a 33 year old woman, who was admitted to the clinic in 30th week of pregnancy for elevated blood pressure and hypokalemia. Arterial hypertension was corrected with the combination of Vasocardin and Dopegyt. In 36th week of pregnancy the patient was admitted to the hospital with premature rupture of membranes. In less than sixteen hours the patient spontaneously gave birth to a healthy girl. Subsequently the patient suffered from abdominal pain. CT scan showed haemoperitoneum and a right adrenal mass and the surgical revision was performed. During the surgery uterine rupture was identified and the patient uderwent abdominal hysterectomy. Based on the endocrinological examination the diagnosis of Cushing’s syndrome was made. Three months after the delivery she underwent laparoscopic right adrenalectomy. Histological examination revealed adrenocortical oncocytoma.
Conclusion:
Cushing’s syndrome is rare in pregnancy and misdiagnosis is common. Symptoms mimic pregnancy complications. The disease is often diagnosed with delay even after delivery. The sonography and magnetic resonance are dominant imaging methods. Laboratory diagnosis is difficult due to pregnancy related changes. In spite of rarity of Cushing’s syndrome in pregnancy we should think about it in terms of the differential diagnosis of hypertension, diabetes, dysbalance of mineralogram and typical cushingoid habitus.
Keywords:
adrenocotical oncocytoma, Cushing’s syndrome in pregnancy, hypertension, uterine rupture
Sources
1. Češka, R., et al. Interna. První vyd. Praha: Triton, 2010, s. 327–329, 351–352.
2. Garla, V., Kheeten, R., Saleem, T. Cushing´s syndrome in pregnancy: A diagnostic conundrum. [online] West Virginia Med J, 2013, 109, p. 34–36.
3. Hamperl, H. Onkocytes and the so called Hurthle cell tumor. Arch Pathol, 1950, 49, p. 563–570.
4. Hána, V., Dokoupilová, M., Marek, J., et al. Reccurent ACTH-independent Cushing´s syndrome in multiple pregnancies and its treatment with metyrapone. [online] Clinl Endocrinol, 2001, 54, p. 277–281.
5. Hofmeyer, GS., Say, L., Gülmezoglu, AM. WHO systematic review of maternal mortality and morbidity: the prevalence of uterine rupture. [Online] BJOG, 2005, 112, p. 1221–1228.
6. Hunt, AB., McConahay, WM. Pregnancy associated with diseases of the adrenal glands. Am J Obstet Gynecol, 1953, 66(5), p. 970–987.
7. Chang, I., Cha, HH., Kim, JH., et al. Cushing syndrome in pregnancy secondary to adrenal adenoma. [online] Obstet Gynecol Sci, 2013, 56(6), p. 400–403.
8. Jaraith, A., Aulakh, BS. Adrenocortical carcinoma in pregnancy: A diagnostic dilemma. [online] Indian J Urol, 2014, 30(3), p. 342–344.
9. Kim, HG., Lee, HK., Je, GH. et al. A case of Cushing´s syndrome in pregnancy secondary to an adrenal cortical adenoma. [online] J Korean Med Sci, 2003, 18, p. 444–446.
10. Kita, M., Sakalidou, M., Saratzis, A., et al. Cushing´s syndrome in pregnancy: Report of a case and review of the literature. [online] Hormones, 2007, 6(3), p. 242–246.
11. Lee, SS., Baek, KH., Lee, YS., et al. Subclinical Cushing’s syndrome associated with an adrenocortical oncocytoma. [online] J Endocrinol Investigation, 2008, 31(7), p. 675–679.
12. Lekarev, O., New, MI. Adrenal disease in pregnancy. [online] Best Pract Res Clin Endocrinol Metab, 2011, 25, p. 959–973.
13. Lim, WH., et al. The medical mangement of Cushing´s syndrome in pregnancy. [online] Eur J Obstet Gynecol Reprod Biol, 2013, 168, p. 1–6.
14. Mavromatidis, G., Karavas, G., Margioula-Siarkou, C., et al. Spontaneous postpartum rupture of an intact uterus: a case report. [online] J Clin Med Res, 2015, 7(1), p. 56–58.
15. Pereira, BD., Rios, ES., Cabrera, RA., et al. Adrenocortical oncocytoma presenting as Cushing’s syndrome: An additional report of a paediatric case. [online] Endocr Pathol, 2014, 25(4), p. 397–403.
16. Sammour, RN., et al. Adrenalectomy for adrenocortical adenoma causing Cushing´s syndrome in pregnancy: a case report and review of literatue. [online] Eur J Obstet Gynecol Reprod Biol, 2012, 165, p. 1–7.
Labels
Paediatric gynaecology Gynaecology and obstetrics Reproduction medicineArticle was published in
Czech Gynaecology
2016 Issue 3
Most read in this issue
- Traumatic symphyseal rupture by vaginal delivery,report of a rare case
- The possibility of antepartal prevention of episiotomy and perineal tears during delivery
- Spontaneus delivery after two previous caesarean sections – case report
- Surgical treatment of endometriomas and ovarian reserve