CASE REPORT Bilateral Laparoscopic Adrenalectomy in a Pregnant Woman with Cushing’s Syndrome Mohammad Aslzare,1 Mohammad Alipour,2 Morteza Taghavi,3 Alireza Ghoreifi4* Keywords: adrenal cortex neoplasms; adrenalectomy; Cushing syndrome; pregnancy; laparoscopy. INTRODUCTION Cushing’s syndrome (CS) occurs rarely during pregnancy.(1) Surgical treatment is the principal therapy for CS in preg- nancy, with medical treatments constituting the second choice. The most common cause of CS in pregnancy is adrenal gland adenoma that may be treated by unilateral adrenalectomy during gestation.(2,3) Herein we present a 31 years old pregnant woman with CS who underwent bilateral laparoscopic adrenalectomy in her 18th week of pregnancy. Our Medline search revealed that this is the first “bilateral” laparoscopic adrenalectomy during pregnancy which has been reported. CASE REPORT A 31-year old primigravid woman was referred to our clinic at 4 months gestation following the diagnosis of CS from about three months ago. She has been treated medically but her blood pressure was not well controlled. The patient also had a history of cardiac ablation because of paroxysmal supraventricular tachycardia about 4 months ago when she was unaware of her pregnancy. Her laboratory data showed urine free cortisol level of 730 µgr/24hr (normal range, 10-100) and her late-night salivary cortisol level was 60 nmol/L (normal range, 6.2-19.4). Ultrasonography showed increased size of adrenals with an 18 weeks normal fetus (Figure 1). Abdominal magnetic resonance imaging (MRI) showed adrenal hyperplasia (Figure 2) but brain MRI was normal. We decided to perform bilateral laparoscopic adrenalectomy. Laparoscopy was done under general anesthesia with anterior transperitoneal approach using three working trocars. After extraction of left ad- renal gland (Figure 3) the position was changed to right lateral decubitus and right adrenal gland was similarly excised successfully (Figure 4). The time of surgery was about 4.5 hours. Post-operative view of the abdomen is shown in Figure 5. The operation was uneventful and the patient and fetus were well during post-operative period. The patient was discharged after 4 days with oral medications. After surgery baseline serum cortisol level decreased from 60 to 2.8 nmol/L, and 24-hour urine cortisol decreased from 730 to 29 µg. After 3 months the patient underwent cesarean section because of fetoplacental abnormality. A preterm infant with intrauterine growth retardation (IUGR) without any malformation was born. The patient was discharged without any complication but her infant was remained under observation in neonatal intensive care unit (NICU) for Figure 1. Ultrasonography of the patient. Figure 2. Magnetic resonance imaging of the patient. Departments of Urology1, Anesthesiology2 and Endocrinology3, Ghaem Hospital, Mashhad University of Medical Sciences, Mashhad, Iran. 4 Department of Urology, Imam Reza Hospital, Mashhad University of Medical Sciences, Mashhad, Iran. * Correspondence: Imam Reza Hospital, Mashhad University of Medical Sciences, Mashhad, Iran. Tel: +98 511 8022553. E-mail: aghoreifi@yahoo.com. Received February 2014 & Accepted June 2014 Vol 11. No 05 Sept-Oct 2014 1911 40 days. The baby had good condition after discharge and remained normal during his two years follow up. DISCUSSION Pregnancy is rare in women with CS, with fewer than 150 cases reported in the literature. It is because that the hyperandrogenism and hypercortisolism status during pregnancy suppress pituitary secretion of gonadotropins. (4,5) However, because CS results in increased fetal and maternal complications, its early diagnosis and treatment are critical.(1,2) The etiology of CS in pregnant women is different from that in of non-pregnant women. Adrenal adenomas cause approximately 40-50% and 17-29% of CS in pregnant and non-pregnant women, respectively. (4,6,7) In contrast, Cushing disease is less common in preg- nancy; with rates of 63-72% in the general population compared with 33% in pregnant women.(4,6) The etiology of CS in our case was bilateral adrenal hyperplasia. There are many overlapping features between normal pregnancy and CS, so the clinical diagnosis of CS in preg- nancy may be difficult and unfortunately is often not de- tected until 12-26 weeks of gestation.(4,6,7) The biochem- ical diagnosis of CS in pregnancy is difficult because of the normal hypercortisolism during pregnancy.(6) Adrenal ultrasonography or MRI can be safely performed during pregnancy for detection of adrenal tumor. MRI can also be useful in locating pituitary tumors.(3) When CS is diagnosed during pregnancy, therapeutic options depend on the underlying etiology, including, surgical treatment, conservative management, medi- cal treatment, and delay of surgery until after delivery. (5) When contemplating surgical treatment for a pregnant patient with suspected adrenocortical adenoma, the surgi- cal approach and the optimal time for surgery need to be determined. Surgery is the treatment of choice for CS in pregnancy, except perhaps late in the third trimester, with medical treatment being a second choice. There is no ra- tionale for supportive treatment alone.(4) The commonest cause of CS in pregnancy, adrenal gland adenoma, may be treated by unilateral adrenalectomy during gestation. Open or laparoscopic methods have been performed in practice. The end of the first trimester and the first half of the second trimester are considered the best time for surgery. In the third trimester, conservative treatment and early delivery are preferred. However Aishima and colleagues and Sammoura and colleagues successfully treated their patients with CS at this gestational stage by retroperitoneal laparoscopic adrenalectomy.(5,8) We found 24 cases of adrenalectomy in literature due to CS caused by adrenocortical adenoma in pregnancy, but we didn’t find a case of bilateral laparoscopic adrenalectomy in a pregnant woman. In our case, laparoscopy was done by transperitoneal approach. Although the patient was obese with a history of severe hypertension and cardiac abla- tion, she didn’t have any complication perioperatively. CONCLUSION We demonstrated that bilateral laparoscopic adrenalecto- my is possible during pregnancy and may be considered safe and minimally invasive in selected patients. CONFLICT OF INTEREST None declared. REFERENCES 1. Kita M, Sakalidou M, Saratzis A, Ioannis S, Avramidis A. Cushing’s syndrome in pregnan- Figure 3. Laparoscopic views. Left: The pregnant uterus. Right: The adrenal during excision. Figure 5. Abdomen at the end of surgery. Figure 4. Adrenal specimens after excision. Bilateral Laparoscopic Adrenalectomy in Pregnancy-Aslzare et al Case Report 1912 cy: report of a case and review of the literature. Hormones (Athens). 2007;6:242-6. 2. 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;90:3077-83. 3. Bednarek-Tupikowska G, Kubicka E, Sicińs- ka-Werner T, et al. A case of Cushing’s syn- drome in pregnancy. Endokrynol Pol. 2011;62:181-5. 4. Vilar L, Freitas Mda C, Lima LH, Lyra R, Kater CE. Cushing’s syndrome in pregnancy: an over- view. Arq Bras Endocrinol Metabol. 2007;51:1293-302. 5. Sammour RN, Saiegh L, Matter I, et al. Adre- nalectomy for adrenocortical adenoma causing Cushing’s syndrome in pregnancy: a case report and review of literature. Eur J Obstet Gynecol Reprod Biol. 2012;165:1-7. 6. Lindsay JR, Nieman LK. The hypothalamic- pituitary-adrenal axis in pregnancy: challenges- in disease detection and treatment. Endocr Rev. 2005;26:775-99. 7. Buescher MA, McClamrock HD, Adashi EY. Cushing’s syndrome in pregnancy. Obstet Gy- necol. 1992;79:130-7. 8. Aishima M, Tanaka M, Haraoka M, Naito S. Retroperitoneal laparoscopic adrenalectomy in a pregnant woman with Cushing’s syndrome. J Urol. 2000;164:770-1. Bilateral Laparoscopic Adrenalectomy in Pregnancy-Aslzare et al Vol 11. No 05 Sept-Oct 2014 1913