Pregnancy-induced Cushing’s syndrome with an adrenocortical adenoma overexpressing LH/hCG receptors: a case report

Author:

Li Shaohua,Yang Chen,Fan Jing,Yao Yao,Lv Xiaomei,Guo Ying,Zhang Shaoling

Abstract

Abstract Background Pregnancy-induced Cushing’s syndrome (CS) with an adrenocortical adenoma overexpressing luteinizing hormone (LH)/human choriogonadotropin (hCG) receptors (LHCGR) has been rarely reported in the literatures. This peculiar condition challenges the canonical diagnosis and management of CS. Case presentation A 27-year-old woman (G2P0A1) presented at 20 weeks gestational age (GA) with overt Cushingoid clinical features. Adrenocorticotropic hormone (ACTH)-independent CS was diagnosed based on undetectable ACTH and unsuppressed cortisol levels by dexamethasone. Magnetic resonance imaging (MRI) scanning without contrast revealed a left adrenal nodule while pituitary MRI scanning was normal. A conservative treatment strategy of controlling Cushingoid comorbidities was conducted. At 36 weeks GA, a caesarean operation was performed and a live female infant was delivered. At 8 weeks after parturition, our patient achieved normalization of blood pressure, blood glucose, serum potassium, and urinary cortisol level spontaneously. During non-pregnancy period, stimulation testing with exogenous hCG significantly evoked a cortisol increase. The woman underwent resection of the adrenal tumor at 6 months after parturition. Immunohistochemistry (IHC) showed the tumor tissue that stained positive for luteinizing hormone (LH)/human choriogonadotropin (hCG) receptor (LHCGR), whereas negative for both melanocortin 2 receptor (MC2R) and G protein-coupled receptor-1 (GPER-1). Conclusions Stimulation test with exogenous hCG after parturition is necessary for the diagnosis of pregnancy-induced CS. LHCGR plays an essential role in the pathogenesis of this rare condition.

Funder

National Natural Science Foundation of China

Natural Science Foundation of Guangdong Province

Publisher

Springer Science and Business Media LLC

Subject

General Medicine,Endocrinology, Diabetes and Metabolism

Reference25 articles.

1. Wieland RG, Shaffer MB Jr, Glove RP. Cushing's syndrome complicating pregnancy. A case report. Obstet Gynecol. 1971;38(6):841–3.

2. Reschini E, Giustina G, Crosignani PG, D'Alberton A. Spontaneous remission of Cushing syndrome after termination of pregnancy. Obstet Gynecol. 1978;51(5):598–602.

3. Aron DC, Schnall AM, Sheeler LR. Spontaneous resolution of Cushing's syndrome after pregnancy. Am J Obstet Gynecol. 1990;162(2):472–4.

4. Close CF, Mann MC, Watts JF, Taylor KG. ACTH-independent Cushing's syndrome in pregnancy with spontaneous resolution after delivery: control of the hypercortisolism with metyrapone. Clin Endocrinol. 1993;39(3):375–9.

5. Caticha O, Odell WD, Wilson DE, Dowdell LA, Noth RH, Swislocki AL, Lamothe JJ, Barrow R. Estradiol stimulates cortisol production by adrenal cells in estrogen-dependent primary adrenocortical nodular dysplasia. J Clin Endocrinol Metab. 1993;77(2):494–7.

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