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