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A Case of ACTH-Independent Macronodular Adrenal Hyperplasia: Simultaneous Expression of Several Aberrant Hormone Receptors in the Adrenal Gland

机译:一个不依赖ACTH的大结节性肾上腺皮质增生病例:肾上腺中几种异常激素受体的同时表达

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ACTH-independent macronodular adrenal hyperplasia (AIMAH) is a rare cause of Cushing's syndrome. Recently, aberrant expression of adrenal receptors for various hormones and/or cytokines has been identified in several cases with AIMAH, which may act as a pathogenetic factor for the disorder. We report here an AIMAH patient with a Rathke's cleft cyst. Endocrinological examinations revealed that the pituitary cyst had no hormonal secretion. Administrations of either AVP or isoproterenol provoked cortisol production in the patient, whereas DDAVP, mosapride or endogenous LH induced by GnRH did not. Reverse transcriptional-PCR analysis of total RNA obtained from the patient's adrenal tissue revealed the expression of mRNA of receptors for V1a, V1b, V2, and LH/hCG. Three of these receptors except for V1 a receptor were not expressed in normal adrenal tissue. Hyperosmolar saline infusion promoted the patient's cortisol secretion through the increase in endogenous AVP (peak plasma AVP level reached 90.4 pg/ml during the test). These results suggest that endogenous AVP and catecholamines are involved in the pathophysiology of the patient. Further study will be necessary to clarify the molecular mechanisms that regulate tissue-specific expression of these receptors and their role in the overgrowth of adrenal in AIMAH.
机译:独立于ACTH的大结节性肾上腺皮质增生(AIMAH)是库欣综合征的罕见原因。近来,在几种情况下,已经确认了与AIMAH有关的各种激素和/或细胞因子的肾上腺受体的异常表达,这可能是该疾病的致病因素。我们在这里报告一名有Rathke裂隙囊肿的AIMAH患者。内分泌检查发现垂体囊肿无激素分泌。 AVP或异丙肾上腺素的给药会引起患者皮质醇的产生,而GnRH诱导的DDAVP,mosapride或内源性LH则不会。从患者肾上腺组织获得的总RNA的逆转录PCR分析显示V1a,V1b,V2和LH / hCG受体的mRNA表达。在正常的肾上腺组织中除V1a受体外,这些受体中的三个均未表达。高渗盐水输注通过内源性AVP的增加来促进患者的皮质醇分泌(在测试过程中血浆AVP峰值达到90.4 pg / ml)。这些结果表明内源性AVP和儿茶酚胺参与了患者的病理生理。有必要进行进一步的研究以阐明调节这些受体的组织特异性表达及其在AIMAH中肾上腺过度生长中的作用的分子机制。

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