Davis P B, Dieckman L, Boat T F, Stern R C, Doershuk C F
J Clin Invest. 1983 Jun;71(6):1787-95. doi: 10.1172/jci110934.
Intact lymphocytes from patients with cystic fibrosis (CF) produce significantly (P less than 0.001) less adenosine 3':5' cyclic monophosphate (cAMP) than normal lymphocytes in response to isoproterenol (10(-8)-10(-4) M), although the basal cAMP content and the response to prostaglandin E1 are normal. Obligate heterozygotes for CF have significantly (P less than 0.005) reduced cAMP response to isoproterenol as well, suggesting a genetic component in the beta adrenergic deficiency in CF. The number of beta adrenergic receptors, as determined by equilibrium binding of [3H]dihydroalprenolol to lymphocyte particulates, is the same in normal lymphocytes (969 +/- 165 receptors/cell) and lymphocytes from patients with CF (1,333 +/- 263 receptors/cell). Binding properties of the receptor for both antagonist and agonist, as assessed by KD for dihydroalprenolol and Ki for (-)-isoproterenol, are also normal in the CF lymphocytes. Similarly, in granulocytes from patients with CF, the cAMP response to isoproterenol (10(-8)-10(-4) M) is significantly reduced compared with healthy controls (P less than 0.03), as is the response of granulocytes from obligate heterozygotes (P less than 0.05). Again, the basal cAMP levels and the response to prostaglandin E1 are normal. The number of beta adrenergic receptors, as determined by equilibrium binding of [3H]dihydroalprenolol to granulocyte particulates, was the same in normal (1,462 +/- 249 receptors/cell) and CF (1,621 +/- 221 receptors/cell) preparations. Binding properties of the receptor for both agonist and antagonist, as assessed by KD for dihydroalprenolol and Ki for isoproterenol, are normal in CF granulocyte particulates. The lymphocyte and granulocyte beta adrenergic defect in CF cannot be explained by abnormalities of the beta adrenergic receptor or of adenylate cyclase itself. Receptor-cyclase coupling is the most likely site of the heritable beta adrenergic defect in CF.
囊性纤维化(CF)患者的完整淋巴细胞在对异丙肾上腺素(10⁻⁸ - 10⁻⁴M)产生反应时,产生的3':5'环磷酸腺苷(cAMP)明显(P小于0.001)少于正常淋巴细胞,尽管基础cAMP含量以及对前列腺素E1的反应是正常的。CF的 obligate 杂合子对异丙肾上腺素的cAMP反应也明显(P小于0.005)降低,这表明CF中β肾上腺素能缺陷存在遗传因素。通过[³H]二氢阿普洛尔与淋巴细胞颗粒的平衡结合测定的β肾上腺素能受体数量,在正常淋巴细胞(969 ± 165个受体/细胞)和CF患者的淋巴细胞(1333 ± 263个受体/细胞)中是相同的。通过二氢阿普洛尔的KD和( - ) - 异丙肾上腺素的Ki评估的受体对拮抗剂和激动剂的结合特性,在CF淋巴细胞中也是正常的。同样,在CF患者的粒细胞中,与健康对照相比,对异丙肾上腺素(10⁻⁸ - 10⁻⁴M)的cAMP反应明显降低(P小于0.03),CF的 obligate 杂合子的粒细胞反应也是如此(P小于0.05)。基础cAMP水平以及对前列腺素E1的反应再次正常。通过[³H]二氢阿普洛尔与粒细胞颗粒的平衡结合测定的β肾上腺素能受体数量,在正常(1462 ± 249个受体/细胞)和CF(1621 ± 221个受体/细胞)制剂中是相同的。通过二氢阿普洛尔的KD和异丙肾上腺素的Ki评估的受体对激动剂和拮抗剂的结合特性,在CF粒细胞颗粒中是正常的。CF中淋巴细胞和粒细胞的β肾上腺素能缺陷不能用β肾上腺素能受体或腺苷酸环化酶本身的异常来解释。受体 - 环化酶偶联是CF中遗传性β肾上腺素能缺陷最可能的位点。