Santbergen Bart, van der Heul Cees
Department of Internal Medicine, St. Elisabeth Hospital, Tilburg, The Netherlands.
BMJ Case Rep. 2014 Mar 5;2014:bcr2014203701. doi: 10.1136/bcr-2014-203701.
We describe two patients, a father and a son, presenting with erythrocytosis. Evaluation showed no pulmonal or cardial disorders. Owing to an elevated erythropoietin level after phlebotomy, a physiological secondary polycythaemia was suspected. A haemoglobin electrophoresis showed that our patients have a haemoglobinopathy with high affinity for oxygen, called Hb-Malmö (exon 3: c.294 C>G p.His98Gln). Hb-Malmö is a congenital disorder located on a gene at chromosome 11, in the B-chain on codon 97, decoding the α-subunit and β-subunit of the haemoglobin. Through a mutation (CAC→CAG), histidine is replaced by glutamine. The mutation causes a disorder in the connection between the α1-subunit and β2-subunit of the haemoglobin structure. These connections are important sites for binding oxygen. Mutated haemoglobin has a preference for an oxygenated status, which implicates that there is an increased binding and decreased release of oxygen. To compensate, there will be an erythrocytosis to transport sufficient oxygen to the peripheral tissues.
我们描述了两名患有红细胞增多症的患者,分别为一对父子。评估显示他们没有肺部或心脏疾病。由于放血后促红细胞生成素水平升高,怀疑是生理性继发性红细胞增多症。血红蛋白电泳显示,我们的患者患有对氧具有高亲和力的血红蛋白病,称为Hb-马尔默(外显子3:c.294 C>G,p.His98Gln)。Hb-马尔默是一种先天性疾病,位于11号染色体上的一个基因上,在密码子97的B链上,负责解码血红蛋白的α亚基和β亚基。通过一个突变(CAC→CAG),组氨酸被谷氨酰胺取代。该突变导致血红蛋白结构中α1亚基和β2亚基之间的连接出现紊乱。这些连接是结合氧气的重要位点。突变的血红蛋白更倾向于氧合状态,这意味着氧气的结合增加而释放减少。为了进行补偿,将会出现红细胞增多症,以便将足够的氧气输送到外周组织。