Simon Sebastian, Resch Heinrich
Department of Orthopedics and Trauma Surgery, Vienna General Hospital, Medical University of Vienna, Vienna, Austria.
St. Vincent Hospital-2nd Medical Department, The VINFORCE Study Group, Academic Teaching Hospital of the Medical University of Vienna, Vienna, Austria.
Wien Med Wochenschr. 2020 Apr;170(5-6):112-115. doi: 10.1007/s10354-020-00736-3. Epub 2020 Feb 18.
Hypophosphatasia (HPP) is a rare disorder with perinatal, infantile, childhood, and adult presentations. Severe forms are autosomal recessive with an early onset, whereas milder forms have a later onset. The underlying cause of the disease is a mutation based on a genetic disorder of the tissue non-specific alkaline phosphatase (TNSALP) gene, leading on the one hand to decreased activity of the TNSALP enzyme, and on the other hand to accumulation of TNSALP substrates. Symptoms like non-traumatic and non-healing fractures, musculoskeletal pain, chondrocalcinosis, seizures, premature loss of fully rooted teeth or delayed development of milk teeth, respiratory insufficiency, and calcinosis in muscles, kidneys, and joints occur. Supportive treatment is important for HPP patients, including mechanical ventilation, accurate fracture treatment, physical therapy, dental monitoring, and follow-up care to avoid subsequent problems. A causal enzyme therapy replacement with asfotase-alfa was approved by the Food and Drug Administration (FDA) in 2015. Asfotase-alfa improves respiratory insufficiency, bone mineralization, and long-term survival, and has a very good safety profile.
低磷性骨软化症(HPP)是一种罕见的疾病,有围产期、婴儿期、儿童期和成人期表现。严重形式为常染色体隐性遗传且发病早,而较轻形式发病较晚。该疾病的根本原因是基于组织非特异性碱性磷酸酶(TNSALP)基因的遗传紊乱发生的突变,一方面导致TNSALP酶活性降低,另一方面导致TNSALP底物蓄积。会出现非创伤性且不愈合的骨折、肌肉骨骼疼痛、软骨钙质沉着、癫痫发作、恒牙过早脱落或乳牙萌出延迟、呼吸功能不全以及肌肉、肾脏和关节钙化等症状。支持性治疗对HPP患者很重要,包括机械通气、准确的骨折治疗、物理治疗、牙科监测以及后续护理以避免出现后续问题。2015年,食品药品监督管理局(FDA)批准了用阿法骨化醇进行因果性酶替代治疗。阿法骨化醇可改善呼吸功能不全、骨矿化和长期生存率,并且安全性非常好。