Center for Oral Health in Rare Conditions, Aarhus University Hospital, Aarhus, Denmark.
Am J Med Genet A. 2011 Nov;155A(11):2654-60. doi: 10.1002/ajmg.a.34242. Epub 2011 Sep 30.
Hypophosphatemic rickets (HR) are diseases characterized by deficient mineralization of bone due to abnormal renal wasting of phosphate. Deformation of bony structures of cartilaginous origin has been described as a major characteristic in patients with HR, but little is known about the impact on bony structures of intramembranous origin. The aim of the present study was to describe the osseous morphology of the craniofacial structures in patients with HR compared to healthy controls, and to investigate the impact of different bone origin on the osseous morphology. Fifty-three patients with HR (17 males, 36 females), aged 3-74 yrs, were included. Fifty HR patients had dominant X-linked disease, and in three patients no mutations were identified. A total of 79 healthy individuals (37 males, 42 females), aged 6-79 yrs, with normal occlusion served as controls. Significant cephalometric differences were found between HR patients and controls. In HR patients, the cranial base was flattened and the depth of the posterior cranial fossa was decreased. The anterior height of the cranium, the angle nasion-sella-frontale, and the thickness of theca were increased. The length of the nasal bone and the height of the maxilla were reduced. In contrast, the vertical as well as the sagittal relation between the jaws were unaffected in HR patients compared to controls. In conclusion, we found that the cranial structures of cartilaginous origin as well as the structures of intramembraneous origin were affected in patients with HR.
低磷血症性佝偻病(HR)是一种由于肾脏对磷酸盐的异常排泄而导致骨骼矿化不足的疾病。骨骼结构的变形已被描述为 HR 患者的主要特征,但对于膜内起源的骨骼结构的影响知之甚少。本研究的目的是描述与健康对照组相比 HR 患者的颅面结构的骨骼形态,并探讨不同骨骼起源对骨骼形态的影响。共纳入 53 例 HR 患者(男 17 例,女 36 例),年龄 3-74 岁。53 例 HR 患者中,有 50 例为 X 连锁显性疾病,3 例患者未发现突变。共有 79 名年龄在 6-79 岁、具有正常咬合的健康个体(男 37 名,女 42 名)作为对照组。HR 患者与对照组之间存在显著的头影测量差异。在 HR 患者中,颅底扁平,后颅窝深度减小。颅前高、鼻根蝶鞍额点角和颅腔厚度增加。鼻骨长度和上颌高度减小。相比之下,HR 患者的颌骨垂直和矢状关系与对照组相比无明显差异。总之,我们发现 HR 患者的软骨起源的颅面结构和膜内起源的结构均受到影响。