Rhumatologie, Hôpital Henri Mondor, 51 Avenue du Maréchal de Lattre de Tassigny, 94010, Créteil, France.
Centre d'investigation clinique, Hôpital Henri Mondor, 51 Avenue du Maréchal de Lattre de Tassigny, 94010, Créteil, France.
Calcif Tissue Int. 2021 Jun;108(6):738-745. doi: 10.1007/s00223-021-00807-6. Epub 2021 Feb 8.
There is an increased risk of osteoporosis and an abnormal bone turn over in neurofibromatosis 1 (NF1). Our objective is to evaluate bone status in NF1 and to look for associations with cutaneous phenotype. We conducted a descriptive, monocentric study. We included 60 NF1 women, 18-51 years old, non-menopausal, divided in 2 groups: «at risk phenotype» (ARP) composed by 30 patients with at least 2 subcutaneous neurofibromas (SC-NF) and «classical phenotype» (CP) composed by 30 patients with none or 1 SC-NF. We evaluated low bone mineral density (BMD) risk factors and measured BMD, calcium and phosphorus homeostasis and bone turnover markers. Before 50 years old, Z-score has to be used to assess BMD. Z-score < - 2 is below expected range and represents 2.5% of the population. There was no difference between the two groups. Overall, Z-scores were low and 5 patients had a Z-score < - 2 (8.3%), which is 3 times general population low BMD frequency. 10 fragility fractures occurred in 8 patients, among which 2 were vertebral fractures. 85% had low calcium intake. 12 patients had hypophosphoremia, 25 elevated PTH. Vitamin D levels were low for 86.4%. 41 patients (69.5%) had at least one abnormal bone turnover markers. Low BMD is 3.3 times more frequent in NF1 than in general population, with high fracture risk, regardless of the skin phenotype, classical or at risk, because of high bone turn over and secondary hyperparathyroidism due to vitamin D deficiency and poor calcium intake.
神经纤维瘤病 1 型(NF1)患者存在骨质疏松症风险增加和骨代谢异常。本研究旨在评估 NF1 患者的骨状况,并寻找与皮肤表型的相关性。这是一项描述性、单中心研究。我们纳入了 60 名 NF1 女性患者,年龄 18-51 岁,均为绝经前,分为两组:“高危表型”(ARP)组 30 例,至少有 2 个皮下神经纤维瘤(SC-NF);“经典表型”(CP)组 30 例,无或仅有 1 个 SC-NF。我们评估了低骨密度(BMD)的危险因素,并测量了 BMD、钙磷代谢和骨转换标志物。在 50 岁之前,需要使用 Z 评分来评估 BMD。Z 评分 < -2 表示低于预期范围,代表 2.5%的人群。两组间无差异。总体而言,Z 评分较低,5 名患者的 Z 评分 < -2(8.3%),这是一般人群中低 BMD 频率的 3 倍。8 名患者中有 10 例脆性骨折,其中 2 例为椎体骨折。85%的患者钙摄入量低。12 例患者存在低磷血症,25 例患者甲状旁腺激素升高。86.4%的患者维生素 D 水平较低。41 名患者(69.5%)至少有一种骨转换标志物异常。NF1 患者的低 BMD 发生率是一般人群的 3.3 倍,骨折风险较高,与皮肤表型无关,无论是经典表型还是高危表型,这是由于骨转换增加和维生素 D 缺乏及钙摄入不足导致的继发性甲状旁腺功能亢进。