Endocrinology Research Centre, Moscow, Russia.
I.M. Sechenov First Moscow State Medical University of the Ministry of Health of the Russian Federation (Sechenov University), Moscow, Russia.
Osteoporos Int. 2021 Jun;32(6):1227-1231. doi: 10.1007/s00198-020-05758-6. Epub 2020 Nov 27.
We report a case of a young male patient with clinical signs of dyskeratosis congenita who presented with multiple bilateral low-traumatic hip fractures. Whole exome sequencing (WES) showed a previously unreported mutation in the poly(A)-specific ribonuclease (PARN) gene. Zoledronic acid 5 mg over 3 years was effective at preventing further fractures. A male patient was referred to our clinic at age 24 due to multiple bilateral hip fractures. At the time of admission, the patient's height was 160 cm and weight 40 kg; bone mineral density (BMD) at the lumbar spine was normal (L1-L4 0.0 Z-score). The patient was found to have abnormal skin pigmentation, hyperkeratosis of palms and soles, nail dystrophy, and signs of bone marrow failure (BMF). Bone fragility first presented at 5 years old with a wrist fracture, followed by multiple bilateral low-traumatic hip fractures without falls from 14 to 24 years. WES showed a previously unreported mutation (NM_002582.3: c.1652delA; p.His551fs) in the poly(A)-specific ribonuclease (PARN) gene. Flow fish telomere measurement result was 5.9 (reference range 8.0-12.6), which is consistent with the DC diagnosis. Permanent fixation with internal metal rods and zoledronic acid 5 mg over 3 years was effective at preventing further fractures over 4 years of follow-up. Additionally, BMF did not progress over 4 years of observation. DC associated with PARN gene mutations might predispose to low-traumatic multiple hip fractures in adolescents and young adults. Treatment with zoledronic acid in this case was effective and safe at preventing further fractures.
我们报告了一例具有先天性角化不良临床特征的年轻男性患者,他表现为多发性双侧低创伤性髋部骨折。全外显子组测序(WES)显示,在多聚(A)特异性核糖核酸酶(PARN)基因中存在先前未报道的突变。唑来膦酸 5mg 治疗 3 年可有效预防进一步骨折。一名 24 岁男性因多发性双侧髋部骨折就诊于我院。入院时,患者身高 160cm,体重 40kg;腰椎骨密度(BMD)正常(L1-L4 0.0 Z 评分)。患者存在皮肤色素异常、手掌和足底过度角化、指甲营养不良和骨髓衰竭(BMF)迹象。脆性首先在 5 岁时表现为腕部骨折,随后在 14 至 24 岁期间无跌倒发生多发性双侧低创伤性髋部骨折。WES 显示在多聚(A)特异性核糖核酸酶(PARN)基因中存在先前未报道的突变(NM_002582.3:c.1652delA;p.His551fs)。流式鱼端粒测量结果为 5.9(参考范围 8.0-12.6),与 DC 诊断相符。4 年随访期间,永久性内金属棒固定和唑来膦酸 5mg 治疗 3 年可有效预防进一步骨折。此外,4 年内 BMF 无进展。PARN 基因突变相关的 DC 可能导致青少年和年轻成人低创伤性多发性髋部骨折。在本例中,唑来膦酸治疗可有效且安全地预防进一步骨折。