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一例诺南综合征样疾病伴生长期毛发松动的病例报告,经重组人生长激素治疗。

A case report of Noonan syndrome-like disorder with loose anagen hair 2 treated with recombinant human growth hormone.

机构信息

Growth, Development, and Mental health of Children and Adolescence Center, Children's Hospital of Chongqing Medical University, Chongqing, China.

Chongqing Key Laboratory of Child Health and Nutrition, Chongqing, China.

出版信息

Am J Med Genet A. 2020 Aug;182(8):1967-1971. doi: 10.1002/ajmg.a.61638. Epub 2020 Jun 1.

Abstract

Protein phosphatase 1 catalytic subunit beta (PPP1CB) is a disease-causing gene of Noonan-like syndrome, which acts via the RAS/MAPK pathway. To date, only 17 patients diagnosed with PPP1CB-related Noonan-like syndrome have been reported around the world, with few reports in Asia. Twelve reported patients are of short stature and only one patient was treated with growth hormone (GH); however, follow-up data is lacking. To the best of our knowledge, this is the first reported patient with complete recombinant human growth hormone (rhGH) treatment follow-up data; the patient has a de novo c.146C>G (p.Pro49Arg) mutation in the PPP1CB gene. The hair pattern of the patient (coarse, curly, slow growing, and fragile) combined with Noonan dysmorphic features, developmental delay, and congenital heart disease, are highly consistent with the typical features observed in Noonan syndrome-like disorder with loose anagen hair 2 (NSLH2). rhGH treatment, administered for 3 years and 8 months, promoted the patient's linear growth. Our findings expand the data regarding the treatment of short stature in patients with NSLH2 caused by PPP1CB mutation. Clinical manifestation, growth and development process, and rhGH therapy effect data will aid in future revision of the relevant diagnosis and treatment guidelines.

摘要

蛋白磷酸酶 1 催化亚基 β(PPP1CB)是类诺南综合征的致病基因,通过 RAS/MAPK 途径发挥作用。迄今为止,全世界仅报道了 17 例诊断为 PPP1CB 相关类诺南综合征的患者,亚洲报道的病例较少。12 例报告患者身材矮小,仅 1 例患者接受生长激素(GH)治疗;然而,缺乏随访数据。据我们所知,这是首例报道的接受完整重组人生长激素(rhGH)治疗随访数据的患者;该患者在 PPP1CB 基因中存在新发生的 c.146C>G(p.Pro49Arg)突变。患者的毛发模式(粗糙、卷曲、生长缓慢且脆弱)结合诺南畸形特征、发育迟缓以及先天性心脏病,与具有稀疏生长期毛发 2(NSLH2)的类诺南综合征高度一致。rhGH 治疗 3 年 8 个月后,促进了患者的线性生长。我们的发现扩展了 PPP1CB 突变引起的 NSLH2 患者矮小症治疗的数据。临床表现、生长发育过程和 rhGH 治疗效果数据将有助于未来修订相关的诊断和治疗指南。

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