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2
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本文引用的文献

1
Treatment of Short Stature in Aggrecan-deficient Patients With Recombinant Human Growth Hormone: 1-Year Response.聚集蛋白聚糖缺乏症患者用重组人生长激素治疗身材矮小:1 年应答。
J Clin Endocrinol Metab. 2022 Apr 19;107(5):e2103-e2109. doi: 10.1210/clinem/dgab904.
2
GH Responsiveness in Children With Noonan Syndrome Compared to Turner Syndrome.与 Turner 综合征相比,Noonan 综合征患儿的 GH 反应性。
Front Endocrinol (Lausanne). 2021 Nov 9;12:737893. doi: 10.3389/fendo.2021.737893. eCollection 2021.
3
Novel PRMT7 mutation in a rare case of dysmorphism and intellectual disability.罕见的畸形和智力残疾病例中的新型PRMT7突变。
J Hum Genet. 2022 Jan;67(1):19-26. doi: 10.1038/s10038-021-00955-5. Epub 2021 Jul 9.
4
Diagnosis, Genetics, and Therapy of Short Stature in Children: A Growth Hormone Research Society International Perspective.儿童身材矮小的诊断、遗传学和治疗:生长激素研究学会国际视角。
Horm Res Paediatr. 2019;92(1):1-14. doi: 10.1159/000502231. Epub 2019 Sep 12.
5
Prenatal and postnatal presentation of PRMT7 related syndrome: Expanding the phenotypic manifestations.PRMT7 相关综合征的产前和产后表现:扩展表型表现。
Am J Med Genet A. 2019 Jan;179(1):78-84. doi: 10.1002/ajmg.a.6. Epub 2018 Dec 4.
6
Pseudohypoparathyroidism.假性甲状旁腺功能减退症。
Endocrinol Metab Clin North Am. 2018 Dec;47(4):865-888. doi: 10.1016/j.ecl.2018.07.011. Epub 2018 Oct 12.
7
Further delineation of the phenotype caused by loss of function mutations in PRMT7.PRMT7功能丧失突变所导致表型的进一步描绘。
Eur J Med Genet. 2019 Mar;62(3):182-185. doi: 10.1016/j.ejmg.2018.07.007. Epub 2018 Jul 10.
8
Efficacy and safety of two doses of Norditropin (somatropin) in short stature due to Noonan syndrome: a 2-year randomized, double-blind, multicenter trial in Japanese patients.两剂诺德人体生长激素(生长激素)治疗努南综合征所致身材矮小的疗效与安全性:一项针对日本患者的为期2年的随机、双盲、多中心试验
Endocr J. 2018 Feb 26;65(2):159-174. doi: 10.1507/endocrj.EJ17-0313. Epub 2017 Nov 7.
9
Expanding the clinical and molecular spectrum of PRMT7 mutations: 3 additional patients and review.扩展 PRMT7 突变的临床和分子谱:3 例额外患者及综述。
Clin Genet. 2018 Mar;93(3):675-681. doi: 10.1111/cge.13137. Epub 2018 Feb 5.
10
Loss of the arginine methyltranserase PRMT7 causes syndromic intellectual disability with microcephaly and brachydactyly.精氨酸甲基转移酶PRMT7的缺失会导致伴有小头畸形和短指(趾)畸形的综合征性智力障碍。
Clin Genet. 2017 May;91(5):708-716. doi: 10.1111/cge.12884. Epub 2016 Nov 30.

PRMT7 基因突变导致的身材矮小:生长激素治疗反应的首次证据。

Short stature in PRMT7 Mutations: first evidence of response to growth hormone treatment.

机构信息

Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Endocrinology Unit, Milan, Italy.

Department of Clinical Sciences and Community Health, University of Milan, Milan, Italy.

出版信息

Eur J Hum Genet. 2023 Feb;31(2):195-201. doi: 10.1038/s41431-022-01220-9. Epub 2022 Nov 9.

DOI:10.1038/s41431-022-01220-9
PMID:36348013
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC9905056/
Abstract

Protein arginine methyltransferase 7 (PRMT7) pathogenetic variants have been associated with the human disorder of Short Stature, Brachydactyly, Intellectual Developmental Disability and Seizures syndrome (SBIDDS). Only 15 cases have been described in the literature. Here we report two female dizygotic twins with novel compound heterozygous deleterious variants of PRMT7 and describe the associated endocrine manifestations and short-term response to recombinant growth hormone (rGH) treatment. They were born at 36 + 3 weeks from a dichorionic diamniotic twin pregnancy. Twin A was appropriate for gestational age while Twin B was small for gestational age. Whole exome sequencing analyses showed the same novel compound heterozygous genetic defects in the PRMT7 gene (c.1220 G > A of maternal origin; c.1323 + 2 T > G of paternal origin, Fig. 1). Due to severe short stature and growth impairment, at six years of age, endocrine investigations were performed to rule out growth hormone (GH) deficiency, and revealed GH deficiency (GHD) in Twin A and an appropriate GH response in Twin B. Therefore, both started rGH, albeit at different dosages according to the underlying diagnosis. Both showed a satisfactory short-term response to treatment with height gain (∆HT) of +0.52 SDS (Twin A) and +0.88 SDS (Twin B) during the first year. In conclusion, our findings expand the knowledge about the endocrine manifestations associated with PRMT7 pathogenetic variants, including GH deficiency and rGH response. Further studies are needed to investigate long-term outcomes and establish whether PRMT7 genetic defects can be included among syndromic short stature treatable with rGH.

摘要

精氨酸甲基转移酶 7(PRMT7)致病性变异与人类短身材、短指(趾)畸形、智力发育障碍和癫痫综合征(SBIDDS)有关。文献中仅描述了 15 例。本文报道了两例具有 PRMT7 新型复合杂合有害变异的女性双卵双胎,并描述了相关的内分泌表现和短期重组人生长激素(rGH)治疗反应。她们均在 36+3 孕周时从双绒毛膜双羊膜囊的双胞胎妊娠中分娩。双胞胎 A 为适期分娩,而双胞胎 B 为小于胎龄儿。全外显子组测序分析显示 PRMT7 基因存在相同的新型复合杂合遗传缺陷(母源性 c.1220 G>A;父源性 c.1323 +2 T>G,图 1)。由于严重身材矮小和生长障碍,在 6 岁时进行了内分泌检查以排除生长激素(GH)缺乏症,并发现双胞胎 A 存在 GH 缺乏症(GHD),而双胞胎 B 的 GH 反应正常。因此,两人均开始接受 rGH 治疗,但根据潜在诊断,剂量不同。在第一年,两人均表现出对治疗的满意短期反应,身高增长(∆HT)分别为+0.52 SDS(双胞胎 A)和+0.88 SDS(双胞胎 B)。总之,我们的研究结果扩展了与 PRMT7 致病性变异相关的内分泌表现的知识,包括 GH 缺乏症和 rGH 反应。需要进一步研究以探讨长期结果,并确定 PRMT7 遗传缺陷是否可以被纳入 rGH 可治疗的综合征性身材矮小症中。