Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Endocrinology Unit, Milan, Italy.
Department of Clinical Sciences and Community Health, University of Milan, Milan, Italy.
Eur J Hum Genet. 2023 Feb;31(2):195-201. doi: 10.1038/s41431-022-01220-9. Epub 2022 Nov 9.
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.
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。