Department of Cardiovascular Surgery, University Heart & Vascular Center Hamburg, Hamburg, Germany.
Institute of Human Genetics, University Medical Center Hamburg-Eppendorf, Hamburg, Germany.
Sci Rep. 2022 Mar 16;12(1):4489. doi: 10.1038/s41598-022-08476-7.
MASS phenotype is a connective tissue disorder clinically overlapping with Marfan syndrome and caused by pathogenic variants in FBN1. We report four patients from three families presenting with a MASS-like phenotype consisting of tall stature, arachnodactyly, spinal deformations, dural ectasia, pectus and/or feet deformations, osteoarthritis, and/or high arched palate. Gene panel sequencing was negative for FBN1 variants. However, it revealed likely pathogenic missense variants in three individuals [c.3936G > T p.(Lys1312Asn), c.193G > A p.(Asp65Asn)] and a missense variant of unknown significance in the fourth patient [c.4013G > A p.(Ser1338Asn)] in propeptide coding regions of COL2A1. Pathogenic COL2A1 variants are associated with type II collagenopathies comprising a remarkable clinical variablility. Main features include skeletal dysplasia, ocular anomalies, and auditory defects. A MASS-like phenotype has not been associated with COL2A1 variants before. Thus, the identification of likely pathogenic COL2A1 variants in our patients expands the phenotypic spectrum of type II collagenopathies and suggests that a MASS-like phenotype can be assigned to various hereditary disorders of connective tissue. We compare the phenotypes of our patients with related disorders of connective tissue and discuss possible pathomechanisms and genotype-phenotype correlations for the identified COL2A1 variants. Our data recommend COL2A1 sequencing in FBN1-negative patients suggestive for MASS/Marfan-like phenotype (without aortopathy).
MASS 表型是一种结缔组织疾病,临床上与马凡综合征重叠,由 FBN1 中的致病性变异引起。我们报告了来自三个家庭的四名患者,他们表现出类似于 MASS 的表型,包括身材高大、蜘蛛指(趾)、脊柱畸形、硬脊膜膨出、鸡胸和/或足部畸形、骨关节炎和/或高拱形腭。基因panel 测序未发现 FBN1 变异。然而,它在三个人中发现了可能的致病错义变异[c.3936G>T p.(Lys1312Asn), c.193G>A p.(Asp65Asn)],在第四位患者中发现了一个意义不明的错义变异[c.4013G>A p.(Ser1338Asn)],这些变异位于 COL2A1 前肽编码区。致病性 COL2A1 变异与包含显著临床变异性的 II 型胶原病有关。主要特征包括骨骼发育不良、眼部异常和听觉缺陷。以前没有将 MASS 样表型与 COL2A1 变异联系起来。因此,我们患者中 COL2A1 变异的鉴定扩展了 II 型胶原病的表型谱,并表明 MASS 样表型可以归因于各种结缔组织遗传性疾病。我们将患者的表型与相关的结缔组织疾病进行了比较,并讨论了所鉴定的 COL2A1 变异的可能发病机制和基因型-表型相关性。我们的数据建议在提示 MASS/马凡综合征样表型(无主动脉病变)的 FBN1 阴性患者中进行 COL2A1 测序。