Departments of Pediatric Genetics.
Medical Genetics.
Psychiatr Genet. 2022 Oct 1;32(5):194-198. doi: 10.1097/YPG.0000000000000323. Epub 2022 Sep 14.
Coffin-Lowry syndrome (CLS) is a rare X-linked disorder that, usually affects males, presenting with intellectual disability, short stature, growth retardation, short hands, hyperextensible fingers and progressive kyphoscoliosis. Due to skewed X chromosome inactivation, the clinical presentations of the affected females vary greatly and clinical manifestations could range from mild intellectual disability to typical features of CLS in males. Here, we reported two different novel RPS6KA3 gene mutations in two unrelated CLS patients and also described concomitant compulsive eyebrow-pulling behavior in one of these cases for the first time in the literature.
Coffin-Lowry 综合征(CLS)是一种罕见的 X 连锁疾病,通常影响男性,表现为智力障碍、身材矮小、生长迟缓、短手、手指过度伸展和进行性脊柱后凸。由于 X 染色体失活偏斜,受影响女性的临床表现差异很大,临床表现可从轻度智力障碍到男性 CLS 的典型特征不等。在这里,我们报道了两名无关 CLS 患者的两个不同的 RPS6KA3 基因突变,并首次在文献中描述了其中一名患者同时存在强制性拔眉行为。