Department of Respiratory Medicine and Infectious Diseases, Niigata University Graduate School of Medical and Dental Sciences, Japan.
Department of Molecular Endocrinology, National Research Institute for Child Health and Development, Japan.
Intern Med. 2021 Jun 15;60(12):1921-1926. doi: 10.2169/internalmedicine.5479-20. Epub 2021 Feb 1.
A 31-year-old woman who was clinically diagnosed with Silver-Russell syndrome (SRS) in childhood was admitted with complaints of dyspnea. She had hypercapnic respiratory failure accompanied by nocturnal hypoventilation. Computed tomography revealed systemic muscle atrophy and superior mesenteric artery syndrome; however, the bilateral lung fields were normal. She was treated with nocturnal noninvasive positive pressure ventilation and showed improvement of respiratory failure. In this case, loss of methylation on chromosome 11p15 and maternal uniparental disomy of chromosome 7, which are the common causes of SRS, were not detected. This is a rare case of adult SRS manifesting as chronic hypercapnic respiratory failure.
一位 31 岁女性,儿童时期临床诊断为 Silver-Russell 综合征(SRS),因呼吸困难入院。她患有高碳酸血症性呼吸衰竭,伴有夜间通气不足。计算机断层扫描显示全身肌肉萎缩和肠系膜上动脉综合征;然而,双肺野正常。她接受了夜间无创正压通气治疗,呼吸衰竭得到改善。在这种情况下,未检测到 SRS 的常见原因,即 11p15 号染色体甲基化缺失和 7 号染色体母源单亲二体。这是一例罕见的成年 SRS 表现为慢性高碳酸血症性呼吸衰竭的病例。