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婴儿型庞贝病的症状缺乏特异性,可能会延迟诊断和治疗的开展。

Non-specificity of symptoms in infantile-onset Pompe disease may delay the diagnosis and institution of treatment.

机构信息

Department of Biochemistry, Faculty of Medical Sciences, University of Sri Jayewardenepura, Nugegoda, Sri Lanka

Department of Chemical Pathology, Lady Ridgeway Hospital for Children, Borella, Sri Lanka.

出版信息

BMJ Case Rep. 2022 Mar 9;15(3):e247312. doi: 10.1136/bcr-2021-247312.

Abstract

Pompe disease is an autosomal-recessive inherited disorder of glycogen metabolism due to lysosomal acid alpha-glucosidase deficiency. The infantile-onset form is rapidly fatal if left untreated and presents with respiratory symptoms, a typical encounter during infancy. We discuss two infants presenting with respiratory symptoms since early infancy and found to have cardiomegaly, hypotonia, elevated muscle enzymes, leading to the diagnosis of Pompe disease with genetic confirmation. However, both infants expired before the enzyme replacement therapy due to complications of irreversible muscle damage despite supportive medical care. Presentation with respiratory symptoms common during childhood, absence of alarming symptoms such as hypoglycaemia, ketoacidosis or encephalopathy, and relative rarity of Pompe disease can contribute to lapses in the early diagnosis as observed in the index patients. Thus, these cases emphasise the importance of vigilant assessment of common paediatric presentations, which may be presenting symptoms of underlying sinister pathologies.

摘要

庞贝病是一种由于溶酶体酸性α-葡萄糖苷酶缺乏导致的糖原代谢常染色体隐性遗传疾病。如果不治疗,婴儿期发病的快速进展型会迅速致命,并出现呼吸症状,这是婴儿期常见的表现。我们讨论了两名婴儿,他们从婴儿早期开始就出现呼吸症状,并发现有心包肥大、肌张力低下、肌肉酶升高,最终通过基因确诊为庞贝病。然而,尽管进行了支持性治疗,由于不可逆肌肉损伤的并发症,这两名婴儿在酶替代疗法之前都死亡了。在儿童时期,呼吸症状很常见,没有低血糖、酮症酸中毒或脑病等警报症状,而且庞贝病相对罕见,这可能导致早期诊断的遗漏,正如这两个索引病例所观察到的那样。因此,这些病例强调了对常见儿科表现进行警惕评估的重要性,这些表现可能是潜在严重病理的首发症状。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5d32/8915381/5819f6090c57/bcr-2021-247312f01.jpg

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