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一名儿科患者中2型糖原贮积病与5型先天性肌无力综合征的共病:病例报告

Co-occurrence of Glycogen Storage Disease Type 2 and Congenital Myasthenic Syndrome Type 5 in a Pediatric Patient: A Case Report.

作者信息

Al-Sharif Fawzia, Alamer Mohammed F, Taher Hussein O, Gazzaz Raneem Y, AlRuwaithi Asma O, Miliany Tuleen T, Alrufaihi Mohammed A, Al Amer Abdullah F

机构信息

Pediatrics, Saudi German Hospital Jeddah, Jeddah, SAU.

College of Medicine, Batterjee Medical College, Jeddah, SAU.

出版信息

Cureus. 2022 Jun 26;14(6):e26345. doi: 10.7759/cureus.26345. eCollection 2022 Jun.

Abstract

Glycogen storage disease type 2 (also known as Pompe disease) is a metabolic disorder characterized by an accumulation of glycogen within lysosomes. Pathophysiologically, this condition is caused by an autosomal recessive deficiency of the lysosomal acid alpha-glucosidase enzyme, resulting in defects in lysosomal metabolism. Glycogen accumulation causes advanced muscle weakness (myopathy) throughout the body, including the heart, skeletal muscles, liver, and the neurological system. Currently, there is no definitive treatment for Pompe disease. However, recent studies have indicated that enzyme replacement therapy (ERT) can be effective. Myasthenia gravis (MG) is an autoimmune illness that affects the postsynaptic acetylcholine receptors and causes fatigue that can be eased by rest. MG is frequently accompanied by a thymoma. Dyspnea and/or bulbar symptoms can indicate an imminent crisis requiring immediate intervention. Here, we present a rare case of a four-year-old female patient who initially presented at the age of one month with the infantile form of Pompe disease and congenital myasthenia syndrome type 5. The patient presented with bradycardia, poor suckling, respiratory distress, and respiratory failure requiring assisted ventilation, subglottic stenosis, and tachypnea. Whole exome sequencing was used for definitive diagnosis. ERT (Myozyme) was administered with good results. We propose that early identification and management of Pompe disease with Myozyme can improve patients' condition and ultimately increase the possibility of survival.

摘要

2型糖原贮积病(也称为庞贝病)是一种代谢紊乱疾病,其特征是溶酶体内糖原蓄积。在病理生理学上,这种疾病是由溶酶体酸性α-葡萄糖苷酶的常染色体隐性缺乏引起的,导致溶酶体代谢缺陷。糖原蓄积会导致全身包括心脏、骨骼肌、肝脏和神经系统在内的进行性肌无力(肌病)。目前,庞贝病尚无确切的治疗方法。然而,最近的研究表明酶替代疗法(ERT)可能有效。重症肌无力(MG)是一种自身免疫性疾病,会影响突触后乙酰胆碱受体,并导致疲劳,休息后可缓解。MG常伴有胸腺瘤。呼吸困难和/或延髓症状可能表明即将发生危机,需要立即干预。在此,我们报告一例罕见病例,一名4岁女性患者,1个月大时首次出现婴儿型庞贝病和5型先天性肌无力综合征。患者表现为心动过缓、吸吮无力、呼吸窘迫、需要辅助通气的呼吸衰竭、声门下狭窄和呼吸急促。采用全外显子组测序进行明确诊断。给予ERT(美而赞)治疗,效果良好。我们建议,早期识别并用美而赞治疗庞贝病可以改善患者状况,并最终增加生存可能性。

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Pompe disease: from pathophysiology to therapy and back again.庞贝病:从病理生理学到治疗再到回归。
Front Aging Neurosci. 2014 Jul 23;6:177. doi: 10.3389/fnagi.2014.00177. eCollection 2014.
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Two cases of Pompe's disease: case report and review of literature.两例庞贝氏病:病例报告及文献综述
Indian Heart J. 2012 Mar-Apr;64(2):214-6. doi: 10.1016/S0019-4832(12)60067-4. Epub 2012 Apr 28.

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