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低张性新生儿的杆状体肌病:早期检测与管理的诊断方法

Nemaline Myopathy in a Hypotonic Neonate: Diagnostic Approach for Early Detection and Management.

作者信息

Vu Annie, Nanda Subah, Chassee Todd

机构信息

Medicine, Michigan State University College of Human Medicine, East Lansing, USA.

Emergency Medicine, Helen DeVos Children's Hospital, Grand Rapids, USA.

出版信息

Cureus. 2024 Mar 25;16(3):e56866. doi: 10.7759/cureus.56866. eCollection 2024 Mar.

Abstract

Neonatal hypotonia presents with low muscle tone and an array of symptoms that vary depending on the etiology. The differential diagnosis for this condition is complex. It is crucial to exclude life-threatening causes before following a diagnostic algorithm and performing additional tests. Given the wide range of clinical symptoms and etiologies for neonatal hypotonia, rapid genetic testing has the potential to expedite diagnosis, reduce invasive testing such as muscle biopsy, reduce hospital stays, and guide condition management.  A four-week-old girl was admitted to the emergency department (ED) with a one-day history of lethargy, poor feeding, congestion, cough, and hypoxemia. Given positive rhino-enterovirus testing and high inflammatory markers, antibiotics were administered. Imaging, venous blood gas, and blood cultures were negative, and the patient was admitted to the pediatric intensive care unit (PICU) for hypoxemia. After speech-language pathology (SLP) and occupational therapy (OT) evaluation, weak orofacial muscles and feeding issues resulted in a nasogastric tube placement. A swallow study revealed decreased pharyngeal contraction and post-swallow liquid residue. Laryngoscopy showed mild laryngomalacia and dysphagia with aspiration. Genetic testing identified an ACTA1 mutation and confirmed nemaline myopathy (NM). The patient's oxygen levels dropped further during sleep, resulting in diagnoses of severe obstructive and moderate-severe central sleep apnea. Treatment included oxygen therapy, SLP, physical therapy, albuterol, and cough assists. After discharge, the patient was frequently re-admitted with chronic respiratory failure and bronchiolitis and later had gastrostomy and tracheostomy tubes inserted.  This specific case highlights the importance of implementing a diagnostic algorithm for neonatal hypotonia. It is also important for physicians, especially emergency medicine (EM) providers, to first exclude infection, sepsis, and cardiac and respiratory organ failure before looking into other tests. Then, physicians should evaluate for more rare etiologies. In this patient's case, the hypotonia was due to a rare genetic disease, nemaline myopathy, and a multidisciplinary approach was used for this patient's care.

摘要

新生儿低张力表现为肌张力低下以及一系列因病因不同而各异的症状。这种病症的鉴别诊断很复杂。在遵循诊断流程并进行额外检查之前,排除危及生命的病因至关重要。鉴于新生儿低张力的临床症状和病因范围广泛,快速基因检测有潜力加快诊断、减少诸如肌肉活检等侵入性检查、缩短住院时间并指导病情管理。一名四周大的女孩因嗜睡、喂养困难、鼻塞、咳嗽和低氧血症一天的病史被收入急诊科。鉴于鼻肠道病毒检测呈阳性且炎症指标高,给予了抗生素治疗。影像学检查、静脉血气分析和血培养均为阴性,患者因低氧血症被收入儿科重症监护病房。经过言语病理学(SLP)和职业治疗(OT)评估,口面部肌肉无力和喂养问题导致放置了鼻胃管。吞咽研究显示咽收缩减弱和吞咽后液体残留。喉镜检查显示轻度喉软化和吞咽困难伴误吸。基因检测发现ACTA1突变并确诊为杆状体肌病(NM)。患者在睡眠期间氧水平进一步下降,导致诊断为重度阻塞性和中重度中枢性睡眠呼吸暂停。治疗包括氧疗、SLP、物理治疗、沙丁胺醇和咳嗽辅助治疗。出院后,患者因慢性呼吸衰竭和细支气管炎频繁再次入院,后来插入了胃造口管和气管造口管。这个具体病例凸显了实施新生儿低张力诊断流程的重要性。对于医生,尤其是急诊医学(EM)提供者来说,在进行其他检查之前首先排除感染、败血症以及心脏和呼吸器官衰竭也很重要。然后,医生应评估更罕见的病因。在该患者的病例中,低张力是由一种罕见的遗传疾病杆状体肌病引起的,并且对该患者的护理采用了多学科方法。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/66fa/11040521/6de6513a2608/cureus-0016-00000056866-i01.jpg

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