A rare cause of respiratory distress in preterm infants: a case report of acquired subglottic cysts.

IF 3.2 3区 医学 Q1 PEDIATRICS
Luca Barchi, Giulia Russo, Sara Donvito, Giulia Barbato, Francesco Leo, Elisa Iannella, Angelo Ghidini, Lorenzo Iughetti, Giancarlo Gargano
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Abstract

Background: The Subglottic Cysts (SGCs) are a rare cause of respiratory distress in infants. Typical risk factors include male gender, extreme prematurity, gastro-oesophageal reflux and invasive ventilation, the latter being associated with mucosal damage and blockage of the subglottic cysts' ducts. We describe a case of acquired subglottic cysts in a premature infants presented with respiratory distress.

Case presentation: A premature male infant was born at 25 weeks + 2 days with a history of monochorionic diamniotic twin pregnancy with twin-to-twin transfusion syndrome. During hospitalization, invasive mechanical ventilation was necessary for a total of 18 days; the patient was discharged at postmenstrual age of 40 weeks + 1 day in good condition. At 43 weeks post-menstrual age, he presented to our department with mixed stridor and worsening of respiratory dynamics. A laryngotracheoscopy evaluation was performed. The exam showed the presence of multiple SGCs causing an almost complete obstruction of the airway. Because of the significant reduction of the airway's patency, the child underwent a tracheotomy and thereafter cysts' removal using cold steel microinstruments. A better airway patency was restored although a slight glottic edema persisted. The histopathology confirmed the benign nature of the lesions. Successive controls showed a completely patent airway and absence of SGCs.

Conclusion: In conclusion, SGCs should be considered in preterm infants with respiratory distress previously intubated, which cannot be explained by the most common causes. Early diagnosis and treatment are fundamental to reducing the morbidity and mortality associated with this disease.

早产儿呼吸窘迫的罕见病因:获得性声门下囊肿病例报告。
背景:声门下囊肿(SGC)是导致婴儿呼吸窘迫的罕见原因。典型的危险因素包括男性、极度早产、胃食管反流和有创通气,后者与粘膜损伤和声门下囊肿导管堵塞有关。我们描述了一例出现呼吸窘迫的早产儿获得性声门下囊肿病例:一名早产男婴于 25 周+2 天时出生,患有单绒毛膜双羊膜妊娠和双胎输血综合征。住院期间,患者需要进行有创机械通气共 18 天;患者于月经后 40 周+1 天出院,情况良好。月经后 43 周时,他因混合性哮鸣音和呼吸动力学恶化来到我科就诊。我们对他进行了喉气管镜检查。检查结果显示,多发性 SGCs 几乎完全阻塞了气道。由于气道的通畅性明显下降,患儿接受了气管切开术,随后使用冷钢微型器械切除了囊肿。虽然声门仍有轻微水肿,但气道的通畅性已经恢复。组织病理学证实病变为良性。连续的对照显示气道完全通畅,没有 SGCs:总之,早产儿出现呼吸困难并插管后,如果无法用最常见的原因来解释,则应考虑 SGCs。早期诊断和治疗是降低该病发病率和死亡率的基础。
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来源期刊
CiteScore
6.10
自引率
13.90%
发文量
192
审稿时长
6-12 weeks
期刊介绍: Italian Journal of Pediatrics is an open access peer-reviewed journal that includes all aspects of pediatric medicine. The journal also covers health service and public health research that addresses primary care issues. The journal provides a high-quality forum for pediatricians and other healthcare professionals to report and discuss up-to-the-minute research and expert reviews in the field of pediatric medicine. The journal will continue to develop the range of articles published to enable this invaluable resource to stay at the forefront of the field. Italian Journal of Pediatrics, which commenced in 1975 as Rivista Italiana di Pediatria, provides a high-quality forum for pediatricians and other healthcare professionals to report and discuss up-to-the-minute research and expert reviews in the field of pediatric medicine. The journal will continue to develop the range of articles published to enable this invaluable resource to stay at the forefront of the field.
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