21三体患者COVID-19与塑料支气管炎的诊断混淆1例

Maricruz J. Montelongo-Rodriguez , Julio C. Acuña-Castro , Antonio Muñiz-Buenrostro
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引用次数: 0

摘要

塑性支气管炎(PB)是一种罕见但可能危及生命的肺部疾病,其特征是形成有凝聚力的支气管铸型,能够阻塞气道并引起严重的呼吸窘迫。虽然它更常与儿科人群相关,特别是那些患有先天性心脏病的人群,但PB已越来越多地在成人中得到认可,通常具有多种且未被充分认识的病因。区分炎症型(I型)和淋巴型(II型)铸造亚型对指导诊断和治疗至关重要。本报告描述了一例罕见的21三体患者成人发病的PB,这种关联以前没有很好的文献记载。病例表现:女性,30岁,21三体,表现为急性低氧血症,口周发绀,咳咳伴白色支气管痰。既往有COVID-19肺炎病史,无先天性心脏病或复发性感染。最初的胸部影像显示马赛克灌注和心包积液,支气管镜检查发现白色丝状物质部分阻塞支气管。铸型组织病理学检查显示纤维蛋白带伴炎性细胞,符合I型炎性PB。患者最初对皮质类固醇治疗有反应;然而,停药后症状复发,需要重复类固醇疗程。支气管镜下取出铸型导致症状改善。没有发现其他传染或结构性原因。结论本病例强调了在成人急性低氧性呼吸衰竭的鉴别诊断中考虑可塑性支气管炎的必要性,特别是当临床和影像学表现与COVID-19肺炎相似时。它强调了结合影像学、支气管镜检查和组织病理学诊断的重要性,以及皮质类固醇在治疗炎症性PB中的治疗作用。据我们所知,这是21三体成人中首次报道的PB病例之一,扩大了对其发生的认识,超越了传统的儿科和先天性心脏病背景。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Diagnostic confusion of COVID-19 versus plastic bronchitis in a patient with trisomy 21: A case report

Introduction

Plastic bronchitis (PB) is a rare but potentially life-threatening pulmonary disorder characterized by the formation of cohesive bronchial casts capable of obstructing airways and causing severe respiratory distress. Although it is more frequently associated with pediatric populations, particularly those with congenital heart disease, PB has been increasingly recognized in adults, often with diverse and underrecognized etiologies. Distinguishing between inflammatory (Type I) and lymphatic (Type II) cast subtypes is critical for guiding diagnosis and management. This report describes a rare case of adult-onset PB in a patient with trisomy 21, an association not previously well-documented.

Case presentation

A 30-year-old female with trisomy 21 presented with acute hypoxemia, perioral cyanosis and productive cough with expectoration of whitish bronchial casts. She had a prior history of COVID-19 pneumonia but no congenital heart disease or recurrent infections. Initial chest imaging revealed mosaic perfusion and pericardial effusion, and bronchoscopy identified whitish filamentous material partially obstructing the bronchi. Histopathological examination of the casts revealed fibrin bands with inflammatory cells, consistent with Type I inflammatory PB. The patient responded initially to corticosteroid therapy; however, symptoms recurred upon withdrawal, necessitating repeated steroid courses. Bronchoscopic removal of casts led to symptomatic improvement. No alternative infectious or structural causes were identified.

Conclusion

This case highlights the need to consider plastic bronchitis in the differential diagnosis of adults presenting with acute hypoxemic respiratory failure, particularly when clinical and imaging findings mimic COVID-19 pneumonia. It underscores the importance of combining imaging, bronchoscopy, and histopathology for diagnosis, and the therapeutic role of corticosteroids in managing inflammatory PB. To our knowledge, this is one of the first reported cases of PB in an adult with trisomy 21, expanding awareness of its occurrence beyond traditional pediatric and congenital heart disease contexts.
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