婴儿囊性纤维化患者的鼻窦粘液囊肿导致单侧突出:文献回顾和病例报告研究。

IF 1.7 Q2 MEDICINE, GENERAL & INTERNAL
Annals of Medicine and Surgery Pub Date : 2024-11-07 eCollection Date: 2024-12-01 DOI:10.1097/MS9.0000000000002700
Narges Lashkarbolouk, Mahdi Mazandarani, Parastou Biazar, Lobat Shahkar
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引用次数: 0

摘要

导言和重要性:囊性纤维化(CF)是一种广泛存在的缩短寿命的隐性遗传病,可表现为鼻窦粘液囊肿。鼻窦粘液囊是一种罕见病,单侧突眼的发病率数据有限:作者介绍了一例 19 个月大的 CF 男孩的病例,他的右眼突眼和外斜视不断加重。脑部和眼眶核磁共振成像检查显示:弥漫性息肉状粘膜增厚,可能有致密的真菌沉积,中面部畸形,尤其是右侧,右叶内侧和下壁隆起更明显,右侧乙状粘液瘤导致眼球移位,内侧直肌受压,视神经受压。眼底检查显示眼盘水肿和血管充血。内窥镜鼻窦手术成功引流了粘液瘤,并使用抗生素和皮质类固醇治疗,症状在三周内得到改善,突眼症状也得到缓解:粘液囊是儿童患者中与 CF 相关的一种不常见的并发症。因此,任何出现此问题的儿童都应接受 CF 评估。研究这种罕见病症的潜在机制和后果可能会改善治疗方法和受影响患者的治疗效果:结论:CF 患者鼻窦粘液囊伴有单侧突眼的情况并不常见,内窥镜鼻窦手术似乎是治疗这种并发症的有效方法,即使在 CF 患者等高风险儿科人群中也是如此。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Sinus mucocele leads to unilateral proptosis in an infant patient with cystic fibrosis: a literature review and a case report study.

Introduction and importance: Cystic fibrosis (CF) is a widespread life-shortening recessive genetic disease and can present with sinus mucocele. Sinus mucocele is a rare condition, with limited prevalence data on unilateral proptosis.

Case presentation: The authors present a case of a 19-month-old boy with CF who experienced worsening proptosis and exotropia in his right eye. A brain and orbit MRI revealed diffuse polypoid mucosal thickening, possible dense fungal deposit, deformity of the mid face, especially on the right side, with more prominent bulging of medical and inferior walls of the right lobe, a right ethmoidal mucocele causing ocular globe displacement, medial rectus compression, and optic nerve. An examination of the eye fundus showed disc edema and vascular congestion. Endoscopic sinus surgery successfully drained the mucocele, and treatment with antibiotics and corticosteroids led to symptom improvement and resolution of proptosis within 3 weeks.

Clinical discussion: Mucoceles represent an uncommon complication associated with CF in pediatric patients. Consequently, any child presenting with this issue should undergo evaluation for CF. Investigating this infrequent condition's underlying mechanisms and consequences may improve treatment approaches and outcomes for those impacted.

Conclusion: Sinus mucocele with unilateral proptosis in CF patients is uncommon, and endoscopic sinus surgery appears to be an effective cure for this complication, even in the pediatric population at high risk, like CF patients.

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Annals of Medicine and Surgery
Annals of Medicine and Surgery MEDICINE, GENERAL & INTERNAL-
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