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双侧上颌窦黏液囊肿:囊性纤维化的一种罕见表现。

Bilateral maxillary mucoceles: an unusual presentation of cystic fibrosis.

作者信息

Qureishi A, Lennox P, Bottrill I

机构信息

Department of Otolaryngology and Head and Neck Surgery, John Radcliffe Hospital, Oxford, UK.

出版信息

J Laryngol Otol. 2012 Mar;126(3):319-21. doi: 10.1017/S002221511100329X. Epub 2012 Jan 12.

Abstract

OBJECTIVE

We report a rare case of bilateral maxillary mucoceles. This was the primary presentation in a child with an underlying diagnosis of cystic fibrosis.

CASE REPORT

A five-month-old infant presented with isolated symptoms of nasal congestion. Initial examination and investigation with computed tomography identified the cause to be bilateral maxillary mucoceles. The child was investigated for cystic fibrosis and found to be positive. Given the young age and anatomy, a modified surgical approach was used to treat the mucoceles, comprising endoscopic and bilateral uncapping of the maxillary mucocele, uncinectomy, and middle meatal antrostomy. The child was then followed up with appropriate treatment for cystic fibrosis. At six-week follow up, there was no recurrence of symptoms.

CONCLUSION

Maxillary mucoceles are extremely rare but can present in patients with chronic sinusitis or cystic fibrosis. To our knowledge, there are no prior reports of this condition being the primary indicator of underlying cystic fibrosis in the absence of upper respiratory tract symptoms. Doctors should be aware that mucoceles can present in very young individuals and may indicate an underlying diagnosis of cystic fibrosis. A high index of clinical suspicion will avoid delays in diagnosis and long-term management.

摘要

目的

我们报告一例罕见的双侧上颌窦黏液囊肿病例。这是一名患有囊性纤维化潜在诊断的儿童的主要表现。

病例报告

一名五个月大的婴儿出现孤立的鼻塞症状。最初的检查和计算机断层扫描调查确定病因是双侧上颌窦黏液囊肿。对该儿童进行了囊性纤维化检查,结果呈阳性。鉴于患儿年龄小及解剖结构特点,采用了一种改良的手术方法来治疗黏液囊肿,包括内镜下双侧上颌窦黏液囊肿开窗、钩突切除术和中鼻道上颌窦造瘘术。然后对该儿童进行了适当的囊性纤维化治疗随访。在六周的随访中,症状没有复发。

结论

上颌窦黏液囊肿极为罕见,但可出现在慢性鼻窦炎或囊性纤维化患者中。据我们所知,在没有上呼吸道症状的情况下,此前尚无这种情况作为潜在囊性纤维化主要指标的报道。医生应意识到黏液囊肿可出现在非常年幼的个体中,可能提示潜在的囊性纤维化诊断。高度的临床怀疑指数将避免诊断和长期管理的延误。

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