Bilateral maxillary mucoceles: An unusual presentation of cystic fibrosis

Department of Otolaryngology and Head and Neck Surgery, John Radcliffe Hospital, Oxford, UK.
The Journal of Laryngology & Otology (Impact Factor: 0.67). 03/2012; 126(3):319-21. DOI: 10.1017/S002221511100329X
Source: PubMed


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

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    • "Patients commonly present with pain, nasal congestion, diplopia, and exophthalmos depending on the location of the lesion and the extent of bone erosion. The most widely accepted treatment protocol is endoscopic marsupialization of the mucocele [3]. Here, the clinical properties, radiological findings, treatment protocols , and medium-long-term results of patients undergoing surgery for sinus mucocele are discussed. "
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    ABSTRACT: Objectives: We present the clinical and radiological features, treatment protocols, and medium-long-term results of our patients following surgery for paranasal sinus mucocele, along with a review of the relevant literature. Materials and methods: A total of 18 patients (11 women and 7 men) who underwent surgery for paranasal sinus mucocele at Kocaeli University Faculty of Medicine, Department of Otolaryngology, between 2006 and 2013 were examined retrospectively. The mean patient age was 41 (range 4-73). Demographic and radiological features, symptoms, treatment protocols, and postoperative outcomes were recorded. Results: The most frequently affected sinus was the maxillary sinus (n=9, 50%) followed by the frontal sinus (n=6, 33%) and sphenoidal sinus (n=3, 16%). The main symptom was headache. Endoscopic marsupialization of the mucocele was applied in all 18 patients, while frontal sinus exploration with the osteoplastic flap procedure was performed in one patient and the Caldwell-Luc operation was performed in another patient. The Caldwell-Luc procedure was subsequently required in one patient (6%) and endoscopic revision surgery was required in another patient (6%). Conclusion: Sinus mucocele that enlarges, eroding the surrounding bone tissue, and induces various clinical symptoms due to the impression of the expansile mass, is treated surgically, and must be planned carefully to prevent serious complications.
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    ABSTRACT: The maxillary sinus may be involved in a wide variety of disorders. Many of these share common presenting symptoms but some have unique features. This article reviews some of the recent publications in this area. The majority of isolated maxillary sinus disease has been previously described. Some recent data on the microbiology of sinusitis have been published. The review also highlights the growing role of endoscopic surgical management due to improved instrumentations and techniques. On the basis of the review, diagnosing isolated maxillary sinus disease can still be delayed due to late presentation. When suspected, it is advisable to investigate early with computed tomography scanning and proceeding to MRI if needed. Often these will show certain features with clues to the diagnosis. Final diagnosis is frequently only obtained on histological examination. The majority of these disorders can now be managed by endoscopic techniques alone with open surgery required in a small number of cases.
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