J Clin Exp Dent. 2019;11(2):e208-12. Sphenoid sinus mucocele in a North African woman Journal section: Oral Surgery doi:10.4317/jced.55161 Publication Types: Case Report http://dx.doi.org/10.4317/jced.55161 Sphenoid sinus mucocele: an unusual complication of head and neck irradiation in a North African woman Aina Brunet-Garcia 1, Mª Dolores Costa-Climent 2, Maria Pujol-Rodríguez 3, Laia Brunet-Garcia 4, Marta Fau­bel-Serra 5 1 MD, Department of Otorhinolaryngology, Hospital Universitar Parc Taulí. Sabadell, Barcelona. Universitat Autònoma de Barce- lona. Universitat de València. Spain 2 MD, Department of Otorhinolaryngology, Hospital General Universitari de Castelló. Castelló de la Plana, Castelló, Spain 3 MD, Department of Otorhinolaryngology, Hospital Universitari Vall d’Hebron. Barcelona. Spain 4 MD, Department of Paediatric Cardiology, Hospital de Mataró. Department of Pediatrics, Hospital Sant Joan de Déu, Barcelona. Universitat de Barcelona. Spain 5 MD, PhD. Head of Otorhinolaryngology department, Hospital General Universitari de Castelló. Castelló de la Plana, Castelló, Spain Correspondence: Servei d’Otorrinolaringologia Hospital Universitari Parc Taulí Parc Taulí, 1 Brunet-Garcia A, Costa-Climent MD, Pujol-Rodríguez M, Brunet-Garcia 08203 Sabadell (Barcelona, Spain) L, Faubel-Serra M. Sphenoid sinus mucocele: an unusual complication abrunet@tauli.cat of head and neck irradiation in a North African woman. J Clin Exp Dent. 2019;11(2):e208-12. http://www.medicinaoral.com/odo/volumenes/v11i2/jcedv11i2p208.pdf Received: 20/07/2018 Accepted: 17/12/2018 Article Number: 55161 http://www.medicinaoral.com/odo/indice.htm © Medicina Oral S. L. C.I.F. B 96689336 - eISSN: 1989-5488 eMail: jced@jced.es Indexed in: Pubmed Pubmed Central® (PMC) Scopus DOI® System Abstract Mucocele is a common benign lesion otherwise rarely located in the sphenoid sinus. Some complications after head and neck irradiation have been described in the literature until now. To our knowledge, this is the first report of a sphenoid sinus mucocele in a North African patient treated some years before with radiotherapy for a nasophary- ngeal carcinoma (NPC). We extend the literature review about this infrequent finding, of which head and neck surgeons should be aware. Key words: Mucocele, sphenoid sinus mucocele, nasopharyngeal carcinoma, radiotherapy, North African. Introduction that developed sphenoid sinus mucocele 11 years after Isolated sphenoid sinus mucocele is a rare entity. Its in- receiving radiotherapy for a nasopharyngeal carcinoma. cidence has been reported to be 1-3% of all mucoceles. Diagnosis of sphenoid sinus mucocele is often dela- Case Report yed due to its late symptoms that are not evident un- A 49 year-old female patient originally from Maghreb til it expands and compresses neighbouring structures. was referred to our Department due to some abnormali- Furthermore, this location makes it impossible to direct ties found on a magnetic resonance imaging (MRI) de- examination (1). Correlation between radiotherapy and veloped for temporomandibular joint dysfunction. mucocele formation has rarely been described in the The patient had undergone radiotherapy for a NPC 11 literature. We report a case of a North African woman years before. e208 J Clin Exp Dent. 2019;11(2):e208-12. Sphenoid sinus mucocele in a North African woman MRI revealed a cystic mass located in the clivus area by the ethmoid (20-30%), maxilar (10%) and sphenoid (Fig. 1a,b). Contrast computed tomography scan (CT) sinus (1-3%) (2). showed a well-defined mass with low attenuation and Mucoceles are considered to arise due to sinus ostium unenhanced by contrast agents, located in the sphenoid obstruction, preceded by fibrosis, infection, trauma, sur- bone (Fig. 1c,d). Nasal endoscopic examination showed gery (35-66%), fibrous dysplasia, osteoma or ossifying a cystic mass in the upper side of the cavum. fibroma. Radiotherapy for a NPC is a rare ethiology for The patient had gone to the Emergency Unit complai- this entity; scarcely any case has been described in the ning about visual disturbances some weeks before. literature before (2). Under general anaesthesia marsupialisation of the mu- Although being rare in Caucasian, it is endemic in southern cocele was performed, and a thick yellowish mucopuru- China. Southeast Asia, North Africa, the Middle East and lent fluid was aspired from the mass (Fig. 2). the Arctic are considered intermediate-risk regions (3). Pathological findings of the lining mucosa showed a mu- Nasal endoscopy followed by a CT scan or an MRI are cous retention cyst with no evidence of tumor cells (Fig. useful tools for the diagnosis, since physical examina- 3). Intraoperative cultures were sterile. Visual symptoms tion might be normal. CT scan would be characterized disappeared after surgery. The patient had regular fo- by a sinus homogeneous isodense mass which does llow-ups with no evidence of recurrence during 4 years. not enhance with contrast administration; it totally fills Informed consent was obtained by the patient. the sinus cavity and surrounding structures. Mucoceles usually show moderate or low signal intensity on T1 Discussion weighted MRI images and high signal intensity on T2 Mucoceles are considered the most frequent expansive weighted images, although in some cases there is a high lesions of the paranasal sinuses (1). They are most com- protein content and density, which leads to T1 high in- monly located in the frontal sinus (60-65%), followed tensity images, as in the present case (4). Fig. 1: Radiological Images: Magnetic Ressonance (MR). Mucocele shows high signal in- tensity in both T1 and T2 weighted images. a: T2 axial MR, b: T1 coronal MR, c: sagital CT, d: axial CT. e209 J Clin Exp Dent. 2019;11(2):e208-12. Sphenoid sinus mucocele in a North African woman Fig. 2: Surgical images. a: detritus material inside sphenoid sinus (star), b: suction of the mu- cocele, c,d: empty sphenoid sinus cavity after mucocele excision (arrow). Fig. 3: Histopathological examination images. a: Cyst lined by respiratory epithelium, b: Inflammatory cells and cholesterol crystals (arrow), c: inflammatory cells, d: hemorrage areas. e210 J Clin Exp Dent. 2019;11(2):e208-12. Sphenoid sinus mucocele in a North African woman Porter et al. analysed paranasal sinuses CT scans before presentation signs were headache (7,8) and visual distur- and after radiotherapy in patients with NPC. They could bances (1,8-11). To our knowledge this complication of find an increase in chronic sinus disease due to mucous radiotherapy has never been mentioned before in a Nor- membrane atrophy, as a late complication of radiothera- th African patient. The role of radiotherapy as a primary py (4). The most common side effects of radiation the- cause of this entity was first described by Rejab et al. in rapy for a NPC are xerostomia (98%), eustachian tube 1991. In that case authors supported the idea that sinus fibrosis causing serous otitis media, fibrosis of the tem- ostium was occluded by scarred mucosa following ra- poromandibular joint leading to the appearance of tris- diotherapy, leading to mucocele formation (9), similarly mus, and neck fibrosis causing neck stiffness, with an to the present case. incidence from 40 to 70% (2). Local recurrence was improbable in this case due to CT Conclusions and MRI characteristics, endoscopic examination fin- Sphenoid sinus mucocele developing after radiothe- dings, the presence of a previous nasosinusal surgery, rapy for NPC has rarely been reported and it has been and long time (11 years) after radiotherapy, since only a mostly described in chinese man. Its presentation may 9% of recurrence arising 5 years after radiotherapy has sometimes mimic recurrence and hence, clinical history, been reported (5). examination, CT scanning and MRI are useful tools for Endoscopic drainage, which is effective and secure, is the diagnosis. It is crucial for oral surgeons and otorhi- considered the outstanding treatment for this entity (6). nolaryngologists to keep this uncommon entity in mind Only few cases of sphenoid mucocele after irradiation in order to allow early diagnosis and treatment and pre- for NPC have been reported in the literature, most of vent possible complications developing in the orbits or them being chinese male patients (Table 1). Common intracranially. Table 1: Patients that developed sphenoid sinus mucocele after radiotherapy for nasopharyneal carcinoma (NPC). Author Year of Ethiology Sex Age Symptoms Period from publishing NPC Rejab (9) 1991 Chinese Male 30 Blurring vision 16 years Wong (12) 1997 Chinese Male 33 Yawning attacks 13 years Wong (10) 2001 Chinese Male 49 Diplopia 21 years Mark (7) 2008 - Male 49 Frontal Headache 18 months - Male 64 Giddiness 7 years Headache - Male 49 Occipital pain 5 years Lin JS (11) 2004 Chinese Male 46 Blurred vision 7 years Soon S (1) 2009 Chinese Male 66 Visual loss - Chinese Male 53 None - Chinese Female 45 Nasal discharge - Mnejja (8) 2011 French Female 30 Diplopia, headache 9 years French Female 56 Fronto-orbital mass 5 years References third nerve paralysis: CT and MR findings. Dentomaxillofac Radiol. 1. Soon SR, Lim CM, Singh H, Sethi DS. Sphenoid sinus mucocele: 10 2004;33:342–4. cases and literature review. J Laryngol Otol. 2010;124:44–7. 5. Lee AW, Foo W, Law SC, Poon YF, Sze WM, O SK, et al. Recurrent 2. 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