Root resorption caused by a maxillary sinus mucocele: a case report

Root resorption caused by a maxillary sinus mucocele: a case report

Root resorption caused by a maxillary sinus mucocele: a case report José Marques, DDS,a Rui Figueiredo, DDS,b José Manuel Aguirre-Urizar, DDS, MD, PhD...

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Root resorption caused by a maxillary sinus mucocele: a case report José Marques, DDS,a Rui Figueiredo, DDS,b José Manuel Aguirre-Urizar, DDS, MD, PhD,c Leonardo Berini-Aytés, DDS, MD, PhD,d and Cosme Gay-Escoda, MD, DDS, PhD,e Barcelona and Leioa, Spain UNIVERSITY OF BARCELONA, IDIBELL RESEARCH INSTITUTE, AND UNIVERSITY OF THE BASQUE COUNTRY

A maxillary sinus mucocele is an infrequent but benign lesion that develops from the obstruction of a seromucous glandular duct of the maxillary sinus mucosa. This clinical entity is generally asymptomatic and selflimited. Mucoceles are described as rounded dome-shaped soft tissue masses frequently located on the floor of the maxillary sinus. In this paper, we present a case of a slightly radiopaque well defined shadow arising from the left maxillary sinus floor that produced the root resorption of the upper second left molar. After the surgical removal of the lesion through a Caldwell-Luc approach, histologic study confirmed the initial diagnosis of mucocele. This case report emphasizes the need of clinical and radiologic follow-up to detect any complications associated with these benign lesions, because, in rare occasions, they can show an aggressive growth pattern. (Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2011;111:e37-e40)

Mucoceles develop when a duct of a seromucous gland of the maxillary sinus mucosa becomes obstructed, resulting in a cystic dilation of the gland. This continued mucous secretion might lead to the development of an expanding epithelial-lined mass which thins and ultimately erodes the bone sinus margins.1-3 The reported incidence of these lesions is 1.4%9.6%, occurring primarily during the third and fourth decades of life, and it is usually discovered when rouSupported by an educational assistance agreement for oral surgery among the University of Barcelona, the Consorci Sanitari Integral, and the Servei Català de la Salut-Generalitat de Catalunya (Catalan Health Service). This study was researched and compiled by the consolidated Research Group in Dental and Maxillofacial Pathology and Treatment of the Institut d‘Investigació Biomèdica de Bellvitge (IDIBELL). a Fellow, Master of Oral Surgery and Orofacial Implantology, School of Dentistry, University of Barcelona. b Associate Professor of Oral Surgery and Professor, Master of Oral Surgery and Orofacial Implantology, School of Dentistry, University of Barcelona; IDIBELL Research Group. c Professor and Chairman, Unit of Oral Medicine, Unit of Oral and Maxillofacial Pathology, Faculty of Medicine and Dentistry, University of the Basque Country/EHU, Leioa, Spain. d Professor of Oral Surgery and Professor, Master of Oral Surgery and Orofacial Implantology, School of Dentistry, University of Barcelona; IDIBELL Research Group. e Chairman and Full Professor, Oral and Maxillofacial Surgery, and Director, Master of Oral Surgery and Orofacial Implantology, School of Dentistry, University of Barcelona; IDIBELL Research Group; Oral and Maxillofacial Surgeon, Teknon Medical Center. Received for publication Dec. 8, 2010; accepted for publication Dec. 15, 2010. 1079-2104/$ - see front matter © 2011 Mosby, Inc. All rights reserved. doi:10.1016/j.tripleo.2010.12.008

tine radiologic examinations are taken.3-4 Generally, mucoceles are a self-limiting condition, with a rate of spontaneous regression and disappearance of 17.6%38%.4 The etiology underlying mucocele formation is multifactorial. Impediments to sinus ostium ventilation are thought to be the primary cause, resulting from anatomical obstruction, mucosal hyperplasia, mass lesions, or other mechanical factors.5,6 Fu et al.6 classify paranasal sinus mucoceles as primary and secondary, based on their anatomic and invasive characteristics. The proposed mechanisms for primary mucocele formation are the inflammatory blockage of mucus drainage, secretory duct obstruction and cystic degeneration of polyps. Furthermore, it is suggested that the retention of residual mucosa in the wound and long-term contact of tissue fluid could also lead to the formation of secondary mucoceles. The same study states that primary mucoceles have a greater potential to cause intraorbital extension compared with secondary mucoceles.6 These lesions are relatively common complications of sinusitis and are seen most often in the frontal and ethmoid regions.7 Radiographically, the mucocele is a rounded dome-shaped soft tissue mass frequently located on the floor of the maxillary sinus and usually filled with clear yellowish fluid.7-9 It is generally asymptomatic, but it can cause facial swelling, nasal obstruction, postnasal drip, nasal discharge, headache, or periorbital or dental pain due to pressure exerted on the mucosa lining. In rare cases, it can also grow and encroach on the inferior orbital floor, causing ocular displacement, nerve compression, lower lid distortion, ptosis, and proptosis.10 e37

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Fig. 1. A, Preoperative panoramic radiography, showing an opacified lesion in the left maxillary sinus floor, as well as the root resorption of the left upper second molar. B, Panoramic reconstruction of a computerized tomographic scan, showing a rounded dome-shaped radiopacified mass with smooth clear-cut margins of bone erosions occurring in the sinus walls.

Commonly, these lesions do not require treatment, unless symptoms appear.4 Traditionally, mucoceles have been treated by means of direct puncture and aspiration through the inferior meatus or natural ostium, or removed by using the Caldwell-Luc approach. More recently, endoscopic intranasal sinus surgery has been used to remove mucoceles of the maxillary sinus.9,11,12 However, this approach requires further training and appropriate equipment, raising treatment costs. Furthermore, the patient’s anatomy can increase the difficulty of this procedure.13,14 The present paper describes the diagnosis and treatment of a case of a maxillary sinus mucocele that was producing the root resorption of the upper second left molar. A review of the literature focusing on the main clinicopathologic and therapeutic aspects is also provided. CASE REPORT A 39-year-old female, allergic to latex and metamizole, showing no systemic pathology, reported to her dentist to extract the left lower first molar. In the panoramic radiography, a slightly radiopaque well defined shadow arising from the left maxillary sinus floor, compatible with mucocele, was detected (Fig. 1, A). The roots of the left upper second molar,

Fig. 2. A, Removal of the cystic lesion through the window previously made in the lateral wall of the left maxillary sinus. B, Appearance of the removed lesion.

which were in direct contact with the lesion, presented a considerable degree of resorption. Nevertheless, pulp vitality was maintained for this tooth. The clinical examination did not show any relevant changes, and the patient was completely asymptomatic. A computerized tomographic (CT) scan of the paranasal sinus confirmed the existence of an opacified lesion in the left maxillary sinus floor (Fig. 1, B). Because this lesion showed an aggressive pattern, especially considering the resorption of the adjacent second molar, it was decided to remove it by using the Caldwell-Luc approach under local anesthesia (articaine in a 4% solution with epinephrine 1:100,000 [Ultracain; Normon, Madrid, Spain]). A horizontal incision was made in the alveolar ridge, with vertical releasing incisions at the level of the canine and the second molar. After raising a full-thickness flap, the bone was removed from the lateral wall of the maxillary sinus with sterile low-speed handpieces using a diamond drill, under profuse sterile saline irrigation. The sinus mucosa was raised and perforated through the window to dissect and remove the cystic lesion (Fig. 2). A reabsorbable collagen membrane (BioGide; Geistlich Biomaterials, Wolhusen, Switzerland)

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was then used to seal the sinus membrane perforation, as well as the lateral wall of the maxillary sinus. The mucoperiosteal flap was detached to facilitate stress-free repositioning, and 4/0 silk sutures (Silkam; Braun, Tuttlingen, Germany) were used to close the wound. After the surgical procedure, the patient was prescribed an antibiotic (amoxicillin 875 mg and clavulanate 125 mg every 8 hours for 10 days [Augmentine 875/125 mg; GlaxoSmithKline, Madrid, Spain]), a nonsteroidal antiinflammatory drug (ibuprofen 600 mg every 8 hours for 7 days [Algiasdin 600; Esteve; Barcelona, Spain]), a single dose of a corticosteroidal drug (methylprednisolone 40 mg [Urbason 40 mg; Aventis Pharma, Madrid, Spain]), and a mouthrinse (0.12% chlorhexidine digluconate every 12 hours for 15 days [Clorhexidina Lacer; Lacer, Barcelona, Spain]). Postoperative instructions together with explanations on the prescribed drugs were given verbally as well as written information to the patient. No complications were registered. The histologic study confirmed a definitive diagnosis of mucocele. The cyst wall was ⬃1 mm thick and showed a thin layer of soft connective tissue, with a moderate lymphoplasmacytic inflammatory infiltrate, as well as foaming macrophages and congested vascular structures. The sample was coated by a ciliated pseudostratified epithelium with some areas of squamous metaplasia phenomena (Fig. 3). Six months after surgery, the patient did not present any clinical or radiographic complications. The thermal pulp vitality tests of the affected molar showed no alterations, and no pathologic mobility of the tooth was observed.

DISCUSSION Mucoceles of the paranasal sinuses are benign cystlike expansible lesions lined with a secretory respiratory mucosa of pseudostratified columnar epithelium. This kind of lesion grows slowly, and could be originated from an obstruction of the sinus outflow in combination with superimposed infection, which can cause the release of cytokines from lymphocytes and monocytes. The cytokine release would stimulate fibroblasts to secrete prostaglandins and collagenases, which could eventually lead to bone resorption.5,10 The diagnosis of mucocele is made on the basis of symptoms, imaging, surgical exploration, and, foremost, histologic confirmation. The symptoms commonly associated with the presence of mucoceles are related to their expansion, usually through the leastresistant path, and subsequent pressure on surrounding anatomic structures.4,5 In the present case, the patient did not present any symptoms related to this lesion; however, root resorption of the second left upper molar was taking place. This is a quite uncommon phenomenon, and we failed to find in the literature any published cases showing similar findings. CT is paramount for the diagnosis of mucoceles. It not only demonstrates sinus involvement, but it also provides information about bone erosion and other effects on neighboring structures. CT shows mucocele as a homogeneous lesion with smooth clear-cut margins of

Fig. 3. Histologic images of the removed maxillary sinus mucocele (hematoxylin and eosin stain). The cyst is lined by respiratory epithelium and shows a mild chronic inflammatory infiltrate. Magnification: A, ⫻40; B, ⫻50.

bone erosions occurring in the sinus walls. In contrast, malignant lesions usually present irregular shapes, erosions or destruction of the sinus walls, infiltration into the surrounding soft tissues, and irregular margins of bone absorption.5,9,10 It is very important to perform a correct differential diagnosis, including mucoceles and other radiopaque lesions, benign or malign, of the maxillary sinus. Benign lesions refer to neurofibroma, dermoid, epidermoid, and cementifying fibroma, angiofibroma, inverting papiloma, and cylindroma. Malignant lesions include adenoid cystic carcinoma, plasmocytoma, embryonal rhabdomyosarcoma, lymphoma, schwannoma, and odontogenic tumors.5,9 In the absence of bone erosions, mucoceles must be differentiated from several conditions, such as retention cysts, chronic sinusitis, antrochoanal polyp, and polyposis of the paranasal cavities.5,9

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Wang et al.4 suggested the following guidelines for management of mucoceles of the maxillary sinus. After the initial detection and in the absence of symptoms, follow-up radiography should be performed ⬃48 months later. If the lesion has not changed significantly in size at that time there is a low probability that it will increase in the longer term. Nevertheless, if the lesion has enlarged significantly after 48 months, it is likely to increase further. Therefore, a second follow-up within 48 months is required. However, unless the mucocele causes any complications, there is no need for any surgical or medical treatment.4 In the present case report, the need for surgical removal was obvious, because it was producing the root resorption of the adjacent molar. There are some surgical options available to eliminate these lesions. Some authors mention that intranasal endoscopy techniques offer good results with very low morbidity.9,11,12 Nevertheless, these procedures require very specific equipment, not usually available in dental offices, and the need for an experienced surgeon. A conventional lateral wall approach also has some advantages, in our opinion. It is a simple and safe technique, with a very low complication rate, and that allows a good exploration of the maxillary sinus. Furthermore, it permits performing sinus augmentation techniques in the same surgical procedure. The present case demonstrates that benign disorders such as mucoceles of the maxillary sinus can, in rare occasions, show an aggressive growth pattern. Therefore, these lesions should be included in the differential diagnosis of pathologies that produce root resorption of maxillary premolars and molars. The surgical removal of this lesion allowed the preservation of the upper second molar, and no relapse was observed after 6 months. REFERENCES 1. Marks SC, Latoni JD, Mathog RH. Mucoceles of the maxillary sinus. Otolaryngol Head Neck Surg 1997;117:18-21.

2. Patrocinio LG, Damasceno PG, Patrocinio JA. Maxillary mucocele in a 4-month infant. Braz J Otorhinolaryngol 2008;74:479. 3. Hadar T, Shvero J, Nageris BI, Yaniv E. Mucus retention cyst of the maxillary sinus: the endoscopic approach. Br J Oral Maxillofac Surg 2000;38:227-9. 4. Wang JH, Jang YJ, Lee BJ. Natural course of retention cysts of the maxillary sinus: long-term follow-up results. Laryngoscope 2007;117:341-4. 5. Caylakli F, Yavuz H, Cagici AC, Ozluoglu LN. Endoscopic sinus surgery for maxillary sinus mucoceles. Head Face Med 2006;2:29. 6. Fu CH, Chang KP, Lee TJ. The difference in anatomical and invasive characteristics between primary and secondary paranasal sinus mucoceles. Otolaryngol Head Neck Surg 2007;136:621-5. 7. Whyte A, Chapeikin G. Opaque maxillary antrum: a pictorial review. Australas Radiol 2005;49:203-13. 8. Mardinger O, Manor I, Mijiritsky E, Hirshberg A. Maxillary sinus augmentation in the presence of antral pseudocyst: a clinical approach. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007;103:180-4. 9. Har-El G. Endoscopic management of 108 sinus mucoceles. Laryngoscope 2001;111:2131-4. 10. Martin RJ, Jackman DS, Philbert RF, McCoy JM. Massive proptosis of the globe. J Oral Maxillofac Surg 2000;58:794-9. 11. Matheny KE, Duncavage JA. Contemporary indications for the Caldwell-Luc procedure. Curr Opin Otolaryngol Head Neck Surg 2003;11:23-6. 12. Busaba NY, Salman SD. Maxillary sinus mucoceles: clinical presentation and long-term results of endoscopic surgical treatment. Laryngoscope 1999;109:1446-9. 13. Costa F, Emanuelli E, Robiony M, Zerman N, Polini F, Politi M. Endoscopic surgical treatment of chronic maxillary sinusitis of dental origin. J Oral Maxillofac Surg 2007;65:223-8. 14. Chiu AG, Kennedy DW. Disadvantages of minimal techniques for surgical management of chronic rhinosinusitis. Curr Opin Otolaryngol Head Neck Surg 2004;12:38-42. Reprint requests: Rui Figueiredo Facultat d’Odontología Universitat de Barcelona Carrer Feixa Llarga s/n Pavelló Govern, Despatx 2.9 08907 L’Hospitalet de Llobregat Barcelona Spain http://www.ruibf.com [email protected]