Brief Clinical Studies
The Journal of Craniofacial Surgery
Nasal Cavity and Paranasal Sinus Diseases Affecting Orbit Kahraman Serif Samil, Cokkeser Yasar,y Akbay Ercan,z Bayarogullari Hanifi,§ and Kahraman Hilal, MDjj Objective: The aim of the authors was to discuss orbital complications of nasal cavity and paranasal sinus diseases. Material and Method: Patients with nasal and paranasal sinus diseases that affected orbit were retrospectively reviewed. Patients with primary orbital abnormality and those without radiologic orbital signs were excluded. Data regarding age and gender distribution, orbital and ocular findings, radiologic findings, and presence of an additional sinonasal disease were analyzed. Results: Disorders affecting orbit were categorized into 6 categories. Mean age was 41.25 22.14 (range: 6–88) years and male : female ratio was 23 : 18. Overall, there were 41 patients including 11 patients with mucocele, 9 patients with sinusitis, 7 patients with fibrous dysplasia, 4 patients with nasal polyp, 4 patients with paranasal osteoma, and 6 patients with neoplasm. Major clinical presentation was proptosis in these patients. Conclusions: Otolaryngologists should consider the possibility of sinonasal diseases to affect orbit because of vicinity of nasal cavity and paranasal sinuses to orbit. Radiologic imaging is essential to determine the extent, extension, relation with surrounding structures, and initial diagnosis of the disease, and to plan multidisciplinary management. Key Words: Fibrous dysplasia, mucocele, orbit, paranasal osteoma, paranasal sinus, rhinosinusitis
O
rbit is a cavity with a pyramid-like apex at posterior and close vicinity to paranasal sinuses and nasal cavity. It is adjacent to frontal sinus at superior, maxillary sinus at inferior, and nasal cavity and ethmoid sinus at medial. Because of this vicinity, we aimed to present information about which are the most common orbital symptoms those caused by a sinonasal diseases, in what frequency could orbital symptoms be encountered in these diseases, incidence of sinonasal diseases that affect orbit, and outcomes and data of patients who underwent surgery.
MATERIAL AND METHOD Of the patients who were presented to ear-nose-throat (ENT) outpatient clinic with nasal and paranasal disorders, and found to have From the Faculty of Medicine, Department of Otolaryngology-Head and Neck Surgery, Mustafa Kemal University, Antakya; yDepartment of Otolaryngology-Head and Neck Surgery, Gaziantep Primer Hospital, Gaziantep; zDepartment of Otolaryngology-Head and Neck Surgery, Kirikkale University, Kirikkale; §Faculty of Medicine, Department of Radiology, Mustafa Kemal University; and jjDepartment of Ophtalmology, Antakya State Hospital, Antakya, Turkey. Received January 19, 2015. Accepted for publication February 19, 2015. Address correspondence and reprint requests to Kahraman Serif Samil, Faculty of Medicine, Department of Otolaryngology-Head and Neck Surgery, Mustafa Kemal University, Antakya, Turkey;. E-mail: [email protected] The authors report no conflicts of interest. Copyright # 2015 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001827
Volume 26, Number 4, June 2015
orbital symptoms, thus, consulted to ophthalmology department, and the patients who were decided to have orbital symptoms secondary to a primary ENT disorder were included to the study. It was found that 41 patients fulfilled these criteria. Patients with primary orbital disorder or those without positive radiologic findings were excluded. Distribution of age and sex, associated orbital symptoms, comorbid diseases, and radiologic findings were noted. Findings of ENT and eye examinations were recorded in all of the patients; in addition, the findings of postoperative ENT and eye examinations were recorded in patients who underwent surgery. Computed tomography (CT) scans and magnetic resonance imaging were used to identify paranasal diseases causing orbital findings and to determine the extent of diseases. Additional sinonasal disorders, diagnostic biopsies, patients who underwent surgery, recurrent diseases, and referrals for further management radiotherapy (RT ) were comprehensively recorded. Main orbital symptoms included proptosis, eyelid edema, sight disorders, diplopia, limitation in eye movements, and orbital mass. The diseases causing orbital symptoms were classified into 6 categories including mucocele, chronic rhinosinusitis, fibrous dysplasia, nasal polyps, paranasal osteoma, and malignancy.
RESULTS Overall, 41 patients were detected who had a disorder at paranasal and nasal region progressing with orbital findings. It was found that there was mucocele in 11 patients, sinusitis in 9 patients, fibrous dysplasia in 7 patients, nasal polyp in 4 patients, paranasal osteoma in 4 patients, and neoplasm at either nasal cavity or paranasal sinus in 6 patients (Table 1). Given the clinical findings, it was detected that there was proptosis in 23 patients (56.9%), limitation in eye movements in 7 patients (17.07%), eyelid edema in 7 patients (17.07%), diplopia in 5 patients (12.19%), mass at the medial to orbit in 2 patients (4.87%), and loss of vision in 1 patient (2.43%) (Table 2). Mean age was 41.25 22.14 years, ranging from 6 to 88 years. Male : female ratio was 23 : 18 (1.27). Of the 41 patients, 29 patients underwent either nasal or paranasal sinus surgery. In addition, biopsy was performed in 3 patients with suspicious malignity. These patients who were reported to have mesenchymal malignant tumors at advanced stage were referred for RT CT. Additional sinonasal disorders were present in 2 patients with mucocele at maxillary sinus. Of these, 1 had fibrous dysplasia at ethmoid region, whereas other had inverted papilloma. Orbital bone destruction was detected in 11 patients with mucocele as being 5 at frontoethmoid region, 4 at frontal sinus, 1 at maxillary sinus, and 1 at sphenoid sinus. There was proptosis in 10 patients, diplopia in 4 patients, limitation in eye movements in 3 patients, and loss of vision in 1 patient. There was acute mucopyocele in 2 patients (sphenoid and maxillary sinus). In a patient with mucocele at sphenoid sinus, there was loss of vision caused by optic nerve injury because of erosion at the posterior wall. Visual acuity was improved from 1/10 to 9/10 on the week 2 after surgery. TABLE 1. Sinonasal Disorders Affecting Orbit and Their Incidence Sinonasal Disorders Mucocele Sinusitis Fibrous dysplasia Nasal polyp Paranasal osteoma Neoplasm Total
N (%) 11 9 7 4 4 6
(26.82) (21.95) (17.07) (09.75) (09.75) (14.63) 41
# 2015 Mutaz B. Habal, MD e348 Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery
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TABLE 2. Clinical Symptoms and Findings in Patients Ocular Symptoms
N (%)
Proptosis Eyelid edema Limitation in eye movements Diplopia Orbital mass Loss of vision Total
23 7 7 5 2 1 41
(56.09) (17.07) (17.07) (12.19) (4.87) (2.43) (100)
All of the patients underwent surgery and nearly complete resolution was detected in symptoms after surgery. Two years after surgery, a recurrent fibrous dysplasia was detected in 1 patient, whereas distance metastasis in another patient. Overall, there were 9 rhinosinusitis, bacterial in 7 patients and fungal in 2 patients. Of the patients with bacterial rhinosinusitis, subperiosteal abscess was detected in 4 patients, whereas preseptal cellulitis in 3 patients. Endoscopic sinus surgery was performed in 3 of 4 patients with bacterial rhinosinusitis. In the follow-up, it was seen that clinical finding were completely recovered after surgery. Symptoms were recovered after medical treatment in patients with preseptal cellulitis. Erosion was observed at lamina papyracea in 2 patients considered as fungal rhinosinusitis by clinical findings. Mucormycosis was the causative agent in 1 patient, whereas no agent was detected in the other patient. There was proptosis in 4 of the 7 patients diagnosed as fibrous dysplasia. Orbital fossa stenosis was detected in 5 patients; whereas extension to superior orbital fissure in 3 patients and optic canal stenosis in 3 patients. There was mucocele formation at maxillary sinus secondary to fibrous dysplasia in a patient. On the CT scan, orbital fossa stenosis was also detected in the same patient caused by fibrous dysplasia seen at periorbital area. There was an aneurismal bone cyst with fibrous dysplasia at ethmoid sinus in 1 patient. Three patients underwent surgery; however, recurrence was detected in 1 patient. No problem was observed in other patients during follow-up period. Of the 4 patients with nasal polyp, there was proptosis in 1 patient and erosion at lamina papyracea in 3 patients. Polypectomy was performed in 3 patients. Only 1 patient underwent second surgery, which was performed because of polyp rather than orbital disorder. No recurrence was observed in other patients during follow-up. Of the 4 patients with paranasal osteoma causing proptosis, osteoma was localized at medial to orbit in 2 patients, whereas it was localized at apex and medial wall of orbit in 2 patients. Surgical excision was performed in 3 patients but not in 1 patient who refused operation. Symptoms were completely recovered in these patients. Of the 6 patients diagnosed with nasal cavity and paranasal sinus neoplasm (2 epidermoid carcinoma, 1 chordoma, 1 angiofibroma, 1 malignant melanoma, 1 adenoid cystic carcinoma), there was proptosis in 2 patients. Diagnostic biopsy was performed in 3 patients, whereas curative surgery was performed in 3 patients. A distance metastasis was detected at the month 6 after surgery in a patient who underwent surgery. No problem was observed in other patients during follow-up.
Brief Clinical Studies
symptoms such as frontal headache, swelling at upper eyelid, and diplopia are seen in the frontal mucocele.1 Frontoethmoid mucocele may cause proptosis and increased intraocular pressure by mass effect.2 Loss of vision caused by being pushed is termed as dynamic proptosis in literature.3 Sphenoid sinus mucoceles are usually asymptomatic, which are incidentally diagnosed after CT scan or magnetic resonance imaging.4 Frequent clinical findings of sphenoid sinus mucocele are diplopia, proptosis, Horner syndrome, multiple cranial paralyses, and panhypopituitarism.5 It has been thought that mucopyocele content of sphenoid sinus mucocele may cause loss of vision by its neurotoxic effect. In addition, compression by mucocele to optic canal may also result in this finding. Surgical drainage within 24 hours is recommended to the patients in whom visual acuity is worse than finger counting.6 Proptosis and diplopia may be seen in frontoethmoid and frontal sinus mucoceles caused by mass effect.1 Majority of our patients in this group had frontal and frontoethmoid mucoceles (Fig. 1). In agreement to literature, proptosis was the most prominent orbital finding in our patients with mucocele. Acute inflammation of paranasal sinus may cause orbital infection by progression. Of our patients, subperiosteal abscess was detected in 4 patients, whereas preseptal cellulitis in 3 patients. There was no patient with orbital abscess or cavernous sinus thrombosis. The widely accepted point of view in subperiosteal abscess at childhood is that antibiotic therapy should be used in the initial phase of disease.7,8 Patients with loss of vision as first symptom, those with rapid progression in loss of vision over time, and those without improvement in ocular symptoms within 48 hours despite systemic antibiotic therapy are candidates for immediate surgical intervention.8,9 Of the 4 patients with subperiosteal abscess in our study, there were an adult patient and 3 children. In 1 child, no surgical intervention was performed as symptoms were recovered with medical treatment. Surgical intervention was performed in remaining 2 children and 1 adult, as no improvement was observed in ocular within 48 hours despite systemic antibiotic therapy (Fig. 2). Fibrous dysplasia of craniofacial region usually involves maxilla, mandible, sphenoid, ethmoid, and frontal bones and, less frequently, temporal and occipital bones. Firstly, it causes expansion and thinning in these bones; then, followed by sclerosis. As a result, it causes cranial asymmetry and facial deformities. Fibrous dysplasia of orbital and periorbital regions may result in hypertelorism, diplopia, and blindness.10–13
FIGURE 1. A-B, computed tomography and C-D, magnetic resonance images of a frontoethmoid mucocele affecting right orbit from medial and superior.
DISCUSSION Orbital symptoms may occur because of extension of mucocele in paranasal sinuses surrounding orbit. Several symptoms may appear depending on the localization of paranasal sinus. Ophthalmologic #
2015 Mutaz B. Habal, MD
FIGURE 2. A, A patient with a subperiosteal abscess at left orbit; B, computed tomography and C-D, magnetic resonance images of the same patient.
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Brief Clinical Studies
The Journal of Craniofacial Surgery
FIGURE 3. A-C, Computed tomography images in a patient with fibrous dysplasia extended to medial to orbit and skull base.
Surgical indications in fibrous dysplasia include vision disorders caused by optic nerve compression, alteration of globe position, and occurrence of great facial deformities caused by extension of disease. Radical resection of fibro-osseous tissue, decompression of optic canal, and graft reconstruction of resection site are performed in the surgical treatment. Recurrence may be seen during follow-up. In the fronto-orbital fibrous dysplasia, devastating outcomes such as blindness may occur because of higher recurrence rate, particularly after incomplete resection. Detection of scotoma in visual field is an indicator suggesting that optic nerve is affected before onset of visual symptoms. Vision is an important predictor and visual impairment is usually irreversible. Thus, a radical and comprehensive approach is recommended in the treatment of fibrous dysplasia.14 Surgical intervention was performed because of a lesion adjacent to optic nerve in 1 patient, erosion at orbital wall (Fig. 3) and extension to skull base in another patient, and erosion at orbital wall and fibrous dysplasia associated by a mucocele in a distinct patient. There was recurrence in the patient with fibrous dysplasia associated by mucocele, whereas no recurrence was observed in remaining 2 patients. Reoperation was recommended to patient with recurrence; however, patient did not accept the surgery. In the nasal polyp, weak areas may occur at orbit after surgical intervention as a result of weakened supporting tissue and symptoms may develop because of recurrent inflammations. We also observed proptosis in the patient with recurrence caused by weakened tissue. Paranasal sinus osteoma is usually detected as incidentaloma on CT scan performed after complaints including chronic sinusitis, hyposmia, or pressure sense on face, as it expresses a slow growth pattern. An osteoma may remain asymptomatic unless it is large enough to close sinus ostium or destructs close areas such as orbit or cranium. Osteomas rarely involve orbit, which occurs via direct extension from sinus adjacent to orbit. It causes proptosis and diplopia by compressing orbit.15 Surgical treatment is rarely indicated in an asymptomatic patient. It is performed in the presence of cosmetic problems related to osteoma, in osteomas with a diameter enough to cause mucocele and chronic sinusitis, and in those causing orbital or intracranial complications.16,17 It can also be done, however, in young patients to prevent possible long-term complications of the lesions adjacent to critical anatomic structures.
FIGURE 4. A-C, Computed tomography and D, three-dimensional computed tomography images of an osteoma extending into orbit from right frontal sinus.
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Endoscopic or external surgical interventions are preferred in the treatment of osteoma. Selection of surgical technique depends on the size and localization of osteoma and experience of surgeon. Although external surgery is gold standard in the treatment of frontal sinus osteomas, endoscopic treatment becomes easier with advances in surgical instruments (drill and angled-telescopes, etc.) in recent years.9,18 External surgery was performed in 3 patients because of osteoma localized at frontal sinus and large size osteoma (Fig. 4). In these patients, no recurrence was seen at follow-up period. In the endonasal surgeries, attempts to improve surgical exposure may interfere to natural drainage paths and normal anatomy. Additionally, endoscopy use may prove helpful in improving vision during external surgical resection of frontal sinus osteomas.19 Sinonasal malign tumors may cause orbital complications when bone erosion occurs because of their extension.20 In sinonasal malignancies, incidence of orbital invasion is related to areas where tumor arises, tumor histology, and tumor pathology (Fig. 5). Although frequency of orbital finding varies depending on the area where tumor arises, epiphora, proptosis, and diplopia may be seen in 50% of the patients.21 Displacement of globe provides information about tumor localization. For example, globe will displace to inferior in tumors originating from frontal sinus, whereas to lateral in ethmoid sinus tumors. Some tumors, such as chondrosarcoma, can result in bilateral blindness because of tendency of infiltration to skull base. Sinonasal tumors reach orbit by either neurovascular route or local invasion. Local invasion of tumors are frequently seen in ethmoid tumors because of its thin lamina papyracea. Orbital wall invasion rate varies from 66% to 82% in ethmoid tumors.22,23 This rate ranges from 60% to 80% in maxillary sinus tumors.24 Orbital periosteum is a good barrier against tumor invasion. In the literature, there is no study assessing disorders of paranasal region causing orbital complications together that were discussed by classifying into 6 categories in our study. There are studies demonstrating orbital effects of each disorder. In the current study, we aimed to present comprehensive data by assessing sinonasal disorders collectively. Malik et al25 evaluated 54 patients with a mean age of 45 years. Authors classified these patients as follows: sinusitis (64.81%), neoplasm (20.4%), nasal polyp (7.4%), granulomatous infection (5.56%), and mucocele (1.85%). In that study, the most common clinical findings were found as proptosis (66.6%), visual disorder (25.9%), limitation of eye movements (11.1%), and diplopia (9.26%).26 In our study, proptosis was the major clinical finding in agreement to that study; however, the largest patient group consisted of patients with mucocele in our study. Fibrous dysplasia and osteoma were discussed in our study, which have not been mentioned in the study by Malik et al; however, we had no patient presenting signs of granulomatous infection.
CONCLUSION Orbital complications may be commonly observed because of vicinity of paranasal sinus and nasal cavity to orbit. Orbital status
FIGURE 5. A-B, Magnetic resonance and C-D, computed tomography images of a malign melanoma extending to medial and inferior parts of orbit.
#
2015 Mutaz B. Habal, MD
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery
Volume 26, Number 4, June 2015
should be taken into consideration while planning the medical or surgical management in primary disorders of paranasal sinus and nasal cavity. Moreover, an ophthalmologist should remember that there may be a sinonasal disorder while investigating etiopathogenesis in patients with proptosis, diplopia, epiphora, or loss of vision. Orbital invasion is an indicator of poorer prognosis and directly influences planning of surgical process. In addition, presence of orbital involvement significantly decreases the survival in ethmoid and maxillary sinus tumors. It is important to detect paranasal sinus diseases, such as osteoma and fibrous dysplasia, by radiology before orbital invasion. Early surgical resection can prevent orbital complications in these diseases. Symptoms that patients may present should be kept in mind and meticulous assessment should be performed for surgery. Radiologic imaging is also essential to plan multidisciplinary management.
REFERENCES 1. Herndon M, McMains KC, Kountakis SE. Presentation and management of extensivefronto-orbital-ethmoid mucoceles. Am J Otolaryngol 2007;28:145–147 2. Loo JL, Looi AL, Seah LL. Visual outcomes in patients with paranasal mucoceles. Ophthal Plast Reconstr Surg 2009;25:126–129 3. Bullock JD, Bartley GB. Dynamic proptosis. Am J Ophtalmol 1986;102:104–110 4. Friedman A, Batra PS, Fakhri S, et al. Isolated sphenoid sinus disease: etiology and management. Otolaryngol Head and Neck Surg 2005;133:544–550 5. Sundar U, Sharma AL, Yeolekar ME, et al. Sphenoidal sinus mucocoele presenting as mono-ocular painless loss of vision. Postgrad Med J 2004;80:40 6. Yumoto E, Hyodo M, Kawakita S, et al. Effect of sinus surgery on visual disturbance caused by spheno-ethmoid mucoceles. Am J Rhinol 1997;11:337–343 7. Rahbar R, Robson CD, Petersen RA, et al. Management of orbital subperiosteal abscess in children. Arch Otolaryngol Head Neck Surg 2001;127:281–286 8. Goodwin WJ Jr, Weinshall M, Chandler JR. The role of highresolution computerized tomography and standardized ultrasound in the evaluation of orbital cellulitis. Laryngoscope 1982;92:729–731 9. Souliere CR Jr, Antoine GA, Martin MP, et al. Selective non-surgical management of subperiosteal abscess of the orbit: computerized tomograph and clinical course as indication for surgical drainage. Int J Pediatr Otorhinol 1990;19:109–119 10. Altuna X, Gorostiaga F, Algaba J. Fibrous monostotic dysplasia of frontal sinus. About a case. ORL-DIPS 2004;31:84–87 11. Alves AL, Canavarros F, Vilela DS, et al. Fibrous displasia: Report of three cases. Rev Bras Otorrinol 2002;68:288–292 12. Lustig LR, Holliday MJ, McCarthy EF, et al. Fibrous dysplasia involving the skull base and temporal bone. Arch Otolaryngol Head Neck Surg 2001;127:1239–1247 ´ , Rodrı´guez-Pereira C, et al. Frontal sinus 13. Fuster MA, Martı´n JA monostotic fibrous dysplasia with orbital extension. Acta Otorrinolaringol Esp 2002;53:203–206 14. Yavuzer R, Bone H, Jackson IT. Fronto-orbital fibrous dysplasia. Orbit 2000;19:119–128 15. Gillman GS, Lampe HB, Allen LH. Orbitoethmoid osteoma: case report of an uncommon presentation of an uncommon tumour. Otolaryngol Head Neck Surg 1997;117:218–220 16. Eller R, Sillers M. Common fibro-osseous lesions of the paranasal sinuses. Otolaryngol Clin North Am 2006;39:585–600 17. Schick B, Steigerwald C, el Rehman el Tahan A, et al. The role ofendonasal surgery in the management of frontoethmoidal osteomas. Rhinology 2001;39:66–70 18. Chiu AG, Schipor I, Cohen NA, et al. Surgical decisions in the management of frontal sinus osteomas. Am J Rhinol 2005;19:191–197 19. Bignami M, Dallan I, Terranova P, et al. Frontal sinus osteomas: the window of endonasal endoscopic approach. Rhinology 2007;45:315– 320 #
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20. Keane WM, Atkins JP Jr, Wetmore R, et al. Epidemiology of head and neck cancer. Laryngoscope 1981;91:2037–2045 21. Lund VJ. Malignant tumours of the nasal cavity and paranasal sinuses. ORL J Otorhinolaryngol Relat Spec 1983;45:1–12 22. Iannetti G, Valentini V, Rinna C, et al. Ethmoido-orbital tumors: our experience. J Craniofac Surg 2005;16:1085–1091 23. Ganly I, Patel SG, Singh B, et al. Craniofacial resection for malignant paranasal sinus tumors: report of an international collaborative study. Head Neck 2005;27:575–584 24. Carrau RL, Segas J, Nuss DW, et al. Squamous cell carcinoma of the sinonasal tract invading the orbit. Laryngoscope 1999;109:230–235 25. Malik TG, Rashid A, Farooq H. Paranasal sinuses and nasal cavity; the notorious neighbours of orbit. Professional Med J Mar 2011;18: 154–159
Osteomyelitis of the Temporomandibular Joint Following Canal Wall Down Mastoidectomy Chang-Woo Kim Abstract: Osteomyelitis of the temporomandibular joint (TMJ) is a rare disease that is characterized by osteitis of the mandibular condyle or glenoid fossa. Although this condition is usually associated with trauma, dental disease, or extension of a local infection, it may also occur as a complication of otologic infection. However, osteomyelitis of the TMJ as a complication of otologic surgery has not yet been reported. I present the case of a 54-year-old woman who developed a polyp on the superior portion of the tympanic membrane 9 weeks after canal wall down mastoidectomy. This is an extremely rare case of a patient with osteomyelitis of the TMJ as a complication of otologic surgery. Key Words: Mastoidectomy, osteomyelitis, postoperative complication, temporomandibular joint
INTRODUCTION
O
steomyelitis of the temporomandibular joint (TMJ) is a rare disease.1 It is usually associated with trauma, dental disease, or bacterial seeding through direct extension of local infection. Despite the relative proximity of the TMJ to the external auditory canal (EAC), TMJ involvement is a rare complication of otologic infection.2,3 Among otologic infections, malignant otitis externa is the most common cause of osteomyelitis of the TMJ.2,4,5 Chronic From the Department of Otorhinolaryngology-Head & Neck Surgery, College of Medicine, Hallym University, Seoul, Korea. Received August 31, 2014. Accepted for publication February 19, 2015. Address correspondence and reprint requests to Chang-Woo Kim, MD, Associate Professor, 445 Gil-Dong, Gangdong-gu, Department of Otorhinolaryngology-Head & Neck Surgery, Hallym University College of Medicine, Seoul 134–701, Korea;. E-mail: [email protected] The authors report no conflicts of interest. Copyright # 2015 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001821
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Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery
Nasal Cavity and Paranasal Sinus Diseases Affecting Orbit Kahraman Serif Samil, Cokkeser Yasar,y Akbay Ercan,z Bayarogullari Hanifi,§ and Kahraman Hilal, MDjj Objective: The aim of the authors was to discuss orbital complications of nasal cavity and paranasal sinus diseases. Material and Method: Patients with nasal and paranasal sinus diseases that affected orbit were retrospectively reviewed. Patients with primary orbital abnormality and those without radiologic orbital signs were excluded. Data regarding age and gender distribution, orbital and ocular findings, radiologic findings, and presence of an additional sinonasal disease were analyzed. Results: Disorders affecting orbit were categorized into 6 categories. Mean age was 41.25 22.14 (range: 6–88) years and male : female ratio was 23 : 18. Overall, there were 41 patients including 11 patients with mucocele, 9 patients with sinusitis, 7 patients with fibrous dysplasia, 4 patients with nasal polyp, 4 patients with paranasal osteoma, and 6 patients with neoplasm. Major clinical presentation was proptosis in these patients. Conclusions: Otolaryngologists should consider the possibility of sinonasal diseases to affect orbit because of vicinity of nasal cavity and paranasal sinuses to orbit. Radiologic imaging is essential to determine the extent, extension, relation with surrounding structures, and initial diagnosis of the disease, and to plan multidisciplinary management. Key Words: Fibrous dysplasia, mucocele, orbit, paranasal osteoma, paranasal sinus, rhinosinusitis
O
rbit is a cavity with a pyramid-like apex at posterior and close vicinity to paranasal sinuses and nasal cavity. It is adjacent to frontal sinus at superior, maxillary sinus at inferior, and nasal cavity and ethmoid sinus at medial. Because of this vicinity, we aimed to present information about which are the most common orbital symptoms those caused by a sinonasal diseases, in what frequency could orbital symptoms be encountered in these diseases, incidence of sinonasal diseases that affect orbit, and outcomes and data of patients who underwent surgery.
MATERIAL AND METHOD Of the patients who were presented to ear-nose-throat (ENT) outpatient clinic with nasal and paranasal disorders, and found to have From the Faculty of Medicine, Department of Otolaryngology-Head and Neck Surgery, Mustafa Kemal University, Antakya; yDepartment of Otolaryngology-Head and Neck Surgery, Gaziantep Primer Hospital, Gaziantep; zDepartment of Otolaryngology-Head and Neck Surgery, Kirikkale University, Kirikkale; §Faculty of Medicine, Department of Radiology, Mustafa Kemal University; and jjDepartment of Ophtalmology, Antakya State Hospital, Antakya, Turkey. Received January 19, 2015. Accepted for publication February 19, 2015. Address correspondence and reprint requests to Kahraman Serif Samil, Faculty of Medicine, Department of Otolaryngology-Head and Neck Surgery, Mustafa Kemal University, Antakya, Turkey;. E-mail: [email protected] The authors report no conflicts of interest. Copyright # 2015 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001827
Volume 26, Number 4, June 2015
orbital symptoms, thus, consulted to ophthalmology department, and the patients who were decided to have orbital symptoms secondary to a primary ENT disorder were included to the study. It was found that 41 patients fulfilled these criteria. Patients with primary orbital disorder or those without positive radiologic findings were excluded. Distribution of age and sex, associated orbital symptoms, comorbid diseases, and radiologic findings were noted. Findings of ENT and eye examinations were recorded in all of the patients; in addition, the findings of postoperative ENT and eye examinations were recorded in patients who underwent surgery. Computed tomography (CT) scans and magnetic resonance imaging were used to identify paranasal diseases causing orbital findings and to determine the extent of diseases. Additional sinonasal disorders, diagnostic biopsies, patients who underwent surgery, recurrent diseases, and referrals for further management radiotherapy (RT ) were comprehensively recorded. Main orbital symptoms included proptosis, eyelid edema, sight disorders, diplopia, limitation in eye movements, and orbital mass. The diseases causing orbital symptoms were classified into 6 categories including mucocele, chronic rhinosinusitis, fibrous dysplasia, nasal polyps, paranasal osteoma, and malignancy.
RESULTS Overall, 41 patients were detected who had a disorder at paranasal and nasal region progressing with orbital findings. It was found that there was mucocele in 11 patients, sinusitis in 9 patients, fibrous dysplasia in 7 patients, nasal polyp in 4 patients, paranasal osteoma in 4 patients, and neoplasm at either nasal cavity or paranasal sinus in 6 patients (Table 1). Given the clinical findings, it was detected that there was proptosis in 23 patients (56.9%), limitation in eye movements in 7 patients (17.07%), eyelid edema in 7 patients (17.07%), diplopia in 5 patients (12.19%), mass at the medial to orbit in 2 patients (4.87%), and loss of vision in 1 patient (2.43%) (Table 2). Mean age was 41.25 22.14 years, ranging from 6 to 88 years. Male : female ratio was 23 : 18 (1.27). Of the 41 patients, 29 patients underwent either nasal or paranasal sinus surgery. In addition, biopsy was performed in 3 patients with suspicious malignity. These patients who were reported to have mesenchymal malignant tumors at advanced stage were referred for RT CT. Additional sinonasal disorders were present in 2 patients with mucocele at maxillary sinus. Of these, 1 had fibrous dysplasia at ethmoid region, whereas other had inverted papilloma. Orbital bone destruction was detected in 11 patients with mucocele as being 5 at frontoethmoid region, 4 at frontal sinus, 1 at maxillary sinus, and 1 at sphenoid sinus. There was proptosis in 10 patients, diplopia in 4 patients, limitation in eye movements in 3 patients, and loss of vision in 1 patient. There was acute mucopyocele in 2 patients (sphenoid and maxillary sinus). In a patient with mucocele at sphenoid sinus, there was loss of vision caused by optic nerve injury because of erosion at the posterior wall. Visual acuity was improved from 1/10 to 9/10 on the week 2 after surgery. TABLE 1. Sinonasal Disorders Affecting Orbit and Their Incidence Sinonasal Disorders Mucocele Sinusitis Fibrous dysplasia Nasal polyp Paranasal osteoma Neoplasm Total
N (%) 11 9 7 4 4 6
(26.82) (21.95) (17.07) (09.75) (09.75) (14.63) 41
# 2015 Mutaz B. Habal, MD e348 Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery
Volume 26, Number 4, June 2015
TABLE 2. Clinical Symptoms and Findings in Patients Ocular Symptoms
N (%)
Proptosis Eyelid edema Limitation in eye movements Diplopia Orbital mass Loss of vision Total
23 7 7 5 2 1 41
(56.09) (17.07) (17.07) (12.19) (4.87) (2.43) (100)
All of the patients underwent surgery and nearly complete resolution was detected in symptoms after surgery. Two years after surgery, a recurrent fibrous dysplasia was detected in 1 patient, whereas distance metastasis in another patient. Overall, there were 9 rhinosinusitis, bacterial in 7 patients and fungal in 2 patients. Of the patients with bacterial rhinosinusitis, subperiosteal abscess was detected in 4 patients, whereas preseptal cellulitis in 3 patients. Endoscopic sinus surgery was performed in 3 of 4 patients with bacterial rhinosinusitis. In the follow-up, it was seen that clinical finding were completely recovered after surgery. Symptoms were recovered after medical treatment in patients with preseptal cellulitis. Erosion was observed at lamina papyracea in 2 patients considered as fungal rhinosinusitis by clinical findings. Mucormycosis was the causative agent in 1 patient, whereas no agent was detected in the other patient. There was proptosis in 4 of the 7 patients diagnosed as fibrous dysplasia. Orbital fossa stenosis was detected in 5 patients; whereas extension to superior orbital fissure in 3 patients and optic canal stenosis in 3 patients. There was mucocele formation at maxillary sinus secondary to fibrous dysplasia in a patient. On the CT scan, orbital fossa stenosis was also detected in the same patient caused by fibrous dysplasia seen at periorbital area. There was an aneurismal bone cyst with fibrous dysplasia at ethmoid sinus in 1 patient. Three patients underwent surgery; however, recurrence was detected in 1 patient. No problem was observed in other patients during follow-up period. Of the 4 patients with nasal polyp, there was proptosis in 1 patient and erosion at lamina papyracea in 3 patients. Polypectomy was performed in 3 patients. Only 1 patient underwent second surgery, which was performed because of polyp rather than orbital disorder. No recurrence was observed in other patients during follow-up. Of the 4 patients with paranasal osteoma causing proptosis, osteoma was localized at medial to orbit in 2 patients, whereas it was localized at apex and medial wall of orbit in 2 patients. Surgical excision was performed in 3 patients but not in 1 patient who refused operation. Symptoms were completely recovered in these patients. Of the 6 patients diagnosed with nasal cavity and paranasal sinus neoplasm (2 epidermoid carcinoma, 1 chordoma, 1 angiofibroma, 1 malignant melanoma, 1 adenoid cystic carcinoma), there was proptosis in 2 patients. Diagnostic biopsy was performed in 3 patients, whereas curative surgery was performed in 3 patients. A distance metastasis was detected at the month 6 after surgery in a patient who underwent surgery. No problem was observed in other patients during follow-up.
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symptoms such as frontal headache, swelling at upper eyelid, and diplopia are seen in the frontal mucocele.1 Frontoethmoid mucocele may cause proptosis and increased intraocular pressure by mass effect.2 Loss of vision caused by being pushed is termed as dynamic proptosis in literature.3 Sphenoid sinus mucoceles are usually asymptomatic, which are incidentally diagnosed after CT scan or magnetic resonance imaging.4 Frequent clinical findings of sphenoid sinus mucocele are diplopia, proptosis, Horner syndrome, multiple cranial paralyses, and panhypopituitarism.5 It has been thought that mucopyocele content of sphenoid sinus mucocele may cause loss of vision by its neurotoxic effect. In addition, compression by mucocele to optic canal may also result in this finding. Surgical drainage within 24 hours is recommended to the patients in whom visual acuity is worse than finger counting.6 Proptosis and diplopia may be seen in frontoethmoid and frontal sinus mucoceles caused by mass effect.1 Majority of our patients in this group had frontal and frontoethmoid mucoceles (Fig. 1). In agreement to literature, proptosis was the most prominent orbital finding in our patients with mucocele. Acute inflammation of paranasal sinus may cause orbital infection by progression. Of our patients, subperiosteal abscess was detected in 4 patients, whereas preseptal cellulitis in 3 patients. There was no patient with orbital abscess or cavernous sinus thrombosis. The widely accepted point of view in subperiosteal abscess at childhood is that antibiotic therapy should be used in the initial phase of disease.7,8 Patients with loss of vision as first symptom, those with rapid progression in loss of vision over time, and those without improvement in ocular symptoms within 48 hours despite systemic antibiotic therapy are candidates for immediate surgical intervention.8,9 Of the 4 patients with subperiosteal abscess in our study, there were an adult patient and 3 children. In 1 child, no surgical intervention was performed as symptoms were recovered with medical treatment. Surgical intervention was performed in remaining 2 children and 1 adult, as no improvement was observed in ocular within 48 hours despite systemic antibiotic therapy (Fig. 2). Fibrous dysplasia of craniofacial region usually involves maxilla, mandible, sphenoid, ethmoid, and frontal bones and, less frequently, temporal and occipital bones. Firstly, it causes expansion and thinning in these bones; then, followed by sclerosis. As a result, it causes cranial asymmetry and facial deformities. Fibrous dysplasia of orbital and periorbital regions may result in hypertelorism, diplopia, and blindness.10–13
FIGURE 1. A-B, computed tomography and C-D, magnetic resonance images of a frontoethmoid mucocele affecting right orbit from medial and superior.
DISCUSSION Orbital symptoms may occur because of extension of mucocele in paranasal sinuses surrounding orbit. Several symptoms may appear depending on the localization of paranasal sinus. Ophthalmologic #
2015 Mutaz B. Habal, MD
FIGURE 2. A, A patient with a subperiosteal abscess at left orbit; B, computed tomography and C-D, magnetic resonance images of the same patient.
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The Journal of Craniofacial Surgery
FIGURE 3. A-C, Computed tomography images in a patient with fibrous dysplasia extended to medial to orbit and skull base.
Surgical indications in fibrous dysplasia include vision disorders caused by optic nerve compression, alteration of globe position, and occurrence of great facial deformities caused by extension of disease. Radical resection of fibro-osseous tissue, decompression of optic canal, and graft reconstruction of resection site are performed in the surgical treatment. Recurrence may be seen during follow-up. In the fronto-orbital fibrous dysplasia, devastating outcomes such as blindness may occur because of higher recurrence rate, particularly after incomplete resection. Detection of scotoma in visual field is an indicator suggesting that optic nerve is affected before onset of visual symptoms. Vision is an important predictor and visual impairment is usually irreversible. Thus, a radical and comprehensive approach is recommended in the treatment of fibrous dysplasia.14 Surgical intervention was performed because of a lesion adjacent to optic nerve in 1 patient, erosion at orbital wall (Fig. 3) and extension to skull base in another patient, and erosion at orbital wall and fibrous dysplasia associated by a mucocele in a distinct patient. There was recurrence in the patient with fibrous dysplasia associated by mucocele, whereas no recurrence was observed in remaining 2 patients. Reoperation was recommended to patient with recurrence; however, patient did not accept the surgery. In the nasal polyp, weak areas may occur at orbit after surgical intervention as a result of weakened supporting tissue and symptoms may develop because of recurrent inflammations. We also observed proptosis in the patient with recurrence caused by weakened tissue. Paranasal sinus osteoma is usually detected as incidentaloma on CT scan performed after complaints including chronic sinusitis, hyposmia, or pressure sense on face, as it expresses a slow growth pattern. An osteoma may remain asymptomatic unless it is large enough to close sinus ostium or destructs close areas such as orbit or cranium. Osteomas rarely involve orbit, which occurs via direct extension from sinus adjacent to orbit. It causes proptosis and diplopia by compressing orbit.15 Surgical treatment is rarely indicated in an asymptomatic patient. It is performed in the presence of cosmetic problems related to osteoma, in osteomas with a diameter enough to cause mucocele and chronic sinusitis, and in those causing orbital or intracranial complications.16,17 It can also be done, however, in young patients to prevent possible long-term complications of the lesions adjacent to critical anatomic structures.
FIGURE 4. A-C, Computed tomography and D, three-dimensional computed tomography images of an osteoma extending into orbit from right frontal sinus.
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Endoscopic or external surgical interventions are preferred in the treatment of osteoma. Selection of surgical technique depends on the size and localization of osteoma and experience of surgeon. Although external surgery is gold standard in the treatment of frontal sinus osteomas, endoscopic treatment becomes easier with advances in surgical instruments (drill and angled-telescopes, etc.) in recent years.9,18 External surgery was performed in 3 patients because of osteoma localized at frontal sinus and large size osteoma (Fig. 4). In these patients, no recurrence was seen at follow-up period. In the endonasal surgeries, attempts to improve surgical exposure may interfere to natural drainage paths and normal anatomy. Additionally, endoscopy use may prove helpful in improving vision during external surgical resection of frontal sinus osteomas.19 Sinonasal malign tumors may cause orbital complications when bone erosion occurs because of their extension.20 In sinonasal malignancies, incidence of orbital invasion is related to areas where tumor arises, tumor histology, and tumor pathology (Fig. 5). Although frequency of orbital finding varies depending on the area where tumor arises, epiphora, proptosis, and diplopia may be seen in 50% of the patients.21 Displacement of globe provides information about tumor localization. For example, globe will displace to inferior in tumors originating from frontal sinus, whereas to lateral in ethmoid sinus tumors. Some tumors, such as chondrosarcoma, can result in bilateral blindness because of tendency of infiltration to skull base. Sinonasal tumors reach orbit by either neurovascular route or local invasion. Local invasion of tumors are frequently seen in ethmoid tumors because of its thin lamina papyracea. Orbital wall invasion rate varies from 66% to 82% in ethmoid tumors.22,23 This rate ranges from 60% to 80% in maxillary sinus tumors.24 Orbital periosteum is a good barrier against tumor invasion. In the literature, there is no study assessing disorders of paranasal region causing orbital complications together that were discussed by classifying into 6 categories in our study. There are studies demonstrating orbital effects of each disorder. In the current study, we aimed to present comprehensive data by assessing sinonasal disorders collectively. Malik et al25 evaluated 54 patients with a mean age of 45 years. Authors classified these patients as follows: sinusitis (64.81%), neoplasm (20.4%), nasal polyp (7.4%), granulomatous infection (5.56%), and mucocele (1.85%). In that study, the most common clinical findings were found as proptosis (66.6%), visual disorder (25.9%), limitation of eye movements (11.1%), and diplopia (9.26%).26 In our study, proptosis was the major clinical finding in agreement to that study; however, the largest patient group consisted of patients with mucocele in our study. Fibrous dysplasia and osteoma were discussed in our study, which have not been mentioned in the study by Malik et al; however, we had no patient presenting signs of granulomatous infection.
CONCLUSION Orbital complications may be commonly observed because of vicinity of paranasal sinus and nasal cavity to orbit. Orbital status
FIGURE 5. A-B, Magnetic resonance and C-D, computed tomography images of a malign melanoma extending to medial and inferior parts of orbit.
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2015 Mutaz B. Habal, MD
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should be taken into consideration while planning the medical or surgical management in primary disorders of paranasal sinus and nasal cavity. Moreover, an ophthalmologist should remember that there may be a sinonasal disorder while investigating etiopathogenesis in patients with proptosis, diplopia, epiphora, or loss of vision. Orbital invasion is an indicator of poorer prognosis and directly influences planning of surgical process. In addition, presence of orbital involvement significantly decreases the survival in ethmoid and maxillary sinus tumors. It is important to detect paranasal sinus diseases, such as osteoma and fibrous dysplasia, by radiology before orbital invasion. Early surgical resection can prevent orbital complications in these diseases. Symptoms that patients may present should be kept in mind and meticulous assessment should be performed for surgery. Radiologic imaging is also essential to plan multidisciplinary management.
REFERENCES 1. Herndon M, McMains KC, Kountakis SE. Presentation and management of extensivefronto-orbital-ethmoid mucoceles. Am J Otolaryngol 2007;28:145–147 2. Loo JL, Looi AL, Seah LL. Visual outcomes in patients with paranasal mucoceles. Ophthal Plast Reconstr Surg 2009;25:126–129 3. Bullock JD, Bartley GB. Dynamic proptosis. Am J Ophtalmol 1986;102:104–110 4. Friedman A, Batra PS, Fakhri S, et al. Isolated sphenoid sinus disease: etiology and management. Otolaryngol Head and Neck Surg 2005;133:544–550 5. Sundar U, Sharma AL, Yeolekar ME, et al. Sphenoidal sinus mucocoele presenting as mono-ocular painless loss of vision. Postgrad Med J 2004;80:40 6. Yumoto E, Hyodo M, Kawakita S, et al. Effect of sinus surgery on visual disturbance caused by spheno-ethmoid mucoceles. Am J Rhinol 1997;11:337–343 7. Rahbar R, Robson CD, Petersen RA, et al. Management of orbital subperiosteal abscess in children. Arch Otolaryngol Head Neck Surg 2001;127:281–286 8. Goodwin WJ Jr, Weinshall M, Chandler JR. The role of highresolution computerized tomography and standardized ultrasound in the evaluation of orbital cellulitis. Laryngoscope 1982;92:729–731 9. Souliere CR Jr, Antoine GA, Martin MP, et al. Selective non-surgical management of subperiosteal abscess of the orbit: computerized tomograph and clinical course as indication for surgical drainage. Int J Pediatr Otorhinol 1990;19:109–119 10. Altuna X, Gorostiaga F, Algaba J. Fibrous monostotic dysplasia of frontal sinus. About a case. ORL-DIPS 2004;31:84–87 11. Alves AL, Canavarros F, Vilela DS, et al. Fibrous displasia: Report of three cases. Rev Bras Otorrinol 2002;68:288–292 12. Lustig LR, Holliday MJ, McCarthy EF, et al. Fibrous dysplasia involving the skull base and temporal bone. Arch Otolaryngol Head Neck Surg 2001;127:1239–1247 ´ , Rodrı´guez-Pereira C, et al. Frontal sinus 13. Fuster MA, Martı´n JA monostotic fibrous dysplasia with orbital extension. Acta Otorrinolaringol Esp 2002;53:203–206 14. Yavuzer R, Bone H, Jackson IT. Fronto-orbital fibrous dysplasia. Orbit 2000;19:119–128 15. Gillman GS, Lampe HB, Allen LH. Orbitoethmoid osteoma: case report of an uncommon presentation of an uncommon tumour. Otolaryngol Head Neck Surg 1997;117:218–220 16. Eller R, Sillers M. Common fibro-osseous lesions of the paranasal sinuses. Otolaryngol Clin North Am 2006;39:585–600 17. Schick B, Steigerwald C, el Rehman el Tahan A, et al. The role ofendonasal surgery in the management of frontoethmoidal osteomas. Rhinology 2001;39:66–70 18. Chiu AG, Schipor I, Cohen NA, et al. Surgical decisions in the management of frontal sinus osteomas. Am J Rhinol 2005;19:191–197 19. Bignami M, Dallan I, Terranova P, et al. Frontal sinus osteomas: the window of endonasal endoscopic approach. Rhinology 2007;45:315– 320 #
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20. Keane WM, Atkins JP Jr, Wetmore R, et al. Epidemiology of head and neck cancer. Laryngoscope 1981;91:2037–2045 21. Lund VJ. Malignant tumours of the nasal cavity and paranasal sinuses. ORL J Otorhinolaryngol Relat Spec 1983;45:1–12 22. Iannetti G, Valentini V, Rinna C, et al. Ethmoido-orbital tumors: our experience. J Craniofac Surg 2005;16:1085–1091 23. Ganly I, Patel SG, Singh B, et al. Craniofacial resection for malignant paranasal sinus tumors: report of an international collaborative study. Head Neck 2005;27:575–584 24. Carrau RL, Segas J, Nuss DW, et al. Squamous cell carcinoma of the sinonasal tract invading the orbit. Laryngoscope 1999;109:230–235 25. Malik TG, Rashid A, Farooq H. Paranasal sinuses and nasal cavity; the notorious neighbours of orbit. Professional Med J Mar 2011;18: 154–159
Osteomyelitis of the Temporomandibular Joint Following Canal Wall Down Mastoidectomy Chang-Woo Kim Abstract: Osteomyelitis of the temporomandibular joint (TMJ) is a rare disease that is characterized by osteitis of the mandibular condyle or glenoid fossa. Although this condition is usually associated with trauma, dental disease, or extension of a local infection, it may also occur as a complication of otologic infection. However, osteomyelitis of the TMJ as a complication of otologic surgery has not yet been reported. I present the case of a 54-year-old woman who developed a polyp on the superior portion of the tympanic membrane 9 weeks after canal wall down mastoidectomy. This is an extremely rare case of a patient with osteomyelitis of the TMJ as a complication of otologic surgery. Key Words: Mastoidectomy, osteomyelitis, postoperative complication, temporomandibular joint
INTRODUCTION
O
steomyelitis of the temporomandibular joint (TMJ) is a rare disease.1 It is usually associated with trauma, dental disease, or bacterial seeding through direct extension of local infection. Despite the relative proximity of the TMJ to the external auditory canal (EAC), TMJ involvement is a rare complication of otologic infection.2,3 Among otologic infections, malignant otitis externa is the most common cause of osteomyelitis of the TMJ.2,4,5 Chronic From the Department of Otorhinolaryngology-Head & Neck Surgery, College of Medicine, Hallym University, Seoul, Korea. Received August 31, 2014. Accepted for publication February 19, 2015. Address correspondence and reprint requests to Chang-Woo Kim, MD, Associate Professor, 445 Gil-Dong, Gangdong-gu, Department of Otorhinolaryngology-Head & Neck Surgery, Hallym University College of Medicine, Seoul 134–701, Korea;. E-mail: [email protected] The authors report no conflicts of interest. Copyright # 2015 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001821
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