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Isolated sphenoid sinus disease: An overlooked cause of headache
Journal of Cranio-Maxillo-Facial Surgery xxx (2015) 1e4
Contents lists available at ScienceDirect
Journal of Cranio-Maxillo-Facial Surgery journal homepage: www.jcmfs.com
Isolated sphenoid sinus disease: An overlooked cause of headache Fatih Celenk*, Secaattin Gulsen, Burhanettin Gonuldas, Elif Baysal, Cengiz Durucu, Muzaffer Kanlikama, Semih Mumbuc Department of Otorhinolaryngology, Gaziantep University Faculty of Medicine, Gaziantep, Turkey
a r t i c l e i n f o
a b s t r a c t
Article history: Paper received 3 June 2015 Accepted 26 August 2015 Available online xxx
Objectives: The aim of this study was to evaluate patients who underwent endoscopic sphenoid sinus surgery for isolated sphenoid sinus disease. We also investigated the impact of sphenoid sinus surgery on headache intensity. Material and methods: Twenty-one consecutive patients who underwent endoscopic sphenoidotomy for isolated sphenoid sinus disease were included in the study. Diagnosis of isolated sphenoid sinus pathology was based on history, physical examination, and radiologic evaluation. All patients had headache with various localizations. Pre- and postoperative headache intensity of patients was scored using a visual analogue scale (VAS). Results: The most common location of headache was the vertex (24%). The preoperative and postoperative mean VAS scores for headache were 8.24 ± 0.94 and 2.67 ± 1.49, respectively. Statistical comparison revealed a significant improvement in headache intensity (p < 0.01). Polyps (33.3%) were the most common pathology, followed by inflammation (23.8%). Conclusions: The most common presenting symptom of isolated sphenoid sinus disease is headache. In this study, we demonstrated that headache induced by isolated sphenoid disease can be relieved by endoscopic sphenoidotomy. Sphenoid sinus disease should be considered in the differential diagnosis of patients presenting with subacute or chronic headache. © 2015 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.
Keywords: Sphenoid sinus Headache Polyp Fungal infection
1. Introduction Isolated sphenoid sinus disease is a rare clinical entity, with a reported incidence between 1% and 3% among patients with paranasal sinus disease (Lawson and Reino, 1997; Metson and Gliklich, 1996; Hnatuk et al., 1994). The deep location of the sphenoid sinus is suggested to make it less accessible to infectious agents (Lawson and Reino, 1997). However, there are many vital structures, including the middle cranial fossa, optic nerve and chiasm, cavernous sinuses, and cranial nerves adjacent to the sphenoid sinus (Wang et al., 2002). Therefore, neglected sphenoid sinus disease may lead to serious complications. Inflammatory and neoplastic processes may affect the sphenoid sinus. A sphenoid sinus disease is difficult to diagnose with history and physical examination due to the anatomical location of the sphenoid sinus. The signs and symptoms associated with isolated
* Corresponding author. Tel.: þ90 5053912781. E-mail address: [email protected] (F. Celenk).
sphenoid sinus disease may be unclear and nonspecific (Ng and Sethi, 2011). The most common presenting symptom of patients with isolated sphenoid sinus disease is headache, which is reported in 70%e90% of patients (Kim et al., 2008). In this study, we evaluated patients who underwent endoscopic sphenoid sinus surgery for isolated sphenoid sinus disease. We also investigated the impact of sphenoid sinus surgery on headache intensity. 2. Material and methods In this retrospective study, we evaluated patients who underwent endoscopic sinus surgery for isolated sphenoid sinus disease. Data collected from the charts of the patients included patient demographics, presenting signs and symptoms, intensity of headache, location of headache, duration of headache, operative findi/ ngs, radiologic findings, and histopathologic outcome. Diagnosis of isolated sphenoid sinus pathology was based on history, physical examination, and radiologic evaluation. Patients with persistent symptoms suggestive of paranasal sinus pathology
http://dx.doi.org/10.1016/j.jcms.2015.08.025 1010-5182/© 2015 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.
Please cite this article in press as: Celenk F, et al., Isolated sphenoid sinus disease: An overlooked cause of headache, Journal of Cranio-MaxilloFacial Surgery (2015), http://dx.doi.org/10.1016/j.jcms.2015.08.025
2
F. Celenk et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (2015) 1e4
underwent radiologic evaluation. This series also included patients whose disease was detected incidentally either by other departments or by the otolaryngology department while investigating other symptoms and pathologies. After a detailed history, all patients underwent a nasal endoscopy using a rigid, 4-mm nasal endoscope or a fiberoptic endoscope. Endoscopic nasal examination findings were noted. All patients underwent computed tomography (CT) for evaluation of the sphenoid sinuses and other paranasal sinuses (Fig. 1). Axial and coronal projections were used to assess sphenoid sinuses. Magnetic resonance imaging (MRI) was performed in patients suspected to have fungal infections or malignancy. Patients who had other sinus involvements or who had tumors extending out of the sphenoid sinus and arising from surrounding tissues were excluded from the study. All patients with intractable headache consulted a neurologist. All patients underwent endoscopic transnasal or transethmoid sphenoidotomy in a head-flexed position under general anesthesia. Before the surgery, nasal mucosa was decongested by placing pledgets soaked in topical decongestants in the nasal cavity. Subsequently, topical anesthetic agent was infiltrated into the nasal septum and middle turbinate. A 4-mm rigid endoscope was used during the operation. The sphenoid sinus was approached by advancing the endoscope between the middle turbinate and nasal septum. The sphenoid sinus ostium was identified 1.5 cm above the choana. The sphenoid sinus ostium was widened, and the pathology was removed. A nasal pack was placed if necessary. Nasal packs were removed 24e48 h after the surgery. All patients were followed up for at least 6 months. Pre- and postoperative headache intensity of patients was scored using a visual analogue scale (VAS). On the VAS, 0 was considered “no pain” and 10 was considered “extreme pain.” The Statistical Package for Social Science (SPSS) version 22 was used for the statistical analysis. Pre- and postoperative VAS scores were compared using the Wilcoxon test. A p value of less than 0.05 was defined as statistically significant. 3. Results Twenty-one consecutive patients who underwent endoscopic sphenoidotomy for isolated sphenoid sinus disease were included in the study. There were 13 (61.9%) female and 8 (38.1%) male patients with ages ranging from 11 to 63 years (mean, 43.14 ± 13.15 years). Sphenoid sinus involvement was unilateral in 8 (38.1%) patients and bilateral in 13 (61.9%) patients. All patients had headache with various localizations. The distribution of locations of headaches was as follows (Fig. 2): vertex
Fig. 1. Coronal computed tomographic scan showing a heterogenous opacity involving the left sphenoid sinus.
Fig. 2. Headache locations.
(n ¼ 5, 24%), hemicranial (n ¼ 4, 19%), retroorbital (n ¼ 3, 14%), diffuse (n ¼ 3, 14%), bifrontal (n ¼ 3, 14%), occipital (n ¼ 2, 10%), and fronto-orbital (n ¼ 1, 5%). Duration of headache ranged from 3 to 48 months, with a mean of 25.57 ± 14.18 months. The preoperative and postoperative mean VAS score for headache were 8.24 ± 0.94 and 2.67 ± 1.49, respectively (Fig. 3). Statistical comparison revealed a significant improvement in headache intensity (p < 0.01). Other symptoms were nasal discharge (n ¼ 5, 23.8%), nasal blockage (n ¼ 5, 23.8%), and episodic nosebleed (n ¼ 1, 4.8%). Ten (47.6%) patients presented with headache as the only symptom. Eight (31.8%) patients also had allergic symptoms. Fifteen (71.4%) patients showed abnormal nasal endoscopic findings, and 6 (28.6%) patients had normal findings. Endoscopic examination findings were nasal purulent secretion in 5 (23.8%) patients, nasal polyp in 4 (19%) patients (Fig. 4), nasal mucosal edema in 5 (23.8%) patients, and hemorrhagic crusts in 1 (4.8%) patient. Histopathologic analysis revealed nasal polyp in 7 (33.3%) patients, chronic inflammation in 5 (23.8%) patients, mucocele in 4 (19%) patients, fungal infection in 3 (14.1%) patients, and malignancy in 2 (9.5%) patients. Eight (38.1%) patients had accompanying systemic disease such as diabetes mellitus or asthma. All patients
Fig. 3. Pre- and postoperative visual analogue scale (VAS) scores of patients for headache.
Please cite this article in press as: Celenk F, et al., Isolated sphenoid sinus disease: An overlooked cause of headache, Journal of Cranio-MaxilloFacial Surgery (2015), http://dx.doi.org/10.1016/j.jcms.2015.08.025
F. Celenk et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (2015) 1e4
Fig. 4. Endoscopic view of a sphenoid sinus polyp.
were immunocompetent at the time of surgery. Demographic data and clinical parameters of patients are summarized in Table 1. 4. Discussion The most common presenting symptom of isolated sphenoid sinus disease is headache that is atypical, unresponsive to analgesics, and exacerbated by head movements (Ruoppi et al., 2000; Gilony et al., 2007). Localization of headache related to sphenoid sinus disease is quite variable; vertex, frontal, temporal, periorbital, and occipital regions are common sites for headache (Ruoppi et al., 2000). This is explained by sensory innervation of the sphenoid sinus. The sphenoid sinus is innervated by the fifth cranial nerve and afferent fibers from the sphenopalatine ganglion. Cakmak et al. (Cakmak et al., 2000) reported on one of the largest documented series of isolated sphenoid sinus disease, and found that 72.5% of patients presented with headache. In a
3
retrospective study, Wang et al. (Wang et al., 2002) included 122 patients with isolated sphenoid sinus disease and reported headache as the initial symptom in 62% of patients. In another retrospective study, conducted by Kim et al. (Kim et al., 2008), headache was reported in 65.8% of patients with isolated sphenoid sinus disease. Headache was diffuse and nonspecific in 50% of patients. Castelnuovo et al. (Castelnuovo et al., 2005) operated on 41 patients with isolated sphenoid sinus disease and reported headache in 73% of patients. The frontal region was the most common site for headache. Socher et al. (Socher et al., 2008) reported on 109 patients with isolated sphenoid sinus disease, and found frontal or retroorbital headache in 71.5% of patients. In another retrospective study that included 72 patients with isolated sphenoid sinus, 85% of patients presented with headache (Gilony et al., 2007). The most common location of headache was the frontal area. Sethi (Sethi, 1999) reported on 21 patients with isolated sphenoid sinus disease and found headache as the presenting symptom in 71.4% of cases. The most common location of headache was the retroorbital area, followed by the vertex. In the current study, we included 21 patients with isolated sphenoid sinus; all of the patients (100%) had headache as the primary presenting symptom. The most common location of headache was the vertex. Other possible symptoms of isolated sphenoid sinus disease are nasal obstruction, nasal discharge, ocular symptoms, and nosebleed. Nasal obstruction is the second most common presenting symptom, according to most current literature. (Kim et al., 2008; Gilony et al., 2007; Castelnuovo et al., 2005; Socher et al., 2008) We found nasal obstruction as well as nasal discharge as the second most common presenting symptom in our study. Visual disturbances were found to be the second most common complaint in several studies (Lawson and Reino, 1997; Wang et al., 2002). Nasal endoscopic examination allows direct observation of the nasal cavity, and should be a part of the evaluation of patients with isolated sphenoid sinus disease. Sphenochoanal polyps, nasal discharge, and tumor extensions into the nasal cavity may be identified during nasal endoscopy. Sethi (Sethi, 1999) reported that nasal endoscopy contributed significantly to the diagnosis of sphenoid pathology in 76.2% of patients. Wang et al. (Wang et al., 2002) reported abnormal nasal endoscopic findings in 79 of 122 patients with isolated sphenoid sinus disease. In the current study, we found abnormal endoscopic findings including nasal polyps, nasal secretions, mucosal edema, and hemorrhagic crusts in 71.4%
Table 1 Demographic and clinical characteristics of patients. Patient
Age (y)
Gender
Duration of headache (mo)
Localization of headache
Histopathologic outcome
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21
50 36 56 35 49 26 23 61 46 37 45 58 52 63 39 44 50 35 36 54 11
Female Female Female Male Male Female Male Female Female Female Male Female Male Male Female Female Female Male Female Female Male
36 3 48 36 36 6 48 24 48 12 24 24 30 36 12 24 30 30 6 12 12
Vertex Hemicranial Diffuse Hemicranial Vertex Occipital Diffuse Hemicranial Vertex Diffuse Frontoorbital Vertex Occipital Vertex Retroorbital Retroorbital Bifrontal Retroorbital Bifrontal Hemicranial Bifrontal
Polyp Chronic inflammation Squamous cell carcinoma Chronic inflammation Polyp Chronic inflammation Mucocele Polyp Polyp Neuroendocrine tumor Mucocele Fungal infection Mucocele Fungal infection Polyp Fungal infection Chronic inflammation Polyp Mucocele Polyp Chronic inflammation
Please cite this article in press as: Celenk F, et al., Isolated sphenoid sinus disease: An overlooked cause of headache, Journal of Cranio-MaxilloFacial Surgery (2015), http://dx.doi.org/10.1016/j.jcms.2015.08.025
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F. Celenk et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (2015) 1e4
of patients. However, in a series reported by Kim et al. (Kim et al., 2008), only 22.2% of patients showed abnormal endoscopic findings. Accordingly, nasal endoscopic examination findings may not contribute to the diagnosis of isolated sphenoid sinus disease in a significant percentage of patients. Normal findings obtained during nasal endoscopy do not exclude the presence of sphenoid sinus disease. Therefore, radiologic evaluation is the most important step in diagnosing isolated sphenoid disease. CT is an essential tool for making diagnoses in patients suspected of having sphenoid sinus disease. Axial and coronal CT scans can demonstrate sphenoid sinus lesions and extension of the disease beyond the sphenoid sinus bony walls. MRI may be helpful in differential diagnosis of neoplastic processes and fungal infections (Socher et al., 2008). If there are CT findings suggesting a neoplasm, MRI should be performed to detect the extent of the disease (Lawson and Reino, 1997; Wang et al., 2002). MRI is also helpful in differentiating cysts and polyps from mucoceles and encephaloceles (Lawson and Reino, 1997; Ng and Sethi, 2011; Martin et al., 2002). Sometimes isolated sphenoid sinus disease may be found incidentally during investigation for headache or other unrelated symptoms. Novel endoscopic surgical approaches to isolated sphenoid sinus disease include transnasal, transetmoid, and transseptal approaches (Martin et al., 2002). Endoscopic sphenoidotomy is an effective method to eliminate the sphenoid sinus disease and to relieve headache. In the current study, all patients underwent transetmoid or transnasal endoscopic sphenoidotomy. We also investigated the impact of sphenoid sinus surgery on headache intensity by utilization of pre- and postoperative VAS. There was a statistically significant improvement in headache intensity after the surgical treatment. Most of the sphenoid sinus diseases are inflammatory in origin, followed by mucocele (Lawson and Reino, 1997; Ng and Sethi, 2011; Kim et al., 2008). Castelnuovo et al. (Castelnuovo et al., 2005) found that fungal sinusitis is the most common sphenoid sinus lesion. Mucocele was the most common pathology, followed by sphenochoanal polyp, in a series of 109 patients with isolated sphenoid sinus disease (Socher et al., 2008). In our study, polyps were the most common pathology, followed by inflammation. In this consecutive case series of patients with isolated sphenoid sinus disease, headache was the most common initial symptom, and all patients reported headache, with various localizations and degrees of severity. Our study differs from other studies in the way that we found headache as the primary symptom in all patients. The higher headache incidence that we found in this study may be explained by the fact that most of the cases were detected radiologically while investigating the etiology of chronic or subacute headache. Headache might be incidentally accompanied by other additional symptoms in some patients. However, significant
improvement in headache severity after surgical treatment confirmed that sphenoid sinus disease was the main cause of headache in our case series. Accordingly, our study indicated that headache is the most common presenting symptom of isolated sphenoid sinus disease, and that the frequency may be as high as 100%. 5. Conclusion The diagnosis of isolated sphenoid sinus disease is mainly based on radiologic evaluation. Symptoms are usually unclear and nonspecific. Headache is the most common presenting symptom followed by nasal obstruction and nasal discharge. Nasal endoscopic examination does not always significantly contribute to diagnosis. In this study, we demonstrated that headache induced by isolated sphenoid disease can be relieved by endoscopic sphenoidotomy. Sphenoid sinus disease should be considered in the differential diagnosis of patients presenting with subacute or chronic headache. Conflict of interest The authors declare that there is no conflict of interest in regard to this work. References Cakmak O, Shohet MR, Kern EB: Isolated sphenoid sinus lesions. Am J Rhinol 14: 13e19, 2000 Castelnuovo P, Pagella F, Semino L, De Bernardi F, Delù G: Endoscopic treatment of the isolated sphenoid sinus lesions. Eur Arch Otorhinolaryngol 262: 142e147, 2005 Gilony D, Talmi YP, Bedrin L, Ben-Shosan Y, Kronenberg J: The clinical behavior of isolated sphenoid sinusitis. Otolaryngol Head Neck Surg 136: 610e615, 2007 Hnatuk LA, Macdonald RE, Papsin BC: Isolated sphenoid sinusitis: the Toronto Hospital for Sick Children experience and review of the literature. J Otolaryngol 23: 36e41, 1994 Kim SW, Kim DW, Kong IG, Kim DY, Park SW, Rhee CS, et al: Isolated sphenoid sinus diseases: report of 76 cases. Acta Otolaryngol 128(4): 455e459, 2008 Apr Lawson W, Reino AJ: Isolated sphenoid sinus disease: an analysis of 132 cases. Laryngoscope 107(12 Pt 1): 1590e1595, 1997 Dec Martin TJ, Smith TL, Smith MM, Loehrl TA: Evaluation and surgical management of isolated sphenoid sinus disease. Arch Otolaryngol Head Neck Surg 128: 1413e1419, 2002 Metson R, Gliklich RE: Endoscopic treatment of sphenoid sinusitis. Otolaryngol Head Neck Surg 114(6): 736e744, 1996 Jun Ng YH, Sethi DS: Isolated sphenoid sinus disease: differential diagnosis and management. Curr Opin Otolaryngol Head Neck Surg 19: 16e20, 2011 Ruoppi P, Seppa J, Pukkila M, Nuutinen J: Isolated sphenoid sinus diseases: report of 39 cases. Arch Otolaryngol Head Neck Surg 126(6): 777e781, 2000 Jun Sethi DS: Isolated sphenoid lesions: diagnosis and management. Otolaryngol Head Neck Surg 120: 730e736, 1999 Socher JA, Cassano M, Filheiro CA, Cassano P, Felippu A: Diagnosis and treatment of isolated sphenoid sinus disease: a review of 109 cases. Acta Otolaryngol 128(9): 1004e1010, 2008 Sep Wang ZM, Kanoh N, Dai CF, Kutler DI, Xu R, Chi FL, et al: Isolated sphenoid sinus disease: an analysis of 122 cases. Ann Otol Rhinol Laryngol 111: 323e327, 2002
Please cite this article in press as: Celenk F, et al., Isolated sphenoid sinus disease: An overlooked cause of headache, Journal of Cranio-MaxilloFacial Surgery (2015), http://dx.doi.org/10.1016/j.jcms.2015.08.025
Contents lists available at ScienceDirect
Journal of Cranio-Maxillo-Facial Surgery journal homepage: www.jcmfs.com
Isolated sphenoid sinus disease: An overlooked cause of headache Fatih Celenk*, Secaattin Gulsen, Burhanettin Gonuldas, Elif Baysal, Cengiz Durucu, Muzaffer Kanlikama, Semih Mumbuc Department of Otorhinolaryngology, Gaziantep University Faculty of Medicine, Gaziantep, Turkey
a r t i c l e i n f o
a b s t r a c t
Article history: Paper received 3 June 2015 Accepted 26 August 2015 Available online xxx
Objectives: The aim of this study was to evaluate patients who underwent endoscopic sphenoid sinus surgery for isolated sphenoid sinus disease. We also investigated the impact of sphenoid sinus surgery on headache intensity. Material and methods: Twenty-one consecutive patients who underwent endoscopic sphenoidotomy for isolated sphenoid sinus disease were included in the study. Diagnosis of isolated sphenoid sinus pathology was based on history, physical examination, and radiologic evaluation. All patients had headache with various localizations. Pre- and postoperative headache intensity of patients was scored using a visual analogue scale (VAS). Results: The most common location of headache was the vertex (24%). The preoperative and postoperative mean VAS scores for headache were 8.24 ± 0.94 and 2.67 ± 1.49, respectively. Statistical comparison revealed a significant improvement in headache intensity (p < 0.01). Polyps (33.3%) were the most common pathology, followed by inflammation (23.8%). Conclusions: The most common presenting symptom of isolated sphenoid sinus disease is headache. In this study, we demonstrated that headache induced by isolated sphenoid disease can be relieved by endoscopic sphenoidotomy. Sphenoid sinus disease should be considered in the differential diagnosis of patients presenting with subacute or chronic headache. © 2015 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.
Keywords: Sphenoid sinus Headache Polyp Fungal infection
1. Introduction Isolated sphenoid sinus disease is a rare clinical entity, with a reported incidence between 1% and 3% among patients with paranasal sinus disease (Lawson and Reino, 1997; Metson and Gliklich, 1996; Hnatuk et al., 1994). The deep location of the sphenoid sinus is suggested to make it less accessible to infectious agents (Lawson and Reino, 1997). However, there are many vital structures, including the middle cranial fossa, optic nerve and chiasm, cavernous sinuses, and cranial nerves adjacent to the sphenoid sinus (Wang et al., 2002). Therefore, neglected sphenoid sinus disease may lead to serious complications. Inflammatory and neoplastic processes may affect the sphenoid sinus. A sphenoid sinus disease is difficult to diagnose with history and physical examination due to the anatomical location of the sphenoid sinus. The signs and symptoms associated with isolated
* Corresponding author. Tel.: þ90 5053912781. E-mail address: [email protected] (F. Celenk).
sphenoid sinus disease may be unclear and nonspecific (Ng and Sethi, 2011). The most common presenting symptom of patients with isolated sphenoid sinus disease is headache, which is reported in 70%e90% of patients (Kim et al., 2008). In this study, we evaluated patients who underwent endoscopic sphenoid sinus surgery for isolated sphenoid sinus disease. We also investigated the impact of sphenoid sinus surgery on headache intensity. 2. Material and methods In this retrospective study, we evaluated patients who underwent endoscopic sinus surgery for isolated sphenoid sinus disease. Data collected from the charts of the patients included patient demographics, presenting signs and symptoms, intensity of headache, location of headache, duration of headache, operative findi/ ngs, radiologic findings, and histopathologic outcome. Diagnosis of isolated sphenoid sinus pathology was based on history, physical examination, and radiologic evaluation. Patients with persistent symptoms suggestive of paranasal sinus pathology
http://dx.doi.org/10.1016/j.jcms.2015.08.025 1010-5182/© 2015 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.
Please cite this article in press as: Celenk F, et al., Isolated sphenoid sinus disease: An overlooked cause of headache, Journal of Cranio-MaxilloFacial Surgery (2015), http://dx.doi.org/10.1016/j.jcms.2015.08.025
2
F. Celenk et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (2015) 1e4
underwent radiologic evaluation. This series also included patients whose disease was detected incidentally either by other departments or by the otolaryngology department while investigating other symptoms and pathologies. After a detailed history, all patients underwent a nasal endoscopy using a rigid, 4-mm nasal endoscope or a fiberoptic endoscope. Endoscopic nasal examination findings were noted. All patients underwent computed tomography (CT) for evaluation of the sphenoid sinuses and other paranasal sinuses (Fig. 1). Axial and coronal projections were used to assess sphenoid sinuses. Magnetic resonance imaging (MRI) was performed in patients suspected to have fungal infections or malignancy. Patients who had other sinus involvements or who had tumors extending out of the sphenoid sinus and arising from surrounding tissues were excluded from the study. All patients with intractable headache consulted a neurologist. All patients underwent endoscopic transnasal or transethmoid sphenoidotomy in a head-flexed position under general anesthesia. Before the surgery, nasal mucosa was decongested by placing pledgets soaked in topical decongestants in the nasal cavity. Subsequently, topical anesthetic agent was infiltrated into the nasal septum and middle turbinate. A 4-mm rigid endoscope was used during the operation. The sphenoid sinus was approached by advancing the endoscope between the middle turbinate and nasal septum. The sphenoid sinus ostium was identified 1.5 cm above the choana. The sphenoid sinus ostium was widened, and the pathology was removed. A nasal pack was placed if necessary. Nasal packs were removed 24e48 h after the surgery. All patients were followed up for at least 6 months. Pre- and postoperative headache intensity of patients was scored using a visual analogue scale (VAS). On the VAS, 0 was considered “no pain” and 10 was considered “extreme pain.” The Statistical Package for Social Science (SPSS) version 22 was used for the statistical analysis. Pre- and postoperative VAS scores were compared using the Wilcoxon test. A p value of less than 0.05 was defined as statistically significant. 3. Results Twenty-one consecutive patients who underwent endoscopic sphenoidotomy for isolated sphenoid sinus disease were included in the study. There were 13 (61.9%) female and 8 (38.1%) male patients with ages ranging from 11 to 63 years (mean, 43.14 ± 13.15 years). Sphenoid sinus involvement was unilateral in 8 (38.1%) patients and bilateral in 13 (61.9%) patients. All patients had headache with various localizations. The distribution of locations of headaches was as follows (Fig. 2): vertex
Fig. 1. Coronal computed tomographic scan showing a heterogenous opacity involving the left sphenoid sinus.
Fig. 2. Headache locations.
(n ¼ 5, 24%), hemicranial (n ¼ 4, 19%), retroorbital (n ¼ 3, 14%), diffuse (n ¼ 3, 14%), bifrontal (n ¼ 3, 14%), occipital (n ¼ 2, 10%), and fronto-orbital (n ¼ 1, 5%). Duration of headache ranged from 3 to 48 months, with a mean of 25.57 ± 14.18 months. The preoperative and postoperative mean VAS score for headache were 8.24 ± 0.94 and 2.67 ± 1.49, respectively (Fig. 3). Statistical comparison revealed a significant improvement in headache intensity (p < 0.01). Other symptoms were nasal discharge (n ¼ 5, 23.8%), nasal blockage (n ¼ 5, 23.8%), and episodic nosebleed (n ¼ 1, 4.8%). Ten (47.6%) patients presented with headache as the only symptom. Eight (31.8%) patients also had allergic symptoms. Fifteen (71.4%) patients showed abnormal nasal endoscopic findings, and 6 (28.6%) patients had normal findings. Endoscopic examination findings were nasal purulent secretion in 5 (23.8%) patients, nasal polyp in 4 (19%) patients (Fig. 4), nasal mucosal edema in 5 (23.8%) patients, and hemorrhagic crusts in 1 (4.8%) patient. Histopathologic analysis revealed nasal polyp in 7 (33.3%) patients, chronic inflammation in 5 (23.8%) patients, mucocele in 4 (19%) patients, fungal infection in 3 (14.1%) patients, and malignancy in 2 (9.5%) patients. Eight (38.1%) patients had accompanying systemic disease such as diabetes mellitus or asthma. All patients
Fig. 3. Pre- and postoperative visual analogue scale (VAS) scores of patients for headache.
Please cite this article in press as: Celenk F, et al., Isolated sphenoid sinus disease: An overlooked cause of headache, Journal of Cranio-MaxilloFacial Surgery (2015), http://dx.doi.org/10.1016/j.jcms.2015.08.025
F. Celenk et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (2015) 1e4
Fig. 4. Endoscopic view of a sphenoid sinus polyp.
were immunocompetent at the time of surgery. Demographic data and clinical parameters of patients are summarized in Table 1. 4. Discussion The most common presenting symptom of isolated sphenoid sinus disease is headache that is atypical, unresponsive to analgesics, and exacerbated by head movements (Ruoppi et al., 2000; Gilony et al., 2007). Localization of headache related to sphenoid sinus disease is quite variable; vertex, frontal, temporal, periorbital, and occipital regions are common sites for headache (Ruoppi et al., 2000). This is explained by sensory innervation of the sphenoid sinus. The sphenoid sinus is innervated by the fifth cranial nerve and afferent fibers from the sphenopalatine ganglion. Cakmak et al. (Cakmak et al., 2000) reported on one of the largest documented series of isolated sphenoid sinus disease, and found that 72.5% of patients presented with headache. In a
3
retrospective study, Wang et al. (Wang et al., 2002) included 122 patients with isolated sphenoid sinus disease and reported headache as the initial symptom in 62% of patients. In another retrospective study, conducted by Kim et al. (Kim et al., 2008), headache was reported in 65.8% of patients with isolated sphenoid sinus disease. Headache was diffuse and nonspecific in 50% of patients. Castelnuovo et al. (Castelnuovo et al., 2005) operated on 41 patients with isolated sphenoid sinus disease and reported headache in 73% of patients. The frontal region was the most common site for headache. Socher et al. (Socher et al., 2008) reported on 109 patients with isolated sphenoid sinus disease, and found frontal or retroorbital headache in 71.5% of patients. In another retrospective study that included 72 patients with isolated sphenoid sinus, 85% of patients presented with headache (Gilony et al., 2007). The most common location of headache was the frontal area. Sethi (Sethi, 1999) reported on 21 patients with isolated sphenoid sinus disease and found headache as the presenting symptom in 71.4% of cases. The most common location of headache was the retroorbital area, followed by the vertex. In the current study, we included 21 patients with isolated sphenoid sinus; all of the patients (100%) had headache as the primary presenting symptom. The most common location of headache was the vertex. Other possible symptoms of isolated sphenoid sinus disease are nasal obstruction, nasal discharge, ocular symptoms, and nosebleed. Nasal obstruction is the second most common presenting symptom, according to most current literature. (Kim et al., 2008; Gilony et al., 2007; Castelnuovo et al., 2005; Socher et al., 2008) We found nasal obstruction as well as nasal discharge as the second most common presenting symptom in our study. Visual disturbances were found to be the second most common complaint in several studies (Lawson and Reino, 1997; Wang et al., 2002). Nasal endoscopic examination allows direct observation of the nasal cavity, and should be a part of the evaluation of patients with isolated sphenoid sinus disease. Sphenochoanal polyps, nasal discharge, and tumor extensions into the nasal cavity may be identified during nasal endoscopy. Sethi (Sethi, 1999) reported that nasal endoscopy contributed significantly to the diagnosis of sphenoid pathology in 76.2% of patients. Wang et al. (Wang et al., 2002) reported abnormal nasal endoscopic findings in 79 of 122 patients with isolated sphenoid sinus disease. In the current study, we found abnormal endoscopic findings including nasal polyps, nasal secretions, mucosal edema, and hemorrhagic crusts in 71.4%
Table 1 Demographic and clinical characteristics of patients. Patient
Age (y)
Gender
Duration of headache (mo)
Localization of headache
Histopathologic outcome
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21
50 36 56 35 49 26 23 61 46 37 45 58 52 63 39 44 50 35 36 54 11
Female Female Female Male Male Female Male Female Female Female Male Female Male Male Female Female Female Male Female Female Male
36 3 48 36 36 6 48 24 48 12 24 24 30 36 12 24 30 30 6 12 12
Vertex Hemicranial Diffuse Hemicranial Vertex Occipital Diffuse Hemicranial Vertex Diffuse Frontoorbital Vertex Occipital Vertex Retroorbital Retroorbital Bifrontal Retroorbital Bifrontal Hemicranial Bifrontal
Polyp Chronic inflammation Squamous cell carcinoma Chronic inflammation Polyp Chronic inflammation Mucocele Polyp Polyp Neuroendocrine tumor Mucocele Fungal infection Mucocele Fungal infection Polyp Fungal infection Chronic inflammation Polyp Mucocele Polyp Chronic inflammation
Please cite this article in press as: Celenk F, et al., Isolated sphenoid sinus disease: An overlooked cause of headache, Journal of Cranio-MaxilloFacial Surgery (2015), http://dx.doi.org/10.1016/j.jcms.2015.08.025
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of patients. However, in a series reported by Kim et al. (Kim et al., 2008), only 22.2% of patients showed abnormal endoscopic findings. Accordingly, nasal endoscopic examination findings may not contribute to the diagnosis of isolated sphenoid sinus disease in a significant percentage of patients. Normal findings obtained during nasal endoscopy do not exclude the presence of sphenoid sinus disease. Therefore, radiologic evaluation is the most important step in diagnosing isolated sphenoid disease. CT is an essential tool for making diagnoses in patients suspected of having sphenoid sinus disease. Axial and coronal CT scans can demonstrate sphenoid sinus lesions and extension of the disease beyond the sphenoid sinus bony walls. MRI may be helpful in differential diagnosis of neoplastic processes and fungal infections (Socher et al., 2008). If there are CT findings suggesting a neoplasm, MRI should be performed to detect the extent of the disease (Lawson and Reino, 1997; Wang et al., 2002). MRI is also helpful in differentiating cysts and polyps from mucoceles and encephaloceles (Lawson and Reino, 1997; Ng and Sethi, 2011; Martin et al., 2002). Sometimes isolated sphenoid sinus disease may be found incidentally during investigation for headache or other unrelated symptoms. Novel endoscopic surgical approaches to isolated sphenoid sinus disease include transnasal, transetmoid, and transseptal approaches (Martin et al., 2002). Endoscopic sphenoidotomy is an effective method to eliminate the sphenoid sinus disease and to relieve headache. In the current study, all patients underwent transetmoid or transnasal endoscopic sphenoidotomy. We also investigated the impact of sphenoid sinus surgery on headache intensity by utilization of pre- and postoperative VAS. There was a statistically significant improvement in headache intensity after the surgical treatment. Most of the sphenoid sinus diseases are inflammatory in origin, followed by mucocele (Lawson and Reino, 1997; Ng and Sethi, 2011; Kim et al., 2008). Castelnuovo et al. (Castelnuovo et al., 2005) found that fungal sinusitis is the most common sphenoid sinus lesion. Mucocele was the most common pathology, followed by sphenochoanal polyp, in a series of 109 patients with isolated sphenoid sinus disease (Socher et al., 2008). In our study, polyps were the most common pathology, followed by inflammation. In this consecutive case series of patients with isolated sphenoid sinus disease, headache was the most common initial symptom, and all patients reported headache, with various localizations and degrees of severity. Our study differs from other studies in the way that we found headache as the primary symptom in all patients. The higher headache incidence that we found in this study may be explained by the fact that most of the cases were detected radiologically while investigating the etiology of chronic or subacute headache. Headache might be incidentally accompanied by other additional symptoms in some patients. However, significant
improvement in headache severity after surgical treatment confirmed that sphenoid sinus disease was the main cause of headache in our case series. Accordingly, our study indicated that headache is the most common presenting symptom of isolated sphenoid sinus disease, and that the frequency may be as high as 100%. 5. Conclusion The diagnosis of isolated sphenoid sinus disease is mainly based on radiologic evaluation. Symptoms are usually unclear and nonspecific. Headache is the most common presenting symptom followed by nasal obstruction and nasal discharge. Nasal endoscopic examination does not always significantly contribute to diagnosis. In this study, we demonstrated that headache induced by isolated sphenoid disease can be relieved by endoscopic sphenoidotomy. Sphenoid sinus disease should be considered in the differential diagnosis of patients presenting with subacute or chronic headache. Conflict of interest The authors declare that there is no conflict of interest in regard to this work. References Cakmak O, Shohet MR, Kern EB: Isolated sphenoid sinus lesions. Am J Rhinol 14: 13e19, 2000 Castelnuovo P, Pagella F, Semino L, De Bernardi F, Delù G: Endoscopic treatment of the isolated sphenoid sinus lesions. Eur Arch Otorhinolaryngol 262: 142e147, 2005 Gilony D, Talmi YP, Bedrin L, Ben-Shosan Y, Kronenberg J: The clinical behavior of isolated sphenoid sinusitis. Otolaryngol Head Neck Surg 136: 610e615, 2007 Hnatuk LA, Macdonald RE, Papsin BC: Isolated sphenoid sinusitis: the Toronto Hospital for Sick Children experience and review of the literature. J Otolaryngol 23: 36e41, 1994 Kim SW, Kim DW, Kong IG, Kim DY, Park SW, Rhee CS, et al: Isolated sphenoid sinus diseases: report of 76 cases. Acta Otolaryngol 128(4): 455e459, 2008 Apr Lawson W, Reino AJ: Isolated sphenoid sinus disease: an analysis of 132 cases. Laryngoscope 107(12 Pt 1): 1590e1595, 1997 Dec Martin TJ, Smith TL, Smith MM, Loehrl TA: Evaluation and surgical management of isolated sphenoid sinus disease. Arch Otolaryngol Head Neck Surg 128: 1413e1419, 2002 Metson R, Gliklich RE: Endoscopic treatment of sphenoid sinusitis. Otolaryngol Head Neck Surg 114(6): 736e744, 1996 Jun Ng YH, Sethi DS: Isolated sphenoid sinus disease: differential diagnosis and management. Curr Opin Otolaryngol Head Neck Surg 19: 16e20, 2011 Ruoppi P, Seppa J, Pukkila M, Nuutinen J: Isolated sphenoid sinus diseases: report of 39 cases. Arch Otolaryngol Head Neck Surg 126(6): 777e781, 2000 Jun Sethi DS: Isolated sphenoid lesions: diagnosis and management. Otolaryngol Head Neck Surg 120: 730e736, 1999 Socher JA, Cassano M, Filheiro CA, Cassano P, Felippu A: Diagnosis and treatment of isolated sphenoid sinus disease: a review of 109 cases. Acta Otolaryngol 128(9): 1004e1010, 2008 Sep Wang ZM, Kanoh N, Dai CF, Kutler DI, Xu R, Chi FL, et al: Isolated sphenoid sinus disease: an analysis of 122 cases. Ann Otol Rhinol Laryngol 111: 323e327, 2002
Please cite this article in press as: Celenk F, et al., Isolated sphenoid sinus disease: An overlooked cause of headache, Journal of Cranio-MaxilloFacial Surgery (2015), http://dx.doi.org/10.1016/j.jcms.2015.08.025