ORL 2015;77:162–170 DOI: 10.1159/000381918 Received: September 17, 2014 Accepted: March 24, 2015 Published online: July 4, 2015

© 2015 S. Karger AG, Basel 0301–1569/15/0773–0162$39.50/0 www.karger.com/orl

Original Paper

Clinical Features of Patients Treated with Endoscopic Sinus Surgery for Posttraumatic Paranasal Sinus Mucocele Yusuke Kojima Kenzo Tsuzuki Yoriko Yukitatsu Hironori Takebayashi Masafumi Sakagami

Hideki Oka

Department of Otolaryngology – Head and Neck Surgery, Hyogo College of Medicine, Hyogo, Japan

Key Words Posttraumatic paranasal sinus mucocele · Frontal sinus · Endoscopic sinus surgery Abstract Aim: The purpose of this study was to analyze the clinical features of patients with posttraumatic paranasal sinus mucocele (PSM). Subjects and Methods: Between 2009 and 2013, we performed endoscopic sinus surgery (ESS) on 68 patients with PSM at the Department of Otolaryngology – Head and Neck Surgery at Hyogo College of Medicine. Five male patients (age range, 45–76 years) with posttraumatic PSM were analyzed retrospectively. Diagnosis was based on the history of injury and radiological findings. Results: Posttraumatic PSM was found in 7% (5/68) of patients. The mean interval from injury to diagnosis was 28.4 years. All patients had frontal sinus mucocele. Four patients had symptoms of headache, diplopia, visual field defect, and forehead swelling, and 1 patient was asymptomatic. ESS was performed under general anesthesia in all cases, and the symptoms improved postoperatively. Reoperation was required in 1 patient (20%) because headache developed with obstruction of the frontal drainage route 7 months after ESS. Conclusions: Posttraumatic PSM was the least frequent form of PSM and was located predominantly in the frontal sinus, causing symptoms long after the forehead injury. The important lessons to be learned for treating posttraumatic PSM are to obtain a detailed history and to enlarge the route to the cyst to avoid its recurrence. © 2015 S. Karger AG, Basel

Introduction

Kenzo Tsuzuki, MD, PhD Department of Otolaryngology – Head and Neck Surgery Hyogo College of Medicine 1-1 Mukogawa, Nishinomiya, Hyogo 663-8501 (Japan) E-Mail kenzo @ hyo-med.ac.jp

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Paranasal sinus mucocele (PSM) is a cyst caused by secretions accumulated from the sinus mucosa due to obstruction of sinus drainage routes. In Japan, the pathogenesis of PSM is classified into idiopathic, postoperative, and posttraumatic cysts [1]. Postoperative

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DOI: 10.1159/000381918

Kojima et al.: Clinical Features of Patients Treated with Endoscopic Sinus Surgery for Posttraumatic Paranasal Sinus Mucocele

Posttraumatic PSM n = 5 (7%) Idiopatic PSM n = 11 (16%)

Postoperative PSM n = 52 (77%)

Fig. 1. Pathogenesis of PSM. Five patients with posttraumatic PSM were analyzed in this study.

and posttraumatic PSMs were identified when patients had clearly undergone sinus surgery or suffered a traumatic injury, respectively. Patients without any apparent cause of PSM were classified as having idiopathic PSM. Postoperative PSM is reported as the most common type and posttraumatic PSM as the least common type [2, 3]. The paranasal sinuses are adjacent to the orbita and skull base. Large PSMs may cause symptoms such as headache, diplopia, exophthalmos, and blurred vision rather than nasal symptoms, due to increased pressure and inflammation. Because PSMs grow slowly, a long time usually passes before symptoms arise. Surgical treatment is commonly selected to resolve symptoms. Endoscopic sinus surgery (ESS), extranasal sinus surgery, or two-way approaches that utilize a combination of intra- and extranasal sinus surgeries are performed as surgical treatments [4]. Although many cases of idiopathic and/or postoperative PSMs have been reported [2, 3, 5–9], few case reports have described posttraumatic PSM [10–12]. When we previously studied PSMs, we had to exclude posttraumatic PSM because the diagnosis was definitively achieved in only 1 patient at that time (1995–2008) [13]. The present study thus focused on and analyzed patients with definitively diagnosed posttraumatic PSM who underwent sinus surgery.

We performed ESS in 68 patients with PSMs at the Department of Otolaryngology – Head and Neck Surgery at Hyogo College of Medicine between January 2009 and December 2013 (fig. 1). Among those, 5 male patients were definitively diagnosed with posttraumatic PSM. The mean age was 57 years (range, 45–76). The clinical course was investigated based on the clinical and operative records. This study conformed to the regulations of the Ethics Committee of Hyogo College of Medicine (approval No. 1512). Patients with idiopathic or postoperative PSMs or a history of any sinonasal surgery were excluded from this study. In this study, posttraumatic PSM was identified based on clear documentation in the clinical records that the patient had suffered a traumatic injury to the head and/or face. Posttraumatic PSM was also diagnosed on the basis of a detailed history of the injury, preoperative radiological findings from computed tomography (CT) and magnetic resonance imaging (MRI), and intraoperative endoscopic sinonasal findings. We diagnosed PSM when the images delineated well-circumscribed, homogeneous, and expansile lesions [8, 14]. As postoperative treatment, medical therapy with antibiotics, mucinolytics, antiallergic agents, and nasal irrigation with saline was performed for each patient until the postoperative sinonasal mucosal condition had stabilized, as we reported previously [15]. Postoperative intranasal findings were evaluated using the endoscopic score (E score) [16]. The endoscopic appearance of the operated sinuses and olfactory

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Patients and Methods

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Kojima et al.: Clinical Features of Patients Treated with Endoscopic Sinus Surgery for Posttraumatic Paranasal Sinus Mucocele

Table 1. Clinical features of the 5 male patients with posttraumatic PSM Case No.

Age, years

First visited department

Symptoms

History of traumatic injury

Elapsed time PSM Postoperative after injury, years locations period, months

1 2

65 48

neurosurgery neurosurgery

headache visual field defect

60 3

F + AE F

3

49

otolaryngology

symptomless

20

F

17

4 5

76 45

otolaryngology ophthalmology

forehead swelling diplopia

frontal bone fracture facial bruise without bony fracture facial bruise without bony fracture frontal bone fracture frontal bone fracture

30 29

F F

5 7

7 1

F = Frontal sinus; AE = anterior ethmoid sinus.

clefts was scored as follows: 0 = normal condition; 1 = sinus only partially observable due to occupation by polyps, edematous mucosa, and/or discharge; 2 = unobservable due to complete occupation by polyps and/ or discharge. This study retrospectively analyzed the evident opportunity, symptoms, first visit to a clinical department, time elapsed since injury, location of mucoceles, surgical procedures, and postoperative course in patients with posttraumatic PSM.

Results

Case 1 A 65-year-old man suffering from headache initially visited a neurosurgeon. He had a history of frontal bone fracture at 5 years of age. Traumatic injury to the head at that time was clearly documented in the clinical records. CT delineated a well-circumscribed cystic lesion

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We performed sinus surgery in 68 patients with PSM at our department between 2009 and 2013. Idiopathic, postoperative, and posttraumatic PSM was present in 16% (11/68), 77% (52/68), and 7% (5/68) of patients, respectively. Posttraumatic PSM was thus the least frequent form of PSM (fig. 1). The clinical characteristics of the 5 patients with posttraumatic PSM are summarized in table 1. Regarding the evident opportunity, head trauma with depressed fracture of the frontal bone to the frontal sinus was identified in 3 patients, while facial bruise without bony fracture was observed in 2 patients. Most patients experienced headaches, visual field defects, forehead swelling, and diplopia, but no patients presented with nasal symptoms. One patient without symptoms was diagnosed because a cystic lesion was identified on imaging. Three of the 5 patients (60%) visited neurosurgeons or ophthalmologists, whereas 2 patients (40%) visited otolaryngologists first. The mean interval from head injury to PSM diagnosis was 28.4 years (range, 3–60). All patients showed frontal sinus mucoceles, and 1 patient had both frontal and anterior ethmoid sinus mucoceles. ESS was performed in all 5 patients under general anesthesia. The Kolibri navigation system (Brainlab, Tokyo, Japan) was used for 3 patients (cases 1, 4, and 5) because the PSM had expanded into the orbita and skull base with bony wall destruction. Symptoms disappeared in each patient after ESS (cases 1, 3, 4, and 5). However, 1 patient (case 5) who had headache since postoperative month 5 required additional ESS. In this patient, no closure of the PSM drainage route has been observed for 7 months after the second surgery.

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Kojima et al.: Clinical Features of Patients Treated with Endoscopic Sinus Surgery for Posttraumatic Paranasal Sinus Mucocele

Fig. 2. Radiological findings in case 1. A mucocele (arrows) was observed from the frontal sinus to the anterior ethmoid sinus with bony destruction (arrowheads) on axial (a) and coronal (b) CT as well as on axial (c) and coronal (d) T2-WI MRI. Axial (e) and coronal (f) CT show pneumatization of the sinuses 7 months after ESS. The ocular deviation between the operated and nonoperated sides was 3.33 mm (a), improving to a maximum of 1.02 mm postoperatively (e). Lines indicate the operated (a) and nonoperated (b) side.

a

b

c

d

e

f

Case 2 A 48-year-old man complaining of visual field defect initially visited a neurosurgeon. He had suffered facial bruising in a traffic accident at the age of 45 years. CT delineated a wellcircumscribed cystic lesion from the frontal sinus (fig. 3). The mucocele was widely opened by ESS under general anesthesia. The drainage route for the mucocele has remained open for 1 month postoperatively.

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from the frontal sinus to the anterior ethmoid sinus (fig. 2a, b). The mucocele was homogeneous, expansive, and accompanied by bony destruction. MRI confirmed a mucocele from the frontal sinus to the anterior ethmoid sinus (fig. 2c, d). The lesion was well-circumscribed and hypointense on T1-weighted imaging (WI) and hyperintense on T2-WI. Based on all the evidence, we diagnosed posttraumatic PSM. The mucocele was endoscopically opened and drained using a navigation system under general anesthesia. The patient was discharged on postoperative day 5 without any severe complications after ESS. The mucocele has not recurred on CT as of 7 months postoperatively (fig. 2e, f). The ocular deviation between the lesional and contralateral sides improved from 3.33 mm (fig. 2a) to 1.02 mm (fig. 2e) postoperatively.

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Kojima et al.: Clinical Features of Patients Treated with Endoscopic Sinus Surgery for Posttraumatic Paranasal Sinus Mucocele

a

b

a

b

c

d

Fig. 4. Radiological and endoscopic findings in case 4. Axial (a) and coronal (b) CT show that the right frontal sinus is occupied with bone destruction (arrowheads). Sagittal (c) and coronal (d) T2-WI MRI confirm the frontal sinus mucocele (arrows) without any invasion to the brain. The 0-degree (e) and 70-degree (f) endoscopic findings (arrowheads) confirm that the frontal sinus mucocele was completely open in the right nasal cavity during ESS. g Endoscopy shows that the mucocele has remained open for 5 months after ESS. S = Septum; MT = middle turbinate.

e

f

Color version available online

Fig. 3. Radiological findings in case 2. A mucocele (arrows) is apparent in the left frontal sinus with bony destruction (arrowheads) on axial (a) and coronal (b) CT.

g

Case 4 A 76-year-old man complaining of forehead swelling visited an otolaryngologist. He had also had a depressed fracture of the frontal bone at 46 years of age. CT delineated a wellcircumscribed cystic lesion with bony defect in the right frontal sinus (fig. 4a, b). MRI confirmed

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Case 3 A 49-year-old man was identified with sensorineural hearing loss on pure-tone audiometry by an otolaryngologist. Suspecting a cerebellopontine angle tumor, MRI was performed. Although no cerebellopontine angle tumor was detected, a cystic lesion in the frontal sinus was pointed out. Based on a history of facial bruising in a traffic accident at 29 years of age, we diagnosed posttraumatic PSM. The mucocele was drained by ESS under general anesthesia. The drainage route for the PSM has remained open for 17 months postoperatively.

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a

b

c

d

Fig. 5. Radiological findings in case 5. Coronal images show the left frontal sinus occupied with bone destruction (arrows) and a partition wall in the PSM (arrowheads) before the first (a, b) and second (c, d) surgeries. a–c CT. d T2-WI MRI.

that the cystic lesion was hypointense on T1-WI and isointense on T2-WI (fig. 4c, d). ESS was performed with a navigation system under general anesthesia, and the mucocele was opened widely (fig. 4e, f). The patient was discharged on postoperative day 4 without an eventful course. The drainage route for the mucocele has remained open for 5 months postoperatively (fig. 4g). The frontal sinus was opened without discharge and swollen mucosa, as the postoperative E score [14] was 0%. Case 5 A 45-year-old man suffering from diplopia initially visited an ophthalmologist. He had also sustained a depressed fracture of the frontal bone from a quarrel at the age of 16 years. CT delineated a well-circumscribed cystic lesion with bony defect in the left frontal sinus (fig. 5a, b). The partition wall in the PSM was observed on CT (fig. 5a, b). During the primary ESS, we did not remove the membranous partition wall in the frontal sinus because we confirmed that the PSM was a unilocular cyst and achieved a wide opening. However, headache appeared 5 months postoperatively. The recurrence of the PSM was confirmed on CT and MRI (fig. 5c, d). The partition wall in the recurrent PSM was not observed on CT (fig. 5c). However, T2-WI MRI delineated the membrane-like closure (fig. 5d). During reoperation using a navigation system, the membranous partition wall was removed with forceps. The drainage route for the PSM has remained open for 7 months postoperatively.

This study presented the clinical features of patients with posttraumatic PSMs. PSM is a cystic lesion caused by secretions from the paranasal mucosa and the blockage of the associated drainage routes. In terms of pathogenesis, posttraumatic PSM is thought to be the least

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Discussion

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frequent type, whereas postoperative PSM is the most common [17]. Only a few cases of posttraumatic PSM have been reported previously [10–12]. In our previous retrospective study about 219 patients with PSM treated over 13 years, posttraumatic PSM was found in only 1 patient (0.5%), compared to postoperative PSM which was found in 79.0% (n = 173) and idiopathic PSM in 20.5% (n = 45) [13]. We could not accurately analyze the clinical features of patients with posttraumatic PSM at that time [13]. A lesson learned from our previous study was that obtaining a detailed history of the injury is crucial to the diagnosis of posttraumatic PSM. Posttraumatic PSM comprised 7% of all PSMs that we encountered over a recent 5-year period in this study, which is similar to a previous report [17]. We still cannot deny that some cases of idiopathic PSM might have been caused by a head injury. The frequency of posttraumatic PSM might be increased in the future if patients were interviewed in greater detail regarding their history of head injury. Radiological imaging tests are indispensable to diagnosis. PSM is defined by the characteristic homogeneous, isodense, and expansile well-circumscribed mass [13, 16, 17]. The intensity on MRI varies depending on the contents of the mucocele [18]. We have previously reported that postoperative PSM is predominantly located in the maxillary sinus, and idiopathic PSM predominantly involves the anterior ethmoid sinus [13]. The majority of patients (approximately 80%) with postoperative PSM had undergone Caldwell-Luc procedures. This is one reason why postoperative PSM is predominantly located in the maxillary sinus in the healing process. With regard to idiopathic PSM, blockage of anterior ethmoidal drainage routes might result from inflammation of the middle nasal meatus mucosa, along with contributing factors such as infection (bacterial, vial, and/or fungal), allergy, and mucociliary impairments [19]. Posttraumatic PSM has been reported to be located mainly in the frontal sinus [12]. All patients with posttraumatic PSM in this study also showed a mucocele located in the frontal sinus. Since the frontal sinus is located in the anterosuperior part of the paranasal sinuses, it may be susceptible to traumatic injury. Furthermore, because the nasofrontal duct at the ostium of the frontal sinus is narrow and long, it is prone to obstruction. Posttraumatic PSMs are thus thought to predominantly involve the frontal sinus. Symptoms developed due to inflammatory conditions resulting from an infection in the mucoceles and increased secretion volume and pressure. Acute infection in the mucoceles results in local heat, headache, and forehead redness. Symptoms from increased pressure also included exophthalmos, double vision and forehead swelling, in addition to headache. Blurred vision, diplopia, visual field defects and forehead swelling due to pressure symptoms from the mucocele were observed in 3 patients in this study, and headache due to infection was seen in 1 patient. Patients with PSM may not only visit otolaryngologists but also neurosurgeons and ophthalmologists. Cooperation among these specialists is thus very important for the valid management of PSM. ESS, extranasal sinus surgery, or a two-way combined approach can be performed for PSM [4]. ESS is a relatively less-invasive surgery, so that all 5 cases in this study could be discharged within 1 week after surgery (postoperative days 4–5). To avoid recurrent obstruction of the frontal sinus drainage route, the mucoceles must be widely opened. For the endoscopic approach, Draf IIb and III drainage procedures were required to enlarge the outflow tract from the frontal sinus [20–22]. The opened drainage route appeared to be narrow in case 4 (fig. 4g), but the patient had not shown any symptoms after ESS. Case 5 experienced headache caused by reclosure of the route to the PSM. We should therefore have performed Draf IIb or III procedures in cases 4 and 5. Furthermore, a two-way combined approach is required when the PSM is located laterally in the frontal sinus to open and dilate the PSM. Reoperation was required in 1 patient (20%). If we could accumulate more patients with posttraumatic PSM, the ratio of recurrence might be decreased. Previous studies have also

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Kojima et al.: Clinical Features of Patients Treated with Endoscopic Sinus Surgery for Posttraumatic Paranasal Sinus Mucocele

devised methods such as insertion of silicone tubes [23, 24] or stents [25], obliteration, and complete removal of the cyst and replacement with fat [26]. We did not apply any tubes to the patients in this study. Examining the usefulness of tube insertion to prevent the obstruction of drainage routes is also necessary in the future. Periodic follow-up on a long-term basis using endoscopy is necessary. Postoperative E scores could be applied to evaluate the condition of surgically opened drainage routes [16]. An E score of 0% indicates that the sinuses have been widely opened without any fluid. On the other hand, an E score of 100% means that the sinuses have closed and/or filled with fluid, and radiological imaging is required. Some patients might not have visited us if their symptoms had resolved completely. Some patients who lived far from our hospital were followed up by other doctors in other hospitals. It was difficult to observe the patients for a longer follow-up period, and facilitating the data collection of patients’ clinical courses was important in such situations. In conclusion, symptoms of patients with posttraumatic PSM developed long after the injury. The important lessons to be learned for the management of posttraumatic PSM are the need to obtain a detailed history at the first visit to achieve a definitive diagnosis and a surgical strategy to enlarge the route of cyst drainage to resolve symptoms and avoid recurrence. Furthermore, postoperative long-term follow-up is also important to avoid overlooking the recurrence of PSM. Disclosure Statement The authors have no conflicts of interest to declare.

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Clinical Features of Patients Treated with Endoscopic Sinus Surgery for Posttraumatic Paranasal Sinus Mucocele.

The purpose of this study was to analyze the clinical features of patients with posttraumatic paranasal sinus mucocele (PSM)...
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