Eur Arch Otorhinolaryngol DOI 10.1007/s00405-013-2538-5
RHINOLOGY
Effect of endoscopic marsupialization of paranasal sinus mucoceles involving the orbit: a review of 27 cases Il Gyu Kang • Seon Tae Kim • Joo Hyun Jung • Ju Young Paik • Joo Hyun Woo • Heung Eog Cha Mi Jung Chi • Sung Min Jin • Kyung Chul Lee
•
Received: 23 December 2012 / Accepted: 25 April 2013 Ó Springer-Verlag Berlin Heidelberg 2013
Abstract Paranasal sinus mucoceles (PSMs) can involve the orbit because the topographic anatomies of the paranasal sinuses and orbit are interrelated. We encountered 27 patients with PSMs involving the orbit that caused orbital symptoms. In this study, we evaluated the frequent symptoms and signs of PSMs involving the orbit, and report postoperative changes of orbital symptoms including the effects on visual acuity. A retrospective chart review, radiologic evaluation, and interviews were conducted after Institutional Review Board approval was obtained. Over the past 11 years, we encountered 27 patients with PSMs involving the orbit. We classified the patients according to orbital symptoms and PSM origin, and evaluated the surgical outcomes. A total of 27 patients (17 males and 10 females) with PSMs involving the orbit were included in the present study. The mean patient age was 51.0 ± 9.7 years (range 32–90) and the mean follow-up period was 20.5 months (range 2–84). Proptosis (15/27, 56 %) was the most common symptom. Other common symptoms included orbital pain (9/27, 33 %), decreased visual acuity or vision loss (9/27, 33 %), and diplopia (7/27, 26 %). All symptoms except for vision loss were improved by I. G. Kang S. T. Kim J. H. Jung J. Y. Paik J. H. Woo H. E. Cha Department of Otolaryngology, Head and Neck Surgery, Gil Medical Center, Gachon University, Incheon, South Korea M. J. Chi Department of Ophthalmology, Gil Medical Center, Gachon University, Incheon, South Korea S. M. Jin K. C. Lee (&) Department of Otorhinolaryngology, Kangbuk Samsung Hospital, Sungkyunkwan University School of Medicine, 108, Pyung-dong, Jongro-gu, Seoul, South Korea e-mail:
[email protected]
endoscopic marsupialization regardless of the disease period. Four out of five patients with decreased visual acuity experienced complete recovery. The remaining patient showed partial improvement after surgery. Four patients who were blind when they initially visited the hospital did not show any improvement after surgery. For PSM patients with decreased visual acuity, we can predict that vision will improve after surgery regardless of the disease duration. However, blindness will probably not resolve after endoscopic marsupialization. Even if the orbital symptoms (except for blindness) have persisted for a long time, surgery could still produce positive outcomes. Keywords Paranasal sinus Mucocele Visual acuity Blind Orbit Ophthalmic manifestations
Introduction Paranasal sinus mucoceles (PSMs) develop due to obstruction of the paranasal sinuses (idiopathic or secondary to trauma), inflammation, or tumors. Primary PSMs arise from anatomic obstructions caused by masses, mucosal hyperplasia, or mechanical factors. Formation of primary PSMs may be attributed to the blockage of mucus drainage due to inflammation, secretory duct obstruction, cystic dilatation of the mucosal glands, and cystic degeneration of polyps [1–3]. Alternatively, secondary PSMs can arise from the sequestration of residual mucosa in a wound or long-term retention of fluid in tissues [2, 3]. There have been no reported differences in PSM incidence associated with race or gender although these lesions usually develop in patients in their 40s and 70s, and are rare in children [4]. PSM growth is typically unilateral and insidious, and may involve the paranasal bony structure, orbital wall, and skull base.
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Fig. 1 a CT scans showing a mucocele in the right sphenoid sinus that had compressed the optic nerve. b The postoperative CT scan
PSM symptoms are variable and depend on the direction of lesion extension into the neighboring structures [5]. Thus, characteristics of the anatomic sites affected by PSMs are important. The ethmoid and frontal sinuses are common sites of PSM development while the maxillary and sphenoid sinuses are relatively uncommon locations [6, 7]. Orbital symptoms, such as proptosis, frontal headaches, and limited orbital movement, are related to compression by PSMs [8–10]. Surgical techniques for ameliorating PSMs include radical excision and endoscopic marsupialization. Currently, endoscopic marsupialization is generally performed to treat patients with PSMs. In this retrospective study, we reviewed 27 cases of PSMs involving the orbit encountered over the past 11 years. Clinical symptoms and signs, especially visual acuity, were analyzed. We also examined the associations between common orbital symptoms, PSM location, and the results of endoscopic marsupialization.
Fig. 2 CT image showing a mucocele in an Onodi cell along with dehiscence of the lateral cell wall and optic nerve (arrow)
Materials and methods Patients and data acquisition The present study was approved by the Institutional Review Board of Gachon University Gil Medical Center. This study included 27 patients with PSMs involving the orbit who underwent endoscopic marsupialization between 2000 and 2011. Charts and radiologic data were reviewed. Telephone interviews and meetings with the patients were conducted to evaluate the present symptoms or surgical
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outcomes. Orbital extension was assessed based on a combination of physical examination results and CT findings with or without MRI data. In addition, we obtained information regarding visual disturbances, PSM origins, previous surgeries or trauma experienced by the patients, and surgical results. Intraorbital extension was defined as a PSM affecting or damaging the lamina papyracea, periorbita, orbital muscle, or optic nerve (Figs. 1a, 2). Visual acuity of all patients was documented by an ophthalmologist before and after endoscopic marsupialization.
Eur Arch Otorhinolaryngol Table 1 Clinical symptoms of paranasal sinus mucoceles (PSMs) involving the orbit, symptom duration, and endoscopic marsupialization outcomes Patient no.
Patient gender/ age
Surgical history
Mucocele site
Presenting symptoms
Visual acuity
1
M/56
No
SS
DP
0.22
0.05
1 week
FR of DP
2
M/42
BOF
FS
PS
0
0.05
2 months
FR of PS
3 4
M/41 M/65
No C-L
SS PES
PS VD
0.1 3
0.15 0.15
1 year 2 days
FR of PS NR of VD FR of PS
5
F/39
No
PES
PS, VD
0.4
1.4
3 years
PR of VD (1.4 to [0.7)
6
F/64
No
FS
PS, OP
0.3
0.4
1 week
FR of PS, OP
7
F/66
ESS
FS
PS, OP
0.22
0.3
1 year
FR of PS, OP
8
M/48
ESS
AES
PS, DP
0.15
0
1 month
FR of PS, DP
9
M/57
No
SS
VD
0.52
0.05
1 year
FR of VD (0.52 to [0)
10
F/56
No
PES
PS, VD
3
0.15
PS: 5 months VD: 3 years
FR of PS, NR of VD
11
F/32
No
AES
OP
0.1
0.1
2 days
FR of OP
12
M/36
ESS
FS
PS, DP
0
0
3 days
FR of PS, DP
13
F/43
No
AES
PS, OP
0.15
0
5 days
FR of PS, OP
14
M/47
No
SS
PS, VD, Seizure
0.1
3
PS: 15 years VD: 5 years
FR of PS, NR of VD
15
M/65
No
FS
PS, VD
0.4
3
2 days
FR of PS, NR of VD
16
M/47
No
PES
PS, DP
0.15
0.1
1 month
FR of PS, DP
17
F/38
No
ES
DP, OP
0
-0.08
DP: 3 days OP: 1 day
FR of DP, OP
18
M/90
ESS
FS
PS, VD, OP
0.4
1
1 day
FR of PS, OP, VD (1 to [0.52)
19
M/47
No
SS
OP
0.05
0.1
2 years
FR of OP
20
M/57
No
ES
OP
0.1
0.15
4 months
FR of OP
21 22
M/49 M/52
No C-L
AES MS
BV OP
0 0.3
0 0.15
1 year 14 days
FR of BV FR of OP
23
M/36
No
AES
PS
0
0.05
7 days
FR of PS
24
F/58
No
FS
PS
0.3
0.15
3 days
FR of PS
25
F/52
No
SS
VD
1
0.1
2 years
FR of VD (1 to [0.15)
26
M/56
No
FE
DP
0.15
0.1
1 month
FR of DP
27
F/38
No
PES
VD, DP
0.1
0.3
1 day
FR of DP, VD (0.3 to [0.1)
R
L
Duration of symptoms
Surgical outcomes
BOF blowout fracture, FS frontal sinus, ES ethmoid sinus, AES anterior ethmoid sinus, PES posterior ethmoid sinus, SS sphenoid sinus, FE fronto-ethmoid sinus, ESS endoscopic sinus surgery, C-L Caldwell-Luc operation, PS proptosis, OP orbital pain, VD visual disturbance, DP diplopia, FR full recovery, PR partial recovery, NR no recovery, BV blurred vision
Surgical procedures
Results
Endoscopic marsupialization was performed as follows. With the patient under local or general anesthesia, the nasal cavity was packed with cotton pledgets saturated with epinephrine and lidocaine for 10 min. Using an endoscope (0°, 30°, and 45°), the anteroinferior wall of the PSM was removed with a curette and Blacksley forceps so that the lesion could be sufficiently drained before the contents were completely removed by suctioning. Postoperative packing was then performed. Vision, diplopia, and proptosis were assessed before and after endoscopic marsupialization by an ophthalmologist.
A total of 27 patients (17 males and 10 females) with PSMs involving the orbit were included in our retrospective analysis. The mean patient age was 51.0 ± 9.7 years (range 32–90) and the mean follow-up period was 20.5 months (range 2–84). Seven out of the 27 patients had previously undergone nasal surgery. Four had undergone endoscopic sinus surgery, two had undergone a CaldwellLuc operation, and one patient had undergone surgery to treat a blowout fracture (Table 1). The sites of PSM origin included the ethmoidal (44 %), frontal (26 %), sphenoidal (22 %), and maxillary (4 %) sinuses. One patient had
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Eur Arch Otorhinolaryngol Table 2 Sites of the PSM lesions and clinical symptoms of the patients Frontal sinus (%)
Ethmoid sinus (%)
Sphenoid sinus (%)
Maxillary sinus (%)
Visual disturbance
2 (25)
5 (38)
3 (50)
0
Proptosis
7 (88)
7 (54)
2 (33)
0
Diplopia
2a (25)
5a (38)
1 (17)
0
Orbital pain
3 (38)
4 (31)
1 (17)
1 (100)
a
Fronto–ethmoid mucoceles
Table 3 Endoscopic marsupialization outcomes for nine patients with decreased visual acuity or blindness with and without optic canal dehiscence due to mucoceles Visual acuity improvement
No visual acuity improvement
Optic canal dehiscence
3 (50)
3 (50)
No optic canal dehiscence
2 (66.7)
1 (33.3)
previously experienced paranasal area trauma and a PSM developed in that region. Proptosis (15/27, 56 %) was the most common symptom. Other commonly observed symptoms were orbital pain (9/27, 33 %), decreased visual acuity or vision loss (9/27, 33 %), and diplopia (7/27, 26 %). Symptoms varied according to which sinus was involved (Table 2). PSMs affecting the ethmoid sinus presented as proptosis (54 %, 7/13), decreased visual acuity or vision loss (38 %), diplopia (38 %), and orbital pain (31 %). Frontal sinus mucoceles presented as proptosis (88 %, 7/8), orbital pain (38 %), diplopia (25 %), and decreased visual acuity (25 %). PSMs arising in the sphenoid sinus were associated with decreased visual acuity (50 %, 3/6), proptosis (33 %), orbital pain (17 %), and diplopia (17 %). Thirteen (48 %) out of the 27 patients had multiple orbital symptoms. PSM locations associated with decreased visual acuity or visual loss included the posterior ethmoidal (4/9, 44 %), sphenoidal (3/9, 33 %), and frontal (2/9, 22 %) sinuses. Two individuals had bilateral PSMs, but only lesions arising on one side induced orbital symptoms. All patients underwent endoscopic marsupialization. Proptosis, orbital pain, and diplopia were completely resolved in all patients after treatment. Among the individuals with decreased visual acuity, four out of the five completely improved after surgery. The remaining patient showed partial improvement. Patients who experienced a loss of vision did not improve after surgery (Table 1). In cases of decreased visual acuity, partial or complete recovery was achieved with surgery even if the PSMs had persisted for long period of time. For example, a patient with 3-year history of the disease achieved partial recovery of visual acuity after surgery. However, blindness was
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unaffected by surgery even when the disease had persisted for only 2 days. Out of nine individuals with decreased visual acuity or blindness, six had optic canal dehiscence due to a PSM. Three out of the six PSM patients experienced improved visual acuity after endoscopic marsupialization while the other three did not. Among the three patients without optic canal dehiscence, two experienced improved visual acuity after endoscopic marsupialization while the remaining one did not (Table 3).
Discussion PSMs with orbital extension result in orbital displacement, proptosis, diplopia, and decreased visual acuity. In the present study, PSMs were associated with diverse orbital symptoms regardless of origin. Previous investigations have demonstrated that PSMs occur in all sinuses including the frontal (65–75 %) and anterior ethmoidal (30 %), but rarely develop in the posterior and sphenoidal sinuses [11– 13]. In our analysis, the most common PSM sites were the ethmoid and frontal sinuses. This is because we analyzed cases of PSMs according to orbital symptoms and signs. Since the ethmoid sinus space is narrower than that of any other sinuses, PSMs arising in this location have a greater tendency to extend into the orbit and are more closely associated with orbital symptoms. Furthermore, ethmoid mucoceles have a higher potential for intraorbital extension while sphenoid mucoceles tend to extend intracranially [6]. Intranasal drainage and marsupialization of mucoceles using endoscopic surgical techniques have emerged as ideal methods for definitive disease management with minimal morbidity and a low recurrence rate [4]. The recurrence rate of mucoceles after osteoplastic and obliteration procedures has been reported to be 19 % (9/47) [14]. In comparison, the recurrence rate was low with minimal complications for two previous endoscopic series [4, 15]. In the present study, seven of the 27 patients had a history of nasal surgery. Four had undergone endoscopic sinus surgery, two had undergone a Caldwell-Luc operation, and one had undergone surgery to correct a blowout fracture. It
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is important for surgeons to maintain sinus drainage so as not to obstruct the sinuses after surgery. This is because accumulated contents cause the bone walls to distend and become thinner when the sinus opening is closed for a long time. In addition, a large amount of mucous exudate leads to the development of mucoceles [16]. In general, the prolonged onset of decreased visual acuity does not improve after surgery [17, 18]. A previous study found that visual improvement occurs in patients with visual disturbances that had persisted for less than 6 months although a shorter interval between symptom onset and surgery did not guarantee an earlier recovery [17]. This same study additionally determined that vision recovery not only depends on the interval between symptom onset and surgery but also the severity of visual disturbance. However, two patients in our study who had developed decreased visual acuity over a long period of time showed improvement after surgery. One had experienced reduced vision for 3 years and improved partially after marsupialization. In the other patient, visual acuity had started to decrease 2 years prior to surgery but improved completely after marsupialization. The mechanisms underlying decreased vision or blindness caused by PSMs include direct mechanical compression of the optic nerve and inflammation due to infection as the mucocele extends to the optic nerve [19]. Prognosis depends on how long the condition has persisted. Lee et al. [17] reported that patients with visual disturbances existing for longer than 6 months fail to recover. In addition, Moriyama et al. [18] showed that patient prognosis is worse when disease period exceeds 2 months. However, our study showed that visual acuity of patients with PSMs could be partially or fully restored even in individuals with substantial visual disturbances and disease periods longer than 2–3 years. These findings clearly indicate that marsupialization to treat PSMs should not be delayed or omitted even for patients with a long history of the disease (even for several years) and visual acuity so weak that counting fingers is challenging or impossible.
Conclusions Based on the results of our retrospective analysis, we made the following conclusions. For PSM patients who experienced gradual decreases in visual acuity, surgical treatment may improve vision regardless of the length of PSM persistence. Therefore, endoscopic marsupialization should be performed as soon as possible. For individuals experiencing a rapid decrease of visual acuity, urgent endoscopic
marsupialization should be performed immediately. However, if the first symptom of PSM is blindness, favorable surgical outcomes cannot be expected. Overall, endoscopic marsupialization is sufficient for improving orbital symptoms associated with PSMs except for blindness. Acknowledgments This work was supported by the Gachon University research fund of 2012.
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