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Silent sinus syndrome causing cyclovertical diplopia masquerading as superior oblique paresis in the fellow eye
Silent sinus syndrome causing cyclovertical diplopia masquerading as superior oblique paresis in the fellow eye Cheng Zhang, MD, Pittaya Phamonvaechavan, MD, Alex Christoff, CO, and David L. Guyton, MD
Silent sinus syndrome is an insidious maxillary sinus inflammatory disease causing a lowering, thinning, or even absorption of the orbital floor. Patients usually present with progressive enophthalmos and hypoglobus. We report a 41-year-old man with silent sinus syndrome who presented with cyclovertical diplopia masquerading as superior oblique muscle paresis in the fellow eye. Inferior oblique myectomy in the fellow eye resulted in excellent alignment.
Case Report
A
41-year-old healthy white male pilot developed new binocular cyclovertical double vision 9 months previously. An oculoplastic surgeon measured 4 mm of right enophthalmos and 4–5 mm of right hypoglobus (Figure 1). Intermittent vertical diplopia increased in right gaze. A computed tomographic scan showed maxillary sinus inflammation and loss of bone and bowing downward of the right orbital floor, establishing the diagnosis of silent sinus syndrome (Figure 2). An ear, nose, and throat surgeon performed right endoscopic surgery to clear the infection and inflammation from the right maxillary sinus. The diplopia did not improve and continued to worsen. Prior to planned elevation of the orbital floor with an implant, the patient was referred to our institution. Visual acuity was 20/20 in each eye, and with a slight chin-up head posture, the patient had 50 arcsec stereopsis. There was a 2-mm downward displacement of the right globe with mild enophthalmos (Figure 1C). Ductions appeared full, and versions showed 12 apparent overaction of the left inferior oblique muscle (Figure 1A) and 1 apparent underaction of the left superior oblique muscle. In straight ahead gaze (Figure 3), there was a left hyperdeviation (LHT) of 7D at distance and 4D at near. Right gaze measured LHT 20D and left gaze measured no shift. Head tilt to the right showed LHT 8D; head tilt to the left showed LHT 16D. Fundus examination revealed 11 (mild) extorsion of the right fundus and trace extorsion of the left fundus.1 Author affiliations: The Krieger Children’s Eye Center at the Wilmer Eye Institute, The Johns Hopkins University School of Medicine, Baltimore, Maryland Submitted February 14, 2010. Revision accepted July 23, 2010. Reprint requests: Cheng Zhang, MD, The Wilmer Institute 233, The Johns Hopkins Hospital, 600 N. Wolfe Street, Baltimore, MD 21287-9028 (email: [email protected]). J AAPOS 2010;14:450-452. Copyright Ó 2010 by the American Association for Pediatric Ophthalmology and Strabismus. 1091-8531/$36.00 doi:10.1016/j.jaapos.2010.07.004
450
A Lancaster red-green test2 was performed (Figure 4A). With right eye fixation, the test showed a pattern suggestive of a left superior oblique paresis, but with subjective extorsion only of the right eye, which increased in upgaze. A small V-pattern was present. Based on clinical and radiological findings, a diagnosis of right silent sinus syndrome was made. The cyclovertical double vision, which appeared to be the result of a left superior oblique paresis, was most likely the result of downward displacement of the right globe, tightening the right inferior rectus and inferior oblique muscles, and loosening the right superior rectus and superior oblique muscles, leading to the right hypodeviation and extorsion of the right eye. Neither elevation of the right orbital floor nor simple recession of the right inferior rectus muscle was felt likely to reverse the prominent extorsion. A Harada-Ito procedure on the right superior oblique tendon was considered but was rejected because of the hyperdeviation being worse on right gaze. Guided by the pattern of an apparent overacting left inferior oblique muscle, therefore, we recommended and performed a large myectomy of the left inferior oblique muscle. On forced ductions at surgery, the right inferior rectus muscle was slightly tight. Six weeks later, realignment was practically complete (Figure 4B), with diplopia only in extremes of gaze. Ductions appeared full and versions comitant. There was orthophoria at distance and near, with only an eso flick in right and left gaze.
Discussion Silent sinus syndrome was first described by Soparkar and colleagues in 1994.3 It was found to occur commonly in the third to fifth decade of life, with an average hypoglobus measuring 3.4 mm.4 The evolution of the disease process is usually slow, likely due to the asymptomatic chronic maxillary sinus inflammation. Negative pressure has been documented in the obstructed maxillary sinus, possibly causing shrinkage of the sinus with subsequent lowering of the orbital floor.5,6 Patients with silent sinus syndrome can present with intermittent or constant vertical double vision, with or without limitation of ocular rotations. Cases with limited ispilateral supraduction may be related to fibrosis or loss of the elasticity of the inferior rectus muscle.7 One patient reported by Borruat and colleagues4 had a variable right hypertropia with silent sinus syndrome on the left side, but the authors did not describe the strabismus in detail.
Journal of AAPOS
Volume 14 Number 5 / October 2010
Zhang et al
451
LHT 14
LHT 20
LHT 7
No shift
No shift
LHT 8
LHT 16
FIG 3. Measurements of misalignment in various directions of gaze, and on right and left head tilts (measurements in prism diopters).
FIG 1. A, The patient’s left eye showed 12 apparent inferior oblique overaction in right gaze (arrow). B, Normal right inferior oblique muscle action in left gaze (arrow). C, The right eye showed mild enophthalmos and hypoglobus on looking straight ahead with the nonpreferred left eye. (Color version of figure is available online.)
FIG 4. Lancaster red-green plots (dark streaks: right eye; light streak: left eye): preoperative (A) and postoperative (B).
FIG 2. Computed tomographic scan, prior to sinus surgery, showing absence of bone and bowing downward of the floor of the right orbit (arrow), with downward displacement of the globe and of the right inferior rectus muscle.
The ocular motility in our patient was abnormal ipsilateral to the sinus disease; that is, there was mild limitation of supraduction of the right eye presumably from fibrosis of the inferior rectus muscle and/or its surrounding connective tissue, or simply from tightening of the inferior rectus
Journal of AAPOS
muscle from the downwardly displaced globe. This created depression of the right eye in right gaze, mimicking an underacting left superior oblique muscle with an apparent overaction of the left inferior oblique muscle. The 3-step test was consistent with a left superior oblique paresis. The only inconsistency was more extorsion of the right fundus than the left, undoubtedly due to the tightening of both the right inferior rectus and the inferior oblique muscles. With preferred fixation with the right eye, the patient presented with a hyperdeviation of his left eye. Previous studies have described mild cases of silent sinus syndrome in which the enophthalmos and hypoglobus can be corrected by simple opening and drainage of the sinus, obviating the need for orbital floor reconstruction. In virtually all of these cases, the diplopia improved.8 Iseli and colleagues9 reported 1 case of mild silent sinus syndrome with vertical diplopia successfully treated with prism. In cases with severe hypoglobus, however, improvement of the diplopia has occurred only after orbital floor reconstruction. Our patient showed decreased enophthalmos and hypoglobus after the sinus procedure, but with persistent vertical diplopia. Guided by the pattern of
452
Zhang et al
Volume 14 Number 5 / October 2010
misalignment masquerading as the pattern of a left superior oblique paresis, we were able to cure the misalignment by a balancing procedure on the left eye, a large left inferior oblique myectomy.
Literature Search A review of the literature was performed using the PubMed database with the following search terms: silent sinus syndrome AND strabismus, silent sinus syndrome AND diplopia. References 1. Guyton DL. Clinical assessment of ocular torsion. Am Orthopt J 1983; 33:7-15. 2. Christoff A, Guyton DL. The Lancaster red-green test. Am Orthopt J 2006;56:157-65.
3. Soparkar CN, Patrinely JR, Cuaycong MJ, et al. The silent sinus syndrome: A cause of spontaneous enophthalmos. Ophthalmology 1994; 101:772-8. 4. Borruat FX, Jaques B, Du¨rig J. Transient vertical diplopia and silent sinus disorder. J Neuroophthalmol 1999;19:173-75. 5. Davidson JK, Soparkar CN, Williams JB, Patrinely JR. Negative sinus pressure and normal predisease imaging in silent sinus syndrome. Arch Ophthalmol 1999;117:1653-4. 6. Numa WA, Desai U, Gold DR, Heher KL, Annino DJ. Silent sinus syndrome: A case presentation and comprehensive review of all 84 reported cases. Ann Otol Rhinol Laryngol 2005;114:688-94. 7. Yip CC, McCulley TJ, Kersten RC, Tami TA, Kulwin DR. Silent sinus syndrome as a cause of diplopia in a child. J Pediatr Ophthalmol Strabismus 2003;40:309-11. 8. Wan MK, Francis IC, Carter PR, Griffits R, van Rooijen ML, Coroneo MT. The spectrum of presentation of silent sinus syndrome. J Neuroophthalmol 2000;20:207-12. 9. Iseli HP, Hafezi F, Mojon DS. Conservative treatment of vertical diplopia in a patient with silent sinus syndrome. Ophthalmologica 2003; 217:308-9.
Journal of AAPOS
Silent sinus syndrome is an insidious maxillary sinus inflammatory disease causing a lowering, thinning, or even absorption of the orbital floor. Patients usually present with progressive enophthalmos and hypoglobus. We report a 41-year-old man with silent sinus syndrome who presented with cyclovertical diplopia masquerading as superior oblique muscle paresis in the fellow eye. Inferior oblique myectomy in the fellow eye resulted in excellent alignment.
Case Report
A
41-year-old healthy white male pilot developed new binocular cyclovertical double vision 9 months previously. An oculoplastic surgeon measured 4 mm of right enophthalmos and 4–5 mm of right hypoglobus (Figure 1). Intermittent vertical diplopia increased in right gaze. A computed tomographic scan showed maxillary sinus inflammation and loss of bone and bowing downward of the right orbital floor, establishing the diagnosis of silent sinus syndrome (Figure 2). An ear, nose, and throat surgeon performed right endoscopic surgery to clear the infection and inflammation from the right maxillary sinus. The diplopia did not improve and continued to worsen. Prior to planned elevation of the orbital floor with an implant, the patient was referred to our institution. Visual acuity was 20/20 in each eye, and with a slight chin-up head posture, the patient had 50 arcsec stereopsis. There was a 2-mm downward displacement of the right globe with mild enophthalmos (Figure 1C). Ductions appeared full, and versions showed 12 apparent overaction of the left inferior oblique muscle (Figure 1A) and 1 apparent underaction of the left superior oblique muscle. In straight ahead gaze (Figure 3), there was a left hyperdeviation (LHT) of 7D at distance and 4D at near. Right gaze measured LHT 20D and left gaze measured no shift. Head tilt to the right showed LHT 8D; head tilt to the left showed LHT 16D. Fundus examination revealed 11 (mild) extorsion of the right fundus and trace extorsion of the left fundus.1 Author affiliations: The Krieger Children’s Eye Center at the Wilmer Eye Institute, The Johns Hopkins University School of Medicine, Baltimore, Maryland Submitted February 14, 2010. Revision accepted July 23, 2010. Reprint requests: Cheng Zhang, MD, The Wilmer Institute 233, The Johns Hopkins Hospital, 600 N. Wolfe Street, Baltimore, MD 21287-9028 (email: [email protected]). J AAPOS 2010;14:450-452. Copyright Ó 2010 by the American Association for Pediatric Ophthalmology and Strabismus. 1091-8531/$36.00 doi:10.1016/j.jaapos.2010.07.004
450
A Lancaster red-green test2 was performed (Figure 4A). With right eye fixation, the test showed a pattern suggestive of a left superior oblique paresis, but with subjective extorsion only of the right eye, which increased in upgaze. A small V-pattern was present. Based on clinical and radiological findings, a diagnosis of right silent sinus syndrome was made. The cyclovertical double vision, which appeared to be the result of a left superior oblique paresis, was most likely the result of downward displacement of the right globe, tightening the right inferior rectus and inferior oblique muscles, and loosening the right superior rectus and superior oblique muscles, leading to the right hypodeviation and extorsion of the right eye. Neither elevation of the right orbital floor nor simple recession of the right inferior rectus muscle was felt likely to reverse the prominent extorsion. A Harada-Ito procedure on the right superior oblique tendon was considered but was rejected because of the hyperdeviation being worse on right gaze. Guided by the pattern of an apparent overacting left inferior oblique muscle, therefore, we recommended and performed a large myectomy of the left inferior oblique muscle. On forced ductions at surgery, the right inferior rectus muscle was slightly tight. Six weeks later, realignment was practically complete (Figure 4B), with diplopia only in extremes of gaze. Ductions appeared full and versions comitant. There was orthophoria at distance and near, with only an eso flick in right and left gaze.
Discussion Silent sinus syndrome was first described by Soparkar and colleagues in 1994.3 It was found to occur commonly in the third to fifth decade of life, with an average hypoglobus measuring 3.4 mm.4 The evolution of the disease process is usually slow, likely due to the asymptomatic chronic maxillary sinus inflammation. Negative pressure has been documented in the obstructed maxillary sinus, possibly causing shrinkage of the sinus with subsequent lowering of the orbital floor.5,6 Patients with silent sinus syndrome can present with intermittent or constant vertical double vision, with or without limitation of ocular rotations. Cases with limited ispilateral supraduction may be related to fibrosis or loss of the elasticity of the inferior rectus muscle.7 One patient reported by Borruat and colleagues4 had a variable right hypertropia with silent sinus syndrome on the left side, but the authors did not describe the strabismus in detail.
Journal of AAPOS
Volume 14 Number 5 / October 2010
Zhang et al
451
LHT 14
LHT 20
LHT 7
No shift
No shift
LHT 8
LHT 16
FIG 3. Measurements of misalignment in various directions of gaze, and on right and left head tilts (measurements in prism diopters).
FIG 1. A, The patient’s left eye showed 12 apparent inferior oblique overaction in right gaze (arrow). B, Normal right inferior oblique muscle action in left gaze (arrow). C, The right eye showed mild enophthalmos and hypoglobus on looking straight ahead with the nonpreferred left eye. (Color version of figure is available online.)
FIG 4. Lancaster red-green plots (dark streaks: right eye; light streak: left eye): preoperative (A) and postoperative (B).
FIG 2. Computed tomographic scan, prior to sinus surgery, showing absence of bone and bowing downward of the floor of the right orbit (arrow), with downward displacement of the globe and of the right inferior rectus muscle.
The ocular motility in our patient was abnormal ipsilateral to the sinus disease; that is, there was mild limitation of supraduction of the right eye presumably from fibrosis of the inferior rectus muscle and/or its surrounding connective tissue, or simply from tightening of the inferior rectus
Journal of AAPOS
muscle from the downwardly displaced globe. This created depression of the right eye in right gaze, mimicking an underacting left superior oblique muscle with an apparent overaction of the left inferior oblique muscle. The 3-step test was consistent with a left superior oblique paresis. The only inconsistency was more extorsion of the right fundus than the left, undoubtedly due to the tightening of both the right inferior rectus and the inferior oblique muscles. With preferred fixation with the right eye, the patient presented with a hyperdeviation of his left eye. Previous studies have described mild cases of silent sinus syndrome in which the enophthalmos and hypoglobus can be corrected by simple opening and drainage of the sinus, obviating the need for orbital floor reconstruction. In virtually all of these cases, the diplopia improved.8 Iseli and colleagues9 reported 1 case of mild silent sinus syndrome with vertical diplopia successfully treated with prism. In cases with severe hypoglobus, however, improvement of the diplopia has occurred only after orbital floor reconstruction. Our patient showed decreased enophthalmos and hypoglobus after the sinus procedure, but with persistent vertical diplopia. Guided by the pattern of
452
Zhang et al
Volume 14 Number 5 / October 2010
misalignment masquerading as the pattern of a left superior oblique paresis, we were able to cure the misalignment by a balancing procedure on the left eye, a large left inferior oblique myectomy.
Literature Search A review of the literature was performed using the PubMed database with the following search terms: silent sinus syndrome AND strabismus, silent sinus syndrome AND diplopia. References 1. Guyton DL. Clinical assessment of ocular torsion. Am Orthopt J 1983; 33:7-15. 2. Christoff A, Guyton DL. The Lancaster red-green test. Am Orthopt J 2006;56:157-65.
3. Soparkar CN, Patrinely JR, Cuaycong MJ, et al. The silent sinus syndrome: A cause of spontaneous enophthalmos. Ophthalmology 1994; 101:772-8. 4. Borruat FX, Jaques B, Du¨rig J. Transient vertical diplopia and silent sinus disorder. J Neuroophthalmol 1999;19:173-75. 5. Davidson JK, Soparkar CN, Williams JB, Patrinely JR. Negative sinus pressure and normal predisease imaging in silent sinus syndrome. Arch Ophthalmol 1999;117:1653-4. 6. Numa WA, Desai U, Gold DR, Heher KL, Annino DJ. Silent sinus syndrome: A case presentation and comprehensive review of all 84 reported cases. Ann Otol Rhinol Laryngol 2005;114:688-94. 7. Yip CC, McCulley TJ, Kersten RC, Tami TA, Kulwin DR. Silent sinus syndrome as a cause of diplopia in a child. J Pediatr Ophthalmol Strabismus 2003;40:309-11. 8. Wan MK, Francis IC, Carter PR, Griffits R, van Rooijen ML, Coroneo MT. The spectrum of presentation of silent sinus syndrome. J Neuroophthalmol 2000;20:207-12. 9. Iseli HP, Hafezi F, Mojon DS. Conservative treatment of vertical diplopia in a patient with silent sinus syndrome. Ophthalmologica 2003; 217:308-9.
Journal of AAPOS