Original Contribution
Laughter-Induced Transient Vision Loss in a Patient With Silent Sinus Syndrome Sara N. Reggie, MD, Krishna Kalyam, MD, John B. Holds, MD, Sophia M. Chung, MD
Background: To report a patient with silent sinus syndrome (SSS) who experienced transient ipsilateral monocular vision loss during intense laughter. Methods: Case report. Results: Our patient’s transient vision loss completely resolved after maxillary sinus decompression and during 7 months of follow-up. Conclusions: Although the precise mechanism of our patient’s vision loss remains undetermined, we suspect that the vascular supply to the eye and/or the optic nerve was compromised as the result of the combination of laughter (causing Valsalva maneuver and increased intrathoracic pressure) and SSS. Journal of Neuro-Ophthalmology 2017;0:1–2 doi: 10.1097/WNO.0000000000000536 © 2017 by North American Neuro-Ophthalmology Society
S
ilent sinus syndrome (SSS) is characterized by blockage of the maxillary osteomeatal complex, resulting in negative maxillary sinus pressure with atelectasis and eventual collapse of the maxillary antrum. We describe a patient with SSS who experienced transient monocular vision loss during laughter or yelling.
up to 20 times in 10 minutes. These episodes were not associated with pain, tearing, flashes, or floaters. She denied lightheadedness, syncope, or similar episodes associated with any other precipitating factors. She also experienced daily left-sided periorbital headaches for 1 year. Computed tomography showed complete opacification of the right maxillary sinus with atelectasis (Fig. 1). The patient was referred to our neuro-ophthalmology service for evaluation of her transient right monocular vision loss. On examination, the patient’s visual acuity was 20/20 in both eyes. Pupillary reactions, color vision, and ocular motility were normal. Exophthalmology measurements were 15 mm, right eye, and 16 mm, left eye. Intraocular pressures were normal bilaterally and the appearance of the anterior and posterior segments was unremarkable. Automated visual fields were full in both eyes. The patient’s medical history was noncontributory. Her only medication was oral contraceptives and she denied the use of tobacco.
CASE REPORT A 32-year-old woman reported a 7-month history of transient vision loss in the right eye that occurred during episodes of intense laughter or yelling. She described the vision loss as brief episodes during which the entire right visual field became dark, lasting a few seconds but occurring Department of Ophthalmology (SNR, JBH, SMC), St. Louis University School of Medicine, St. Louis, Missouri; Department of Ophthalmology and Visual Sciences (KK), Washington University in St. Louis, St. Louis, Missouri; and Ophthalmic Plastic and Cosmetic Surgery Inc. (JBH), St. Louis, Missouri. The authors report no conflicts of interest. Address correspondence to Sara N. Reggie, MD, Department of Ophthalmology, St. Louis University Eye Institute, 1755 South Grand Boulevard, St. Louis, MO 63104; E-mail: [email protected] Reggie et al: J Neuro-Ophthalmol 2017; 0: 1-2
FIG. 1. Coronal orbital computed tomography shows total opacification of the right maxillary sinus and collapse of the right orbital floor.
1
Copyright Ó North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited.
Original Contribution The patient underwent right maxillary sinus decompression via an endoscopic antrostomy. One week postoperatively, she noted improvement in headaches and no recurrence of transient vision loss with intense laughter or yelling and remained asymptomatic over the ensuing 7 months.
DISCUSSION SSS, also known as imploding antrum syndrome or chronic maxillary atelectasis, is due to an obstruction of the osteomeatal complex, which impairs maxillary sinus ventilation and causes entrapment of mucous secretions. This leads to resorption of gases in the sinus causing a negative pressure gradient and collapse of the maxillary antrum (1–4). SSS is found equally in men and women, most commonly in the fourth and fifth decades of life, and it equally affects right and left maxillary sinuses. Patients may present with painless and progressive enophthalmos, hypoglobus, and diplopia (1–8). Neuroimaging is essential to establish the correct diagnosis and usually shows neartotal opacification and contraction of the maxillary sinus, infundibular obstruction, and downward retraction of the orbital floor. In addition, there may be expansion of the orbital volume, bone loss of the orbital floor, and collapse of the uncinate process (3,9–11). Management of SSS involves surgical drainage of the maxillary sinus with or without removal of the ethmoid uncinate process for better visualization and aspiration and culture of sinus secretions. There is a continued debate whether orbital reconstruction should be performed simultaneously during sinus drainage or postponed as a second surgery (1–4,6–8,12–14). We were unable to find similar reports of transient monocular vision loss precipitated by laughing episodes in a patient with SSS. There are, however, reported cases of laughter-induced syncope in patients with vascular disease, which were attributed to a combination of poor cerebral blood flow coupled with stenosis of the external carotid artery (15–17). In addition, laughter leads to a Valsalva maneuver which, in turn, causes increased intrathoracic pressure. If the autonomic nervous system fails to increase heart rate and activate peripheral vasoconstriction, syncope may occur (16,18). Although our patient did not have any known vascular disease, we suspect that her SSS contributed to compromise of retinal blood flow (19). This may have occurred because of decreased internal carotid artery blood flow from increased intrathoracic pressure, lack of autonomic compensation, and compromise of external carotid collateral flow to the ophthalmic artery due to changes in orbital anatomy from SSS. In addition, disturbance in venous outflow has been demonstrated in patients with transient monocular visual loss (20). Although vascular studies would have been helpful in supporting our hypothesis, we are still missing a proven link as to 2
the mechanism of vision loss in our otherwise young, healthy patient. We do not believe that traction on the optic nerve from SSS caused the patient’s vision loss, as she did not have significant enophthalmos or hypoglobus to induce optic nerve stretching. Although the combination of signs and symptoms are rare, laughter-induced transient monocular vision loss may indicate the pressure of SSS and should be considered during patient evaluation.
REFERENCES 1. Soparkar CN, Patrinely JR, Cuaycong MJ, Dailey RA, Kersten RC, Rubin PA, Linberg JV, Howard GR, Donovan DT, Matoba AY, Holds JB. The silent sinus syndrome. A cause of spontaneous enophthalmos. Ophthalmology. 1994;101:772–778. 2. Pula JH, Mehta M. Silent sinus syndrome. Curr Opin Ophthalmol. 2014;25:480–484. 3. 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;114:688–694. 4. Wan MK, Francis IC, Carter PR, Griffits R, van Rooijen ML, Coroneo MT. The spectrum of presentation of silent sinus syndrome. J Neuroophthalmol. 2000;20:207–212. 5. Singman EL, Matta NS, Silbert DI. Silent sinus syndrome presenting after a roller coaster ride: a case report. Am Orthopt J. 2014;64:123–125. 6. Buono LM. The silent sinus syndrome: maxillary sinus atelectasis with enophthalmos and hypoglobus. Curr Opin Ophthalmol. 2004;15:486–489. 7. Arikan OK, Onaran Z, Muluk NB, Yilmazbas P, Yazici I. Enophthalmos due to atelectasis of the maxillary sinus: silent sinus syndrome. J Craniofac Surg. 2009;20:2156–2159. 8. Bahgat M, Bahgat Y, Bahgat A. Silent sinus syndrome. BMJ Case Rep. 2012:2012. doi: 10.1136/bcr-2012-007198. 9. Illner A, Davidson HC, Harnsberger HR, Hoffman J. The silent sinus syndrome: clinical and radiographic findings. AJR Am J Roentgenol. 2002;178:503–506. 10. Gaudino S, Di Lella GM, Piludu F, Martucci M, Schiarelli C, Africa E, Salvolini L, Colosimo C. CT and MRI diagnosis of silent sinus syndrome. Radiol Med. 2013;118:265–272. 11. Bas A, Tutar O, Samanci C, Kepek F. Silent sinus syndrome: CT and MRI findings. BMJ Case Rep. 2012;2012. doi: 10.1136/ bcr-2012-007492. 12. Brandt MG, Wright ED. The silent sinus syndrome is a form of chronic maxillary atelectasis: a systematic review of all reported cases. Am J Rhinol. 2008;22:68–73. 13. Monos T, Levy J, Lifshitz T, Puterman M. The silent sinus syndrome. Isr Med Assoc J. 2005;7:333–335. 14. Cobb AR, Murthy R, Cousin GC, El-Rasheed A, Toma A, Uddin J, Manisali M. Silent sinus syndrome. Br J Oral Maxillofac Surg. 2012;50:e81–e85. 15. Cox SV, Eisenhauer AC, Hreib K. “Seinfeld syncope”. Cathet Cardiovasc Diagn. 1997;42:242. 16. Sarzi Braga S, Manni R, Pedretti RF. Laughter-induced syncope. Lancet. 2005;366:426. 17. Shah AA, Gelber AC. Laughter-induced syncope. Am J Med. 2010;123:609–611. 18. Totah AR, Benbadis SR. Gelastic syncope mistaken for cataplexy. Sleep Med. 2002;3:77–78. 19. Macchi C, Catini C. The anatomy and clinical significance of the collateral circulation between the internal and external carotid arteries through the ophthalmic artery. Ital J Anat Embryol. 1993;1993:23–29. 20. Hsu HY, Chao AC, Chen YY, Yang FY, Chung CP, Sheng WY, Yen MY, Hu HH. Reflux of jugular and retrobulbar venous flow in transient monocular blindness. Ann Neurol. 2008;63:247–253.
Reggie et al: J Neuro-Ophthalmol 2017; 0: 1-2
Copyright Ó North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited.
Laughter-Induced Transient Vision Loss in a Patient With Silent Sinus Syndrome Sara N. Reggie, MD, Krishna Kalyam, MD, John B. Holds, MD, Sophia M. Chung, MD
Background: To report a patient with silent sinus syndrome (SSS) who experienced transient ipsilateral monocular vision loss during intense laughter. Methods: Case report. Results: Our patient’s transient vision loss completely resolved after maxillary sinus decompression and during 7 months of follow-up. Conclusions: Although the precise mechanism of our patient’s vision loss remains undetermined, we suspect that the vascular supply to the eye and/or the optic nerve was compromised as the result of the combination of laughter (causing Valsalva maneuver and increased intrathoracic pressure) and SSS. Journal of Neuro-Ophthalmology 2017;0:1–2 doi: 10.1097/WNO.0000000000000536 © 2017 by North American Neuro-Ophthalmology Society
S
ilent sinus syndrome (SSS) is characterized by blockage of the maxillary osteomeatal complex, resulting in negative maxillary sinus pressure with atelectasis and eventual collapse of the maxillary antrum. We describe a patient with SSS who experienced transient monocular vision loss during laughter or yelling.
up to 20 times in 10 minutes. These episodes were not associated with pain, tearing, flashes, or floaters. She denied lightheadedness, syncope, or similar episodes associated with any other precipitating factors. She also experienced daily left-sided periorbital headaches for 1 year. Computed tomography showed complete opacification of the right maxillary sinus with atelectasis (Fig. 1). The patient was referred to our neuro-ophthalmology service for evaluation of her transient right monocular vision loss. On examination, the patient’s visual acuity was 20/20 in both eyes. Pupillary reactions, color vision, and ocular motility were normal. Exophthalmology measurements were 15 mm, right eye, and 16 mm, left eye. Intraocular pressures were normal bilaterally and the appearance of the anterior and posterior segments was unremarkable. Automated visual fields were full in both eyes. The patient’s medical history was noncontributory. Her only medication was oral contraceptives and she denied the use of tobacco.
CASE REPORT A 32-year-old woman reported a 7-month history of transient vision loss in the right eye that occurred during episodes of intense laughter or yelling. She described the vision loss as brief episodes during which the entire right visual field became dark, lasting a few seconds but occurring Department of Ophthalmology (SNR, JBH, SMC), St. Louis University School of Medicine, St. Louis, Missouri; Department of Ophthalmology and Visual Sciences (KK), Washington University in St. Louis, St. Louis, Missouri; and Ophthalmic Plastic and Cosmetic Surgery Inc. (JBH), St. Louis, Missouri. The authors report no conflicts of interest. Address correspondence to Sara N. Reggie, MD, Department of Ophthalmology, St. Louis University Eye Institute, 1755 South Grand Boulevard, St. Louis, MO 63104; E-mail: [email protected] Reggie et al: J Neuro-Ophthalmol 2017; 0: 1-2
FIG. 1. Coronal orbital computed tomography shows total opacification of the right maxillary sinus and collapse of the right orbital floor.
1
Copyright Ó North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited.
Original Contribution The patient underwent right maxillary sinus decompression via an endoscopic antrostomy. One week postoperatively, she noted improvement in headaches and no recurrence of transient vision loss with intense laughter or yelling and remained asymptomatic over the ensuing 7 months.
DISCUSSION SSS, also known as imploding antrum syndrome or chronic maxillary atelectasis, is due to an obstruction of the osteomeatal complex, which impairs maxillary sinus ventilation and causes entrapment of mucous secretions. This leads to resorption of gases in the sinus causing a negative pressure gradient and collapse of the maxillary antrum (1–4). SSS is found equally in men and women, most commonly in the fourth and fifth decades of life, and it equally affects right and left maxillary sinuses. Patients may present with painless and progressive enophthalmos, hypoglobus, and diplopia (1–8). Neuroimaging is essential to establish the correct diagnosis and usually shows neartotal opacification and contraction of the maxillary sinus, infundibular obstruction, and downward retraction of the orbital floor. In addition, there may be expansion of the orbital volume, bone loss of the orbital floor, and collapse of the uncinate process (3,9–11). Management of SSS involves surgical drainage of the maxillary sinus with or without removal of the ethmoid uncinate process for better visualization and aspiration and culture of sinus secretions. There is a continued debate whether orbital reconstruction should be performed simultaneously during sinus drainage or postponed as a second surgery (1–4,6–8,12–14). We were unable to find similar reports of transient monocular vision loss precipitated by laughing episodes in a patient with SSS. There are, however, reported cases of laughter-induced syncope in patients with vascular disease, which were attributed to a combination of poor cerebral blood flow coupled with stenosis of the external carotid artery (15–17). In addition, laughter leads to a Valsalva maneuver which, in turn, causes increased intrathoracic pressure. If the autonomic nervous system fails to increase heart rate and activate peripheral vasoconstriction, syncope may occur (16,18). Although our patient did not have any known vascular disease, we suspect that her SSS contributed to compromise of retinal blood flow (19). This may have occurred because of decreased internal carotid artery blood flow from increased intrathoracic pressure, lack of autonomic compensation, and compromise of external carotid collateral flow to the ophthalmic artery due to changes in orbital anatomy from SSS. In addition, disturbance in venous outflow has been demonstrated in patients with transient monocular visual loss (20). Although vascular studies would have been helpful in supporting our hypothesis, we are still missing a proven link as to 2
the mechanism of vision loss in our otherwise young, healthy patient. We do not believe that traction on the optic nerve from SSS caused the patient’s vision loss, as she did not have significant enophthalmos or hypoglobus to induce optic nerve stretching. Although the combination of signs and symptoms are rare, laughter-induced transient monocular vision loss may indicate the pressure of SSS and should be considered during patient evaluation.
REFERENCES 1. Soparkar CN, Patrinely JR, Cuaycong MJ, Dailey RA, Kersten RC, Rubin PA, Linberg JV, Howard GR, Donovan DT, Matoba AY, Holds JB. The silent sinus syndrome. A cause of spontaneous enophthalmos. Ophthalmology. 1994;101:772–778. 2. Pula JH, Mehta M. Silent sinus syndrome. Curr Opin Ophthalmol. 2014;25:480–484. 3. 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;114:688–694. 4. Wan MK, Francis IC, Carter PR, Griffits R, van Rooijen ML, Coroneo MT. The spectrum of presentation of silent sinus syndrome. J Neuroophthalmol. 2000;20:207–212. 5. Singman EL, Matta NS, Silbert DI. Silent sinus syndrome presenting after a roller coaster ride: a case report. Am Orthopt J. 2014;64:123–125. 6. Buono LM. The silent sinus syndrome: maxillary sinus atelectasis with enophthalmos and hypoglobus. Curr Opin Ophthalmol. 2004;15:486–489. 7. Arikan OK, Onaran Z, Muluk NB, Yilmazbas P, Yazici I. Enophthalmos due to atelectasis of the maxillary sinus: silent sinus syndrome. J Craniofac Surg. 2009;20:2156–2159. 8. Bahgat M, Bahgat Y, Bahgat A. Silent sinus syndrome. BMJ Case Rep. 2012:2012. doi: 10.1136/bcr-2012-007198. 9. Illner A, Davidson HC, Harnsberger HR, Hoffman J. The silent sinus syndrome: clinical and radiographic findings. AJR Am J Roentgenol. 2002;178:503–506. 10. Gaudino S, Di Lella GM, Piludu F, Martucci M, Schiarelli C, Africa E, Salvolini L, Colosimo C. CT and MRI diagnosis of silent sinus syndrome. Radiol Med. 2013;118:265–272. 11. Bas A, Tutar O, Samanci C, Kepek F. Silent sinus syndrome: CT and MRI findings. BMJ Case Rep. 2012;2012. doi: 10.1136/ bcr-2012-007492. 12. Brandt MG, Wright ED. The silent sinus syndrome is a form of chronic maxillary atelectasis: a systematic review of all reported cases. Am J Rhinol. 2008;22:68–73. 13. Monos T, Levy J, Lifshitz T, Puterman M. The silent sinus syndrome. Isr Med Assoc J. 2005;7:333–335. 14. Cobb AR, Murthy R, Cousin GC, El-Rasheed A, Toma A, Uddin J, Manisali M. Silent sinus syndrome. Br J Oral Maxillofac Surg. 2012;50:e81–e85. 15. Cox SV, Eisenhauer AC, Hreib K. “Seinfeld syncope”. Cathet Cardiovasc Diagn. 1997;42:242. 16. Sarzi Braga S, Manni R, Pedretti RF. Laughter-induced syncope. Lancet. 2005;366:426. 17. Shah AA, Gelber AC. Laughter-induced syncope. Am J Med. 2010;123:609–611. 18. Totah AR, Benbadis SR. Gelastic syncope mistaken for cataplexy. Sleep Med. 2002;3:77–78. 19. Macchi C, Catini C. The anatomy and clinical significance of the collateral circulation between the internal and external carotid arteries through the ophthalmic artery. Ital J Anat Embryol. 1993;1993:23–29. 20. Hsu HY, Chao AC, Chen YY, Yang FY, Chung CP, Sheng WY, Yen MY, Hu HH. Reflux of jugular and retrobulbar venous flow in transient monocular blindness. Ann Neurol. 2008;63:247–253.
Reggie et al: J Neuro-Ophthalmol 2017; 0: 1-2
Copyright Ó North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited.
