648
Shah et al
Volume 17 Number 6 / December 2013
Isolated oculomotor nerve palsy in a child caused by an internal carotid aneurysm Vedang Shah, MBBS, Mahesh Kumar, DO, DNB, Kowsalya Akkayasamy, DO, DNB, and Kunal Rana, DO, DNB, FICO
Common causes for acquired isolated oculomotor nerve palsy in children are trauma, inflammation, and neoplasia. We report a case of isolated oculomotor nerve palsy in a 13-year-old boy secondary to intracranial carotid aneurysm.
Case Report
A
13-year-old south Indian boy presented at the Aravind Eye Hospital, Madurai, with chief symptoms of headache associated with vomiting of 2 week’s duration, right eye pain of 1 week’s duration, and ptosis of the right upper eyelid and binocular double vision for the previous 2 days. There was no history of fever, ocular trauma, or head injury. General physical examination was unremarkable. On ophthalmological examination, visual acuity was 6/6 in both eyes. Ptosis in the right eye was complete; the pupil was dilated and fixed at 5 mm. Extraocular movements in the right eye were limited in all directions except abduction (Figure 1A). Diplopia and Hess charts were suggestive of oculomotor nerve palsy in the right eye. Left eye findings were normal. Dilated fundus examination was normal in both eyes. Color vision and central field assessment were normal in both eyes. Neurological examination was otherwise normal. Magnetic resonance imaging (MRI) and magnetic resonance angiography (MRA) revealed a lesion of mixed signal intensity that involved distal narrowing of the petrous, cavernous, and supraclinoid parts of the internal carotid artery. Digital subtraction angiography revealed a wide-neck saccular, lobulated right cavernous internal carotid artery aneurysm measuring 12.7 7.4 mm, with narrowing of the proximal cavernous internal carotid
Author affiliations: Aravind Eye Hospital, Madurai, Tamil Nadu, India Submitted April 8, 2013. Revision accepted August 30, 2013. Published online November 7, 2013. Correspondence: Vedang Shah, MBBS, Aravind Eye Hospital.1, Anna nagar, Madurai625020, Tamil Nadu, India (email: [email protected]). J AAPOS 2013;17:648-649. Copyright Ó 2013 by the American Association for Pediatric Ophthalmology and Strabismus. 1091-8531/$36.00 http://dx.doi.org/10.1016/j.jaapos.2013.08.008
artery (Figure 2A). Good cross circulation was noted in the anterior communicating artery on right internal carotid artery compression. Based on the clinical history and imaging, the boy was diagnosed with pupilinvolving, oculomotor nerve palsy due to internal carotid artery aneurysm. The patient was referred to a neurosurgeon for further management. He underwent balloon-assisted parent artery occlusion of the right cavernous aneurysm using coils and glue. At 1 month’s follow-up, recovery of the right oculomotor nerve palsy was complete, and ptosis and ophthalmoplegia had resolved completely, with minimally sluggish pupillary reaction. No signs of aberrant regeneration were observed (Figures 2B and 1B).
Discussion Isolated, acquired oculomotor nerve palsy secondary to intracranial aneurysms are extremely rare in children.1 Fox2 reported a similar case in a 6-year-old with oculomotor nerve palsy due to a giant cavernous carotid aneurysm. Posterior communicating aneurysms have been reported in children 7-11 years of age by Wolin and Saunders,3 Liu and colleagues,1 and Branley and colleagues.4 Tamhankar and colleagues5 reported an 8-month-old girl with internal carotid artery aneurysm that was left untreated due to its fusiform nature. The present case was a saccular aneurysm, which is the least common form of cerebral aneurysm. The cavernous location of the aneurysm was also very uncommon and accounts for only 2% of all intracranial aneurysms. These aneurysms most commonly involve the abducens nerve, as the nerve traverses just lateral to the internal carotid artery in the cavernous sinus. Management of internal carotid artery aneurysms require a multidisciplinary approach involving ophthalmologists, radiologists, and neurosurgeons. Treatment depends on the symptoms, size, and location of the aneurysm. Linskey and colleagues6 reported improvement in 25%-40% of cases with conservative treatment. The decision to intervene typically centers around the persistence of pain, subarachnoid hemorrhage, progressive or unresolving ophthalmoplegia, and visual loss. Our patient recovered completely following successful
Journal of AAPOS
Volume 17 Number 6 / December 2013
Shah et al
649
FIG 1. Clinical photographs of patient at presentation (A) and after surgery (B).
closure with endovascular coiling. Fulkerson and colleagues7 have also demonstrated successful closure of traumatic skull base aneurysm with endovascular coiling in 3 adolescent patients. Mavilio and colleauges8 have also shown complete recovery after endovascular packing of an aneurysm of the posterior communicating artery of the internal carotid artery in adults.
References
FIG 2. Preoperative digital subtraction angiography showing the wide-necked, saccular aneurysm before surgery (A) and carotid artery occlusion with coils and glue postoperatively (B).
Journal of AAPOS
1. Liu GT, Mehkri IA, Awner S, Olitsky SE, et al. Double vision in a child. Surv Ophthalmology 1999;44:45-52. 2. Fox AJ. Angiography for third nerve palsy in children. J Clin Neuroophthalmol 1989;9:37-8. 3. Wolin MJ, Saunders RA. Aneurysmal oculomotor nerve palsy in an 11-year-old boy. J Clin Neuro-ophthalmol 1992;12:178-80. 4. Branley MG, Wright KW, Borchert MS. Third nerve palsy due to cerebral artery aneurysm in a child. Aust N Z J Ophthalmol 1992;20: 137-40. 5. Tamhankar MA, Liu GT, Young TL, Sutton LN, Hurst RW. Acquired, isolated third nerve palsies in infants with cerebrovascular malformations. Am J Ophthalmology 2004;138:484-6. 6. Linskey ME, Sekhar LN, Hirsch WL Jr, Yonas H, Horton JA. Aneurysms of the intracavernous carotid artery: natural history and indications for treatment. Neurosurgery 1990;26:933-7; discussion 937-8. 7. Fulkerson DH, Voorhies JM, McCanna SP, et al. Endovascular treatment and radiographic follow-up of proximal traumatic intracranial aneurysms in adolescents: case series and review of the literature. Childs Nerve Syst 2010;26:613-20. 8. Mavilio N, Pisani R, Rivano C, Testa V, Spaziante R, Rosa M. Recovery of third nerve palsy after endovascular packing of internal carotid-posterior communicating artery aneurysms. Interv Neuroradiol 2000;6:203-9.
Shah et al
Volume 17 Number 6 / December 2013
Isolated oculomotor nerve palsy in a child caused by an internal carotid aneurysm Vedang Shah, MBBS, Mahesh Kumar, DO, DNB, Kowsalya Akkayasamy, DO, DNB, and Kunal Rana, DO, DNB, FICO
Common causes for acquired isolated oculomotor nerve palsy in children are trauma, inflammation, and neoplasia. We report a case of isolated oculomotor nerve palsy in a 13-year-old boy secondary to intracranial carotid aneurysm.
Case Report
A
13-year-old south Indian boy presented at the Aravind Eye Hospital, Madurai, with chief symptoms of headache associated with vomiting of 2 week’s duration, right eye pain of 1 week’s duration, and ptosis of the right upper eyelid and binocular double vision for the previous 2 days. There was no history of fever, ocular trauma, or head injury. General physical examination was unremarkable. On ophthalmological examination, visual acuity was 6/6 in both eyes. Ptosis in the right eye was complete; the pupil was dilated and fixed at 5 mm. Extraocular movements in the right eye were limited in all directions except abduction (Figure 1A). Diplopia and Hess charts were suggestive of oculomotor nerve palsy in the right eye. Left eye findings were normal. Dilated fundus examination was normal in both eyes. Color vision and central field assessment were normal in both eyes. Neurological examination was otherwise normal. Magnetic resonance imaging (MRI) and magnetic resonance angiography (MRA) revealed a lesion of mixed signal intensity that involved distal narrowing of the petrous, cavernous, and supraclinoid parts of the internal carotid artery. Digital subtraction angiography revealed a wide-neck saccular, lobulated right cavernous internal carotid artery aneurysm measuring 12.7 7.4 mm, with narrowing of the proximal cavernous internal carotid
Author affiliations: Aravind Eye Hospital, Madurai, Tamil Nadu, India Submitted April 8, 2013. Revision accepted August 30, 2013. Published online November 7, 2013. Correspondence: Vedang Shah, MBBS, Aravind Eye Hospital.1, Anna nagar, Madurai625020, Tamil Nadu, India (email: [email protected]). J AAPOS 2013;17:648-649. Copyright Ó 2013 by the American Association for Pediatric Ophthalmology and Strabismus. 1091-8531/$36.00 http://dx.doi.org/10.1016/j.jaapos.2013.08.008
artery (Figure 2A). Good cross circulation was noted in the anterior communicating artery on right internal carotid artery compression. Based on the clinical history and imaging, the boy was diagnosed with pupilinvolving, oculomotor nerve palsy due to internal carotid artery aneurysm. The patient was referred to a neurosurgeon for further management. He underwent balloon-assisted parent artery occlusion of the right cavernous aneurysm using coils and glue. At 1 month’s follow-up, recovery of the right oculomotor nerve palsy was complete, and ptosis and ophthalmoplegia had resolved completely, with minimally sluggish pupillary reaction. No signs of aberrant regeneration were observed (Figures 2B and 1B).
Discussion Isolated, acquired oculomotor nerve palsy secondary to intracranial aneurysms are extremely rare in children.1 Fox2 reported a similar case in a 6-year-old with oculomotor nerve palsy due to a giant cavernous carotid aneurysm. Posterior communicating aneurysms have been reported in children 7-11 years of age by Wolin and Saunders,3 Liu and colleagues,1 and Branley and colleagues.4 Tamhankar and colleagues5 reported an 8-month-old girl with internal carotid artery aneurysm that was left untreated due to its fusiform nature. The present case was a saccular aneurysm, which is the least common form of cerebral aneurysm. The cavernous location of the aneurysm was also very uncommon and accounts for only 2% of all intracranial aneurysms. These aneurysms most commonly involve the abducens nerve, as the nerve traverses just lateral to the internal carotid artery in the cavernous sinus. Management of internal carotid artery aneurysms require a multidisciplinary approach involving ophthalmologists, radiologists, and neurosurgeons. Treatment depends on the symptoms, size, and location of the aneurysm. Linskey and colleagues6 reported improvement in 25%-40% of cases with conservative treatment. The decision to intervene typically centers around the persistence of pain, subarachnoid hemorrhage, progressive or unresolving ophthalmoplegia, and visual loss. Our patient recovered completely following successful
Journal of AAPOS
Volume 17 Number 6 / December 2013
Shah et al
649
FIG 1. Clinical photographs of patient at presentation (A) and after surgery (B).
closure with endovascular coiling. Fulkerson and colleagues7 have also demonstrated successful closure of traumatic skull base aneurysm with endovascular coiling in 3 adolescent patients. Mavilio and colleauges8 have also shown complete recovery after endovascular packing of an aneurysm of the posterior communicating artery of the internal carotid artery in adults.
References
FIG 2. Preoperative digital subtraction angiography showing the wide-necked, saccular aneurysm before surgery (A) and carotid artery occlusion with coils and glue postoperatively (B).
Journal of AAPOS
1. Liu GT, Mehkri IA, Awner S, Olitsky SE, et al. Double vision in a child. Surv Ophthalmology 1999;44:45-52. 2. Fox AJ. Angiography for third nerve palsy in children. J Clin Neuroophthalmol 1989;9:37-8. 3. Wolin MJ, Saunders RA. Aneurysmal oculomotor nerve palsy in an 11-year-old boy. J Clin Neuro-ophthalmol 1992;12:178-80. 4. Branley MG, Wright KW, Borchert MS. Third nerve palsy due to cerebral artery aneurysm in a child. Aust N Z J Ophthalmol 1992;20: 137-40. 5. Tamhankar MA, Liu GT, Young TL, Sutton LN, Hurst RW. Acquired, isolated third nerve palsies in infants with cerebrovascular malformations. Am J Ophthalmology 2004;138:484-6. 6. Linskey ME, Sekhar LN, Hirsch WL Jr, Yonas H, Horton JA. Aneurysms of the intracavernous carotid artery: natural history and indications for treatment. Neurosurgery 1990;26:933-7; discussion 937-8. 7. Fulkerson DH, Voorhies JM, McCanna SP, et al. Endovascular treatment and radiographic follow-up of proximal traumatic intracranial aneurysms in adolescents: case series and review of the literature. Childs Nerve Syst 2010;26:613-20. 8. Mavilio N, Pisani R, Rivano C, Testa V, Spaziante R, Rosa M. Recovery of third nerve palsy after endovascular packing of internal carotid-posterior communicating artery aneurysms. Interv Neuroradiol 2000;6:203-9.
