678
Case Reports / Journal of Clinical Neuroscience 21 (2014) 678–680
Parkinson’s disease. Nonetheless, the position of the electrodes in relation to the MFB, MMT and DLF could help explain the effects on diaphoresis. 4. Conclusion We stipulate that activation of the nearby hypothalamospinal tract is the cause of observed reversible deep brain stimulator DBS-induced diaphoresis. Conflict of interest/disclosure The authors declare that they have no financial or other conflicts of interest in relation to this research and its publication.
References [1] Weaver FM, Follett KF, Stern M, et al. Bilateral deep brain stimulation vs best medical therapy for patients with advanced Parkinson disease: a randomized controlled trial. JAMA 2009;301:63–73. [2] Swinn L, Schrag A, Viswanathan R, et al. Sweating dysfunction in Parkinson’s disease. Mov Disord 2003;18:1459–63. [3] Halim A, Baumgartner L, Binder DK. Effect of deep brain stimulation on autonomic dysfunction in patients with Parkinson’s disease. J Clin Neurosci 2011;18:804–6. [4] Trachani E, Constantoyannis C, Sirrou V, et al. Effects of subthalamic nucleus deep brain stimulation on sweating function in Parkinson’s disease. Clin Neurol Neurosurg 2010;112:213–7. [5] Wolz M, Hauschild J, Fauser M, et al. Immediate effects of deep brain stimulation of the subthalamic nucleus on nonmotor symptoms in Parkinson’s disease. Parkinsonism Relat Disord 2012;18:994–7.
http://dx.doi.org/10.1016/j.jocn.2013.05.016
Panophthalmoplegia and vision loss after cosmetic nasal dorsum injection Shi Nae Kim, Dae Sup Byun, Jung Hyun Park, Sang Won Han ⇑, Jong Sam Baik, Jeong Yeon Kim, Jae Hyeon Park Department of Neurology, Sanggye Paik Hospital, Inje University College of Medicine, 761-1 Sanggye 7, Nowon-gu, Seoul 139-707, South Korea
a r t i c l e
i n f o
Article history: Received 28 August 2012 Accepted 26 May 2013
Keywords: Blindness Hyaluronic acid Injections Opthalmoplegia Stroke
a b s t r a c t We report a case of unilateral blindness and panophthalmoplegia after hyaluronic acid injection into the dorsum of the nose in a healthy young woman. Microspheres of hyaluronic acid are popular fillers for facial rejuvenation. While ocular side effects from injections in the nose and face have been reported following turbinate injection, rhinoplasty and infraorbital nerve block, ocular side effects from injection into the dorsum of the nose are extremely rare. We presume that the symptoms were due to obstruction of the branches of the ophthalmic artery. Under high injection pressure, the microspheres travelled to the ophthalmic artery and were propelled by the blood flow to the central retinal artery and the anterior and posterior long ciliary arteries, leading to her symptoms. Alternatively, there are several arterio-venous anastomotic channels in the nasal mucosa that aid heat exchange. These may have been the conduit for reflux of the filler into the arterial side of the regional circulation. Physicians must remain aware of serious complications during cosmetic injections to this region. Ó 2013 Elsevier Ltd. All rights reserved.
1. Introduction Microspheres of hyaluronic acid are popular fillers for facial rejuvenation. While ocular side effects from injections in the nose and face have been reported following turbinate injection, rhinoplasty and infraorbital nerve block, ocular side effects from injection into the dorsum of nose are extremely rare [1–4]. We report a case of unilateral blindness and panophthalmoplegia after hyaluronic acid injection into the dorsum of the nose in a healthy young woman. 2. Case report A young woman presented to the emergency department of the hospital with ptosis, ophthalmoplegia and vision loss in her right eye. Six hours prior to her arrival at the hospital, she had received a hyaluronic acid-containing filler injection into the dorsum of her nose for dorsal augmentation. Immediately after the injection, she
⇑ Corresponding author. Tel.: +82 2950 1090; fax: +82 2950 1955. E-mail address: [email protected] (S.W. Han).
felt sudden, severe periocular pain and had complete vision loss in her right eye. The procedure was performed by a general surgeon at his hospital under local anesthesia. During neurologic examination, ptosis and panophthalmoplegia were observed in her right eye. The direct and indirect pupil light reflex was absent. Visual acuities were 6/6 in the left eye, and there was no perception of light in the right. A fundus examination of the right eye revealed a pale optic disc with a blurred margin and central retinal artery occlusion (Fig. 1). The left eye was normal. MRI of the brain revealed multifocal punctuate high signal intensity lesions in both frontal areas on diffusion-weighted imaging (Fig. 2). The brain magnetic resonance angiography was normal. She was taking no medication at the time of the procedure and had no history of coagulopathy or other systemic disease. Systemic investigations, including electrocardiography, chest radiograph and blood tests for coagulopathy were normal. The transthoracic and transesophageal echocardiographic studies were normal. She was treated with high doses intravenous corticosteroids. Five days after symptom onset, lateral gaze in the right eye was partially improved. There was no evidence of regional tissue necrosis. At the 6 month follow-up visit, visual acuity and ophthalmoplegia in the right eye had not improved.
Case Reports / Journal of Clinical Neuroscience 21 (2014) 678–680
Fig. 1. Fundus examination of the right eye revealing a pale optic disc with a blurred margin and central retinal artery occlusion.
3. Discussion The internal carotid artery (ICA) sends branches to the forehead and nose. The ophthalmic artery, the first branch of the ICA, is the major source of blood supply to the orbit. The branches of the ophthalmic artery are divided into an orbital group, which distributes blood to the orbit and surrounding parts, and an ocular group, which distributes blood to the muscles and bulb of the eye. The orbital group consists of the lacrimal artery, supraorbital artery, posterior ethmoidal artery, anterior ethmoidal artery, internal palpebral artery, frontal artery and nasal artery. The ocular group consists of the long ciliary artery, short ciliary artery, anterior ciliary artery, central retinal artery and muscular artery [5]. These multiple branches projecting to the nose and forehead were most likely
679
responsible for the ocular accident experienced by the patient following cosmetic injections into this region. Several cases of visual loss after cosmetic filler injection in the glabellar area or nasolabial fold have been reported [1–4]. However our patient had unilateral blindness and panophthalmoplegia after hyaluronic acid injection into the dorsum of her nose. We presume that her symptoms were due to obstruction of the branches of the ophthalmic artery, including the supratrochlear or dorsal nasal artery. Under high injection pressure, the microspheres travelled to the ophthalmic artery and were propelled by the blood flow to the central retinal artery and the anterior and posterior long ciliary arteries, leading to the symptoms [1–4]. Alternatively, there are several arterio-venous anastomotic channels in the nasal mucosa that aid heat exchange. These may have been the conduit for reflux of the filler into the arterial side of the regional circulation. In this patient, brain MRI showed multifocal punctuated high signal intensity lesions in both frontal areas. The reflux of the filler material under high injecting force may go to the ICA through the anastomotic braches to the ophthalmic artery or any cerebral artery branching from the external carotid artery and cross at the circle of Willis. This may have caused the iatrogenic embolization in the intracranial circulation observed in our patient. Thus, physicians must remain aware of serious complications of cosmetic injections into the forehead and nose.
Conflict of interest/disclosure The authors declare that they have no financial or other conflicts of interest in relation to this research and its publication.
References [1] Silva MT, Curi AL. Blindness and total ophthalmoplegia after aesthetic polymethylmethacrylate injection: case report. Arq Neuropsiquiatr 2004;62:873–4.
Fig. 2. Axial diffusion-weighted brain MRI showing multifocal punctuate high signal intensity lesions in both frontal areas, suggesting emboli in the intracranial circulation.
680
Case Reports / Journal of Clinical Neuroscience 21 (2014) 680–683
[2] Matsuo T, Fujiwara H, Gobara H, et al. Central retinal and posterior ciliary artery occlusion after intralesional injection of sclerosant to glabellar subcutaneous hemangioma. Cardiovasc Intervent Radiol 2009;32:341–6. [3] Egido JA, Arroyo R, Marcos A, et al. Middle cerebral artery embolism and unilateral visual loss after autologous fat injection into the glabellar area. Stroke 1993;24:615–6.
[4] Feinendegen DL, Baumgartner RW, Schroth G, et al. Middle cerebral artery occlusion AND ocular fat embolism after autologous fat injection in the face. J Neurol 1998;245:53–4. [5] Hayreh SS. The Ophthalmic Artery: III. Branches. Br J Ophthalmol 1962;46:212–47.
http://dx.doi.org/10.1016/j.jocn.2013.05.018
Neurosurgical management for complicated catastrophic antiphospholipid syndrome Doniel Drazin a,⇑, H. Westley Phillips a, Ali Shirzadi a, Noam Drazin b, Wouter Schievink a a b
Department of Neurosurgery, Cedars-Sinai Medical Center, 8631 West Third Street, Suite 800E, Los Angeles, CA 90048, USA Department of Medicine, Division of Hematology/Oncology, Cedars-Sinai Medical Center, Los Angeles, CA, USA
a r t i c l e
i n f o
Article history: Received 16 February 2013 Accepted 15 May 2013
Keywords: Antiphospholipid syndrome Catastrophic illness Cerebellar subdural hemorrhage Intracerebral hemorrhage Laminectomy Suboccipital craniotomy
a b s t r a c t Antiphospholipid syndrome (APS) is an autoimmune condition involving arterial and venous thrombosis. An unusual APS variant, catastrophic antiphospholipid syndrome (CAPS), includes rapid multi-organ failure from widespread small vessel thrombosis. Central nervous system complications arise in one-third of CAPS patients. In rare cases, CAPS co-manifests with cerebellar hemorrhage presenting a neurosurgical emergency. We present a 65-year-old woman with CAPS-related cerebellar hematoma, co-morbid idiopathic thrombocytopenic purpura, deep vein thrombosis and altered mental status, with treatment complicated by thrombocytopenia. The patient suddenly deteriorated, secondary to a cerebellar subdural hematoma, and underwent decompression and excision of the hematoma. After recovery in the intensive care unit, she developed a new spontaneous epidural hematoma requiring additional surgery. Management of these patients is hematologically complex and often requires a multi-disciplinary team of physicians. This patient provides an important learning point for clinicians – consider CAPS when hemorrhage and thrombosis are present. Ó 2013 Elsevier Ltd. All rights reserved.
1. Introduction Antiphospholipid syndrome (APS) is an autoimmune disease with vasculature thromboses and pregnancy complications wherein autoantibodies target membrane phospholipids and phospholipid binding proteins [1]. Catastrophic antiphospholipid syndrome (CAPS), a rare APS variant, is characterized by multiple small vessel thrombosis, potentially leading to multi-organ failure [2]. Cerebral involvement is present in one-third of CAPS patients [3]. Cerebellar hematomas (CH) are associated with 5–13% of intracranial hemorrhage patients and high mortality rates [4–6]. Neurosurgical intervention is a viable treatment option [5,6]. We present to our knowledge the first reported patient with CAPS and spontaneous acute CH requiring urgent neurosurgical intervention. The high mortality rate associated with CAPS makes its documentation in the literature important.
2. Illustrative patient 2.1. History A 65-year-old woman presented with urosepsis and altered mental status. Medical history included deep vein thrombosis since age 17 and pre-eclampsia during two pregnancies. She was taking enoxaparin, following 10 years of warfarin. She was recently diagnosed with idiopathic thrombocytopenic purpura and APS; she had high anticardiolipin immunoglobulin (Ig) G/IgM levels (141/83 IgG phospholipid units/IgM phospholipid units, normal
Case Reports / Journal of Clinical Neuroscience 21 (2014) 678–680
Parkinson’s disease. Nonetheless, the position of the electrodes in relation to the MFB, MMT and DLF could help explain the effects on diaphoresis. 4. Conclusion We stipulate that activation of the nearby hypothalamospinal tract is the cause of observed reversible deep brain stimulator DBS-induced diaphoresis. Conflict of interest/disclosure The authors declare that they have no financial or other conflicts of interest in relation to this research and its publication.
References [1] Weaver FM, Follett KF, Stern M, et al. Bilateral deep brain stimulation vs best medical therapy for patients with advanced Parkinson disease: a randomized controlled trial. JAMA 2009;301:63–73. [2] Swinn L, Schrag A, Viswanathan R, et al. Sweating dysfunction in Parkinson’s disease. Mov Disord 2003;18:1459–63. [3] Halim A, Baumgartner L, Binder DK. Effect of deep brain stimulation on autonomic dysfunction in patients with Parkinson’s disease. J Clin Neurosci 2011;18:804–6. [4] Trachani E, Constantoyannis C, Sirrou V, et al. Effects of subthalamic nucleus deep brain stimulation on sweating function in Parkinson’s disease. Clin Neurol Neurosurg 2010;112:213–7. [5] Wolz M, Hauschild J, Fauser M, et al. Immediate effects of deep brain stimulation of the subthalamic nucleus on nonmotor symptoms in Parkinson’s disease. Parkinsonism Relat Disord 2012;18:994–7.
http://dx.doi.org/10.1016/j.jocn.2013.05.016
Panophthalmoplegia and vision loss after cosmetic nasal dorsum injection Shi Nae Kim, Dae Sup Byun, Jung Hyun Park, Sang Won Han ⇑, Jong Sam Baik, Jeong Yeon Kim, Jae Hyeon Park Department of Neurology, Sanggye Paik Hospital, Inje University College of Medicine, 761-1 Sanggye 7, Nowon-gu, Seoul 139-707, South Korea
a r t i c l e
i n f o
Article history: Received 28 August 2012 Accepted 26 May 2013
Keywords: Blindness Hyaluronic acid Injections Opthalmoplegia Stroke
a b s t r a c t We report a case of unilateral blindness and panophthalmoplegia after hyaluronic acid injection into the dorsum of the nose in a healthy young woman. Microspheres of hyaluronic acid are popular fillers for facial rejuvenation. While ocular side effects from injections in the nose and face have been reported following turbinate injection, rhinoplasty and infraorbital nerve block, ocular side effects from injection into the dorsum of the nose are extremely rare. We presume that the symptoms were due to obstruction of the branches of the ophthalmic artery. Under high injection pressure, the microspheres travelled to the ophthalmic artery and were propelled by the blood flow to the central retinal artery and the anterior and posterior long ciliary arteries, leading to her symptoms. Alternatively, there are several arterio-venous anastomotic channels in the nasal mucosa that aid heat exchange. These may have been the conduit for reflux of the filler into the arterial side of the regional circulation. Physicians must remain aware of serious complications during cosmetic injections to this region. Ó 2013 Elsevier Ltd. All rights reserved.
1. Introduction Microspheres of hyaluronic acid are popular fillers for facial rejuvenation. While ocular side effects from injections in the nose and face have been reported following turbinate injection, rhinoplasty and infraorbital nerve block, ocular side effects from injection into the dorsum of nose are extremely rare [1–4]. We report a case of unilateral blindness and panophthalmoplegia after hyaluronic acid injection into the dorsum of the nose in a healthy young woman. 2. Case report A young woman presented to the emergency department of the hospital with ptosis, ophthalmoplegia and vision loss in her right eye. Six hours prior to her arrival at the hospital, she had received a hyaluronic acid-containing filler injection into the dorsum of her nose for dorsal augmentation. Immediately after the injection, she
⇑ Corresponding author. Tel.: +82 2950 1090; fax: +82 2950 1955. E-mail address: [email protected] (S.W. Han).
felt sudden, severe periocular pain and had complete vision loss in her right eye. The procedure was performed by a general surgeon at his hospital under local anesthesia. During neurologic examination, ptosis and panophthalmoplegia were observed in her right eye. The direct and indirect pupil light reflex was absent. Visual acuities were 6/6 in the left eye, and there was no perception of light in the right. A fundus examination of the right eye revealed a pale optic disc with a blurred margin and central retinal artery occlusion (Fig. 1). The left eye was normal. MRI of the brain revealed multifocal punctuate high signal intensity lesions in both frontal areas on diffusion-weighted imaging (Fig. 2). The brain magnetic resonance angiography was normal. She was taking no medication at the time of the procedure and had no history of coagulopathy or other systemic disease. Systemic investigations, including electrocardiography, chest radiograph and blood tests for coagulopathy were normal. The transthoracic and transesophageal echocardiographic studies were normal. She was treated with high doses intravenous corticosteroids. Five days after symptom onset, lateral gaze in the right eye was partially improved. There was no evidence of regional tissue necrosis. At the 6 month follow-up visit, visual acuity and ophthalmoplegia in the right eye had not improved.
Case Reports / Journal of Clinical Neuroscience 21 (2014) 678–680
Fig. 1. Fundus examination of the right eye revealing a pale optic disc with a blurred margin and central retinal artery occlusion.
3. Discussion The internal carotid artery (ICA) sends branches to the forehead and nose. The ophthalmic artery, the first branch of the ICA, is the major source of blood supply to the orbit. The branches of the ophthalmic artery are divided into an orbital group, which distributes blood to the orbit and surrounding parts, and an ocular group, which distributes blood to the muscles and bulb of the eye. The orbital group consists of the lacrimal artery, supraorbital artery, posterior ethmoidal artery, anterior ethmoidal artery, internal palpebral artery, frontal artery and nasal artery. The ocular group consists of the long ciliary artery, short ciliary artery, anterior ciliary artery, central retinal artery and muscular artery [5]. These multiple branches projecting to the nose and forehead were most likely
679
responsible for the ocular accident experienced by the patient following cosmetic injections into this region. Several cases of visual loss after cosmetic filler injection in the glabellar area or nasolabial fold have been reported [1–4]. However our patient had unilateral blindness and panophthalmoplegia after hyaluronic acid injection into the dorsum of her nose. We presume that her symptoms were due to obstruction of the branches of the ophthalmic artery, including the supratrochlear or dorsal nasal artery. Under high injection pressure, the microspheres travelled to the ophthalmic artery and were propelled by the blood flow to the central retinal artery and the anterior and posterior long ciliary arteries, leading to the symptoms [1–4]. Alternatively, there are several arterio-venous anastomotic channels in the nasal mucosa that aid heat exchange. These may have been the conduit for reflux of the filler into the arterial side of the regional circulation. In this patient, brain MRI showed multifocal punctuated high signal intensity lesions in both frontal areas. The reflux of the filler material under high injecting force may go to the ICA through the anastomotic braches to the ophthalmic artery or any cerebral artery branching from the external carotid artery and cross at the circle of Willis. This may have caused the iatrogenic embolization in the intracranial circulation observed in our patient. Thus, physicians must remain aware of serious complications of cosmetic injections into the forehead and nose.
Conflict of interest/disclosure The authors declare that they have no financial or other conflicts of interest in relation to this research and its publication.
References [1] Silva MT, Curi AL. Blindness and total ophthalmoplegia after aesthetic polymethylmethacrylate injection: case report. Arq Neuropsiquiatr 2004;62:873–4.
Fig. 2. Axial diffusion-weighted brain MRI showing multifocal punctuate high signal intensity lesions in both frontal areas, suggesting emboli in the intracranial circulation.
680
Case Reports / Journal of Clinical Neuroscience 21 (2014) 680–683
[2] Matsuo T, Fujiwara H, Gobara H, et al. Central retinal and posterior ciliary artery occlusion after intralesional injection of sclerosant to glabellar subcutaneous hemangioma. Cardiovasc Intervent Radiol 2009;32:341–6. [3] Egido JA, Arroyo R, Marcos A, et al. Middle cerebral artery embolism and unilateral visual loss after autologous fat injection into the glabellar area. Stroke 1993;24:615–6.
[4] Feinendegen DL, Baumgartner RW, Schroth G, et al. Middle cerebral artery occlusion AND ocular fat embolism after autologous fat injection in the face. J Neurol 1998;245:53–4. [5] Hayreh SS. The Ophthalmic Artery: III. Branches. Br J Ophthalmol 1962;46:212–47.
http://dx.doi.org/10.1016/j.jocn.2013.05.018
Neurosurgical management for complicated catastrophic antiphospholipid syndrome Doniel Drazin a,⇑, H. Westley Phillips a, Ali Shirzadi a, Noam Drazin b, Wouter Schievink a a b
Department of Neurosurgery, Cedars-Sinai Medical Center, 8631 West Third Street, Suite 800E, Los Angeles, CA 90048, USA Department of Medicine, Division of Hematology/Oncology, Cedars-Sinai Medical Center, Los Angeles, CA, USA
a r t i c l e
i n f o
Article history: Received 16 February 2013 Accepted 15 May 2013
Keywords: Antiphospholipid syndrome Catastrophic illness Cerebellar subdural hemorrhage Intracerebral hemorrhage Laminectomy Suboccipital craniotomy
a b s t r a c t Antiphospholipid syndrome (APS) is an autoimmune condition involving arterial and venous thrombosis. An unusual APS variant, catastrophic antiphospholipid syndrome (CAPS), includes rapid multi-organ failure from widespread small vessel thrombosis. Central nervous system complications arise in one-third of CAPS patients. In rare cases, CAPS co-manifests with cerebellar hemorrhage presenting a neurosurgical emergency. We present a 65-year-old woman with CAPS-related cerebellar hematoma, co-morbid idiopathic thrombocytopenic purpura, deep vein thrombosis and altered mental status, with treatment complicated by thrombocytopenia. The patient suddenly deteriorated, secondary to a cerebellar subdural hematoma, and underwent decompression and excision of the hematoma. After recovery in the intensive care unit, she developed a new spontaneous epidural hematoma requiring additional surgery. Management of these patients is hematologically complex and often requires a multi-disciplinary team of physicians. This patient provides an important learning point for clinicians – consider CAPS when hemorrhage and thrombosis are present. Ó 2013 Elsevier Ltd. All rights reserved.
1. Introduction Antiphospholipid syndrome (APS) is an autoimmune disease with vasculature thromboses and pregnancy complications wherein autoantibodies target membrane phospholipids and phospholipid binding proteins [1]. Catastrophic antiphospholipid syndrome (CAPS), a rare APS variant, is characterized by multiple small vessel thrombosis, potentially leading to multi-organ failure [2]. Cerebral involvement is present in one-third of CAPS patients [3]. Cerebellar hematomas (CH) are associated with 5–13% of intracranial hemorrhage patients and high mortality rates [4–6]. Neurosurgical intervention is a viable treatment option [5,6]. We present to our knowledge the first reported patient with CAPS and spontaneous acute CH requiring urgent neurosurgical intervention. The high mortality rate associated with CAPS makes its documentation in the literature important.
2. Illustrative patient 2.1. History A 65-year-old woman presented with urosepsis and altered mental status. Medical history included deep vein thrombosis since age 17 and pre-eclampsia during two pregnancies. She was taking enoxaparin, following 10 years of warfarin. She was recently diagnosed with idiopathic thrombocytopenic purpura and APS; she had high anticardiolipin immunoglobulin (Ig) G/IgM levels (141/83 IgG phospholipid units/IgM phospholipid units, normal
