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Hypersexuality after bilateral deep brain stimulation of the subthalamic nucleus for Parkinson’s disease Sir, Parkinson’s disease (PD) is a degenerative disorder of the central nervous system.[1] The cardinal symptoms are bradykinesia, rigidity, postural instability, and resting tremor. However, cognitive, mood, and behavioral problems may also arise either as a part of course of the disease or triggered by dopamine replacement therapy.[2] While deep brain stimulation of the subthalamic nucleus (DBS-STN) might be a therapeutic option for either PD or medication-related behavior, it has been reported that DBS-STN might also induce pathologic hypersexuality.[3-6] Here, we report a case of PD patient, who developed pathologic hypersexuality commencing after DBS-STN, despite remarkable motor improvement. A 58-year-old man suffering from PD was admitted for bilateral STN-DBS placement. After magnetic resonance imaging, a stereotactic frame (Integra CRW, New Jersey, USA) was placed onto the patient’s head on the day of the surgery and examined the brain with computed tomography. Later both images were auto-fused to target the anterior lateral side of left STN and right STN through image fusion program (Atlas Integra Software, New Jersey, USA). Bilateral micro-recording test (Alfa Omega Micro Recording System, Nazareth, Israel) was done. DBS electrode leads were placed at predefined targets (St. Jude Medical 6149 40 cm lead, Minnesota, USA) bilaterally to STN anteriolateral sides [Figure 1 left and right]. Then impulse generator (St. Jude Medical Libra XP 6644, Minnesota, USA) was implanted and connected to DBS electrode leads. The day after the surgery, the implanted impulse generator was turned on. The stimulation parameters are shown in Table 1. Preoperative drug regime was resumed. Postoperatively, there was remarkable improvement in his symptoms and according to the Unified Parkinson’s Disease Rating Scale III (UPDRS III), motor and total outcome. Both pre- and post-operative examinations are mentioned in Table 1. At the first follow-up visit, the patient’s wife mentioned that while he did not have any sexual gratification more than 2 years, after DBS-STN, every day he would insist on it and he would become very aggressive if denied. Although, he approved to be hypersexual and sometimes have uncontrollable behavior, he did not allow any intervention. Pathologic hypersexuality has been defined as ‘the need for sexual behavior that consumes so much money, time, concentration, and energy that the patient describes Neurology India | Mar-Apr 2014 | Vol 62 | Issue 2
Figure 1: Postoperative control cranial computed tomography
Table 1: UPDRS III in total and motor outcomes according to preoperative and postoperative of the bilateral STN-DBS and stimulation parameters of the patient with two channels and eight contacts constant current stimulator Preoperative STN-DBS Postoperative STN-DBS
Channel (left) Channel (right)
Motor
Total
72 34
114 63
Active contact
Amplitude (mA)
Width (uS)
Frequency (Hz)
3 2
3 2, 5
78 65
142 130
STN-DBS - Subthalamic nucleus deep brain stimulation, UPDRS III - Unified Parkinson’s disease rating scale III
himself as out of control; intrusive unwanted paraphiliac thoughts prevent concentration on other life demands and are the source of anxiety, and orgasm does not produce satiety in the way it typically does for age mates’.[7] Overall estimated prevalence of pathologic hypersexuality in PD varied between 2% and 10%, which can be induced even following DBS-STN.[3] In the literature, five cases of new onset pathologic hypersexuality after DBS-STN with no previous history of any impulse control disorder have been documented.[3-5] The role of the STN in the basal ganglia motor circuits is having a central position within the basal ganglia-thalamocortical associative and limbic circuits. It is functionally a potent regulator of these pathways.[8] Various pathophysiological mechanisms have been proposed to explain the impairment of cognition after DBS-STN. The electrodes are relatively large in comparison with the small size of the STN and it may be difficult to selectively influence the motor part of the STN without affecting domains within the STN or in neighboring structures that are associated with motivational, emotional, and cognitive functions.[9] Modulation of STN hyperactivity with DBS may improve impaired proactive inhibition in PD patients, but may induce impairment of reactive inhibition or cognitive 233
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control that leads to premature and impulsive responses, such as hypersexuality.[3,4,10] In our case, while the patient had remarkable motor improvement with significant UPDRS motor score increase, he displayed clinical features of pathologic hypersexuality on follow-up examinations. In conclusion, DBS-STN may induce hypersexuality in rare occasions. Further careful evaluation is required to elucidate the mechanisms through which DBS-STN might affect sexual function.
Akın Akakın, Baran Yılmaz, Kamran Urgun, Murat Şakir Ekşi1, Nazire Afşar2, Türker Kılıç Departments of Neurosurgery, and 2Neurology, Bahcesehir Unıversity, Istanbul, Turkey, 1Department of Orthopedics, University of California, Spine Center, San Francisco, California, USA E-mail: [email protected]
References Li Q, Qian ZM, Arbuthnott GW, Ke Y, Yung WH. Cortical effects of deep brain stimulation: Implications for pathogenesis and treatment of Parkinson disease. JAMA Neurol 2014;71:100-3. 2. Merims D, Giladi N. Dopamine dysregulation syndrome, addiction and behavioral changes in Parkinson’s disease. Parkinsonism Relat Disord 2008;14:273-80. 3. Broen M, Duits A, Visser-Vandewalle V, Temel Y, Winogrodzka A. Impulse control and related disorders in Parkinson’s disease patients treated with bilateral subthalamic nucleus stimulation: A review. Parkinsonism Relat Disord 2011;17:413-7. 4. Demetriades P, Rickards H, Cavanna AE. Impulse control disorders following deep brain stimulation of the subthalamic nucleus in Parkinson’s disease: Clinical aspects. Parkinsons Dis 2011;2011:658415. 5. Doshi P, Bhargava P. Hypersexuality following subthalamic nucleus stimulation for Parkinson’s disease. Neurol India 2008;56:474-6. 6. Chang CH, Chen SY, Hsiao YL, Tsai ST, Tsai HC. Hypomania with hypersexuality following bilateral anterior limb stimulation in obsessive-compulsive disorder. J Neurosurg 2010;112:1299-300. 7. Klos KJ, Bower JH, Josephs KA, Matsumoto JY, Ahlskog JE. Pathological hypersexuality predominantly linked to adjuvant dopamine agonist therapy in Parkinson’s disease and multiple system atrophy. Parkinsonism Relat Disord 2005;11:381-6. 8. Temel Y, Blokland A, Steinbusch HW, Visser-Vandewalle V. The functional role of the subthalamic nucleus in cognitive and limbic circuits. Prog Neurobiol 2005;76:393-413. 9. Lim SY, O’Sullivan SS, Kotschet K, Gallagher DA, Lacey C, Lawrence AD, et al. Dopamine dysregulation syndrome, impulse control disorders and punding after deep brain stimulation surgery for Parkinson’s disease. J Clin Neurosci 2009;16:1148-52. 10. Ballanger B, van Eimeren T, Moro E, Lozano AM, Hamani C, Boulinguez P, et al. Stimulation of the subthalamic nucleus and impulsivity: Release your horses. Ann Neurol 2009;66:817-24.
Actively leaking ruptured acom aneurysm: Fountain sign Sir, We describe an interesting imaging finding in a patient with ruptured anterior communicating artery (Acom) aneurysm. The ruptured Acom aneurysm was actively leaking as observed in digital subtraction angiography (DSA), giving the appearance of a fountain, i.e. “the fountain sign” [Figure 1]. Axial computed tomography (CT) angiography image showed contrast-filled outpouching in the Acom region suggestive of Acom aneurysm [Figure 2]. DSA Towns view left internal carotid artery (ICA) injection showed Acom aneurysm with anterior cerebral artery (ACA) and terminal ICA vasospasm. There was contrast seen actively leaking out of the ruptured Acom aneurysm, giving the appearance of a fountain. In Video 1, the DSA running images Towne’s view left ICA injection give better
1.
Figure 1: Axial CT angiography image showing contrast-filled outpouching in the Acom region suggestive of acom aneurysm
Access this article online Quick Response Code:
Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.132453
Received: 26-01-2014 Review completed: 28-01-2014 Accepted: 06-04-2014 234
Figure 2: DSA angiography Towns view left ICA injection showing acom aneurysm with ACA and terminal ICA vasospasm. There is contrast seen actively leaking out of the ruptured Acom aneurysm, giving the appearance of a fountain
Neurology India | Mar-Apr 2014 | Vol 62 | Issue 2
Copyright of Neurology India is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
Hypersexuality after bilateral deep brain stimulation of the subthalamic nucleus for Parkinson’s disease Sir, Parkinson’s disease (PD) is a degenerative disorder of the central nervous system.[1] The cardinal symptoms are bradykinesia, rigidity, postural instability, and resting tremor. However, cognitive, mood, and behavioral problems may also arise either as a part of course of the disease or triggered by dopamine replacement therapy.[2] While deep brain stimulation of the subthalamic nucleus (DBS-STN) might be a therapeutic option for either PD or medication-related behavior, it has been reported that DBS-STN might also induce pathologic hypersexuality.[3-6] Here, we report a case of PD patient, who developed pathologic hypersexuality commencing after DBS-STN, despite remarkable motor improvement. A 58-year-old man suffering from PD was admitted for bilateral STN-DBS placement. After magnetic resonance imaging, a stereotactic frame (Integra CRW, New Jersey, USA) was placed onto the patient’s head on the day of the surgery and examined the brain with computed tomography. Later both images were auto-fused to target the anterior lateral side of left STN and right STN through image fusion program (Atlas Integra Software, New Jersey, USA). Bilateral micro-recording test (Alfa Omega Micro Recording System, Nazareth, Israel) was done. DBS electrode leads were placed at predefined targets (St. Jude Medical 6149 40 cm lead, Minnesota, USA) bilaterally to STN anteriolateral sides [Figure 1 left and right]. Then impulse generator (St. Jude Medical Libra XP 6644, Minnesota, USA) was implanted and connected to DBS electrode leads. The day after the surgery, the implanted impulse generator was turned on. The stimulation parameters are shown in Table 1. Preoperative drug regime was resumed. Postoperatively, there was remarkable improvement in his symptoms and according to the Unified Parkinson’s Disease Rating Scale III (UPDRS III), motor and total outcome. Both pre- and post-operative examinations are mentioned in Table 1. At the first follow-up visit, the patient’s wife mentioned that while he did not have any sexual gratification more than 2 years, after DBS-STN, every day he would insist on it and he would become very aggressive if denied. Although, he approved to be hypersexual and sometimes have uncontrollable behavior, he did not allow any intervention. Pathologic hypersexuality has been defined as ‘the need for sexual behavior that consumes so much money, time, concentration, and energy that the patient describes Neurology India | Mar-Apr 2014 | Vol 62 | Issue 2
Figure 1: Postoperative control cranial computed tomography
Table 1: UPDRS III in total and motor outcomes according to preoperative and postoperative of the bilateral STN-DBS and stimulation parameters of the patient with two channels and eight contacts constant current stimulator Preoperative STN-DBS Postoperative STN-DBS
Channel (left) Channel (right)
Motor
Total
72 34
114 63
Active contact
Amplitude (mA)
Width (uS)
Frequency (Hz)
3 2
3 2, 5
78 65
142 130
STN-DBS - Subthalamic nucleus deep brain stimulation, UPDRS III - Unified Parkinson’s disease rating scale III
himself as out of control; intrusive unwanted paraphiliac thoughts prevent concentration on other life demands and are the source of anxiety, and orgasm does not produce satiety in the way it typically does for age mates’.[7] Overall estimated prevalence of pathologic hypersexuality in PD varied between 2% and 10%, which can be induced even following DBS-STN.[3] In the literature, five cases of new onset pathologic hypersexuality after DBS-STN with no previous history of any impulse control disorder have been documented.[3-5] The role of the STN in the basal ganglia motor circuits is having a central position within the basal ganglia-thalamocortical associative and limbic circuits. It is functionally a potent regulator of these pathways.[8] Various pathophysiological mechanisms have been proposed to explain the impairment of cognition after DBS-STN. The electrodes are relatively large in comparison with the small size of the STN and it may be difficult to selectively influence the motor part of the STN without affecting domains within the STN or in neighboring structures that are associated with motivational, emotional, and cognitive functions.[9] Modulation of STN hyperactivity with DBS may improve impaired proactive inhibition in PD patients, but may induce impairment of reactive inhibition or cognitive 233
[Downloaded free from http://www.neurologyindia.com on Thursday, May 15, 2014, IP: 202.177.173.189] || Click here to download free Android application for this journal Letters to Editor
control that leads to premature and impulsive responses, such as hypersexuality.[3,4,10] In our case, while the patient had remarkable motor improvement with significant UPDRS motor score increase, he displayed clinical features of pathologic hypersexuality on follow-up examinations. In conclusion, DBS-STN may induce hypersexuality in rare occasions. Further careful evaluation is required to elucidate the mechanisms through which DBS-STN might affect sexual function.
Akın Akakın, Baran Yılmaz, Kamran Urgun, Murat Şakir Ekşi1, Nazire Afşar2, Türker Kılıç Departments of Neurosurgery, and 2Neurology, Bahcesehir Unıversity, Istanbul, Turkey, 1Department of Orthopedics, University of California, Spine Center, San Francisco, California, USA E-mail: [email protected]
References Li Q, Qian ZM, Arbuthnott GW, Ke Y, Yung WH. Cortical effects of deep brain stimulation: Implications for pathogenesis and treatment of Parkinson disease. JAMA Neurol 2014;71:100-3. 2. Merims D, Giladi N. Dopamine dysregulation syndrome, addiction and behavioral changes in Parkinson’s disease. Parkinsonism Relat Disord 2008;14:273-80. 3. Broen M, Duits A, Visser-Vandewalle V, Temel Y, Winogrodzka A. Impulse control and related disorders in Parkinson’s disease patients treated with bilateral subthalamic nucleus stimulation: A review. Parkinsonism Relat Disord 2011;17:413-7. 4. Demetriades P, Rickards H, Cavanna AE. Impulse control disorders following deep brain stimulation of the subthalamic nucleus in Parkinson’s disease: Clinical aspects. Parkinsons Dis 2011;2011:658415. 5. Doshi P, Bhargava P. Hypersexuality following subthalamic nucleus stimulation for Parkinson’s disease. Neurol India 2008;56:474-6. 6. Chang CH, Chen SY, Hsiao YL, Tsai ST, Tsai HC. Hypomania with hypersexuality following bilateral anterior limb stimulation in obsessive-compulsive disorder. J Neurosurg 2010;112:1299-300. 7. Klos KJ, Bower JH, Josephs KA, Matsumoto JY, Ahlskog JE. Pathological hypersexuality predominantly linked to adjuvant dopamine agonist therapy in Parkinson’s disease and multiple system atrophy. Parkinsonism Relat Disord 2005;11:381-6. 8. Temel Y, Blokland A, Steinbusch HW, Visser-Vandewalle V. The functional role of the subthalamic nucleus in cognitive and limbic circuits. Prog Neurobiol 2005;76:393-413. 9. Lim SY, O’Sullivan SS, Kotschet K, Gallagher DA, Lacey C, Lawrence AD, et al. Dopamine dysregulation syndrome, impulse control disorders and punding after deep brain stimulation surgery for Parkinson’s disease. J Clin Neurosci 2009;16:1148-52. 10. Ballanger B, van Eimeren T, Moro E, Lozano AM, Hamani C, Boulinguez P, et al. Stimulation of the subthalamic nucleus and impulsivity: Release your horses. Ann Neurol 2009;66:817-24.
Actively leaking ruptured acom aneurysm: Fountain sign Sir, We describe an interesting imaging finding in a patient with ruptured anterior communicating artery (Acom) aneurysm. The ruptured Acom aneurysm was actively leaking as observed in digital subtraction angiography (DSA), giving the appearance of a fountain, i.e. “the fountain sign” [Figure 1]. Axial computed tomography (CT) angiography image showed contrast-filled outpouching in the Acom region suggestive of Acom aneurysm [Figure 2]. DSA Towns view left internal carotid artery (ICA) injection showed Acom aneurysm with anterior cerebral artery (ACA) and terminal ICA vasospasm. There was contrast seen actively leaking out of the ruptured Acom aneurysm, giving the appearance of a fountain. In Video 1, the DSA running images Towne’s view left ICA injection give better
1.
Figure 1: Axial CT angiography image showing contrast-filled outpouching in the Acom region suggestive of acom aneurysm
Access this article online Quick Response Code:
Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.132453
Received: 26-01-2014 Review completed: 28-01-2014 Accepted: 06-04-2014 234
Figure 2: DSA angiography Towns view left ICA injection showing acom aneurysm with ACA and terminal ICA vasospasm. There is contrast seen actively leaking out of the ruptured Acom aneurysm, giving the appearance of a fountain
Neurology India | Mar-Apr 2014 | Vol 62 | Issue 2
Copyright of Neurology India is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
