- Email: [email protected]
Dopamine dysregulation syndrome and psychosis in 24-h intestinal levodopa infusion for Parkinson’s disease
Parkinsonism and Related Disorders 31 (2016) 144e145
Contents lists available at ScienceDirect
Parkinsonism and Related Disorders journal homepage: www.elsevier.com/locate/parkreldis
Correspondence
Dopamine dysregulation syndrome and psychosis in 24-h intestinal levodopa infusion for Parkinson’s disease
Keywords: Dopamine dysregulation syndrome Capgras syndrome Othello syndrome Parkinson’s disease Levodopa-carbidopa intestinal gel infusion
Dear Editor, We read with interest the paper of Ricciardi et al. [1] describing a male patient affected by advanced Parkinson’s disease (PD) who developed a dopamine dysregulation syndrome (DDS) associated with Othello and Capgras Syndromes in the context of a severe psychosis while on 24-h levodopa/carbidopa intestinal gel (LCIG). While Othello syndrome is a paranoid delusion supported by the absolute certainty of the infidelity of the partner, Capgras syndrome is a delusional misidentification syndrome (DMS) in which the patients are convinced that a familiar person has been replaced by an impostor or a double who is physically very similar to the original. We previously described the presence of Othello Syndrome in PD patients as a dopaminergic-induced psychosis not necessarily associated with cognitive impairment [2], while Capgras syndrome has been scarcely explored. The merits of the paper of Ricciardi et al. lie precisely in the demonstration of a DDS associated with the appearance of these delusional syndromes, triggered by unnecessary dosages of LCIG, with a review of several cases of Capgras syndrome in PD patients, suggesting a dopaminergic-induced psychosis in the setting of cognitive impairment. However, several fundamental issues, especially on dopaminergic treatment, remain not discussed. First of all, it is not clear why the patient, with a previous history of levodopa/entacapone visual hallucinations triggered at a dosage of 400/600 mg/day, was started on a levodopa dose of 48 mg/h for 16 h (768 mg/day) which appears excessive in comparison with his previous treatment (even considering the possible use of extra doses self-administered by the patient), although the authors described a virtual elimination of OFF time and no side effects. Secondly, we were relatively surprised that the same patient on overnight LCIG, may have self-administered a levodopa dosage of 2030 mg/day within six months, and maintained for other two http://dx.doi.org/10.1016/j.parkreldis.2016.06.022 1353-8020/© 2016 Elsevier Ltd. All rights reserved.
months (which means more of one cassette per day) without any evidence of dyskinesias at follow-up visits. In our clinical practice, the choice of an overnight administration of LCIG requires a more frequent and stricter control of motor and non motor complications. As described by the same authors, we were the first to describe two patients who developed DDS on LCIG and both developed delusional disorders, mainly of paranoid type [3]. These patients did not respect follow-up schedule, preferring telephonic contacts to visits, with an inappropriate monitoring of the treatment and insufficient therapeutical adjustment [3]. According to author’s opinion, although the use of LCIG in PD patients is generally a safe and efficacious treatment option in advanced PD patients, this case emphasizes that a correct management of this anti-parkinsonian infusional treatment is needed. Moreover, we think that the authors reported erroneously as “two cases of DDS have been reported recently after LCIG initiation” [3,4] citing both our first description of two patients who developed DDS on LCIG and the case of a patient with a previous history of DDS who subsequently relapsed on LCIG. Indeed, the cases previously described are four, because these authors have missed the more recent paper of Salomone et al. [5], who described another patient with DDS and punding on LCIG. Finally it seems worth noting that the review of previous cases of PD patients with Capgras syndrome reported in the literature (seven cases reported plus the case of the paper) is largely incomplete, as at least four cases were not included [6e8]. These cases not only confirm the key role of the cognitive impairment in the development of Capgras syndrome, but also add new elements, given that neurosurgical procedures as pallidotomy intervention [6] and deep brain stimulation [7] may be associated with Capgras syndrome. Aside these criticisms on the incomplete review of the literature, we are in strong agreement with the general view of the authors on the crucial need of treatment monitoring for the prevention and management of behavioural disorders such as DDS in PD patients on LCIG, particularly in patients with previous dopaminergicrelated abnormal behaviours, and high total daily dosage of dopaminergic drugs. We retain that PD patients on 24-h administration of LCIG should be more carefully monitorized with a more frequent and tighter control of motor and non motor complications. Authors’ role 1. Research project: A. Conception, B. Organization, C. Execution; 2. Statistical Analysis: A. Design, B. Execution, C. Review and Critique; 3. Manuscript Preparation: A. Writing of the first draft,
Correspondence / Parkinsonism and Related Disorders 31 (2016) 144e145
B. Review and Critique. PS: 1ABC, 3AB. AC: 1B, 3B. MM: 1B, 3B. FM: 1B, 3B. Conflicts of interest The authors declare that they have no conflict of interest. Disclosures P. Solla has received institutional research funding from the University of Cagliari, received funding from the Fondazione Banco di Sardegna, and received research grant from the Dystonia Europe. A. Cannas reports no disclosures relevant to the manuscript. M. Meloni reports no disclosures relevant to the manuscript. F. Marrosu reports no disclosures relevant to the manuscript. References [1] L. Ricciardi, K.J. Espay, R. Krikorian, A. Fasano, A.J. Espay, Dopamine dysregulation syndrome with psychosis in 24-hour intestinal levodopa infusion for Parkinson’s disease, Park. Relat. Disord. 28 (2016) 152e154. [2] A. Cannas, P. Solla, G. Floris, P. Tacconi, F. Marrosu, M.G. Marrosu, Othello syndrome in parkinson disease patients without dementia, Neurologist 15 (1) (2009) 34e36. [3] A. Cannas, P. Solla, M.G. Marrosu, F. Marrosu, Dopamine dysregulation syndrome in Parkinson’s disease patients on duodenal levodopa infusion, Mov.
1
All the authors contributed equally to this study.
145
Disord. 28 (6) (2013) 840e841. [4] R. Cilia, C. Siri, M. Canesi, A.L. Zecchinelli, D. De Gaspari, F. Natuzzi, S. Tesei, N. Meucci, C.B. Mariani, G. Sacilotto, M. Zini, C. Ruffmann, G. Pezzoli, Dopamine dysregulation syndrome in Parkinson’s disease: from clinical and neuropsychological characterisation to management and long-term outcome, J. Neurol. Neurosurg. Psychiatry 85 (3) (2014 Mar) 311e318. [5] G. Salomone, M. Marano, L. di Biase, J.M. Melgari, V. Di Lazzaro, Dopamine dysregulation syndrome and punding in levodopa-carbidopa intestinal gel (LCIG) infusion: a serious but preventable complication, Park. Relat. Disord. 21 (9) (2015) 1124e1125. [6] H. Apaydin, G. Benbir, Capgras syndrome during the course of Parkinson disease dementia, J. Neuropsychiatry Clin. Neurosci. 26 (3) (2014) E46. [7] C.R. Kyrtsos, M.C. Stahl, P. Eslinger, T. Subramanian, E.B. Lucassen, Capgras syndrome in a patient with Parkinson’s disease after bilateral subthalamic nucleus deep brain stimulation: a case report, Case Rep. Neurol. 7 (2) (2015) 127e133. [8] H. Shiotsuki, Y. Motoi, S. Nakamura, Y. Mizuno, N. Hattori, Dopamine deficiency may lead to Capgras syndrome in Parkinson’s disease with dementia, J. Neuropsychiatry Clin. Neurosci. 22 (3) (2010), 352i.e14e352.e15.
Paolo Solla*,1, Antonino Cannas1, Mario Meloni1, Francesco Marrosu1 Movement Disorders Center, Department of Public Health, Clinical and Molecular Medicine, University of Cagliari, Cagliari, Italy *
Corresponding author. Movement Disorders Center, Department of Public Health, Clinical and Molecular Medicine, University of Cagliari, SS 554 Bivio per Sestu, 09042 Monserrato, Cagliari, Italy. E-mail address: [email protected] (P. Solla). 7 June 2016
Contents lists available at ScienceDirect
Parkinsonism and Related Disorders journal homepage: www.elsevier.com/locate/parkreldis
Correspondence
Dopamine dysregulation syndrome and psychosis in 24-h intestinal levodopa infusion for Parkinson’s disease
Keywords: Dopamine dysregulation syndrome Capgras syndrome Othello syndrome Parkinson’s disease Levodopa-carbidopa intestinal gel infusion
Dear Editor, We read with interest the paper of Ricciardi et al. [1] describing a male patient affected by advanced Parkinson’s disease (PD) who developed a dopamine dysregulation syndrome (DDS) associated with Othello and Capgras Syndromes in the context of a severe psychosis while on 24-h levodopa/carbidopa intestinal gel (LCIG). While Othello syndrome is a paranoid delusion supported by the absolute certainty of the infidelity of the partner, Capgras syndrome is a delusional misidentification syndrome (DMS) in which the patients are convinced that a familiar person has been replaced by an impostor or a double who is physically very similar to the original. We previously described the presence of Othello Syndrome in PD patients as a dopaminergic-induced psychosis not necessarily associated with cognitive impairment [2], while Capgras syndrome has been scarcely explored. The merits of the paper of Ricciardi et al. lie precisely in the demonstration of a DDS associated with the appearance of these delusional syndromes, triggered by unnecessary dosages of LCIG, with a review of several cases of Capgras syndrome in PD patients, suggesting a dopaminergic-induced psychosis in the setting of cognitive impairment. However, several fundamental issues, especially on dopaminergic treatment, remain not discussed. First of all, it is not clear why the patient, with a previous history of levodopa/entacapone visual hallucinations triggered at a dosage of 400/600 mg/day, was started on a levodopa dose of 48 mg/h for 16 h (768 mg/day) which appears excessive in comparison with his previous treatment (even considering the possible use of extra doses self-administered by the patient), although the authors described a virtual elimination of OFF time and no side effects. Secondly, we were relatively surprised that the same patient on overnight LCIG, may have self-administered a levodopa dosage of 2030 mg/day within six months, and maintained for other two http://dx.doi.org/10.1016/j.parkreldis.2016.06.022 1353-8020/© 2016 Elsevier Ltd. All rights reserved.
months (which means more of one cassette per day) without any evidence of dyskinesias at follow-up visits. In our clinical practice, the choice of an overnight administration of LCIG requires a more frequent and stricter control of motor and non motor complications. As described by the same authors, we were the first to describe two patients who developed DDS on LCIG and both developed delusional disorders, mainly of paranoid type [3]. These patients did not respect follow-up schedule, preferring telephonic contacts to visits, with an inappropriate monitoring of the treatment and insufficient therapeutical adjustment [3]. According to author’s opinion, although the use of LCIG in PD patients is generally a safe and efficacious treatment option in advanced PD patients, this case emphasizes that a correct management of this anti-parkinsonian infusional treatment is needed. Moreover, we think that the authors reported erroneously as “two cases of DDS have been reported recently after LCIG initiation” [3,4] citing both our first description of two patients who developed DDS on LCIG and the case of a patient with a previous history of DDS who subsequently relapsed on LCIG. Indeed, the cases previously described are four, because these authors have missed the more recent paper of Salomone et al. [5], who described another patient with DDS and punding on LCIG. Finally it seems worth noting that the review of previous cases of PD patients with Capgras syndrome reported in the literature (seven cases reported plus the case of the paper) is largely incomplete, as at least four cases were not included [6e8]. These cases not only confirm the key role of the cognitive impairment in the development of Capgras syndrome, but also add new elements, given that neurosurgical procedures as pallidotomy intervention [6] and deep brain stimulation [7] may be associated with Capgras syndrome. Aside these criticisms on the incomplete review of the literature, we are in strong agreement with the general view of the authors on the crucial need of treatment monitoring for the prevention and management of behavioural disorders such as DDS in PD patients on LCIG, particularly in patients with previous dopaminergicrelated abnormal behaviours, and high total daily dosage of dopaminergic drugs. We retain that PD patients on 24-h administration of LCIG should be more carefully monitorized with a more frequent and tighter control of motor and non motor complications. Authors’ role 1. Research project: A. Conception, B. Organization, C. Execution; 2. Statistical Analysis: A. Design, B. Execution, C. Review and Critique; 3. Manuscript Preparation: A. Writing of the first draft,
Correspondence / Parkinsonism and Related Disorders 31 (2016) 144e145
B. Review and Critique. PS: 1ABC, 3AB. AC: 1B, 3B. MM: 1B, 3B. FM: 1B, 3B. Conflicts of interest The authors declare that they have no conflict of interest. Disclosures P. Solla has received institutional research funding from the University of Cagliari, received funding from the Fondazione Banco di Sardegna, and received research grant from the Dystonia Europe. A. Cannas reports no disclosures relevant to the manuscript. M. Meloni reports no disclosures relevant to the manuscript. F. Marrosu reports no disclosures relevant to the manuscript. References [1] L. Ricciardi, K.J. Espay, R. Krikorian, A. Fasano, A.J. Espay, Dopamine dysregulation syndrome with psychosis in 24-hour intestinal levodopa infusion for Parkinson’s disease, Park. Relat. Disord. 28 (2016) 152e154. [2] A. Cannas, P. Solla, G. Floris, P. Tacconi, F. Marrosu, M.G. Marrosu, Othello syndrome in parkinson disease patients without dementia, Neurologist 15 (1) (2009) 34e36. [3] A. Cannas, P. Solla, M.G. Marrosu, F. Marrosu, Dopamine dysregulation syndrome in Parkinson’s disease patients on duodenal levodopa infusion, Mov.
1
All the authors contributed equally to this study.
145
Disord. 28 (6) (2013) 840e841. [4] R. Cilia, C. Siri, M. Canesi, A.L. Zecchinelli, D. De Gaspari, F. Natuzzi, S. Tesei, N. Meucci, C.B. Mariani, G. Sacilotto, M. Zini, C. Ruffmann, G. Pezzoli, Dopamine dysregulation syndrome in Parkinson’s disease: from clinical and neuropsychological characterisation to management and long-term outcome, J. Neurol. Neurosurg. Psychiatry 85 (3) (2014 Mar) 311e318. [5] G. Salomone, M. Marano, L. di Biase, J.M. Melgari, V. Di Lazzaro, Dopamine dysregulation syndrome and punding in levodopa-carbidopa intestinal gel (LCIG) infusion: a serious but preventable complication, Park. Relat. Disord. 21 (9) (2015) 1124e1125. [6] H. Apaydin, G. Benbir, Capgras syndrome during the course of Parkinson disease dementia, J. Neuropsychiatry Clin. Neurosci. 26 (3) (2014) E46. [7] C.R. Kyrtsos, M.C. Stahl, P. Eslinger, T. Subramanian, E.B. Lucassen, Capgras syndrome in a patient with Parkinson’s disease after bilateral subthalamic nucleus deep brain stimulation: a case report, Case Rep. Neurol. 7 (2) (2015) 127e133. [8] H. Shiotsuki, Y. Motoi, S. Nakamura, Y. Mizuno, N. Hattori, Dopamine deficiency may lead to Capgras syndrome in Parkinson’s disease with dementia, J. Neuropsychiatry Clin. Neurosci. 22 (3) (2010), 352i.e14e352.e15.
Paolo Solla*,1, Antonino Cannas1, Mario Meloni1, Francesco Marrosu1 Movement Disorders Center, Department of Public Health, Clinical and Molecular Medicine, University of Cagliari, Cagliari, Italy *
Corresponding author. Movement Disorders Center, Department of Public Health, Clinical and Molecular Medicine, University of Cagliari, SS 554 Bivio per Sestu, 09042 Monserrato, Cagliari, Italy. E-mail address: [email protected] (P. Solla). 7 June 2016