Catherine M K E de Cuba1, Alberto Albanese2, Angelo Antonini3, Giovanni Cossu4, Günther Deuschl5, Roberto Eleopra6, Alejandro Galati7, Carel F E Hoffmann8, Karina Knudsen5, Andrea Landi9, Michele Maria R Lanotte10, Andrea Marcante3, Arne Mosch11, Manuela Pilleri12, Martin M Reich13, Valeria Ricchi4, Sara Rinaldo6, Luigi M Romito14, Felipe S Saba15, Horacio E Sacristan16, P Richard Schuurman1, Andrea Trezza9, Pepijn van den Munckhof1, Jens Volkmann13, Maurizio Zibetti10, Maria Fiorella Contarino17. 1. Department of Neurosurgery, Academic Medical Center, Meibergdreef 9, 1105 AZ, Amsterdam, The Netherlands. 2. Department of Neurology, Istituto Neurologico Carlo Besta, Via Giovanni Celoria 11, 20133, Milan, Italy; Department of Neurology, Istituto Clinico Humanitas, Via Alessandro Manzoni 113, Rozzano, Milano, Italy. 3. UO Parkinson -IRCCS San Camillo, Via Alberoni 70, 30126, Venice, Italy. 4. Neurology Unit, Brotzu General Hospital, Piazzale Ricchi 1, Cagliari, Italy. 5. Department of Neurology, Universitätsklinik Schleswig-Holstein, Christian-Albrechts University, Christian-Albrechts-Platz 4, 24118, Kiel, Germany. 6. Neurological Unit, S. Maria della Misericordia University Hospital, Piazzale Santa Maria della Misericordia 15, Udine, Italy. 7. Department of Neurosurgery, Prof. Dr. Bernardo A. Houssay Hospital, Pres. Hipólito Yrigoyen 1757, Buenos Aires, Argentina. 8. Department of Neurosurgery, Haga Teaching Hospital, Leyweg 275, 2545 CH, The Hague, The Netherlands. 9. Department of Neurosurgery, San Gerardo Hospital, Via Giambattista Pergolesi 33, 20052, Monza, Italy. 10. Department of Neuroscience, University of Turin, Via Cherasco 15, Turin, Italy. 11. Department of Neurology, Haga Teaching Hospital, Leyweg 275, 2545 CH, The Hague, The Netherlands. 12. UO Parkinson -IRCCS San Camillo, Via Alberoni 70, 30126, Venice, Italy; UF Neurologia Casa di Cura Villa Margherita, Via Costacolonna 6, 36057, Vicenza, Italy. 13. Department of Neurology, University Clinic of Würzburg, Josef-Schneider-Str. 11, 97080, Würzburg, Germany. 14. Department of Neurology, Istituto Neurologico Carlo Besta, Via Giovanni Celoria 11, 20133, Milan, Italy. 15. Department of Neurology, Istituto Neurologico Carlo Besta, Via Giovanni Celoria 11, 20133, Milan, Italy; Departmento de neurociências en ciências do comportamento, Universidade de São Paulo, Faculdade de Medicina de Riberão Preto - Serviço de Neurocirurgia Funcional, Hospital do Coração, R. Abílio Soares, 250 - Paraíso, São Paulo, SP, 04005-000, Brazil. 16. Department of Neurology, Prof. Dr. Bernardo A. Houssay Hospital, Pres. Hipólito Yrigoyen 1757, Buenos Aires, Argentina. 17. Department of Neurology, Haga Teaching Hospital, Leyweg 275, 2545 CH, The Hague, The Netherlands; Department of Neurology, Leiden University Medical Centre, Albinusdreef 2, 2333 ZA Leiden, The Netherlands. Electronic address: m.f.contarino@lumc.nl.
Abstract
INTRODUCTION: Deep brain stimulation (DBS) is effective for some neurological and psychiatric conditions. Idiopathic delayed-onset edema (IDE) surrounding the leads has been anecdotally reported. The etiology, predisposing factors and prognosis of this complication are unknown. We present a multicenter case series of patients with IDE, and a systematic literature review, aimed at defining the pathophysiology and identifying appropriate treatment strategies. METHODS: IDE was defined as edema along the DBS lead, occurring ≥72 h postoperatively, in absence of trauma, vascular events or infection. Information on patients with IDE was collected in a standardized way. A systematic search was performed in Pubmed. RESULTS: Twelve new patients presenting with 14 episodes of IDE are described. From the literature, 38 patients were identified. No common surgical aspects or patient-related factors were identified as risk predictors for the onset of IDE. Symptoms included deterioration of the stimulation effect, seizures and focal neurological signs. Although the condition is self-limiting, with symptoms resolution in 28.5 days on average, three patients underwent surgical revision and seven received antibiotics. CONCLUSIONS: IDE is a rare complication of DBS procedures, presenting from few days to months after surgery. Symptoms can be mild and not-specific, and the condition is self-limiting. The diagnosis of IDE is made after exclusion of vascular events or infections. The pathophysiology is still unexplained. The recognition of this complication can help avoiding unnecessary surgical procedures (system explantation) and antibiotic treatment.
INTRODUCTION: Deep brain stimulation (DBS) is effective for some neurological and psychiatric conditions. Idiopathic delayed-onset edema (IDE) surrounding the leads has been anecdotally reported. The etiology, predisposing factors and prognosis of this complication are unknown. We present a multicenter case series of patients with IDE, and a systematic literature review, aimed at defining the pathophysiology and identifying appropriate treatment strategies. METHODS: IDE was defined as edema along the DBS lead, occurring ≥72 h postoperatively, in absence of trauma, vascular events or infection. Information on patients with IDE was collected in a standardized way. A systematic search was performed in Pubmed. RESULTS: Twelve new patients presenting with 14 episodes of IDE are described. From the literature, 38 patients were identified. No common surgical aspects or patient-related factors were identified as risk predictors for the onset of IDE. Symptoms included deterioration of the stimulation effect, seizures and focal neurological signs. Although the condition is self-limiting, with symptoms resolution in 28.5 days on average, three patients underwent surgical revision and seven received antibiotics. CONCLUSIONS: IDE is a rare complication of DBS procedures, presenting from few days to months after surgery. Symptoms can be mild and not-specific, and the condition is self-limiting. The diagnosis of IDE is made after exclusion of vascular events or infections. The pathophysiology is still unexplained. The recognition of this complication can help avoiding unnecessary surgical procedures (system explantation) and antibiotic treatment.