Marco Cenzato1, Davide Colistra1, Giorgia Iacopino2, Christian Raftopoulos3, Ulrich Sure4, Marcos Tatagiba5, Robert F Spetzler6, Alexander N Konovalov7, Andriy Smolanka8, Volodymir Smolanka8, Roberto Stefini9, Carlo Bortolotti10, Paolo Ferroli11, Giampietro Pinna12, Angelo Franzini11, Philipp Dammann4, Georgios Naros5, Davide Boeris1, Paolo Mantovani10, Domenico Lizio13, Mariangela Piano14, Enrica Fava1,15. 1. 1Neurosurgery, Great Metropolitan Hospital Niguarda, Milan. 2. 2Neurosurgery, Department of Human Neurosciences, Sapienza University, Rome, Italy. 3. 3Department of Neurosurgery, Saint-Luc University Clinic, Catholic University of Louvain, Brussels, Belgium. 4. 4Department of Neurosurgery, University Hospital Essen, University of Duisburg-Essen. 5. 5Department of Neurosurgery, University Hospital Tübingen, Germany. 6. 6Neurosurgery, Barrow Neurological Institute, Phoenix, Arizona. 7. 7Neurosurgery, Burdenko Neurosurgical Institute, Moscow, Russia. 8. 8Neurosurgery, University Hospital Uzhhorod, Ukraine. 9. 9Department of Neurosurgery, Legnano Hospital, Milan. 10. 10Department of Neurologic Surgery, Bellaria Hospital, Institute of Neurological Sciences of Bologna. 11. 11Department of Neurosurgery, Carlo Besta Neurological Institute, Milan. 12. 12Neurosurgery Department, University Hospital Verona. 13. Departments of13Medical Physics and. 14. 14Neuroradiology, Great Metropolitan Hospital Niguarda, Milan; and. 15. 15Department of Medical Biotechnologies and Translational Medicine, University of Milan, Italy.
Abstract
OBJECTIVE: In this paper, the authors aimed to illustrate how Holmes tremor (HT) can occur as a delayed complication after brainstem cavernoma resection despite strict adherence to the safe entry zones (SEZs). METHODS: After operating on 2 patients with brainstem cavernoma at the Great Metropolitan Hospital Niguarda in Milan and noticing a similar pathological pattern postoperatively, the authors asked 10 different neurosurgery centers around the world to identify similar cases, and a total of 20 were gathered from among 1274 cases of brainstem cavernomas. They evaluated the tremor, cavernoma location, surgical approach, and SEZ for every case. For the 2 cases at their center, they also performed electromyographic and accelerometric recordings of the tremor and evaluated the post-operative tractographic representation of the neuronal pathways involved in the tremorigenesis. After gathering data on all 1274 brainstem cavernomas, they performed a statistical analysis to determine if the location of the cavernoma is a potential predicting factor for the onset of HT. RESULTS: From the analysis of all 20 cases with HT, it emerged that this highly debilitating tremor can occur as a delayed complication in patients whose postoperative clinical course has been excellent and in whom surgical access has strictly adhered to the SEZs. Three of the patients were subsequently effectively treated with deep brain stimulation (DBS), which resulted in complete or almost complete tremor regression. From the statistical analysis of all 1274 brainstem cavernomas, it was determined that a cavernoma location in the midbrain was significantly associated with the onset of HT (p < 0.0005). CONCLUSIONS: Despite strict adherence to SEZs, the use of intraoperative neurophysiological monitoring, and the immediate success of a resective surgery, HT, a severe neurological disorder, can occur as a delayed complication after resection of brainstem cavernomas. A cavernoma location in the midbrain is a significant predictive factor for the onset of HT. Further anatomical and neurophysiological studies will be necessary to find clues to prevent this complication.
OBJECTIVE: In this paper, the authors aimed to illustrate how Holmes tremor (HT) can occur as a delayed complication after brainstem cavernoma resection despite strict adherence to the safe entry zones (SEZs). METHODS: After operating on 2 patients with brainstem cavernoma at the Great Metropolitan Hospital Niguarda in Milan and noticing a similar pathological pattern postoperatively, the authors asked 10 different neurosurgery centers around the world to identify similar cases, and a total of 20 were gathered from among 1274 cases of brainstem cavernomas. They evaluated the tremor, cavernoma location, surgical approach, and SEZ for every case. For the 2 cases at their center, they also performed electromyographic and accelerometric recordings of the tremor and evaluated the post-operative tractographic representation of the neuronal pathways involved in the tremorigenesis. After gathering data on all 1274 brainstem cavernomas, they performed a statistical analysis to determine if the location of the cavernoma is a potential predicting factor for the onset of HT. RESULTS: From the analysis of all 20 cases with HT, it emerged that this highly debilitating tremor can occur as a delayed complication in patients whose postoperative clinical course has been excellent and in whom surgical access has strictly adhered to the SEZs. Three of the patients were subsequently effectively treated with deep brain stimulation (DBS), which resulted in complete or almost complete tremor regression. From the statistical analysis of all 1274 brainstem cavernomas, it was determined that a cavernoma location in the midbrain was significantly associated with the onset of HT (p < 0.0005). CONCLUSIONS: Despite strict adherence to SEZs, the use of intraoperative neurophysiological monitoring, and the immediate success of a resective surgery, HT, a severe neurological disorder, can occur as a delayed complication after resection of brainstem cavernomas. A cavernoma location in the midbrain is a significant predictive factor for the onset of HT. Further anatomical and neurophysiological studies will be necessary to find clues to prevent this complication.