Holmes tremor: a delayed complication after resection of brainstem cavernomas

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  • 1 Neurosurgery, Great Metropolitan Hospital Niguarda, Milan;
  • 2 Neurosurgery, Department of Human Neurosciences, Sapienza University, Rome, Italy;
  • 3 Department of Neurosurgery, Saint-Luc University Clinic, Catholic University of Louvain, Brussels, Belgium;
  • 4 Department of Neurosurgery, University Hospital Essen, University of Duisburg-Essen;
  • 5 Department of Neurosurgery, University Hospital Tübingen, Germany;
  • 6 Neurosurgery, Barrow Neurological Institute, Phoenix, Arizona;
  • 7 Neurosurgery, Burdenko Neurosurgical Institute, Moscow, Russia;
  • 8 Neurosurgery, University Hospital Uzhhorod, Ukraine;
  • 9 Department of Neurosurgery, Legnano Hospital, Milan;
  • 10 Department of Neurologic Surgery, Bellaria Hospital, Institute of Neurological Sciences of Bologna;
  • 11 Department of Neurosurgery, Carlo Besta Neurological Institute, Milan;
  • 12 Neurosurgery Department, University Hospital Verona;
  • 13 Departments of Medical Physics and
  • 14 Neuroradiology, Great Metropolitan Hospital Niguarda, Milan; and
  • 15 Department of Medical Biotechnologies and Translational Medicine, University of Milan, Italy
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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.

ABBREVIATIONS DBS = deep brain stimulation; DTT = dentatothalamic tract; EMG = electromyographic; FSL = Functional MRI of the Brain Software Library; FTOZ = frontotemporoorbitozygomatic; HT = Holmes tremor; Max = maximum intensity; NST = nigrostriatal tract; ROI = region of interest; ROT = rubroolivary tract; SEZ = safe entry zone; VIM = ventral intermediate nucleus.

Supplementary Materials

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Contributor Notes

Correspondence Davide Colistra: Great Metropolitan Hospital Niguarda, Milan, Italy. colistra.davide@gmail.com.

INCLUDE WHEN CITING Published online December 11, 2020; DOI: 10.3171/2020.7.JNS201352.

Disclosures The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper.

M.C. and D.C. contributed equally to this work.

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