technical note Use of a contoured bioresorbable plate with a hemostatic plug to control life-threatening bleeding from the superior sagittal sinus during hemispherotomy: technical note Gerald F. Tuite, MD,1,3 Carolyn M. Carey, MD,1,3 William W. Nelson, MD,4 Scott J. Raffa, MD,1,3 and S. Parrish Winesett, MD2,3 Divisions of 1Pediatric Neurosurgery and 2Neurology, Neuroscience Institute, All Children’s Hospital/Johns Hopkins Medicine, St. Petersburg; 3Department of Neurosurgery and Brain Repair, Morsani College of Medicine, University of South Florida, Tampa; and 4Division of Anesthesiology, Department of Surgery, All Children’s Hospital/Johns Hopkins Medicine, St. Petersburg, Florida Profuse bleeding originating from an injured cerebral sinus can be a harrowing experience for any surgeon, particularly during an operation on a young child. Common surgical remedies include sinus ligation, primary repair, placement of a hemostatic plug, and patch or venous grafting that may require temporary stenting. In this paper the authors describe the use of a contoured bioresorbable plate to hold a hemostatic plug in place along a tear in the inferomedial portion of a relatively inaccessible part of the posterior segment of the superior sagittal sinus in an 11-kg infant undergoing hemispherotomy for epilepsy. This variation on previously described hemostatic techniques proved to be easy, effective, and ultimately lifesaving. Surgeons may find this technique useful in similar dire circumstances when previously described techniques are ineffective or impractical. http://thejns.org/doi/abs/10.3171/2016.5.PEDS1633 Key Words superior sagittal sinus; cerebral sinus; injury; repair; hemispherotomy; hemorrhage; technique U profuse bleeding from the superior sagittal sinus (SSS) during surgery can provoke anxiety for even the most seasoned neurosurgeon. Bleeding from far anterior sites can be definitively halted with SSS ligation with minimal risk of cerebral ischemia or hemorrhage. However, more posterior ligation of the SSS is known to carry a significant risk of brain injury due to stroke, hemorrhage, or raised intracranial pressure, thereby necessitating repair when possible.28,29,31,34 Repairing a copiously bleeding cerebral sinus can be trepidatious because temporary proximal and distal control of the vessel can be difficult to obtain. Surgeons are burdened with controlling potentially life-threatening hemorrhage while remaining acutely aware of the potential consequences of exsanguination or severe brain injury if the repair is not successful.28,33,35 Primary suture is the optimal repair solution, but this is sometimes not feasible.2,23,26,33,35 Packing the surface of the nexpected sinus with thrombogenic materials in combination with a fibrin sealant will often suffice when the tear is small and when direct pressure can be maintained on the hemostatic plug.2,4,11–14 For large tears, repair with adjacent dura, a separate autologous or allograft material, or even a vein graft can be performed, with or without the assistance of a temporary flow diversion stent.8,18,22,26,30,33 Emergency endovascular solutions may offer a viable alternative in some select cases in the ideal setting, but this method has not been widely reported.36 In this paper we report our solution to life-threatening intraoperative bleeding originating from a hole in the inferomedial side of the SSS during hemispherotomy in an 11-kg infant. After considering and attempting many of the typical options, we were able to successfully stop the hemorrhage by fashioning a bioresorbable plate to hold packing material firmly against the medial surface of the SSS. Abbreviations SSS = superior sagittal sinus. SUBMITTED January 18, 2016. ACCEPTED May 4, 2016. include when citing Published online July 8, 2016; DOI: 10.3171/2016.5.PEDS1633. ©AANS, 2016 J Neurosurg Pediatr July 8, 2016 1 G. F. Tuite et al. Case Report Initial Operation This 11-kg, 11-month-old boy was selected for left periinsular hemispherotomy3,6,7,17,19,27,38 due to medically refractory epilepsy related to a large, left hemisphere, perinatal stroke (Fig. 1). The surgical approach proceeded without incident through a left frontotemporoparietal craniotomy that encompassed the insular area, with the medial margin of the craniotomy approximately 4 cm from the midline. A curvilinear dural opening was made, exposing an abnormal left hemisphere, consistent with imaging performed prior to surgery. The brain was noted to be bulging through the dural opening more than we would have expected in a child with brain atrophy (Fig. 2). While the operating microscope was being draped, before any resection of the brain, brisk venous bleeding was noted to be originating from the subdural space. There was no clear cause for the bleeding. The opening had been uneventful, no brain resection had been performed, and the patient was not undergoing hyperventilation. Dural elevation and subdural exploration to locate the source of bleeding resulted in nearly uncontrollable bleeding and massive blood loss, leading to severe hypotension and a cardiac arrhythmia. The resultant hypotension did reduce the bleeding enough to allow direct visualization of the bleeding site, which was determined to be an approximately 4-mm hole in the inferomedial side of the SSS. It appeared that a large cortical draining vein, possibly the vein of Trolard, had avulsed directly from the SSS along the middle one-third of the SSS.24 Repair of the Bleeding Coagulation of the bleeding cortical vein and direct compression of the hole in the SSS with Surgicel (Johnson & Johnson) followed by Floseal (Baxter), microfibrillar collagen (Avitene, CR Bard), and cottonoid patties stopped all bleeding, allowing the anesthesia team to transfuse packed red blood cells and fresh frozen plasma. Over the ensuing 30 minutes, direct pressure was maintained on the SSS while the child was stabilized. Brisk bleeding resumed every time the cotton patties were removed, regardless of the thickness of the hemostatic plugs, despite multiple attempts over subsequent hours. Because the bleeding could be controlled with direct pressure, we chose to fashion a bioresorbable plate (Re­ sorbX Mesh, KLS Martin LP) to hold the packing material in place over the defect in the SSS. The bioresorbable plate was contoured so it could be attached to the outer portion of the skull, bent 90°, and then shaped to maintain pressure on the packing material that was in place along the medial surface of the SSS (Fig. 3). To more securely hold the packing material in place, a long, thick, folded piece of dural substitute (Durepair, Medtronic) was placed between the hemostatic plug and the plate. We initially tried to fashion a titanium plate to hold the packing in place, but it fractured as it was bent excessively. Temporal and occipital lobectomies were performed while the hemostatic plug was manually held in place by an assistant. All bleeding stopped after the bioresorbable 2 J Neurosurg Pediatr July 8, 2016 Fig. 1. Axial T2-weighted MR image of the infant prior to surgery, illustrating the extensive left hemispheric injury related to perinatal ischemia. plate and packing material were in position. The dura was closed around the plate and the bone was replaced. The total operative time was 455 minutes exclusive of the anesthetic induction and preparation time. The total blood loss was 1400 ml and the child received 1069 ml (3 adult units, or 1.2 blood volumes) of packed red blood cells, 520 ml of fresh frozen plasma, 40 ml of platelets, 40 ml of cryoprecipitate, as well as Factor VIIa (NovoSeven, Novo Nordisk) and vitamin K. He was taken to the intensive care unit intubated, where he recovered. Postoperative Course The patient had an uneventful hospitalization for a child who had undergone a life-threatening operation. He was readmitted 4 weeks after surgery for Enterobacter cloacae meningitis, which required removal of the packing Fig. 2. Photograph of hemisphere bulging through the dural opening before any resection began, which may have led to the venous avulsion from the SSS. Figure is available in color online only. Resorbable plate with hemostatic plug to repair sinus injury Fig. 3. Intraoperative photograph (left) taken just prior to closure, showing the bioresorbable plate contoured to hold packing material firmly against the falx and SSS. Coronal contrast-enhanced CT scan (right) performed 1 day after surgery, showing the bioresorbable plate holding Durepair and a hemostatic plug against a patent SSS. Figure is available in color online only. material, the bioresorbable plate, and the bone flap. There was no bleeding from the SSS when the plate and packing material were removed. We realized there was a risk of bleeding associated with removal of the plate and packing, but we chose to return to surgery and accept that risk, without a lengthy trial of antibiotics, because the child had a serious infection with definite signs of sepsis. We believed that enough time had elapsed to allow sufficient healing of the sinus injury to allow attempted removal of the foreign body, which could have been contributing to the severity of the infection. CT venography performed the day after the initial surgery showed a patent SSS, but venous imaging 1 month after surgery showed a partially occluded SSS (Fig. 4). The child was treated with enoxaparin (Lovenox, Sanofi) for 6 months. By 12 months, the posterior portion of the SSS showed recanalization but the anterior portion remained patent yet diminutive (Fig. 4). Fig. 4. CT venography performed the day after surgery (A and B), and at 1 month (C and D) and 1 year (E and F) after surgery. The anterior portion of the SSS is widely patent 1 day after surgery (A), occluded 1 month later (C), and partially recanalized at 1 year (E). Similarly, the posterior portion of the SSS is patent 1 day after surgery (B), mostly occluded at 1 month (D), and mostly patent (F) 1 year after surgery. J Neurosurg Pediatr July 8, 2016 3 G. F. Tuite et al. No injury occurred on the contralateral hemisphere, and the boy’s neurological examination did not change after surgery (Fig. 5). An extensive hematological evaluation also failed to show any evidence of coagulopathy. The child has remained seizure free since surgery 1 year ago, without any antiepileptic medications for 6 months. Discussion Hemispherectomy and its surgical variations have been successfully used for the control of medically refractory epilepsy since it was initially advocated by McKenzie more than 75 years ago.3,7,19,32,38 Excellent seizure control can be achieved in carefully selected patients, but early enthusiasm for the procedure was tempered by high complication rates, particularly related to acute and chronic bleeding.39 Refinements in surgical technique have reduced hemorrhagic complications, but recent reviews still point to a 0.9%–2% overall mortality rate after hemispherectomy.17,21,37 Seizure control with hemispherectomy can be particularly dramatic and gratifying in infants with some of the most severe forms of epilepsy,1,3,5,8,10,15–17,20,21,25,27,37 but the risk of hemorrhage is greatest in babies with small total blood volumes and “immature brain and blood vessels that are fragile and vulnerable to bleeding.”10 While intraoperative death due to bleeding is exceedingly uncommon with modern anesthetic and resuscitative techniques, coagulopathy is common and even cardiac arrest can occur during hemispherectomy in the modern age of surgery.1,10 The current case illustrates the fragility of the blood vessels in a young infant as well as the consequences of sudden, profuse hemorrhage in a baby with a small blood volume. Surgical trauma during the opening, brain manipulation, or the bulging of the brain through the dural opening (Fig. 2) may have led to the avulsion of a large draining vein from the SSS, which resulted in life-threatening hemorrhage, cardiac arrhythmia, and coagulopathy within a very short period of time. Based on this experience, we recommend efforts to maintain the normal position of the brain after dural opening. It is possible that a mild degree of hyperventilation and/or modifications of patient positioning (neck less turned, head up slightly) might have kept our patient’s brain from bulging through the dural opening, which may have prevented a venous avulsion. Fig. 5. Axial (left) and coronal (right) CT scans of the infant 1 year after surgery, demonstrating typical postoperative changes after temporal and occipital lobectomies with an unaffected contralateral hemisphere. 4 J Neurosurg Pediatr July 8, 2016 We chose to use a contoured bioresorbable plate to maintain pressure on the hemostatic material that was placed over the hole in the SSS only after the typical options to control bleeding were considered and many were attempted. We do not believe the utilization of a bioresorbable plate to maintain pressure on the hemostatic plug has been previously described, but the concept is a variation on techniques that have been described by other authors.2,4,11,12,14 Other authors have also reported excellent hemostatic efficacy with long-term sinus patency utilizing a fibrin sealant with a structural thrombogenic material. Most authors emphasize the importance of maintaining pressure on the plug, often utilizing a dural tenting hitch stitch to pull the dura firmly to the overlying bone.2,4,11,12,14 Dural tenting was not technically feasible in our case because the SSS injury occurred along the inferomedial aspect of the SSS, in a location where a tenting dural stitch was not possible. As a substitute for a tenting suture, the bioresorbable plate proved easy and effective. Before utilizing the bioresorbable plate to hold the packing material in place, other hemostatic techniques were considered and many were attempted. Complete ligation of the SSS was not considered prudent because of the risk of bilateral ischemia that might have resulted from ligation of the SSS so far posteriorly.28,29,31,34 Primary suturing was not believed to be possible due to the width of the opening and the inelasticity of the sinus. Medial extension of the craniotomy with suturing of a dural leaflet over the hole in the SSS and falx was considered, but they were believed to be too hazardous given the risk of significant blood loss during such a maneuver in an 11-kg infant. Direct placement of a temporary stent was not possible because the hole in the sinus was not large enough to accommodate available stents. It could be argued that the presence of the plate increased the risk of infection, leading to the child’s meningitis. However, we believe the long duration of the procedure, the extraordinary blood loss with resultant transfusions, and the prolonged hypotension likely played a pivotal role in the child’s infection.15,17,37 Conclusions Serious bleeding from an injury to the inferomedial wall of the SSS during hemispherectomy was effectively controlled with a thrombotic plug held in place with a bioresorbable plate. This plate was attached to the outer skull layer and contoured into an S-shape to allow maintenance of compression of a hemostatic plug on the injured inferomedial SSS. The use of the bioresorbable plate proved to be an effective, efficient, and potentially lifesaving technique. This technique may be applicable to other surgical scenarios in which robust cerebral sinus bleeding from poorly accessible locations is encountered, when standard hemostatic techniques prove difficult. Acknowledgments We would like to thank Allyn DiVito for his assistance with the figure annotations. William Nelson, MD, the anesthesiologist caring for this child during surgery, deserves credit for his exem- Resorbable plate with hemostatic plug to repair sinus injury plary resuscitative efforts and for suggesting we use a resorbable plate, which was much easier to contour than our attempts with a metal plate. References 1. 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Brain 93:147–180, 1970 Disclosures The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper. 6 J Neurosurg Pediatr July 8, 2016 Author Contributions Conception and design: all authors. Acquisition of data: all authors. Analysis and interpretation of data: all authors. Drafting the article: Tuite. Critically revising the article: all authors. Reviewed submitted version of manuscript: all authors. Approved the final version of the manuscript on behalf of all authors: Tuite. Administrative/technical/material support: Tuite. Study supervision: Tuite. Correspondence Gerald Tuite, Division of Pediatric Neurosurgery, Neuroscience Institute, All Children’s Hospital/Johns Hopkins Medicine, 601 5th St. S, Ste. 511, St. Petersburg, FL 33701. email: geraldtuite@ gmail.com.