Journal of Perinatology (2015) 35, 460–462 © 2015 Nature America, Inc. All rights reserved 0743-8346/15 www.nature.com/jp
PERINATAL/NEONATAL CASE PRESENTATION
Acute paraplegia in a preterm infant with cerebral sinovenous thrombosis J Hobbs1, A Tekes2,3, J Klein3,4, M Lemmon3,4, RJ Felling3,4 and R Chavez-Valdez1,3 We report the case of a 1-month old, 28-week gestational age infant who presented with acute paraplegia after cardiopulmonary arrest. Later imaging confirms cerebral sinovenous thrombosis (CSVT) and a suspected infarction in the conus medullaris of the spinal cord. A prothrombotic state may explain the numerous areas of infarction visualized on neuroimaging. To our knowledge this is the first case report of acute and persistent paraplegia in an infant with CSVT and conus medullaris injury, which may be due to venous infarction of the spinal cord. Journal of Perinatology (2015) 35, 460–462. doi:10.1038/jp.2015.26
INTRODUCTION Cerebral sinovenous thrombosis (CSVT) is an uncommon, but increasingly recognized cause of neonatal stroke.1 Depending on the neuroimaging modality, the reported incidence of neonatal CSVT varies from 0.6 to 12 for every 100 000 live births.2 In premature infants, CSVT incidence has been reported as high as 4.4% using head ultrasound (HUS) with Doppler scan through the mastoid fontanelle confirmed with magnetic resonance imaging (MRI).3 The superior sagittal and transverse lateral sinuses are the most common CSVT sites.4,5 CSVT accompanied by spinal cord injury has never been reported in premature neonates. Most neonates with CSVT present within 48 h with seizures, irritability and hypotonia, and less often with apnea, motor weakness and cranial nerve palsies.5–8 The spectrum of CSVT presentation in neonates ranges from asymptomatic (13%)2,7 to deathly (6 to 25%).2,4,5,9 Pre-eclampsia, premature rupture of membranes and neonatal dehydration, infection, polycythemia, trauma, hypoxic ischemic injury and prothrombotic states are the main risk factors for neonatal CSVT.2,4,5 We report a case of a 25-day-old premature infant with acute paraplegia following cardiorespiratory arrest at day of life (DOL) 13 and diagnosed with cerebral, cerebellar and spinal cord hemorrhagic infarcts associated with extensive CSVT. CASE This 850-g extreme low birth weight infant was born via cesarean section at 28 2/7 weeks secondary to chronic hypertension with superimposed pre-eclampsia. Prenatal laboratories were unremarkable. Rupture of membranes occurred at the time of delivery. Apgar scores were 8 at both 1 and 5 min. Supplemental oxygen was provided at 5 min of life for oxygen saturations below Neonatal Resuscitation Program guidelines. The infant received antibiotics until DOL 2 and minimal invasive ventilator support until DOL 9. The infant was initiated on total parenteral nutrition and caffeine citrate on DOL 1, and trophic
feeds on DOL 3. On DOL 9, soon after endotracheal tube removal, patient developed increased events of apnea, bradycardia, oxygen desaturations and feeding intolerance with no hemodynamic instability. Management included nothing per os (NPO), vancomycin and gentamicin and adjustment on high-flow nasal cannula support. All abdominal radiographs were negative for pneumatosis intestinalis or peritoneal free air. Blood work results did not support infection. Trophic feeds were resumed by DOL 13. At the end of DOL 13, the infant had a cardiopulmonary arrest receiving positive pressure ventilation, chest compressions and intravenous epinephrine. The treating team obtained: bacterial and viral cultures of blood, urine and cerebrospinal fluid (CSF), amplitude integrated electroencephalogram (aEEG), HUS and twodimensional echocardiogram. The infant was transitioned to high-frequency oscillatory ventilation, initiated on amphotericin, acyclovir, vancomycin and cefotaxime, and treated with fluid boluses and inotropes for systemic hypotension. HUSs on DOL 3, 13 (day of cardiorespiratory event) and 17 were reported as within normal limits. Echocardiogram on DOL 13 showed mild pulmonary hypertension but normal biventricular function and no evidence of patent ductus arteriosus. Electrocardiogram tracing showed sinus rhythm before and after cardiorespiratory event. aEEG showed no evidence of seizure activity or abnormal background for 72 h after the event. CSF studies were within normal limits and all cultures were negative. The infant’s neurologic exam on DOL 13 demonstrated symmetric spontaneous movement, and normal tone, deep tendon and primitive reflexes after the event. By DOL 20, infant was transitioned to nasal cannula, weaned off vasopressors and resumed on enteral feeds. The infant completed 2 days of acyclovir, 7 days of amphotericin and 14 days of antibiotics. On DOL 25, infant developed acute absence of spontaneous movements, reflexes and pain responses in lower extremities with decreased rectal tone and anal wink. Primitive reflexes and cranial nerve functions were preserved. Contrast-enhanced brain MRI and venography demonstrated bilateral CSVT of transverse sinuses to
1 Division of Neonatology, Department of Pediatrics, Johns Hopkins University School of Medicine, Baltimore, MD, USA; 2Department of Pediatric Neuroradiology, Johns Hopkins University School of Medicine, Baltimore, MD, USA; 3Neurosciences Intensive Care Nursery, Johns Hopkins Hospital, Baltimore, MD, USA and 4Department of Neurology, Pediatric Stroke Program Johns Hopkins University School of Medicine, Baltimore, MD, USA. Correspondence: Dr R Chavez-Valdez, Division of Neonatology, Department of Pediatrics, Johns Hopkins University School of Medicine, 600 North Wolfe St CMSC 6-104, Baltimore, MD 21287, USA. E-mail: [email protected] Received 4 October 2014; revised 29 December 2014; accepted 10 February 2015
Paraplegia in a preterm infant J Hobbs et al
461 the torcula and hemorrhagic infarcts in thalami and supra/infratentorial white matter (Figure 1). Additionally, MRI revealed mild increase in T2 signal and focal area of leptomeningeal contrast enhancement in the conus medullaris (Figure 2). At second review, thalamic echogenicity in HUS on DOL 17 may be consistent with ischemia and hemorrhage. A prothrombotic state was proposed based on imaging and history of pre-eclampsia and prematurity. After extensive discussion with the hematology service and due to the severity of presentation, heparin anticoagulation treatment was started. Protein-C, protein-S and factor VIII activities, antithrombin III levels, activated protein-C resistance, and anti-phospholipid antibody panel and prothrombin G20210A mutation were negative or within known normal values. Serial HUSs showed no further bleeding while receiving anticoagulation. On DOL 88, the infant was discharged home, on oxygen via nasal cannula, medications for chronic lung disease and gastroesophageal reflux and enoxaparin. The infant had persistent flaccid paralysis of the lower extremities and no plantar grasp or response to touch at time of discharge.
Figure 1. First row: axial T2-weighted image through the posterior fossa with corresponding post contrast T1-weighted image. Note the thick, curvilinear dark T2 signal in right transverse sinus extending into the torcula representing thrombus. Post contrast T1-weighted image demonstrates enhancement of the thrombus. In addition, note areas of dark T2 signal in the cerebellar parenchyma with corresponding enhancement, representing venous hemorrhagic ischemic injury. Second row: axial T2-weighted image through the basal ganglia and lateral ventricles with corresponding post contrast T1-weighted image. Note the dark T2 signal in the posterior aspect of the basal ganglia and thalami representing thrombus. Contrast enhancement of the thrombus in the basal ganglia and thalami are noted. In addition note linear areas of enhancement in the periventricular region representing enhancing thrombus in the intramedullary veins.
DISCUSSION Our patient shares risk factors to those described in CSVT case reports. However, the delayed presentation and spinal cord injury are unusual. With the concomitant CSVT, we speculated that an inherent or transient prothrombotic state may have also caused a venous thrombosis explaining the conus medullaris injury. Although not specific, the increased T2 signal along with leptomeningeal enhancement in the conus was attributed to venous infarction. Standard HUS technique misses 63% of CSVT cases,7 but addition of color Doppler imaging through the mastoid window improves sensitivity for transverse lateral sinuses.3 However, subtle changes in a HUS (DOL 17) were not obvious in this case until retrospective review, delaying further testing and therapy. Computed tomography for CSVT detection has also suboptimal sensitivity and high ionizing radiation exposure.2,10 Hence, MRI has the highest resolution to detect thrombus and associated paren-
Figure 2. Sagittal T2 weighted demonstrates minimal increased T2 signal in the central aspect of the conus medullaris. Sagittal post contrast T1-weighted image demonstrates contrast enhancement in the posterior aspect of the conus medullaris. Axial T2-weighted image (top) shows normal signal intensity slightly above the conus medullaris. Axial T2-weighted image (below) shows increased central T2 signal at the conus medullaris. Note lack of intradural mass or extra-axial collection that rules out cord compression. © 2015 Nature America, Inc.
Journal of Perinatology (2015), 460 – 462
Paraplegia in a preterm infant J Hobbs et al
462 chymal lesions and is the most suitable modality for neonatal CSVT detection. Neonatal CSVT has been linked to protein-C, protein-S and antithrombin III deficiencies and methylenetetrahydrofolate reductase (MTHFR), factor-V Leiden and PAI-1 mutations.11 However, the value of any early prothrombotic workup is controversial in preterm neonates due to the lack of reference values, underlining the need for delayed additional testing. Thus, a prothrombotic disorder cannot be fully ruled out in our patient even after extensive early hematology studies. Even more controversial is the use, duration and dosing of anticoagulation for neonatal CSVT, in the setting of unconfirmed prothrombotic disorder.2,3 The little available evidence suggests that withholding anticoagulation increases the risk of thrombus propagation,2 whereas anticoagulation therapy does not increase new bleeding. In the Canadian Pediatric Ischemic Stroke Registry (CPISR), no infant died or showed deterioration from bleeding while receiving anticoagulation.12 The American College of Chest Physicians recommends unfractionated heparin or low-molecular weight heparin (LMWH) until anticoagulation is achieved, followed by LMWH or vitamin K antagonists for 6 to 12 weeks in neonates with CSVT without significant intracranial hemorrhage. For infants with significant hemorrhage, anticoagulation is recommended if thrombus extension occurs by 5 to 7 days after diagnosis.2,3,12 The CPISR reports of those neonates with CSVT: (i) 40 to 80% have poor neurological outcome; (ii) 30 to 80% have associated intracranial hemorrhage; (iii) 30 to 50% receive anticoagulation therapy; and (iv) 0 to 8% develop anticoagulant-related bleeding.4 Contrast-enhanced MRI cannot rule out spinal cord infarction, a rare entity not linked to venous thrombosis. Although our patient never had an umbilical arterial catheter, the most important coexisting circumstantial risk factor, other risk factors are unknown. In the setting of an acute hypoxic–ischemic event during cardiorespiratory arrest, we speculate that the absence or failure of spinal cord blood flow autoregulation secondary to systemic hypotension and prematurity may explain the conus medullaris ischemic injury in our patient.13,14 Our patient shared risk factors reported in other CSVT case studies; however, the late presentation, persistent paraplegia, and conus medullaris injury have never been reported with CSVT. Our case shows the value of entire neuroaxis MRI to document spinal cord injury.
Journal of Perinatology (2015), 460 – 462
CONFLICT OF INTEREST The authors declare no conflict of interest.
ACKNOWLEDGMENTS We thank Drs Frances J Northington, Adam Hartman, Thierry AGM Huisman and the Neuroscience Intensive care Nursery Program group at Johns Hopkins University for their support. Support has been provided by Sheila S and Lawrence C Pakula, M.D. Endowment for Neonatal Research.
REFERENCES 1 Dlamini N, Billinghurst L, Kirkham FJ. Cerebral venous sinus (sinovenous) thrombosis in children. Neurosurg Clin N Am 2010; 21(3): 511–527. 2 van der Aa N, Benders M, Groenendaal F, de Vries L. Neonatal stroke: a review of the current evidence on epidemiology, pathogenesis, diagnostics and therapeutic options. Acta Paediatr 2014; 103(4): 356–364. 3 Raets MM, Sol JJ, Govaert P, Lequin MH, Reiss IK, Kroon AA et al. Serial cranial US for detection of cerebral sinovenous thrombosis in preterm infants. Radiology 2013; 269(3): 879–886. 4 Moharir MD, Shroff M, Pontigon AM, Askalan R, Yau I, Macgregor D et al. A prospective outcome study of neonatal cerebral sinovenous thrombosis. J Child Neurol 2011; 26(9): 1137–1144. 5 Hedlund GL. Cerebral sinovenous thrombosis in pediatric practice. Pediatr Radiol 2013; 43(2): 173–188. 6 Khurana DS, Buonanno F, Ebb D, Krishnamoorthy KS. The role of anticoagulation in idiopathic cerebral venous thrombosis. J Child Neurol 1996; 11(3): 248–250. 7 Berfelo FJ, Kersbergen KJ, van Ommen CH, Govaert P, van Straaten HL, Poll-The BT et al. Neonatal cerebral sinovenous thrombosis from symptom to outcome. Stroke 2010; 41(7): 1382–1388. 8 deVeber G, Andrew M, Adams C, Bjornson B, Booth F, Buckley DJ et al. Cerebral sinovenous thrombosis in children. N Engl J Med 2001; 345(6): 417–423. 9 Goldenberg NA. Long-term outcomes of venous thrombosis in children. Curr Opin Hematol 2005; 12(5): 370–376. 10 Kersbergen KJ, Groenendaal F, Benders MJ, de Vries LS. Neonatal cerebral sinovenous thrombosis: neuroimaging and long-term follow-up. J Child Neurol 2011; 26(9): 1111–1120. 11 Golomb MR. The contribution of prothrombotic disorders to peri- and neonatal ischemic stroke. Semin Thromb Hemost 2003; 29(4): 415–424. 12 Cnossen MH, van Ommen CH, Appel IM. Etiology and treatment of perinatal stroke; a role for prothrombotic coagulation factors? Semin Fetal Neonatal Med 2009; 14(5): 311–317. 13 Singer R, Joseph K, Gilai AN, Meyer S. Nontraumatic, acute neonatal paraplegia. J Pediatr Orthop 1991; 11(5): 588–593. 14 Sladky JT, Rorke LB. Perinatal hypoxic/ischemic spinal cord injury. Pediatr Pathol 1986; 6(1): 87–101.
© 2015 Nature America, Inc.
PERINATAL/NEONATAL CASE PRESENTATION
Acute paraplegia in a preterm infant with cerebral sinovenous thrombosis J Hobbs1, A Tekes2,3, J Klein3,4, M Lemmon3,4, RJ Felling3,4 and R Chavez-Valdez1,3 We report the case of a 1-month old, 28-week gestational age infant who presented with acute paraplegia after cardiopulmonary arrest. Later imaging confirms cerebral sinovenous thrombosis (CSVT) and a suspected infarction in the conus medullaris of the spinal cord. A prothrombotic state may explain the numerous areas of infarction visualized on neuroimaging. To our knowledge this is the first case report of acute and persistent paraplegia in an infant with CSVT and conus medullaris injury, which may be due to venous infarction of the spinal cord. Journal of Perinatology (2015) 35, 460–462. doi:10.1038/jp.2015.26
INTRODUCTION Cerebral sinovenous thrombosis (CSVT) is an uncommon, but increasingly recognized cause of neonatal stroke.1 Depending on the neuroimaging modality, the reported incidence of neonatal CSVT varies from 0.6 to 12 for every 100 000 live births.2 In premature infants, CSVT incidence has been reported as high as 4.4% using head ultrasound (HUS) with Doppler scan through the mastoid fontanelle confirmed with magnetic resonance imaging (MRI).3 The superior sagittal and transverse lateral sinuses are the most common CSVT sites.4,5 CSVT accompanied by spinal cord injury has never been reported in premature neonates. Most neonates with CSVT present within 48 h with seizures, irritability and hypotonia, and less often with apnea, motor weakness and cranial nerve palsies.5–8 The spectrum of CSVT presentation in neonates ranges from asymptomatic (13%)2,7 to deathly (6 to 25%).2,4,5,9 Pre-eclampsia, premature rupture of membranes and neonatal dehydration, infection, polycythemia, trauma, hypoxic ischemic injury and prothrombotic states are the main risk factors for neonatal CSVT.2,4,5 We report a case of a 25-day-old premature infant with acute paraplegia following cardiorespiratory arrest at day of life (DOL) 13 and diagnosed with cerebral, cerebellar and spinal cord hemorrhagic infarcts associated with extensive CSVT. CASE This 850-g extreme low birth weight infant was born via cesarean section at 28 2/7 weeks secondary to chronic hypertension with superimposed pre-eclampsia. Prenatal laboratories were unremarkable. Rupture of membranes occurred at the time of delivery. Apgar scores were 8 at both 1 and 5 min. Supplemental oxygen was provided at 5 min of life for oxygen saturations below Neonatal Resuscitation Program guidelines. The infant received antibiotics until DOL 2 and minimal invasive ventilator support until DOL 9. The infant was initiated on total parenteral nutrition and caffeine citrate on DOL 1, and trophic
feeds on DOL 3. On DOL 9, soon after endotracheal tube removal, patient developed increased events of apnea, bradycardia, oxygen desaturations and feeding intolerance with no hemodynamic instability. Management included nothing per os (NPO), vancomycin and gentamicin and adjustment on high-flow nasal cannula support. All abdominal radiographs were negative for pneumatosis intestinalis or peritoneal free air. Blood work results did not support infection. Trophic feeds were resumed by DOL 13. At the end of DOL 13, the infant had a cardiopulmonary arrest receiving positive pressure ventilation, chest compressions and intravenous epinephrine. The treating team obtained: bacterial and viral cultures of blood, urine and cerebrospinal fluid (CSF), amplitude integrated electroencephalogram (aEEG), HUS and twodimensional echocardiogram. The infant was transitioned to high-frequency oscillatory ventilation, initiated on amphotericin, acyclovir, vancomycin and cefotaxime, and treated with fluid boluses and inotropes for systemic hypotension. HUSs on DOL 3, 13 (day of cardiorespiratory event) and 17 were reported as within normal limits. Echocardiogram on DOL 13 showed mild pulmonary hypertension but normal biventricular function and no evidence of patent ductus arteriosus. Electrocardiogram tracing showed sinus rhythm before and after cardiorespiratory event. aEEG showed no evidence of seizure activity or abnormal background for 72 h after the event. CSF studies were within normal limits and all cultures were negative. The infant’s neurologic exam on DOL 13 demonstrated symmetric spontaneous movement, and normal tone, deep tendon and primitive reflexes after the event. By DOL 20, infant was transitioned to nasal cannula, weaned off vasopressors and resumed on enteral feeds. The infant completed 2 days of acyclovir, 7 days of amphotericin and 14 days of antibiotics. On DOL 25, infant developed acute absence of spontaneous movements, reflexes and pain responses in lower extremities with decreased rectal tone and anal wink. Primitive reflexes and cranial nerve functions were preserved. Contrast-enhanced brain MRI and venography demonstrated bilateral CSVT of transverse sinuses to
1 Division of Neonatology, Department of Pediatrics, Johns Hopkins University School of Medicine, Baltimore, MD, USA; 2Department of Pediatric Neuroradiology, Johns Hopkins University School of Medicine, Baltimore, MD, USA; 3Neurosciences Intensive Care Nursery, Johns Hopkins Hospital, Baltimore, MD, USA and 4Department of Neurology, Pediatric Stroke Program Johns Hopkins University School of Medicine, Baltimore, MD, USA. Correspondence: Dr R Chavez-Valdez, Division of Neonatology, Department of Pediatrics, Johns Hopkins University School of Medicine, 600 North Wolfe St CMSC 6-104, Baltimore, MD 21287, USA. E-mail: [email protected] Received 4 October 2014; revised 29 December 2014; accepted 10 February 2015
Paraplegia in a preterm infant J Hobbs et al
461 the torcula and hemorrhagic infarcts in thalami and supra/infratentorial white matter (Figure 1). Additionally, MRI revealed mild increase in T2 signal and focal area of leptomeningeal contrast enhancement in the conus medullaris (Figure 2). At second review, thalamic echogenicity in HUS on DOL 17 may be consistent with ischemia and hemorrhage. A prothrombotic state was proposed based on imaging and history of pre-eclampsia and prematurity. After extensive discussion with the hematology service and due to the severity of presentation, heparin anticoagulation treatment was started. Protein-C, protein-S and factor VIII activities, antithrombin III levels, activated protein-C resistance, and anti-phospholipid antibody panel and prothrombin G20210A mutation were negative or within known normal values. Serial HUSs showed no further bleeding while receiving anticoagulation. On DOL 88, the infant was discharged home, on oxygen via nasal cannula, medications for chronic lung disease and gastroesophageal reflux and enoxaparin. The infant had persistent flaccid paralysis of the lower extremities and no plantar grasp or response to touch at time of discharge.
Figure 1. First row: axial T2-weighted image through the posterior fossa with corresponding post contrast T1-weighted image. Note the thick, curvilinear dark T2 signal in right transverse sinus extending into the torcula representing thrombus. Post contrast T1-weighted image demonstrates enhancement of the thrombus. In addition, note areas of dark T2 signal in the cerebellar parenchyma with corresponding enhancement, representing venous hemorrhagic ischemic injury. Second row: axial T2-weighted image through the basal ganglia and lateral ventricles with corresponding post contrast T1-weighted image. Note the dark T2 signal in the posterior aspect of the basal ganglia and thalami representing thrombus. Contrast enhancement of the thrombus in the basal ganglia and thalami are noted. In addition note linear areas of enhancement in the periventricular region representing enhancing thrombus in the intramedullary veins.
DISCUSSION Our patient shares risk factors to those described in CSVT case reports. However, the delayed presentation and spinal cord injury are unusual. With the concomitant CSVT, we speculated that an inherent or transient prothrombotic state may have also caused a venous thrombosis explaining the conus medullaris injury. Although not specific, the increased T2 signal along with leptomeningeal enhancement in the conus was attributed to venous infarction. Standard HUS technique misses 63% of CSVT cases,7 but addition of color Doppler imaging through the mastoid window improves sensitivity for transverse lateral sinuses.3 However, subtle changes in a HUS (DOL 17) were not obvious in this case until retrospective review, delaying further testing and therapy. Computed tomography for CSVT detection has also suboptimal sensitivity and high ionizing radiation exposure.2,10 Hence, MRI has the highest resolution to detect thrombus and associated paren-
Figure 2. Sagittal T2 weighted demonstrates minimal increased T2 signal in the central aspect of the conus medullaris. Sagittal post contrast T1-weighted image demonstrates contrast enhancement in the posterior aspect of the conus medullaris. Axial T2-weighted image (top) shows normal signal intensity slightly above the conus medullaris. Axial T2-weighted image (below) shows increased central T2 signal at the conus medullaris. Note lack of intradural mass or extra-axial collection that rules out cord compression. © 2015 Nature America, Inc.
Journal of Perinatology (2015), 460 – 462
Paraplegia in a preterm infant J Hobbs et al
462 chymal lesions and is the most suitable modality for neonatal CSVT detection. Neonatal CSVT has been linked to protein-C, protein-S and antithrombin III deficiencies and methylenetetrahydrofolate reductase (MTHFR), factor-V Leiden and PAI-1 mutations.11 However, the value of any early prothrombotic workup is controversial in preterm neonates due to the lack of reference values, underlining the need for delayed additional testing. Thus, a prothrombotic disorder cannot be fully ruled out in our patient even after extensive early hematology studies. Even more controversial is the use, duration and dosing of anticoagulation for neonatal CSVT, in the setting of unconfirmed prothrombotic disorder.2,3 The little available evidence suggests that withholding anticoagulation increases the risk of thrombus propagation,2 whereas anticoagulation therapy does not increase new bleeding. In the Canadian Pediatric Ischemic Stroke Registry (CPISR), no infant died or showed deterioration from bleeding while receiving anticoagulation.12 The American College of Chest Physicians recommends unfractionated heparin or low-molecular weight heparin (LMWH) until anticoagulation is achieved, followed by LMWH or vitamin K antagonists for 6 to 12 weeks in neonates with CSVT without significant intracranial hemorrhage. For infants with significant hemorrhage, anticoagulation is recommended if thrombus extension occurs by 5 to 7 days after diagnosis.2,3,12 The CPISR reports of those neonates with CSVT: (i) 40 to 80% have poor neurological outcome; (ii) 30 to 80% have associated intracranial hemorrhage; (iii) 30 to 50% receive anticoagulation therapy; and (iv) 0 to 8% develop anticoagulant-related bleeding.4 Contrast-enhanced MRI cannot rule out spinal cord infarction, a rare entity not linked to venous thrombosis. Although our patient never had an umbilical arterial catheter, the most important coexisting circumstantial risk factor, other risk factors are unknown. In the setting of an acute hypoxic–ischemic event during cardiorespiratory arrest, we speculate that the absence or failure of spinal cord blood flow autoregulation secondary to systemic hypotension and prematurity may explain the conus medullaris ischemic injury in our patient.13,14 Our patient shared risk factors reported in other CSVT case studies; however, the late presentation, persistent paraplegia, and conus medullaris injury have never been reported with CSVT. Our case shows the value of entire neuroaxis MRI to document spinal cord injury.
Journal of Perinatology (2015), 460 – 462
CONFLICT OF INTEREST The authors declare no conflict of interest.
ACKNOWLEDGMENTS We thank Drs Frances J Northington, Adam Hartman, Thierry AGM Huisman and the Neuroscience Intensive care Nursery Program group at Johns Hopkins University for their support. Support has been provided by Sheila S and Lawrence C Pakula, M.D. Endowment for Neonatal Research.
REFERENCES 1 Dlamini N, Billinghurst L, Kirkham FJ. Cerebral venous sinus (sinovenous) thrombosis in children. Neurosurg Clin N Am 2010; 21(3): 511–527. 2 van der Aa N, Benders M, Groenendaal F, de Vries L. Neonatal stroke: a review of the current evidence on epidemiology, pathogenesis, diagnostics and therapeutic options. Acta Paediatr 2014; 103(4): 356–364. 3 Raets MM, Sol JJ, Govaert P, Lequin MH, Reiss IK, Kroon AA et al. Serial cranial US for detection of cerebral sinovenous thrombosis in preterm infants. Radiology 2013; 269(3): 879–886. 4 Moharir MD, Shroff M, Pontigon AM, Askalan R, Yau I, Macgregor D et al. A prospective outcome study of neonatal cerebral sinovenous thrombosis. J Child Neurol 2011; 26(9): 1137–1144. 5 Hedlund GL. Cerebral sinovenous thrombosis in pediatric practice. Pediatr Radiol 2013; 43(2): 173–188. 6 Khurana DS, Buonanno F, Ebb D, Krishnamoorthy KS. The role of anticoagulation in idiopathic cerebral venous thrombosis. J Child Neurol 1996; 11(3): 248–250. 7 Berfelo FJ, Kersbergen KJ, van Ommen CH, Govaert P, van Straaten HL, Poll-The BT et al. Neonatal cerebral sinovenous thrombosis from symptom to outcome. Stroke 2010; 41(7): 1382–1388. 8 deVeber G, Andrew M, Adams C, Bjornson B, Booth F, Buckley DJ et al. Cerebral sinovenous thrombosis in children. N Engl J Med 2001; 345(6): 417–423. 9 Goldenberg NA. Long-term outcomes of venous thrombosis in children. Curr Opin Hematol 2005; 12(5): 370–376. 10 Kersbergen KJ, Groenendaal F, Benders MJ, de Vries LS. Neonatal cerebral sinovenous thrombosis: neuroimaging and long-term follow-up. J Child Neurol 2011; 26(9): 1111–1120. 11 Golomb MR. The contribution of prothrombotic disorders to peri- and neonatal ischemic stroke. Semin Thromb Hemost 2003; 29(4): 415–424. 12 Cnossen MH, van Ommen CH, Appel IM. Etiology and treatment of perinatal stroke; a role for prothrombotic coagulation factors? Semin Fetal Neonatal Med 2009; 14(5): 311–317. 13 Singer R, Joseph K, Gilai AN, Meyer S. Nontraumatic, acute neonatal paraplegia. J Pediatr Orthop 1991; 11(5): 588–593. 14 Sladky JT, Rorke LB. Perinatal hypoxic/ischemic spinal cord injury. Pediatr Pathol 1986; 6(1): 87–101.
© 2015 Nature America, Inc.
