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Pediatric otogenic lateral sinus thrombosis recanalization
International Journal of Pediatric Otorhinolaryngology 75 (2011) 850–853
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Pediatric otogenic lateral sinus thrombosis recanalization§ Ryan E. Neilan, Brandon Isaacson *, J. Walter Kutz Jr., Kenneth H. Lee, Peter S. Roland Department of Otolaryngology-Head and Neck Surgery, University of Texas Southwestern Medical Center, 5323 Harry Hines Boulevard, Dallas, TX 75390-9035, United States
A R T I C L E I N F O
A B S T R A C T
Article history: Received 9 January 2011 Received in revised form 23 March 2011 Accepted 24 March 2011 Available online 22 April 2011
Objective: To describe the recovery outcomes in pediatric patients with otogenic lateral sinus and internal jugular vein thrombosis. Methods: An inpatient database from a tertiary care pediatric hospital was queried from 1999 to 2010 for the diagnosis code [325] thrombosis of intracranial venous sinus. Demographics, extent of thrombosis, surgical intervention, use of anticoagulation, and the presence of recanalization on follow-up imaging was collected. Results: Fifteen patients (10 male, 5 female) were identified with otogenic lateral sinus thrombosis. Eleven patients (73.3%) had evidence of thrombus in the transverse sinus, while 10 patients (66.7%) had thrombus in the internal jugular vein, and one patient (6.7%) had thrombus in the cavernous sinus. Five patients (33%) had otitic hydrocephalus. Twelve patients (80%) underwent operative manipulation of the lateral sinus including: three decompressions 20%, three needle aspirations 20%, and six venotomies with evacuation of clot or pus 40%. Twelve patients were anticoagulated with low molecular weight heparin, and three patients were not anticoagulated. Ten of fourteen patients (71.4%) who underwent follow-up magnetic resonance venography had evidence of partial (57.1%) or complete (14.3%) recanalization of the lateral sinus. All five patients with otitic hydrocephalus recovered as determined by a normal fundoscopic exam and recovery of abducens paresis. Conclusion: Recanalization of the lateral intracranial venous sinus occurred in the majority of the patients in this series. The role of operative intervention and/or anticoagulation remains unclear. ß 2011 Elsevier Ireland Ltd. All rights reserved.
Keywords: Lateral sinus thrombosis Otitis media Otitic hydrocephalus
1. Introduction Lateral sinus thrombosis is the second most common intracranial complication of otitis media after intracranial abscesses [1]. In the pre-antibiotic era, complications of otitis media such as lateral sinus thrombosis commonly led to serious neurologic sequelae and septic emboli. Infections in the mastoid can result in dural venous thrombosis by several mechanisms. The close proximity of the sigmoid sinus allows adjacent inflammation to activate platelets and fibrin resulting in a mural thrombus [2]. Such thrombi may then propagate to the adjacent dural venous sinuses (transverse, inferior petrosal, superior petrosal), or the internal jugular vein. Dural venous sinus thrombosis may result in reduced resorption of cerebrospinal fluid (CSF) from arachnoid granulations. Reduced resorption of CSF then results in increased intracranial pressure (ICP), which is known as otitic hydrocephalus. Patients with otogenic sigmoid sinus thrombosis typically present with
§ Oral presentation: American Academy of Otolaryngology-Head and Neck Surgery Annual Meeting, 26–29 September 2010, Boston, MA. * Corresponding author. Tel.: +1 214 648 3437; fax: +1 214 648 9122. E-mail address: [email protected] (B. Isaacson).
0165-5876/$ – see front matter ß 2011 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2011.03.024
high spiking fevers, otorrhea, and post auricular tenderness Griesinger’s sign [3]. With the introduction of antibiotics as the primary treatment for otitis media this ‘‘classic’’ presentation has become less common and replaced with symptoms and findings such as headache, nausea and vomiting, low grade fevers, and signs of increased ICP [4]. The diagnosis of sigmoid sinus thrombosis has been aided by the use of non invasive imaging, most notably MRI/ MRV or less reliably CT [5]. According to a study by Proctor in 1966, the use of antibiotics in conjunction with surgical intervention has decreased the mortality from 40% to essentially 0 [6]. The use of anticoagulation in the setting of otogenic lateral sinus thrombosis remains controversial [7,8]. The objective of this retrospective study is to examine treatment outcomes in pediatric patients with lateral sinus thrombosis including sinus recanalization and resolution of otitic hydrocephalus. 2. Methods A retrospective chart review was performed at a tertiary children’s hospital in a major metropolitan area. All inpatient admissions from 1999 to 2010 were queried for the ICD-9 code [325] thrombosis of intracranial venous sinus. Ninety-three patients were initially identified. Included in our series were patients that were found to have thrombosis documented on MRI and from an otogenic
R.E. Neilan et al. / International Journal of Pediatric Otorhinolaryngology 75 (2011) 850–853
source. All subjects with otogenic sigmoid sinus thrombosis were preceded by and temporally related to an episode of acute otitis media. Those with thrombosis from aseptic sources such as stroke and trauma were excluded. Careful chart review found 15 patients of which the thrombosis was of otogenic origin. Information on age, gender, length of hospital stay, type of sigmoid sinus treatment, overall surgical treatment, whether purulence was encountered, presence of otitic hydrocephalus, venous sinuses involved, use of anticoagulation, length of anticoagulation treatment if rendered, state of sinus on follow up MRI, the interval between diagnosis and follow up MRI, and any other complications from the disease process were recorded. Exclusion criteria included any patients older than 18 years of age and medical records that were incomplete or of questionable accuracy. Trace flow detected in at least one of the following was classified as partial recanalization: sigmoid sinus, transverse sinus, or internal jugular vein. Normal flow through all thrombosed venous segments was classified as complete recanalization. This study was approved by the Institutional Review Board at the University of Texas Southwestern Medical Center. 3. Results Otogenic lateral sinus thrombosis was diagnosed in 10 male patients and 5 female patients in study period. The age of subjects ranged from 6 months to 14 years of age with a mean of 4.87 years. The mean length of symptoms prior to presentation was 8.6 days. Length of hospital stay ranged from 4 days to 48 days with a mean of 17 days. Thrombi frequently extended from the sigmoid sinus into the transverse sinus (11 patients, 73.3%), internal jugular vein (10 patients, 66.7%) and cavernous sinus (1 patient, 6.7%) (Table 1). All 15 patients underwent a mastoidectomy and tympanostomy tube placement. Mastoid purulence was encountered in 80% of these patients. The sigmoid sinus was addressed by needle decompression in 3 patients (20%), osseous decompression of the sigmoid plate in 3 patients (20%), and venotomy in 6 patients (40%). Venotomy consisted of incising the vein and draining any purulence and evacuating some clot, but not to the point of bleeding. No packing or vein ligation was performed. Two of the six patients undergoing venotomy were found to have intraluminal pus. In 3 patients (20%) the sigmoid sinus was
851
not manipulated or exposed (simple mastoidectomy only). Management of the thrombosed sigmoid sinus varied according to surgeon’s discretion. An algorithm for the operative management of the sigmoid sinus did not exist secondary to the retrospective nature of the study. Cholesteatoma was identified in one patient (6%). Intra operative cultures grew out Group A Streptococcus in four patients (27%), Streptococcus pneumoniae in three patients (20%), methicillin resistant Staphylococcus aureus in one patient (6.5%), and Streptococcus intermedius in one patient (6.5%). Six of the cultures demonstrated no growth (40%). The effects of culture results on outcomes was not assessed due to the limited number of cases. Otitic hydrocephalus was identified in five patients (33%) all of whom presented with diplopia from a 6th cranial nerve palsy. All five patients with otitic hydrocephalus eventually recovered as evidenced by resolution of their 6th nerve palsy and/or papilledema on ophthalmologic examination. Twelve patients (85%) were treated with low molecular weight heparin postoperatively, and 3 (15%) were not anticoagulated. The mean duration of anticoagulation was 3.4 months. Fourteen of the fifteen patients had follow up MRI examinations, one patient did not undergo MRI, and no patients were lost to follow up. The mean time interval from surgical treatment to when the last MRI was completed was 5.7 months with a range from 1 to 26 months. In the ten patients who demonstrated some degree of recanalization the mean length of time was 6.2 months with a range from 1 to 26 months. Persistent total occlusion of the sigmoid sinus was identified in 4 (28.6%) patients, while complete recanalization was noted in 2 patients (14.3%), and partial recanalization was found in 8 patients (57.1%) (Figs. 1 and 2). Five of eight patients with partial recanalization showed flow signal in all previously occluded segments restoring venous continuity. Three of eight patients had partial recanalization which did not have restoration of venous continuity through all previously involved segments. Seven of fourteen (50%) patients with follow imaging demonstrated restoration of venous continuity whether recanalization was found to be partial or complete. Eleven of the 14 (79%) patients that had follow-up MRIs were anticoagulated, some degree of recanalization was seen in eight patients (73%) with three patients (27%) still demonstrating no flow. Of the three (21%) which were not anticoagulated two (66.6%)
Table 1 Patient demographic, treatment, and outcome data. Patient
Age (years)
Length of stay (days)
Side
Sigmoid sinus treatment
Otitic hydrocephalus
Location of thrombus
Anti coagulation
Length of treatment
F/U MRI
MRI interval (months)
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15
9 6 6 0.67 3 2 1.5 3 6 5 12 14 0.5 1.5 3
48 7 11 10 5 7 20 42 12 4 17 13 8 44 8
Right Left Right Left Left Left Left Right Right Right Left Right Left Left Right
Needle Decomp Venotomy Venotomy Venotomy Venotomy Venotomy Needle Decomp None None None Venotomy Needle Decomp
Yes, No No No No No Yes, Yes, No No No Yes, Yes, No No
SS, SS SS, SS, SS SS, SS, SS, SS, SS, SS, SS, SS, SS, SS,
LMWH LMWH LMWH LMWH None LMWH LMWH LMWH LMWH None None LMWH LMWH LMWH LMWH
3 months 6 weeks 3 months 4 months N/A 3 months 3 months 6 months 3 months N/A N/A 3 months 6 months 8 months 3 months
No Flow Complete Partial Partial Complete Partial Partial Partial No MRI No Flow Partial Partial No Flow Partial No Flow
2 26 1 3 4 3 2 1 N/A 2 7 5 12 10 2
LRP, Right
LRP, Right LRP, Bilateral
LRP, Left LRP, Bilateral
TS, IJ TS, IJ TS, IJ TS, IJ IJ TS TS, IJ TS TS, IJ TS, IJ TS Cav, IJ TS, IJ
LOS: length of stay; needle: the osseous sigmoid plate was removed and the sinus was needle aspirated; decomp (decompressed): the osseous sigmoid plate was removed with removal of granulation tissue and pus, but the sinus was not opened; venotomy: the osseous sigmoid plate was removed and the sinus was opened and clot and purulence were evacuated; none: no manipulation of the sinus; LRP: lateral rectus palsy; SS: sigmoid sinus; TS: transverse sinus; IJ: internal jugular vein; Cav: cavernous sinus; no flow: no flow was identified on the follow up MRV; partial: recanalization with a more narrow caliber lumen involving at least one or all of the thrombosed segments of the lateral sinus; complete: complete recanalization to normal caliber.
[()TD$FIG]
852
R.E. Neilan et al. / International Journal of Pediatric Otorhinolaryngology 75 (2011) 850–853
heparin, developed a treatment related small surgical site hematoma, which was incised and evacuated in clinic with local anesthesia. Patients developing additional complications did not differ in presentation from those subjects that did not. 4. Discussion
Fig. 1. Pre-treatment reconstructed coronal view of MRV demonstrating flow void in the left sigmoid sinus and internal jugular vein.
[()TD$FIG]
Fig. 2. Post-treatment reconstructed coronal MRV of same patient showing recanalization through previously thrombosed segments.
demonstrated recanalization with one (33.3%) demonstrating persistent thrombosis. Of the two patients who demonstrated complete recanalization; one received anticoagulation and the other did not. Two patients developed septic hip joints during the course of their illness presumably from septic emboli. There were no mortalities in this series of fifteen patients. One patient, whose blood and wound cultures grew out Fusobacterium necrophorum, developed a cerebral hemispheric ischemic stroke from an ipsilateral internal carotid artery thrombosis. This patient was treated with low molecular weight heparin, and eventually regained use of his paretic lower extremity, but continues to have upper extremity paralysis and spasticity. One patient who was also administered low molecular weight heparin developed an endophthalmitis. One patient, treated with low molecular weight
Temporal bone surgery, modern imaging (i.e., MRI and CT), and antibiotics are largely responsible for the reduced incidence of intratemporal and intracranial complications of otitis media [1]. Despite these advances, complications of otitis media still occur, and if not recognized and treated in a timely fashion, may result in significant morbidity and mortality [9]. Otogenic lateral sinus thrombosis is an infrequent complication that is now rarely associated with mortality or permanent morbidity in areas with access to care [10,11]. Goldenberg found that 66% of the otolaryngology departments they surveyed had no episodes of sigmoid sinus thrombosis in the 5 years preceding the survey. The incidence of sigmoid sinus thrombosis from the 83 departments surveyed was found to be 0.625 cases per department for the 5 years preceding the survey [12]. Patients may present with a myriad of symptoms including headache, fever, nuchal rigidity, post auricular pain, and diplopia [4]. An ongoing or recent history of middle ear infection is usually elicited. Typical physical exam findings in patients with lateral sinus thrombosis include serous or purulent middle ear effusion, post auricular tenderness/edema, lateral rectus palsy, and papilledema [4,13,14]. Otitic hydrocephalus is not uncommon in the setting of dural venous sinus thrombosis and occurred in 5 out of the 15 (33.3%) patients in our series. A decrease in CSF resorption from arachnoid granulations secondary to thrombosis in a dominant lateral dural venous sinus is the purported pathophysiologic mechanism for otitic hydrocephalus [15]. In the present series, the 5 patients with otitic hydrocephalus presented with diplopia secondary to a 6th nerve palsy. One patient was noted to have papilledema, and another patient had an elevated opening pressure during a lumbar puncture. As in previous reports, complete resolution of otitic hydrocephalus was noted in all five patients even in the setting of persistent lateral sinus thrombosis. Recovery from otitic hydrocephalus is thought to be secondary to development of compensatory collateral venous drainage [4,15]. A number of questions remain with respect to the management of lateral sinus thrombosis including the need for and the extent of operative intervention, as well as the utility of anticoagulation. Management of lateral sinus thrombosis has included observation, pressure equalization tube placement, simple mastoidectomy, removal of the sigmoid plate with or without perisinus exploration, needle aspiration, and venotomy with partial or complete evacuation of thrombus, resection of the thrombosed sigmoid sinus, bulb and internal jugular vein, and internal jugular vein ligation [5,13,14,16–18]. Based on prior reports, it appears that manipulation of the lateral sinus from needle aspiration to resection have all been associated with symptom resolution in the vast majority of cases [4,5,18,19]. A definitive cause and effect relationship between lateral sinus operative intervention and symptom resolution has not been established. Recanalization of the lateral sinus has been demonstrated in patients undergoing non-operative medical management, perisinus exploration, needle aspiration, and venotomy with or without thrombectomy [4,10,19]. Resection of the involved sinus and or ligation of the internal jugular vein precludes recanalization [5]. Internal jugular vein ligation may be considered in the setting of septic emboli which is unresponsive to medical management. There is currently no evidence for or against the use of thrombectomy to reduce recovery time and increase the likelihood of recanalization. In agreement with previous studies, the present series demonstrated
R.E. Neilan et al. / International Journal of Pediatric Otorhinolaryngology 75 (2011) 850–853
no obvious correlation with sinus manipulation and recanalization [4,10,14,20]. A trend towards more conservative operative management was noted later in the time course of the present study. The data from eight studies of patients with lateral sinus thrombosis was reviewed and pooled for type of treatment, and recanalization rates. 45 out of 52 (90.4%) patients had a follow up MRI or ultrasound imaging to assess the patency of the affected sinuses [4,8,10,11,14,15,18,19]. Thirty six of fifty two (69.2%) patients were anticoagulated with either low molecular weight heparin, or a combination of heparin and coumadin. Twenty nine of forty seven (61.7%) patients with follow up imaging demonstrated partial or complete recanalization which approximates the incidence of partial or complete recanalization in the present study (71.4%). Anticoagulation is sometimes utilized in the setting of septic emboli, or propagation of thrombus to adjacent sinuses or the internal jugular vein [7]. A majority of the patients in the present series were anticoagulated with low molecular weight heparin as recommended by the pediatric hematology or neurology service. Patients with lateral sinus thrombosis at our institution are routinely treated with anticoagulation per recommendations by the pediatric hematology service. A number of studies have examined the pros and cons of anticoagulation for lateral sinus thrombosis, but due to the rarity of this condition insufficient evidence exists to support or reject its use. One patient in this series had a presumed anticoagulation related post surgical hematoma which resolved with incision and drainage. Forty-seven patients from eight studies in addition to the present study had follow up imaging data to determine the rate of recanalization with respect to the use of anticoagulation [4,8,10,11,14,15,18,19]. Twenty of 30 (66.7%) patients who were administered anticoagulation showed evidence of recanalization. Nine of seventeen (52.9%) patients who were not given anticoagulation showed evidence of recanalization. A Fisher Exact test showed no significant effect of anticoagulation on lateral sinus recanalization (p = 0.53). The lack of effect seen with anticoagulation on sinus recanalization may be related to the small sample size despite combining data from the present study with seven prior reports [4,8,10,11,15,16,19,20]. These previous reports demonstrate recanalization rates of 0–72% of patients who were anticoagulated and 0–100% of those in whom no anticoagulation was administered [4,8,11,15,16,19,20]. In the present study 79% of patients were anticoagulated resulting in 73% partial or complete recanalization. 5. Conclusion Lateral sinus thrombosis is a rare complication of acute and chronic otitis media. The appropriate management of this complication remains controversial with respect to the necessity and extent of surgical intervention and the use of anticoagulation. Mortality is rare from otogenic lateral sinus thrombosis, but
853
significant morbidity still occurs. In some cases, follow up imaging demonstrates partial or complete lateral sinus recanalization. How anticoagulation and surgical intervention affect recanalization remains unclear. Conflicts of interest Peter Roland, MD – Consultant for Alcon; Ryan Neilan MD – none; J. Walter Kutz Jr., MD – none; Kenneth H. Lee, MD, PhD – none; Brandon Isaacson, MD – Consultant and Course Instructor for Medtronic Midas Rex Institute. References [1] B. Isaacson, C. Mirabal, J.W. Kutz Jr., K.H. Lee, P.S. Roland, Pediatric otogenic intracranial abscesses, Otolaryngol. Head Neck Surg. 142 (2010) 434–437. [2] C. Bianchini, C. Aimoni, S. Ceruti, D.L. Grasso, A. Martini, Lateral sinus thrombosis as a complication of acute mastoiditis, Acta Otorhinolaryngol. Ital. 28 (2008) 30– 33. [3] R.F. Wetmore, Complications of otitis media, Pediatr. Ann. 29 (2000) 637–646. [4] C.B. Bales, S. Sobol, R. Wetmore, L.M. Elden, Lateral sinus thrombosis as a complication of otitis media: 10-year experience at the children’s hospital of Philadelphia, Pediatrics 123 (2009) 709–713. [5] S. Manolidis, J.W. Kutz Jr., Diagnosis and management of lateral sinus thrombosis, Otol. Neurotol. 26 (2005) 1045–1051. [6] C.A. Proctor, Intracranial complications of otitic origin, Laryngoscope 76 (1966) 288–308. [7] D.T. Bradley, G.T. Hashisaki, J.C. Mason, Otogenic sigmoid sinus thrombosis: what is the role of anticoagulation? Laryngoscope 112 (2002) 1726–1729. [8] U.K. Shah, T.F. Jubelirer, J.D. Fish, L.M. Elden, A caution regarding the use of lowmolecular weight heparin in pediatric otogenic lateral sinus thrombosis, Int. J. Pediatr. Otorhinolaryngol. 71 (2007) 347–351. [9] S.P. Dubey, V. Larawin, Complications of chronic suppurative otitis media and their management, Laryngoscope 117 (2007) 264–267. [10] N. Christensen, J. Wayman, J. Spencer, Lateral sinus thrombosis: a review of seven cases and proposal of a management algorithm, Int. J. Pediatr. Otorhinolaryngol. 73 (2009) 581–584. [11] N. de Oliveira Penido, J.R. Testa, D.P. Inoue, O.L. Cruz, Presentation, treatment, and clinical course of otogenic lateral sinus thrombosis, Acta Otolaryngol. 129 (2009) 729–734. [12] R.A. Goldenberg, Lateral sinus thrombosis. Medical or surgical treatment? Arch. Otolaryngol. 111 (1985) 56–58. [13] H. Seven, A.E. Ozbal, S. Turgut, Management of otogenic lateral sinus thrombosis, Am. J. Otolaryngol. 25 (2004) 329–333. [14] D.M. Kaplan, M. Kraus, M. Puterman, A. Niv, A. Leiberman, D.M. Fliss, Otogenic lateral sinus thrombosis in children, Int. J. Pediatr. Otorhinolaryngol. 49 (1999) 177–183. [15] J. Kuczkowski, M. Dubaniewicz-Wybieralska, T. Przewozny, W. Narozny, B. Mikaszewski, Otitic hydrocephalus associated with lateral sinus thrombosis and acute mastoiditis in children, Int. J. Pediatr. Otorhinolaryngol. 70 (2006) 1817–1823. [16] E.E. Tov, A. Leiberman, I. Shelef, D.M. Kaplan, Conservative nonsurgical treatment of a child with otogenic lateral sinus thrombosis, Am. J. Otolaryngol. 29 (2008) 138–141. [17] E.H. Ooi, M. Hilton, G. Hunter, Management of lateral sinus thrombosis: update and literature review, J. Laryngol. Otol. 117 (2003) 932–939. [18] A. Kutluhan, M. Kiris, V. Yurttas, A.F. Kiroglu, O. Unal, When can lateral sinus thrombosis be treated conservatively? J. Otolaryngol. 33 (2004) 107–110. [19] A. Agarwal, P. Lowry, G. Isaacson, Natural history of sigmoid sinus thrombosis, Ann. Otol. Rhinol. Laryngol. 112 (2003) 191–194. [20] M. Iseri, O. Aydin, E. Ustundag, G. Keskin, A. Almac, Management of lateral sinus thrombosis in chronic otitis media, Otol. Neurotol. 27 (2006) 1098–1103.
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Pediatric otogenic lateral sinus thrombosis recanalization§ Ryan E. Neilan, Brandon Isaacson *, J. Walter Kutz Jr., Kenneth H. Lee, Peter S. Roland Department of Otolaryngology-Head and Neck Surgery, University of Texas Southwestern Medical Center, 5323 Harry Hines Boulevard, Dallas, TX 75390-9035, United States
A R T I C L E I N F O
A B S T R A C T
Article history: Received 9 January 2011 Received in revised form 23 March 2011 Accepted 24 March 2011 Available online 22 April 2011
Objective: To describe the recovery outcomes in pediatric patients with otogenic lateral sinus and internal jugular vein thrombosis. Methods: An inpatient database from a tertiary care pediatric hospital was queried from 1999 to 2010 for the diagnosis code [325] thrombosis of intracranial venous sinus. Demographics, extent of thrombosis, surgical intervention, use of anticoagulation, and the presence of recanalization on follow-up imaging was collected. Results: Fifteen patients (10 male, 5 female) were identified with otogenic lateral sinus thrombosis. Eleven patients (73.3%) had evidence of thrombus in the transverse sinus, while 10 patients (66.7%) had thrombus in the internal jugular vein, and one patient (6.7%) had thrombus in the cavernous sinus. Five patients (33%) had otitic hydrocephalus. Twelve patients (80%) underwent operative manipulation of the lateral sinus including: three decompressions 20%, three needle aspirations 20%, and six venotomies with evacuation of clot or pus 40%. Twelve patients were anticoagulated with low molecular weight heparin, and three patients were not anticoagulated. Ten of fourteen patients (71.4%) who underwent follow-up magnetic resonance venography had evidence of partial (57.1%) or complete (14.3%) recanalization of the lateral sinus. All five patients with otitic hydrocephalus recovered as determined by a normal fundoscopic exam and recovery of abducens paresis. Conclusion: Recanalization of the lateral intracranial venous sinus occurred in the majority of the patients in this series. The role of operative intervention and/or anticoagulation remains unclear. ß 2011 Elsevier Ireland Ltd. All rights reserved.
Keywords: Lateral sinus thrombosis Otitis media Otitic hydrocephalus
1. Introduction Lateral sinus thrombosis is the second most common intracranial complication of otitis media after intracranial abscesses [1]. In the pre-antibiotic era, complications of otitis media such as lateral sinus thrombosis commonly led to serious neurologic sequelae and septic emboli. Infections in the mastoid can result in dural venous thrombosis by several mechanisms. The close proximity of the sigmoid sinus allows adjacent inflammation to activate platelets and fibrin resulting in a mural thrombus [2]. Such thrombi may then propagate to the adjacent dural venous sinuses (transverse, inferior petrosal, superior petrosal), or the internal jugular vein. Dural venous sinus thrombosis may result in reduced resorption of cerebrospinal fluid (CSF) from arachnoid granulations. Reduced resorption of CSF then results in increased intracranial pressure (ICP), which is known as otitic hydrocephalus. Patients with otogenic sigmoid sinus thrombosis typically present with
§ Oral presentation: American Academy of Otolaryngology-Head and Neck Surgery Annual Meeting, 26–29 September 2010, Boston, MA. * Corresponding author. Tel.: +1 214 648 3437; fax: +1 214 648 9122. E-mail address: [email protected] (B. Isaacson).
0165-5876/$ – see front matter ß 2011 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2011.03.024
high spiking fevers, otorrhea, and post auricular tenderness Griesinger’s sign [3]. With the introduction of antibiotics as the primary treatment for otitis media this ‘‘classic’’ presentation has become less common and replaced with symptoms and findings such as headache, nausea and vomiting, low grade fevers, and signs of increased ICP [4]. The diagnosis of sigmoid sinus thrombosis has been aided by the use of non invasive imaging, most notably MRI/ MRV or less reliably CT [5]. According to a study by Proctor in 1966, the use of antibiotics in conjunction with surgical intervention has decreased the mortality from 40% to essentially 0 [6]. The use of anticoagulation in the setting of otogenic lateral sinus thrombosis remains controversial [7,8]. The objective of this retrospective study is to examine treatment outcomes in pediatric patients with lateral sinus thrombosis including sinus recanalization and resolution of otitic hydrocephalus. 2. Methods A retrospective chart review was performed at a tertiary children’s hospital in a major metropolitan area. All inpatient admissions from 1999 to 2010 were queried for the ICD-9 code [325] thrombosis of intracranial venous sinus. Ninety-three patients were initially identified. Included in our series were patients that were found to have thrombosis documented on MRI and from an otogenic
R.E. Neilan et al. / International Journal of Pediatric Otorhinolaryngology 75 (2011) 850–853
source. All subjects with otogenic sigmoid sinus thrombosis were preceded by and temporally related to an episode of acute otitis media. Those with thrombosis from aseptic sources such as stroke and trauma were excluded. Careful chart review found 15 patients of which the thrombosis was of otogenic origin. Information on age, gender, length of hospital stay, type of sigmoid sinus treatment, overall surgical treatment, whether purulence was encountered, presence of otitic hydrocephalus, venous sinuses involved, use of anticoagulation, length of anticoagulation treatment if rendered, state of sinus on follow up MRI, the interval between diagnosis and follow up MRI, and any other complications from the disease process were recorded. Exclusion criteria included any patients older than 18 years of age and medical records that were incomplete or of questionable accuracy. Trace flow detected in at least one of the following was classified as partial recanalization: sigmoid sinus, transverse sinus, or internal jugular vein. Normal flow through all thrombosed venous segments was classified as complete recanalization. This study was approved by the Institutional Review Board at the University of Texas Southwestern Medical Center. 3. Results Otogenic lateral sinus thrombosis was diagnosed in 10 male patients and 5 female patients in study period. The age of subjects ranged from 6 months to 14 years of age with a mean of 4.87 years. The mean length of symptoms prior to presentation was 8.6 days. Length of hospital stay ranged from 4 days to 48 days with a mean of 17 days. Thrombi frequently extended from the sigmoid sinus into the transverse sinus (11 patients, 73.3%), internal jugular vein (10 patients, 66.7%) and cavernous sinus (1 patient, 6.7%) (Table 1). All 15 patients underwent a mastoidectomy and tympanostomy tube placement. Mastoid purulence was encountered in 80% of these patients. The sigmoid sinus was addressed by needle decompression in 3 patients (20%), osseous decompression of the sigmoid plate in 3 patients (20%), and venotomy in 6 patients (40%). Venotomy consisted of incising the vein and draining any purulence and evacuating some clot, but not to the point of bleeding. No packing or vein ligation was performed. Two of the six patients undergoing venotomy were found to have intraluminal pus. In 3 patients (20%) the sigmoid sinus was
851
not manipulated or exposed (simple mastoidectomy only). Management of the thrombosed sigmoid sinus varied according to surgeon’s discretion. An algorithm for the operative management of the sigmoid sinus did not exist secondary to the retrospective nature of the study. Cholesteatoma was identified in one patient (6%). Intra operative cultures grew out Group A Streptococcus in four patients (27%), Streptococcus pneumoniae in three patients (20%), methicillin resistant Staphylococcus aureus in one patient (6.5%), and Streptococcus intermedius in one patient (6.5%). Six of the cultures demonstrated no growth (40%). The effects of culture results on outcomes was not assessed due to the limited number of cases. Otitic hydrocephalus was identified in five patients (33%) all of whom presented with diplopia from a 6th cranial nerve palsy. All five patients with otitic hydrocephalus eventually recovered as evidenced by resolution of their 6th nerve palsy and/or papilledema on ophthalmologic examination. Twelve patients (85%) were treated with low molecular weight heparin postoperatively, and 3 (15%) were not anticoagulated. The mean duration of anticoagulation was 3.4 months. Fourteen of the fifteen patients had follow up MRI examinations, one patient did not undergo MRI, and no patients were lost to follow up. The mean time interval from surgical treatment to when the last MRI was completed was 5.7 months with a range from 1 to 26 months. In the ten patients who demonstrated some degree of recanalization the mean length of time was 6.2 months with a range from 1 to 26 months. Persistent total occlusion of the sigmoid sinus was identified in 4 (28.6%) patients, while complete recanalization was noted in 2 patients (14.3%), and partial recanalization was found in 8 patients (57.1%) (Figs. 1 and 2). Five of eight patients with partial recanalization showed flow signal in all previously occluded segments restoring venous continuity. Three of eight patients had partial recanalization which did not have restoration of venous continuity through all previously involved segments. Seven of fourteen (50%) patients with follow imaging demonstrated restoration of venous continuity whether recanalization was found to be partial or complete. Eleven of the 14 (79%) patients that had follow-up MRIs were anticoagulated, some degree of recanalization was seen in eight patients (73%) with three patients (27%) still demonstrating no flow. Of the three (21%) which were not anticoagulated two (66.6%)
Table 1 Patient demographic, treatment, and outcome data. Patient
Age (years)
Length of stay (days)
Side
Sigmoid sinus treatment
Otitic hydrocephalus
Location of thrombus
Anti coagulation
Length of treatment
F/U MRI
MRI interval (months)
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15
9 6 6 0.67 3 2 1.5 3 6 5 12 14 0.5 1.5 3
48 7 11 10 5 7 20 42 12 4 17 13 8 44 8
Right Left Right Left Left Left Left Right Right Right Left Right Left Left Right
Needle Decomp Venotomy Venotomy Venotomy Venotomy Venotomy Needle Decomp None None None Venotomy Needle Decomp
Yes, No No No No No Yes, Yes, No No No Yes, Yes, No No
SS, SS SS, SS, SS SS, SS, SS, SS, SS, SS, SS, SS, SS, SS,
LMWH LMWH LMWH LMWH None LMWH LMWH LMWH LMWH None None LMWH LMWH LMWH LMWH
3 months 6 weeks 3 months 4 months N/A 3 months 3 months 6 months 3 months N/A N/A 3 months 6 months 8 months 3 months
No Flow Complete Partial Partial Complete Partial Partial Partial No MRI No Flow Partial Partial No Flow Partial No Flow
2 26 1 3 4 3 2 1 N/A 2 7 5 12 10 2
LRP, Right
LRP, Right LRP, Bilateral
LRP, Left LRP, Bilateral
TS, IJ TS, IJ TS, IJ TS, IJ IJ TS TS, IJ TS TS, IJ TS, IJ TS Cav, IJ TS, IJ
LOS: length of stay; needle: the osseous sigmoid plate was removed and the sinus was needle aspirated; decomp (decompressed): the osseous sigmoid plate was removed with removal of granulation tissue and pus, but the sinus was not opened; venotomy: the osseous sigmoid plate was removed and the sinus was opened and clot and purulence were evacuated; none: no manipulation of the sinus; LRP: lateral rectus palsy; SS: sigmoid sinus; TS: transverse sinus; IJ: internal jugular vein; Cav: cavernous sinus; no flow: no flow was identified on the follow up MRV; partial: recanalization with a more narrow caliber lumen involving at least one or all of the thrombosed segments of the lateral sinus; complete: complete recanalization to normal caliber.
[()TD$FIG]
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heparin, developed a treatment related small surgical site hematoma, which was incised and evacuated in clinic with local anesthesia. Patients developing additional complications did not differ in presentation from those subjects that did not. 4. Discussion
Fig. 1. Pre-treatment reconstructed coronal view of MRV demonstrating flow void in the left sigmoid sinus and internal jugular vein.
[()TD$FIG]
Fig. 2. Post-treatment reconstructed coronal MRV of same patient showing recanalization through previously thrombosed segments.
demonstrated recanalization with one (33.3%) demonstrating persistent thrombosis. Of the two patients who demonstrated complete recanalization; one received anticoagulation and the other did not. Two patients developed septic hip joints during the course of their illness presumably from septic emboli. There were no mortalities in this series of fifteen patients. One patient, whose blood and wound cultures grew out Fusobacterium necrophorum, developed a cerebral hemispheric ischemic stroke from an ipsilateral internal carotid artery thrombosis. This patient was treated with low molecular weight heparin, and eventually regained use of his paretic lower extremity, but continues to have upper extremity paralysis and spasticity. One patient who was also administered low molecular weight heparin developed an endophthalmitis. One patient, treated with low molecular weight
Temporal bone surgery, modern imaging (i.e., MRI and CT), and antibiotics are largely responsible for the reduced incidence of intratemporal and intracranial complications of otitis media [1]. Despite these advances, complications of otitis media still occur, and if not recognized and treated in a timely fashion, may result in significant morbidity and mortality [9]. Otogenic lateral sinus thrombosis is an infrequent complication that is now rarely associated with mortality or permanent morbidity in areas with access to care [10,11]. Goldenberg found that 66% of the otolaryngology departments they surveyed had no episodes of sigmoid sinus thrombosis in the 5 years preceding the survey. The incidence of sigmoid sinus thrombosis from the 83 departments surveyed was found to be 0.625 cases per department for the 5 years preceding the survey [12]. Patients may present with a myriad of symptoms including headache, fever, nuchal rigidity, post auricular pain, and diplopia [4]. An ongoing or recent history of middle ear infection is usually elicited. Typical physical exam findings in patients with lateral sinus thrombosis include serous or purulent middle ear effusion, post auricular tenderness/edema, lateral rectus palsy, and papilledema [4,13,14]. Otitic hydrocephalus is not uncommon in the setting of dural venous sinus thrombosis and occurred in 5 out of the 15 (33.3%) patients in our series. A decrease in CSF resorption from arachnoid granulations secondary to thrombosis in a dominant lateral dural venous sinus is the purported pathophysiologic mechanism for otitic hydrocephalus [15]. In the present series, the 5 patients with otitic hydrocephalus presented with diplopia secondary to a 6th nerve palsy. One patient was noted to have papilledema, and another patient had an elevated opening pressure during a lumbar puncture. As in previous reports, complete resolution of otitic hydrocephalus was noted in all five patients even in the setting of persistent lateral sinus thrombosis. Recovery from otitic hydrocephalus is thought to be secondary to development of compensatory collateral venous drainage [4,15]. A number of questions remain with respect to the management of lateral sinus thrombosis including the need for and the extent of operative intervention, as well as the utility of anticoagulation. Management of lateral sinus thrombosis has included observation, pressure equalization tube placement, simple mastoidectomy, removal of the sigmoid plate with or without perisinus exploration, needle aspiration, and venotomy with partial or complete evacuation of thrombus, resection of the thrombosed sigmoid sinus, bulb and internal jugular vein, and internal jugular vein ligation [5,13,14,16–18]. Based on prior reports, it appears that manipulation of the lateral sinus from needle aspiration to resection have all been associated with symptom resolution in the vast majority of cases [4,5,18,19]. A definitive cause and effect relationship between lateral sinus operative intervention and symptom resolution has not been established. Recanalization of the lateral sinus has been demonstrated in patients undergoing non-operative medical management, perisinus exploration, needle aspiration, and venotomy with or without thrombectomy [4,10,19]. Resection of the involved sinus and or ligation of the internal jugular vein precludes recanalization [5]. Internal jugular vein ligation may be considered in the setting of septic emboli which is unresponsive to medical management. There is currently no evidence for or against the use of thrombectomy to reduce recovery time and increase the likelihood of recanalization. In agreement with previous studies, the present series demonstrated
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no obvious correlation with sinus manipulation and recanalization [4,10,14,20]. A trend towards more conservative operative management was noted later in the time course of the present study. The data from eight studies of patients with lateral sinus thrombosis was reviewed and pooled for type of treatment, and recanalization rates. 45 out of 52 (90.4%) patients had a follow up MRI or ultrasound imaging to assess the patency of the affected sinuses [4,8,10,11,14,15,18,19]. Thirty six of fifty two (69.2%) patients were anticoagulated with either low molecular weight heparin, or a combination of heparin and coumadin. Twenty nine of forty seven (61.7%) patients with follow up imaging demonstrated partial or complete recanalization which approximates the incidence of partial or complete recanalization in the present study (71.4%). Anticoagulation is sometimes utilized in the setting of septic emboli, or propagation of thrombus to adjacent sinuses or the internal jugular vein [7]. A majority of the patients in the present series were anticoagulated with low molecular weight heparin as recommended by the pediatric hematology or neurology service. Patients with lateral sinus thrombosis at our institution are routinely treated with anticoagulation per recommendations by the pediatric hematology service. A number of studies have examined the pros and cons of anticoagulation for lateral sinus thrombosis, but due to the rarity of this condition insufficient evidence exists to support or reject its use. One patient in this series had a presumed anticoagulation related post surgical hematoma which resolved with incision and drainage. Forty-seven patients from eight studies in addition to the present study had follow up imaging data to determine the rate of recanalization with respect to the use of anticoagulation [4,8,10,11,14,15,18,19]. Twenty of 30 (66.7%) patients who were administered anticoagulation showed evidence of recanalization. Nine of seventeen (52.9%) patients who were not given anticoagulation showed evidence of recanalization. A Fisher Exact test showed no significant effect of anticoagulation on lateral sinus recanalization (p = 0.53). The lack of effect seen with anticoagulation on sinus recanalization may be related to the small sample size despite combining data from the present study with seven prior reports [4,8,10,11,15,16,19,20]. These previous reports demonstrate recanalization rates of 0–72% of patients who were anticoagulated and 0–100% of those in whom no anticoagulation was administered [4,8,11,15,16,19,20]. In the present study 79% of patients were anticoagulated resulting in 73% partial or complete recanalization. 5. Conclusion Lateral sinus thrombosis is a rare complication of acute and chronic otitis media. The appropriate management of this complication remains controversial with respect to the necessity and extent of surgical intervention and the use of anticoagulation. Mortality is rare from otogenic lateral sinus thrombosis, but
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significant morbidity still occurs. In some cases, follow up imaging demonstrates partial or complete lateral sinus recanalization. How anticoagulation and surgical intervention affect recanalization remains unclear. Conflicts of interest Peter Roland, MD – Consultant for Alcon; Ryan Neilan MD – none; J. Walter Kutz Jr., MD – none; Kenneth H. Lee, MD, PhD – none; Brandon Isaacson, MD – Consultant and Course Instructor for Medtronic Midas Rex Institute. References [1] B. Isaacson, C. Mirabal, J.W. Kutz Jr., K.H. Lee, P.S. Roland, Pediatric otogenic intracranial abscesses, Otolaryngol. Head Neck Surg. 142 (2010) 434–437. [2] C. Bianchini, C. Aimoni, S. Ceruti, D.L. Grasso, A. Martini, Lateral sinus thrombosis as a complication of acute mastoiditis, Acta Otorhinolaryngol. Ital. 28 (2008) 30– 33. [3] R.F. Wetmore, Complications of otitis media, Pediatr. Ann. 29 (2000) 637–646. [4] C.B. Bales, S. Sobol, R. Wetmore, L.M. Elden, Lateral sinus thrombosis as a complication of otitis media: 10-year experience at the children’s hospital of Philadelphia, Pediatrics 123 (2009) 709–713. [5] S. Manolidis, J.W. Kutz Jr., Diagnosis and management of lateral sinus thrombosis, Otol. Neurotol. 26 (2005) 1045–1051. [6] C.A. Proctor, Intracranial complications of otitic origin, Laryngoscope 76 (1966) 288–308. [7] D.T. Bradley, G.T. Hashisaki, J.C. Mason, Otogenic sigmoid sinus thrombosis: what is the role of anticoagulation? Laryngoscope 112 (2002) 1726–1729. [8] U.K. Shah, T.F. Jubelirer, J.D. Fish, L.M. Elden, A caution regarding the use of lowmolecular weight heparin in pediatric otogenic lateral sinus thrombosis, Int. J. Pediatr. Otorhinolaryngol. 71 (2007) 347–351. [9] S.P. Dubey, V. Larawin, Complications of chronic suppurative otitis media and their management, Laryngoscope 117 (2007) 264–267. [10] N. Christensen, J. Wayman, J. Spencer, Lateral sinus thrombosis: a review of seven cases and proposal of a management algorithm, Int. J. Pediatr. Otorhinolaryngol. 73 (2009) 581–584. [11] N. de Oliveira Penido, J.R. Testa, D.P. Inoue, O.L. Cruz, Presentation, treatment, and clinical course of otogenic lateral sinus thrombosis, Acta Otolaryngol. 129 (2009) 729–734. [12] R.A. Goldenberg, Lateral sinus thrombosis. Medical or surgical treatment? Arch. Otolaryngol. 111 (1985) 56–58. [13] H. Seven, A.E. Ozbal, S. Turgut, Management of otogenic lateral sinus thrombosis, Am. J. Otolaryngol. 25 (2004) 329–333. [14] D.M. Kaplan, M. Kraus, M. Puterman, A. Niv, A. Leiberman, D.M. Fliss, Otogenic lateral sinus thrombosis in children, Int. J. Pediatr. Otorhinolaryngol. 49 (1999) 177–183. [15] J. Kuczkowski, M. Dubaniewicz-Wybieralska, T. Przewozny, W. Narozny, B. Mikaszewski, Otitic hydrocephalus associated with lateral sinus thrombosis and acute mastoiditis in children, Int. J. Pediatr. Otorhinolaryngol. 70 (2006) 1817–1823. [16] E.E. Tov, A. Leiberman, I. Shelef, D.M. Kaplan, Conservative nonsurgical treatment of a child with otogenic lateral sinus thrombosis, Am. J. Otolaryngol. 29 (2008) 138–141. [17] E.H. Ooi, M. Hilton, G. Hunter, Management of lateral sinus thrombosis: update and literature review, J. Laryngol. Otol. 117 (2003) 932–939. [18] A. Kutluhan, M. Kiris, V. Yurttas, A.F. Kiroglu, O. Unal, When can lateral sinus thrombosis be treated conservatively? J. Otolaryngol. 33 (2004) 107–110. [19] A. Agarwal, P. Lowry, G. Isaacson, Natural history of sigmoid sinus thrombosis, Ann. Otol. Rhinol. Laryngol. 112 (2003) 191–194. [20] M. Iseri, O. Aydin, E. Ustundag, G. Keskin, A. Almac, Management of lateral sinus thrombosis in chronic otitis media, Otol. Neurotol. 27 (2006) 1098–1103.