J Neurol Surg B Skull Base 2021; 82(02): 233-243
DOI: 10.1055/s-0040-1713105
Original Article

Predicting Anticoagulation Need for Otogenic Intracranial Sinus Thrombosis: A Machine Learning Approach

1   Boston University School of Medicine, Boston, Massachusetts, United States
,
Philip Ryan Camilon
1   Boston University School of Medicine, Boston, Massachusetts, United States
2   Department of Otolaryngology, Boston University, Boston, Massachusetts, United States
,
Jessica R. Levi
1   Boston University School of Medicine, Boston, Massachusetts, United States
2   Department of Otolaryngology, Boston University, Boston, Massachusetts, United States
,
1   Boston University School of Medicine, Boston, Massachusetts, United States
2   Department of Otolaryngology, Boston University, Boston, Massachusetts, United States
3   Department of Neurological Surgery and Ophthalmology, Boston, Massachusetts, United States
4   Institute for Health System Innovation and Policy, Boston University, Boston, Massachusetts, United States
› Author Affiliations

Abstract

Objective The role of anticoagulation (AC) in the management of otogenic cerebral venous sinus thrombosis (OCVST) remains controversial. Our study aims to better define when AC is used in OCVST.

Methods MEDLINE, EMBASE, and The Cochrane Library were searched from inception to February 14, 2019 for English and English-translated articles. References cited in publications meeting search criteria were searched. Titles and abstracts were screened and identified in the literature search, assessing baseline risk of bias on extracted data with the methodological index for nonrandomized studies (MINORS) scale. Random effects meta-regression followed by random forest machine learning analysis across 16 moderator variables between AC and nonanticoagulated (NAC) cohorts was conducted.

Results A total of 92% of treated patients were free of neurologic symptoms at the last follow-up (mean 29.64 months). Four percent of AC and 14% of NAC patients remained symptomatic (mean 18.72 and 47.10 months). 3.5% of AC patients experienced postoperative wound hematomas. AC and NAC recanalization rates were 81% (34/42) and 63% (five-eights), respectively. OCVST was correlated with cholesteatoma and intracranial abscess. Among the analyzed covariates, intracranial abscess was most predictive of AC and cholesteatoma was most predictive of NAC. Comorbid intracranial abscess and cholesteatoma were predictive of AC.

Conclusion The present study is the first to utilize machine learning algorithms in approaching OCVST. Our findings support the therapeutic use of AC in the management of OCVST when complicated by thrombophilia, intracranial abscess, and cholesteatoma. Patients with intracranial abscess and cholesteatoma may benefit from AC and surgery. Patients with cholesteatoma can be managed with NAC and surgery.



Publication History

Received: 19 November 2019

Accepted: 15 April 2020

Article published online:
05 October 2020

© 2020. Thieme. All rights reserved.

Georg Thieme Verlag KG
Rüdigerstraße 14, 70469 Stuttgart, Germany

 
  • References

  • 1 Bales CB, Sobol S, Wetmore R, Elden LM. Lateral sinus thrombosis as a complication of otitis media: 10-year experience at the children's hospital of Philadelphia. Pediatrics 2009; 123 (02) 709-713
  • 2 Manolidis S, Kutz Jr JW. Diagnosis and management of lateral sinus thrombosis. Otol Neurotol 2005; 26 (05) 1045-1051
  • 3 Dentali F, Gianni M, Crowther MA, Ageno W. Natural history of cerebral vein thrombosis: a systematic review. Blood 2006; 108 (04) 1129-1134
  • 4 Anthonsen K, Høstmark K, Hansen S. et al. Acute mastoiditis in children: a 10-year retrospective and validated multicenter study. Pediatr Infect Dis J 2013; 32 (05) 436-440
  • 5 Ghosh PS, Ghosh D, Goldfarb J, Sabella C. Lateral sinus thrombosis associated with mastoiditis and otitis media in children: a retrospective chart review and review of the literature. J Child Neurol 2011; 26 (08) 1000-1004
  • 6 Mattos JL, Colman KL, Casselbrant ML, Chi DH. Intratemporal and intracranial complications of acute otitis media in a pediatric population. Int J Pediatr Otorhinolaryngol 2014; 78 (12) 2161-2164
  • 7 Wong BY, Hickman S, Richards M, Jassar P, Wilson T. Management of paediatric otogenic cerebral venous sinus thrombosis: a systematic review. Clin Otolaryngol 2015; 40 (06) 704-714
  • 8 Mather M, Musgrave K, Dawe N. Is anticoagulation beneficial in acute mastoiditis complicated by sigmoid sinus thrombosis?. Laryngoscope 2018; 128 (11) 2435-2436
  • 9 Moher D, Liberati A, Tetzlaff J, Altman DG. PRISMA Group. Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. PLoS Med 2009; 6 (07) e1000097
  • 10 Inokuchi G, Tsutsumi N, Komatsu H, Fujita T, Sawada N, Kumoi K. Persistent petrosquamosal sinus: underlying cause of otitic hydrocephalus with lateral sinus thrombosis. Int J Pediatr Otorhinolaryngol 2013; 77 (11) 1908-1911
  • 11 Holzmann D, Huisman TA, Linder TE. Lateral dural sinus thrombosis in childhood. Laryngoscope 1999; 109 (04) 645-651
  • 12 Koitschev A, Simon C, Löwenheim H, Kumpf M, Besch D, Ernemann U. Delayed otogenic hydrocephalus after acute otitis media in pediatric patients: the changing presentation of a serious otologic complication. Acta Otolaryngol 2005; 125 (11) 1230-1235
  • 13 Lee JH, Choi SJ, Park K, Choung YH. Managements for lateral sinus thrombosis: does it need the ligation of internal jugular vein or anticoagulants?. Eur Arch Otorhinolaryngol 2009; 266 (01) 51-58
  • 14 Zangari P, Messia V, Viccaro M. et al. Genetic prothrombotic factors in children with otogenic lateral sinus thrombosis: five case reports. Blood Coagul Fibrinolysis 2012; 23 (02) 158-163
  • 15 Csákányi Z, Rosdy B, Kollár K, Móser J, Kovács E, Katona G. Timely recanalization of lateral sinus thrombosis in children: should we consider hypoplasia of contralateral sinuses in treatment planning?. Eur Arch Otorhinolaryngol 2013; 270 (07) 1991-1998
  • 16 Singh GB, Rai AK, Singh S, Sahu R, Arora R. Management of otogenic lateral sinus thrombosis. Auris Nasus Larynx 2014; 41 (02) 143-147
  • 17 Zanoletti E, Cazzador D, Faccioli C, Sari M, Bovo R, Martini A. Intracranial venous sinus thrombosis as a complication of otitis media in children: critical review of diagnosis and management. Int J Pediatr Otorhinolaryngol 2015; 79 (12) 2398-2403
  • 18 Schneider S, Kapelushnik J, Kraus M, El Saied S, Levi I, Kaplan DM. The association between otogenic lateral sinus thrombosis and thrombophilia—a long-term follow-up. Am J Otolaryngol 2018; 39 (03) 299-302
  • 19 Gold S, Kamerer DB, Hirsch BE, Cass SP. Hypercoagulability in otologic patients. Am J Otolaryngol 1993; 14 (05) 327-331
  • 20 Leiberman A, Lupu L, Landsberg R, Fliss DM. Unusual complications of otitis media. Am J Otolaryngol 1994; 15 (06) 444-448
  • 21 Sennaroğlu L, Kaya S, Gürsel B, Saatçi I. Role of MRI in the diagnosis of otitic hydrocephalus. Am J Otol 1996; 17 (05) 784-786
  • 22 Unsal EE, Ensari S, Koç C. A rare and serious complication of chronic otitis media: lateral sinus thrombosis. Auris Nasus Larynx 2003; 30 (03) 279-282
  • 23 Omer Unal F, Sennaroğlu L, Saatçi I. Otitic hydrocephalus: role of radiology for diagnosis. Int J Pediatr Otorhinolaryngol 2005; 69 (07) 897-901
  • 24 Kuczkowski J, Dubaniewicz-Wybieralska M, Przewoźny T, Narozny W, Mikaszewski B. Otitic hydrocephalus associated with lateral sinus thrombosis and acute mastoiditis in children. Int J Pediatr Otorhinolaryngol 2006; 70 (10) 1817-1823
  • 25 Redaelli de Zinis LO, Gasparotti R, Campovecchi C, Annibale G, Barezzani MG. Internal jugular vein thrombosis associated with acute mastoiditis in a pediatric age. Otol Neurotol 2006; 27 (07) 937-944
  • 26 Shah UK, Jubelirer TF, Fish JD, Elden LM. A caution regarding the use of low-molecular weight heparin in pediatric otogenic lateral sinus thrombosis. Int J Pediatr Otorhinolaryngol 2007; 71 (02) 347-351
  • 27 Sneed WF. Lateral sinus thrombosis. Am J Otol 1983; 4 (03) 258-262
  • 28 Zalzal GH. Acute mastoiditis complicated by sigmoid sinus thrombosis in congenital aural atresia. Int J Pediatr Otorhinolaryngol 1987; 14 (01) 31-39
  • 29 Tovi F, Hirsch M, Gatot A. Superior vena cava syndrome: presenting symptom of silent otitis media. J Laryngol Otol 1988; 102 (07) 623-625
  • 30 Doyle KJ, Jackler RK. Otogenic cavernous sinus thrombosis. Otolaryngol Head Neck Surg 1991; 104 (06) 873-877
  • 31 Oyarzabal MF, Patel KS, Tolley NS. Bilateral acute mastoiditis complicated by lateral sinus thrombosis. J Laryngol Otol 1992; 106 (06) 535-537
  • 32 Garcia RD, Baker AS, Cunningham MJ, Weber AL. Lateral sinus thrombosis associated with otitis media and mastoiditis in children. Pediatr Infect Dis J 1995; 14 (07) 617-623
  • 33 Lubianca Neto JF, Saffer M, Rotta FT, Arrarte JL, Brinckmann CA, Ferreira P. Lateral sinus thrombosis and cervical abscess complicating cholesteatoma in children: case report and review. Int J Pediatr Otorhinolaryngol 1998; 42 (03) 263-269
  • 34 Spandow O, Gothefors L, Fagerlund M, Kristensen B, Holm S. Lateral sinus thrombosis after untreated otitis media. A clinical problem—again?. Eur Arch Otorhinolaryngol 2000; 257 (01) 1-5
  • 35 Ram B, Meiklejohn DJ, Nunez DA, Murray A, Watson HG. Combined risk factors contributing to cerebral venous thrombosis in a young woman. J Laryngol Otol 2001; 115 (04) 307-310
  • 36 McMullan R, McConville C, Clarke JC, Adams DA, Hedderwick S. Lemierre syndrome: remember the forgotten disease. Ulster Med J 2004; 73 (02) 123-125
  • 37 Barbara M, Consagra C, Buongiorno G, Monini S, Bandiera G, Filipo R. Genetically-induced deep venous thrombosis presenting as acute mastoiditis. J Laryngol Otol 2005; 119 (04) 308-310
  • 38 Ozdemir D, Cakmakci H, Ikiz AO. et al. Sigmoid sinus thrombosis following mastoiditis: early diagnosis enhances good prognosis. Pediatr Emerg Care 2005; 21 (09) 606-609
  • 39 Masterson T, El-Hakim H, Magnus K, Robinson J. A case of the otogenic variant of Lemierre's syndrome with atypical sequelae and a review of pediatric literature. Int J Pediatr Otorhinolaryngol 2005; 69 (01) 117-122
  • 40 Dorn M, Liener K, Rozsasi A, Keck T. Prolonged diplopia following sinus vein thrombosis mimicking Gradenigo's syndrome. Int J Pediatr Otorhinolaryngol 2006; 70 (04) 741-743
  • 41 Vlastos IM, Helmis G, Athanasopoulos I, Houlakis M. Acute mastoiditis complicated with bezold abscess, sigmoid sinus thrombosis and occipital osteomyelitis in a child. Eur Rev Med Pharmacol Sci 2010; 14 (07) 635-638
  • 42 Scardapane A, Del Torto M, Nozzi M, Elio C, Breda L, Chiarelli F. Gradenigo's syndrome with lateral venous sinus thrombosis: successful conservative treatment. Eur J Pediatr 2010; 169 (04) 437-440
  • 43 Rosenberg JD, Parikh SR. Anticoagulation therapy as a supplement to recanalization for the treatment of sigmoid sinus thrombosis: a case report. Ear Nose Throat J 2011; 90 (09) 418-422
  • 44 Lee SJ, Weon YC, Cha HJ. et al. A case of atypical skull base osteomyelitis with septic pulmonary embolism. J Korean Med Sci 2011; 26 (07) 962-965
  • 45 Mustafa A, Toçi B, Thaçi H, Gjikolli B, Baftiu N. Acute mastoiditis complicated with concomitant Bezold's abscess and lateral sinus thrombosis. Case Rep Otolaryngol 2018; 2018: 8702532
  • 46 Zapanta PE, Chi DH, Faust RA. A unique case of Bezold's abscess associated with multiple dural sinus thromboses. Laryngoscope 2001; 111 (11 Pt 1): 1944-1948
  • 47 de Oliveira Penido N, Testa JR, Inoue DP, Cruz OL. Presentation, treatment, and clinical course of otogenic lateral sinus thrombosis. Acta Otolaryngol 2009; 129 (07) 729-734
  • 48 Slim K, Nini E, Forestier D, Kwiatkowski F, Panis Y, Chipponi J. Methodological index for non-randomized studies (minors): development and validation of a new instrument. ANZ J Surg 2003; 73 (09) 712-716
  • 49 Stijnen T, Hamza TH, Ozdemir P. Random effects meta-analysis of event outcome in the framework of the generalized linear mixed model with applications in sparse data. Stat Med 2010; 29 (29) 3046-3067
  • 50 R Core Team. R: A language and environment for statistical computing. R Foundation for Statistical Computing; Vienna, Austria: 2018. . Accessed October 24, 2018 at: https://www.R-project.org/
  • 51 RStudio Team. RStudio: integrated development for R. RStudio, Inc.; Boston, MA: 2016. . Accessed October 24, 2018 at: http://www.rstudio.com
  • 52 van Lissa CJ. metaforest: exploring heterogeneity in meta-analysis using random forests. R package version 0.1.2. 2018 . Accessed October 24, 2018 at: https://CRAN.R-project.org/package=metaforest
  • 53 Janitza S, Celik E, Boulesteix A. A computationally fast variable importance test for random forests for high-dimensional data. Adv Data Anal Classif 2018; 12 (04) 885-915
  • 54 Viechtbauer W. Conducting meta-analyses in R with the metafor package. J Stat Softw 2010; 36 (03) 1-48
  • 55 Marvin N, Wright AZ. ranger: a fast implementation of random forests for high dimensional data in C++ and R. J Stat Softw 2017; 77 (01) 1-17
  • 56 Kuhn M, Wing J, Weston S. et al. caret: classification and regression training. R package version 6.0–81. 2018 . Accessed October 24, 2018 at: https://CRAN.R-project.org/package=caret
  • 57 Newall F, Branchford B, Male C. Anticoagulant prophylaxis and therapy in children: current challenges and emerging issues. J Thromb Haemost 2018; 16 (02) 196-208
  • 58 Greene LA, Law C, Jung M. et al. Lack of anti-factor Xa assay standardization results in significant low molecular weight heparin (enoxaparin) dose variation in neonates and children. J Thromb Haemost 2014; 12 (09) 1554-1557
  • 59 Romantsik O, Bruschettini M, Zappettini S, Ramenghi LA, Calevo MG. Heparin for the treatment of thrombosis in neonates. Cochrane Database Syst Rev 2016; 11: CD012185
  • 60 Goto T, Camargo Jr CA, Faridi MK, Freishtat RJ, Hasegawa K. Machine learning-based prediction of clinical outcomes for children during emergency department triage. JAMA Netw Open 2019; 2 (01) e186937
  • 61 Thompson SG, Higgins JP. How should meta-regression analyses be undertaken and interpreted?. Stat Med 2002; 21 (11) 1559-1573
  • 62 Winham SJ, Freimuth RR, Biernacka JM. A weighted random forests approach to improve predictive performance. Stat Anal Data Min 2013; 6 (06) 496-505
  • 63 Tang F, Ishwaran H. Random forest missing data algorithms. Stat Anal Data Min 2017; 10 (06) 363-377
  • 64 Connors JM. Thrombophilia testing and venous thrombosis. N Engl J Med 2017; 377 (12) 1177-1187
  • 65 Scherer A, Jea A. Pediatric otogenic sigmoid sinus thrombosis: case report and literature reappraisal. Glob Pediatr Health 2017; 4: X17738837
  • 66 Ryan JT, Pena M, Zalzal GH, Preciado DA. Otogenic lateral sinus thrombosis in children: a review of 7 cases. Ear Nose Throat J 2016; 95 (03) 108-112
  • 67 Singh GB, Arora R, Garg S, Kumar D, Ranjan S. Septic lateral sinus thrombosis: sinus exploration is unnecessary. Case Rep Otolaryngol 2016; 2016: 4349538
  • 68 Moharir MD, Shroff M, Stephens D. et al. Anticoagulants in pediatric cerebral sinovenous thrombosis: a safety and outcome study. Ann Neurol 2010; 67 (05) 590-599
  • 69 Coutinho J, de Bruijn SF, Deveber G, Stam J. Anticoagulation for cerebral venous sinus thrombosis. Cochrane Database Syst Rev 2011; (08) CD002005