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Otogenic lateral sinus thrombosis in children: a review of 7 cases.

Abstract

Otogenic lateral sinus thrombosis (LST) is a rare but serious intracranial complication of acute or chronic otitis media. Reported mortality rates have ranged from 8 to 25%; the pediatric mortality rate might be as low as 5%. Controversy still exists over the medical and surgical management of this condition. We conducted a retrospective chart review of 7 cases of pediatric otogenic LST that were treated at our institution over a period of 8 years. We hypothesized that good outcomes in very sick patients can beachievedby aggressively managing the mastoid cavity and without the need for a thrombectomy. Our study group was made up of 4 boys and 3 girls, aged 6 to 15 years (mean: 11.1). All patients received intravenous antibiotics and underwent mastoidectomy with unroofing of the sigmoid sinus and placement of a tympanostomy tube. Sinus exploration with thrombectomy was not performed in any patient. Anticoagulation was used perioperatively in 5 patients (71%) without complication. All patients recovered well without major sequelae, which supports our hypothesis. We also describe the case of a patient with multiple concomitant intracranial comorbidities associated with this rare condition.

Introduction

Otogenic lateral sinus thrombosis (LST) is a rare and serious intracranial complication of acute or chronic otitis media. Reported mortality rates in most larger case series have ranged from 8 to 25%,14 but the pediatric mortality rate may be as low as 5%. (5)

Controversy still exists over the medical and surgical management of this condition. Management has traditionally included intravenous antibiotic therapy and mastoidectomy with thrombus removal. (6-10) Anticoagulation is used perioperatively in some cases, and its use continues to be an area of debate. Internal jugular vein ligation, historically a common part of surgical intervention, is no longer routinely employed. (11-13)

In this article, we present our small, descriptive case series of children with otogenic LST. We hypothesized that good outcomes in very sick patients can be achieved by aggressively managing the mastoid cavity and not performing thrombectomy. We also describe a case that involved multiple concomitant intracranial comorbidities associated with this rare condition.

Patients and methods

We conducted a retrospective chart review of all cases of pediatric otogenic LST that had been treated at our institution over a period of 8 years. We compiled information on presentation, coexisting intracranial complications, treatment, cultured organisms, and outcomes.

Our study group was made up of 4 boys and 3 girls, aged 6 to 15 years (mean: 11.1) (table 1). The most common presenting signs and symptoms were fever in 5 patients, otalgia in 5, and mastoid tenderness, nuchal rigidity, and otorrhea in 3 (table 2). Coexisting intracranial complications were present in 4 patients (57%), the most common of which were epidural abscess in 2 patients and otitic hydrocephalus in 2 (table 3).

All patients had been treated with intravenous antibiotics, and all had undergone mastoidectomy with unroofing of the sigmoid sinus and placement of a tympanostomy tube. Sinus exploration with thrombectomy was not performed in any patient. Anticoagulation was used perioperatively in 5 patients (71%) without complication. The most common organisms isolated were Streptococcus spp (table 1).

Institutional review board approval was obtained for the study protocol.

After treatment, all patients recovered well, and none experienced a major sequela. One patient with cavernous sinus thrombosis and otitic hydrocephalus experienced a persistent right visual-field deficit.

Case report

A10-year old girl (patient 6) presented to the emergency room with a history of blurred vision and ataxia. On physical examination, she was noted to have papilledema and right-sided otorrhea. Computed tomography (CT) demonstrated mastoid cavity and ethmoid sinus opacification on the right. A triangle of contrast-enhanced dura was seen at the level of the sigmoid sinus, which represents the so-called delta sign (figure 1). Magnetic resonance venography (MRV) demonstrated right sigmoid and transverse sinus thrombosis, right proximal internal jugular vein thrombosis, and bilateral cavernous sinus thrombosis (figure 2).

The patient underwent an urgent right tympanomastoidectomy with unroofing of the sigmoid sinus, rightsided endoscopic sinus surgery, and a bilateral myringotomy with tube placement. In addition to intravenous antibiotics, she was anticoagulated postoperatively.

The patient recovered well, but she was left with a persistent right visual-field deficit. Repeat MRV 2 months postoperatively showed a persistent thrombosis. An MRV obtained 9 months after surgery demonstrated improved flow on the right side, but the thrombosis had not fully resolved.

Discussion

Otogenic LST as a complication of acute and chronic otitis media is believed to occur via one of two mechanisms. (1,4,11,14,15)

* Acute or chronic otitis media can cause erosion of the bone covering the sigmoid sinus, which then results in inflammation of the outer and inner walls of the sinus, which in turn eventually leads to thrombus formation.

* Thrombus formation occurs as a result of osteothrombophlebitic extension via small venules.

The clinical signs and symptoms of otogenic LST include high-grade fever, otalgia, mastoid tenderness, neck stiffness, otorrhea, papilledema, and headache." (14,16,17) "Picket fence" fevers are classically described but rarely seen.

A previous report of a case of LST in a child with congenital aural atresia illustrated the difficulties one can encounter in diagnosing this entity. (15) The diagnosis is based on a high degree of clinical suspicion, and it is generally confirmed with imaging studies. CT with contrast demonstrates temporal bone pathology, as well as any associated bony erosion. An empty triangle at the level of the sigmoid sinus surrounded by contrast-enhanced dura (the delta sign) is diagnostic.

Magnetic resonance imaging (MRI) is considered to be more sensitive than CT in detecting sigmoid sinus thrombosis. The clot appears initially as hypointense on T2-weighted imaging, and then it transitions to hyperintense on T1 and T2 weighting, which reflects the characteristics of methemoglobin. MRV demonstrates an absence of flow in the affected sinus.

MRI allows for noninvasive evaluation of the brain for detecting associated swelling, ischemia, hemorrhage, or inflammation. Improvements in imaging techniques and the increased availability of MRI have dramatically reduced the need for invasive cerebral angiography. (5,18,19)

Management of LST has traditionally included broad-spectrum intravenous antibiotics, tympanostomy tube placement, and mastoid surgery with opening of the sinus and removal of the thrombus after needle aspiration to confirm the diagnosis. (6-10) Perioperative anticoagulation is used in selected cases.

Few reports have specifically addressed the role of anticoagulation. Bradley et al suggested that patients with a thrombus confined to the sigmoid sinus may be considered for treatment without anticoagulation in order to avoid its associated risks, but they advised that patients with evidence of thrombus progression or extension may benefit from systemic anticoagulation; the decision should be made on a case-by-case basis. (20)

Shah et al urged caution regarding the use of perioperative anticoagulation in view of the increased risk of bleeding-related complications. (21) In a recent review of case series in the literature over a 20-year period, Au et al recommended management with a broad-spectrum antibiotic and surgical removal of all perisinus infection. (22) The role of anticoagulation was unclear.

Internal jugular vein ligation was originally recommended in the preantibiotic era as a way to prevent septic emboli and thrombus extension, but it is now reserved for rare cases of thrombus extension, infection and thrombosis of the internal jugular vein, and persistent septicemia and pulmonary complications despite initial medical and surgical management. (11-13)

In recent years there has been a trend toward more conservative surgical management of LST, and several other authors have reported successful outcomes without performing thrombectomy as part of the surgical intervention. Kutluhan et al described 4 cases of LST in which 2 patients were successfully managed with sinus aspiration during surgery to confirm thrombosis; no further intervention was required. (17) Agarwal et al presented the case of a 3-year-old girl with otogenic LST who was treated with mastoidectomy. (23) In that case, the sinus was neither aspirated nor opened. The thrombus was monitored by ultrasonography via a window in the parieto-occipital bone, and recannulation of the transverse and sigmoid sinuses was demonstrated at week 5. Anticoagulation was not used.

In the report by Bradley et al, the authors reviewed their experience with 9 patients with otogenic LST; 7 of those patients underwent mastoidectomy, while only 2 had the sigmoid sinus opened. (20) Ooi et al reported on the management of 4 cases of otogenic LST in patients between the ages of 13 and 25 years. (12) All of them underwent surgical intervention with mastoidectomy. Thrombectomy was performed in 2 of 4 patients, and anticoagulation was used in 1. The authors recommended always removing the sinus plate, and they concluded that conservative surgical treatment of the sinus did not seem to affect the overall prognosis in their small series.

Syms et al reported 6 cases of otogenic LST in which all patients underwent surgical intervention with mastoidectomy, exposure of the lateral sinus, and needle aspiration to confirm the diagnosis. (16) No sinus exploration or thrombectomy was performed, and no therapeutic anticoagulation was used.

Singh described the management of 36 cases of otogenic LST in South Africa; 33 of these patients were younger than 20 years. (24) All 36 patients were treated with intravenous antibiotics and exploratory mastoidectomy. The diagnosis was confirmed at the time of surgery by direct observation or sinus aspiration. Thrombectomy was not performed in any patient. Nine patients did not respond to initial therapy because of resistant bacteria, but all of them responded to a change in antibiotic regimen. No deaths were reported in this series.

Christensen et al (25) and Bales et al (26) reported their respective institution's experience with otogenic LST. Christensen et al obtained good results in 7 patients treated with intravenous antibiotics, simple mastoidectomy with unroofing of the sigmoid sinus, and tympanostomy tube placement. (25) Bales et al reported good outcomes in 13 patients with intravenous antibiotics, simple mastoidectomy, tympanostomy tube placement and, in most patients, anticoagulation. (26)

There have been additional recent reports of successful nonsurgical management in highly selected cases. Tov et al reported the case of a 5-year-old boy with otogenic LST who demonstrated a prompt response to intravenous antibiotics, and who therefore was successfully treated without surgical intervention. (18) Wong et al described 3 cases in which patients were managed conservatively with tympanostomy tube placement and antibiotics, thereby avoiding mastoidectomy. (5) They recommended intravenous antibiotics, drainage of the middle ear with tympanostomy tube placement, and close monitoring, with mastoidectomy being reserved for refractory cases only.

Finally, Garcia et al presented a case of otogenic TST that was successfully treated with intravenous antibiotics, tympanostomy tube placement, and anticoagulation. (11) Gradual resolution of the thrombus was observed on serial imaging studies. Garcia et al also reviewed the literature on pediatric otogenic TST from 1960 to 1995 and found that 95% of 58 patients had undergone surgical intervention. They suggested that the medical therapy they described can be sufficient in highly selected cases.

In conclusion, our study demonstrated good outcomes with emergent treatment of otogenic TST with mastoidectomy and unroofing of the sigmoid sinus, tympanostomy tube placement, intravenous antibiotics, and selective anticoagulation. We did not find throm bectomy necessary, which supports the trend toward more conservative surgical management.

References

(1.) Samuel J, Fernandes CM, Steinberg JL. Intracranial otogenic complications: A persisting problem. Laryngoscope 1986;96(3):272-8.

(2.) Mathews TJ. Lateral sinus pathology (22 cases managed at Groote Schuur Hospital). J Laryngol Otol 1988;102(2):118-20.

(3.) Teichgraeber JF, Per-Lee JH, Turner JS Jr. Lateral sinus thrombosis: A modern perspective. Laryngoscope 1982;92(7 Pt 1):744-51.

(4.) Kangsanarak J, Navacharoen N, Fooanant S, Ruckphaopunt K. Intracranial complications of suppurative otitis media: 13 years' experience. Am J Otol 1995;16(1):104-9.

(5.) Wong I, Kozak FK, Poskitt K, et al. Pediatric lateral sinus thrombosis: Retrospective case series and literature review. J Otolaryngol 2005;34(2):79-85.

(6.) Samuel J, Fernandes CM. Lateral sinus thrombosis (a review of 45 cases). J Laryngol Otol 1987;101(12):1227-9.

(7.) Kuczkowski J, Mikaszewski B. Intracranial complications of acute and chronic mastoiditis: Report of two cases in children. Int J Pediatr Otorhinolaryngol 2001;60(3):227-37.

(8.) Azzi W, Saliba I, Forest VI, Abela A. Lateral sinus thrombosis: Serious complication of otitis media. J Otolaryngol 2005;34(6):427-31.

(9.) Lubianca Neto JF, Satfer M, Rotta FT, et al. Lateral sinus thrombosis and cervical abscess complicating cholesteatoma in children: Case report and review. Int J Pediatr Otorhinolaryngol 1998;42(3):263-9.

(10.) Kaplan DM, Kraus M, Puterman M, et al. Otogenic lateral sinus thrombosis in children. Int J Pediatr Otorhinolaryngol 1999;49(3): 177-83.

(11.) Garcia RD, Baker AS, Cunningham MF, Weber AL. Lateral sinus thrombosis associated with otitis media and mastoiditis in children. Pediatr Infect Dis J 1995; 14(7):617-23.

(12.) Ooi EH, Hilton M, Hunter G. Management of lateral sinus thrombosis: Update andliterature review. J Laryngol Otol 2003; 117(12):932-9.

(13.) Holzmann D, Huisman TA, Linder TE. Lateral dural sinus thrombosis in childhood. Laryngoscope 1999;109(4):645-51.

(14.) Agrawal S, Husein M, MacRae D. Complications of otitis media: An evolving state. J Otolaryngol 2005;34(Suppl l):S33-9.

(15.) Zalzal GH. Acute mastoiditis complicated by sigmoid sinus thrombosis in congenital aural atresia. Int J Pediatr Otorhinolaryngol 1987;14(l):31-9.

(16.) Syms MJ, Tsai PD, Holtel MR. Management oflateral sinus thrombosis. Laryngoscope 1999;109(10):1616-20.

(17.) Kutluhan A, Kiris M, Yurttas V, et al. When can lateral sinus thrombosis be treated conservatively? J Otolaryngol 2004;33(2):107-10.

(18.) Tov EE, Leiberman A, Shelef I, Kaplan DM. Conservative nonsurgical treatment of a child with otogenic lateral sinus thrombosis. Am J Otolaryngol 2008;29(2):138-41.

(19.) van den Bosch MA, Vos JA, de Letter MA, et al. MRI findings in a child with sigmoid sinus thrombosis following mastoiditis. Pediatr Radiol 2003;33(12):877-9.

(20.) Bradley DT, Hashisaki GT, Mason JC. Otogenic sigmoid sinus thrombosis: What is the role of anticoagulation? Laryngoscope 2002;112(10):1726-9.

(21.) 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 (2):347-51.

(22.) Au JK, Adam SI, Michaelides EM. Contemporary management of pediatric lateral sinus thrombosis: A twenty year review. Am J Otolaryngol 2013;34(2):145-50.

(23.) Agarwal A, Lowry P, Isaacson G. Natural history of sigmoid sinus thrombosis. Ann Otol Rhinol Laryngol 2003;112(2):191-4.

(24.) Singh B. The management of lateral sinus thrombosis. J Laryngol Otol 1993;107(9):803-8.

(25.) Christensen N, Wayman J, Spencer J. Lateral sinus thrombosis: A review of seven cases and proposal of a management algorithm. Int J Pediatr Otorhinolaryngol 2009;73(4):581-4.

(26.) 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(2):709-13.

Jesse T. Ryan, MD; Maria Pena, MD; George H. Zalzal, MD; Diego A. Preciado, MD, PhD

From the Department of Otolaryngology and Communication Sciences, Upstate University Hospital, State University of New York, Syracuse (Dr. Ryan); and the Division of Pediatric Otolaryngology-Head and Neck Surgery, Childrens National Medical Center, Washington, D.C. (Dr. Pena, Dr. Zalzal, and Dr. Preciado). The study described in this article was conducted at the Children's National Medical Center.

Corresponding author: Jesse T. Ryan, MD, Department of Otolaryngology and Communication Sciences, Upstate University Hospital, Campus West Bldg., 750 E. Adams St., Room 241, Syracuse, NY 13210. Email: ryanje@upstate.edu Results

Table 1. Summary of presenting symptoms, intracranial
complications, treatments, organisms cultured, and outcomes

Pt.   Age/sex   Presentation              Intracranial complications

1      11/M     Fever, otalgia, right     Right lateral, transverse,
                nuchal rigidity,          and internal jugular vein
                frontal headache          thrombosis, left trans-
                                          verse sinus thrombosis

2       8/M     Fever, otalgia,           Left lateral sinus
                mastoid                   thrombosis, left perisinus
                tenderness                epidural abscess

3      15/M     Fever, otalgia,           Right lateral and transverse
                mastoid                   sinus thrombosis, internal
                tenderness                jugular vein thrombosis

4      15/M     Fever, otalgia, nuchal    Right sigmoid sinus
                pain and rigidity,        thrombosis, meningitis
                otorrhea headache

5       6/F     Ophthalmoplegia           Bilateral lateral sinus
                and papilledema           thrombosis, right internal
                present for 4 days        jugular vein thrombosis,
                after discharge from      bilateral posterior fossae
                initial hospitalization   epidural abscesses, right
                                          middle fossa epidural
                                          abscess, otitic
                                          hydrocephalus

6      10/F     Blurred vision,           Right sigmoid and transverse
                ataxia, papilledema,      sinus thrombosis, right
                right-sided otorrhea      proximal internal jugular
                                          vein thrombosis, bilateral
                                          cavernous sinus thrombosis,
                                          otitic hydrocephalus

7      13/F     Fever, otalgia,           Right sigmoid and
                mastoid tenderness,       transverse sinus thrombosis,
                nuchal rigidity,          right internal jugular
                otorrhea, altered         vein thrombosis with
                mental status, nausea,    extension into the right
                vomiting, diarrhea        innominate vein

Pt.   Treatment                        Organism          Outcome

1     Right simple mastoidectomy,      --                No sequelae
      right myringotomy with tube
      placement, right maxillary
      sinus irrigation,
      anticoagulation

2     Left complete mastoidectomy      No growth         No sequelae
      with drainage of perisinus
      epidural abscess, left myring-
      otomy with tube placement

3     Right simple mastoidectomy,      Streptococcus     No sequelae
      right myringotomy with tube      pneumoniae
      placement

4     Right simple mastoidectomy,      Group A           No sequelae
      right myringotomy with tube      streptococci
      placement, anticoagulation

5     Bilateral complete mastoid-      No growth         No sequelae
      ectomies with drainage of
      abscesses, anticoagulation,
      bilateral myringotomy with
      tube placement

6     Right tympanomastoidectomy,      No growth         Right visual-
      right endoscopic sinus                             field deficit
      surgery, bilateral myringotomy
      with tube placement,
      anticoagulation

7     Bilateral myringotomy with       Proteus           No sequelae
      tube placement, incision         mirabilis,
      and drainage of subperiosteal    Corynebacterium
      abscess, staged tympano-         spp
      mastoidectomy (delayed
      due to low hematocrit),
      anticoagulation

Table 2. Most common presenting signs and
symptoms (N = 7)

Fever                5
Otalgia              5
Mastoid tenderness   3
Nuchal rigidity      3
Otorrhea             3
Papilledema          2
Headache             2

Table 3. Coexisting intracranial complications (N = 7)

Epidural abscess             2
Otitic hydrocephalus         2
Meningitis                   1
Cavernous sinus thrombosis   1
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Title Annotation:ORIGINAL ARTICLE
Author:Ryan, Jesse T.; Pena, Maria; Zalzal, George H.; Preciado, Diego A.
Publication:Ear, Nose and Throat Journal
Article Type:Clinical report
Date:Mar 1, 2016
Words:2934
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