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ANL-1978; No. of Pages 4 Auris Nasus Larynx xxx (2015) xxx–xxx

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A case of lateral sinus insufficiency with acute otitis media: Early surgical intervention for prevention of lateral sinus thrombosis Kuniyuki Takahashi *, Yutaka Yamamoto, Manabu Ogi, Shinsuke Ohshima, Yuka Morita, Sugata Takahashi Department of Otolaryngology, Head and Neck Surgery, Niigata University Faculty of Medicine, Japan

A R T I C L E I N F O

A B S T R A C T

Article history: Received 19 January 2015 Accepted 27 April 2015 Available online xxx

Objective: To describe a case of lateral sinus insufficiency, presumably occurring just prior to lateral sinus thrombosis (LST), and to discuss the importance of early surgical intervention and the pathophysiology of full-blown LST. Case report: A 4-year-old boy developed headaches and vomiting after exhibiting typical symptoms of acute otitis media. Contrast-enhanced CT revealed narrowing of the sigmoid sinus medially by gas and low-density material. We diagnosed the patient with suspected LST and consequently performed mastoidectomy. A large amount of bloody pus was found in the mastoid cavity and below the bony sinus plate. Sigmoid sinus blood flow was completely restored after drainage of the hemorrhagic and purulent material, and the patient recovered fully. Conclusion: Because of the anatomical features of the dural venous sinus, hemorrhage per diapedesis may be strongly associated with the development of LST. In the patient with suspected LST, early diagnosis and surgery prior to the development of intravenous thrombus are key for full recovery from this condition. ß 2015 Elsevier Ireland Ltd. All rights reserved.

Keywords: Lateral sinus thrombosis Hemorrhage per diapedesis Intravenous thrombosis

1. Introduction

2. Case report

Otogenic intracranial complications have decreased since the introduction of antibiotics. In particular, lateral sinus thrombosis (LST) has become a rare intracranial complication and is uncommonly encountered, even by otolaryngologists [1,2]. When LST develops and completely occludes the sinus, it frequently causes irreversible neural damage and may be life-threatening. Early diagnosis and treatment are mandatory. It is not clear how inflammation in the mastoid cavity influences lateral sinus blood flow and how thrombus formation occurs inside the lateral sinus. We described a rare case of acute lateral sinus insufficiency secondary to acute mastoiditis, which clinically mimicked LST. Our case may represent a preliminary step in the development of LST and may shed further light on the pathogenesis of LST.

A 4-year-old boy presented to his pediatrician with headaches and vomiting. The onset of high fever and right otalgia were noted 1 day prior to presentation. Laboratory data revealed leukocytosis (white blood cell count 20,000/mm3, neutrophils 86%) and elevated C-reactive protein level (11.78 mg/dl). His pediatrician suspected a diagnosis of meningitis; however, the cerebrospinal fluid examination was normal. Computed tomography (CT) findings were consistent with acute mastoiditis, and the patient was brought to our hospital the next day. On arrival, the patient exhibited no vertigo or any disturbance in his level of consciousness, but had severe headache accompanied by nausea and vomiting. Although cranial nerve IX, X, or XII palsies were not identified, his eye movements and visual acuity could not be evaluated. Otoscopy revealed redness and swelling on the posterior wall of the external ear canal and ear drum with slight otorrhea in his right ear. There was no retroauricular swelling or auricular displacement detected. High-resolution CT of the temporal bone revealed a soft tissue density in the welldeveloped right mastoid cavity and abnormal gas patterns in the sigmoid groove (Fig. 1A). Contrast-enhanced CT of the temporal bone demonstrated that the gas in the sigmoid groove was inside

* Corresponding author at: Department of Otolaryngology, Niigata University Faculty of Medicine, 1 Asahi-machi, Chuo-ku, Niigata 951-8510, Japan. Tel.: +81 25 227 2306; fax: +81 25 227 0786. E-mail address: [email protected] (K. Takahashi). http://dx.doi.org/10.1016/j.anl.2015.04.007 0385-8146/ß 2015 Elsevier Ireland Ltd. All rights reserved.

Please cite this article in press as: Takahashi K, et al. A case of lateral sinus insufficiency with acute otitis media: Early surgical intervention for prevention of lateral sinus thrombosis. Auris Nasus Larynx (2015), http://dx.doi.org/10.1016/j.anl.2015.04.007

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Fig. 1. High-resolution CT of the temporal bone. Bone density CT revealed the deterioration of the sigmoid sinus bony plate and abnormal gas (arrow head) in the sigmoid groove (A). Contrast-enhanced CT showed gas and soft tissue density narrowing the dural venous sinus (arrow heads) (B). This was not an empty delta sign.

the bony sinus plate, but outside the dural sinus. The sigmoid sinus was narrowed, and blood flow to the sigmoid sinus was present but markedly diminished. The jugular bulb was normally enhanced (Fig. 1B). MRI, MR angiography (MRA), and MR venography (MRV) were not performed. After hospital admission, the patient was administered panipenem/betamipron intravenously and underwent emergency surgery under general anesthesia. No defect in the cortical bone was observed during surgery; however, a large amount of bloody pus filled the mastoid cavity. We performed simple mastoidectomy and drained the pus. We subsequently were able to observe the dark-colored sigmoid sinus through the thin eroded bone (Fig. 2A). We gently skeletonized and removed the bony sinus plate with a diamond burr. Clots and pus appeared on the surface of the sigmoid sinus following removal of the thin bony plate (Fig. 2B). The sigmoid sinus appeared to have been narrowed by the clots from the outside, but no defect was observed within the dural sinus (Fig. 2C). We removed as much clot as possible, and confirmed the restoration of sigmoid sinus blood flow (Fig. 2D). Two drainage tubes were placed in the mastoid cavity and a tympanic ventilation tube was inserted into the tympanic membrane. Bacterial cultures of the otorrhea, intraoperative pus, and clots were negative;

Fig. 2. Photograph of the right mastoid cavity during surgery. A dark-colored sigmoid sinus (white arrow heads) was observed under the thin bony plate (A). Many clots and a large amount of pus were found after the bony plate was removed (B). The dural sinus with an intact blood flow (yellow arrow) was detected under the clots (C). The recovered bulging sigmoid sinus at the conclusion of surgery (D). (For interpretation of the references to color in this figure legend, the reader is referred to the web version of the article.)

Fig. 3. Contrast-enhanced CT 2 days after surgery. Narrowing of the sigmoid sinus has nearly resolved to an almost normal condition (A), but a soft tissue density (arrow heads) is observed lateral to the jugular bulb (B).

however, a blood culture performed at the previous hospital revealed penicillin-intermediate resistant Streptococcus pneumoniae (PISP). The patient’s headache resolved immediately after surgery. CT 2 days after surgery showed the amelioration of sigmoid sinus narrowing (Fig. 3A), but a partial low density remained on the jugular bulb (Fig. 3B). The patient was administered intravenous panipenem/betamipron for 9 days. No other antibiotics, steroids, or anticoagulants were administered. His general condition and laboratory data markedly improved, and he was discharged 9 days after surgery. There have been no complications or signs of recurrent otitis media. CT demonstrated the complete recovery of sigmoid sinus blood flow 5 months later (Fig. 4).

3. Discussion LST was a severe complication of middle ear infection in the pre-antibiotic era. If untreated, mortality was very high. As LST became more widely recognized and as antibiotics and surgical techniques developed mortality decreased to 0–16% [3–6]. Although LST is typically observed in children with acute otitis media, it has also been reported in adults or patients with chronic middle ear disease [7,8]. Because otolaryngologists now rarely encounter this condition, the timely and correct diagnosis of LST has become more difficult. Most patients with LST exhibit severe headaches, as well as nausea and vomiting, following the typical symptoms of acute otitis media [9,10]. Headaches, nausea, and vomiting are all caused by an increase in intracranial pressure (ICP). Increased ICP secondary to otitis media is called otitic hydrocephalus (OH). Neither ventricular dilation nor focal neurological signs, which are both often found in hydrocephalus, have been reported in OH [11,12]. Although the pathophysiology of OH remains unclear, it is often accompanied by LST clinically, especially if blood flow

Fig. 4. Contrast-enhanced CT 5 months after surgery. The soft tissue density around the sigmoid sinus has disappeared and blood flow has completely recovered.

Please cite this article in press as: Takahashi K, et al. A case of lateral sinus insufficiency with acute otitis media: Early surgical intervention for prevention of lateral sinus thrombosis. Auris Nasus Larynx (2015), http://dx.doi.org/10.1016/j.anl.2015.04.007

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insufficiency occurs in the dominant lateral sinus [11,13]. OH also causes abducens nerve palsy and papilledema and patients with LST frequently develop diplopia and/or photophobia [9,12]. It is important to recognize abnormalities in the patient’s vision as signs of LST. Identifying atypical symptoms in the clinical course of otitis media is important for an accurate and timely diagnosis of LST. High-resolution and contrast-enhanced CT of the temporal bone should be performed if LST is suspected. In dural sinus thrombosis including LST, contrast-enhanced CT often shows an empty delta sign [14,15]. This sign consists of a triangular area of enhancement with a low attenuating center. The enhanced area reflects the weak maintenance of blood flow and a thickened dura, while the low attenuating center is regarded as the thrombus. In our case, the enhanced area shifted medially with a low density laterally. This was not the empty delta sign typical of the intrasinus thrombus. The pathophysiology of LST remains unclear, especially the mechanism of thrombus formation in the dural venous sinus. A previous study reported that LST was preceded by an extradural abscess in most cases, while other cases have developed from retrograde phlebitis in the mastoid emissary vein [16]. Although our patient did not develop intrasinus thrombosis, he demonstrated nearly the same symptoms. We believe that if the patient went untreated, he would have developed full-blown LST. Our case allows speculation on the pathophysiology of LST. The dural venous sinuses are intracranial blood drainage channels that exist between two layers of the dura mater. The outer layer serves as the endocranium, which adheres tightly to the periosteum of the skull (periosteal layer). The inner layer surrounds the arachnoid mater (meningeal layer). The walls of the dural venous sinuses are composed of dura mater lined with a vascular endothelium, and blood flows between these two layers. They differ from endocranial veins because they lack a tunica media. Therefore, once inflammation destroys the mastoid bone, it spreads directly to the vascular endothelium. As a result, vascular permeability increases, causing hemorrhage per diapedesis. Hemorrhage between the bony sinus plate and dural venous sinus narrows the sinus itself, which then causes blood stasis. We believe that this is a step just prior to full-blown LST as seen in our case (Fig. 5A). Damage to the vascular endothelium also activates a coagulation cascade intravenously. These phenomena of blood stasis by hemorrhage per diapedesis and the activation of a coagulation cascade lead to intrasinus thrombus (Fig. 5B). This may represent another pathophysiological mechanism for the development of LST. The aim of surgery should be to eradicate the infectious disease in the mastoid cavity and, if possible, recanalize the lateral sinus. Even when a patient is thought to have LST, they may clinically have an LST-like pathological condition simply due to an extradural clot pressing on the lateral sinus rather than congestion due to a sinus thrombus, as in our case. Therefore, we believe that early diagnosis and surgical intervention before the patient develops full-blown LST are key for the treatment of this disease. We propose that simple mastoidectomy, in which the bony sinus plate, perisinusal abscess, and clots on the sigmoid sinus are removed as much as possible, should be the primary surgery. Though metastatic emboli, represented by pulmonary thrombosis, often occurred in the pre-antibiotic era, they have rarely occurred since the introduction of antibiotics [3]. Further, it is not necessary to totally remove clots that remain on the sinus because they disappear once the infection is controlled [12,17]. If the inside of the lateral sinus is likely to be completely thrombosed, lateral sinus puncture with aspiration, thrombus removal with incision, jugular vein ligation to prevent the spread of infection, and anticoagulant therapy may be considered. These procedures may pose some risk for intracranial complications and compelling evidence of their effectiveness has not yet been demonstrated [3,12,18–20]. In our

3

Fig. 5. (A) The schema of pathophysiology in our case. This condition seems to be an initial stage of lateral sinus insufficiency caused by hemorrhage per diapedesis secondary to damage to the endothelial cells. This may lead to full-blown LST (B). (B) A second schema for a pathophysiological mechanism for the formation of intramural thrombus. This condition is generally known as full-blown LST.

case, the clots existed only outside the dural sinus, and the patient required only the administration of antibiotics after surgery; the use of these advanced procedures was not indicated. 4. Conclusion  We reported a case of acute lateral sinus insufficiency mimicking the symptoms of LST, which was thought to represent a stage just prior to full-blown LST.  Based on the anatomical features of the dural venous sinus, hemorrhage per diapedesis due to endothelial cell damage causes blood stasis. This may lead to the development of otitic hydrocephalus.  After developing blood stasis due to hemorrhage per diapedesis, endothelial cell damage may activate a coagulation cascade leading to full-blown LST. This may represent another pathophysiological mechanism for the development of LST.  If LST is suspected, but the empty delta sign is absent on CT scan, mastoidectomy should be performed as soon as possible. Conflict of interest There were no conflicts of interest with regard to this work. Acknowledgements This work received no specific grant from any funding agency, commercial, or not-for-profit sector. References [1] Dhooge IJ, Albers FW, Van Cauwenberge PB. Intratemporal and intracranial complications of acute suppurative otitis media in children: renewed interest. Int J Pediatr Otorhinolaryngol 1999;49(Suppl. 1):S109–14. [2] Go C, Bernstein JM, de Jong AL, Sulek M, Friedman EM. Intracranial complications of acute mastoiditis. Int J Pediatr Otorhinolaryngol 2000;52:143–8. [3] Ooi EH, Hilton M, Hunter G. Management of lateral sinus thrombosis: update and literature review. J Laryngol Otol 2003;117:932–9.

Please cite this article in press as: Takahashi K, et al. A case of lateral sinus insufficiency with acute otitis media: Early surgical intervention for prevention of lateral sinus thrombosis. Auris Nasus Larynx (2015), http://dx.doi.org/10.1016/j.anl.2015.04.007

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K. Takahashi et al. / Auris Nasus Larynx xxx (2015) xxx–xxx

[4] Samuel J, Fernandes CM, Steinberg JL. Intracranial otogenic complications: a persisting problem. Laryngoscope 1986;96:272–8. [5] Mathews TJ. Lateral sinus pathology (22 cases managed at Groote Schuur Hospital). J Laryngol Otol 1988;102:118–20. [6] Teichgraeber JF, Per-Lee JH, Turner JS. Lateral sinus thrombosis: a modern perspective. Laryngoscope 1982;92(Pt 1):744–51. [7] Leskinen K, Jero J. Acute complications of otitis media in adults. Clin Otolaryngol 2005;30:511–6. [8] Manolidis S, Kutz JW. Diagnosis and management of lateral sinus thrombosis. Otol Neurotol 2005;26:1045–51. [9] 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;73:581–4. [10] 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;123:709–13. [11] Tomkinson A, Mills RG, Cantrell PJ. The pathophysiology of otitic hydrocephalus. J Laryngol Otol 1997;111:757–9. [12] Kuczkowski J, Dubaniewicz-Wybieralska M, Przewoz´ny T, Narozny W, Mikaszewski B. Otitic hydrocephalus associated with lateral sinus thrombosis and acute mastoiditis in children. Int J Pediatr Otorhinolaryngol 2006;70:1817–23.

[13] Doyle KJ, Brackmann DE, House JR. Pathogenesis of otitic hydrocephalus: clinical evidence in support of Symonds’ (1937) theory. Otolaryngol Head Neck Surg 1994;111(Pt 1):323–7. [14] Kaplan DM, Kraus M, Puterman M, Niv A, Leiberman A, Fliss DM. Otogenic lateral sinus thrombosis in children. Int J Pediatr Otorhinolaryngol 1999;49:177–83. [15] Rao KC, Knipp HC, Wagner EJ. Computed tomographic findings in cerebral sinus and venous thrombosis. Radiology 1981;140:391–8. [16] Smith JA, Danner CJ. Complications of chronic otitis media and cholesteatoma. Otolaryngol Clin North Am 2006;39:1237–55. [17] Davison SP, Facer GW, McGough PF, McCaffrey TV, Reder PA. Use of magnetic resonance imaging and magnetic resonance angiography in diagnosis of sigmoid sinus thrombosis. Ear Nose Throat J 1997;76:436–41. [18] Shah UK, Jubelirer TF, Fish JD, Elden LM. A caution regarding the use of lowmolecular weight heparin in pediatric otogenic lateral sinus thrombosis. Int J Pediatr Otorhinolaryngol 2007;71:347–51. [19] Wong I, Kozak FK, Poskitt K, Ludemann JP, Harriman M. Pediatric lateral sinus thrombosis: retrospective case series and literature review. J Otolaryngol 2005;34:79–85. [20] Mallur PS, Harirchian S, Lalwani AK. Preoperative and postoperative intracranial complications of acute mastoiditis. Ann Otol Rhinol Laryngol 2009;118:118–23.

Please cite this article in press as: Takahashi K, et al. A case of lateral sinus insufficiency with acute otitis media: Early surgical intervention for prevention of lateral sinus thrombosis. Auris Nasus Larynx (2015), http://dx.doi.org/10.1016/j.anl.2015.04.007

A case of lateral sinus insufficiency with acute otitis media: Early surgical intervention for prevention of lateral sinus thrombosis.

To describe a case of lateral sinus insufficiency, presumably occurring just prior to lateral sinus thrombosis (LST), and to discuss the importance of...
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