Management of otogenic lateral sinus thrombosis

Auris Nasus Larynx 41 (2014) 143–147

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Management of otogenic lateral sinus thrombosis Gautam Bir Singh a,b,*, Anil K. Rai a, Sarvejeet Singh a, Radhamadhab Sahu a, Rubeena Arora a a b

Department of Otorhinolaryngology & Head-Neck Surgery, Lady Hardinge Medical College & Associated Hospitals, New Delhi 110001, India VMMC & Safdarjung Hospital, New Delhi, India

A R T I C L E I N F O

A B S T R A C T

Article history: Received 8 March 2013 Accepted 3 October 2013 Available online 20 November 2013

Objective: To evaluate the changing clinical course and trends in management of otogenic lateral sinus thrombosis (OLST), in view of the rarity of the said lesion and antibiotic abuse. Methods: A retrospective case study was done in 6 patients referred to our tertiary care centre with OLST over a period of three years from May 2007 to May 2010: Department of Otorhinolaryngology, Vardhman Mahavir Medical College and Safdarjung Hospital, New Delhi – a tertiary care university teaching hospital under central government of India. Medical records of all the patients were scrutinized and data pertaining to patient profile, type of chronic suppurative otitis media (CSOM), clinical course with any other complication and management were tabulated. The said data was scientifically analyzed w.r.t. current medical literature on the subject. Results: Clinically, it was observed that all patients had protracted CSOM – attico antral type, with history of rampant misuse of antibiotics. The attack of OLST in each case was triggered off by an acute attack of suppurative otitis media. Moreover, in all the cases OLST was masquerading as an intra-cranial complication, with no specific clinical features thereby causing missed diagnosis initially. All these patients were diagnosed by CT scan, and underwent modified radical mastoidectomy (MRM) with needle aspiration of sinus under antibiotic cover. The said treatment protocol resulted in excellent prognosis with no morbidity or mortality whatsoever. Pathologically, cholesteatoma was detected in 5 of the six cases with one case having only granulations. Conclusions: In this era of antibiotic abuse, the clinical presentation of OLST has altered substantially, and mimics other intra-cranial complication with vague signs and symptoms. There are conflicting views in contemporary medical literature regarding management of OLST. In our experience, MRM with needle aspiration under antibiotic cover is the treatment of choice for all cases of OLST associated with CSOM attico antral disease/cholesteatoma disease. ß 2013 Elsevier Ireland Ltd. All rights reserved.

Keywords: Otogenic lateral sinus thrombosis Chronic suppurative otitis media

1. Introduction Otogenic lateral sinus thrombosis (OLST) is a rare intracranial complication of suppurative otitis media [1–3]. The treatment of suppurative otitis media with highly potent antibiotics in the modern era has led to the dramatic decrease in the incidence of the said complication [1–4]. Thus, a marked paucity of literature exists on the cited subject in modern otology. However, the clinical course of this dreaded complication has also been altered by these antibiotics leading to a more subtle presentation of OLST. In addition, it is still associated with a mortality and morbidity of 10% and 30% respectively [2,3]. With this background, we present our

modest experience in a series of 6 patients who successfully underwent treatment for OLST. This study highlights the paradigm shift in the clinical course and management of OLST. 2. Materials and method The clinical records of 6 patients of OLST, which were referred to our institution Vardhman Mahavir Medical College and Safdarjung Hospital, New Delhi: a tertiary care university teaching hospital over a period of three years i.e. may 2007 to may 2010 were analyzed in this retrospective study design. 3. Results

* Corresponding author at: House No: 1433/Sector: 15, Faridabad 121007 (National Capital Region), Haryana, India. Tel.: +91 0129 4012368/4007550; mobile: +91 9818836242. E-mail addresses: [email protected], [email protected] (G.B. Singh). 0385-8146/$ – see front matter ß 2013 Elsevier Ireland Ltd. All rights reserved. http://dx.doi.org/10.1016/j.anl.2013.10.005

All the 6 cases that reported to us were young and less than 25 years of age [mean age: 19 years], 2 were in paediatric age group, i.e. less than 12 years of age, rest were all adults. There were 2 females and 4 males in the study group.

G.B. Singh et al. / Auris Nasus Larynx 41 (2014) 143–147

144 Table 1 Otogenic lateral sinus thrombosis [clinical course]. Patient profile [age/sex]

Social background

Ear diagnosis

Case I 15 M

Rural

Case II 12 M Case III 25F Case IV 20 M Case V 22 F

Rural

Case VI 10 M

Rural

CSOM-B/E R/E-AA L/E-TT CSOM-R/E AA CSOM-L/E AA CSOM-L/E AA CSOM-B/E L/E-AA R/E-TT CSOM-B/E R/E-AA L/E-TT

Rural Rural Rural

H/O antibiotic intake

S/S of intracranial complication

Referral period (days)

6

Yes

Present

10

3

Yes

Present

12

3

Yes

Present

14

10

Yes

Present

23

8

Yes

Present

10

5

Yes

Present

16

Duration of CSOM (yrs)

M: male; F: female; CSOM: chronic suppurative otitis media; B/E: both ear; L/E: left ear; R/E: right ear; AA: attico antral; TT: tubotympanic; yrs: years. Table 2 Laboratory investigations [meninigitis]. Case profile

Case I 15 M Case II 12 M Case III 25 F Case IV 20 M Case V 22 F Case VI 10 M

CSF Gross examination

Proteins (normal: 15–45 mg/dl)

Glucose (normal: 40–70 mg/dl)

Leucocyte counts (normal: 0–5 cu mm) neutrophil

Turbid

60

18

Turbid

80

20

Turbid

76

16

Turbid

60

18

Turbid

73

14

Turbid

62

12

650 N92 550 N92 1010 N84 650 N88 750 N93 2000 N90

hs-CRP (high sensitivity C-reactive proteins) (normal: 0.00–0.30 mg/dl)

30 24 Not available Not available 20 28

Note: All the case had pyogenic meningitis: CSF had an increase in proteins and decrease in glucose with leucocytosis [predominantly nuetrophills]

All these cases had protracted chronic suppurative otitis media (CSOM) with a history of intermittent discharge for years [for a minimum 3 years and maximum of 10 years, mean being: 5 years and 10 months]. All the patients gave the history of use of antibiotics during their protracted course of disease to stop the discharge. Unfortunately, the details of this antibiotic treatment were not available: as most of our patients belong to a rural background, and had no medical record of their treatment. It was an attack of acute suppurative otitis media in each case that finally led to the aetiopathogenesis of lateral sinus thrombosis. Initially all the patients had symptoms of headache, nausea and vomiting with dizziness or vertigo along with ear discharge which prompted them to seek medical advice (Table 1). All of our cases had fever at some point of time during the course of their illness, but none had the characteristic picket fence fever mentioned in the medical text. As we could only examine the medical records of the patients, only two cases had a positive history of neck rigidity, and only in one case brudzinskis sign was documented to be positive. Initially all cases were diagnosed as meningitis and started on I/V antibiotics. Table 2 documents the CSF findings and hs-CRF, i.e. high sensitivity C reactive proteins values. The evaluation of the table delineates the severity of the infection in these cases However, later on as the condition of the patient deteriorated and CT scan revealed a thrombus in the sinus; these cases were referred to our ENT department. In two cases the patients had multiple referrals, i.e. the patient had visited two or more than two hospitals before being referred to a tertiary care institute. The time from the start of aforesaid symptoms of intracranial involvement to the transfer to our tertiary care hospital varied from a minimum of 10

days to a maximum of 23 days, mean: 14 days (Table 1). Two cases were also referred from the departments of our institution, one from neurosurgery and the other from general medicine. On examination, three cases had bilateral CSOM; other three had unilateral ear disease. In all the patients, the ear on the side of lateral sinus thrombosis was having attico antral perforation. The other ear in bilateral CSOM patients was harbouring mucosal disease only (Table 1). One female patient had a neck swelling with cord sign positive (Fig. 1). Cord sign is defined as an induration corresponding to the

Fig. 1. Patient’s photograph showing swelling [abscess] in neck.

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Table 3 Otogenic lateral sinus thrombosis [management]. Case profile

Diagnosis OLST

Hypercoagubility

Pus culture

Any other complication

Treatment

Case I 15 M Case II 12 M Case III 25 F

CT scan

Absent

Sterile

Meningitis

MRM + Needle aspiration of sinus + Tympanoplasty type III + antibiotics

CT scan

Absent

Sterile

MRM + Needle aspiration of sinus + Tympanoplasty type III + antibiotics

CT scan

Absent

Sterile

Case IV 20 M Case V 22 F Case VI 10 M

CT scan

Absent

Sterile

Meningitis + IJV thrombosis Meningitis + IJV thrombosis + carotid space abscess Meningitis

CT scan

Absent

Sterile

Meningitis

MRM + Needle aspiration of sinus + Tympanoplasty type III + antibiotics

CT scan

Absent

Sterile

Meningitis

MRM + Needle aspiration of sinus + Tympanoplasty type III + antibiotics

MRM + Needle aspiration of sinus + Tympanoplasty type III + antibiotics

MRM + Needle aspiration of sinus + Tympanoplasty type III+ antibiotics

M: male; F: female; OLST: otogenic lateral sinus thrombosis; IJV: internal jugular venous thrombosis; MRM: modified radical mastoidectomy.

course of internal jugular vein beneath the anterior border of sternocleidomastoid [5]. This patient was diagnosed to have an associated carotid abscess with IJV thrombosis (Fig. 2). One paediatric patient also had an IJV thrombosis (Table 3). All the patients in our medical institution underwent CT scan (Table 3) on the basis of which the final diagnosis of OLST was made (Fig. 3). In accordance with our treatment protocol, all the patients underwent: modified radical mastoidectomy with delineation of the sinus and needle aspiration [till frank blood is aspirated] under intravenous antibiotics (amoxycillin + clavulinic acid, along with metrogyl) for 14 days, followed by systemic antibiotics for another 3 weeks. No anti-coagulent treatment was given in any patient. The erosion of sinus plate was seen only in three cases and all these cases had perisinus abscess. Pathologically, of the six patients 5 patients had cholesteatoma disease, one patient had frank granulations and one patient had granulations along with cholesteatoma. A brief synopsis of the surgical management is given in Table 4. All the patients had an excellent outcome with no morbidity related to OLST. They were subsequently discharged after 3 weeks on oral antibiotics and were kept on a regular follow-up for 3 months in the ENT OPD, with no untoward incident to report.

probably attributed to the ignorance about the seriousness of persistent ear discharge in our rural population. From this case study it is clear that OLST has no distinct clinical expression; a fact widely accepted in medical literature and attributed to the early use of potent antibiotics for treatment of suppurative otitis media [3,9–11]. All the cases in this series were diagnosed by accident during the investigative image study. However, all our patients prior to having LST had meningitis with clear clinical features of intra cranial complication. It is thus prudent to re-evaluate all cases of intracranial complication with CSOM not responding to medical treatment. From the above account it is clear that cases of OLST may not be always seen

4. Discussion The study and analysis of the aforesaid data brings forth some interesting facts regarding the complication OLST. All the CSOM patients who were referred to us with lateral sinus thrombosis were young patients (age <25 years). This is in no way to suggest that young patients have a greater chance of having this complication, but it is the protracted course of the disease which probably made these young patients vulnerable to the said complication. Moreover all patients had been taking antibiotics off and on [no clinical record of the medicines prescribed was available], many a time’s self medication was practiced in rural areas. Whether this rampant misuse of antibiotics led to a cultivation of resistant flora in the ear, subsequently causing this complication in this series is however open to debate. Nevertheless, it would be pertinent to note that antibiotic resistance has been recognized as a main factor for aetiopathogenesis of OLST [2,6]. Further, it would also be important to note that an acute attack of suppurative otitis media with characteristic features of pain and discharge did initiate the complication in all the cases. Several other studies have also ascertained the role of acute suppurative otitis media in intracranial complications [7–9]. This delineates the importance of judicious management of longstanding CSOM presenting with acute ear discharge. The reason why suppurative otitis media complications are still encountered in developing countries is

Fig. 2. CT scan showing internal jugular venous thrombosis with carotid abscess.

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Fig. 3. CT scan showing lateral sinus thrombosis.

primarily by an ENT surgeon, but by a medical/neurospecialist as the complication usually masquerades as an intracranial complication. This study thus emphasizes the importance of a close inter specialist co-operation and heightened awareness of the changing presentation of OLST, so that delayed diagnosis as a result of misdiagnosis is avoided in this life threatening complication. Another point merits mention: although literature is replete with references that suggest hypercoagulability as an important predisposing factor for OLST [1,2,9,12,13], yet no hypercoagulability state was detected in any patient in this case series. All the patients of suspected lateral sinus thrombosis in our institute undergo a CT scan to confirm diagnosis. In this series of patients CT scan served as a gold standard, though MRI is regarded more sensitive to diagnose OLST [2,4,14]. Despite the advantages of MRI, its cost and selective availability especially in developing countries, limits its use. It is thus mandatory only in those suspicious cases where the performed CT scan fails to demonstrate the thrombus. This view has also been endorsed by other otologists [4]. MR venography/arteriography now supersedes all other investigations for the identification of the thrombus in the sigmoid sinus as evidenced by flow void [14,15]. Interestingly, the characteristic delta sign [central non enhancing clot surrounded by enhancing dural sinus wall] for OLST mentioned in literature was not detected in any case [12]. Though our treatment protocol (MRM + needle aspiration under antibiotic cover) gave us excellent results in all the cases, yet there is considerable controversy regarding management of OLST. The recent review of literature now postulates that the basic principal for management of OLST is the treatment of the source of infection. It is reasoned that formation of thrombus is a protective mechanism attempting to localize infection and natural history of OLST is of resolution, thus once the source of infection is eradicated the thrombus resolves [4,9]. This has led to a strong argument against exploration of the sigmoid sinus and many otologists have reported a favourable outcome in patients of OLST where the clot was left untouched [16–18]. There are also sporadic reports of management of OLST with antibiotics and anticoagulant therapy alone with no mastoid surgery at all in cases of CSOM-mucosal disease, especially in children [4,19,20]. Nevertheless, we would like to emphasize that in cases of CSOM-cholesteatoma disease with OLST: mastoid

Table 4 Synopsis of surgical treatment. Treatment protocol: all patients underwent modified radical mastoidectomy + needle aspiration of sinus + functional Type III tympanoplasty + antibiotics Case profile

Ear pathology

Case I 15 M Case II 12 M

Cholesteatoma: antrum, aditus and attic Cholesteatoma: antrum, aditus, posterior superior part attic and adjoining area of hypotympanum involving sinus tympani and facial recess Cholesteatoma: antrum, aditus and attic along with post superior quadrant of hypoympanum Granulations: antrum, aditus, attic, hypotympanum and ant extending upto ET area Cholesteatoma: antrum, aditus and attic Cholesteatoma + gganulations: antrum aditus and PSQ of hypotympanum involving sinus tympani and facial recess

Case III 25 F

Case IV 20 M Case V 22 F Case VI 10 M

Post operative period: in all the cases was uneventful.

Ossicular status

Tympanoplasty

Necrosed: Handle of malleus, long process of incus Necrosed: handle of malleus, long process of incus

Functional Type III [temporalis fascia graft on stapes] Functional Type III [temporalis fascia graft on stapes]

Present

Necrosed: long pocess of incus and stapes suprastructure (partially)

Functional Type III [temporalis fascia graft on remanent stapes suprastructure]

Absent

Except stapes (partial necrosis of suprastructure seen) no other ossicle found

Absent

Except stapes no other ossicle found

Present

Handle of malleus, almost whole of incus (partially eroded body of incus seen) and stapes suprastructure necrosed

Functional Type III [temporalis fascia graft on remanent stapes suprastructure] Functional Type III [temporalis fascia graft on stapes] Augmented Type III; a cartilage placed on stapes footplate and graft put on it

Erosion of sinus plate with perisinus abscess Absent Present

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exploration is the treatment of choice. However, in view of the aforesaid facts it is not possible to discern whether all cases should undergo sinus exploration for evacuation of the clot. This controversial aspect of treatment certainly warrants research in accordance with the concept of evidence based medicine. Interestingly, two of our cases had internal jugular venous (IJV) thrombosis. One of these cases even had an associated carotid abscess probably attributed to reactionary inflammatory response to the thrombus and IJV infection. This neck swelling was an ominous sign of advanced stage of OLST, clearly missed by all treating physicians. Both the cases were managed with mastoid surgery with needle aspiration of sinus under I/V antibiotic cover. No anticoagulant therapy or IJV ligation was required. The use of anti-coagulants in LST is controversial [2,3,12]. The general consensus is that, concomitant use of anti-coagulants with antibiotics is best avoided in septic conditions as it leads to dissemination of the emboli. Moreover, the absence of hypercoagulable state in the said case also prompted us to eliminate anticoagulants all together from the management protocol. The medical literature however, cites the importance of this modality of treatment to prevent complications attributed to thrombus persistence and its possible propagation [2,21]. Hence, whether the early prophylactic use of anti-coagulant therapy could have averted the complication of IJV thrombosis and carotid space abscess is speculative. In this context it would be important to note that in modern otology, IJV ligation is reserved for persistent septicemia even after mastoidectomy or septic pulmonary or extra pulmonary embolization [22]. 5. Conclusions From the preceding discussion we conclude that: 1. Though antibiotic resistance is regarded as an important factor for the aetiopathogenesis of OLST, the same is difficult to discern on the basis of our study in accordance with evidence based medicine. However, the protracted course of CSOM and history of rampant abuse of antibiotics in this series does indicate that this could be an important factor in the aetiopathogenesis of OLST. 2. An attack of acute supurative otitis media is an important predisposing factor for OLST. 3. Hypercoagulability was not found to be an important factor in aetiopathogenesis of OLST. 4. All patients of OLST present with non-specific intra-cranial symptoms. Hence, a close professional co-operation between ENT surgeon and physician is mandatory for the diagnosis of this complication. 5. A CT scan usually sufficient to confirm the diagnosis. 6. As most of the cases are of attico antral disease with cholesteatoma: a mastoid exploration is a must, but there is ambiguity regarding the exploration of sinus in medical literature. 7. As patients of OLST with IJV were managed successfully without any anticoagulent treatment, no definite role for this modality of treatment could be defined in this study.

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Conflict of interest None of the authors has any conflict of interest, financial or otherwise. All the doctors in this study are employed by ‘‘Government of India’’ and paid salary by the Government of India, and the medical college is a ‘‘Central Government Institution’’. The present study was a thesis conducted in the department of ENT. References [1] Levine SC, Desouza C. Intracranial complications of otitis media. In: Gulya JA, Minor LB, Poed DS, editors. Glasscock-Shambaugh Surgery of the Ear. 6th ed., Connecticut: Peoples Medical Publishing House; 2010. p. 458. [2] Bianchini C, Aimoni C, Ceruti S, Grasso DL, Martini A. Lateral sinus thrombosis as a complication of acute mastoiditis. Acta Otorhinolaryngol Italica 2008;28: 30–3. [3] Iseri M, Aydin O, Ustundag E, Keskin G, Almac A. Management of lateral sinus thrombosis in chronic otitis media. Otol Neurotol 2006;27:1098–103. [4] Tov EE, Leiberman A, Shelef I, Kaplan DM. Conservative non surgical treatment of a child with otogenic lateral sinus thrombosis. Am J Otolaryngol 2008;29: 138–41. [5] Tovi F, Fliss DM, Noyek AM. Septic jugular venous thrombosis. J Otolaryngol 1993;22:415–20. [6] Luntz M, Brodsky A, Nusen S. Acute mastoiditis-antibiotic era: a multicentric study. Int J Paediatr Otorhinolaryngol 2001;57:1–9. [7] 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;115:307–10. [8] Garcia RD, Baker AS, Cunnigham MJ, Weber AL. Lateral sinus thrombosis associated with otitis media and mastoiditis in children. Pediatr Infect Dis J 1995;14:617–23. [9] Redaelli De Zenis LO, Gasparotti, Campovecchi C, Annibale G, Barezzani MG. Internal jugular vein thrombosis associated with acute mastoiditis in a pediatric patient. Otol Neurotol 2006;27:937–44. [10] Jose J, Coatesworth PA, Anthony R, Reilly PG. Life threatening complication after partially treated mastoiditis. Br Med J 2003;327:41–2. [11] Rocha JL, Kondo W, Garcia CM, Baptista MI, Buchele G, da Cunha CA, et al. Central venous sinus thrombosis following mastoiditis: report of 4 cases and literature review. Braz J infect Dis 2000;4:307–12. [12] Bradley DT, Hashisaki GT, Mason JC. Otogenic sigmoid sinus thrombosis: what is the role of anticoagulation? Laryngoscope 2002;112:1726–9. [13] Unsal EE, Ensari S, Koc C. A rare case and serious complication of chronic otitis media: lateral sinus thrombosis. Auris Nasus Larynx 2003;30:279–82. [14] Penido NO, Toledo RN, Silveria PAL, Lei Muhoz MS, Testa JRG, Cruz OCM. Sigmoid sinus thrombosis associated to chronic otitis media. Rev Bras Otorrinolaringol 2007;73(2):165–70. [15] Davison SP, Facer GW, McGough PF, McCaffrey TV, Rder PA. Use of magnetic resonance imaging and magnetic resonance angiography in diagnosis of sigmoid sinus thrombosis. Ear Nose Throat JS 1997;76(7):436–41. [16] Ooi EH, Hilton M. Management of lateral sinus thrombosis: update and literature review. J Laryngol Otol 2003;117:932–9. [17] Syms MJ, Tsai PD, Holten MR. Management of lateral sinus thrombosis. Laryngoscope 1999;109(10):1616–20. [18] Kutluhan A, Kuris M, Yurttas V, Kyroolu AF, Unal O. When can lateral sinus thrombosis be treated conservatively? J Otolaryngol 2003;33:107–10. [19] 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;257:1–5. [20] Wong I, Kozak FK, Poskitt K, Ludemann JP, Harrimen M. Paediatric lateral sinus thrombosis: retrospective case series and literature review. J Otolaryngol 2005;34:79–85. [21] Agarwal A, lowry P, Issacson G. Natural history of sigmoid sinus thrombosis. Ann Otol Rhinol Laryngol 2003;112(2):191–4. [22] Neto JFL, Saffer M, Rotta FT, Arrarte JLF, Brinckmann CA, Ferriera P. Lateral sinus thrombosis and cervical abscess complicating cholestetoma in children: case report and review. Int J Pediatric Otorhinolaryngol 1998;42:263–9.