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Spontaneous bilateral and concurrent cerebrospinal fluid otorrhoea: case presentation and literature review

Published online by Cambridge University Press:  05 July 2016

T Tikka*
Affiliation:
Department of ENT, New Cross Hospital, Royal Wolverhampton Hospitals NHS Trust, UK
A Opeodu
Affiliation:
Department of ENT, Queen Elizabeth Hospital, University Hospitals Birmingham NHS Foundation Trust, UK
R Irving
Affiliation:
Department of ENT, Queen Elizabeth Hospital, University Hospitals Birmingham NHS Foundation Trust, UK
J Murphy
Affiliation:
Department of ENT, New Cross Hospital, Royal Wolverhampton Hospitals NHS Trust, UK
*
Address for correspondence: Miss Theofano Tikka, Department of ENT, New Cross Hospital, Wolverhampton WV10 0QP, UK E-mail: [email protected]

Abstract

Background:

Cerebrospinal fluid otorrhoea is a rare entity. Only a few cases of spontaneous bilateral cerebrospinal fluid otorrhoea have been reported. In all cases, there was a definite time interval between the two (left and right) presentations.

Objectives:

To raise awareness and report on the very rare entity of bilateral spontaneous cerebrospinal fluid otorrhoea.

Case report:

This paper reports the case of a bilateral, synchronous, spontaneous cerebrospinal fluid otorrhoea in a 44-year-old female. The patient had grommets surgically inserted on two separate occasions for treatment of otitis media with effusion, and received several courses of oral and topical antibiotics. Five years following the patient's initial presentation, a suspicion of concurrent bilateral cerebrospinal fluid otorrhoea was raised. The otorrhoea sample collected proved to be cerebrospinal fluid. Cross-sectional imaging revealed bilateral defects in the tegmen tympani of the skull base. She underwent staged middle fossa craniotomies to repair the defects.

Conclusion:

Careful observation of the middle-ear fluid characteristics following myringotomy can allow for prompt diagnosis.

Type
Clinical Record
Copyright
Copyright © JLO (1984) Limited 2016 

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References

1 Standring, S, ed. Gray's Anatomy: The Anatomical Basis of Clinical Practice, 40th edn. China: Elsevier, 2008 Google Scholar
2 Mokri, B. Spontaneous CSF leaks: low CSF volume syndromes. Neurol Clin 2014;32:397422 Google Scholar
3 Graff-Radford, SB, Schievink, WI. High-pressure headaches, low-pressure syndromes, and CSF leaks: diagnosis and management. Headache 2014;54:394401 CrossRefGoogle ScholarPubMed
4 Soler, ZM, Schlosser, RJ. Spontaneous cerebrospinal fluid leak and management of intracranial pressure. Adv Otorhinolaryngol 2013;74:92103 Google Scholar
5 Kou, YF, Allen, KP, Isaacson, B. Recurrent meningitis secondary to a petrous apex meningocele. Am J Otolaryngol 2014;35:405–7Google Scholar
6 Battal, B, Castillo, M. Brain herniations into the dural venous sinuses or calvarium: MRI of a recently recognized entity. Neuroradiol J 2014;27:5562 CrossRefGoogle ScholarPubMed
7 Rao, N, Redleaf, M. Spontaneous middle cranial fossa cerebrospinal fluid otorrhea in adults. Laryngoscope 2016;126:464–8Google Scholar
8 Zakaryan, A, Poulsgaard, L, Hollander, C, Fugleholm, K. Spontaneous cerebrospinal fluid otorrhea from a persistent tympanomeningeal fissure presenting as recurrent serous otitis media. J Neurol Surg Rep 2015;76:e11719 Google Scholar
9 Son, HJ, Karkas, A, Buchanan, P, Giurintano, JP, Theodosopoulos, P, Pensak, ML et al. Spontaneous cerebrospinal fluid effusion of the temporal bone: repair, audiological outcomes, and obesity. Laryngoscope 2014;124:1204–8Google Scholar
10 Allen, KP, Perez, CL, Isaacson, B, Roland, PS, Duong, TT, Kutz, JW. Superior semicircular canal dehiscence in patients with spontaneous cerebrospinal fluid otorrhea. Otolaryngol Head Neck Surg 2012;147:1120–4CrossRefGoogle ScholarPubMed
11 Rajkumar, KT, Orabi, AA, Timms, MS. Spontaneous cerebrospinal fluid leak presenting as unilateral (left-sided) middle ear effusion. Ear Nose Throat J 2008;87:7980 Google Scholar
12 Brown, NE, Grundfast, KM, Jabre, A, Megerian, CA, O'Malley, BW Jr, Rosenberg, SI. Diagnosis and management of spontaneous cerebrospinal fluid--middle ear effusion and otorrhea. Laryngoscope 2004;114:800–5Google Scholar
13 Kutz, JW Jr, Husain, IA, Isaacson, B, Roland, PS. Management of spontaneous cerebrospinal fluid otorrhea. Laryngoscope 2008;118:2195–9Google Scholar
14 Wills, AD, Biggs, N. External auditory canal meningoencephalocele with spontaneous cerebrospinal otorrhea. Otolaryngol Head Neck Surg 2008;139:478–9Google Scholar
15 Toh, A, De, R. Spontaneous cerebrospinal fluid otorrhoea presenting as otitis externa. Eur Arch Otorhinolaryngol 2007;264:689–91Google Scholar
16 Rao, AK, Merenda, DM, Wetmore, SJ. Diagnosis and management of spontaneous cerebrospinal fluid otorrhea. Otol Neurotol. 2005;26:1171–5Google Scholar
17 Teo, DT, Tan, TY, Eng, SP, Chan, YM. Spontaneous cerebrospinal fluid otorrhoea via oval window: an obscure cause of recurrent meningitis. J Laryngol Otol 2004;118:717–20Google Scholar
18 Welge-Luessen, A, Probst, R. Spontaneous cerebrospinal fluid otorrhea in the posterior fossa as a rare cause of adult bacterial meningitis. Otolaryngol Head Neck Surg 2004;130:375–7CrossRefGoogle ScholarPubMed
19 Gacek, RR, Gacek, MR, Tart, R. Adult spontaneous cerebrospinal fluid otorrhea: diagnosis and management. Am J Otol 1999;20:770–6Google ScholarPubMed
20 Kuhweide, R, Casselman, JW. Spontaneous cerebrospinal fluid otorrhea from a tegmen defect: transmastoid repair with minicraniotomy. Ann Otol Rhinol Laryngol 1999;108:653–8Google Scholar
21 May, JS, Mikus, JL, Matthews, BL, Browne, JD. Spontaneous cerebrospinal fluid otorrhea from defects of the temporal bone: a rare entity? Am J Otol 1995;16:765–71Google Scholar
22 Wetmore, SJ, Herrmann, P, Fisch, U. Spontaneous cerebrospinal fluid otorrhea. Am J Otol 1987;8:96102 Google Scholar
23 Myer, CM 3rd, Miller, GW, Ball, JB Jr. Spontaneous cerebrospinal fluid otorrhea. Ann Otol Rhinol Laryngol 1985;94:96–7Google Scholar
24 Neely, JG, Neblett, CR, Rose, JE. Diagnosis and treatment of spontaneous cerebrospinal fluid otorrhea. Laryngoscope 1982;92:609–12Google Scholar
25 Patil, S, Trinidade, A, Yung, M, Donnelly, N. Cerebrospinal fluid otorrhoea following grommet insertion: management using a multi-layered graft technique and an important lesson revisited. J Laryngol Otol 2013;127:70–2Google Scholar
26 Ferguson, BJ, Wilkins, RH, Hudson, W, Farmer, J Jr. Spontaneous CSF otorrhea from tegmen and posterior fossa defects. Laryngoscope 1986;96:635–44Google Scholar
27 Merchant, SN, McKenna, MJ. Neurotologic manifestations and treatment of multiple spontaneous tegmental defects. Am J Otol 2000;21:234–9Google Scholar
28 Lundy, LB, Graham, MD, Kartush, JM, LaRouere, MJ. Temporal bone encephalocele and cerebrospinal fluid leaks. Am J Otol 1996;17:461–9Google Scholar
29 Honda, N, Okouchi, Y, Sato, H, Sanuki, T, Hato, N, Yanagihara, N et al. Bilateral spontaneous cerebrospinal fluid otorrhea. Am J Otolaryngol 2004;25:6872 Google Scholar
30 Rotilio, A, Andrioli, GC, Scanarini, M, Zuccarello, M, Fiore, DL. Concurrent spontaneous CSF otorrhea and rhinorrhea. Eur Neurol 1982;21:7783 Google Scholar
31 Yi, HJ, Zhao, LD, Guo, W, Wu, N, Li, JN, Ren, LL et al. The diagnosis and surgical treatment of occult otogenic CSF leakage. Acta Otolaryngol 2013;133:130–5Google Scholar
32 Park, CW, Jin, BJ, Jeong, SW, Min, HJ, Shin, JH, Lee, SH. CSF otorrhea resulting from osteoradionecrosis of the temporal bone in a patient with recurrent meningioma. Clin Exp Otorhinolaryngol 2009;2:97–9Google Scholar
33 Stenzel, M, Preuss, S, Orloff, L, Jecker, P, Mann, W. Cerebrospinal fluid leaks of temporal bone origin: etiology and management. ORL J Otorhinolaryngol Relat Spec 2005;67:51–5Google Scholar
34 Wolfowitz, B. Spontaneous CSF otorrhea simulating serous otitis. Arch Otolaryngol 1979;105:496–9Google Scholar
35 Vivas, EX, McCall, A, Raz, Y, Fernandez-Miranda, JC, Gardner, P, Hirsch, BE. ICP, BMI, surgical repair, and CSF diversion in patients presenting with spontaneous CSF otorrhea. Otol Neurotol 2014;35:344–7Google Scholar
36 Rosenfeld, E, Dotan, G, Kimchi, TJ, Kesler, A. Spontaneous cerebrospinal fluid otorrhea and rhinorrhea in idiopathic intracranial hypertension patients. J Neuroophthalmol 2013;33:113–16Google Scholar
37 Brainard, L, Chen, DA, Aziz, KM, Hillman, TA. Association of benign intracranial hypertension and spontaneous encephalocele with cerebrospinal fluid leak. Otol Neurotol 2012;33:1621–4CrossRefGoogle ScholarPubMed
38 Stucken, EZ, Selesnick, SH, Brown, KD. The role of obesity in spontaneous temporal bone encephaloceles and CSF leak. Otol Neurotol 2012;33:1412–17Google Scholar
39 LeVay, AJ, Kveton, JF. Relationship between obesity, obstructive sleep apnea, and spontaneous cerebrospinal fluid otorrhea. Laryngoscope 2008;118:275–8Google Scholar
40 Lloyd, KM, DelGaudio, JM, Hudgins, PA. Imaging of skull base cerebrospinal fluid leaks in adults. Radiology 2008;248:725–36Google Scholar
41 Shetty, PG, Shroff, MM, Sahani, DV, Kirtane, MV. Evaluation of high-resolution CT and MR cisternography in the diagnosis of cerebrospinal fluid fistula. Am J Neuroradiol 1998;19:633–9Google Scholar
42 Komotar, RJ, Starke, RM, Raper, DM, Anand, VK, Schwartz, TH. Endoscopic endonasal versus open repair of anterior skull base CSF leak, meningocele, and encephalocele: a systematic review of outcomes. J Neurol Surg A Cent Eur Neurosurg 2013;74:239–50CrossRefGoogle ScholarPubMed
43 Pelosi, S, Bederson, JB, Smouha, EE. Cerebrospinal fluid leaks of temporal bone origin: selection of surgical approach. Skull Base 2010;20:253–9CrossRefGoogle ScholarPubMed
44 Savva, A, Taylor, MJ, Beatty, CW. Management of cerebrospinal fluid leaks involving the temporal bone: report on 92 patients. Laryngoscope 2003;113:50–6Google Scholar