Case Report Late Pneumolabyrinth May Be Induced by Old ...downloads.hindawi.com/journals/criot/2015/506484.pdf · : Follow-up audiogram performed years later. Profound sensorineural

Case ReportLate Pneumolabyrinth May Be Induced byOld Penetrating Injury Possibility of UndiagnosedPosttraumatic Perilymphatic Fistula

Takahiro Nakashima1 Keiji Matsuda1 Takumi Okuda2 Tetsuya Tono1 Minoru Takaki2

Tamon Hayashi2 and Yutaka Hanamure2

1Department of Otorhinolaryngology Head and Neck Surgery Faculty of Medicine University of Miyazaki 5200 KiharaKiyotakecho Miyazaki 889-1692 Japan2Department of Otorhinolaryngology Head and Neck Surgery Kagoshima City Hospital No 20-17 KajiyachoKagoshima 892-8580 Japan

Correspondence should be addressed to Takahiro Nakashima maypapatnakagmailcom

Received 28 December 2014 Accepted 21 March 2015

Academic Editor Tamas Karosi

Copyright copy 2015 Takahiro Nakashima et al This is an open access article distributed under the Creative Commons AttributionLicense which permits unrestricted use distribution and reproduction in any medium provided the original work is properlycited

Traumatic pneumolabyrinth is a relatively rare entity We report the case of a unilaterally deaf woman with pneumolabyrinth whohad suffered penetrating injury 15 years ago This past history indicated that the case was late pneumolabyrinth occurring fromundiagnosed old posttraumatic perilymphatic fistula In Japan most cases of traumatic pneumolabyrinth are caused by penetratinginjury with an ear pick Dizziness often improves within several months Immediate surgical intervention is recommended forhearing loss but the hearing outcome is not satisfactory An appropriate strategy should be selected based on the interval to surgerybone conduction hearing level at disease onset stapes lesions and location of air

1 Introduction

Pneumolabyrinth is a condition caused by air in the labyrinthwhich indicates that a perilymphatic fistula (PLF) is presentin the affected ear However patients with traumatic PLFseldom develop pneumolabyrinth Here we report a caseof pneumolabyrinth that may have been caused by a pene-trating injury of the tympanic membrane that occurred 15years before presentation This interesting case suggests thatpneumolabyrinth may be among the differential diagnosesfor recurrent dizziness

2 Case Report

A 25-year-old woman was admitted to hospital with a com-plaint of longstanding right-sided profound hearing loss Herhearing impairment had begun 15 years ago At that time shesuffered traumatic tympanic membrane perforation due topenetration by an ear pick She had sudden profound hearing

loss at the onset of injury and it was unclear that she hadfelt vertigo after injury Conservative therapy was performedat ENT clinic nearby Although perforation of tympanicmembrane was healed completely profound hearing loss hadremained She mentioned the degree of hearing loss had notchanged She had felt lightheadedness about 7 years ago

An otoscopic examination revealed no perforation in herright ear but pure tone audiometry showed right profoundhearing loss (Figure 1) Imaging of temporal bone using cone-beam computed tomography (CBCT) showed a round lowdensity area in the vestibule and intrusion of stapes intothe vestibule (Figure 2) A diagnosis of pneumolabyrinth wasmade and PLF was suspected The patient refused surgicalintervention and chose to receive outpatient follow-up andno medication

Two years later she was admitted again for follow-up ofhearing loss She sometimes had dizziness or lightheaded-ness Pure tone audiometry showed right profound hearingloss similar to before (Figure 3) Equilibrium tests showed no

Hindawi Publishing CorporationCase Reports in OtolaryngologyVolume 2015 Article ID 506484 4 pageshttpdxdoiorg1011552015506484

2 Case Reports in Otolaryngology

120125 250 500 1000 2000 4000 8000

40

Frequency (Hz)

Hea

ring

thre

shol

d le

vel (

dB)

0

10

20

30

110

50

60

70

80

90

100

darr

darr

darr darr

darr

darr

darr

darr

minus10

]

[

[[ [

[

]] ]

998666

998666

998666

998666 998666998666

998666

times times times times]times times

times

Figure 1 Audiogram on hospital on day 1 The right ear showedprofound sensorineural hearing loss

positional or positioning vertigo A caloric test indicated thatright vestibular function was slightly decreased but not abol-ished Reexamination by CBCT showed that air that had beenpresent in the right vestibule had disappeared (Figure 4) Werecommended probe tympanotomy to confirm and cure PLFbut the patient rejected surgical intervention and requestedcourse observation

3 Discussion

The number of reports on pneumolabyrinth is smaller thanthose on PLF but has shown a slight increase due to improvedaccuracy of imaging modalities Hidaka et al [1] suggestedthat about 40 of cases of pneumolabyrinth are caused byblunt trauma to the head followed by penetrating injuryIn Japan however penetrating injury is the most commonetiology because of a habit of cleaning the external ear canalusing an ear pick

According to medical interview profound hearing lossoccurred immediately after injury and was supposed tobe permanent Intrusion of stapes found in CBCT imageindicated the possibility of old posttraumatic PLF Symptomsassociated with PLF tend to appear rapidly and the degreeof hearing loss varies from mild to profound Subacutedeterioration of hearing level has been documented in severalreports [2ndash6] and all cases showed progression of hearing losswithin 2 weeks One case had an apparent causative event andall received conservative therapy

The unique aspect of this case is the long interval betweeninjury and diagnosis of pneumolabyrinth by CBCT It wasimpossible to make sure that pneumolabyrinth had occurredsoon after injury Intrusion of stapes suggested that there hadbeen posttraumatic PLF There was no causative event otherthan penetrating injury by ear pick 15 years ago Air that

(a)

(b)

Figure 2 Axial views of the right ear on cone-beam computedtomography on hospital on day 1 showing a round low density area(a) and intrusion of stapes (b) (black arrows)

enters the inner ear usually disappears within several weeksPneumolabyrinth detected by CBCT suggested that air wastrapped recently and that PLF did not heal completely In thepatient with PLF elevated atmospheric pressure in themiddleear cavity may induce fainting and dizziness If excessiveelevation of the middle ear pressure happens entrapmentof air in the vestibule may occur Nurre et al [7] describeda case of traumatic pneumolabyrinth due to a temporalbone fracture that happened 2 years earlier Pneumolabyrinthmay be a clue to find undiagnosed PLF that is among thedifferential diagnoses for dizziness

Treatment of dizziness has a good prognosis but treat-ment of hearing loss is difficult and often unsuccessfulMost reports have shown unsatisfactory hearing outcomesregardless of the treatment strategyHidaka et al [1] suggestedthat there is no difference in efficacy between conservativetherapy and surgical therapy Tsubota et al [5] proposed threepredictive factors for hearing improvement the interval untilsurgery bone conduction hearing level at onset of diseaseand stapes lesions Probe tympanotomy should be performed

Case Reports in Otolaryngology 3

120125 250 500 1000 2000 4000 8000

Frequency (Hz)

Hea

ring

thre

shol

d le

vel (

dB)

0

10

20

30

40

50

60

70

80

90

100

110

minus10

]

[

[[ [

[

]

darr

darr

darrdarr darr

darr

darr

darr darr darr darr

darr

times times] times] times]times times times

998666

998666

998666 998666 998666 998666

998666

Figure 3 Follow-up audiogram performed 2 years later Profoundsensorineural hearing loss in her right ear had not changed

Figure 4 Axial view of the right ear on cone-beam computedtomography obtained 2 years after initial presentation The airbubble apparent in Figure 2(a) had disappeared

as soon as possible and several authors have recommendedsurgical intervention within 2 weeks from disease onset

Tsubota et al [5] found that a bone conduction level(BCL) of gt65 dB HL at onset of disease resulted in apoor hearing outcome Hidaka et al [1] found no BCLdeterioration over 10 dB in patients with a preoperative BCLgt65 dB HL but a rate of worsening of 29 in those withBCL lt65 dB HL preoperatively however this may reflectdifferences in the selected cases Tsubota et al [5] described15 cases up to 2009 and Hidaka et al [1] added another 16cases reported since 2009 The earlier 15 cases had worseBCL and more of the cases were treated surgically Regardingstapes lesions Sarac et al [8] pointed out that cases with noor mild stapes intrusion had good hearing outcomes after

surgery whereas those with deeply intruded stapes may nothave improved hearing after surgery and may suffer fromadditional inner ear damage due to surgery It was alsoconcluded that stapes surgery should be performed early toavoid a late complication of inner ear dysfunction due tofibrosis in the labyrinth

Hidaka et al [1] also found that the location of air inthe labyrinth is an important prognostic factor In cases withair in the cochlea a good hearing outcome can be expectedwhereas hearing improvement may not always be achieved incases with air trapped in the vestibule Poor hearing resultsare also likely if air is present in both the cochlea and thevestibule These clinical results are supported by findings ofKobayashi et al [9 10] using measurements of endocochlearDC potential cochlear microphonic potential and summat-ing potential in guinea pig under conditions of air in thescala vestibuli and scala tympani All potentials decreasedtemporarily and recovered with air in the scala tympaniIn contrast all potentials decreased without recovery withair in the scala vestibuli In histological analysis structuressuch as the organ of Corti Reissnerrsquos membrane spiralganglion and stria vascularis retained normal morphologyin animals with air in the scala tympani whereas collapseof the Reissnerrsquos membrane was observed in those withair in the scala vestibuli Based on these results it wassuggested that air entrapped from an oval window mightcause greater inner ear damage than that from a roundwindow in pneumolabyrinth These findings provide usefulinformation on the pathogenesis of pneumolabyrinth

Prevention from worsening of hearing loss is importantAchache et al [11] emphasize that labyrinthine concussionshould be systematically suspected in cranial trauma Pene-trating injury by an ear pick is sometimes accompanied withdizziness If there is mixed or sensorineural hearing losscareful follow-up to check hearing status is mandatory Addi-tionally thin-sliced computed tomography of the temporalbone should be performed to examine the condition of stapesand inner ear

4 Conclusion

Pneumolabyrinthmay occur in case PLF is present even if theinterval between diagnosis and causative event is very longAn appropriate decision on the treatment strategy requiresconsideration of the timing of surgery the bone conductionlevel at onset of disease stapes lesions and location of air inthe inner ear

Conflict of Interests

The authors declare that there is no conflict of interestsregarding the publication of this paper

References

[1] H Hidaka M Miyazaki T Kawase and T Kobayashi ldquoTrau-matic pneumolabyrinth air location and hearing outcomerdquoOtology and Neurotology vol 33 no 2 pp 123ndash131 2012


[2] R Uemura and M Kawaura ldquoTraumatic perilymphatic fistulacaused by an earpickrdquo ORL Tokyo vol 38 pp 724ndash728 1995(Japanese)

[3] S Nishiike Y Hyo and H Fukushima ldquoStapediovestibulardislocation with pneumolabyrinthrdquo Journal of Laryngology andOtology vol 122 no 4 pp 419ndash421 2008

[4] A Hatano M Rikitake M Komori T Irie and H MoriyamaldquoTraumatic perilymphatic fistula with the luxation of the stapesinto the vestibulerdquo Auris Nasus Larynx vol 36 no 4 pp 474ndash478 2009

[5] M Tsubota H Shojaku and Y Watanabe ldquoPrognosis of innerear function in pneumolabyrinth case report and literaturereviewrdquo The American Journal of OtolaryngologymdashHead andNeck Medicine and Surgery vol 30 no 6 pp 423ndash426 2009

[6] T Ueno M Ito H Sugimoto and T Yoshizaki ldquoUsabilityof multiplanar reconstruction two case reports of traumaticperilymphatic fistula with pneumolabyrinthrdquo Otology Japanvol 22 pp 833ndash838 2012 (Japanese)

[7] J W Nurre G W Miller and J B Ball Jr ldquoPneumolabyrinth asa late sequela of temporal bone fracturerdquoThe American Journalof Otology vol 9 no 6 pp 489ndash493 1988

[8] S Sarac S Cengel and L Sennaroglu ldquoPneumolabyrinthfollowing traumatic luxation of the stapes into the vestibulerdquoInternational Journal of Pediatric Otorhinolaryngology vol 70no 1 pp 159ndash161 2006

[9] T Kobayashi Z Itoh T Sakurada N Shiga and T TakasakaldquoEffect of perilymphatic air perfusion on cochlear potentialsrdquoActa Oto-Laryngologica vol 110 no 3-4 pp 209ndash216 1990

[10] T Kobayashi T Sakurada K Ohyama and T Takasaka ldquoInnerear injury caused by air intrusion to the scala vestibuli of thecochleardquo Acta Oto-Laryngologica vol 113 no 6 pp 725ndash7301993

[11] M Achache M Sanjuan Puchol L Santini et al ldquoLate pneu-molabyrinth after undiagnosed post-traumatic perilymphaticfistula Case report illustrating the importance of systematicemergency managementrdquo European Annals of Otorhinolaryn-gology Head andNeckDiseases vol 130 no 5 pp 283ndash287 2013

Submit your manuscripts athttpwwwhindawicom

Stem CellsInternational

Hindawi Publishing Corporationhttpwwwhindawicom Volume 2014


MEDIATORSINFLAMMATION

of


Behavioural Neurology

EndocrinologyInternational Journal of



Disease Markers


BioMed Research International

OncologyJournal of



Oxidative Medicine and Cellular Longevity


PPAR Research

The Scientific World JournalHindawi Publishing Corporation httpwwwhindawicom Volume 2014

Immunology ResearchHindawi Publishing Corporationhttpwwwhindawicom Volume 2014

Journal of

ObesityJournal of



Computational and Mathematical Methods in Medicine

OphthalmologyJournal of


Diabetes ResearchJournal of



Research and TreatmentAIDS


Gastroenterology Research and Practice


Parkinsonrsquos Disease

Evidence-Based Complementary and Alternative Medicine

Volume 2014Hindawi Publishing Corporationhttpwwwhindawicom


120125 250 500 1000 2000 4000 8000

40

Frequency (Hz)

Hea

ring

thre

shol

d le

vel (

dB)

0

10

20

30

110

50

60

70

80

90

100

darr

darr

darr darr

darr

darr

darr

darr

minus10

]

[

[[ [

[

]] ]

998666

998666

998666

998666 998666998666

998666

times times times times]times times

times

Figure 1 Audiogram on hospital on day 1 The right ear showedprofound sensorineural hearing loss

positional or positioning vertigo A caloric test indicated thatright vestibular function was slightly decreased but not abol-ished Reexamination by CBCT showed that air that had beenpresent in the right vestibule had disappeared (Figure 4) Werecommended probe tympanotomy to confirm and cure PLFbut the patient rejected surgical intervention and requestedcourse observation

3 Discussion

The number of reports on pneumolabyrinth is smaller thanthose on PLF but has shown a slight increase due to improvedaccuracy of imaging modalities Hidaka et al [1] suggestedthat about 40 of cases of pneumolabyrinth are caused byblunt trauma to the head followed by penetrating injuryIn Japan however penetrating injury is the most commonetiology because of a habit of cleaning the external ear canalusing an ear pick

According to medical interview profound hearing lossoccurred immediately after injury and was supposed tobe permanent Intrusion of stapes found in CBCT imageindicated the possibility of old posttraumatic PLF Symptomsassociated with PLF tend to appear rapidly and the degreeof hearing loss varies from mild to profound Subacutedeterioration of hearing level has been documented in severalreports [2ndash6] and all cases showed progression of hearing losswithin 2 weeks One case had an apparent causative event andall received conservative therapy

The unique aspect of this case is the long interval betweeninjury and diagnosis of pneumolabyrinth by CBCT It wasimpossible to make sure that pneumolabyrinth had occurredsoon after injury Intrusion of stapes suggested that there hadbeen posttraumatic PLF There was no causative event otherthan penetrating injury by ear pick 15 years ago Air that

(a)

(b)

Figure 2 Axial views of the right ear on cone-beam computedtomography on hospital on day 1 showing a round low density area(a) and intrusion of stapes (b) (black arrows)

enters the inner ear usually disappears within several weeksPneumolabyrinth detected by CBCT suggested that air wastrapped recently and that PLF did not heal completely In thepatient with PLF elevated atmospheric pressure in themiddleear cavity may induce fainting and dizziness If excessiveelevation of the middle ear pressure happens entrapmentof air in the vestibule may occur Nurre et al [7] describeda case of traumatic pneumolabyrinth due to a temporalbone fracture that happened 2 years earlier Pneumolabyrinthmay be a clue to find undiagnosed PLF that is among thedifferential diagnoses for dizziness

Treatment of dizziness has a good prognosis but treat-ment of hearing loss is difficult and often unsuccessfulMost reports have shown unsatisfactory hearing outcomesregardless of the treatment strategyHidaka et al [1] suggestedthat there is no difference in efficacy between conservativetherapy and surgical therapy Tsubota et al [5] proposed threepredictive factors for hearing improvement the interval untilsurgery bone conduction hearing level at onset of diseaseand stapes lesions Probe tympanotomy should be performed


120125 250 500 1000 2000 4000 8000

Frequency (Hz)

Hea

ring

thre

shol

d le

vel (

dB)

0

10

20

30

40

50

60

70

80

90

100

110

minus10

]

[

[[ [

[

]

darr

darr

darrdarr darr

darr

darr

darr darr darr darr

darr


998666

998666

998666 998666 998666 998666

998666








4 Conclusion




References


















of






Disease Markers



OncologyJournal of





PPAR Research



Journal of

ObesityJournal of

















120125 250 500 1000 2000 4000 8000

Frequency (Hz)

Hea

ring

thre

shol

d le

vel (

dB)

0

10

20

30

40

50

60

70

80

90

100

110

minus10

]

[

[[ [

[

]

darr

darr

darrdarr darr

darr

darr

darr darr darr darr

darr


998666

998666

998666 998666 998666 998666

998666








4 Conclusion




References


















of






Disease Markers



OncologyJournal of





PPAR Research



Journal of

ObesityJournal of
































of






Disease Markers



OncologyJournal of





PPAR Research



Journal of

ObesityJournal of





















of






Disease Markers



OncologyJournal of





PPAR Research



Journal of

ObesityJournal of
















Documents

Case Report Late Pneumolabyrinth May Be Induced by Old ...downloads.hindawi.com/journals/criot/2015/506484.pdf · : Follow-up audiogram performed years later. Profound sensorineural