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1 구자원외 3 인. 후반고리관단독이상을보인돌발성난청환자 Research in Vestibular Science Vol. 16, No. 4, December 2017 Case Report pissn , eissn 후반고리관기능이상만을보인현훈을동반한돌발성난청환자 1 예 서남대학교의과대학명지병원이비인후과 구자원, 신용국, 송미현, 심대보 A Patient with Sudden Hearing Loss with Vertigo Showing Exclusive Posterior Semicircular Canal Abnormality Ja Won Gu, Yong Gook Shin, Mee Hyun Song, Dae Bo Shim Department of Otorhinolaryngology, Myongji Hospital, Seonam University College of Medicine, Goyang, Korea Received Oct 16, 2017 Revised Nov 1, 2017 Accepted Nov 17, 2017 Corresponding Author: Dae Bo Shim Department of Otorhinolaryngology, Myongji Hospital, Seonam University College of Medicine, 55 Hwasu-ro 14beon-gil, Deokyang-gu, Goyang 10475, Korea Tel: Fax: lovend77@gmail.com Copyright c 2017 by The Korean Balance Society. All rights reserved. This is an open access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. It is known that about 30% of patients with sudden hearing loss present with vertigo or dizziness. In clinical practice, this is called sudden hearing loss with vertigo (SHLV) although definite diagnostic criteria have not been established. Dizziness in SHLV is known to be caused by the dysfunction of the vestibular end-organs as well as the superior vestibular nerve or both vestibular nerve divisions. Lesions of the inferior vestibular nerve or a single semicircular canal have also been reported in these patients. Herein we report a 71-year-old male patient with SHLV who demonstrated vestibular dysfunction involving only the posterior semicircular canal. The patient showed normal results in the bithermal caloric test and the cervical vestibular evoked myogenic potentials test as well as positional test. Video head impulse test showed decreased gain only in the posterior semicircular canal. This case is significant in showing that dizziness in SHLV patients can occur by an abnormality involving only a single semicircular canal. Res Vestib Sci 2017;16(4): Keywords: Sudden hearing loss; Vertigo; Posterior semicircular canal 서론돌발성난청 (sudden sensorineural hearing loss, SHL) 은명확한원인없이 3일이내에갑자기발생하며, 세개이상의연속된주파수에서 30 db 이상의청력감소를보이는질환으로정의된다 [1]. 돌발성난청환자의 30% 내외에서현훈이나어지럼을동반하는것으로알려져있으며 [2,3], 임상에서돌발성난청에현훈이나어지럼이동반된환자들에대한명확한진단기준이정립되지않은상태로 현훈을동반한돌발성난청 (sudden hearing loss with vertigo, SHLV) 으 로명명하고있으며, 미로염 (labyrinthitis) 이나신경미로염 (neurolabyrinthitis) 등과혼용하여사용하고있다 [4,5]. 일반적으로 SHLV의원인은인접하여주행하는전정신경과와우신경이같이영향을받거나미로자체의문제로추정하고있고, SHLV는다양한원인에의해발생할수있어비슷한증상을나타낼수있는메니에르병, 전하소뇌동맥경색 (anterior inferior cerebellar artery stroke), 이성대상포진 (herpes zoster oticus), 전정신경초종과같은다른원인을감별해야한다. 전정신경염 (vestibular neuritis) 은대부분상전정신경의 161

2 Res Vestib Sci Vol. 16, No. 4, Dec 이상에의한것으로알려진것에비해, SHLV는전정신경염에비해하전정신경의이상이관찰되는경우가상대적으로높은것으로보고되고있다 [5,6]. 하지만 SHLV에서한개의반고리관이상만을동반하는경우는매우드물다고보고하고있다 [7]. 이에저자들은 SHLV 환자에서구형낭의기능은정상이나 후반고리관 (posterior semicircular canal) 의기능이상만동반한환자를경험하여문헌고찰과함께보고하는바이다. 증례 71세남자환자가내원전날부터발생한좌측의난청및 Fig. 1. Results of vestibular function test. (A) Result of video-nystagmographic recordings showed left beating horizontal spontaneous nystagmus (3.6 day/sec) on the initial visit day of admission. (B) Bithermal caloric test did not show canal paresis on both sides on the second day of admission. CP, canal paresis; DP, directional preponderance. (C) Cervical vestibular evoked myogenic potential showed normal response on the second day of admission. (D) Ocular vestibular evoked myogenic potential also showed normal response on the second day of admission. 162

3 구자원외 3 인. 후반고리관단독이상을보인돌발성난청환자 구토를동반한현훈을주소로외래를방문하였다. 환자는고혈압, 당뇨, 고지혈증등의내과적과거력은없었다. 이학적검사에서양측고막은정상이었고, 비디오안진검사에서좌측으로향하는 3.6 day/sec의약한수평성자발안진이관찰되었으나, 두진후안진검사에서안진강도의증가소견및나안두부충동검사에서교정성단속운동이관찰되지않았다 (Fig. 1A). 체위검사에서도특이소견은관찰되지않았으며, 신경학적진찰에서중추신경계이상을의심할만한소견은관찰되지않았다. 순음청력검사에서우측의기도청력역치는 34 db로경도의감각신경성난청소견을보였으며, 좌측의기도청력역치는 100 db 이상의전농소견을보였다. 어음청력검사에서어음명료도가우측은 100%, 좌측은 0% 로나타났다 (Fig. 2A). 내원당시시행한혈액검사에서특이소견은보이지않았고, 자발안진이있으나두진후안진과두부충동검사에서중추성원인의감별이필요한소견이관찰되어시행한뇌확산강조자기공명영상및뇌자기공명영상에서뇌간및소뇌의이상소견은관찰되지않았다. 환자는 SHLV로진단후, 입원하여 7일간고용량스테로이드 (Dexamethasone 0.15 mg/kg/day) 와 5일간항바이러스제 (Acyclovir 750 mg/day) 의투여및현훈의증상완화를위해약물치료 (Dimenhydrinate 120 mg/day, Cinnarizine 60 mg/day, Kallidinogenase 150 IU/day) 를시행하였다. 입원 2일째, 안정시의회전성어지럼은전날보다많이호전되었으나, 환자는앉은상태에서좌측으로 45 정도머리를돌리고뒤로젖히는동작을하면회전성어지럼이발생하는비특이적인체위현훈을호소하였다. 좌측난청및 이명은전날과차이가없었다. 같은날시행한비디오안진검사에서자발안진은나타나지않았고, 체위검사에서양성돌발두위현훈을의심할만한특징적인체위안진이나체위현훈은관찰되지않았으며, 두진후안진검사만에서좌측으로향하는 4.7 day/sec의수평성안진이관찰되었다. 비디오두부충동검사에서좌측후반고리관에서교정성단속운동과함께 0.34로이득이감소된소견외에는모두정상소견으로확인되었으며 (Fig. 3A), 냉온교대온도안진검사에서우측 4% 반고리관마비, 좌측 24% 방향우위성을보였으며 (Fig. 1B), 경부및외안근전정유발근전위검사에서양이간진폭차이비 (interaural amplitude difference ratio, IAD ratio) 는각각 6%, 5% 로정상이었다 (Fig. 1C, D). 입원 7일째, 환자의안정시어지럼은완전히회복되었으며, 좌측 45 에서발생하는체위현훈도많이호전되었으나난청과이명은지속되었다. 같은날시행한청력검사에서는내원첫날결과와변화없이좌측의전농소견관찰되었다. 비디오안진검사에서자발안진은관찰되지않았으며, 비디오두부충동검사에서좌측후반고리관에서 0.50으로약간이득이향상된소견관찰되었다 (Fig. 3B). 환자는퇴원후난청의치료를위해일주일에 2회씩총 6차례의좌측고실내스테로이드주입술 (Dexamethasone 5 mg/ml, 0.4 ml) 을시행하였다. 퇴원후 3개월째외래내원시, 환자의좌측난청및이명은남아있으나, 어지럼은완전히호전된상태였다. 같은날시행한순음청력검사에서좌측기도청력역치는 78 db로내원첫날보다는호전된양상의고도난청소견을보였으나, 어음청력검사에서좌측의어음명료도는 0% 로변화가없었다 (Fig. 2B). 비디오두부충동검사에서 Fig. 2. Pure tone audiometry. (A) Initial pure tone audiometry showed profound sensorineural hearing loss on the left side at the initial visit. (B) Follow-up pure tone audiomerty showed mild improvement of hearing threshold on the left ear three months after admission. 163

4 Res Vestib Sci Vol. 16, No. 4, Dec Fig. 3. Serial change of the results of video head impulse test. (A) Initial video head impulse test showed decreased gain (0.34) and catch up saccade on the left posterior semicircular canal. The gains of five semicircular canals other than the left posterior semicircular canal were within the normal limit. (B) Follow-up video head impulse test on the 7th day after admission showed mild improvement of the gain on the left posterior semicircular canal. (C) Three months after admission, follow-up video head impulse test showed increased gain of the left posterior semicircular canal compared with previous results. LARP, left-anterior-right-posterior; RALP, right-anteriorleft-posterior; LA, left anterior semicircular canal; LP, left posterior semicircular canal; RA, right anterior semicircular canal; RP, right posterior semicircular canal. 좌측 후반고리관은 0.58으로 아직 이득이 감소된 소견 관 정신경 모두에 의해 영향을 받아 생기는 것으로 알려져 있 찰되었다(Fig. 3C). 현재 이명 및 난청에 대해 약물치료 중 으며[8,9], 일부 연구들에서는 하전정신경이나 각 개별 반 으로 정기적인 추적 관찰 중이다. 고리관만의 손상을 보고하고 있다[6,7]. SHLV의 자발안진 에 대해 분석한 기존 연구에서는 상전정신경이 주로 침범 고 찰 된 경우에 병변의 반대측으로 향하는 자발안진을 관찰 할 수 있었고[6,8], 하전정신경염의 경우에도 병변 반대측을 본 증례는 SHLV에서 온도안진검사, 경부 및 외안근 전 향하는 회전성 하방안진을 관찰 할 수 있으나[10], 본 증례 정유발근전위검사에서는 정상 소견을 보이나 비디오 두부 에서는 내원 당시 시행한 비디오 안진검사에서 병변측을 충동검사에서 후반고리관에서만 이득이 감소해 있는 환자 향하는 자발안진이 관찰되었고, 하방 안진성분도 관찰되 로, 이는 SHLV 환자에서 후반고리관의 기능에만 영향을 지 않았다. 자발안진은 다음날 시행한 비디오 안진검사에 받은 증례이다. 서 나타나지 않았으며, 이러한 병변측을 향하는 자발안진은 SHLV에서 어지럼의 원인은 상전정신경이나 상 하전 164 미로염의 자극기(irritative phase)에 병변측의 전정핵(vesti-

5 구자원외 3 인. 후반고리관단독이상을보인돌발성난청환자 bular nuclei) 이자극되면서병변측을향하는자발안진이발생하는것으로추정된다 [11]. 수직성분이나회선성분이관찰되지않은것은병변측의마비로인한 마비기안진 (paralytic nystagmus) 이아닌병변측의일시적인자극으로인해발생한 자극기안진 (irritative nystagmus) 으로매우약한수평안진만관찰되었던것으로추정된다. 또한, 본증례의환자에서초기에심한어지럼과함께병변측을향하는약한안진을보였다가입원 2일째에안정기현훈과안진이모두호전되었는데, 한연구에의하면온도안진검사에서반고리관마비가 25% 이하인전정신경염환자군은반고리관마비가 25% 이상인환자군에비해자발안진이관찰되는기간과증상의회복기간이더짧은양상을확인하였고, 이는염증의진행과정이나혈관성손상에의한일시적인자극현상과중추전정계의보상에의한것으로설명하였다 [12]. 본증례의환자도입원 2일째시행한온도안진검사가정상이었으나, 25% 이하의약한손상에의해매우짧은자발안진과현훈경과를보였을가능성도생각해볼수있다. 본증례의환자에서발생한현훈은일반적으로돌발성난청과동반된상전정신경의손상을의심하였다. 그러나, 온도안진검사에서정상소견이관찰되어 SHLV 환자들의일부에서관찰되는하전정신경의이상을의심하였다. 하전정신경염은전정신경염의한종류로하전정신경에만침범하는아형으로이론적으로자발안진이병변반대쪽을향하는회선성분을가지는하방안진 (spontaneous torsional downbeat nystagmus) 으로나타나고, 온도안진검사에서정상소견을보이나, 경부전정유발근전위검사에서이상소견이관찰되며, 비디오두부충동검사에서수평반고리관과전반고리관은정상이나후반고리관에서만비정상소견이관찰된다 [10]. 본증례의환자에서는경부전정유발근전위검사에서정상소견이관찰되었고, 비디오두부충동검사에서는후반고리관에서만이득이감소되고교정성단속운동이관찰되었다. 이에본증례의경우기존의연구들과는다르게상전정신경이나하전정신경을침범하지않고, 후팽대부신경또는후반고리관에만손상을받은것으로확인하였다. 비디오두부충동검사를이용하여 SHLV 환자들의각개별반고리관들의손상을비교한연구에서후반고리관의이상을보이는비율이 74% 로다른반고리관에비해가장높았고, 이는내이도내에서후팽대부신경이와우신경과인접하게주행하기때문에염증이나감염에취약한것이원인으로추정하고있다 [7,10]. 임상적으로 SHLV는전정신경염과난청의유무를통해명확히구분할수있으나, 어지럼의양상이나전정기관의손상에대해서는임상적으로구분이쉽지않다. 두질환모두전정신경이지배하는전정기관들이영향을받게되는데, 어떤전정기관들이영향을받는지에따라차이를나타낸다. 비디오두부충동검사에서후반고리관의이상을보인환자들에대한한연구에서일측의후반고리관의이상을보인 40명의환자들에게서특징적인어지럼증의양상이관찰되지않았고, 전체 40명의환자들중 18명 (45%) 에서만동측의청력저하가관찰되었다 [13]. 따라서, 본증례의환자가경험한비특이적인체위현훈이반드시후반고리관의기능이상과연관성이있다고단정짓기는어렵다. 그러나, SHLV에서온도안진검사만으로는각전정기관들이받는영향을파악하고진단하는데어려움이있을수있으므로, SHLV 환자에서경부및외안근전정유발근전위검사, 비디오두부충동검사와같은추가적전정기능검사를통해어지럼증의정확한원인추정에도움을줄수있을것으로생각된다. 중심단어 : 돌발성난청, 현훈, 후반고리관 CONFLICT OF INTEREST No potential conflict of interest relevant to this article was reported. REFERENCES 1. Stachler RJ, Chandrasekhar SS, Archer SM, Rosenfeld RM, Schwartz SR, Barrs DM, et al. Clinical practice guideline: sudden hearing loss. Otolaryngol Head Neck Surg 2012;146(3 Suppl):S Nosrati-Zarenoe R, Arlinger S, Hultcrantz E. Idiopathic sudden sensorineural hearing loss: results drawn from the Swedish national database. Acta Otolaryngol 2007;127: Kitoh R, Nishio SY, Ogawa K, Kanzaki S, Hato N, Sone M, et al. Nationwide epidemiological survey of idiopathic sudden sensorineural hearing loss in Japan. Acta Otolaryngol 2017;137(suppl 565):S Hotson JR, Baloh RW. Acute vestibular syndrome. N Engl J Med 1998;339: Baloh RW. Clinical practice: vestibular neuritis. N Engl J Med 2003;348: Jung JY, Kim YH, Suh MW. Difference in the nature of dizzi- 165

6 Res Vestib Sci Vol. 16, No. 4, Dec ness between vestibular neuritis and sudden sensorineural hearing loss with vertigo. Otol Neurotol 2012;33: Pogson JM, Taylor RL, Young AS, McGarvie LA, Flanagan S, Halmagyi GM, et al. Vertigo with sudden hearing loss: audio-vestibular characteristics. J Neurol 2016;263: Kim CH, Na BR, Park HJ, Shin JE. Impairment of static vestibular function is limited in patients with sudden sensorineural hearing loss with vertigo. Audiol Neurootol 2013;18: Iwasaki S, Takai Y, Ozeki H, Ito K, Karino S, Murofushi T. Extent of lesions in idiopathic sudden hearing loss with vertigo: study using click and galvanic vestibular evoked myogenic potentials. Arch Otolaryngol Head Neck Surg 2005;131: Kim JS, Kim HJ. Inferior vestibular neuritis. J Neurol 2012;259: Kim SH, Chung WK, Kim BG, Hwang CS, Kim MJ, Lee WS. Periodic alternating nystagmus of peripheral vestibular origin. Laryngoscope 2014;124: Kim HJ, Kim DY, Hwang JH, Kim KS. Vestibular neuritis with minimal canal paresis: characteristics and clinical implication. Clin Exp Otorhinolaryngol 2017;10: Tarnutzer AA, Bockisch CJ, Buffone E, Weber KP. Association of posterior semicircular canal hypofunction on video-head-impulse testing with other vestibulo-cochlear deficits. Clin Neurophysiol 2017;128:

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