대한임상신경생리학회지 17(2):86-90, 2015 eissn 2288-1026 pissn 1229-6414 http://dx.doi.org/10.14253/kjcn.2015.17.2.86 Case Report 대상포진후발생한아급성위팔신경근얼기염 건양대학교의과대학신경과학교실 Subacute Brachial Radiculoplexitis Following Herpes Zoster Infection Jae-Hwan Kim, Yong-Duk Kim, Sang-Jun Na, Kee Ook Lee, Bora Yoon Department of Neurology, Konyang University College of Medicine, Daejeon, Korea Brachial radiculoplexitis is characterized by acute onset of shoulder and arm pain followed by weakness and sensory loss. Brachial radiculoplexitis by herpes zoster is a rare disease, which can be diagnosed by careful history, electrodiagnosis and MRI. It has remained uncertain about clinical characteristics, treatment, and prognosis. Better understanding of this disease helps earlier diagnosis and prompt treatment to minimize neurologic sequale. We present two cases of subacute brachial radiculoplexitis preceded by herpes zoster infection. (Korean J Clin Neurophysiol 2015;17:86-90) Key Words: Brachial plexopathy, Brachial radiculoplexitis, Herpes zoster Received 21 May 2015; received in revised form 28 August 2015; accepted 7 September 2015. 대상포진은수두대상포진바이러스 (varicella-zoster virus) 가일차감염후등뿌리신경절 (dorsal root ganglia) 에잠복해있다가재활성화되면서발생하는질환으로, 1 주로감각신경침범에의한통증과감각이상이주증상이지만말초신경계에서중추신경계까지다양한신경학적합병증을일으킬수있다. 그중운동신경병증을일으킬수있는데뇌신경마비가가장흔하며이외에도신경뿌리병증, 홑신경병증 (mononeuropathy) 의형태로나타날수있다. 2 대상포진에의한신경근얼기염 (radiculoplexitis) 은몇차례보고된바있으나매운드문질환으로알려져있다. 3 저자들은신경전도, 근전도검사및영상학적검사로진단되었으며스테로이드치료에의해호전을보인대상포진에의한아급성위 팔신경근얼기염 2 예를경험하여이를보고한다. Address for correspondence; Bora Yoon Department of Neurology, Konyang University Hospital, 158 Gwanjeodong-ro, Seo-gu, Daejeon 35365, Korea, Tel: +82-42-600-9156 Fax: +82-42-545-0050 E-mail: boradori3@kyuh.ac.kr Copyright 2015 by The Korean Society of Clinical Neurophysiology This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. A Figure 1. Skin lesions of case 1. The patient had multiple erythematous crusted plaques on the right arm (C5-6 dermatome areas). B
Brachial Radiculoplexitis by Herpes Zoster 증례 1. 증례 1 82세남자환자는우측경추 5-6 피부분절대상포진을진단받고피부과에입원하여 1주일동안 acyclovior 정맥주사치료후퇴원했던환자로, 대상포진피부병변발생일로부터 18일 ( 퇴원후 9일 ) 뒤, 우측팔의갑작스런근력저하와경추 5-6 피부분절영역에통증이발생하여 5일뒤에내원 하였다. 환자는고혈압과거력이있었으며우측팔과어깨의신경학적, 근골격계과거력및외상력은없었다. 신체검사에서, 우측경추 5-6 피부분절에다수의가피로덮인홍반발진이관찰되었다 (Fig. 1). 신경학적진찰에서의식은명료하였고뇌신경검사는정상이었으며우측팔의명확한근위축은관찰되지않았다. 근력검사에서우측어깨의벌림 (abduction), 모음 (adduction), 굽힘 (flexion) medical research council (MRC) grade III, 폄 (extension) MRC grade IV-, 우측 Table 1. Nerve conduction study, electromyography of case 1 and case 2 at 10 days after the onset of motor weakness Case 1 Motor Latency (R/L) (ms) CMAP (R/L) (mv) NCV (R/L) (m/s) Axillary 11.4/3.8 0.6/6.0 Musculocutaneous 5.6/4.8 4.0/7.6 Sensory Latency (R/L) (ms) SNAP (R/L) (µv) NCV (R/L) (m/s) Lateral antebrachial cutaneous 4.1/2.3 37.4/23.6 34/52 Medial antebrachial cutaneous 1.7/1.7 20.7/20.7 50/52 Muscle (R) Nerve Root Fib/Psw FlexCarRad Median C6-7 1+ BrachioRad Radial C5-6 1+ Biceps Musculocutaneous C5-6 1+ Supraspinatus Suprascapular C5-6 1+ Infraspinatus Suprascapular C5-6 1+ Deltoid Axillary C5-6 1+ Case 2 Motor Latency (R/L) (ms) CMAP (R/L) (mv) NCV (R/L) (m/s) Median Wrist 3.8/4.1 7.2/8.7 54.0/47.0 Elbow 8.0/8.6 6.6/8.0 61.0/64.0 Axillary 9.9/10.2 6.7/8.1 Axillary 4.3/4.5 2.0/0.2 Sensory Latency (R/L) (ms) SNAP (R/L) (µv) NCV (R/L) (m/s) Median Palm 2.3/2.3 23.4/19.3 31.0/24.0 Wrist 3.1/2.8 15.2/10.1 40.0/36.0 Elbow 3.8/3.7 27.4/15.8 51.0/51.0 Axillar 2.2/2.1 38.2/94.8 50.0/50.0 Radial 2.4/2.2 35.9/16.3 44/42 Muscle (L) Nerve Root Fib/Psw FlexCarRad Median C6-7 1+ Pronator Teres Median C6-7 1+ BrachioRad Radial C5-6 1+ Biceps Musculocutaneous C5-6 1+ Deltoid Axiallary C5-6 1+ NCV; nerve conduction velocity, R; right, L; left, CMAP; compound muscle action potential, SNAP; sensory nerve action potential, FlexCarRad; flexor carpi radialis, BrachioRad; brachioradialis, Fib; fibrillation potential, Psw; positive sharp wave. Korean J Clin Neurophysiol / Volume 17 / December 2015 87
팔꿈치의굽힘, 폄 MRC grade IV, 우측손목의굽힘, 폄 MRC grade IV+, 우측엄지손가락의벌림이 MRC grade IV+ 로저하되어있었다. 감각검사에서우측경추 5-6 피부분절에서감각저하가관찰되었다. 우측의두갈래근반사, 위팔노근반사가저하되어있었으며병적반사는관찰되지않았다. 혈액검사는정상소견이었다. 근력저하발생 10일뒤에신경전도검사와근전도검사를시행하였다. 신경전도검사에서우측겨드랑신경 (axillary nerve) 의말단잠복기 (terminal latency) 연장, 복합근활동전위 (compound muscle action potential) 의진폭감소, 우측근육피부신경 (musculocutaneous nerve) 의복합근활동전위의진폭감소, 우측가쪽아래팔피부신경 (lateral antebrachial cutaneous nerve) 의복합근활동전위의진폭감소, 신경전도속도 (nerve conduction velocity) 감소가관찰되었다 (Table 1). 근전도검사에서우측노쪽손목굽힘근 (flexor carpi radialis muscle), 가시위근 (supraspinatus muscle), 가시아래근 (infraspinatus muscle), 어깨세모근 (deltoid muscle), 위팔두갈래근 (biceps brachii muscle), 위팔노근 (brachioradialis muscle) 에탈신경전위가관찰되었으며척추옆근 (paraspinal muscles) 은정상이었다 (Table 1). 신경전도검사, 근전도검사와임상소견을종합하여우측위줄기위팔신경얼기병증 (upper trunk brachial plexopathy) 으로판단하여위팔신경얼기자기 공명영상을촬영하였다. 자기공명영상에서우측경추 5-6 신경뿌리, 위줄기를포함한위팔신경얼기에광범위한조영증강과비후가관찰되었다 (Fig. 2A-D). 환자의병력, 신경전도검사, 근전도검사, 자기공명영상을토대로대상포진에의한아급성우측위팔신경근얼기염으로최종진단하였다. 이후 5일간 methylprednisolone 1 g의정맥주사치료와함께우측팔의재활치료를진행하였다. 스테로이드정맥주사동안환자는우측팔통증의빠른호전과근력회복을보였다. 환자는퇴원후에도경구 prednisolone을일정기간유지하였고우측팔근력저하는점진적인호전을보였다. 대상포진발병 3달뒤, 근력검사에서우측어깨의벌림, 모임, 굽힘 MRC grade IV+, 우측팔꿈치의굽힘, 폄 MRC grade V, 우측손목의굽힘, 폄, 엄지손가락벌림 MRC grade V로어깨근력을제외하고는상당한호전을보였다. 2. 증례 2 75세여환은좌측경추 5-7 피부분절대상포진진단하에 7일간 acyclovir 정맥주사후퇴원했던환자로, 대상포진피부병변발생 17일 ( 퇴원후 7일 ) 뒤, 갑작스런좌측팔의근력저하와경추 5-7 피부분절영역의통증이발생하여재입원하였다. 환자는당뇨, 고혈압, 고지혈증, 심부전증, 협 A B C D E F G H Figure 2. Brachial plexus magnetic resonance imaging (MRI) of case 1 and 2. They were performed at 10 days after the onset of motor weakness. In case 1 (A-D), abnormal diffuse enhancement and thickening involving right brachial plexus, especially C5-6 root are shown on T2 short tau inversion recovery (STIR) coronal image (A), and modified Dixon contrast enhanced image (B). Moreover, abnormal enhancement of right upper trunk is shown on T2 STIR coronal (C) and 3 mm reconstruction coronal image (D). In case 2 (E-H), T2 STIR coronal (E) and 3 mm reconstruction coronal image (F) demonstrate abnormal diffuse enhancement and thickening involving left brachial plexus, especially C5-7 root. In addition, T2 STIR coronal (G) and 5 mm reconstruction coronal image (H) show enhancement and thickening of left upper and middle trunk. 88 Korean J Clin Neurophysiol / Volume 17 / December 2015
Brachial Radiculoplexitis by Herpes Zoster 심증과거력이있었으며그외의신경학적, 근골격계과거력및외상력이없었다. 신체검사에서좌측경추 5-7 피부분절에다수의가피가덮인홍반성수포가관찰되었다. 신경학적진찰에서의식은명료하였고뇌신경검사는정상이었으며좌측팔의명확한근위축은관찰되지않았다. 근력검사에서좌측어깨의벌림, 모음 MRC grade IV, 굽힘, 폄 MRC grade IV+, 좌측팔꿈치의굽힘, 폄 MRC grade IV+, 좌측손목의굽힘, 폄 MRC grade IV, 좌측손가락굽힘 MRC grade IV+, 좌측엄지손가락의벌림, 모음, 굽힘, 폄, 맞섬이 MRC grade IV+ 로저하되어있었다. 감각검사에서좌측경추 6-7 피부분절에서감각저하가관찰되었다. 좌측의두갈래근반사, 위팔노근반사, 세갈래근반사가저하되어있었으며병적반사는관찰되지않았다. 혈액검사에서특이소견은없었다. 근력저하발생 10일째에시행한신경전도검사에서좌측정중신경 (median nerve) 의말단잠복기의연장과운동신경전도속도와감각신경전도속도감소, 좌측노신경 (radial nerve) 의감각신경활동전위 (sensory nerve action potential) 의진폭감소, 좌측겨드랑신경의복합근활동전위의진폭감소가관찰되었다 (Table 1). 근전도검사에서좌측노쪽손목굽힘근, 원엎침근 (pronator teres muscle), 위팔노근, 위팔두갈래근, 어깨세모근에탈신경전위가관찰되었으며척추옆근은정상이었다 (Table 1). 신경전도검사, 근전도검사, 임상양상을종합하여좌측위, 중간줄기위팔신경얼기병증으로판단하여위팔신경얼기자기공명영상을촬영하였다. 위팔신경얼기자기공명영상에서좌측경추 5-7 신경뿌리, 위, 중간줄기를포함한위팔신경얼기에광범위한조영증강과비후가관찰되었다 (Fig. 2E-H). 환자의병력, 신경전도검사, 근전도검사, 자기공명영상을토대로대상포진에의한아급성좌측위팔신경근얼기염으로최종진단하였으며재활치료와함께 5일간 methylprednisolone 1 g을정맥주사한뒤경구약 (prednisolone) 으로변환하여퇴원하였다. methylprednisolone 정맥주사동안좌측팔의통증은빠르게소실됐고점진적인근력회복을보였다. 대상포진발병 3달뒤, 근력검사에서좌측팔꿈치의굽힘, 좌측손목의굽힘, 폄, 좌측손가락의굽힘이 MRC grade V로회복되었다.. 고찰위팔신경근얼기염은신경통성근위축 (neuralgic amyotrophy), 파르소니지-알드렌-터너증후준 (Parsonage-Aldren- Turner syndrome), 급성상완신경총신경근염 (acute brachial radiculitis), 특발성상완신경총신경병증 (idiopathic brachial neuropathy) 등의여러명칭으로일컬어지는질환으로신경 뿌리및위팔신경얼기의신경을침범하여어깨와상지의통증및근력약화를일으키며원인은외상, 감염, 바이러스성질환, 과도한활동, 수술, 면역주사등이있고드물게유전성을보이는경우도있다. 4 전연령대에서발생가능하나호발연령은 20대와 60대이고, 남자에게서더흔하며, 5 수시간에서 2-3주후소실되는통증과통증이후발생하는근력약화를특징으로한다. 4,5 신경근얼기염의원인으로대상포진은드물어서발생시점, 호발신경부위, 임상증상, 치료반응들에대해서는정확히밝혀지지않았지만발생기전은수두대상포진바이러스에의한염증이등뿌리신경절에서부터신경뿌리나말초신경으로전파되면서발생하는것으로추정된다. 3 이전에보고된증례들은대부분대상포진발생 7일이내에근력저하가발생했으나수개월후에근력저하가발생한증례도있었고 6 침범한피부분절은경추 4번에서흉추 1번까지다양했다. Jones 등 7 이보고한대상포진관련팔다리마비환자 49명분석을보면, 이중 13 명 (26.5%) 이위팔신경얼기염으로나타났는데, 이중 6명이전기생리학적검사에서같은이상소견을보였고, 자기공명영상검사를시행한 6명중에서는 4명이이상소견을보인것으로보고됐다. 본증례처럼대상포진발생아급성기에도전기생리학적검사와함께자기공명영상을시행하는것이정확한진단에매우유용하겠다. 치료에대해서보고된증례마다차이가있었고 Jones 등 7 의보고에따르면대상포진관련사지마비환자의 69% 에서항바이러스제치료를한반면, 스테로이드치료는 12% 에서만시행하여치료에도차이를보이는것을알수있었다. 또한예후도항바이러스제투여후재활치료및통증조절을통한치료를하면서증상이완전히호전된경우도있었으나, 8 평균마비지속기간이 193일정도로비교적길게지속되었고다수에서신경통증이나마비가계속남아있다는 Jones 등 7 의보고를감안할때일관되지않았다. 본증례 2명은공통적으로 70 세이상의고령이었고, 대상포진이경추 5-6 피부분절을침범했고, 대상포진발생 2-3주후피부병변이회복되는과정에서아급성 ( 지연성 ) 으로근력저하가발생했으며, 근력저하와함께다시통증이발생했다. 또한대상포진후아급성기에도자기공명영상에서신경얼기에조영증강을보였으며고용량스테로이드치료가통증을신속하게감소시켰고근력향상에도움이됐다는점이특징적이었다. 대상포진에의한위팔신경근얼기염의치료는대상포진급성기에신속한항바이러스제투여를통해바이러스의복제를억제하여신경손상을줄일수있도록하는것이필수적이다. 또한본두증례처럼보존적인물리치료이외에스테로이드치료를통해광범위한염증을줄이는것이증상회복에 Korean J Clin Neurophysiol / Volume 17 / December 2015 89
도움이될수있겠다. 스테로이드제가면역억제를일으켜바이러스가더활성화되거나기회감염이발생할수있다는위험성이존재하지만본증례는스테로이드제사용전항바이러스제치료를종료하였고급성기가지난시기였기때문에이러한위험을방지할수있었다. 결론적으로본증례를통해급성대상포진감염후아급성위팔신경근얼기염이발생할수있으며, 전기생리학적검사와뇌자기공명영상을같이시행할경우진단의정확성을높일수있겠고, 고용량의스테로이드치료가빠르게통증을경감시키고근력을호전시킬수있음을알수있었다. 대상포진감염이후아급성으로근력약화가발생할경우대상포진에의한신경근얼기염을반드시고려해야하며신속한진단을통한항바이러스제와고용량의스테로이드치료로신경학적후유장애를최소화해야할것이다. REFERENCES J Am Osteopath Assoc 2009;109:2-6. 2. Merchut MP, Gruener G. Segmental zoster paresis of limbs. Electromyogr Clin Neurophysiol 1996;36:369-375. 3. Haanpaa M, Hakkinen V, Nurmikko T. Motor involvement in acute herpes zoster. Muscle Nerve 1997;20:1443-1448. 4. Tsairis P, Dyck PJ, Mulder DW. Natural history of brachial plexus neuropathy. Report on 99 patients. Arch Neurol 1972;27:109-117. 5. Beqhi E, Kurland LT, Mulder DW, Nicolosi A. Brachial plexus neuropathy in the population of Rochester, Minnesota. Ann Nuurol 1985;18:320-323. 6. Ayoub T, Raman V, Chowdhury M. Brachial neuritis caused by varicella-zoster diagnosed by changes in brachial plexus on MRI. J Neurol 2010;257:1-4. 7. Jones LK Jr, Reda H, Watson JC. Clinical, electrophysiologic, and imaging features of zoster-associated limb paresis. Muscle Nerve 2014;50:177-185. 8. Eyigor S, Durmaz B, Karapolat H. Monoparesis with complex regional pain syndrome-like symptoms due to brachial plexopathy caused by the varicella zoster virus: a case report. Arch Phys Med Rehabil 2006;87:1653-1655. 1. Weaver BA. Herpes zoster overview: natural history and incidence. 90 Korean J Clin Neurophysiol / Volume 17 / December 2015