臨床神経 indd

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1 True neurogenic thoracic outlet syndrome TOS TOStrue neurogenic TOS TNTOS disputed neurogenic TOS DNTOS TNTOS 12 TNTOS 4 Th1 TOS 7 6 DNTOS TNTOS Th1 DNTOS TOS TOS Wilbourn Gilliatt 9 TOS true neurogenic TOS; TNTOS Th1 10 disputed neurogenic TOS DNTOS TNTOS TOS

2 132 TNTOS TNTOS 4 TOS TNTOS TOS TNTOS Neuropack 9216, MS 25, Medelec NCS APB ADM MCS SCS MAC SCS SCS 33 MCS SCS MAC SCS SCS 10 Hz 3 khz 3 db MCS 5 ms/div SCS 2 ms/div CMAPSNAP TNTOS TNTOS Halsted Morley Roos 3 X NCS 2 MCS APB ADM CMAP SCS MAC SNAP SNAP 2 NCS 1 T1 APB C8 IOD C7/8 C paresthesia 3

133 1 TNTOS 4 Table 1 Clinical features of the 4 true neurogenic TOS cases 1 2 3 4 45 F 21 M 33 F 13 F APB 3 APB 3 APB 3 APB 4 IOD 4 IOD 4 IOD 3 IOD 5 MMT ADM 4 ADM 4 ADM 3 ADM 5 EDC 5 EDC 5 EDC 5

3 133 1 TNTOS 4 Table 1 Clinical features of the 4 true neurogenic TOS cases F 21 M 33 F 13 F APB 3 APB 3 APB 3 APB 4 IOD 4 IOD 4 IOD 3 IOD 5 MMT ADM 4 ADM 4 ADM 3 ADM 5 EDC 5 EDC 5 EDC 5 EDC 5 FDP1 3 FDP1 4 FDP1 3 FDP1 3 FDP4 5 FDP4 5 FDP4 5 FDP4 5 4, 5 Morley, Adson, Allen, Halsted Halsted, Wright, shoulder bracing Morley, Halsted Morley, Allen MMT Daniels 26 grading 3 2 MMT 3 APB, IOD, ADM EDCFDP1FDP4 MMT manual muscle testing : The influence of the gravity is negligible for all listed muscles, intrinsic hand muscles or finger flexors/extensors, and therefore the MMT grade 3 was defined as complete full range of motion with no resistance according to the definition in the textbook of Daniels 26. This actually corresponds to MMT grade 2 in ordinary muscles, and therefore these muscles with MMT 3 were actually considerably weak. Positive provocative tests were listed. APB, abductor pollicis brevis; IOD, interossei dorsalis first ; ADM, abductor digiti minimi; EDC, extensor digitorum communis; FDP1, first flexor digitorum porfundus for the index finger; FDP4, fourth flexor digitorum profundus for the ring finger. 1 2 X X Fig. 1 Plain X-ray film of the cervical spine of Patient 2 anteriorposterior view. Low shoulders on both sides are evident. Notably, the upper border of the scapula dotted line is lower on the right side the affected side than on the left side, indicating the more caudal location of the right upper limb. The distance between the supposed skin surface line dotted line and the first rib on the film is also narrower on the right side, implying the narrower space of the thoracic outlet TOS TNTOS TOS giving-way weakness hysterical weakness 27 28

4 TNTOS Table 2 Results of nerve conduction studies in TNTOS patients Case 1 Case 2 Case 3 Case 4 LLN Amplitude: affected/unaffected side Median CMAP 2.2/ / / / Ulnar CMAP 10.6/ / / / Median - thumb SNAP 43.3/ / / Median - index finger SNAP 28.1/ / / / Median - ring finger SNAP 10.4/ / / Ulnar SNAP 4.8/ / / / MAC SNAP 0.9/ / / / / CMAP SNAP 50 LLN lower limit of normal SD Case 1 SCS 1, 2, 4 MAC SCS CMAPSNAP MAC The amplitudes of the compound muscle action potential CMAP and sensory nerve action potential SNAP are listed in the order of the affected and unaffected sides. Bold figures indicate abnormal results judged by more than 50 reduction of the amplitude compared to the unaffected side. Underlined figures indicate that the value is lower than the lower limit value, although the side difference does not reach 50. LLN: Lower limit of normal values, which have been established from the examination of 32 to 40 healthy controls at our institution. In Case 1, the thumb, index and ring finger recordings following median nerve stimulation were not performed, and the orthodromic SNAPs following index finger stimulation are written. An orthodromic method was used for MAC SCS also in this case. CMAP, compound muscle action potential; SNAP, sensory nerve action potential; MAC, medial antebrachial cutaneous nerve. MUP poor activation NCS non-organic hysterical weakness 1 Romberg 1 MRI TNTOS TOS 4 TNTOS T1 DNTOS 1 3 Levin 10 TNTOS MCS APB SCS MAC C8 C8 MAC APB T1 TNTOS T1 10 NCS APB

5 T1 MCS MAC SCS C8 MCS SCS SCS C6 C7 SCS MCS SCS CMAPSNAP APB ADM MAC Fig. 2 Nerve conduction results of Patient 2. The waveforms on the affected right side and the unaffected left side are compared. Dominant myotome or dermatome is indicated in the parentheses after each examination. The T1-innervated median-apb CMAP and MAC SCS are the most severely affected, followed by the C8-innervated ulnar MCS, SCS and median-ring finger SNAP. The C6 and C7 innervated median-thumb and median-index finger SNAPs are not affected. MCS, motor-nerve conduction study; SCS, sensory-nerve conduction study; CMAP, compound muscle action potential; SNAP, sensory nerve action potential; APB, abductor pollicis brevis; ADM, abductor digiti minimi; MAC, medial antebrachial cutaneous nerve. ADM MAC C8 SNAP Levin T1 C8 TNTOS MAC MAC C8 20 MAC SNAP MAC NCS T TNTOS TOS 1 3

6 TNTOS 18 TNTOS MCS MAC SCS MCS SCS SCS T1 SCS APB ADM MAC Fig. 3 Localization of the lesion of the true neurogenic thoracic outlet syndrome TNTOS. The courses of the affected nerves in TNTOS are superimposed over the illustration of the brachial plexus modified from the reference number 18 with permission from the publisher. The most severely affected median-mcs and MAC SCS are drawn using solid black lines; the next severely affected ulnar-mcs/scs and median-ring finger IV SCS are drawn using dotted black lines. These results localize the lesion of the TNTOS most probably at the lower trunk, dominant in the T1 component. SCS, sensory-nerve conduction study; APB, abductor pollicis brevis; ADM, abductor digiti minimi; MAC, medial antebrachial cutaneous nerve. Wilbourn disputed neurogenic TOS DNTOS DNTOS TNTOS 3 Roos CTS TOS DNTOS giving-way weakness 27 28

7 TOS CTS TOS CTS CTS TNTOS TOS TOS TOS CTS double crush CTS Roos 3 TOS CTS proximal symptom TOS TNTOS neurogenic TOS 1 : 41: , : 24: , : 16: , Wilbourn AJ: The thoracic outlet syndrome is overdiagnosed. 137 Arch Neurol 47: , Roos DB: The thoracic outlet syndrome is underrated. Arch Neurol 47: , Wilbourn AJ: Thoracic outlet syndrome is overdiagnosed. Muscle Nerve 22: , Roos DB: Thoracic outlet syndrome is underdiagnosed. Muscle Nerve 22: , Wilbourn AJ: Thoracic outlet syndrome surgery causing severe brachial plexopathy. Muscle Nerve 11: 66 74, Gilliatt RW, Le Quesne PM, Logue V, et al: Wasting of the hand associated with a cervical rib or band. J Neurol Neurosurg Psychiatry 33: , Levin KH, Wilbourn AJ, Maggiano HJ: Cervical rib and median sternotomy-related brachial plexopathies: a reassessment. Neurology 50: , : : 44 47, : Scalenus medius band 18: , : 7: , : Thermography. Biomedical Thermology 10: , : : , : 58: , : 22: , : 18: , : 1 21: , : 73: , : 7: , : Clin Neurosci 14: , : dogma. 70: , Higashihara M, Sonoo M, Tsuji S, et al: Two-step technique to optimize the medial antebrachial cutaneous nerve response. Clin Neurophysiol 121: , : 65 Suppl 4 : , Hislop HJ, Mongomery J: Daniels and Worthingham s Muscle Testing: Techniquies of Manual Examination. 7th ed. W.B. Saunders Company, Philadelphia, p 170, 2002.

8 Magee KR: Hysterical hemiplegia and hemianesthesia. Postgrad Med 31: , Haerer AF: Dejong s the Neurologic Examination. 5th ed. J.B. Lippincott Company, Philadelphia, pp , : true neurogenic thoracic outlet syndrome TOS 18 39: 411, Aminoff MJ, Olney RK, Parry GJ, et al: Relative utility of different electrophysiological techniques in the evaluation of brachial plexopathies. Neurology 38: , Cuetter AC, Bartoszek DM: The thoracic outlet syndrome: controversies, overdiagnosis, overtreatment, and recommendations for management. Muscle Nerve 12: , Huang JH, Zager EL: Thoracic outlet syndrome. Neurosurgery 55: , Ferrante MA, Wilbourn AJ: The utility of various sensory nerve conduction responses in assessing brachial plexo pathies. Muscle Nerve 18: , : C8 1 47: , Nishida T, Price SJ, Minieka MM: Medial antebrachial cutaneous nerve conduction in true neurogenic thoracic outlet syndrome. Electromyogr Clin Neurophysiol 33: , Hirayama K, Tsubaki T, Toyokura Y, et al: Juvenile muscular atrophy of unilateral upper extremity. Neurology 13: , : 35: , Wilbourn AJ: Thoracic outlet syndrome. In Syllabus, Course D: Controversies in Entrapment Neuropathies. American Association of Electromyography and Electrodiagnosis, Rochester, pp 28 38, Cherington M, Happer I, Machanic B, et al: Surgery for thoracic outlet syndrome may be hazardous to your health. Muscle Nerve 9: , Costigan DA, Wilbourn AJ: The elevated arm stress test: specificity in the diagnosis of thoracic outlet syndrome. Neurology 35 suppl 1 : 74 75, Plewa MC, Delinger M: The false-positive rate of thoracic outlet syndrome shoulder maneuvers in healthy subjects. Acad Emerg Med 5: , Nord KM, Kapoor P, Fisher J, et al: False positive rate of thoracic outlet syndrome diagnostic maneuvers. Electromyogr Clin Neurophysiol 13: 67 74, : entrapment neuropathy 29: , : 6: , Loong SC: The carpal tunnel syndrome: a clinical and electrophysiological study of 250 patients. Clin Exp Neurol 14: 51 65, Jackson DA, Clifford JC: Electrodiagnosis of mild carpal tunnel syndrome. Arch Phys Med Rehabil 70: , Stevens JC, Smith BE, Weaver AL, et al: Symptoms of 100 patients with electromyographically verified carpal tunnel syndrome. Muscle Nerve 22: , 1999.

9 139 Clinical and electrodiagnostic features of the true neurogenic thoracic outlet syndrome TOS : a comparison with the disputed neurogenic TOS MASAHIRO SONOO 1, TETSUO ANDO 2, AYUMI UCHIBORI 3, OSAMU KAWAKAMI 2, YASUNOBU SHOZAWA 1, YUKI HATANAKA 1, MAKOTO TANIGUCHI 4, MANA HIGASHIHARA 5, CHIZUKO OISHI 3, YASUOMI KAWAMURA 1, JUN-ICHI KUNOKI 6, ATSURO CHIBA 3, TERUO SHIMIZU 1 1 Department of Neurology, Teikyo University School of Medicine 2 Department of Neurology, Anjo Kosei Hospital 3 Department of Neurology, Kyorin University School of Medicine 4 Department of Neurosurgery, Tokyo Metropolitan Neurological Hospital 5 Division of Neurology, Department of Internal Medicine 3, National Defense Medical College 6 Department of Spine and Orthopedic Surgery, Japan Red-Cross Medical Center The concept of thoracic outlet syndrome TOS has been a subject of much controversy in USA. Professor Wilbourn argued that the rare true neurogenic TOS TNTOS presenting with the thenar atrophy is the only established disorder, and the other majority of patients who had been diagnosed as TOS, complaining various head, neck and arm symptoms without documented neurophysiological abnormalities, should be designated as disputed neurogenic TOS DNTOS. Such a controversy has not been popularly known in Japan, and there have been few case reports of TNTOS patients, especially with detailed neuropysiological investigations. In this study, we report clinical and electrodiagnostic features of 4 patients with TNTOS who were experienced at our institute for 12 years, and also presented the clinical features of 7 patients who were referred to the first author under the diagnosis of TOS from other doctors. Presenting symptoms of all four TNTOS patients were thenar atrophy or other motor symptoms. Two patients noticed mild sensory symptoms in the medial aspect of the forearm. Neurological examinations revealed the weakness of abductor pollicis brevis APB, and ulnar-innervated small hand muscles to a lesser extent. The flexor digitorum profundus FDP muscle to the index finger was weak, but that to the little finger was preserved for all patients. All patients had objective sensory loss in the medial aspect of the forearm. Motor nerve conduction studies revealed severe decrease of the amplitude of the compound muscle action potential CMAP from the APB muscle and mild decrease of the CMAP from the abductor digiti minimi muscle for all patients. Sensory nerve conduction studies documented loss or severe depression of the sensory nerve action potential SNAP from the medial antebrachial cutaneous MAC nerve and moderate decrease of the SNAP amplitude from the ulnar nerve and from the ring finger following the median nerve stimulation. Needle EMG, when examined, revealed greater involvement of the APB muscle than the first dorsal intereosseous muscle, and the C7-innervated muscles were normal. These electrophysiological results unequivocally indicated the lower trunk plexopathy that was dominant in the T1 components, which agrees with the localization documented for TNTOS by previous authors. The distribution of the weakness of present patients also revealed the T1 innervation of the median FDP muscles and the C8 innervation of the ulnar FDP muscles, which has not been reported previously. Among 7 other patients referred to the first author under the diagnosis of TOS, 5 showed evident hysterical weakness, and masked depression was evident in another patient. In this regard, these patients were thought to be classified under the category of DNTOS. The first author did not gave the final diagnosis of TOS to any other patient during the investigation period of 12 years. TNTOS is a rare disorder presenting with motor-dominant symptoms, and the electrodiagnosis clearly localizes the lesion at the T1-dominnant upper trunk for these patients. This concept should be strictly discriminated from the DNTOS, the presence of which is still controversial. Key Words true neurogenic thoracic outlet syndrome; disputed neurogenic thoracic outlet syndrome; sensory nerve conduction studies; medial antebrachial cutaneous nerve; flexor digitorum profundus muscle

臨床神経 indd

臨床神経 indd 157 C6 T1 C8/T1 TOS true neurogenic TOS T1 C8 Key Words : brachial plexopathy, sensory nerve conduction studies, needle EMG, thoracic outlet syndrome, neuralgic amyotrophy 1 2 1 3 C5 T1 5 root trunk division