Received September 19, 1996. Accepted in final form March 24, 1997. Address correspondence and reprint requests to Dr. M.G. Hanna, University Department of Clinical Neurology, Institute of Neuralogy, Queen Square, London WClN 3BG, UK. References 1. Heilman KM. Orthostatic tremor. Arch Neurol 1984;41:880881. 2. Britton TC, Thompson PD, van der Kamp W, et al. Primary orthostatic tremor: further observations in six cases. J Neurol 1992;239209-217. Late blink reflex changes in lesions of thalamus and internal capsule 3. McManis PG, Sharbrough FW. Orthostatic tremor: clinical and electrophysiologic characteristics. Muscle Nerve 1993;16: 1254-1260. 4. Pazzaglia p, Sabattini L, Lugaresi E. Su di un singolare disturbo della stazione eretta (osservazione di tre casi). Riv Freniatr 1970;96:450-457. 5. Thompson PD, Rothwell JC, Day BL, et al. The physiology of orthostatic tremor. Arch Neurol 1986;43:584-587. 6. Newsom-Davis J, Mills KR. Immunological associations of acquired neuromyotonia (Isaacs’ syndrome): report of 5 cases and literature review. Brain 1993;116:453-458. 7. Shillito P, Molenaar PC, Vincent A, et al. Acquired neuromyotonia: evidence for antibodies directed against K+ channels of peripheral nerves. Ann Neurol 1995;38:714-722. Article abstract-A 60-year-old man suddenly suffered from left hemiplegia with sensory loss on the left side of his face, trunk, and extremities. Brain CT disclosed hemorrhage in the right thalamus and internal capsule. Blink reflex with electrical stimulation on the left supraorbital nerve showed normal early response (Rl) on the left side but absent late response (R2) on both sides, while stimulation of the right supraorbital nerve showed normal R1 on the right side and normal R2 on both sides. This observation suggests that the impulses of R2 from stimulation of the supraorbital nerve on the affected side may be blocked by contralateral lesions of the thalamus and internal capsule. NEUROLOGY 1997;49:874-876 Lie-Gan Chia, MD Blink reflex evoked in the orbicularis oculi muscles by electrical stimulation of the supraorbital nerve consists of two COmpOnentS, an early oligosynaptic ipsilateral component, R1, and a late polysynaptic bilateral component, R2.12 The afferent limb of this reflex comes from sensory root of the trigeminal nerve and the efferent limb from the facial nerve. It is generally agreed that the central pathway of the R1 may conduct through the p o n ~ . lThe , ~ exact central pathway of the R2 is not entirely understood. It has been presumed that the longer latencies of R2 may travel along the longer pathways up to the opposite thalamus or cortex where they project down to bilateral facial n ~ c l e iThe . ~ longer latencies of R2 are suggested to include R1 latency and to-and-fro duration of somatosensory evoked potential between the upper cervical cord (N14) and the thalamus or cortex (N20) as well as the central delay of many synapse^.^ Although R2 pathways via the lower brainstem to both facial nuclei cannot be e ~ c l u d e d ,this ~ is not supported by the longer latencies of R2.4 In order to verify the pathway of late blink reflex, we studied the blink reflex of a patient with lesions in the thalamus and internal capsule. Case report and electrophysiologic study. A 60year-old man suddenly suffered from left hemiplegia. He was immediately sent t o a local clinic, where brain CT disclosed hemorrhage in right thalamus and internal capsule, and lateral ventricles (figure 1).He was then transferred to our hospital for further evaluation. 874 Copyright 0 1997 by the American Academy of Neurology On admission, he had normal heartbeat and respiration with an elevated blood pressure of 180/120 mm Hg. He was alert and oriented. Neurologic examination showed sensory loss of pain, touch, vibration, and joint position on left side of the face, trunk, and extremities. There was motor Figure 1. CT showing hemorrhage in right thalamus and internal capsule, and laterul uentricles. Recording side Left i L Left supra-orbital nerve R1 Right supra-orbital nerve Jzoopv R1 R2 1Oms Right t Stimulation Stimulation Figure 2. Blink reflex showing norma1 Rl (10.8 ms) on left side, but absent R2 on both sides, as stimulation on left supraorbital nerue; normal R1 (10.4 ms) on right side and normal R2 (32.1 ms, left, and 32.4 ms, right) on both sides, as stimulation on right supraorbital nerue. showed normal R1 (10.4 msec) on the right side and normal R2 (32.4 msec, right; and 32.1 msec, left) on both sides (figure 2). I If stimulation supraorbit. n. sensory nucleus V MEDULLA OBLONGATA \ 111 fibers from dorsal column & ventral spinothalamus tract of spinal cord Figure 3. Diagram showing the presumed location of brainstem-thalamus (internal capsule)-cortex interneurons interpreting the two components of blink reflex. The shaded area (hemorrhage) in right thalamus and internal capsule involves R2 from stimulation of left supraorbital nerue. R1 (early reflex, ipsilateral), R2 (late reflex, bilateral), T h (thalamus), IC (internal capsule), M L (medial lemniscus). weakness on left extremities, especially on the arm. Tendon reflexes were present with slight increase on left side. Extensor plantar responses were present on left side. His upward gaze was slightly limited. Dysarthria and left central facial palsy were noted. Corneal reflex was impaired on left side. He was known to have hypertension without regular treatment for about 1 year, but denied history of diabetes mellitus, heart, liver, or renal diseases. Results from routine laboratory tests of blood, urine, and ECG were all normal. Electrically elicited blink reflex4 was studied with a DISA electromyograph on the fourth hospital day. Blink reflex with electrical stimulation on left supraorbital nerve showed normal R1 (10.8 msec) on left side, but absent R2 on both sides; stimulation on right supraorbital nerve Discussion. It has been suggested that R2 blink reflex is conducted via polysynaptic pathways in the lower brainstem, from the spinal trigeminal nucleus to both facial n ~ c l e i However, .~ it is hard to explain why, over the short distance in the lower brainstem, R2 latency (25-40 msec) is so much longer than R l latency (8-12 msec). It seems reasonable to assume that the impulses of R2 may travel along the longer pathways up to the opposite thalamus o r cortex where they project down to bilateral facial n ~ c l e i . ~ The longer pathway up to the thalamus for R2 is also suggested by animal experiments.6 In the present case, the impulses of R2 from stimulation of left supraorbital nerve were blocked by hemorrhage in right thalamus and internal capsule (figure 3). The absence of R2 may be explained on the basis of loss of hemispheral i n f l u e n ~ e s . ~However, -l~ it is difficult to interpret the present case, which shows the absence of bilateral R2, with the presence of ipsilateral R1. Because hemispheral lesions, which do not involve the brainstem reflex centers, affect the brainstem trigeminal and facial systems of both R1 and R2, the dissociation of R1 and R2 behavior cannot be explained entirely by hemispheral influences. In the present case, the absence of bilateral R2 and the presence of ipsilateral R1 suggest that contralateral lesions of the thalamus and internal capsule block the R2 pathways. From the Section of Neurology, Taichung Veterans General Hospital, Taichung, Taiwan, R.O.C. Received December 26, 1996. Accepted in final form April 21. 1997. Address correspondence and reprint requests to Dr. Lie-Gan Chia. Section of Neurology, Taichung Veterans General Hospital, Taichung, Taiwan (4071, R.O.C. References 1. Kimura J, Powers JM, Van Allen MW. Reflex response of orbicularis oculi muscle to supraorbital nerve stimulation. Study in normal subjects and in peripheral facial paresis. Arch Neurol 1969;21:193-199. 2. Kugelberg E. Facial reflexes. Brain 1952;75:385-396. 3. Namerow NS. The orbicularis oculi reflex in multiple sclerosis. Neurology 1970;20:1200-1203, 4. Chia LG, Shen WC. Wallenberg’s lateral medullary syndrome with loss of pain and temperature sensation on the contralateral face: clinical, MRI and electrophysiological studies. J Neurol 1993;240:462-467. September 1997 NEUROLOGY 49 875 5. Ongerboer De Visser BW, Kuypers HGJM. Late blink reflex changes in lateral medullary lesions: a n electrophysiological and neuroanatomical study of Wallenberg's syndrome. Brain 1978;101:285-294. 6. Tokunaga A, Oka M, Murao T, et al. An experimental study on facial reflex by evoked electromyography. Med J Osaka Univ 1958;9:397-411. 7. Kimura J. Effect of hemispheral lesions on the contralateral blink reflex. Neurology 1974;24:168-174. Cat-scratch encephalopathy I 8. Dengler R, Kossev A, Gippner C, Struppler A. Quantitative analysis of blink reflexes in patients with hemiplegic disorders. EEG Clin Neurophysiol 1982;53:513-524. 9. Fisher MA, Shahani BT, Young RR. Assessing segmental excitability after acute rostra1 lesions: the blink reflex. Neurology 1979;29:45-50. 10. Berardelli A, Accornero N, Cruccu G, Fabian0 F, Guerrisi V. The orbicularis oculi response after hemispheral damage. J Neurol Neurosurg Psychiatry 1983;46:837-843. Article abstract-We present a case of cat-scratch disease in a 9-year-old girl, complicated by encephalopathy and seizures. Bartonella (formerly Rochalimaea) henselae is the causative agent in cat-scratch disease; methods now available for detection of this pleomorphic, gram-negative bacterium, including polymerase chain reaction amplification and indirect fluorescence antibody testing, may lead to changes in standard criteria used to verify a diagnosis of cat-scratch disease. NEUROLOGY 1997;49:876-878 Steven W. Wheeler, MD; Sheldon M. Wolf, MD; and Evan A. Steinberg, MD Cat-scratch disease (CSD) is a bacillary infection that occurs predominantly in children and adolescents. The illness is typically self-limited, characterized by regional lymphadenitis following cutaneous inoculation, and variably accompanied by low-grade fever, malaise, and headache. Neurologic complications associated with CSD are rare, with encephalopathy accounting for the majority of these cases. Bartonella (formerly Rochalimaea) henselae, a pleomorphic, gram-negative bacillus, is the etiologic agent of CSD.1-3Methods now available for detection of this bacterium, including polymerase chain reaction (PCR) amplification, as well as indirect fluorescence antibody testing, may lead to changes in standard criteria used to verify a diagnosis of CSD. We report a case of a child with CSD complicated by encephalopathy and briefly review the neurologic manifestations of this disease. Case report. A previously healthy 9-year-old girl was admitted to the hospital with a 2-week history of rightsided cervical adenitis and a 2-day history of low-grade fevers, diarrhea, and headache. Treatment with oral antibiotics over an 11-day period prior to admission (cephalexin over 3 days, followed by rifampin for 7 days, and amoxacillin-potassium clavulanate during the final 4 days) produced no discernible symptomatic improvement. Two episodes of emesis were temporally associated with ingestion of amoxacillin-potassium clavulanate. Purified protein derivative (PPD) testing was negative, and there was no history of recent travel. Although she had four kittens a t home, there were no known recent scratches or bites from cats. Following needle aspiration of the right cervical mass, the patient was hospitalized for intravenous antibiotics (cefuroxime, gentamicin) plus oral rifampin. On initial examination, vital signs were normal except for a tempera876 Copyright 0 1997 by the American Academy of Neurology ture of 38.0 "C. A 5 X 3-cm, tender superior right cervical mass, and a 1-cm nontender left midcervical mass, were noted. There was no other adenopathy or organomegaly. Neurologic examination was normal. Ten hours after admission, the patient had a generalized tonic-clonic seizure lasting 2 minutes, which aborted with administration of intravenous lorazepam. Postictal combativeness and delirium were followed by stupor and coma. Temperature was 39.2 "C. Neurologic examination demonstrated slight withdrawal of the limbs t o noxious stimulation, with slow roving eye movements, extensor plantar responses, and bilateral ankle clonus. Noninfusion CT brain imaging was negative. EEG revealed generalized theta and delta frequencies, with no evidence of epileptiform activity. Serum WBC, electrolytes, BUN, creatinine, and liver function tests were normal. Erythrocyte sedimentation rate was elevated at 73 m d h . Studies of CSF were normal except for increased protein of 72 mg/dl. Cultures of blood, urine, and CSF were negative. Serologies for Epstein-Barr virus, tularemia, and syphilis were negative. After 24 hours, the patient's mental status began t o improve. No further seizures occurred, and no anticonvulsant medication was given. By the fifth hospital day, neurologic examination was normal. She had intermittent fever until the sixth hospital day and was discharged the following afternoon. Two weeks later, incision and drainage of the right cervical node was performed. The necrotic tissue obtained at surgery was negative for organisms on special staining, and cultures for bacteria, acid-fast bacilli, and fungi were negative. Serum B henselae serology was positive a t 123192. PCR analysis of frozen lymph node tissue, performed by the Centers for Disease Control and Prevention, Atlanta, GA, was positive for B hensalae. The patient has remained seizure-free, with a normal neurologic examination and normal EEG 30 months after initial presentation. Late blink reflex changes in lesions of thalamus and internal capsule Lie-Gan Chia Neurology 1997;49;874-876 DOI 10.1212/WNL.49.3.874 This information is current as of September 1, 1997 Updated Information & Services including high resolution figures, can be found at: http://www.neurology.org/content/49/3/874.full.html References This article cites 10 articles, 2 of which you can access for free at: http://www.neurology.org/content/49/3/874.full.html##ref-list-1 Permissions & Licensing Information about reproducing this article in parts (figures,tables) or in its entirety can be found online at: http://www.neurology.org/misc/about.xhtml#permissions Reprints Information about ordering reprints can be found online: http://www.neurology.org/misc/addir.xhtml#reprintsus Neurology ® is the official journal of the American Academy of Neurology. Published continuously since 1951, it is now a weekly with 48 issues per year. Copyright . All rights reserved. Print ISSN: 0028-3878. Online ISSN: 1526-632X.