Rubral Tremor After Thalamic Infarction in Childhood Hu ¨ seyin Tan, MD*, Gu ¨ zide Turanli, MD † , Hakan Ay, MD ‡ , and Isil Saatc ¸i, MD § The occurrence of tremor after thalamic lesions is well known. Delayed rubral tremor secondary to bilateral thalamic infarction is a rare finding and has not been reported previously in childhood. We present two children with a combined resting-postural-kinetic tremor caused by bithalamic infarction. The first child was a male 14 months of age, and the second was a male 9 years of age. These children come from unre- lated families. On hospital admission of the first pa- tient, generalized seizures and routine electroencepha- logram (EEG) findings with diffuse spike-wave discharges predominantly over the left frontal area were clinically observed, leading to the initial diagnosis of epilepsia partialis continua. However, clinical obser- vation and video-EEG monitoring of the movements revealed nonepileptiform accompaniments, favoring the diagnosis of rubral tremor. In the second patient, EEG revealed no paroxysmal activity and was within normal limits for age. In both patients, cranial mag- netic resonance imaging revealed ischemic lesions in thalami bilaterally but failed to reveal any mesence- phalic lesion. These patients demonstrate that thalamic infarction can cause rubral tremor in childhood. © 2001 by Elsevier Science Inc. All rights reserved. Tan H, Turanli G, Ay H, Saatc ¸i I. Rubral tremor after thalamic infarction in childhood. Pediatr Neurol 2001;25: 409-412. Introduction Tremor is the most common movement disorder. Most patients with tremor have Parkinson’s disease or essential tremor. The remaining types of tremor, consisting of different idiopathic forms that are caused by brain trauma or multiple sclerosis, are uncommon but often give rise to severe disability [1]. The term “rubral tremor” (also called Holmes’ tremor or midbrain tremor) is used to describe a tremor that is revealed at rest and becomes pronounced on goal-directed movement [2,3]. The appearance of tremor after thalamic lesions is well known but infrequent [4,5]. A computer-based scanning of the National Library of Medicine revealed that an association between medial bithalamic infarction and isolated rubral tremor has not been described previously, although thalamic infarction in children (particularly in infancy) has occasionally been reported [6,7]. Therefore we report two children with isolated rubral tremor associated with thalamic infarction. Case Reports Patient 1 A male patient 14 months of age was admitted to the hospital because of high fever and restlessness. He had a 1-year history of Wilms’ tumor, for which a right nephrectomy had been performed 9 months previously. Follow-up abdominal ultrasound scan 3 months postoperatively revealed a left renal mass, 1-2 cm in size. The patient underwent a course of chemotherapy, receiving the last dose 1 week before admission. On admission, the patient was febrile with a temperature of 39.2°C. His pulse was 72/minute, and respiratory rate was 32/minute. Neurologic and physical examinations were otherwise normal. Hematologic and blood chemistry values were in normal limits except for a marked neutropenia (2000 WBC/mm 3 ). An extensive microbiologic analysis, including culture and microscopic examinations of blood, urine, and cerebrospinal fluid, failed to reveal any source of infection. The patient responded well to antibiotic treatment. On the third day of admission, a low-amplitude resting tremor in all limbs was observed. The tremor was more apparent in the distal parts of the upper limbs and the amplitude increased on attempts to reach a target. The following day, additional involuntary movements on the right side of the body, predominantly in the face, neck, and arm, appeared. The amplitude of these 3-Hz clonic movements increased progressively and resulted in abduction and adduction of the right arm at the shoulder joint in the resting position. Neurologic examination revealed synchronous, low-amplitude involuntary movements involving the tongue and the proximal right lower extremity muscles. The movements tended to disappear during sleep, and increased in amplitude on awakening and in response to verbal, tactile, and noxious stimuli. Phenobarbital therapy (5 mg/kg/day) was introduced on the sixth day of admission after two successive afebrile generalized seizures recorded at the time of the abnormal movements. While the movements continued, eight-channel electroencephalogram (EEG) recording revealed bilateral synchronous 3.5-4-Hz spike-and-wave discharges of higher amplitude in the left frontal area. Three-hour video-EEG monitoring revealed the same localized abnormality. A causal relationship between these EEG abnor- malities and the tremor-like movements was difficult to establish. From the *Department of Pediatrics, Atatu ¨rk University Faculty of Medicine, Ankara, Turkey; and the † Department of Pediatric Neurology, the ‡ Department of Neurology, and the § Department of Radiology, Hacettepe University Faculty of Medicine, Ankara, Turkey Communications should be addressed to: Dr. Tan; Hacettepe U ¨ niversitesi; Pediatrik No ¨roloji Bo ¨lu ¨mu ¨; 06100 Sihhiye; Ankara, Turkey. Received January 22, 2001; accepted June 22, 2001. 409 © 2001 by Elsevier Science Inc. All rights reserved. Tan et al: Rubral Tremor in Childhood PII S0887-8994(01)00331-9 ● 0887-8994/01/$—see front matter