J Neurol (2010) 257:478–480 DOI 10.1007/s00415-009-5394-x
LETTER TO THE EDITORS
Persistent pre-sleep behaviour and paroxysmal sweating with a stab lodged in the head Margherita Fabbri • Roberto Vetrugno • Joa˜o Eudes Magalha˜es • Joaquim Jose´ de Souza Costa-Neto • Indira Luz Benevides • Mario Mascalchi Pasquale Montagna
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Received: 2 September 2009 / Revised: 5 November 2009 / Accepted: 9 November 2009 / Published online: 25 November 2009 Ó Springer-Verlag 2009
Dear Sirs, Behavioural, autonomic and sleep disorders may follow traumatic brain injury [1–4]. We report a patient in whom a stab lodged into the brain was associated to chronic headache, excessive daytime sleepiness and recurrent episodes of asymmetric excessive sweating and piloerection. A 26-year-old man received a stab in the forehead during an assault about which he was amnesic. Admitted to an emergency wait, he was however discharged with the stab still lodged in his head. One year later he come to our attention complaining of daily headache, tiredness and constant desire to sleep. According to his wife, his initiative was decreased and he often yawned, stretched and assumed a sleeping posture. However, when given active tasks, he was able to perform them correctly. In addition he had become quiet, ‘‘easy-going’’, kind and overtly religious. Finally, he complained of paroxysmal excessive sweating triggered by exposure to hot temperatures, and rarely by apprehension, when he broke out in profuse sweating and piloerection limited to the left side of the body. Hot-induced excessive sweating was preceded by a
M. Fabbri R. Vetrugno (&) P. Montagna Department of Neurological Sciences, University of Bologna, Via Ugo Foscolo 7, 40123 Bologna, Italy e-mail:
[email protected] J. E. Magalha˜es J. J. de Souza Costa-Neto I. L. Benevides Hospital das Clı´nicas da Universidade Federal de Pernambuco, Recife, Brazil M. Mascalchi Radiodiagnostic Section, Department of Clinical Physiopathology, University of Florence, 50134 Florence, Italy
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feeling of ‘‘inner heat’’, and was a source of considerable fatigue and embarrassment. General examination revealed a linear scar in the left forehead. Body temperature was 36.6°C, heart rate 65 beats/ min and regular, respiratory rate 14 breaths/min, and blood pressure 125/75 mm Hg. Epworth Sleepiness Scale score, for measuring subjective daytime sleepiness, was 12 (n.v. B 10) [5]. Skull X-ray showed a metal stab which from the left frontal region projected just right of the midline into the suprasellar region (Fig. 1a). Marked beam hardening artefacts due to the metal blade hindered brain evaluation with cranial CT. Digital subtraction arteriography revealed that the tip of the stab was located just in front of the distal portion of the A1 segment of the anterior right cerebral artery beside the anterior communicating artery (Fig. 1b). This suggested that the distal portion of the blade had crossed the left diencephalon and reached the right hypothalamus. Neuropsychological evaluation [Wechsler Adult Intelligence Scale III score: 89 (lower limits); Wisconsin Card Sorting Test score: 94 mistakes on 128 (no category was completed); Rey’s 15 Words Immediate Recall Test score: 49/75 (corrected: 44.55/75); Rey’s Words Delayed Recall Test score: 4/15 (corrected: 2.25/15); Rey’s Complex Figure Copy Test score: 36/36 (corrected: 37.03); Rey’s Complex Figure Reproduction Test score: 14/36 (corrected: 11.97/36)] revealed impairment of long-memory and executive functions. Nocturnal sleep was unremarkable, but upon multiple sleep latency test (MSLT) he fell asleep during each session, with a mean sleep latency of 8min 30sec and without sleep onset REM periods. However, between every MSLT session, the patient constantly held a pre-sleep behaviour, all the time lying in bed or sitting on a chair, repetitively yawning and often with closed eyes. However, he could promptly resume full-wake behaviour upon stimulations. Assessment of body core temperature
J Neurol (2010) 257:478–480
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Fig. 1 a Skull X-ray (lateral projection) showing a blade that from the frontal region projects into the suprasellar area. Inset in a indicates a linear vertical scar in the left forehead. b Digital subtraction arteriography of the right internal carotid artery (coronal oblique projection) revealing that the tip of the blade (arrow) is located just in front of the distal portion of the A1 segment of the anterior right
cerebral artery beside the anterior communicating artery. c Excessive sweating evident only on the left side of the face during hot exposure. d Relationship between ambient temperature and sweating spells, showing two sweating episodes (transverse arrows) arising with ambient temperature above 30°C during which tympanic temperature increases in the right but not in the left ear
was done by means of thermistor probes applied to the left and right tympanic membranes, manipulating room temperature from 24 to 32°C. No sweating was observed when room temperature was below 29.5°C. On two occasions, as soon as room temperature reached 30–32°C, sweating and shivering were observed on the left side of the body (Fig. 1c). During the sweating episodes tympanic temperature was lower on the left side (by 1.3 and 0.8°C) (Fig. 1d). The patient was instructed to live in air conditioned ambient at 26°C, however, he preferred to return to his native village in the mountains where the temperature is lower, and is still on a waiting list for surgery. The peculiarity of the present case consists in the fact that the offending traumatic tool remained in place thus enabling us to make inference on the pathophysiology of the symptoms [6]. The personality changes and the results of neuropsychological tests in our patient are in line with frontal lobe
damage [7]. This was combined with compulsive pre-sleep behaviour and excessive daytime sleepiness consistent with diencephalic damage [8]. Also the left side paroxysmal sweating spells pointed to hypothalamic damage. In particular, since the hypothalamus provides a descending pathway that exerts an inhibitory influence on sympathetic neurons activating contralateral sweating [9], the right hypothalamic damage indicated by the radiological location of the blade tip well explained the asymmetric sweating and the derangement of the thermoregulation in our patient.
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