Generalized dystonia following resuscitation from a cardiac arrest: a case report and review of the literature

The clinical case we reported corresponds to a generalized dystonia secondary to a cardiac arrest in an 11-year-old girl. One of the particularities of this case was its rarity and its acute onset. Indeed, there are few cases described in the literatures [2, 4, 5]. A wide variety of movement disorders can be observed after cerebral hypoxia, including akineto-hypertonic syndrome and dystonia [3,4,5]. This latter may be focal, affecting a hand, foot, or cranial region, segmental or even generalized [2]. These disorders can occur acutely or with delay [7]. Indeed, in his review, Bhatt et al. found a delay in the onset of signs ranging from 1 week to 36 months after cardiac arrest [4]. There is an age-related difference in relation to the type of abnormal movements. The akineto-hypertonic syndrome is more frequent in the elderly, whereas dystonia is more frequent in young subjects such as our patient [2, 4,5,6]. However, the study of Scheibe et al. [8] have not confirmed this effect of age in the type of abnormal movements occurring after cardiac arrest. This could be related to the predominant inclusion of adults and the sample size in his study [8]. Pathophysiologically, it is generally accepted that lesions of the putamen are more responsible for dystonia, whereas lesions of the pallidum lead to an akineto-hypertonic syndrome [6, 7, 9]. However, cases of dystonia secondary to lesions of the medial pallidum have been reported in the literature [9, 10]. This could explain the clinical manifestations of our patient who presented with ischaemia of the putamen and pallidum. The vulnerability of the basal ganglia to cerebral hypoxia/anoxia has been reported by several authors with different hypotheses, the main one being their terminal vascularization [4, 10]. The treatment of dystonia induced by cerebral hypoxia/anoxia is symptomatic [1]. Our patient received oral baclofen because anticholinergics were not available. Her evolution was favourable with a clear regression of dystonic movements. On the other hand, Wiltshire et al. and Ray et al. [11] did not find this same evolution and used other therapeutic means in association with baclofen [2].

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