JOURNAL OF THE NEUROLOGICAL SCIENCES ELSEVIER Journal of the Neurological Sciences140( 1996) 123- 128 Short report Bilateral perioral sensory symptom after unilateral stroke: Does it have a localizing value? Jong S. Kim * Department of Neurology, Unii,ersi& of Ulsan. Asan Medical Center, Song-Pa PO Box 145, Seoul 1.38~600, South Korea Received 3 1 October 1995;revised 20 February 1996;accepted26 February 1996 Abstract Bilateral perioral sensory dysfunction due to unilateral cerebral lesion is rare, and has been thought to be causedby midline brainstem lesions. Six patients are described herewith, with bilateral perioral sensory symptoms due to unilateral strokes that do not involve the mid-brainstem region. Brain-computed tomographic scan and magnetic resonanceimaging demonstratedunilateral cerebral strokesin the thalamus, posterior limb of the internal capsule, basal ganglia, fronto-parietal cortex and the insular-frontal subcortical area. This observation suggests that bilateral perioral sensory symptoms do not have a localizing value. Possible pathogenetic mechanisms for this sign are discussed. Keywords: Bilateral: Sensation;Perioral; Cerebral lesion; Cerebrovasculardisorder 1. Introduction Ipsilateral motor dysfunction following unilateral cerebral lesion has been repeatedly documented (Glees and Cole, 1952; Brodal, 1973; Jones et al., 1989; Colebatch and Gandevia, 1989; Yelnik et al., 1993) which supports the presence of the bilateral efferent impulses from the motor cortex (Glees and Cole, 1952; Yakovlev and Rakic, 1966; Tanji et al., 1988; Aizawa et al., 1990; Kim et al., 1993a,b). Since a part of the sensory fibers also travels uncrossed (Tasker et al., 1976; Tasker, 1976; Nieuwenhuys et al., 1988), ipsilateral sensory symptoms are anticipated in patients with unilateral cerebral lesions (Semmes and Mishkin, 1965). However, so far only a few reports have described subtle ipsilateral dysfunction of discriminative sensation detected by meticulous sensory tests (Carmen and Benton, 1969; Corkin and Milner, 1970; Corkin et al., 1973; Kim and Choi-Kwon, 1996), while clinically overt ipsilateral sensory changes have not been reported even in recent studies with a large number of patients presenting with sensory stroke (Fisher, 1982; Kim, 1992; Bassetti et al., 1993). * Corresponding author. Tel: (+ 82-2) 224 3442; Fax: (+ 82-2) 474 4691. The only possible exceptions appear to be bilateral facial sensory symptoms due to lateral medullary infarction (Currier et al., 1961; Caplan et al., 1992; Kim et al., 1994b) or paramedian pontine lesions (Matsumoto et al., 1989; Helgason and Wilbur, 1991; Yasuda et al., 1988; Reutens, 1990; Kim and Lee, 1994; Kim et al., 1994a). These reports, along with previously reported ones before the era of MRI (Caplan and Gorelick, 1983), suggested that bilateral facial or perioral sensory symptoms are a localizing sign of brainstem lesion. However, in the present study, I report 6 patients who developed bilateral perioral sensory disturbances due to lesions located elsewhere, illustrating that bilateral perioral symptoms do not have a localizing value. 2. Case reports 2.1. Patient A 73-year-old hypertensive woman suddenly developed mild left hemiparesis and sensory changes in the left half of her body. On admission, the hemiparesis soon resolved itself. There was mildly decreased sensation in all modalities in the left half of her body including the face. She felt paresthesia in the distal part of her left forearm and 0022-5lOX/96/$15.CXl Copyright 0 1996 Elsevier Science B.V. All rights reserved. PII SOO22-510X(96)00078-0 1 124 J.S. Kim/ Journal of the Neurological Sciences 140 (1996) 123-128 Fig. 3. Patient 3: CT shows an infarct in the right posterior limb of the internal capsule (arrowhead). Fig. 1. Patient 1: T2-weighted MRI shows a small infarct in the right thalamus (arrow). mild paresthesia over the left palm remained, while bilateral perioral paresthesia resolved. 2.2. Patient 2 bilateral perioral area. The paresthesia over these areas persisted although the objective sensory deficit gradually improved. Brain MRI performed 6 days after the onset showed a small right thalamic infarction (Fig. 1). MR angiogram results were unrevealing. Three weeks later, A 61-year-old hypertensive man developed sudden onset of mild left hemiparesis and paresthesia in the left half of his body. These symptoms improved in a few days. However, the paresthesia over the left perioral area remained. The area of perioral paresthesia gradually ex- Fig. 2. Patient 2: CT shows a hemorrhage in the right thalamus. J.S. Kim/Journal of the Neurological Fig. 4. Patient 4: CT shows a hemorrhage in the left putamen. tended to the right side. Brain CT scan performed 4 days after the onset showed right thalamic hemorrhage (Fig. 2). The patient complained of a persistent, uncomfortable sensation described as ‘something thick was covered’ over both sides of the lower perioral area. This symptom was not relieved by carbamazepine and persisted at 7 months of follow up. Repeat brain MRI at this time showed an old 125 Sciences 140 (19%) 123-128 Fig. 5. Patient 5: T2-weighted fronto-parietal area. MRI shows an infarct in the right hemorrhage in the same location without any other new lesions. 2.3. Patient 3 A 62-year-old hypertensive and diabetic woman experienced dizziness followed by left hemiparesis. On admis- Fig. 6. Patient 6: T2-weighted MRI shows an infarct involving the right insular cortex (A) and frontal subcortical area (B). 126 J.S. Kim /Journal of the Neurological Sciences 140 C1996) 123- 128 sion, the patient showed left facial paresis, hemiparesis and left hemianopia. The sensation was slightly decreased in the left face and extremities in all modalities. Brain CT taken 7 days later showed an infarct in the right posterior limb of the internal capsule (Fig. 3). Angiogram results showed mild atherosclerotic narrowings in the both internal carotid arteries. The patient’s motor and sensory symptoms gradually improved, but she complained of persistent paresthesia over the distal part of her left arm and both perioral areas, Pinprick sense was felt slightly altered in those areas. One month later, the perioral paresthesia slightly decreased in severity. 2.4. Patient 4 A 72-year-old hypertensive woman developed sudden right hemiparesis. On examination, she had dysarthria, right facial paresis, hemiparesis and slightly decreased pinprick and touch sensation in the right. Brain CT showed a left putaminal hemorrhage (Fig. 4). The neurologic symptoms gradually improved. However, she gradually developed paresthesia over both perioral areas where objective sensation remained intact. The bilateral perioral paresthesia persisted at 7 months of follow-up. 2.5. Patient 5 A 67-year-old hypertensive woman became drowsy and developed left hemiparesis. On admission, the patient showed left facial paresis, orobuccal apraxia, left hemiparesis, hemineglect, hemianopia and hemihypesthesia of all sensory modalities. Brain MRI showed a large infarct in the right middle cerebral artery territory (Fig. 51. Angiogram was refused by the patient. The patient’s motor and sensory symptoms rapidly improved, but she developed persistent paresthesia over the distal part of her left arm and both perioral areas. At 1 month of follow-up, the patient’s weakness was almost confined to the left hand and fingers. She complained of paresthesia over the left palm where sensation was decreased in all modalities. She also had persistent tingling sensation over both sides of the perioral area where pinprick and vibration sense were slightly decreased. 2.6. Patient 6 A 7 1-year-old hypertensive woman suddenly developed left hemiparesis. On admission, the patient was alert, but dysarthric without aphasia. She showed left facial paresis and mild left hemiparesis worse on the upper extremity. Pinprick, temperature and vibration sensation were normally perceived, but sensory extinction and impaired graphesthesia were noted in the left limbs. Brain MRI performed 3 days later showed an infarction involving the right insular cortex and frontal subcortical area (Fig. 6). MR angiogram findings were unrevealing. The patient’s motor symptoms rapidly resolved, but she gradually developed paresthesia in both perioral areas where objective sensation remained intact. At 4 months of follow-up, the perioral paresthesia was still present. 3. Discussion The 6 patients described here had acute unilateral cerebral lesions, i.e., infarction in 4 and hemorrhage in 2, and experienced distinct sensory changes over the bilateral perioral areas. In all patients, imaging studies and clinical history strongly illustrated that there were no significant lesions in the side contralateral to the main lesion. Relatively large infarcts located on the lateral medulla can cause bilateral facial sensory changes probably due to an involvement of both the descending trigeminal fibers and ascending secondary trigeminal-thalamic tracts (Currier et al., 1961; Caplan et al., 1992; Kim et al., 1994b). Paramedian mid-pontine lesions can also cause bilateral sensory deficit especially in the perioral area (Matsumoto et al., 1989; Helgason and Wilbur, 1991; Yasuda et al., 1988; Kim and Lee, 1994; Kim et al., 1994a). The prevailing explanation for this phenomenon was that the lesions situated in the paramedian pons can involve both sides of the medial parts of the medial lemniscus, where the ascending trigeminal sensory fibers are located. However, our observation illustrated that bilateral perioral sensory symptoms can be caused by thalamic (patients 1 and 21, capsular (patient 31, putaminal (patient 4) and cortical-subcortical (patients 5 and 6) strokes. Thus, although bilateral facial or perioral paresthesia may occur most frequently after brainstem lesions, it does not, in a strict sense, have a localizing value. It is noticeable that our patients had bilateral sensory dysfunction only in the perioral area with the ipsilateral hand or foot being left unaffected. Therefore, the perioral area seems to be more vulnerable for bilateral sensory dysfunction compared to other parts of the body. Picard and Olivier (19831, after electrically stimulating the cortex of the 100 patients, found that the sensory responses were noted in the ipsilatera1 or bilateral area of the tongue in 6 and 7% of the responses respectively. Tasker et al. (1976) observed induction of occasional bilateral paresthesia over the mouth, lip, or face after unilateral stimulation of a certain area of the thalamus. More recently, Walter et al. (1992) investigated cortical activation provoked by movement of foot, thumb, index and mouth using magnetoencephalography, positron emission tomography and MRI, and found that only the mouth movement induced bilateral cortical activation. The anatomical substrate related to the ipsilateral trigeminal sensory projection could be the uncrossed dorsal trigeminothalamic tracts which originate from the dorsomedial part of the principal sensory nucleus (Nieuwenhuys et al., 1988). Alternatively, the bilateral symptoms may be mediated by yet poorly defined medially located sensory pathway, J.S. Kim/Journal of the Neurological Sciences 140 (19%) 123-128 the stimulation of which induces bilateral paresthesia (Nashold et al., 1969). It is noteworthy that the medial part of the midbrain near the aqueduct has fiber tracts related to the central regions of the face, especially the oral and nasal cavities (Nashold et al., 1969). Finally, the sensory symptoms on the ‘good’ side of the face may be related to transhemispheric diaschisis (Andrews, 1991) or bilateral reorganization process of neuronal circuitry after acute focal brain injury (Kucera and Wiesendanger, 1985; Chollet et al., 1991; Fisher, 1992; Weiller et al., 1992). The perioral sensory symptoms our patients experienced were mainly positive one (paresthesia), and may thus be equivalent to recently described hyperkinetic motor behaviors in the side ipsilateral to the unilateral stroke (Ghika et al., 1995). 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