(Stroke. 1996;27:991-995.)
© 1996 American Heart Association, Inc.
Articles |
Late Contralateral Hyperhidrosis in Lateral Medullary Infarcts
From the Service de Rééducation et Convalescence Neurologiques (M.R., C.B.) and the Service de Neurophysiologie Clinique (J.F.H., F.C.), Centre Hospitalier Universitaire, Lille, France.
Correspondence to Dr M. Rousseaux, Service de Rééducation et Convalescence Neurologiques, Hôpital Swynghedauw, Centre Hospitalier Universitaire, 59037 Lille Cedex, France.
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Abstract |
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Background and Purpose This study describes unilateral increases of sweating reactions observed in the months after contralateral medullary infarct; evaluation of sympathetic cutaneous response may help to explain sweating disorders.
Summary of Report After the discovery of the clinical phenomenon in one case, patients admitted between 1990 and 1993 were systematically evaluated clinically and electrophysiologically. In a group of five patients presenting with lateral or dorsal medullary lesions, two exhibited an increase of contralateral sweating reactions that appeared 6 to 8 months after stroke, were elicited by effort and exposure to heat and stress, and were more severe over the forehead, face, and upper trunk. In one case, this was clinically associated with an absence of sweating on the side of the lesion. During the late phase after stroke, in three patients presenting with lateral medullary lesions, electrophysiological evaluation revealed significant asymmetry of the sympathetic skin response, which was higher on the side contralateral to the lesion than on the ipsilateral side. In one patient, no response could be elicited by stimulations applied on the side of the lesion.
Conclusions Contralateral hyperhidrosis can be observed in the late phase after lateral medullary infarct and is likely due to lesion of the sympathetic pathway passing through the lateral medulla, which inhibits sudomotor neurons. Evaluation of sympathetic skin response may help to explain such clinical disorders.
Key Words: autonomic dysfunctions • cerebral infarction • medulla oblongata • sweating • Wallenberg's syndrome
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Introduction |
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In patients presenting with Horner's syndrome, unilateral sweating and flushing have been described over the contralateral side of the face1 and more recently on the chest and limbs.2 Most of the corresponding lesions are localized in the second sympathetic neuron, at the level of the first thoracic segments. In only one previously reported case1 have authors suggested medullary injury, without confirmation by neuroradiological investigation. In lateral medullary infarcts, subclinical increase of contralateral sudoral secretion has been recently observed using evaporation rate evaluation.3
The aim of this study is to demonstrate that (1) contralateral hyperhidrosis can be observed in the first few months after lateral medullary infarction, with or without the more classic decrease of ipsilateral sweating,4 5 6 and (2) as previously suggested,7 electrophysiological evaluation of sympathetic skin response may confirm and help to explain clinical disorders.
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Subjects and Methods |
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After the discovery of increased contralateral sweating in one case, we systematically evaluated patients who were admitted to the neurological rehabilitation unit from 1990 through 1993 (Table 1
). Five patients were included. Three presented
with pure lateral medullary infarct (cases 1 through 3), one with
lateral medullary and cerebellar infarct (case 4), and one with
posterior medullary and cerebellar infarct (case 5). Lesions were
evaluated with MR imaging, using an MR-max machine (General Electric)
with a superconducting magnet operating at 0.5 T.
T2-weighted spin-echo sequences were used (repetition
time, 2000 milliseconds; echo time, 100 milliseconds; slice thickness,
5 mm). Infarcts were localized on horizontal slices8 (Fig 1) at the level of the decussation of the corticospinal
tract, the inferior olive complex, and the upper
medulla.
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Case Reports
Case 1
A 56-year-old man with no past neurological history suddenly
presented with right cervical and occipital pain in August
1992. A few minutes later, diplopia, rotary vertigo, and
right-sided gait deviation occurred. Neurological examination also
revealed mild right-sided limb ataxia, severe left-sided
thermal and pain analgesia, and right Horner's syndrome with ptosis
and miosis. Results of lingual and pharyngeal examinations were normal.
Muscle strength and discriminative sensibilities were spared. MRI
revealed on T2 sequences hyperintensity of the right
lateral medulla. During the rehabilitation period (September 1992),
sweating reactions were clinically normal on the left side of the body.
At follow-up (June 1993 and July 1994), severe left
anesthesia for pain and temperature and discrete miosis
persisted. Six months after stroke, contralateral
hyperhidrosis progressively occurred. This disorder was
more severe over the forehead, face, and upper trunk, extending to the
midline with a well-defined vertical limit. It was elicited by
minor physical effort, heat exposure, and stress or emotion and was
very uncomfortable. Skin color was perfectly normal, without flushing
reaction. On the right side, cutaneous examination at rest and during
effort was normal.
Case 2
In September 1992 a 50-year-old man presented with
abrupt vomiting and left incoordination. Examination revealed right
noxious hypesthesia (sparing face); left discriminative hypesthesia;
left limb and truncal ataxia; left multimodal anesthesia of
the face, tongue, and pharynx; phonation and swallowing disorders
associated with left velar palsy; and horizontal-rotatory
nystagmus. Left Horner's syndrome (ptosis and miosis) was also
observed. Severe lesion of the left lateral medulla was seen on MRI. In
the rehabilitation unit (October 1992), no
hyperhidrosis was observed. At follow-up,
neurological examination (November 1992, September 1993, and September
1994) showed severe right hypesthesia for pain and temperature;
discrete gait imbalance and left nystagmus; hypesthesia of the left
face, tongue, and pharynx; and left ptosis without miosis. Six to 8
months after stroke, severe hyperhidrosis occurred on
the right side of the body, with the same characteristics as in the
first case and without flushing. Furthermore, the skin surface was
severely dry on the left side of the body, at rest and during physical
effort, and the patient reported that prelesional sweating reactions
had disappeared.
Case 3
In November 1992 a 37-year-old patient with no past
neurological history presented with sudden gait imbalance.
Examination revealed left gait and limb ataxia, right pain and thermal
anesthesia, left tactile and kinesthetic hypesthesia,
multimodal hypesthesia on the left side of the face, horizontal
diplopia with right nystagmus, and left Horner's syndrome. Results of
pharyngeal and laryngeal examinations were normal. MRI showed a limited
left lateral medullary hyperecho on T2 sequences, without
other lesion. In the rehabilitation unit (December 1992 to January
1993), no hyperhidrosis or hypohidrosis was observed.
At follow-up examinations (June 1993, December 1993, and December
1994), mild gait imbalance, discrete deficit of right epicritic
sensibility, left facial hypesthesia, and left miosis persisted. During
physical effort, sweating reactions were clinically important and
symmetrical on both sides, and the patient reported that no change had
occurred since the stroke.
Case 4
In May 1990 a 48-year-old patient presented with
vomiting, vertigo, and gait imbalance. Examination revealed left limb
and gait ataxia, horizontal-rotatory nystagmus on left gaze, and
left facial hypesthesia. Results of pharyngeal, laryngeal, and tongue
examinations were normal. Hyperecho of the left inferior
cerebellum and dorsolateral medulla was observed on MRI. During
rehabilitation, hyperhidrosis was not reported.
Recovery was relatively rapid. On later evaluations (July 1990, January
1991, and January 1995), discrete imbalance and left rotatory nystagmus
persisted. Sweating reactions and skin color were normal.
Case 5
A 46-year-old woman presented in October 1991 with
vertigo, vomiting, diplopia, and gait imbalance. Examination revealed
right limb and gait ataxia, left tactile hypesthesia, right Horner's
syndrome, right trigeminal deficit, horizontal-rotatory nystagmus
on right gaze, and discrete swallowing disorder. Sensibilities were
normal. On MRI, hyperecho of the right cerebellum and the right
dorsolateral medulla was observed (T2 sequences). On later
evaluations (December 1992, June 1993, July 1994, and January 1995),
gait imbalance and hypesthesia of the left hand persisted. No
hyperhidrosis and no hypohidrosis were observed.
Evaluation of Sympathetic Skin Responses
Sympathetic skin responses were evaluated in four patients
(Table 2), 742 to 1184 days after stroke, using the
method described by Shahani et al9 and by Knezevic and
Bajada.10
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Methods
Recording was performed in a quiet room, at an ambient
temperature of 23°C to 27°C, with patients awake and comfortably
sitting. Evaluation was made with a Viking II E apparatus
(Nicolet), with a band pass of 2 Hz to 5 kHz, amplification of 100
µV/cm, and sweep speed of 500 ms/cm. Stimulating electrodes (Dantec)
were placed over the median nerve at the wrist; recording
electrodes (Dantec) were placed at the palmar (active) and dorsal
(passive) surfaces of the hands and at the plantar (active) and dorsal
(passive) surfaces of the feet, over the second interphalangeal spaces.
Rectangular shocks (0.1 millisecond; 20 mA) were randomly applied to
stimulate the right and then the left median nerves, with interstimuli
intervals of >20 seconds. The amplitude and latency were recorded
simultaneously for both hands or feet. Three stimulations
were performed for each place of stimulation. Mean latency, mean
peak-to-peak amplitude, and asymmetry index (AI) of mean
latencies and mean amplitudes (AI%=[value on the contralateral
side-value on the ipsilateral side]x200/[value on the
contralateral side+value on the ipsilateral side]) were calculated and
compared with reference values obtained in a group of 10 control
subjects ranging in age from 28 to 48 years (mean, 38.6 years; 4 men
and 6 women). As variation of peak-to-peak amplitude was
relatively important, we took the AIs into account. No patient
presented with diabetes mellitus or clinical signs of
peripheral neuropathy; medications that could
influence the sympathetic system were interrupted at the time of the
evaluation.
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Results |
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In each case latencies were relatively symmetrical, without significant increase. Amplitude was reduced in the first two patients, and this was more severe on the ipsilateral side; then a significant asymmetry was observed. Furthermore, in the second patient no response could be elicited by left stimulations (Fig 2
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Evaluation of the third patient also revealed severe asymmetry of
amplitude, which was higher on the right side, contralateral to the
lesion, than on the left side (Fig 3), but the mean
values were relatively high on the contralateral side. In the fifth
patient, amplitudes and AIs were in the normal ranges.
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Discussion |
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TopAbstractIntroductionSubjects and MethodsResultsDiscussionReferences |
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Of five patients with lateral or posterolateral medullary infarct, two presented with hyperhidrosis on the contralateral side of the body and one with ipsilateral anhidrosis. Furthermore, evaluation of sympathetic skin response revealed a significant increase of the AIs of amplitude in each patient presenting with lateral medullary lesion.
In the first case, initial clinical deficits were discretely different from what is usually observed in Wallenberg's syndrome and corresponded to what has been described in more anterior lesions.11 12 13 In patients 2, 3, and 4, clinical disorders were those of the classic Wallenberg's syndrome; pharyngeal and laryngeal sensorimotor deficit was severe in the second case, suggesting a more internal and superior extension of the lesion,14 15 16 17 18 which was corroborated by MRI.
Anhidrosis on the side of the medullary lesion (including the face) was clinically observed in only one case. Unilateral anhidrosis ("Ross syndrome"17 ) has been reported at the level of the face in sympathetic postganglionic lesions.17 18 19 20 In lateral medullary infarcts, such a disorder is most often associated with Horner's syndrome4 5 and has been initially described over the ipsilateral face and neck.21 22 In our patient the forehead, trunk, and limbs were also clinically affected, as also described by Korpelainen et al6 in nine cases, using evaluation of the evaporation rate. Most authors have attributed this to lesions of the central sympathetic pathways8 in the lateral medulla.14 21 22 Winther3 has suggested that fibers supplying sudomotor neurons are situated between the dorsal part of the nucleus ambiguus and the descending root of the trigeminal nucleus, running more anteriorly than the oculosympathetic pathway, and that fibers supplying neurons for the trunk and limbs are in a more anterior situation. In our second patient, who clinically presented with severe anhidrosis, the lesion extended more medially in the area of the nucleus ambiguus8 ; this also supports, as previously suggested by Winther,3 that pathways controlling sweat reactions over the trunk and limbs are medially situated.
Hyperhidrosis has never been described clinically in patients with lateral medullary lesion. The clinical characteristics of this disorder must be stressed: (1) it became perceptible 6 months after stroke onset, (2) it was contralateral to the lesion and extended to the midline with a clearly defined vertical limit, (3) it was severe over the face and trunk, and (4) it was elicited by heat exposure, effort, and stress. In the first two patients, the increase of sweating reactions was relatively different from what has been described in the "Harlequin syndrome." Lance et al1 reported rapid onset of flushing and sweating on one half of the face in five patients; one of them presented with contralateral ptosis and miosis, and the diagnosis was thought to be a right medullary infarct, but MRI was normal. However, the elicitation of increased sweating reactions by heat exposure, effort, and stress was similar. Clinically perceptible and diffuse contralateral hyperhidrosis has been reported in hemispheric lesions,23 24 25 and it has been shown that in such patients sweating asymmetry would be more frequent after 6 months.26 Two similar cases have been described in pontine infarcts,27 28 and with the use of evaporimetry, subclinical hyperhidrosis and increase of vasomotor responses during deep breath and stress were recently demonstrated on the contralateral side of lateral medullary and pontine lesions.5 Lance et al1 suggested that the contralateral flushing could counterbalance the lack of response on the ipsilateral side of the body induced by the sympathetic deficit, which would maintain normal heat regulation. However, a compensatory mechanism does not clearly explain the delayed occurrence of the contralateral hyperhidrosis, the dissociation between such a disorder and normal clinical sweating on the lesion side, as suggested by our first case, and the increase of sweating during stress or emotion. It could also be hypothesized that this disorder has a neuronal substrate. In cerebral infarcts of the middle cerebral artery territory, Labar et al25 have suggested that the lesion could have disrupted a pathway inhibiting sweating on the contralateral side of the body. This could also be evoked in medullary lesions, since sympathoinhibitory bulbospinal systems have been described in animals.29 However, the clinical observations of the delayed onset of hyperhidrosis also suggest a progressively developing increase of sensitivity of sympathetic neurons controlling contralateral sweating reactions.
Investigations of electrodermal reactions using sympathetic skin response to electric shocks have been recently performed in brain stem lesions. In the series of Linden and Berlit,30 two patients presented with medullary infarct, but a precise description of the results was not provided. Serra et al31 described low amplitude of the response on the side of Horner's syndrome. Korpelainen et al7 reported bilateral increase of the latency at the acute phase and bilateral reduction of the amplitude of the sympathetic skin response, more marked on the ipsilateral hand, at the acute and late phases after stroke. Such results are relatively similar to what we observed in the first and second cases; however, in the third case the increase of the AI was associated with both ipsilateral reduction and contralateral increase of the amplitude. Another noticeable observation was that in the second patient, who presented with more severe and medial medullary injury, no response could be elicited by stimulation of the hand ipsilateral to the lesion; this finding suggests that the main pathway going to the posterior hypothalamus and mesencephalic reticular formation, which control sweat reactions,32 33 was destroyed. Thus, evaluation of the sympathetic skin response can also give information about the integrity of the afferent part of the reflex loop. The normality of the response in the fourth patient who presented with dorsal medullary lesion evoked the integrity of the afferent and efferent pathways implicated in the reflex.
In conclusion, this study shows that contralateral hyperhidrosis may be a long-term consequence of lateral medullary infarct and can be observed clinically in some patients. Electrophysiological evaluation can demonstrate subclinical impairment of the efferent pathway of the sympathetic skin reflex, but it can also suggest a lesion of the afferent pathway going to the mesencephalon and hypothalamus.
Received December 14, 1995; revision received February 8, 1996; accepted February 12, 1996.
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