urinary dysfunction in brown-séquard syndrome

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Neurourology and Urodynamics 20:661–667 (2001) Urinary Dysfunction in Brown-Se ´ quard Syndrome Ryuji Sakakibara, 1 * Takamichi Hattori, 1 Tomoyuki Uchiyama, 1 and Tomonori Yamanishi 2 1 Department of Neurology, Chiba University School of Medicine, Chiba, Japan 2 Department of Urology, Chiba University School of Medicine, Chiba, Japan Brown-Se ´quard syndrome (BS) is a rare but well documented condition of the spinal cord hemisection, comprising hemiparesis with crossed superficial sensory disturbance. However, little is known of micturitional function in BS, although some patients with BS are troubled with severe voiding difficulty and urinary incontinence. We performed urinary questionnaire and urodynamic studies in eight patients with BS, including seven men and one woman, mean age of 41 years. Detailed questionnaire showed that five of the eight patients (63%) had micturitional symptoms, including voiding difficulty in three, urinary retention in two, urinary frequency in two and urge urinary incontinence in one. Urodynamic abnormalities were noted in all five patients with micturitional symptoms, including post-micturition residuals in four (average 149 mL), high urethral closure pressure in two, increased bladder volume at first sensation in one, detrusor hyperreflexia in four, detrusor areflexia on voiding in three and unrelaxing sphincter on voiding in four. Three asymptomatic patients showed normal urodynamic finding. Micturitional symptoms were more common in patients with severe motor paresis (100%) than in those with mild motor paresis (40%), and there was no relation between micturitional disturbance with superficial or deep sensory disturbance. A combi- nation of treatments for the underlying disorders with a-adrenergic blocking agent and clean, intermittent self-catheterization ameliorated the urinary dysfunction in all patients together with neurological dysfunction. In conclusion, micturitional disturbance was not uncommon in our patients with BS, particularly in those with severe motor paresis, which could ameliorate by appropriate therapies. Neurourol. Urodynam. 20:661–667, 2001. ß 2001 Wiley-Liss, Inc. Key words: Brown-Se ´quard syndrome; micturitional disturbance; urinary retention; urodynamic study INTRODUCTION Brown-Se ´quard syndrome (BS) is a rare but well documented condition which results from hemisection of the spinal cord [Brown-Se ´quard, 1849, 1868]. In contrast to hemiparesis of cerebral origin, that of spinal origin is crossed with respect to the associated hemianesthesia, and provided the disorder is due to a single lesion, this finding is pathognomonic of a spinal cord disorder [Rondt, 1969]. The classical syndrome consists of hemiparesis (or monoparesis of the leg) ipsilateral to the lesion (pyramidal tract disorder), contralateral loss of superficial sensation (spinothalamic ß 2001 Wiley-Liss, Inc. *Correspondence to: Ryuji Sakakibara, M.D., Neurology Department Chiba University, 1-8-1 Inohana Chuo-ku, Chiba 260-8670, Japan. E-mail: [email protected] Received 14 May 2000; Accepted 15 October 2000

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Page 1: Urinary dysfunction in Brown-Séquard syndrome

Neurourology and Urodynamics 20:661±667 (2001)

Urinary Dysfunction in Brown-SeÂquardSyndromeRyuji Sakakibara,1* Takamichi Hattori,1 Tomoyuki Uchiyama,1

and Tomonori Yamanishi2

1Department of Neurology, Chiba University School of Medicine, Chiba, Japan2Department of Urology, Chiba University School of Medicine, Chiba, Japan

Brown-SeÂquard syndrome (BS) is a rare but well documented condition of the spinal cordhemisection, comprising hemiparesis with crossed super®cial sensory disturbance. However,little is known of micturitional function in BS, although some patients with BS are troubledwith severe voiding dif®culty and urinary incontinence. We performed urinary questionnaireand urodynamic studies in eight patients with BS, including seven men and one woman, meanage of 41 years. Detailed questionnaire showed that ®ve of the eight patients (63%) hadmicturitional symptoms, including voiding dif®culty in three, urinary retention in two, urinaryfrequency in two and urge urinary incontinence in one. Urodynamic abnormalities were notedin all ®ve patients with micturitional symptoms, including post-micturition residuals in four(average 149 mL), high urethral closure pressure in two, increased bladder volume at ®rstsensation in one, detrusor hyperre¯exia in four, detrusor are¯exia on voiding in three andunrelaxing sphincter on voiding in four. Three asymptomatic patients showed normalurodynamic ®nding. Micturitional symptoms were more common in patients with severemotor paresis (100%) than in those with mild motor paresis (40%), and there was no relationbetween micturitional disturbance with super®cial or deep sensory disturbance. A combi-nation of treatments for the underlying disorders with a-adrenergic blocking agent and clean,intermittent self-catheterization ameliorated the urinary dysfunction in all patients togetherwith neurological dysfunction. In conclusion, micturitional disturbance was not uncommon inour patients with BS, particularly in those with severe motor paresis, which could ameliorateby appropriate therapies. Neurourol. Urodynam. 20:661±667, 2001. ß 2001 Wiley-Liss, Inc.

Key words: Brown-SeÂquard syndrome; micturitional disturbance; urinary retention; urodynamicstudy

INTRODUCTION

Brown-SeÂquard syndrome (BS) is a rare but well documented condition whichresults from hemisection of the spinal cord [Brown-SeÂquard, 1849, 1868]. In contrast tohemiparesis of cerebral origin, that of spinal origin is crossed with respect to theassociated hemianesthesia, and provided the disorder is due to a single lesion, this®nding is pathognomonic of a spinal cord disorder [Rondt, 1969]. The classicalsyndrome consists of hemiparesis (or monoparesis of the leg) ipsilateral to the lesion(pyramidal tract disorder), contralateral loss of super®cial sensation (spinothalamic

ß 2001 Wiley-Liss, Inc.

*Correspondence to: Ryuji Sakakibara, M.D., Neurology Department Chiba University, 1-8-1 InohanaChuo-ku, Chiba 260-8670, Japan. E-mail: [email protected]

Received 14 May 2000; Accepted 15 October 2000

Page 2: Urinary dysfunction in Brown-Séquard syndrome

tract disorder), and ipsilateral loss of deep sensation (dorsal tract disorder) [Dejong,1979]. Depending on the position of the lesion in the transverse plane, however, motoror sensory disturbance will predominate [Koehler and Endtz, 1986]. In addition thereare several other less characteristic and more variable signs which are neverthelessimportant, including radicular type of segmental sensory or motor disturbance (rootdisorder) and paresthesia ipsilateral to the lesion (presumable dorsal tract disorder).

Autonomic ®bres are also components of the spinal tracts. When the sympatheticpathways are interrupted the pilomotor re¯ex is abolished on the side of the lesion andskin temperature is elevated [Rondt, 1969]. However, little is known of micturitionalfunction and no urodynamic data is available to our knowledge, although some patientswith BS are troubled with severe voiding dif®culty and urinary incontinence. We heredescribe results of micturitional questionnaire and urodynamic studies in eight patientswith BS.

MATERIALS AND METHODS

We recruited eight patients with BS, including seven men and one woman, meanage of 41 years ranging 18±60 years (Figure 1). All patients met the clinical criteria ofBS [Rondt, 1969; Dejong, 1979]. The underlying disorders in the patients includedspondylosis and vertebral disc herniation in four, hematomyelia in two, herpes zostermyelitis in one and vertebral anomaly in one, and the duration of diseases was 2 days±12 years. All patients had solely unilateral motor weakness with ipsilateral hyper-re¯exia of the extremities, which was mild (grade 4 or 5 in manual muscle test; walkingwith aid) in four and severe (grades 0±3; wheel chair-bound) in four. However, sensorymanifestations varied in each patient including decreased pain/cold sensation of thecontralateral side in ®ve (mild in one and severe in four), paraesthesia of the ipsilateralside in three and of the contralateral side in one, and decreased position/vibrationsensation of the ipsilateral side in ®ve.

We performed micturitional questionnaire in all patients on admission to ourhospital. It comprised ®lling phase symptoms; diurnal (more than eight times) andnocturnal (more than twice) urinary frequency, urinary urgency and urge urinaryincontinence, and voiding symptoms; voiding dif®culty (urinary hesitation, prolonga-tion, straining, and sensation of residual urine) and urinary retention. Before startingtherapies as described below (within 2 weeks after admission) we performed urody-namic studies in all patients with informed consent. During voluntary voiding wemeasured uro¯owmetry, and maximum and average ¯ow rates were obtained by astandard nomogram [Siroky et al., 1980]. After voiding we measured post-micturitionresiduals and their normal volume is under 30 mL. After inserting a 12 F side-holecatheter into the urethra, we measured urethral pressure pro®lometry by pulling out thecatheter at a rate of 1 cm/min along with infusing sterile water at a rate of 2 mL/min.Maximum urethral closure pressure (UPmax) of more than 41 cm H2O and less than82 cm H2O is considered as normal. After inserting an 8 F transurethral catheter and arectal catheter, medium-®ll (50 mL/min) water cystometry with simultaneous sphincterelectromyography (EMG cystometry) was done with an electromyographic computer(Nihon Kohden; Neuropack S) and a urodynamic computer (Lifetech; Janus). Detrusorhyperre¯exia is de®ned as a phasic contraction during ®lling with the detrusor pressurerise over 10 cm H2O. Detrusor are¯exia is de®ned when the patient is unable to contractthe detrusor on voiding. Unrelaxing sphincter is de®ned when the patient is unable to

662 Sakakibara et al.

Page 3: Urinary dysfunction in Brown-Séquard syndrome

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relax the sphincter on voiding. None of the men had apparent prostatic hypertrophy bydigital examination or ultrasound echography. The methods and de®nitions used for theurodynamic studies were described in detail previously [Sakakibara et al., 2000] andconformed to the standards proposed by the International Continence Society [Abramset al., 1989].

RESULTS

Detailed questionnaire revealed that ®ve of the eight patients (63%) hadmicturitional symptoms (Fig. 1). The most common symptom was voiding dif®culty inthree, and two patients were in a state of urinary retention, followed by urinary fre-quency in two and urge urinary incontinence in one. These symptoms appeared anddeteriorate gradually along with other symptoms. Patients with intramedullary diseases(cases 1±3) (all of those with duration of disease less than 3 weeks) tended to have moresevere motor and micturitional disorders than those with extramedullary diseases (cases4±8) (all of those with duration of disease more than 2 months). Micturitional symptomswere more common in patients with severe motor paresis (100%) than in those with mildmotor paresis (40%) although there was no statistical signi®cance (Fig. 2), and urinaryretention was noted only in patients with severe motor paresis. There was no relationbetween micturitional disturbance with super®cial or deep sensory disturbance. Therewas no relation between laterality of the lesion with micturitional disturbance in thepatients.

Urodynamic abnormalities were noted in all ®ve patients with micturitionalsymptoms. Uro¯owmetry was performed in ®ve of eight patients and maximum andaverage ¯ow rates were decreased in two. Measurement of post-micturition residualswas performed in seven patients and four had residual urine with an average of 149 mL.Urethral pressure pro®lometry was performed in six patients and two had high UPmax.

Fig. 2. Relationship between micturitional symptom and neurological signs. Micturitional symptom wasmore common in patients with sever motor paresis (100%) than in those with mild motor paresis (40%)although there was no statistical signi®cance. There was no relation between micturitional disturbance withsuper®cial or deep sensory disturbance.

664 Sakakibara et al.

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EMG cystometry was performed in all patients, showing increased bladder volume at®rst sensation in one (case 1; 380 mL), detrusor hyperre¯exia during ®lling in four,detrusor are¯exia on voiding in three, and unrelaxing sphincter on voiding in fourpatients. Three asymptomatic patients showed normal urodynamic ®nding.

In the eight patients with BS, two patients with hematomyelia (cases 1,2)underwent steroid pulse therapy (1,000 mg/day of methylprednisolone for 3 succeedingdays), and during a state of urinary retention we performed clean, intermittent self-catheterization (CISC). Both patients showed an improvement in motor weakness andthey became able to urinate. One patient with herpes zoster myelitis (case 3) underwent1,500 mg/day of aciclovir for 2 weeks, and he showed an improvement in motor andsensory disturbances. After the treatment voiding dif®culty gradually improved. Hisurge urinary incontinence once exacerbated, but improved afterwards. One patient withvertebral anomaly (case 4) had a longstanding impairment by BS (12 years). Hereceived 0.3 mg/day of tamsulosin for 3 months, and showed an improvement of urinaryfrequency from 20 to 10 times a day, and his residual urine volume from 120 to 50 ml.Four patients with spondylosis and disc herniation (cases 5±8) underwent cervicaltraction or surgery, which improved voiding dif®culty along with motor and sensorydisturbances.

DISCUSSION

Little is known of micturitional function and no urodynamic data is available toour knowledge in BS, although urinary dysfunction is found in two of 12 patients withBS (`bladder paralysis' and urinary hesitation) in the original description [Brown-SeÂquard, 1868]. However, the present study showed that ®ve of eight patients (63%) hadmicturitional symptoms. These symptoms appeared and deteriorate gradually alongwith other symptoms and no apparent causes such as prostate hypertrophy were noted.As a result, we could attribute these symptoms to BS. The most common symptom wasvoiding dif®culty in three, and two patients were in a state of urinary retention, followedby urinary frequency in two and urge urinary incontinence in one. It is likely thatboth ®lling and voiding disorders may occur in patients with BS, although the latterpredominates as in the other spinal cord disorders [Kaplan et al., 1991; Wyndaele,1997].

Urodynamic studies revealed that four of the ®ve symptomatic patients (80%; twoof those with urinary urgency / frequency) had detrusor hyperre¯exia during ®lling, andthree patients (60%; all of those with urinary retention / voiding dif®culty) had detrusorare¯exia on voiding. The ®ndings indicate detrusor hyperre¯exia and detrusor are¯exiato be major urodynamic abnormalities in BS. Detrusor hyperre¯exia and detrusorare¯exia could be considered as supranuclear type of pelvic nerve dysfunction,probably re¯ecting a lesion of the descending pathway to the sacral intermediolateralcell column [Blaivas, 1982; de Groat et al., 1993]. External sphincter EMG revealed anunrelaxing sphincter in four patients (all of those with urinary retention / voidingdif®culty). Unrelaxing sphincter or detrusor-sphincter dyssynergia could be consideredas supranuclear type of pudendal nerve dysfunction, probably re¯ecting a lesion of thepathway to the sacral Onuf's nucleus [Blaivas, 1982; de Groat et al., 1993]. Maximumurethral closure pressure was increased in two (50%; both with voiding dif®culty) offour symptomatic patients studied. This may indicate a supranuclear type of hypogastricnerve dysfunction innervating the internal urethral sphincter, although increased

Urinary Dysfunction in Brown-SeÂquard Syndrome 665

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pressure in a resting state should not directly re¯ect that in a voiding state. The presentstudy also showed an increased bladder volume at ®rst sensation, indicative ofdecreased bladder sensation [Blaivas, 1982; Wyndaele, 1997].

The present study showed that the intramedullary diseases tended to cause moresevere motor and micturitional disorders than extramedullary diseases. The presentstudy also showed that urinary dysfunction was more common in patients with severemotor paresis (100%) than in those with mild motor paresis (40%), and urinary retentionwas noted only in patients with severe motor paresis. There was no relation betweenmicturitional disturbance with super®cial or deep sensory disturbance in the presentstudy. The results are in accord to our previous study of 128 patients with compressivecervical myelopathy [Sakakibara et al., 1995]. It showed that detrusor hyperre¯exia anddetrusor-sphincter dyssynergia were more common in patients with pyramidal sign thanin those without, indicating descending pathway subserving micturition to be located inthe lateral column [Nathan and Smith, 1958; Inatomi et al., 1998]. Although the lowerurinary tract is bilaterally innervated in the periphery [Morrison, 1987], the results of thepresent study showed that spinal cord hemisection affects micturition function andcauses urinary retention, as known in patients with hemispheric brain disease [Khanet al., 1981; Sakakibara and Fowler, 1999].

In the eight patients with BS, we performed steroid pulse therapy in two patientswith hematomyelia (cases 1,2), antiviral agent (aciclovir) in a patient with herpes zostermyelitis (case 3), and cervical traction or surgery in four patients with spondylosis anddisc herniation (cases 5±8). All patients responded well to the treatment and the urinarydysfunction improved along with neurological dysfunction. It seems likely that urinaryand neurological dysfunction in BS improves better than that in transverse spinal cordlesion [Rondt, 1969; Dejong, 1979; Sakakibara et al., 1996]. During a state of urinaryretention we performed clean, intermittent self-catheterization in two patients toprevent overdistension injury of the bladder. Both patients with urinary retentionbecame able to urinate. One patient with vertebral anomaly (case 4) had a longstandingimpairment by BS (12 years). Therefore, we prescribed tamsulosin, an a-adrenergicblocking agent [Michel et al., 1998], which ameliorated his urinary frequency andvoiding dif®culty. An approach for the treatment of urinary dysfunction in BS patientsmay require a combination of therapies for the underlying disorders with a-adrenergicblocking agents [Michel et al., 1998; Sakakibara et al., 2000] and clean, intermittentself-catheterization for large residuals.

CONCLUSIONS

Micturitional disturbance was not uncommon in our patients with BS, althoughthe number of the patients was too small for generalization. Micturitional disturbancewas commonly noted in patients with severe motor paresis. An approach for thetreatment of urinary dysfunction in BS will require a combination of therapies for theunderlying disorders with a-adrenergic blocking agents and clean, intermittent self-catheterization for large residuals.

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