J UOEH（産業医科大学雑誌）35（ 2 ）: 159－164（2013） 159

［Case Report］

Hypertensive Intracerebral Hemorrhage Due to Autonomic Dysreflexia in a Young Man with Cervical Cord Injury

Tadashi Sumiya

Department of Spinal Care Center, Division of Rehabilitation Medicine 219 Myoji, Katsuragi-cho, Ito-gun, 649-7113, Japan

Abstract : The author reports the case of a 36 year old man with cervical cord injury in whom autonomic dysreflexia developed into intracerebral hemorrhage during inpatient rehabilitation. This patient showed complete quadriplegia (motor below C6 and sensory below C7) due to fracture of the 6th cervical vertebra. An indwelling urethral catheter had been inserted into the bladder for 3 months, diminishing bladder expansiveness. Bladder capacity decreased to 200 ml and the patient frequently experienced headaches whenever his bladder was full. To obtain smoother urine flow, a supra-pubic cystostomy was performed. The headaches were temporarily cured, but soon relapsed with extreme increases in blood pressure, representing typical symptoms of autonomic dysreflexia. However, no poten- tial triggers were identified or removed, and lack of blood pressure management led to left putaminal hemorrhage. Despite operative treatment, the right upper extremity showed progressive increases in muscle tonus and finally formed a frozen shoulder with elbow flexion contracture. Two factors contributed to this serious complication: first, autonomic dysreflexia triggered by minor malfunction and/or irritation from the cystostomy catheter; and second, the medical staff lacked sufficient experience in and knowledge about the management of autonomic dysreflexia. It is of the utmost importance for medical staff engaging in rehabilitation of spinal patients to share information regard- ing triggers of autonomic dysreflexia and to be thorough in ensuring proper medical management.

Keywords : spinal cord injury, urethral catheterization, cystostomy, autonomic dysreflexia, intracerebral hemorrhage.

（Received January 18, 2013, accepted May 2, 2013）

Introduction of prevention is not always sufficiently recognized by medical staff engaging in the rehabilitation of spinal Autonomic dysreflexia, first described by Gutt- patients. This report describes the case of a young man mann and Whitteridge in 1947 [1], is a well-known with cervical cord injury in whom autonomic dysre- syndrome seen in individuals with spinal cord injury flexia developed into intracerebral hemorrhage during above the T4/5 neurological level. The most serious inpatient rehabilitation. complication is hypertensive intracerebral hemor- rhage. Sufficient knowledge and proper medical man- Case report agement are of the utmost importance for preventing this syndrome. Although a small number of reports A 36 year old male worker with no particular past have described hypertensive intracerebral hemorrhage history was involved in a labor accident on September due to autonomic dysreflexia [2-4], the importance 1, 2004. A 100 kg signboard struck him in the occipital

Corresponding author: Tadashi Sumiya, MD, Department of Spinal Care Center, Division of Rehabilitation Medicine, 219 Myoji, Katsuragi-cho, Ito-gun, 649-7113, Japan. Phone: +81-736-22-0066, Fax: +81-736-22-2579, E-mail: [email protected] 160 Tadashi Sumiya

region and caused hyperflexion of the neck, resulting tient quickly overcame orthostatic hypotension, and in immediate quadriplegia without loss of conscious- his blood pressure stabilized to remain at about 90/50 ness. In an emergency hospital, dislocation fracture of mmHg. The pressure ulcer took 3 months to heal with the 6th cervical vertebra was diagnosed and he under- conservative treatment. Spasticity in the trunk and went anterior-posterior cervical fusion on September lower extremity muscles gradually increased and oral 8, 2004. Postoperative magnetic resonance imaging of administration of dantrolene sodium (Dantrium) was the cervical spine showed abnormal signals inside the required to relieve accentuated muscle tonus. spinal cord from the C3 to C7 level, suggesting spinal Long-term use of the indwelling urethral catheter cord malacia [Fig. 1]. diminished bladder expansiveness, which showed a The patient remained quadriplegic 3 months postop- decrease in capacity to 200 ml. The patient frequent- eratively without any neurological recovery, and was ly reported headache whenever his bladder was full, admitted to a rehabilitation hospital on November 24, which was interpreted as a symptom of autonomic 2004. On admission, his neurological level was mo- dysreflexia. For better urinary management, a supra- tor C6 complete and sensory C7 complete bilaterally. pubic cystostomy was performed in March 2005, but The upper extremity showed almost normal muscle this treatment did not immediately cure the frequent tonus, with normal muscle strength in the deltoid and headaches. To make urine flow as smoothly as pos- biceps brachii, good strength in the wrist extensors, sible and avoid irritation of the bladder wall by the poor strength in the triceps, and no strength in the catheter tip, a cystostomy catheter had to be inserted wrist flexors and all finger muscles. Lower extrem- into the bladder to the optimum depth, and occasional- ity muscles were all very spastic without any volun- ly the cystostomy catheter needed to be exchanged for tary movements. An indwelling urethral catheter had a urethral catheter in the event of sustained headache. been placed since emergency hospital admission and In July, the patient finally attained stable urine flow by a pressure ulcer was present in the sacral area. The cystostomy after several attempts and achieved relief patient was dependent on nursing staff for all activities from frequent headaches. After 8 months of hospital- of daily living except feeding. ization, he became an independent manual wheelchair At the beginning of wheelchair training, the pa- user, and discharge in early September with home-visit nursing care was scheduled. On the afternoon of Saturday, August 14, after ram- bling about by wheelchair, he developed headache with a cold sweat. At this time, his blood pressure reached 211/128 mmHg with a regular heart rate of 67 beats/min. The cystostomy catheter was inspected, but no obvious occlusion was apparent. An on-duty doctor could not make a clear diagnosis about these symptoms. To obtain quick relief from the headache, a loxoprofen sodium tablet (Loxonin) was adminis- tered, but had no effect in relieving the headache and hypertension. About an hour later, the patient began salivating from the right corner of the mouth, became inarticulate, and unable to move the right upper ex- tremity at all, and finally entered a semi-comatose state. Computed tomography (CT) of the head re- vealed left putaminal hemorrhage perforating to the Fig. 1. Sagittal T2-weighted MR image of the cervi- 4th ventricle [Fig. 2]. An intravenous drip of nicar- cal spine (taken on October 15, 2004). High intensity sig- dipine hydrochloride (Perdipine) was started at 40 μg/ nals are seen inside the spinal cord from C5 to C7 level. min, and the patient was transferred to an emergency Intracerebral Hemorrhage Due to Autonomic Dysreflexia 161

Fig. 2. CT scan of the head (taken on August 14, 2005). Fig. 3. CT scan of the head (taken on October 11, 2006). Left putaminal hemorrhage perforating to the 4th ventricle is Low-density area from left putamen to corona radiata is to be seen. noted. hospital, where he underwent evacuation of hematoma showed progressive loss of functional movements and by craniotomy. The following day, his consciousness finally formed a frozen shoulder with elbow flexion became clear and physiotherapy was started while he contracture. remained confined to bed. He returned to the former Stroke sequelae in addition to cervical cord injury rehabilitation hospital on October 11, 2005. made rehabilitation very difficult and contributed to The right upper extremity showed partial motor re- the development of a depressive state. For a change covery over the course of 3 months after stroke onset. of pace, he tried staying at home one night during the Using synergic movements, he could flex, abduct, and 2006 New Year holiday, which was very effective for adduct the shoulder, and flex the elbow, but could not improving both his mental and physical status. About extend the elbow or move the wrist at all. He also 4 months after stroke, the patient was partly able to showed right facial paralysis, amnestic aphasia, and feed himself using the left upper extremity, but was slight memory disturbance. He was absolutely de- still unable to control a manual wheelchair unaided. pendent on the nursing staff for activities of daily liv- After arranging for home visit nursing care, the patient ing, including feeding. A CT of the head after stroke was discharged home on March 5, 2006. showed a low-density area from the left putamen to the corona radiate [Fig. 3]. Discussion The author expected the right upper extremity to regain some degree of function, but the muscles of Symptoms of autonomic dysreflexia vary widely shoulder adduction and elbow flexion showed grad- from some feelings of slight involuntary muscle con- ual increases in tonus. Passive flexion/abduction of tractions (crispation) to severe hypertension accompa- the shoulder or passive extension of the elbow con- nied by headache. These symptoms are often a casual sequently induced severe pain, progressively limiting sign of bladder filling, but sometimes develop into a the ranges of joint motion. Motor points of the biceps serious complication. It has been observed that the brachii muscle were repeatedly blocked with 5% phe- higher the injury level, the greater the degree of clini- nol to reduce spasticity, but results proved only briefly cally manifest cardiovascular dysfunction [5, 6]. The effective. The patient was unable to accept muscle completeness of the spinal injury also relates to the stretching either by a physiotherapist or by arm splint- severity of autonomic dysreflexia: only 27% of pa- ing, due to intolerable pain. The right upper extremity tients with incomplete quadriplegia present with signs 162 Tadashi Sumiya

of autonomic dysreflexia, in comparison with 91% of Conclusion patients with quadriplegia with complete lesions [5]. While autonomic dysreflexia occurs more often in the This report presents the case of a young man with chronic stage of spinal cord injury at or above the sixth cervical cord injury who developed hypertensive in- thoracic segment, there is also clinical evidence of epi- tracerebral hemorrhage due to autonomic dysreflexia. sodes of autonomic dysreflexia in the first days and Cystostomy catheter dysfunction probably acted as a weeks after injury [7]. trigger. This serious complication resulted in severe Medical management of autonomic dysreflexia is contracture of the right upper extremity, adding to a well-known issue among medical staff specializ- quadriplegia. Medical staff engaging in the rehabili- ing in patients with cervical cord injury, but may be tation of patients with cervical cord injury should be less familiar to doctors in other specialties not closely thorough in identifying triggers of autonomic dysre- acquainted with characteristic symptoms and proper flexia and providing emergent medical management. treatment of autonomic dysreflexia. Nevertheless, any doctor may have to manage autonomic dysreflexia in References the course of providing primary care for a patient. Due to the potentially serious complications of autonomic 1 . Guttmann L & Whitteridge D (1947): Effects of blad- dysreflexia, medical staff, including doctors, require der distension on autonomic mechanisms after spinal education about autonomic dysreflexia [8]. cord injuries. Brain 70: 361-404 When patients with cervical cord injury complain 2 . Hanowell LH & Wilmot C (1988): Spinal cord injury of headache with hypertension, autonomic dysreflexia leading to intracranial hemorrhage. Crit Care Med 16: should be the first condition suspected. An exhaus- 911-912 tive search for possible triggers must be conducted and 3 . Eltorai I, Kim R, Vulpe M, Kasravi H & Ho W (1992): those identified must be quickly eliminated, or blood Fatal cerebral hemorrhage due to autonomic dysre- pressure should be immediately controlled with anti- flexia in a tetraplegic patient: case report and review. hypertensives [9]. Paraplegia 30: 355-360 In this case, distention of the bladder wall due to 4 . Pan SL, Wang YH, Lin HL, Chang CW, Wu TY & insufficient urine flow and/or irritation of the blad- Hsieh ET (2005): Intracerebral hemorrhage secondary der wall by the tip of the cystostomy catheter were to autonomic dysreflexia in a young person with in- considered strong candidates as potential triggers. A complete C8 tetraplegia: a case report. Arch Phys Med quick change to a urethral catheter should have been Rehabil 86: 591-593 attempted, and if no amelioration had been obtained, 5 . Curt A, Nitsche B, Rodic B, Schurch B & Dietz V an antihypertensive should have been administrated (1997): Assessment of autonomic dysreflexia in pa- immediately. However, it must be very difficult for an tients with spinal cord injury. J Neurol Neurosurg Psy- on-duty doctor to decide on changing a catheter from chiatry 62: 473-477 cystostomy to urethral while urine flow is being main- 6 . Krassioukov A, Warburton DE, Teasell R & Eng JJ tained without apparent occlusion. Moreover, blood (2009): A systematic review of the management of pressure should have been repeatedly monitored to al- autonomic dysreflexia after spinal cord injury. Arch low active decisions on the use of antihypertensives Phys Med Rehabil 90: 682-695 without delay. Information on specific triggers of au- 7 . Silver JR (2000): Early autonomic dysreflexia. Spinal tonomic dysreflexia and proper medical management Cord 38: 229-233 should thus be shared by all medical staff, to ensure 8 . Jackson CR & Acland R (2011): Knowledge of au- that adequate emergent medical care is provided. For tonomic dysreflexia in the emergency department. this purpose, a complete clinical practice guideline Emerg Med J 28: 866-869 should be available on a website [10]. 9 . Erickson RP (1980): Autonomic hyperreflexia: patho- physiology and medical management. Arch Phys Med Rehabil 61: 431-440 Intracerebral Hemorrhage Due to Autonomic Dysreflexia 163

10 . Consortium for Spinal Cord Medicine (2001): Acute 2nd ed. Paralysed Veterans of America Washington DC, management of autonomic dysreflexia; Individual with 40 pp spinal cord injury presenting to health-care facilities.

164 Tadashi Sumiya

頸髄損傷の若年男性に併発した自律神経過反射による高血圧性脳内出血

隅谷 政

和歌山県立医科大学附属病院 紀北分院 リハビリテーション科

要 旨：リハビリテーションのため入院中に自律神経過反射による脳内出血を併発した 36歳男性頸髄損傷者 の1 症例を報告する．本患者は第6頸椎骨折による完全四肢麻痺（第 6頸髄節以下で運動麻痺，第 7頸髄節以下で感 覚麻痺）であった．尿道カテーテルが3ヶ月間膀胱内に留置されていたため膀胱拡張能は低下していた．膀胱容量 は200 mlに減少しており，膀胱充満時にはつねに頭痛を訴えていた．より円滑な尿流を得るために膀胱瘻が造設さ れた．頭痛は一時的に治まったが程なく再発し，血圧の極端な上昇を伴って典型的な自律神経過反射の症状を呈し ていた．しかし，潜在的な引き金が見つからず除去できなかったことと，血圧管理ができていなかったことで，左被 殻出血を併発した．手術療法が行われたにも関わらず，右上肢の筋緊張は進行的に増大し，最終的には肘屈曲拘縮を 伴った凍結肩を呈するに至った．この重大な合併症をもたらした誘因は2つあげられる；第1に，膀胱瘻カテーテル のわずかな機能不全や刺激が自律神経過反射をもたらしたこと；第2に，医療スタッフが自律神経過反射に関する 十分な経験と知識を持ち合わせていなかったことである． 頸髄損傷患者のリハビリテーションに従事する医療ス タッフにとって，自律神経過反射の引き金に関する情報を共有することと，正しい医学的管理を確実に行えるように 徹底させることは極めて重要である．

キーワード：脊髄損傷，尿道カテーテル，膀胱瘻，自律神経過反射，脳内出血．

J UOEH（産業医大誌）35（2）：159－164（2013）