Hemiparesis following sunbathing

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Case Reports

Angiography is the criterion standard diagnostic tool for aortic dissection. Recently, this has been supplanted by transesophageal echocardiography, computed tomography, and magnetic resonance imaging. Transesophageal echocardiography, in experienced hands, is a reliable, safe, and lowcost diagnostic tool. It can be performed at the bedside within minutes, reducing potential mortality [4]. Bedside-specific biomarkers are not in clinical use. Some studies have examined concentrations of soluble elastic compounds or smooth muscle myosin heavy chain [5] in early hours of aortic dissection. d-dimers have also been evaluated as early serum markers. This case emphasizes the need for high index of suspicion for dissection in patients with chest pain. Absence of classical widened mediastinum on chest roentogram and hypertension does not exclude the diagnosis. Aortic dissection may be a differential in patients presenting with chest pain and atrial fibrillation. Huck Chin Chew MD Swee Han Lim MD Department of Emergency Medicine Singapore General Hospital, 169608, Singapore E-mail address: [email protected] doi:10.1016/j.ajem.2005.11.001

later, unresponsive in her chair, her head bent to the left with large blisters covering her right arm and neck. Physical examination disclosed oligoanuria and second-degree sunburn blisters on the right side of the neck, right forearm, and both legs below the knees. There were no signs of head trauma or fall. The patient had a history of hypertension, congestive heart failure, and hypercholesterolemia, for which she takes lisinopril, atorvastatin, furosemide, potassium chloride, and medroxyprogesterone. An initial comprehensive laboratory evaluation revealed leukocytosis, acute renal failure, and rhabdomyolysis with elevated serum ethanol level and liver enzymes (Table 1). An electrocardiogram indicated sinus tachycardia without evidence of myocardial ischemia. Serum toxicological screening for overdosage of salicylate and acetaminophen was negative. Urine analysis was negative for recreational drugs, but consistent with myoglobinuria. Findings on emergency cervical spine and chest radiography, head computed tomography (CT), and carotid ultrasonography were normal. Brain magnetic resonance imaging (MRI) Table 1

Laboratory tests results of the patient at presentation

Laboratory tests

Results

Normal

Hematologic counts and coagulation White blood cells (109/L) Platelets (109/L) Hemoglobin (g/dL) Prothrombin time (s) Partial thromboplastin time

14.6 124 11.5 12.0 23.2

4.0-10.0 150-350 12.0-16.0 b13.0 22.0-35.0

Serum chemistry Sodium (mmol/L) Potassium (mmol/L) Chloride (mmol/L) Carbon dioxide (mmol/L) Anion gap (mmol/L) Urea nitrogen (mg/dL) Creatinine (mg/dL) Serum ethanol (mmol/L)

141 6.7 113 10.5 18 72 3.1 52.2

135-145 3.5-5.0 96-106 22-30 4 -10 8-18 0.5-1.2 b1

Liver parameters Serum alkaline phosphatase (U/L) Serum aspartate aminotransferase (U/L) Serum alanine aminotransferase U/L Serum total bilirubin (lmol/L)

58 2590 1350 5.1

30-130 0-35 0-35 0-20.5

Muscle enzymes Creatine phosphokinase (U/L) Creatine phosphokinase–MB (% ng/mL) Troponin (ng/mL)

3760 40.0 b0.3

24-170 b5.0 b0.3

395 4.1

40-65 b1.5

References [1] Slater EE, DeSanctis RW. The clinical recognition of dissecting aortic aneurysm. Am J Med 1976;60(5):625 - 33. [2] Fradet G, Jamieson WR, Janusz MT, Munro AI, Ling H, Miyagishun RT, et al. Aortic dissection : a 6 year experience with 117 patients. Am J Surg 1988;155(5):697 - 700. [3] Torossor M, Singh A, Fein SA. Clinical presentation, diagnosis and hospital outcome of patients with documented aortic dissection : the Albany Medical Centre experience, 1986 to 1996. Am J Heart 1999;137(1):154 - 61. [4] Nienaber CA, Eagle KA. Aortic dissection: new frontiers in diagnosis and management. Circulation 2003;108(5):628 - 35. [5] Suzuki T, Katoh H, Watanabe M, et al. Novel biochemical diagnostic method for aortic dissection, results of a prospective study using an immunoassay of smooth muscle myosin heavy chain. Circulation 1996;93:1244 - 9.

Hemiparesis following sunbathing In September 2000, a 56-year-old, right-handed, white woman was transferred to the emergency center of our facility in a state of hemodynamic shock (blood pressure = 64/40 mm Hg, heart rate = 120 per minute, and respiratory rate = 40 per minute). Earlier that morning, she was in her usual state of health, without previous neurological deficit or complaints, when she went outside on her patio sunbathing and drinking a bottle of white wine. She was found 5 hours

Pancreatic enzymes Amylase (U/L) Lipase (U/mL) Arterial blood gas analysis pH Pco2 (mm Hg) Po2 (mm Hg) O2 saturation (%)

7.10 28 77 90

7.48-7.52 35- 45 75-100 92-99

Case Reports showed multiple lacunar infarcts, but the diffusion-weighted images ruled out any recent infarct. After fluid resuscitation, the patient recovered consciousness but complained of with new onset of right arm and leg weakness. On examination, the patient had intact cranial nerves except mild right blepharoptosis and miosis. She had diminished grip strength, 3/5; biceps, 4/5; triceps, 4/5; foot dorsiflexion, 3/5; and knee extension, 4/5. Fine touch and pinprick sensory were diminished in right C7 through T1 distribution and right leg, with loss of proprioceptive and vibratory sensation in the right leg, and loss of thermal and nociceptive sensation in the left leg and trunk below the level of the left nipple. She had an absence of abdominal reflexes on the left side, equivocal cutaneoplantar reflexes bilaterally, and a moderate right triceps jerk with increased deep tendon reflexes (3/4) in lower extremities. Bowel/ bladder functions were intact, and the remainder of her examination was essentially normal. These findings were suggestive of Brown-Se´quard syndrome without central cord syndrome. We first considered internal capsule stroke because of her risk factors (acute alcohol intoxication, congestive heart failure, hypercholesterolemia, and hypertension) and compartment syndrome with second-degree sunburn involving the edematous paretic limbs. However, right-sided parasagittal T1-weighted MRI of the cervical spine showed marked congenital spinal stenosis and an intramedullary contusion lesion with right spinal cord hemisection at T1 (Fig. 1, circled). CT scan of the cervical spine showed anterior and posterior osteophyte complexes at the levels of C6, C7, and T1, with bilateral narrowing of the neural foramen (Fig. 2, circled). The spinal CT and MRI findings suggested that the Brown-Se´quard syndrome was caused by the osteophyte compressing the spinal cord as a result of prolonged anterolateral neck flexion secondary to alcohol-induced stupor and muscle relaxation. Alcohol ingestion and statin medications are believed to have caused the patient’s rhabdomyolysis [1] and secondary renal failure. Brown-Se´quard syndrome is characterized by the interruption of the lateral corticospinal and spinal thalamic tracts, with occasional injury to the posterior columns. It has diverse etiologies such as penetrating cervical injury [2], malignant extramedullary or intramedullary spinal cord neoplasm or metastasis [3], idiopathic spinal cord herniation [4], cervical disc herniation [5], cervical spondylosis [6], vasculitis, and spontaneous [7] or traumatic [8,9] epidural hematoma. Horner’s syndrome (blepharoptosis, enophthalmos, miosis, and hemifacial anhidrosis) results from any interruption along the sympathetic innervation to the eye and face. This can occur at the posterolateral hypothalamus, the midbrain, pons, spinal cord, or the cervical sympathetic chain, which branches off to innervate the facial vessels and sweat glands (by accompanying the branches of the external carotid artery) and the eye through the long ciliary nerves (by accompanying the internal carotid artery).

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Fig. 1 Parasagittal T1-weighted magnetic resonance image of the cervicothoracic spine demonstrates a congenital spinal stenosis with acute intramedullary spinal cord lesion at the level of T1 (circled).

Injuries to the intermediolateral cell column of the spinal cord at the level of C8-T2, such as neck trauma, spinal ischemia, or syringomyelia can cause a Horner’s syndrome. Its combination with Brown-Se´quard syndrome, although very rare, can occur when the injury affects the cervicothoracic spinal cord. Why did our patient have a lateral cord injury in flexion? We offer several possibilities. First, the prolonged hypotension the patient experienced could have contributed to the development of the lesion, but would unlikely be the sole reason. Second, we could speculate that a sustained hypotension could theoretically extend a previous lacunar infarction of the spine. Most interestingly, we hypothesize that muscle relaxation, secondary to alcohol intoxication and excessive heat allowed extreme flexion of the cervical spine. Prolonged compression of the spinal cord against the vertebral osteophyte in a critically narrowed spinal canal resulted in a cord contusion below the osteophyte at C7-T1. It is unclear why this occurred at T1 and not at C5-C6, where there is an osteophyte of larger size, but one can hypothesize that the point of flexion of the cervical spine in flexion occurs below C5-C6 level. The prolonged hyper-

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Case Reports Fadi Braiteh MD Phase I Program Division of Cancer Medicine The University of Texas M. D. Anderson Cancer Center Houston, TX 77030-4009, USA James Abrahams MD Department of Neuro-Radiology Yale–New Haven Hospital Yale University School of Medicine New Haven, CT 06520, USA doi:10.1016/j.ajem.2005.11.008

References

Fig. 2 Sagittal CT scan of the cervical spine reveals multiple anterior and posterior osteophyte complexes at the levels of C6, C7, and T1 (circled).

flexion is supported by our findings and by the sunburn on the right side of the neck and both legs. Furosemide, atorvastatin, and medroxyprogesterone are photosensitizers that could have amplified the sunburn [10]. The pressure contusion resulted in a hemorrhagic lesion, as visualized on the MRI, which caused the hemisection of the spinal cord in the right cervical spine region at the C7-T1 level. Although Horner’s syndrome can hypothetically be caused by a T1 root injury or avulsion, we attribute it in this patient to injury of the right intermediolateral cell column at the same level. The combination of Horner’s syndrome and Brown-Se´quard syndrome is very rare but has been reported in a few cases of cervical trauma [9,11-13]. Spinal cord injury without radiographic evidence of an abnormality is reported most frequently in children [14] but has been reported in adults after dynamic, deceleration, or blunt trauma accidents [15-18]. The patient was offered decompressive laminectomy but agreed only to medical treatment with glucocorticoids, burns wound care, and physical therapy. She had rapid recovery of her motor and sensory functions over the following month. We offer this case as the first report illustrating serious morbidity from a benign condition: vertebral osteophyte in osteodegenerative disease of the spine. In medical literature, there has been one report of a case of isolated nontraumatic Horner’s syndrome caused by thoracic vertebral osteophyte [19]. The case we report here highlights the possibility of the rare combination of BrownSe´quard and Horner’s syndromes secondary to an osteophyte-induced pressure contusion to the cervical cord.

[1] Sauret JM, Marinides G, Wang GK. Rhabdomyolysis. Am Fam Physician 2002;65:907 - 12. [2] Firlik AD, Welch WC. Images in clinical medicine. Brown-Se´quard syndrome. N Engl J Med 1999;340:285. [3] Aryan HE, Farin A, Nakaji P, Imbesi SG, Abshire BB. Intramedullary spinal cord metastasis of lung adenocarcinoma presenting as BrownSe´quard syndrome. Surg Neurol 2004;61:72 - 6. [4] Massicotte EM, Montanera W, Ross Fleming JF, Tucker WS, Willinsky K, TerBrugge K, et al. Idiopathic spinal cord herniation: report of eight cases and review of the literature. Spine 2002;27:E233-41. [5] Mastronardi L, Ruggeri A. Cervical disc herniation producing BrownSe´quard syndrome: case report. Spine 2004;29:E28-31. [6] Fujimoto A, Matsumura A, Maruno T, Yasuda S, Nose T. Cervical spondylosis with thoracic level Brown-Se´quard syndrome. J Clin Neurosci 2004;11:898 - 900. [7] Gill D, Ifthikharuddin S, Samkoff LM. Acute Brown-Se´quard syndrome. Arch Neurol 2004;61:131. [8] Stoll A, Sanchez M. Epidural hematoma after epidural block: implications for its use in pain management. Surg Neurol 2002;57:235 - 40. [9] Shen CC, Wang YC, Yang DY, Wang FH, Shen BB. Brown-Se´quard syndrome associated with Horner’s syndrome in cervical epidural hematoma. Spine 1995;20:244 - 7. [10] Moore DE. Drug-induced cutaneous photosensitivity: incidence, mechanism, prevention and management. Drug Saf 2002;25(5):345 - 72. [11] Koehler PJ, Endtz LJ. The Brown-Se´quard syndrome. True or false? Arch Neurol 1986;43:921 - 4. [12] Edwards A, Andrews R. A case of Brown-Se´quard syndrome with associated Horner’s syndrome after blunt injury to the cervical spine. Emerg Med J 2001;18:512 - 3. [13] Garcia-Manzanares MD, Belda-Sanchis JI, Giner-Pascual M, MiguelLeon I, Delgado-Calvo M, Alio y Sanz JL. Brown-Se´quard syndrome associated with Horner’s syndrome after a penetrating trauma at the cervicomedullary junction. Spinal Cord 2000;38:705 - 7. [14] Pang D. Spinal cord injury without radiographic abnormality in children, 2 decades later. Neurosurgery 2004;55:1325 - 43. [15] Samsani SR, Calthorpe D, Geutjens G. Thoracic spinal cord injury without radiographic abnormality in a skeletally mature patient: a case report. Spine 2003;28:E78-80. [16] Gupta SK, Rajeev K, Khosla VK, Sharma BS, Paramjit BS, Mathuriya SN, et al. Spinal cord injury without radiographic abnormality in adults. Spinal Cord 1999;37:726 - 9. [17] Kothari P, Freeman B, Grevitt M, Kerslake R. Injury to the spinal cord without radiological abnormality (SCIWORA) in adults. J Bone Joint Surg Br 2000;82:1034 - 7. [18] Crawford S, Bleetman T. Adult spinal cord injury without radiological abnormality. J Accid Emerg Med 2000;17:374 - 5. [19] Bernad PG, Perlo VP. Horner syndrome with causalgia. Neurology 1980;30:534 - 5.