A 55-year-old male patient was admitted to the pain department of the First Affiliated Hospital of Harbin Medical University for intermittent fever. The maximum fever of 39.0 °C was accompanied by fatigue and waist pain for more than three months. He reported no nausea or vomiting.
Before he came to the First Affiliated Hospital of Harbin Medical University, the patient did not receive any drug but antipyretic analgesics to control the fever.
The patient raised sheep at his home; there had been an abortion and stillbirth of a lamb three months ago. The personal and family history were unremarkable. There was no history of upper respiratory tract infection or diarrhea.
The patient had long-term fever, fatigue, hyperhidrosis, and muscle and joint pain more than three months. He was admitted to the pain department. The doctor performed blood culture indicated an infection with gram-negative bacilli, and the tube agglutination test (SAT) titer was 1:400. He was diagnosed with brucellosis and given anti-brucella treatment. Four days later, the patient's fever gradually eased, but numbness and weakness began to appear on his legs. Symmetrical muscle weakness gradually appeared in the distal extremities, and this patient gradually became unable to move. He was then transferred to the department of infectious diseases.
Once the patient was admitted to our department, cerebrovascular disease, GBS, and neurobrucellosis were highly suspected. We performed a system physical examination including a neurological examination that showed mild paresthesia and muscle weakness. The muscle strength level of the arms was 0, the proximate muscle strength level of the legs was 0, and the distal muscle strength level was 2. A neuroelectrophysiological examination suggested that the limb F wave latency was extended and the nerve sensory conduction velocity of the legs was slowed down. Meningeal irritation signs and pyramidal signs were all negative.
An imaging examination, including magnetic resonance imaging (MRI), showed a degenerative cervical change. There were no infarction or hemorrhage observed in a computed tomography (CT) scan of the brain.
A laboratory examination was performed once the patient was admitted to our hospital. It showed mild liver damage and mild inflammatory activity: 2018-0-18 ALT:33.5 U/L, AST:41.7 U/L, ALB:33.3 g/L, GGT:63.7 U/L, WBC:7.92 × 109/L NEUT:7.01 × 109/L, NEUT%:88.4, MONO%:8, MONO:0.63 × 109/L, EO:0.00 × 109/L, EO%:0.00, CRP: 6.92 mg/L, ESR: PCT:0.11 ng/mL. The brucellosis agglutination test was retested, the titer of which was 1:1600. The IgM antibodies titer of rubella virus, cytomegalovirus, and herpes simplex virus were all negative.
Although anti-brucellosis drugs were given immediately, this patient's symptoms were not effectively controlled. Slight sensory disturbances, such as numbness and burning sensations, occurred at the end of the extremities. Muscle tone and tendon reflexes were absent in all limbs. Clinical manifestations of cranial nerve injury and autonomic nerve dysfunction included dysuria, sweating, dysphagia, ventilator paralysis, and neck weakness also gradually emerged.
Due to the high suspect of GBS and that the disease was progressing rapidly, intravenous immunoglobulin therapy (25 g/day for six days) combined with neurotrophic nutrition were applied to patient on the 6th day after he was transferred to the department of infectious diseases. On the 12th day, we performed a lumbar puncture and some related CSF examinations on the patient (Fig. 1). In addition, the anti-GM1 antibody IgG, IgM and anti-GD1b antibody IgG, IgM of blood, and CSF were all positive. Additionally, the SAT and bacterial culture of the CSF were also negative. These findings further confirmed the diagnosis of AMSAN and excluded other possibilities that can lead to neuropathy (Table 1).
With a diagnosis of brucellosis, the patient was given a course of antibiotic therapy (streptomycin (1 g/d i.m. 2w) + doxycycline (200 mg/d p.o. 12w) + trimethoprim-sulfa-ethoxazole (1.9 2 g/d p.o. 12w) + ceftriaxone sodium (2 g/d ivgtt 2w)). For the treatment of GBS, in addition to intravenous immunoglobulin and neurotrophic therapy, the patient was also given active rehabilitation (including muscle strength enhancement training, range of motion training, and acupuncture), and appropriate psychological counseling.
The patient's wife thought that the treatment of the patient was relatively timely, the patient's condition was not further aggravated, and the symptoms of urinary incontinence were gradually alleviated. Ten days later, the patient could take liquid food without choking and could perform some slight movements of the fingertips. After three months of systemic treatment, the patient's muscle strength of the distal limbs gradually recovered. The numbness of the limbs eased slowly, and urination function and respiratory function returned to normal. He was able to sit alone without assistance.