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Journal Articles Journal of Clinical Microbiology Year : 2015

Severe Ketoalkalosis as Initial Presentation of Imported Human Rabies in France


We report a patient with an unusual initial metabolic presentation of imported human rabies who became symptomatic within 2 weeks of returning from Mali to France. This is the single case of imported human rabies identified in France within the past 11 years and the first report of viral RNA in bronchial secretions. CASE REPORT A 57-year-old male without past medical history presented to an emergency room in March 2014 because of fever and generalized pain. He had been a resident of France for the previous 15 years and had returned 2 weeks earlier following a 6-month stay in Mali. Approximately 1 month before his return to France, he underwent a right foot injury from a tree branch which eventually healed with local care. On admission, he was conscious and cooperative but appeared anxious; he had fever (38.2°C) and abundant sweating and complained of generalized pain, mostly in the lower limbs. A clean, noninflammatory wound scar was noted on the right foot. He had marked tachypnea (rate, 40 inspirations/minute) and periodic deeper inspirations. Chest examination results were unremark-able. Arterial blood gas measurements while the patient was breathing room air were as follows: pH 7.79; partial pressure of carbon dioxide (PaCO 2), 11 mm Hg; PaO 2 , 136 mm Hg; bicar-bonates, 16 mmol/liter; and lactate, 3.7 mmol/liter. The capillary blood glucose was measured at 12 mmol/liter, and a urine dipstick revealed glycosuria (3) and ketonuria (3). Serum creatinine was at 94 mol/liter and sodium at 136 mmol/liter. Thick and thin blood smears were negative for malaria. Chest X-ray results were normal. The patient was transferred to our intensive care unit (ICU) with a presumptive diagnosis of uncontrolled diabetes and unexplained severe respiratory alkalosis. He received intravenous fluids, continuous insulin infusion, and tetanus prevention with vaccination and serotherapy. Hyperventilation, ketonuria, and metabolic abnormalities resolved within 2 days, with normaliza-tion of pH and PaCO 2. Glycated hemoglobin was 6.5%, and the patient required no further insulin administration during his ICU stay. Two days after ICU admission, he developed bouts of hyper-activity, disorientation, and delirium with thoughts of impending death associated with persecution ideas, alternating with periods of drowsiness and returns to normal behavior when he seemed aware of his disorder and criticized it. Hypersalivation was remarkable , and the patient occasionally spat on ICU personnel. Motor weakness, deep tendon reflexes, and limb sensory perceptions were normal. The results of a computed tomography scan and magnetic resonance imaging (MRI) of the brain were unre-markable. Electroencephalogram results showed no epileptic activity. The cerebrospinal fluid (CSF) test result was normal, with a negative PCR test for herpes simplex virus 1 (HSV-1) and HSV-2. Serological tests for human immunodeficiency virus type 1 (HIV-1) and HIV-2 and HIV-1 p24 antigen were negative. Thyroid stimulating hormone and ammonia levels were in the normal range. Syphilis serologic tests, including Venereal Disease Research Laboratory (VDRL) and Treponema Pallidum Hem Agglutination (TPHA) tests, were negative. On day 8 of ICU admission, the patient developed a rapidly extensive flaccid and areflectic tetraparesis without involvement of cranial nerves. His condition deteriorated, with altered consciousness and hypercapnic acidosis, and the patient required mechanical ventilation on day 9. Results of a repeated CSF analysis were normal. Results of a second cerebral and cervical MRI test were normal. Electroneuromyography showed an acute motor ax-onal neuropathy of both upper and lower limbs without sensory impairment. The results of a search for antineuron antibodies were negative, and the urinary porphobilinogen level was normal. The patient received methylprednisolone (1 g/day from day 8 to day 10) and intravenous immunoglobulin (2 g/kg of body weight from day 8 to day 12) for suspected paraneoplastic limbic enceph-alitis or Guillain-Barré syndrome. A search for rabies was performed on a skin biopsy specimen and salivary swabs obtained on day 13. A profound coma with periodic inspiratory spasms persisted , and the patient died on day 19 after ICU admission.
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Dates and versions

pasteur-01430034 , version 1 (09-01-2017)



Damien Contou, Laurent Dacheux, Inès Bendib, Sarah Jolivet, Christophe Rodriguez, et al.. Severe Ketoalkalosis as Initial Presentation of Imported Human Rabies in France. Journal of Clinical Microbiology, 2015, 53 (6), pp.1979-1982. ⟨10.1128/JCM.00219-15⟩. ⟨pasteur-01430034⟩


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