Authors
D. DOOREMONT (1) / [1] AZ Sint-Elisabeth, Zottegem, Belgium, Gastro-Enterologie
Case
When two rare conditions happen at the same time, it can be difficult to determine any relationship and see the connection between the two. In this case report we will give an example. A young female of 19 years old was admitted in our emergency room with symptoms of anorexia and malaise, two months after undergoing a mini gastric bypass. There were also complaints of blurry vision. As medication she took Pantomed 40 mg/d together with a multivitamin. She noted to recently have taken nitrofurantoin for an urinary tract infection. There was no use of nicotine or alcohol. Clinical investigation showed a weight of 100 kg, slightly icteric sclerae and a mild painful right hypochonder during palpation. The blood showed markedly elevated liver enzymes with AST 248 U/L, ALT 508 U/L, gammaGT 133 U/L and total bilirubin of 3,85 mg/dL. The PT was 49% at admission. The thyroid hormones were normal. In the autoimmune serology only ANF was elevated, the other autoimmune tests were negative. The ferritine and ceruloplasmin were normal. Viral serology was negative, only positive IgG for EBV. An ultrasound of the liver showed slightly increased density of the liver, indicating a mild steatosis. The bile ducts weren’t dilated. After given parenteral vitamin K the PT was normalized. A liver biopsy was performed after a couple of days because the liver enzymes didn’t decrease. Anatomopathology revealed presence of microvesicular steatosis around the vene centerolobularis, some Mallory bodies, some loss of hepatocytes and mild bilirubinostasis, most likely caused by a toxic drug reaction. The treatment with nitrofurantoin was already stopped, because of the very slow decline in liver enzymes a treatment with oral budesonide 9 mg a day was given, although IgG and smooth muscle antibodies were negative. Under this treatment the liver enzymes gradually decreased. From admission the neurological signs progressively worsened with confusion, hearing loss and paraparesis. An urgent advice of the ophthalmologist and neurologist was asked. The ophthalmologist saw bilateral acute mild papilledema. As differential diagnosis were benign intracranial hypertension, an intracranial bleeding or tumor, meningitis, Addison, hypoparathyroidy or hypovitaminosis A proposed. A CT scan of the brain was performed immediately, without showing any lesions in the brain. An urgent NMR of the brain didn’t show any pathological findings, although there were major artefacts caused by orthodontic material. Afterwards a lumbar puncture was performed which was normal. There were no arguments for benign intracranial hypertension or meningitis. The MMSE was 24/30 showing impaired cognition. The conclusion of our neurologists was that the neurological disease was caused by encephalopathy from the liver injury, even though the liver showed no progressive liver failure. The clinical state of the patient went progressively worse, she slept a lot, had confusion and even paraparesis. In an epiphany I came up with the diagnosis of thiamine deficiency. Treatment with parenteral thiamine was started promptly. The day after there was already obvious neurological improvement. The vitamin B1 level at that time was 25 nmol/L, confirming the diagnosis. In the literature I could not found any case of nitrofurantoin induced liver failure and thiamine deficiency at the same time in the same patient. As a result of the toxic liver reaction she ate almost nothing, thereby she didn’t take her vitamins. The combination of recent gastric bypass and anorexia led to malnutrition and vitamin B1 deficiency. In the first place is it very important to consider the diagnosis. Wernicke encephalopathy is a serious disorder and is classical described in chronic alcoholics, certainly here in the Western civilization. It has also been identified in malnutrition, prolonged fasting, gastrointestinal malignancies, dialysis, hyperemesis gravidarum, AIDS and bariatric surgery. If left untreated, it may lead to a state of chronic mental dysfunction known as Korsakoff’s syndrome and even death. Nitrofurantoin is a commonly prescribed medication in primary care, side effects include diarrhea, neuropathy, pulmonary fibrosis, interstitial pneumonitis and hypersensitivity reactions. Hepatotoxicity accounts for 12% of reported adverse reactions and 25% of those resulting in death. Significant hepatotoxicity is rare following short courses, like in our case. Immune-mediated liver injury usually occurs in the setting of long-term use. The prognosis is variable.
