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Anesthetic management of a patient with known Dubin Johnson Syndrome — a case report. Correspondence: Ajinu Achi John; E-mail : ajohn39 hamad. The patient underwent laparoscopic appendicectomy under general anesthesia. Aside from elevated serum bilirubin levels, other routine laboratory tests were normal. As it is a rare and uncommon disease, the anesthetic management was considered a challenge. Liver being the organ affected, the drugs, which increases bilirubin, were to be avoided. Selection of drugs to provide analgesia, anesthesia and to reduce perioperative stress was a priority and to prevent the rise in bilirubin levels. The main findings of the disease are hepatomegaly associated with abdominal pain and jaundice. Liver functions are otherwise normal. As a result, the liver may be grossly black in appearance. Case report A 20 y, old woman weighing 60 kg was scheduled for emergency laparoscopic appendectomy. Pre—anesthetic evaluation of the patient showed increased total and direct bilirubin. Bilirubin values were raised with total bilirubin of CT abdomen showed normal size liver with smooth outline and homogenous parenchymal enhancement with no focal lesions. The common bile duct and intrahepatic biliary radicles were not dilated. Portal and hepatic veins were normal. Patient reported a similar icterus and intermittent rise in bilirubin levels during her previous pregnancy. She was then referred to gastroenterologist and consequently diagnosed as DJS based on the history and investigations. Despite diagnosis she had not undergone any treatment for DJS and considered a self-limiting condition with enzymes getting back to normal. For the laparoscopic appendicectomy, patient fasted for 8 hours and scheduled as the first case in the morning list. Anesthetic induction included fentanyl 50 mcg, remifentanil infusion, propofol mg and cis-atracurium 6 mg intravenously. Patient was intubated with size 7 cuffed endotracheal tube using GlideScope blade 3. During laparoscopic visualization, the liver appeared dark and almost black in color Figure 1. This is a pathognomonic sign of DJS. Neostigmine 2. The patient had a smooth extubation and painless recovery period. Patient was monitored in PACU for one hour and subsequently transferred to the standard ward. Two days after surgery, bilirubin levels checked and a downward trend was observed with total bilirubin Discussion DJS is a rare autosomal recessive disorder characterized by chronic intermittent hyper-bilirubinemia occurring in both sexes in all nationalities and races. There is 1. The degree of hyperbilirubinemia may be increased by intercurrent illnesses, oral contraceptives, pregnancy and stress. Frank Johnson and Dr. Dubin discovered DJS, in This pigment is not melanin and is thought to be derived from epinephrine metabolites that have abnormal excretion. In DJS, the defect in cMOAT canalicular multi-specific organic anion transporter protein seems to be responsible for the predominantly conjugated hyperbilirubinemia and the accumulation of pigment in the lysosomes of the hepatocytes. Macroscopically, the liver appears grey or even black Figure 1. Histologically, the cytoplasm of the hepatocytes contains big lysosomal granules packed with a lipochromic pigments. These were mainly located in the centrilobular region which is responsible for this color of the liver. Adachi et al. Propofol was chosen rather than thiopentone or ketamine because of its major extrahepatic clearance. Remifentanil is an ultra-short acting drug and metabolized by blood and tissue esterases. We avoided long-acting narcotic analgesics as well as paracetamol related to their concerns in liver diseases. The initial increase was followed by subsequent decrease. Revising the literature, we could not find a clear evidence about the drug metabolism derangements in DJS. As the liver does not have enzyme deficiencies as in other causes of hyperbilirubinemia, the fact that the liver will affect the drug metabolism is questionable. In this case we were being cautious for concern of the patient safety and took all the measures in selecting our medications. Though in DJS, the serum bilirubin levels are raised significantly, other routine laboratory tests are normal. Conclusion Dubin Johnson Syndrome is a benign intermittent conjugated hyperbilirubinemia with normal transaminases. Hepatic interaction of anesthetic drugs needs to be further studied. However, considering the patient safety from the deleterious effect of hyperbilirubinemia especially in stress situations, safe anesthetic protocol should be employed. Conflict of interests None declared by the authors. Dubin-Johnson syndrome. McGraw-Hill Education; Rabinowitz SS. Dubin-Johnson Syndrome. Available from: emedicine. A case of Dubin-Johnson Syndrome in pregnancy. Best Pract Res Clin Gastroenterol. Chronic idiopathic jaundice with unidentified pigment in liver cells; a new clinicopathologic entity with a report of 12 cases. Medicine Baltimore. Film retakes in digital and conventional radiography. J Coll Physicians Surg Pak. Postoperative management following massive hepatectomy in a patient with Dubin-Johnson syndrome: report of a case. Surg Today. Diagnostic and pathogenetic implications of urinary coproporphyrin excretion in the Dubin-Johnson syndrome. The Dubin-Johnson syndrome: case report and review of literature. Acta Gastroenterol Latinoam. Nihon Rinsho. Thiopentone as a factor in the production of liver dysfunction. Br J Anaesth. Changes in serum enzyme levels following ketamine infusions. Analgesics in patients with hepatic impairment: pharmacology and clinical implications.