Sickle cell intrahepatic cholestasis (SCIC) is a rare but potentially fatal complication of sickle cell disease (SCD), with high mortality, observed mainly in patients with homozygous sickle cell anemia. Herein, we have reported a case of severe SCIC with a poor outcome despite aggressive measures including exchange transfusion and use of vasopressors.The patient was admitted with generalized weakness, confusion, rigors, chills, and signs of hepatic failure, such as hyperbilirubinemia, hypoalbuminemia, and coagulopathy. There was no evidence of viral hepatitis or biliary obstruction. The patient received two exchange transfusions, but he continued to deteriorate clinically despite exchange transfusion and developed hemorrhagic shock and multiorgan failure. The patient was made comfort care as per family wishes. This case emphasizes the importance of early diagnosis of sickle cell intrahepatic cholestasis and poor prognosis despite aggressive measures.
Streptococcus constellatus is a member of Streptococcus milleri group which is a subgroup of Viridans streptococci, first described by Guthof in 1956 after being isolated from dental abscesses. S. constellatus, a gram positive, non-sporing, non-motile, catalase negative cocci, is the normal flora of the oropharyngeal, gastrointestinal and urogenital tract. It is not a commonly encountered pathogen but has a propensity to form abscesses and cause bacteremia in the immunocompromised patient. Here, we report a 78-year-old man with sepsis due to Streptococcus constellatus liver abscess. The patient had a history of hypertension, stroke, benign prostatic hyperplasia, vascular dementia and myocardial infarction status post coronary artery bypass grafting. There has been no particular link between any of these conditions to S. constellatus. However, immunocompromised status predisposes to fulminant infection and formation of abscesses. The patient was febrile with a temperature of 99.1°F, blood pressure of 143/73 mmHg and the heart rate (HR) of 98. Labs revealed a leukocytosis of 16.90 K/uL, hemoglobin 11.8 g/dL, hematocrit 35.8%, total bilirubin 1.7 mg/dL, direct bilirubin 1.0 mg/dL, aspartate aminotransferase (AST) 44 IU/L, alanine aminotransferase (ALT) 28 IU/L, alkaline phosphatase (ALKP) 176 IU/L and lactate dehydrogenase (LDH) was 290 IU/L. He was started on intravenous Maxipime and Unasyn which was switched to Rocephin and Clindamycin based on the Infectious disease recommendations. Metronidazole was also started and the serologies were sent for Entamoeba histolytica. Computerized tomography (CT) scan showed an abscess in the right lobe of the liver which was finally drained using an interventional radiology (IR)guided approach. The cultures from the fluid and blood yielded S. constellatus and thus Metronidazole was discontinued. The patient improved after a few days and the drainage catheter was pulled out and the patient discharged in stable condition.
Caroli disease is a rare congenital disorder resulting from the dilation of large intrahepatic bile ducts. Patients affected with Caroli disease are at increased risk of complications resulting from bile stasis and stone formation. We report the case of a 37-year-old woman with a past surgical history of cholecystectomy who presented to the emergency room with a chief complaint of abdominal pain and nausea. The pain was characteristic of acute pancreatitis but she was hemodynamically stable. Total bilirubin was 4.1 mg/dL with a direct fraction of 3.1 mg/dL, aspartate aminotransferase (AST) and alanine aminotransferase (ALT) were 850 IU/L and 1025 IU/L, respectively. Serum amylase and lipase were elevated at 581 IU/L and 1328 IU/L, respectively. CT scan of abdomen/pelvis without contrast showed common bile duct (CBD) measuring 1.6 cm with intrahepatic biliary system dilation and mild peripancreatic fat stranding. She was diagnosed with acute pancreatitis. On the second day, she developed a temperature of 99.6°F. Hepatitis immunity panel was negative for acute hepatitis. The patient was started on antibiotics (IV ciprofloxacin and metronidazole) for suspicion of acute cholangitis. Endoscopic retrograde cholangiopancreatography (ERCP) was done which showed mild dilated intrahepatic ducts and CBD dilation of 1.6 cm, and a choledochal cyst at CBD. Sphincterotomy was done and good bile drainage was reported. She was later discharged in a stable condition. Caroli disease affects males and females equally and most are diagnosed before the age of 30 years correlated with the onset of symptoms. By far, the most commonly reported symptom is acute cholangitis but pancreatitis occurs rarely. Recurrent bouts of infection lead to portal hypertension, fibrosis of the liver and ultimately end up with an orthotopic liver transplant (OLT). Regular follow-ups are important for disease surveillance and monitoring.
Hypotonic hyponatremia is caused by a serum sodium level of <135 mEq/L in the setting of excess solute loss accompanied by free water retention because of antidiuretic hormone release, subsequent to decreased effective arterial blood volume. Acute hyponatremia can have various neurological manifestations, including drowsiness, lethargy, coma, seizures, respiratory depression, and even death. In this article, we present a case of a 41-year-old man who presented with hyponatremia as a result of sodium containing biliary fluid loss and resultant renal free water retention in response to increased antidiuretic hormone secretion. He underwent placement of a cholecystostomy tube for acalculous cholecystitis and was found to be persistently hyponatremic despite repletion with sodium-containing fluids. Once the cholecystostomy tube was removed, the patient’s sodium levels improved, and his symptoms resolved. Our case highlights choleuresis as an unusual but significant cause of hyponatremia in patients who have external biliary drainage.
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