ABSTRACT
Vanishing bile duct syndrome is a rare acquired condition, characterized by progressive loss of intrahepatic bile ducts leading to ductopenia and cholestasis. It can be associated with infections, ischemia, drug adverse reactions, neoplasms, autoimmune disease, and allograft rejection. Prognosis is variable and depends on the etiology of bile duct injury. We report the case of a 25-year-old female with cholestatic hepatitis and concomitant intakes of hepatotoxic substances, such as garcinia, field horsetail, and ketoprofen. On suspicion of a drug-induced liver injury, the drugs were promptly withdrawn and ursodeoxycholic acid was started with initial clinical and laboratory improvement, and the patient was discharged from the hospital. One month later, she had a new increase in bilirubin levels and canalicular enzymes, requiring a liver biopsy that showed significant loss of intrahepatic bile ducts, which was compatible with vanishing bile duct syndrome. This was confirmed by using cytokeratin 19 on immunohistochemistry. There was subsequent lymph node enlargement in several chains, and relevant weight loss. Histological analysis of a cervical lymph node revealed nodular sclerosis-subtype classic Hodgkin lymphoma. In this setting, vanishing bile duct syndrome was related to Hodgkin lymphoma and a drug-induced liver injury overlap, leading to progressive cholestasis with a worse prognosis. The patient's response to chemotherapy was poor, requiring biological therapy with brentuximab vedotin. It is crucial for physicians to create a broad differential diagnosis in suspected vanishing bile duct syndrome patients, especially to rule out malignancies.
Subject(s)
Bile Ducts, Intrahepatic/pathology , Chemical and Drug Induced Liver Injury/complications , Cholestasis, Intrahepatic/etiology , Hodgkin Disease/complications , Liver/pathology , Lymph Nodes/pathology , Adult , Alanine Transaminase/blood , Anti-Inflammatory Agents, Non-Steroidal/adverse effects , Antineoplastic Combined Chemotherapy Protocols/therapeutic use , Aspartate Aminotransferases/blood , Bilirubin/blood , Biopsy , Chemical and Drug Induced Liver Injury/blood , Chemical and Drug Induced Liver Injury/etiology , Chemical and Drug Induced Liver Injury/pathology , Cholagogues and Choleretics/therapeutic use , Cholestasis, Intrahepatic/blood , Cholestasis, Intrahepatic/drug therapy , Cholestasis, Intrahepatic/pathology , Equisetum/adverse effects , Female , Garcinia/adverse effects , Gastritis/etiology , Hematemesis/etiology , Hodgkin Disease/diagnostic imaging , Hodgkin Disease/drug therapy , Hodgkin Disease/pathology , Humans , Ketoprofen/adverse effects , Positron Emission Tomography Computed Tomography , Tomography, X-Ray Computed , Ursodeoxycholic Acid/therapeutic useABSTRACT
Introducción: las causas más frecuentes de pancreatitis aguda son las litiasis biliares, el consumo de alcohol, el tabaquismo o los tumores. Hay un porcentaje de ellas que quedan sin causa establecida, catalogándose de pancreatitis idiopática. Caso clínico: presentamos el caso de una mujer de 56 años con antecedente de suprarrenalectomía bilateral de las glándulas suprarrenales en tratamiento hormonal sustitutivo con corticoides, que presenta episodios de pancreatitis aguda leve de repetición con estudio etiológico (analítico y pruebas de imagen) sin hallazgos. Se sospecha el origen tóxico, por lo que se retiran los corticoides y se modifica el tratamiento antihipertensivo, pero la clínica persiste. Posteriormente se detecta el consumo habitual de infusiones de cola de caballo. Tras su suspensión la paciente se queda asintomática y no vuelve a presentar nuevos episodios. Discusión: la pancreatitis aguda tóxica es una causa rara de pancreatitis que con cierta frecuencia queda sin diagnosticar por la dificultad de establecer una relación entre el agente tóxico y la pancreatitis. Los fármacos relacionados con las pancreatitis agudas son múltiples, mientras que la información disponible es escasa con los productos de herboristería. Se suelen presentar como episodios leves y recurrentes, sin objetivar la causa en el estudio tanto analítico como por pruebas complementarias (ecografía de abdomen, tomografía computarizada [TC] de abdomen, colangiopancreatografía por resonancia magnética [RMN] y ecoendoscopia). Es importante detectar el origen de estas pancreatitis para evitar su recurrencia (AU)
Introduction: The most frequent causes of acute pancreatitis are biliary stones, alcohol consumption, smoking and tumors. Some of them do not have any established cause, and they are catalogued as idiopathic pancreatitis. Case report: We report the case of a 56-year-old woman with a history of bilateral adrenalectomy on hormone replacement therapy with corticosteroids, who has recurrent episodes of mild acute pancreatitis with an etiologic study (laboratory and imaging tests) without significant findings. A drug-induced etiology was suspected, so corticosteroids were removed and antihypertensive treatment was modified, but the clinical manifestations persisted. Later regular consumption of horsetail infusions was detected, and after their suspension the patient became asymptomatic and has not presented new episodes. Discussion: The drug-induced acute pancreatitis is a strange cause of pancreatitis that is frequently underdiagnosed because of the difficulty to establish a relationship between the drugs and the pancreatitis. Lots of drugs have been related with acute pancreatitis, while the information available for herbal products is limited. They usually present like mild and recurrent episodes, without significant findings in both laboratory and imaging tests (abdominal ultrasound, abdominal computed tomography [CT], cholangiography and endoscopic ultrasound). It is important to detect the origin of this type of pancreatitis to prevent recurrence (AU)
Subject(s)
Humans , Female , Middle Aged , Pancreatitis/complications , Equisetum/adverse effects , Equisetum/toxicity , Hydrocortisone/analysis , Adrenal Cortex Hormones/therapeutic use , Medicinal Herbs Store , Cholangiopancreatography, Magnetic Resonance/methods , Cholangiopancreatography, Magnetic Resonance , Endosonography , Diagnosis, DifferentialABSTRACT
INTRODUCTION: The most frequent causes of acute pancreatitis are biliary stones, alcohol consumption, smoking and tumors. Some of them do not have any established cause, and they are catalogued as idiopathic pancreatitis. CASE REPORT: We report the case of a 56-year-old woman with a history of bilateral adrenalectomy on hormone replacement therapy with corticosteroids, who has recurrent episodes of mild acute pancreatitis with an etiologic study (laboratory and imaging tests) without significant findings. A drug-induced etiology was suspected, so corticosteroids were removed and antihypertensive treatment was modified, but the clinical manifestations persisted. Later regular consumption of horsetail infusions was detected, and after their suspension the patient became asymptomatic and has not presented new episodes. DISCUSSION: The drug-induced acute pancreatitis is a strange cause of pancreatitis that is frequently underdiagnosed because of the difficulty to establish a relationship between the drugs and the pancreatitis. Lots of drugs have been related with acute pancreatitis, while the information available for herbal products is limited. They usually present like mild and recurrent episodes, without significant findings in both laboratory and imaging tests (abdominal ultrasound, abdominal computed tomography [CT], cholangiography and endoscopic ultrasound). It is important to detect the origin of this type of pancreatitis to prevent recurrence.
Subject(s)
Equisetum/adverse effects , Pancreatitis/chemically induced , Plant Extracts/adverse effects , Cholangiopancreatography, Endoscopic Retrograde , Female , Humans , Middle Aged , Pancreatitis/diagnostic imagingABSTRACT
Self-administration of complementary products concurrently with conventional medication is increasingly common. The potential for cytochrome P450 (CYP) inhibition requires investigation. The N-in-one assay with ten probe substrates for nine CYPs was used with human liver microsomes to investigate ten products. CYP inhibition was measured in a single liquid chromatography-tandem mass spectrometry (LC/MS-MS) analysis. Estimated IC(50)-values were determined for the extracts that produced significant inhibition (less than 100 microg ml(-1)). Inhibition of CYP2C19 by dong quai (IC(50) = 13.7-14.3 microg ml(-1) for the methanolic extract) and CYP2D6 by goldenseal (IC(50) = 6.7 and 6.3 microg ml(-1) for the aqueous and methanolic extracts, respectively), are of particular concern as the potential for adverse interactions is high. The inhibition of CYP2C8 by horsetail (IC(50) = 93 microg ml(-1) for the aqueous extract) requires further investigation, as the potential for concurrent use with products that require CYP2C8 for metabolism is significant. CYP3A4 inhibition varied depending on the probe reaction being monitored. The earlier reported findings of inhibition by black cohosh, goldenseal and gotu kola were confirmed. The present work has shown that the N-in-one cocktail is a rapid and reliable method that can be used as an initial screen to help prioritize products that require more detailed investigations and it can also be applied to monitor product variability.