Clinical Case Summary

Hepatic hydatid cyst is caused by parasitic infection with Echinococcus granulosus or Echinococcus multilocularis. Echinococcus primarily affects dogs as definitive hosts, while humans are incidental hosts. Given their diverse radiological presentations, diagnosing hepatic hydatid cysts is particularly challenging in regions with low prevalence. We describe the diagnostic and treatment procedures of Echinococcosis in a Korean adolescent. An 18-year-old Korean male, who had traveled to Mongolia for 1–2 weeks around 15, 11, and 8 years ago, presented at Boramae Medical Center in Seoul, South Korea with symptoms of continuous fever, vomiting, and rash persisting for two weeks. No abnormal findings were observed in the physical examination and blood tests. The initial CT scan showed two hepatic cysts measuring 7 and 6 cm, with the larger one exhibiting a well-defined lesion with marginal contrast enhancement, suspicious of a hepatic abscess. After receiving percutaneous catheter drainage and antibiotic therapy, the larger cyst decreased in size to 4 cm upon discharge. A follow-up CT scan taken two months after discharge revealed that the previously drained cyst had regrown to 7 cm. A subsequent liver ultrasound revealed detached membranes in the cyst, suggesting a hydatid cyst in the transitional stage (CE3) according to the World Health Organization classification. The positive serum test for Echinococcus antibody further indicated the presence of hepatic hydatid disease. Preoperative albendazole treatment was administered, followed by a left hemihepatectomy. The pathological examination confirmed the presence of protoscolices surrounded by the laminated membrane, characteristic of a hydatid cyst. Hydatid cysts can be mistakenly diagnosed as simple cysts or liver abscesses in areas where such cysts are uncommon. Diagnosing Echinococcosis requires a high degree of suspicion based on the patient's travel history and comprehensive follow-up imaging studies.

Clinical Case Summary

Human alveolar echinococcosis (AE) is a parasitic disease, caused by E. multilocularis eggs ingestion. AE is rare and potentially fatal infection, with multisystemic involvement including the formation of tumor-like hepatic lesions. Differentiation of AE from liver malignancy is of paramount importance due to different prognostic and therapeutic implications (1).
A 33-year-old male patient, previously healthy, was referred for liver MRI due to a cholestatic liver lesion. The MRI revealed dilation of the intrahepatic bile ducts and an irregular mass at the hepatic hilum exhibiting radio-morphological features consistent with Klatskin tumor. Endoscopic ultrasound with fine needle aspiration of the hilar mass and enlarged lymph node (LN) was performed, but cytological examination did not reveal malignant cells. CA 19-9 level was normal. Subsequently, ERCP was performed, complicated with biliary leakage, leading to emergency surgery during which enlarged LN alongside the common bile duct, macroscopically suspicious for carcinoma metastasis was extracted. Again, patohistological (PH) examination revealed only reactive changes. Cytobrush collected during ERCP was negative for malignancy as well. A follow-up MRI after 2 months revealed no signs of disease progression, and PET-CT was inconclusive for malignancy.
Subsequently, patient was re-admitted to hospital due to need for further evaluation. Ultrasound-guided percutaneous biopsy of the lesion in the liver hilum was performed, and the final PH finding was indicative for AE. Treatment with albendazole at a dose of 15 mg/kg/day was initiated. Follow-up ultrasound examination did not show disease progression. Patient is clinically asymptomatic with only mild hepatic enzymes elevation consistent with albendazole treatment.
AE is an uncommon yet life-threatening parasitic infection that predominantly targets the liver. Diagnosis and treatment pose challenges, with clinical misinterpretation as malignancy not being uncommon.

References

Kawamura N, Kamiyama T, Sato N, Nakanishi K, Yokoo H, Kamachi H, et al. Long-Term Results of Hepatectomy for Patients with Alveolar Echinococcosis: A Single-Center Experience. J Am Coll Surg. 2011 May;212(5):804–12. doi: 10.1016/j.jamcollsurg.2011.02.007

Clinical Case Summary

Introduction: Albendazole is a widely used anthelmintic, generally considered safe. While mild liver enzyme elevations are reported, severe drug-induced liver injury (DILI) is rare. We present a case of acute hepatotoxicity after a short course of albendazole, highlighting its potential hepatic risks.
Case presentation: A 13-year-old girl presented with abdominal pain, jaundice, nausea, vomiting, and diarrhea for two days. She had no history of travel, fever, or rash. She had received two doses of albendazole for deworming—15 and 3 days before symptom onset—with no other medication use.
Examination revealed left epigastric tenderness without hepatomegaly. Laboratory findings included markedly elevated liver enzymes (AST 2067 IU/L, ALT 2931 IU/L, ALP 135.58 IU/L, bilirubin 51.3 mg/dL) and prolonged INR (1.86). Viral, autoimmune, and metabolic causes were ruled out. Abdominal ultrasound showed mild hepatomegaly, and CT excluded other pathology.
The Roussel Uclaf Causality Assessment Method (RUCAM) score was 9, indicating a high likelihood of albendazole-induced hepatitis. The patient was treated with supportive care, with gradual improvement and full recovery.
Discussion: DILI is a key differential in acute hepatitis when other causes are excluded. Albendazole hepatotoxicity is rare, mainly reported in long-term therapy for hydatid disease. However, our patient developed severe hepatotoxicity after only two doses, suggesting idiosyncratic or immune-mediated injury. The absence of other risk factors strengthens the causal link.
Conclusion: This case highlights the risk of severe albendazole-induced hepatotoxicity even after short-term use. Given its widespread availability, clinicians should remain vigilant for DILI in children. Liver function monitoring should be considered in at-risk patients, and unnecessary prophylactic albendazole use should be discouraged.

References

1. Chai JY, Jung BK, Hong SJ. Albendazole and Mebendazole as Anti-Parasitic and Anti-Cancer Agents: an Update. Korean J Parasitol. 2021;59(3):189-225.
2. Choi GY, Yang HW, Cho SH, et al. Acute drug-induced hepatitis caused by albendazole. J Korean Med Sci. 2008;23(5):903-905.
3. Ríos D, Restrepo JC. Albendazole-induced liver injury: a case report. Colomb Med (Cali). 2013;44(2):118-120.
4. Garcia-Cortes M, Robles-Diaz M, et al. Drug-induced liver injury: an update. Arch Toxicol. 2020;94:3381.
5. Chalasani NP, Maddur H, Russo MW, et al. ACG Clinical Guideline: Diagnosis and Management of Idiosyncratic Drug-Induced Liver Injury. Am J Gastroenterol. 2021;116:878.

Clinical Case Summary

A 48-year-old woman was diagnosed with subacute ataxia. One day after a cycle of treatment with Immunoglobulin, she presented with elevated liver enzymes, with alanine aminotransferase(ALT)levels reaching up to 5 times the normal upper limit. An abdominal ultrasound and CT scan revealed no abnormalities. Viral serologies and autoimmune tests were also normal. She was reevaluated a week later and showed improvement in liver function tests. Due to suspicion of hepatic toxicity from immunoglobulin and worsening neurological symptoms, she started methylprednisolone. Four days after, she was admitted in the emergency department with epigastric pain, nausea, and vomiting. On physical examination, she had a soft abdomen that was tender on palpation of the epigastric and right hypochondriac regions. Laboratory tests showed leukocytosis of 17,500(73% neutrophils), aspartate aminotransferase of 145 U/L, and ALT of 373U/L. An abdominal CT scan revealed multiple pericentimeter areas of enhancement in the arterial phase, more evident in the right hepatic lobe, some of which appeared coalescent and became homogeneous relative to the rest of the liver parenchyma in the venous phase, suggestive of hepatic peliosis. The corticosteroid therapy was suspended. The patient remained asymptomatic with improvement in liver function parameters. An MRI was performed three months later, which showed partial regression of the lesions. An abdominal CT scan was performed three months after that and showed complete resolution of the lesions.
Peliosis hepatis can be associated with various conditions, including infectious diseases, neoplasms, and immunosuppressive drugs such as corticosteroids, as in the presented case. Although it can be an incidental finding in asymptomatic patients, in some cases, it can lead to complications such as intraperitoneal hemorrhage, liver failure, and portal hypertension. Once the diagnosis is established, it is important to identify and remove the underlying cause.

Clinical Case Summary

A 59-years-old caucasian female patient with Crohn’s Disease (CD) A3L3B3 (Montreal Classification), erythema nodosum episodes. Due to lithiasis, while performing a cholecystectomy, a heterogenous liver was found and a biopsy revealed primary sclerosing cholangitis (PSC).
Since the CD’s diagnosis in 1992, the patient has been on mesalamine (5-ASA). During her disease course, she needed treatment with steroids 4 times. She was started on azathioprine but interrupted it due acute pancreatitis. Last time on steroids was on 2012, and since then she had been on clinical remission on 5-ASA. In 2019, the patient presented perianal disease and terminal ileitis. This justified the decision to start Infliximab (IFX) therapy.
Although asymptomatic, routine analysis at the 4th IFX infusion revealed abnormal liver biochemical tests with a hepatocellular pattern: alanine aminotransferase 449U/L(<55); aspartate aminotransferase 284U/L(<34), other enzymes were normal. Subsequent studies revealed only positive antinuclear antibody(1/640). A liver biopsy was performed in order to guide the therapeutical management. The biopsy revealed a cholestatic pattern hepatitis compatible with hepatotoxicity as well as PSC and steatosis, without features of auto-immune hepatitis (AIH). The histologic findings supported the diagnosis of IFX associated AI-like Hepatitis. IFX was stopped with slight improvement of liver enzymes and prednisolone was started. There was complete liver enzymes normalization at 7^th week, the patient was started on Ustekinumab, with no complications after 6 months.
As anti-TNF drugs’ use is rapidly increasing, associated autoimmune diseases may follow. Immune-mediated DILI related to IFX is still a rare entity but, considering that most cases are asymptomatic, monitoring liver function is mandatory for all patients. The differential diagnosis between AIH and IFX AI-like Hepatitis is challenging but essential due to therapeutic implications.

References

Rodrigues S, Lopes S, Magro F, Cardoso H, Horta e Vale AM, Marques M, Mariz E, Bernardes M, Lopes J, Carneiro F, Macedo G. Autoimmune hepatitis and anti-tumor necrosis factor alpha therapy: A single center report of 8 cases. World J Gastroenterol. 2015 Jun 28;21(24):7584-8. doi: 10.3748/wjg.v21.i24.7584. PMID: 26140007; PMCID: PMC4481456.