Case Report: Multicentric Reticulohistiocytosis Associated With Posterior Mediastinal Adenosquamous Carcinoma, Antinuclear Antibody Positivity and Lupus Anticoagulant Positivity

Multicentric reticulohistiocytosis (MRH) is a rare systemic disease of non-Langerhans cell histiocytosis. A number of studies in the literature have documented that it can coexist with malignancy or autoimmune disease, making it difficult to determine the most appropriate therapy. Here, we present a case study of MRH associated with posterior mediastinal adenosquamous carcinoma along with antinuclear antibody positivity and lupus anticoagulant positivity. The patient experienced 6 months of clinical benefit after surgical resection and chemoradiotherapy of the mediastinal malignancy. This case adds to the available literature on multicentric reticulohistiocytosis associated with different types of malignancy and provides supplementary clinical data on the coexistence of this syndrome with malignancy and immune system abnormalities. To the best of our knowledge, this is the first case study describing MRH accompanied by posterior mediastinal adenosquamous carcinoma and lupus anticoagulant positivity. The unknown aetiology and polymorphic clinical presentation of MRH warrants further investigation.

Unresectable primary hepatic adenosquamous carcinoma successfully treated with systemic and transcatheter hepatic arterial injection chemotherapies followed by conversion surgery: a case report and literature review

Background Primary hepatic adenosquamous carcinoma (ASC) is a type of tumor that has the features of both adenocarcinoma and squamous cell carcinoma (SCC). The prognosis for patients with ASC is poor, as the chemotherapy has been ineffective so far. Case presentation Here, we report a case of a 62-year-old male patient who presented with high fever. The tumor marker levels were high, and abdominal dynamic computed tomography showed a liver tumor and distant lymph node metastases. Upon further investigation, needle biopsy of the liver tumor showed a primary hepatic SCC. Because the SCC was unresectable, the patient was treated with tegafur/gimeracil/oteracil (S-1) and transcatheter hepatic arterial injection (TAI) of cisplatin. After chemotherapy, a surgical resection performed on the remaining liver tumor, made the patient cancer-free. After the operation, the liver tumor was confirmed as primary hepatic ASC. Subsequently, the patient was administered postoperative adjuvant chemotherapy, which prevented its recurrence. Conclusions Due to the lack of an effective treatment for primary hepatic ASC, its prognosis is poor. Here, we suggest that a chemotherapy combination of 5-fluorouracil (S-1) and cisplatin along with conversion surgery might be an effective way for treating primary hepatic ASC. Our experience from this case shall be valuable to clinicians around the world involved in the treatment of primary hepatic ASC.

A Rare Case of Primary Hepatic Adenosquamous Carcinoma Mimicking Liver Abscess

A 61-year-old woman with a 3-week history of chills, fevers to 39°C, anorexia, weight loss, and fatigue was referred to our hospital due to the progressive discomforts over the last week. Her documented medical history was notable for well-controlled type 2 diabetes. Physical examination was unremarkable. Notable laboratory tests of the blood revealed a hemoglobin level of 90 g/L, a white blood cell count of 11.7×109/L with neutrophil 90.7%, albumin 31.6 g/L, gamma glutamyl transferase 331 IU/L, alkaline phosphatase 748 IU/L. Direct microbiologic detection and cultures were negative for hepatitis B and C virus, bacterial and fungal pathogens, and human immunodeficiency virus. A computed tomography scan of the chest was unremarkable. Abdominal contrast-enhanced computed tomography discovered an ill-defined and sub-circular lowdensity region measuring 7.6×6.1cm with patchy enhancement in the right lobe of liver (Figure A), which implied a liver abscess. Empiric antibiotics were changed to meropenem combined with metronidazole on day three of hospitalisation. However, the fever did not alleviate with poorly therapeutic effect. Ultrasound-guided biopsy was performed and purulent fluid was collected to make a definitive diagnosis of the liver mass. Surprisingly, The histopathological detection of a needle biopsy sample confirmed neoplastic necrosis with negative bacterial and fungal culture of the purulent fluid. The patient then underwent right hemihepatectomy and histopathologic examination further revealed that the lesion included a mixture of a poorly differentiated adenocarcinoma with mucin production and squamous cell carcinoma with keratinization, as well as large patchy necrosis (Figure B). On immunostaining, the lesion stained strongly with CK5/6, P63, and P40, and was also partially positive for CK7, CK8/18, and Cam5.2 with negative staining for hepatocyte and alpha-fetoprotein, which were consistent with adenosquamous carcinoma of the liver. Primary hepatic adenosquamous carcinoma is a rare variant of intrahepatic cholangiocarcinoma with approximately 10 cases reported within 5 years.1,2 The pathogenesis of adenosquamous carcinoma of the liver is largely unknown owing to limited data in the literature. Fever, jaundice, weight loss, anorexia, and abdominal pain are the most frequently reported clinical manifestations.3 Adenosquamous carcinoma of the liver usually presents atypical imaging characteristics, which is difficult to distinguish it from liver abscess, hepatocellular carcinoma, and liver metastasis. Adenosquamous carcinoma of the liver is a highly aggressive neoplasm and the median survival time is 6 months after surgery.4 Therefore, preoperative diagnosis is of vital importance to predict a poor prognosis. Despite postoperative chemoradiation, the patient passed away 7 months after surgery.