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Biliary hamartomas are tumor-like malformations of the liver. Biliary hamartomas are a type of fibrocystic disorder originating from ductal plate malformation and are typically considered benign, but with the risk of malignant transformation. In this case report, we present a rare occurrence of intrahepatic cholangiocarcinoma (ICC) that developed from biliary hamartomas, along with a literature review. A 76-year-old man with a diagnosis of biliary hamartomas had a history of recurrent cholangitis for 12 years, necessitating cholecystectomy, ERCP, and repeated antibiotic treatments. During his last episode, imaging studies revealed a hypervascular infiltrative mass in the right posterior liver segment. A liver biopsy confirmed adenocarcinoma and subsequent surgical pathology revealed ICC originating from biliary hamartomas. Chronic inflammation in the bile duct associated with biliary hamartomas may serve as a potential trigger for malignant transformation, as observed in this case. Therefore, close surveillance is essential for patients with biliary hamartomas presenting with infectious complications.
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Background@#Research regarding cervical metastasis from an unknown primary tumor (CUP) according to human papillomavirus (HPV) and Epstein-Barr virus (EBV) status in Korea has been sporadic and small-scale. This study aims to analyze and understand the characteristics of CUP in Korea according to viral and p16 and p53 status through a multicenter study. @*Methods@#Ninety-five cases of CUP retrieved from six hospitals in Korea between January 2006 and December 2016 were subjected to high-risk HPV detection (DNA in situ hybridization [ISH] or real-time polymerase chain reaction), EBV detection (ISH), and immunohistochemistry for p16 and p53. @*Results@#CUP was HPV-related in 37 cases (38.9%), EBV-related in five cases (5.3%), and unrelated to HPV or EBV in 46 cases (48.4%). HPV-related CUP cases had the best overall survival (OS) (p = .004). According to the multivariate analysis, virus-unrelated disease (p = .023) and longer smoking duration (p < .005) were prognostic factors for poor OS. Cystic change (p = .016) and basaloid pattern (p < .001) were more frequent in HPV-related cases, and lymphoepithelial lesion was frequent in EBV-related cases (p = .010). There was no significant association between viral status and p53 positivity (p = .341), smoking status (p = .728), or smoking duration (p = .187). Korean data differ from Western data in the absence of an association among HPV, p53 positivity, and smoking history. @*Conclusions@#Virus-unrelated CUP in Korea had the highest frequency among all CUP cases. HPV-related CUP is similar to HPV-mediated oropharyngeal cancer and EBVrelated CUP is similar to nasopharyngeal cancer in terms of characteristics, respectively.
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As more individuals were coronavirus disease 2019 (COVID-19) vaccinated, unexpected side effects appeared. Herein, we present the case of a 30-year-old male patient with myopathy in both extremities after the second dose of the Pfizer-BioNTech (BNT162b2) COVID-19 vaccine. Symptoms, swelling and pain, started from the proximal upper and lower extremities and extended to the distal parts. Although he underwent massive hydration, the muscle enzyme level continuously increased. He complained of dysphagia and dysarthria.Microscopically, muscle biopsy showed multifocal or scattered macrophage infiltration and degenerated myofibers. In contrast to general myopathy including inflammatory myositis and rhabdomyolysis, vaccine-induced inflammatory myositis shows a prolonged increase in muscle enzyme levels and multifocal macrophage infiltration with necrosis of the muscle fibers. Symptoms improved with glucocorticoid and immunosuppressive treatment. If vaccinated individuals experience severe and continuous muscle pain and swelling, clinicians should consider vaccine-induced inflammatory myositis, measure the muscle enzyme levels, and perform muscle biopsy for a definite diagnosis.
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Purpose@#To determine the multidetector CT (MDCT) findings that differentiate adenomyoma of the ampulla of Vater (AOV) from localized adenocarcinoma of the AOV. @*Materials and Methods@#Sixteen and 30 patients with adenomyoma and localized adenocarcinoma of the AOV, respectively, were evaluated using MDCT. We analyzed the size and attenuation value and presence of uniform enhancement of the lesions, diameters of the extrahepatic bile duct (EHD) and main pancreatic duct, presence of regional lymph node enlargement, and laboratory findings. We determined the independent findings for differentiating adenomyoma from localized adenocarcinoma of the AOV using multivariate analysis. @*Results@#The size of the lesion and diameter of the EHD were significantly smaller for adenomyoma than those for localized adenocarcinoma of the AOV (all p < 0.001). In multivariate analyses, a lesion size of ≤ 1.3 cm, an EHD diameter of ≤ 1.3 cm, and an alanine transaminase level of ≤ 31 IU/L significantly differentiated adenomyoma from localized adenocarcinoma of the AOV. When all of these three findings were met, the specificity for adenomyoma of the AOV was 93.3%. @*Conclusion@#MDCT imaging may facilitate the differential diagnosis of adenomyoma and localized adenocarcinoma of the AOV based on the size of the lesion and diameter of the EHD.
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Purpose@#To determine the multidetector CT (MDCT) findings that differentiate adenomyoma of the ampulla of Vater (AOV) from localized adenocarcinoma of the AOV. @*Materials and Methods@#Sixteen and 30 patients with adenomyoma and localized adenocarcinoma of the AOV, respectively, were evaluated using MDCT. We analyzed the size and attenuation value and presence of uniform enhancement of the lesions, diameters of the extrahepatic bile duct (EHD) and main pancreatic duct, presence of regional lymph node enlargement, and laboratory findings. We determined the independent findings for differentiating adenomyoma from localized adenocarcinoma of the AOV using multivariate analysis. @*Results@#The size of the lesion and diameter of the EHD were significantly smaller for adenomyoma than those for localized adenocarcinoma of the AOV (all p < 0.001). In multivariate analyses, a lesion size of ≤ 1.3 cm, an EHD diameter of ≤ 1.3 cm, and an alanine transaminase level of ≤ 31 IU/L significantly differentiated adenomyoma from localized adenocarcinoma of the AOV. When all of these three findings were met, the specificity for adenomyoma of the AOV was 93.3%. @*Conclusion@#MDCT imaging may facilitate the differential diagnosis of adenomyoma and localized adenocarcinoma of the AOV based on the size of the lesion and diameter of the EHD.
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Pancreatic candidiasis can develop in patients with acute pancreatitis, compromised immune responses, or iatrogenic intervention. This paper reports a case of pancreatic candidiasis presenting as a solid pancreatic mass in a patient without the risk factors. A previously healthy 37-year-old man visited the emergency department with left flank pain. Abdominal CT revealed a 5 cm, irregular heterogeneous enhancing mass accompanied by a left adrenal mass. Positron emission tomography-computed tomography and endoscopic ultrasound-guided fine-needle aspiration (EUS-FNA) could not discriminate pancreatic cancer from infectious disease. A laparoscopic exploration was performed for an accurate diagnosis. After distal pancreatectomy with splenectomy and left adrenalectomy, pancreatic candidiasis and adrenal cortical adenoma were diagnosed based on the pathology findings. His condition improved after the treatment with fluconazole. This paper reports a case of primary pancreatic candidiasis mimicking pancreatic cancer in an immunocompetent patient with a review of the relevant literature.
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The World Health Organization 2016 edition assigned anaplastic lymphoma kinase (ALK) rearrangement-associated renal cell carcinoma (ALK-RCC) as an emerging renal tumor entity. Identifying ALK-RCC is important because ALK inhibitors have been shown to be effective in treatment. Here, we report the case of a 14-year-old young man with ALK-RCC. Computed tomography revealed a well-demarcated 5.3-cm enhancing mass at the upper pole of the left kidney. There was no further history or symptoms of the sickle-cell trait. The patient underwent left radical nephrectomy. Pathologically, the mass was diagnosed as an unclassified RCC. Targeted next-generation sequencing identified a TPM3-ALK fusion gene. The present report and literature review demonstrate that TPM3-ALK RCC may be associated with distinct clinicopathological features. Microscopically, the tumors showed diffuse growth and tubulocystic changes with inflammatory cell infiltration. Tumor cells were dis-cohesive and epithelioid with abundant eosinophilic cytoplasm and cytoplasmic vacuoles. If morphological features and TFE3 expression are present in adolescent and young patients, molecular tests for ALK translocation should be performed. This awareness is critically important, because ALK rearrangement confers sensitivity to ALK inhibitors.
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Adolescente , Humanos , Carcinoma de Células Renais , Citoplasma , Eosinófilos , Rearranjo Gênico , Rim , Linfoma , Nefrectomia , Fosfotransferases , Vacúolos , Organização Mundial da SaúdeRESUMO
Background@#Ankylosis in the temporomandibular joint (TMJ) is known to have various etiologies in veterinary medicine. We observed a case of fibrous ankylosis of the TMJ in a newly imported rhesus monkey (Macaca mulatta).Case presentation:Moderate to severe attrition was found in the middle labial portion of the left maxillary canine. No tenderness around the jaw was detected in the physical examination. The WBC count, CRP level, rheumatoid factor level, and other parameters were normal. Irregularity in the joint surface was observed in both TMJs in the radiographic and computed tomographic examinations, but the left TMJ presented more severe irregularity. It was determined that the removal of the locked portion of the left canine would alleviate the case of lockjaw and allow intubation with an endotracheal tube. Canine tooth crown reduction was performed for both canine teeth. The mouth opening distance slightly (approximately 5 mm) increased up to 20 mm. We concluded that the attrition of canine teeth was not the reason for lockjaw and ankyloses originating from TMJ disease. Fibrotic synovial tissue and joint surface irregularity were observed by necropsy. The presence of fibrocartilage in most areas of the TMJ was confirmed by histology. The diagnosis was fibrous ankylosis of the TMJ associated with DJD. @*Conclusions@#To the best of our knowledge, this is the first report of degenerative joint disease of the TMJ in a rhesus monkey with fibrous ankylosis of the TMJ.
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A 59-year-old woman presented with abdominal pain. Abdominal computerized tomography was suggestive of biliary stones. During endoscopic retrograde cholangiopancreatography, adult worms resembling Clonorchis sinensis (C. sinensis) were drained. Eggs were detected in stool using the formalin-ether concentration method and C. sinensis-specific antibody was detected in the serum. A diagnosis of C. sinensis infection was made. The symptoms of the patient gradually resolved after treatment with anti-parasite medication. The patient lived in a non-endemic region for C. sinensis infection and had no history of intake of raw or undercooked freshwater fishes. South Korea is one of the endemic countries for C. sinensis infection and people can be infected via indirect routes of transmission such as cooking utensils. Therefore, the possibility of C. sinensis infection should be considered in patients presenting with biliary diseases in South Korea. We describe the clinical findings of this case with a review of literature.
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Adulto , Feminino , Humanos , Pessoa de Meia-Idade , Dor Abdominal , Colangiopancreatografia Retrógrada Endoscópica , Colangite , Colecistite , Colecistite Aguda , Clonorchis sinensis , Utensílios de Alimentação e Culinária , Diagnóstico , Ovos , Peixes , Água Doce , Coreia (Geográfico) , Métodos , ÓvuloRESUMO
PURPOSE: Malignant pleural effusions (MPEs) are often observed in lung cancer, particularly adenocarcinoma. The aim of this study was to investigate epidermal growth factor receptor (EGFR) mutation status in lung adenocarcinoma-associated MPEs (LA-MPEs) and its correlation with efficacy of EGFR tyrosine kinase inhibitor (TKI) therapy. MATERIALS AND METHODS: Samples comprised 40 cell blocks of pathologically-confirmed LA-MPEs collected before the start of EGFR TKI therapy. EGFR mutation status was re-evaluated by peptide nucleic acid clamping and the clinical outcomes of EGFR TKI-treated patients were analyzed retrospectively. RESULTS: EGFR mutations were detected in 72.5% of LA-MPE cell blocks (29/40). The median progression-free survival for patients with EGFR mutations in LA-MPEs was better than that for patients with wild-type EGFR (7.33 months vs. 2.07 months; hazard ratio, 0.486; 95% confidence interval, 0.206 to 1.144; p=0.032). The objective response rate (ORR) of 26 patients with EGFR mutations in LA-MPEs among the 36 patients with measurable lesions was 80.8%, while the ORR of the 10 patients with wild-type EGFR in LA-MPEs was 10% (p < 0.001). Among the 26 patients with EGFR mutations in LA-MPEs, the ORR of target lesions and LA-MPEs were 88.5% and 61.5%, respectively (p=0.026). CONCLUSION: EGFR mutation status in cell blocks of LA-MPEs confirmed by pathologic diagnosis is highly predictive of EGFR TKI efficacy. For patients with EGFR mutations in LA-MPEs, the response to EGFR TKIs seems to be worse for pleural effusions than for solid tumors.
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Humanos , Adenocarcinoma , Constrição , Diagnóstico , Intervalo Livre de Doença , Neoplasias Pulmonares , Pulmão , Derrame Pleural , Derrame Pleural Maligno , Proteínas Tirosina Quinases , Receptores ErbB , Estudos Retrospectivos , TirosinaRESUMO
Multiple primary malignant neoplasms indicate an occurrence of two or more malignancies in a patient, and double primary cancers are the most common type. When the second primary cancer occurs simultaneously or within 6 months after the first primary cancer is diagnosed, it is called synchronous carcinoma. If the second primary cancer occurs after 6 months, it is defined as metachronous carcinoma. Recently, we experienced a patient who was diagnosed as double primary cancer in the nasopharynx and nasal cavity. The tumors occurred simultaneously and histopathological examinations revealed nasopharyngeal nonkeratinizing carcinoma in the right nasopharynx and extranodal NK/T-cell lymphoma in the left nasal cavity (inferior turbinate). In situ hybridization showed positivity for Epstein-Barr virus, which encoded early RNA in the neoplastic cells of both specimens. We present this rare disease entity with a review of the relevant literature and a survey of the clinical characteristics.
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Humanos , Herpesvirus Humano 4 , Hibridização In Situ , Linfoma , Cavidade Nasal , Nasofaringe , Neoplasias Primárias Múltiplas , Segunda Neoplasia Primária , Doenças Raras , RNARESUMO
BACKGROUND: Immature teratoma (IT) is a tumor containing immature neuroectodermal tissue, primarily in the form of neuroepithelial tubules. However, the diagnosis of tumors containing only cellular neuroglial tissue (CNT) without distinct neuroepithelial tubules is often difficult, since the histological characteristics of immature neuroectodermal tissues remain unclear. Here, we examined the significance of CNT and tried to define immature neuroectodermal tissues by comparing the histological features of neuroglial tissues between mature teratoma (MT) and IT. METHODS: The histological features of neuroglial tissue, including the cellularity, border between the neuroglial and adjacent tissues, cellular composition, mitotic index, Ki-67 proliferation rate, presence or absence of tissue necrosis, vascularity, and endothelial hyperplasia, were compared between 91 MT and 35 IT cases. RESULTS: CNTs with a cellularity grade of ≥ 2 were observed in 96% of IT cases and 4% of MT cases (p < .001); however, CNT with a cellularity grade of 3 in MT cases was confined to the histologically distinct granular layer of mature cerebellar tissue. Moreover, CNT in IT exhibited significantly higher rates of Ki-67 proliferation, mitoses, and necrosis than those in MT (p < .001). Furthermore, an infiltrative border of neuroglial tissue and glomeruloid endothelial hyperplasia were significantly more frequent in IT cases than in MT cases (p < .001). CONCLUSIONS: Our results suggest that if CNT with a cellularity grade of ≥ 2 is not a component of cerebellar tissue, such cases should be diagnosed as IT containing immature neuroectodermal tissue, particularly if they exhibit an infiltrative border, mitoses, necrosis, and increased Ki-67 proliferation.
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Feminino , Diagnóstico , Hiperplasia , Mitose , Índice Mitótico , Necrose , Placa Neural , Neuroglia , Ovário , TeratomaRESUMO
Isolated gastric IgG4-related disease (IgG4-RD) is a very rare tumefactive inflammatory condition, with only a few cases reported to date. A 48-year-old woman was incidentally found to have a subepithelial tumor in the stomach. Given a presumptive diagnosis of gastrointestinal stromal tumor or neuroendocrine tumor, she underwent wedge resection. The lesion was vaguely nodular and mainly involved the submucosa and proper muscle layer. Microscopically, all classical features of type I autoimmune pancreatitis including lymphoplasmacytic infiltration, storiform fibrosis, obliterative phlebitis, and numerous IgG4-positive plasma cells were seen. She had no evidence of IgG4-RD in other organs. Although very rare, IgG4-RD should be considered one of the differential diagnoses in the setting of gastric wall thickening or subepithelial mass-like lesion. Deep biopsy with awareness of this entity might avoid unnecessary surgical intervention.
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Feminino , Humanos , Pessoa de Meia-Idade , Doenças Autoimunes , Biópsia , Diagnóstico , Diagnóstico Diferencial , Fibrose , Tumores do Estroma Gastrointestinal , Granuloma de Células Plasmáticas , Tumores Neuroendócrinos , Pancreatite , Flebite , Plasmócitos , EstômagoRESUMO
As mast cells have been highlighted in the pathogenesis of diarrhea-predominant irritable bowel syndrome, a new term "mastocytic enterocolitis" was suggested by Jakate and colleagues to describe an increase in mucosal mast cells in patients with chronic intractable diarrhea and favorable response to treatment with antihistamines. Although it is not an established disease entity, two cases have been reported in the English medical literature. Here, for the first time in Asia, we report another case of chronic intractable diarrhea caused by gastrointestinal mastocytosis. The patient was a 70-year-old male with chronic intractable diarrhea for 3 months; the cause of the diarrhea remained obscure even after exhaustive evaluation. However, biopsy specimens from the jejunum were found to have increased mast cell infiltration, and the patient was successfully treated with antihistamines.