Epstein-Barr virus-positive inflammatory pseudotumor of the spleen managed with laparoscopic splenectomy

We report a case of Epstein-Barr virus (EBV)-positive splenic inflammatory pseudotumor treated by laparoscopic splenectomy. A 66-year-old female without symptoms was found to have a solitary, avascular, solid tumor of the spleen by contrast-enhanced computed tomography. The tumor was compatible with a primary tumor of the spleen. A benign splenic tumor was the most likely diagnosis, but malignant lymphoma could not be ruled out because the serum-soluble interleukin-2 receptor (sIL-2R) level was elevated. Laparoscopic splenectomy was performed to enable a definite diagnosis. Removal of the whole spleen without injury was possible. Possible contamination of the extirpation orifice by cancer cells was carefully prevented by enclosing the spleen in a plastic bag. Histopathological examination showed the tumor to be an EBV-positive inflammatory pseudotumor. To the best of our knowledge, this is only the second report of an EBV-positive splenic inflammatory pseudotumor with an increased serum sIL-2R level. Although EBV-positive inflammatory pseudotumors have been reported to recur, no sign of recurrence has been detected in the present case in the 17 months following splenectomy.     

A case of splenic inflammatory pseudotumor

Inflammatory pseudotumor of the spleen in a 50-year-old man was encountered in our surgical clinic. This splenic tumor is very rare and in the ninth of the cases in the literature. The removed spleen, weighting 255g, revealed the presence of a firm, circumscribed mass, measuring 4.5 × 4.0 × 4.5 cm, and histologically presenting marked lamination of collagen fibers around the small vessels associated with granulomatous lesions. The implications of its pathogenesis was analyzed with a review of the literature.Gastroenterol Jpn 1991;26.783-787.     

Hepatic inflammatory pseudotumor presenting in an 8-year-old boy: A case report and review of literature

Hepatic inflammatory pseudotumors are uncommon benign lesions. Accurately diagnosing hepatic inflammatory pseudotumor can be very challenging because the clinical presentation and radiological appearances are nonspecific and cannot be certainly distinguished from malignant neoplastic processes. Herein, we present a case of hepatic IPT in an 8-year-old boy who presented to clinic with a 3-mo history of a tender hepatic mass, fever of unknown origin, and 9-kg weight loss. The physical examination was notable for tender hepatomegaly. Laboratory investigations were notable for a normal hepatic profile and elevated erythrocyte sedimentation rate and C-reactive protein. A T2-attenuated magnetic resonance imaging scan of the abdomen showed a 4.7 cm × 4.7 cm × 6.6 cm, contrast-enhancing, hyper-intense, well-defined lesion involving the right hepatic lobe. In view of the unremitting symptoms, tender hepatomegaly, thrombosed right hepatic vein, nonspecific radiological findings, and high suspicion of a deep-seated underlying infection or malignancy, a right hepatic lobectomy was recommended. Microscopically, the hepatic lesion exhibited a mixture of inflammatory cells (histiocytes, plasma cells, mature lymphocytes, and occasional multinucleated giant cells) in a background of dense fibrous tissue. Immunohistochemically, the cells stained negative for SMA, ALK-1, CD-21 and CD-23, diffusely positive for CD-68, and focally positive for IgG4. The final histopathological diagnosis was consistent with hepatic IPT. At the postoperative 4-mo follow-up, the patient was asymptomatic without radiological evidence of recurrence.     

Secondary polycythemia as a paraneoplastic syndrome of testicular seminoma

A 45-year-old man was referred to our hospital because of polycythemia. A physical examination revealed a large tumor in his scrotum enlarged to the size of 13×10 cm. A laboratory examination revealed severe erythrocytosis with a red blood cell count of 6,820×10⁹/L, a hemoglobin concentration of 21.2 g/dL, and a hematocrit of 59.8%. The total red cell volume was increased. A right radical orchidectomy was done with minimum bleeding, and he was diagnosed as having pure seminoma. After the operation, polycythemia improved spontaneously. Polycythemia is a rare complication of seminoma and only two cases have been reported previously. The precise mechanism of polycythemia in our patient could not be clearly evaluated, but clinical course did indicate a close relationship between two distinct disorders.     

肺部炎性假瘤的多层螺旋CT特征性表现的诊断价值

目的探讨肺部炎性假瘤（IPL）的多层螺旋CT（MSCT）特征性表现的诊断价值。方法回顾性分析76例IPL MSCT表现。结果MSCT首次检查即明确诊断正确者15例（19．7％）,其余被提示为肺癌诊断者43例（56．6％）,结核球19例（25．0％）,良性病变12例（15．6％）,错构瘤2例（2．6％）。分析76例肺部肺炎性假瘤MSCT表现,发现肿块位于肺的外周、局限性胸膜增厚粘连、病灶边缘多发性空泡征、病灶下缘散在结节征、桃尖征、平直征及均匀强化等征象,对肺炎性假瘤的MSCT诊断具有一定的价值。结论综合分析肺炎性假瘤特征性的MSCT表现,结合其肺内感染病史有利于提高肺部炎性假瘤的MSCT诊断与鉴别诊断。     

Inflammatory pseudotumor of the liver and peripheral eosinophilia in autoimmune pancreatitis

AIM: Inflammatory pseudotumor (IPT) of the liver is a rare benign lesion, the etiology of which remains obscure. It is not associated with any particular diseases apart from phlebitis and Crohn's disease. METHODS: A middle-aged male with hepatic IPT and peripheral eosinophilia associated with autoimmune pancreatitis (AIP) was selected for this study and review of literature. RESULTS: A 59-year-old male was admitted with obstructive jaundice, marked eosinophilia (1 343/mm3) and hypergammaglobulinemia (4 145 mg/dL). Imaging techniques revealed dilatation of the intrahepatic bile duct, stenosis of the common bile duct with diffuse wall thickening, gallbladder wall thickening, irregular narrowing of the pancreatic duct, and swelling of the pancreatic parenchyma. Multiple liver masses were also demonstrated and diagnosed as IPT by biopsy specimens. Six months later, the abnormal features of the biliary tree remarkably improved by the oral administration of prednisolone, and the liver masses disappeared. The swelling of the pancreatic head also improved. The peripheral eosinophil count normalized. IPT associated with AIP, as we know, has not been reported in the literature. The clinical features of the present case mimicked those of pancreatic cancer with liver metastasis. This case deserves to be documented to prevent misdiagnosis of similar cases.     

Refractory diarrhea: A paraneoplastic syndrome of neuroblastoma

Neuroblastoma(NB) is the most common extracranial solid tumor in children. Diarrheal NB is quite rare and is not easy to diagnose in the early stage. Six cases of diarrheal NB in our hospital treated from 1996 to 2006 were retrospectively analyzed, including characteristics such as electrolyte imbalance, pathologic features, vasoactive intestinal peptide(VIP) immunohistochemical staining results, treatment, and prognosis. All patients were boys with 3-8 loose or watery stools each day and routine fecal tests were normal. Abdominal tumors were identified by B-ultrasound. Drugs were ineffective. Three patients underwent surgery, and the remaining three patients received surgery and chemotherapy. Diarrhea stopped after treatment in five patients. Two patients died due to intractable hypokalemia. The tumor was located in the adrenal gland in four patients, in the upper retroperitoneum in one patient, and in the presacral area in one patient. Pathologic findings were NB and ganglioneuroblastoma. Five patients were at clinical stage Ⅰ-Ⅱ, and one was at stage Ⅲ. Four patients survived(followed-up for 6 mo to 4 years). Immunohistochemical staining for VIP was positive. Refractory diarrhea is a paraneoplastic syndrome of NB and is rare. Patients aged 1-3 years who present with chronic intractable diarrhea should be followed closely. Intractable diarrhea, hypokalemia, and dysplasia are the initial clinical manifestations. Increased VIP is characteristic of this disease. Potassium supplementation plays a vital role in the treatment procedure, especially preoperatively. The prognosis of diarrheal NB is good following appropriate treatment.     

Splenic inflammatory pseudotumor mimicking angiosarcoma

Splenic tumors are rare. Differentiation of the tumors before operation is of great value regarding the outcome. A case of a 32-year-old man with a splenic inflammatory pseudotumor （IPT） mimicking splenic angiosarcoma is described. The tumor was highly suspected of being splenic angiosarcoma based on radiological findings preoperatively. However, after splenectomy, histopathological examinations revealed splenic IPT. Splenic IPT and angiosarcoma are rare and often pose diagnostic difficulties because the clinical and radiological findings are obscure. Due to large differences in prognosis, we briefly reviewed the clinical, radiological, and pathological features of both of the tumors.     

原发性肝癌伴癌综合征的临床分析

目的分析原发性肝癌伴癌综合征（PNS）的临床特征,探讨PNS患者生化及病理参数的改变规律。方法对病理或影像学检查确诊的635例原发性肝癌患者资料进行回顾性分析,比较PNS组与非PNS组临床特征、生化及病理参数等指标方面的差异。结果PNS占同期原发性肝癌住院人数的20．0％（127／635）,男女之比为8．77：1。PNS主要表现为6种异常：血小板增多症（占同期原发性肝癌住院人数的6．14％,下同）,高胆固醇血症（5．83％）,白细胞增多症（5．20％）,红细胞增多症（1．10％）,高钙血症（0．94％）,低血糖症（0．79％）。原发性肝癌PNS组与非PNS组相比,在平均AFP水平、肿瘤负荷、肝炎病毒感染率等方面差异有统计学意义（P〈0．05）。结论原发性肝癌PNS患者平均AFP水平、肿瘤负荷和肝炎病毒感染率较高,其临床表现无特异性,容易漏诊和误诊。     

Hilar inflammatory pseudotumor mimicking hilar cholangiocarcinoma

BACKGROUND:Inflammatory pseudotumor of the biliary tract is a benign disease,and is extremely rare.Its diagnosis often depends on pathological examination after operation. The histopathological examination shows inflammatory lesions with a polymorphous infiltration and variable amounts of fibrous tissue.This study was undertaken to elucidate that an inflammatory pseudotumor in the right hepatic duct is especially difficult to distinguish from hilar cholangiocarcinoma. METHOD:The clinical data of one patient...     

Recurrence of inflammatory pseudotumor in the distal bile duct： Lessons learned from a single case and reported cases

Inflammatory myofibroblastic tumors (IMTs) or inflammatory pseudotumors (IPs) have been extensively discussed in the literature. They are usually found in the lung and upper respiratory tract. However, reporting of cases involving the biliopancreatic region has increased over recent years. Immunohistochemical study of these lesions limited to the pancreatic head or distal bile duct seems to be compatible with those observed in a new entity called autoimmune pancreatitis, but usually intense fibrotic reaction (zonation) predominates producing a mass. When this condition is limited to the pancreatic head, the common bile duct might be involved by the inflammatory process and jaundice may occur often resembling adenocarcinoma of the pancreas. We have previously reported a case of IMT arising from the bile duct associated with autoimmune pancreatitis which is an extremely rare entity. Four years after Kaush-Whipple resection, radiological examination on routine follow-up revealed a tumor mass, suggesting local recurrence. Ultrasound-guided FNA confirmed our suspicious diagnosis. This present case, as others, suggests that persistent follow-up is necessary in order to prevent irreversible liver damage at this specific location.     

基于增强MRI纹理分析鉴别诊断眼眶淋巴瘤和炎性假瘤

目的探讨增强MRI序列纹理分析在鉴别眼眶淋巴瘤和炎性假瘤中的诊断价值。 方法回顾性分析2013年1月至2019年4月于苏北人民医院经病理证实的眼眶淋巴瘤25例和炎性假瘤24例，术前进行MRI常规序列和脂肪抑制T1WI增强扫描，选取每例患者增强图像上肿瘤的最大层面区域，应用Mazda软件勾画肿瘤感兴趣区并计算灰度直方图纹理参数。提取出9个特征参数分别为：均值(mean)、方差(variance)、偏度(skewness)、峰度(kurtosis)和第1、10、50、90、99百分位数(Pere.1%、Pere.10%、Pere.50%、Pere.90%、Pere.99%)，分别比较淋巴瘤和炎性假瘤各个纹理参数之间的差异，其中符合正态分布的参数采用双样本t检验，不符合正态分布的参数采用秩和检验，并采用受试者工作特征曲线(receiver operaling characteristic curve，ROC)分析曲线下面积(area under curve，AUC)、敏感度、特异度。运用多变量Logistic回归分析对有统计学意义的纹理参数进行建模并绘制ROC曲线评价模型效能。 结果2种肿瘤在均值、方差、第50、90、99个百分位数参数中差异有统计学意义(P<0.05)。在偏度、峰度和第1百分位数参数中差异无统计学意义(P>0.05)。2组间纹理参数方差在阈值为113.20时具有最佳诊断效能，对应的敏感度、特异度、AUC分别是95.83%、76.00%、0.930(54.87%～90.64%)。 结论增强MRI序列纹理分析可以用于眼眶淋巴瘤和炎性假瘤的鉴别诊断，且参数方差具有最佳诊断效能。     

Inflammatory pseudotumor of the colon causing intussusception: A case report and literature review

Inflammatory pseudotumor(IPT) is a rare spaceoccupying lesion of unknown etiology that can mimic malignancy on clinic-radiological and pathological examination. We describe a rare case of ileocecal intussusception from clinically suspected malignancy of the right colon where the patient underwent righthemicolectomy. Histopathology of the resected specimen confirmed IPT of the colon. This patient was observed to have abnormally elevated total leukocyte count and platelets before and after surgery. In an adult with intussusception associated with an abdominal mass, the possibility of IPT of the colon should be considered. Considering the abnormally high total leukocyte and platelet counts and colonic IPT, it is necessary to prevent postoperative adverse effects due to these changes. Although IPT of the colon is usually a benign process, controversy regarding its management still exists. We consider hemicolectomy as a safe treatment approach for colonic IPT and review the existing literature.     

Concomitant inflammatory pseudotumor of the liver and spleen

Abstract: We report the case of a 53‐year‐old man with inflammatory pseudotumor (IPT) of the liver and spleen. This concomitant association has rarely been reported. The patient presented with a hypoechoic mass in the liver and a clinical picture of recurrent sepsis; hematochemical exams and imaging data were nonspecific. Antibiotic therapy improved the clinical course, but did not resolve it definitively. After 50 days of therapy, as the hepatic mass decreased a similar lesion appeared in the spleen. The final diagnosis was made on splenectomy and an intra‐operative biopsy of the residual liver lesion. The diagnostic problems encountered in this very rare association of IPT of the liver and spleen were similar to those for isolated IPT in the respective single organ sites. After 15 months of follow‐up, the patient is in good health and no recurrence of symptoms or masses has been observed.     

肺炎性假瘤的诊断与手术治疗

目的探讨肺炎性假瘤的诊断与手术治疗。方法回顾52例肺炎性假瘤的检查和手术进行分析。结果1例术后并发支气管胸膜瘘死亡外,余均治愈。结论手术是治疗肺炎性假瘤的有效方法。     

Regression of inflammatory pseudotumor of the liver under conservative therapy

Summary Inflammatory pseudotumors (IPT) of the liver are rare benign tumors of unknown origin. Most previously reported cases were initially misdiagnosed and treated surgically; thus, their course under medical treatment remains unclear. We report an additional case of hepatic IPT with a favorable outcome under medical treatment without surgical resection. New imaging and pathologic features of this disease are presented that may facilitate preoperative recognition and appropriate treatment.     

脂肪肝对超声造影诊断肝内炎性假瘤的影响

目的 研究脂肪肝对超声造影诊断肝脏炎性假瘤(IPL)的影响。方法 2016年1月～2021年1月华北理工大学附属开滦总医院诊治的IPL患者28例,其中伴有脂肪肝者12例,正常肝内IPL患者16例。所有患者均接受超声造影检查,比较两组血供状态、超声造影增强模式和超声造影定量参数的异同。结果 伴脂肪肝的IPL病灶动脉供血1例(8.3%)、静脉供血11例(91.7%)、富血供3例(25.0%)、乏血供9例(75.0%),IPL病灶动脉供血7例(43.8%)、静脉供血9例(56.3%)、富血供5例(31.3%)、乏血供11例(68.8%),两组供血类型存在显著性差异(P<0.05),而血供丰富程度无显著性差异(P>0.05);伴脂肪肝病灶呈无明显增强型1例、快速廓清型5例、等增强型3例和低增强型3例,IPL病灶呈无明显增强型2例、快速廓清型5例、等增强型7例和低增强型2例,两组超声造影增强模式无显著差异(P=0.661);伴脂肪肝病灶造影剂到达时间为(8.9±1.3)s,显著慢于IPL病灶【((8.0±0.9)s,P<0.05】,峰值强度为(53.2±7.8)dB,显著弱于I...     

Inflammatory pseudotumor of the liver and spleen diagnosed by percutaneous needle biopsy

An inflammatory pseudotumor (IPT) is a relatively rare lesion characterized by chronic infiltration of inflammatory cells and areas of fibrosis. IPTs are difficult to diagnose because of the absence of specific symptoms or of characteristic hematological or radiological findings. In this study, a case of a woman aged over 70 years was reported, who presented with a general malaise lasting more than two months. A computed tomography scan demonstrated a diffusely spread lesion of the liver with a portal vein occlusion and a splenic lesion surrounded by a soft density layer. Since the percutaneous liver biopsy showed findings that suggested an IPT, although the radiological findings did not exclude the possibility of a malignancy, we performed a percutaneous spleen biopsy to enable a more definitive diagnosis. The microscopic findings from the spleen specimen lead us to a diagnosis of IPT involving the liver and spleen. Subsequent steroid pulse therapy was effective, and rapid resolution of the disease was observed.