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The treatment of complicated anterior cerebral artery aneurysms remains challenging. Here, the authors describe a case of ruptured complicated A3 aneurysm, which was treated with trapping and in-situ bypass. A 47-year-old man presented to the emergency department with severe headache and vomiting. Computed tomography illustrated acute intracerebral hemorrhage in the right frontal lobe. Digital subtraction angiography (DSA) confirmed a ruptured fusiform A3 aneurysm with lobulation and a daughter sac. Trapping of the ruptured fusiform A3 aneurysm and distal end-toside A4 anastomosis was performed. DSA on postoperative day 7 showed mild vasospasm to the afferent artery. However, 2 months later, DSA demonstrated that the antegrade flow through the anastomosis site had recovered. Thus, surgeons should be aware of the possibility of postsurgical vasospasm of anastomosed arteries, especially in cases of ruptured aneurysms.
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Acute pancreatitis can range from a mild, self-limiting disease requiring no more than supportive care, to severe disease with life-threatening complications. With the goal of providing a recommendation framework for clinicians to manage acute pancreatitis, and to contribute to improvements in national health care, the Korean Pancreatobiliary Association (KPBA) established the Korean guidelines for acute pancreatitis management in 2013. However, many challenging issues exist which often lead to differences in clinical practices. In addition, with newly obtained evidence regarding acute pancreatitis, there have been great changes in recent knowledge and information regarding this disorder. Therefore, the KPBA committee underwent an extensive revision of the guidelines. The revised guidelines were developed using the Delphi method, and the main topics of the guidelines include the following: diagnosis, severity assessment, initial treatment, nutritional support, convalescent treatment, and the treatment of local complications and necrotizing pancreatitis. Specific recommendations are presented, along with the evidence levels and recommendation grades.
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Background/Aims@#A previous history of post-endoscopic retrograde cholangiopancreatography (ERCP) pancreatitis (PEP) is a risk factor for PEP, suggesting that there may be a genetic predisposition to PEP. However, nothing is known about this yet. The aim of this study was to identify genetic variations associated with PEP. @*Methods@#A cohort of high-risk PEP patients was queried from December 2016 to January 2019. For each PEP case, two propensity score-matched controls were selected. Whole exome sequencing was performed using blood samples. Genetic variants reported to be related to pancreatitis were identified. To discover genetic variants that predispose to PEP, a logistic regression analysis with clinical adjustment was performed. Gene-wise analyses were also conducted. @*Results@#Totals of 25 PEP patients and 50 matched controls were enrolled. Among the genetic variants reported to be associated with pancreatitis, only CASR rs1042636 was identified, and it showed no significant difference between the case and control groups. A total of 54,269 non-synonymous variants from 14,313 genes was identified. Logistic regression analysis of these variants showed that the IRF2BP1 rs60158447 GC genotype was significantly associated with the occurrence of PEP (odds ratio 2.248, FDR q value = 0.005). Gene-wise analyses did not show any significant results. @*Conclusions@#This study found that the IRF2BP1 gene variant was significantly associated with PEP. This genetic variant is a highly targeted PEP risk factor candidate and can be used for screening high-risk PEP groups before ERCP through future validation. (ClinicalTrials.gov no. NCT02928718)
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Objective@#: This study compared the quality of recovery (QoR) after minicraniotomy for clipping of unruptured intracranial aneurysms (UIAs) between patients with and without scalp nerve block (SNB). @*Methods@#: Patients were randomly assigned to the SNB (SNB using ropivacaine with epinephrine, n=27) and control (SNB using normal saline, n=25) groups. SNB was performed at the end of surgery. To assess postoperative QoR, the QoR-40, a patient-reported questionnaire, was used. The QoR-40 scores were measured preoperatively, 1–3 days postoperatively, at hospital discharge, and 1 month postoperatively. Pain and intravenous patient-controlled analgesia (IV-PCA) consumption were evaluated 3, 6, 9, and 12 hours and 1–3 days postoperatively. @*Results@#: All QoR-40 scores, including those measured 1 day postoperatively (primary outcome measure; 155.0 [141.0–176.0] vs. 161.0 [140.5–179.5], p=0.464), did not significantly differ between the SNB and control groups. The SNB group had significantly less severe pain 3 (numeric rating scale [NRS]; 3.0 [2.0–4.0] vs. 5.0 [3.5–5.5], p=0.029), 9 (NRS; 3.0 [2.0–4.0] vs. 4.0 [3.0–5.0], p=0.048), and 12 (NRS; 3.0 [2.0–4.0] vs. 4.0 [3.0–5.0], p=0.035) hours postoperatively. The total amount of IV-PCA consumed was significantly less 3 hours postoperatively in the SNB group (2.0 [1.0–4.0] vs. 4.0 [2.0–5.0] mL, p=0.044). @*Conclusion@#: After minicraniotomy for clipping of UIAs, SNB reduced pain and IV-PCA consumption in the early postoperative period but did not improve the QoR-40 scores.
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Objective@#Several particular morphological factors that contribute to the hemodynamics of the anterior communicating artery (ACoA) have been documented, but no study has investigated the role of the degree of anterior cerebral artery (ACA) rotation on the presence of ACoA aneurysms (ACoAAs). @*Methods@#A retrospective study of an institutional aneurysm database was performed; patients with ruptured or nonruptured ACoAAs were selected. Two sex- and age-matched control groups were identified: control Group A (nonaneurysms) and control Group B (middle cerebral artery aneurysms). Measurements of ACA rotation degree were obtained by using a three-dimensional imaging tool. @*Results@#From 2015 to 2020, 315 patients were identified: 105 in the ACoAA group, 105 in control Group A, and 105 in control Group B. The average age at the time of presentation was 64 years, and 52.4% were female. The ACA rotation degree of the ACoAA group was significantly higher than that of control Group A (p <0.01). The A1 ratio and the A1A2 ratio of the ACoAA group were greater than those of control Group A (p <0.01 and p <0.01, respectively). The ACA rotation degree correlated insignificantly with aneurysm size in ACoAA patients (p=0.78). The ACA rotation degree in the ACoAA group was also insignificantly different from that in control B (p=0.11). @*Conclusions@#The degree of ACA rotation was greater in the ACoAA group than in the nonaneurysm group, and it may serve as an imaging marker for ACoAA.
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In severe acute pancreatitis, accompanied by local complications such as acute peripancreatic fluid collection, pancreatic pseudocyst, acute necrotic collection and walled-off necrosis, the mortality rate is as high as 12-25%. In many cases, interventional procedure or surgical treatment are required at an appropriate time. Conservative treatment is considered for acute peripancreatic fluid collection. Endoscopic drainage could be considered preferentially for the treatment of pancreatic pseudocysts with clinical symptoms or complications. In the case of necrotizing pancreatitis, conservative treatment is preferred, but therapeutic intervention should be considered if infectious pancreatic necrosis with clinical deterioration is suspected. For therapeutic intervention, it is recommended to proceed with a step-up approach in which drainage is first performed and, if necessary, necrosectomy is performed. The optimal timing of intervention is considered 4 weeks after the onset of pancreatitis when necrosis become walled-off, but early drainage within 4 weeks can be considered depending on the patient's condition. This guideline provides an overview of current treatment strategies for local complications of acute pancreatitis.
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Background/Aims@#There has been growing evidence on the utility of neoadjuvant FOLFIRINOX in borderline resectable (BR) or locally advanced (LA) pancreatic cancer. However, factors predicting survival in these patients remain to be identified, and we aimed to identify these prognostic factors. @*Methods@#Between January 2013 and April 2017, patients with BR or LA pancreatic cancer who received FOLFIRINOX as their initial treatment were identified. Demographic data and clinical outcomes, including the chemotherapy response, conversion to resection, and survival, were reviewed. @*Results@#A total of 117 patients with BR (n=39) or LA (n=78) pancreatic cancer were included. Of these patients, 29 (24.8%) underwent curative surgery, and R0 resection was achieved in 21 patients (72.4%). The median progression-free survival and overall survival time of all patients were 11.6 and 19.0 months, respectively. In resected patients, the median relapse-free survival and overall survival times were 14.8 and 28.6 months, respectively. In the multivariate Cox model, the lowest level of serum carbohydrate antigen 19-9 (CA 19-9) and resection after FOLFIRINOX were independent factors for improved overall survival. In the subgroup analysis of patients with initial 18 F-fluorodeoxyglucose-positron emission tomography (FDG-PET) images, the maximum standardized uptake value (SUVmax) of the pancreatic mass was also shown as an independent factor for improved overall survival. @*Conclusions@#In patients with BR or LA pancreatic cancer, FOLFIRINOX is a valuable neoadjuvant treatment that enables curative surgery in approximately one-quarter of patients and significantly improves overall survival. In these patients, the prognosis can be estimated using the lowest level of serum CA 19-9, operative status, and initial FDG-PET SUVmax.
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Background/Aims@#Treatment of residual intraductal lesions after endoscopic papillectomy for ampullary adenomas is relatively difficult. Few studies have been conducted using intraductal radiofrequency ablation (RFA) in the treatment of such lesions, and no study has aimed to reduce the side effects of excessive heat caused by RFA. Recently, a temperature-controlled RFA probe was developed to avoid excessive heat. This study aimed to investigate the safety of this new RFA probe in the treatment of intraductal lesions of ampullary adenoma. @*Methods@#Patients who received RFA for residual intraductal lesions after endoscopic papillectomy between November 2017 and June 2019 were retrospectively reviewed. A novel temperature-controlled probe (ELRA) was used for intraductal RFA, and clinical data including adverse events were collected. @*Results@#Ten patients were included in this study. Intraductal adenomas showed low-grade dysplasia in eight patients and high-grade dysplasia in two patients. The median diameter of intraductal adenomas was 9 mm (range, 5 to 10 mm) in the common bile duct and 5 mm (range, 4 to 11 mm) in the pancreatic duct. Adverse events occurred in three patients (30.0%), of which two were mild pancreatitis and one was asymptomatic biliary stricture. Over a median follow-up period of 253 days, only one patient underwent additional surgery, as the remainder showed no adenomatous lesions on follow-up biopsies. @*Conclusions@#The new temperature-controlled RFA probe can be used with acceptable safety for the treatment of residual intraductal lesions after endoscopic papillectomy. Further evaluation through future prospective studies is needed.
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BACKGROUND/AIMS: Recurrent pyogenic cholangitis (RPC) is a chronic progressive disease frequently accompanied by cholangiocarcinoma (CCA). This study aimed to investigate the natural course of RPC and identify factors associated with CCA. METHODS: From January 2005 to December 2016, 310 patients diagnosed with RPC at Seoul National University Hospital were included. Complications and management during follow-up were recorded. CCA-free probability was estimated by Kaplan-Meier method, and risk factors associated with CCA were analyzed using log-rank test and Cox’s proportional hazard regression model. RESULTS: Mean age at diagnosis was 59.1±10.9 years and mean follow-up duration was 84.0±64.1 months. An intrahepatic duct stone was found in 253 patients (81.6%). Liver atrophy was identified in 185 patients (59.7%) and most commonly located at the left lobe (65.4%). Acute cholangitis, liver abscesses, cirrhotic complications, and CCA developed in 41.3%, 19.4%, 9.7%, and 7.4%, respectively. During follow-up, complete resolution rate after hepatectomy, biliary bypass surgery, and choledocholithotomy with T-tube insertion reached 82.3%, 55.2%, and 42.1%, respectively. None of the patients who maintained complete resolution by the last follow-up day developed CCA. In univariate analysis, female, both-sided intrahepatic duct stones, and liver atrophy at any location were associated with increased risk of CCA. Multivariate analysis revealed that both-sided atrophy significantly increased risk of CCA (hazard ratio, 4.56; 95% confidence interval, 1.48 to 14.09; p=0.008). In 21 patients who developed intrahepatic CCA, tumor was located mostly in the atrophied lobe (p=0.023). CONCLUSIONS: In RPC patients, acute cholangitis, liver abscess, cirrhotic complications, and CCA frequently developed. Both-sided liver atrophy was a significant risk factor for developing CCA.
Subject(s)
Female , Humans , Atrophy , Cholangiocarcinoma , Cholangitis , Cohort Studies , Diagnosis , Fibrosis , Follow-Up Studies , Hepatectomy , Liver , Liver Abscess , Methods , Multivariate Analysis , Prognosis , Retrospective Studies , Risk Factors , SeoulABSTRACT
BACKGROUND: To evaluate the efficacy of modified ventriculolumbar perfusion (VLP) chemotherapy with methotrexate on leptomeningeal carcinomatosis in terms of symptomatic response and side effects. METHODS: Previous infusion rate of 20 mL/h was reduced to 15 mL/h for the purpose of decreasing constitutional side effects of VLP such as nausea/vomiting, insomnia and confusion. The primary outcome was the response rate of increased intracranial pressure (ICP), and the secondary outcome was the occurrence of side effects compared to previous 20 mL/h trial. This interim analysis to validate the reduced infusion rate is not to affect the original effect of VLP chemotherapy. RESULTS: All forty-seven patients were enrolled including 22 patients with increased ICP. Thirteen patients out of these (59%) got normalized ICP after VLP chemotherapy. Moderate to severe (grade 2–3) confusion was observed in 3 patients (6%) and it was significantly reduced compared to those (23%) in the VLP 20 mL/h (p=0.017). Grade 2–3 nausea/vomiting was also reduced from 64% to 45% but failed to reach statistical significance (p=0.08). Median overall survival (OS) was 5.3 months (95% confidence interval, 3.55–7.05) and patients OS, who received maintenance VLP was significantly prolonged compared to patients who underwent induction VLP only (5.8 vs. 3.4 months, p=0.025). CONCLUSION: VLP of reduced perfusion rate (15 mL/h) showed compatible control rate of increased ICP at this interim analysis. Decreased moderate to severe side effects and prolonged OS in patients received maintenance VLP encourage us to evaluate the effectiveness of this trial further.
Subject(s)
Humans , Drug Therapy , Infusions, Intraventricular , Intracranial Pressure , Meningeal Carcinomatosis , Methotrexate , Perfusion , Sleep Initiation and Maintenance DisordersABSTRACT
In acute biliary pancreatitis, endoscopic retrograde cholangiopancreatography (ERCP) and cholecystectomy should be considered to reduce the complications of gallstones including recurrent biliary pancreatitis. If biliary pancreatitis is accompanied by cholangitis or evidence of obvious biliary obstruction, removal of the common bile duct stone via early ERCP (within 24 to 72 hours) is necessary. Less or non-invasive imaging modalities such as endoscopic ultrasound, magnetic resonance cholangiopancreatography can be considered to avoid unnecessary ERCP if suspected biliary obstruction in the absence of cholangitis in patients with biliary pancreatitis. Cholecystectomy in patients with biliary pancreatitis requires a strategy that varies the timing of surgery depending on the severity of pancreatitis. In mild acute biliary pancreatitis, cholecystectomy can be performed safely at the time of initial admission. In moderate to severe biliary pancreatitis, cholecystectomy should be delayed until about 6 weeks when active inflammation subsides and fluid collections resolve or stabilize. Endoscopic sphincterotomy (EST) can be helpful in reducing recurrent pancreatitis in patients who unfit for cholecystectomy. However, even if EST is performed, additional cholecystectomy will further reduce the risk of recurrent pancreatitis, if possible, it is recommended to undergo a cholecystectomy.
Subject(s)
Humans , Cholangiopancreatography, Endoscopic Retrograde , Cholangiopancreatography, Magnetic Resonance , Cholangitis , Cholecystectomy , Common Bile Duct , Gallstones , Inflammation , Pancreatitis , Sphincterotomy, Endoscopic , UltrasonographyABSTRACT
BACKGROUND/AIMS: The combination of nab-paclitaxel and gemcitabine (nab-P/Gem) is widely used for treating meta-static pancreatic cancer (MPC). We aimed to evaluate the therapeutic outcomes and prognostic role of treatment-related peripheral neuropathy in patients with MPC treated with nab-P/Gem in clinical practice. METHODS: MPC patients treated with nab-P/Gem as the first-line chemotherapy were included. All 88 Korean patients underwent at least two cycles of nab-P/Gem combination chemotherapy (125 and 1,000 mg/m2, respectively). Treatment-related adverse events were monitored through periodic follow-ups. Overall survival and progression-free survival were estimated by the Kaplan Meier method, and the Cox proportional hazards regression linear model was applied to assess prognostic factors. To evaluate the prognostic value of treatment-related peripheral neuropathy, the landmark point analysis was used. RESULTS: Patients underwent a mean of 6.7±4.2 cycles during 6.3±4.4 months. The median overall survival and progression-free survival rates were 14.2 months (95% confidence interval [CI], 11.8 to 20.3 months) and 8.4 months (95% CI, 7.1 to 13.2 months), respectively. The disease control rate was 84.1%; a partial response and stable disease were achieved in 30 (34.1%) and 44 (50.0%) patients, respectively. Treatment-related peripheral neuropathy developed in 52 patients (59.1%), and 13 (14.8%) and 16 (18.2%) patients experienced grades 2 and 3 neuropathy, respectively. In the landmark model, at 6 months, treatment-related peripheral neuropathy did not have a significant correlation with survival (p=0.089). CONCLUSIONS: Nab-P/Gem is a reasonable choice for treating MPC, as it shows a considerable disease control rate while the treatment-related peripheral neuropathy was tolerable. The prognostic role of treatment-related neuropathy was limited.
Subject(s)
Humans , Disease-Free Survival , Drug Therapy , Drug Therapy, Combination , Follow-Up Studies , Linear Models , Methods , Neoplasm Metastasis , Pancreatic Neoplasms , Peripheral Nervous System DiseasesABSTRACT
Gallbladder cancer is the most common malignancy of the biliary tract and carries very poor prognosis. Surgery is an only curative modality of treatment in the gallbladder cancer. However, as most of the gallbladder cancers are diagnosed at advanced stages, surgery can be attempted in a very limited number of patients. In advanced stage, treatment option is confined to a palliative systemic chemotherapy, and biliary decompression is needed when cholangitis is suspected. We report a case of 49-year-old patient with metastatic gallbladder cancer treated with successful curative resection after several courses of palliative chemotherapy and biliary decompression.
Subject(s)
Humans , Middle Aged , Antineoplastic Agents , Biliary Tract , Cholangitis , Cholecystectomy , Decompression , Drainage , Drug Therapy , Gallbladder Neoplasms , Gallbladder , Neoplasm Metastasis , PrognosisABSTRACT
Although Dieulafoy lesion can occur in any part of the gastrointestinal tract, its occurrence in the rectum is rare. Rectal Dieulafoy lesions have been associated with advanced age, renal failure, burns, liver transplantation and cirrhosis. Here, we report on a case of massive bleeding from a rectal Dieulafoy lesion after lung decortication surgery in a 57-year-old male patient with alcoholic cirrhosis. Although rare, a rectal Dieulafoy lesion should be included in the differential diagnosis of massive lower gastrointestinal bleeding in a patient with cirrhosis.
Subject(s)
Humans , Male , Middle Aged , Alcoholics , Burns , Diagnosis, Differential , Fibrosis , Gastrointestinal Tract , Hemorrhage , Liver Cirrhosis , Liver Cirrhosis, Alcoholic , Liver Transplantation , Lung , Rectum , Renal InsufficiencyABSTRACT
Although Dieulafoy lesion can occur in any part of the gastrointestinal tract, its occurrence in the rectum is rare. Rectal Dieulafoy lesions have been associated with advanced age, renal failure, burns, liver transplantation and cirrhosis. Here, we report on a case of massive bleeding from a rectal Dieulafoy lesion after lung decortication surgery in a 57-year-old male patient with alcoholic cirrhosis. Although rare, a rectal Dieulafoy lesion should be included in the differential diagnosis of massive lower gastrointestinal bleeding in a patient with cirrhosis.
Subject(s)
Humans , Male , Middle Aged , Alcoholics , Burns , Diagnosis, Differential , Fibrosis , Gastrointestinal Tract , Hemorrhage , Liver Cirrhosis , Liver Cirrhosis, Alcoholic , Liver Transplantation , Lung , Rectum , Renal InsufficiencyABSTRACT
OBJECTIVE: The purpose of this study is to describe the detailed surgical technique and short-term clinical and radiological outcomes of lateral lumbar interbody fusion (LLIF) and in situ lateral screw fixation using a conventional minimally invasive screw fixation system (MISF) for revision surgery to treat rostral lumbar adjacent segment disease. METHODS: The medical and radiological records were retrospectively reviewed. The surgery was indicated in 10 consecutive patients with rostral adjacent segment stenosis and instability. After the insertion of the interbody cage, lateral screws were inserted into the cranial and caudal vertebra using the MISF through the same LLIF trajectory. The radiological and clinical outcomes were assessed preoperatively and at 1, 3, 6, and 12 months postoperatively. RESULTS: The median follow-up period was 13 months (range, 3–48 months). Transient sensory changes in the left anterior thigh occurred in 3 patients, and 1 patient experienced subjective weakness; however, these symptoms normalized within 1 week. Back and leg pain were significantly improved (p<0.05). In the radiological analysis, both the segmental angle at the operated segment and anterior disc height were significantly increased. At 6 months postoperatively, solid bony fusion was confirmed in 7 patients. Subsidence and mechanical failure did not occur in any patients. CONCLUSION: This study demonstrates that LLIF and in situ lateral screw fixation may be an alternative surgical option for rostral lumbar adjacent segment disease.
Subject(s)
Humans , Constriction, Pathologic , Follow-Up Studies , Leg , Minimally Invasive Surgical Procedures , Reoperation , Retrospective Studies , Spinal Fusion , Spinal Stenosis , Spine , ThighABSTRACT
Rhabdomyolysis results from acute damage to skeletal muscles caused by various conditions, of which hypokalemia is a recognized but rare example. Although primary aldosteronism may cause severe hypokalemia leading to rhabdomyolysis, the potassium level of such patients can be within the normal range. Hypokalemia is most frequently triggered when these patients are exposed to an additional insult, such as diuretic stress. Similarly, overzealous consumption of ionic beverages with osmotic diuretic effects can cause hypokalemia. Here, we describe a patient with an aldosterone-secreting adrenal adenoma, who presented with acute rhabdomyolysis secondary to severe hypokalemia triggered by consumption of a large volume of ionic beverage for 3 weeks.
Subject(s)
Humans , Adenoma , Adrenocortical Adenoma , Beverages , Diuresis , Diuretics , Hyperaldosteronism , Hypokalemia , Muscle, Skeletal , Potassium , Reference Values , RhabdomyolysisABSTRACT
BACKGROUND: Prevalence of adenoma in males aged 40-49 years in Korea was higher than expected. The aim of this study was to investigate the prevalence and risk factors of colorectal adenoma in males aged 40-49 years. METHODS: Total 1,902 asymptomatic subjects with a mean age of 47.9±6.7 years, who underwent a screening colonoscopy in a health promotion center of Myongji Hospital from 2010 to 2013 were enrolled in this study. We conducted a case-control study to determine the risk factors for adenoma. The subjects were classified into two groups (adenoma vs. controls). To validate the diagnostic value of carcinoembryonic antigen (CEA) for adenoma, area under the receiver operating characteristic curve (AUROC) was calculated. RESULTS: At least one colorectal adenoma was identified in 385 subjects (20.2%). Among these 385 subjects, 372 subjects were found to have a non-advanced adenoma, 13 subjects had an invasive adenoma. One subject had cancer. Male sex, age, smoking, metabolic syndrome, and elevated CEA level were significantly associated with a colorectal adenoma in univariate analysis. However, metabolic syndrome was not significant in multivariate analysis. In the male group, the AUROC of CEA for colorectal adenoma was 0.600 (0.543 to 0.656) in non-smokers under 50 years of age, and 0.615 (0.540 to 0.690) in smokers under 50 years of age. CONCLUSION: Male sex, smoking, and high levels of CEA seem to be associated with colorectal adenoma. High levels of CEA and smoking may be diagnostic markers for any colorectal adenoma in Korean males aged 40-49 years.
Subject(s)
Humans , Male , Adenoma , Carcinoembryonic Antigen , Case-Control Studies , Colonoscopy , Health Promotion , Korea , Mass Screening , Multivariate Analysis , Prevalence , Risk Factors , ROC Curve , Smoke , SmokingABSTRACT
A variety of diseases are associated with the development of unilateral pleural effusion. Although unilateral pleural effusion is common, refractory unilateral pleural effusion is rare. It is important to make an accurate diagnosis using proper diagnostic tools. Thrombocytopenia is one of the rare conditions occurring from various diseases such as severe infection or autoimmune diseases. It can be life-threatening if accurate diagnosis and treatment are delayed and be a clue to accurate diagnosis in differential diagnosis from refractory pleural effusion. Kasabach-Merrit syndrome (KMS) is often accompanied by extensive vascular tumors and characterized by consumptive coagulopathy with profound thrombocytopenia. It is also important to have a high index of suspicion for the diagnosis. We report a case of KMS in a 2-month-old female infant with a vascular tumor on her left intrathoracic cage, who had presented refractory unilateral pleural effusion and thrombocytopenia. Initially, the patient was diagnosed as having complications of severe infection, and a chest tube was inserted for aggressive treatment. However, her unilateral pleural effusion persisted, and thrombocytopenia and hypofibrinogenemia were refractory. Chest imaging revealed an infiltrating large vascular tumor involving the cardiac border, diaphragm, and chest wall. The patient’s unilateral pleural effusion was misidentified as an infectious condition at the initial stage. As a result of the ultrasonography-guided biopsy, it was revealed to be Kaposiform hemangioendothelioma. The patient was cured after treatment for KMS.