BACKGROUND Primary splenic lesions are rare and often detected incidentally through imaging,biopsy,or autopsy,typically without distinct clinical symptoms.Although imaging can help differentiate benign from malignant ...BACKGROUND Primary splenic lesions are rare and often detected incidentally through imaging,biopsy,or autopsy,typically without distinct clinical symptoms.Although imaging can help differentiate benign from malignant lesions,splenic hamartomas,and angiosarcomas may exhibit overlapping features,making diagnosis challenging.This report presents a case of splenic hamartoma suspected to be an angiosarcoma based on preoperative imaging.Splenic hamartomas that mimic angiosarcomas are exceedingly rare.CASE SUMMARY A 33-year-old male presented to the Department of Emergency with frank red blood hematemesis and a 1-week history of epigastric pain.On arrival,he was alert and hemodynamically stable.Contrast-enhanced abdominal computed tomography revealed splenomegaly with significant engorgement of the portal and splenic veins,along with a diffuse nodular splenic lesion measuring 8.2 cm×6.2 cm.Following esophageal varix ligation,abdominal magnetic resonance imaging demonstrated iso-to high-signal intensity within the splenic mass and multiple hypervascular lesions in the right hepatic lobe,raising suspicion for splenic an-giosarcoma with hepatic metastases.18F-fluorodeoxyglucose positron emission tomography-computed tomography showed diffusely mild increased metabolic activity in the spleen.The patient subsequently underwent splenectomy and liver biopsy.Histopathological examination revealed chronic inflammation in the liver,and the splenic lesion was confirmed to be a splenic hamartoma.The patient successfully returned to work and remains in good health.CONCLUSION This rare case of splenic hamartoma mimicking angiosarcoma highlights the importance of differential diagnosis in managing splenic tumors.展开更多
BACKGROUND Breast hamartomas are rare benign breast tumors,with an incidence rate of 0.8%-4.8%.Further,the coexistence of hamartomas and carcinoma is also uncommon.Our case report presents a unique instance where inva...BACKGROUND Breast hamartomas are rare benign breast tumors,with an incidence rate of 0.8%-4.8%.Further,the coexistence of hamartomas and carcinoma is also uncommon.Our case report presents a unique instance where invasive ductal carcinoma(IDC)and ductal carcinoma in situ were found both inside and outside a breast hamartoma.This is the second case reported in the literature.CASE SUMMARY A 51-year-old woman presented with a 6.0 cm breast tumor on mammography and ultrasound,with suspicious areas indicative of malignant transformation.Biopsy of the suspicious area confirmed IDC with intraductal carcinoma.Breast magnetic resonance imaging showed typical hamartoma changes with irregular areas of abnormal enhancement both inside and outside.A breast-conserving surgery was performed,and postoperative pathology confirmed mammary hamartoma,concurrent with IDC and intraductal carcinoma occurring both inside and outside the hamartoma.Subsequently,appropriate adjuvant therapy was initiated.Currently,the patient is in good condition.Breast cancer may be located both inside and outside the ipsilateral mammary hamartoma,which is difficult to detect preoperatively,especially when there is a focus of intraductal carcinoma,requiring accurate assessment of the tumor extent by modern imaging techniques.Early detection of the coexistence of cancer is clinically important as it can alter patient management.CONCLUSION This case emphasizes the importance of modern imaging techniques in accurately evaluating mammary hamartomas associated with malignancies prior to surgery.展开更多
Splenic hamartomas(SHs)are uncommon,benign vascular lesions of unclear etiology and are mostly found incidentally on abdominal images,at surgery,or at autopsy.Since the first case description,in 1861,less than 50 pedi...Splenic hamartomas(SHs)are uncommon,benign vascular lesions of unclear etiology and are mostly found incidentally on abdominal images,at surgery,or at autopsy.Since the first case description,in 1861,less than 50 pediatric SH cases have been reported in the literature.In this article,we have performed an analysis of all SH cases in children published in the literature to date and presented our case of an 8-year-old male with SH.These lesions in children were shown to cause symptoms more often than in the adult population.The observed SH sizes in children ranged from a few millimeters to 18 cm,and the symptomatic lesions were mostly larger or multiple.The most common clinical finding was splenomegaly.Signs of hypersplenism were present in children with a single SH larger than 4.5 cm(diameter range:4.5-18.0 cm)and in those with multiple hamartomas,ranging from a few millimeters to 5 cm.Eighty percent of patients with available laboratory findings had hematological abnormalities such as anemia,thrombocytopenia,or pancytopenia.Other symptoms and signs included abdominal pain,recurrent infections,fever,night sweats,lethargy,growth retardation,and weight loss.The use of multiple imaging modalities may suggest the preoperative diagnosis of a splenic mass in children and determine the therapeutic approach.However,the final diagnosis of SH relies on histopathological evaluation.Surgery,including total or partial splenectomy(PS),is the mainstay of SH management.Milickovic M et al.Splenomas in children WJCC https://www.wjgnet.com 1910 April 16,2024 Volume 12 Issue 11 Although total splenectomy carries a greater risk of overwhelming post-splenectomy infection than PS it has remained the most performed surgical procedure in children with SH.In the majority of pediatric patients with symptomatic SH,resolution of symptoms and resolution or improvement of cytopenias occurred after surgical treatment.展开更多
Background: Hypothalamic hamartoma is a rare non-neoplastic lesion, typically identified in early childhood during investigations for precocious puberty and/or gelastic seizures. However, cases of diagnosis even earli...Background: Hypothalamic hamartoma is a rare non-neoplastic lesion, typically identified in early childhood during investigations for precocious puberty and/or gelastic seizures. However, cases of diagnosis even earlier or during fetal development have been documented. Case Presentation: A newborn girl was diagnosed with hydrocephalus during pregnancy. An MRI revealed a large oval hypothalamic process, which suggested a hypothalamic hamartoma. At the age of 2, she experienced alternating crying and laughing seizures, followed by a decrease in visual acuity. Due to involvement of the optic pathways, surgery was not performed and she underwent antiepileptic medicines and gamma knife radiotherapy. At the age of 5 years and 3 months, she presented with breast development and laboratory tests confirmed central precocious puberty. Quarterly injections of GnRH agonists have since been administered with favorable results. Conclusion: Early-diagnosed hypothalamic hamartomas require close monitoring, by an experienced multidisciplinary, to promptly detect and treat potential complications, especially precocious puberty, and prevent any undesirable impact on final height.展开更多
BACKGROUND Splenic hamartoma(SH)is a rare,benign vascular proliferation that is often found incidentally.It may be misdiagnosed as a splenic aneurysm or splenic malignancy.CASE SUMMARY A 21-year-old male patient was a...BACKGROUND Splenic hamartoma(SH)is a rare,benign vascular proliferation that is often found incidentally.It may be misdiagnosed as a splenic aneurysm or splenic malignancy.CASE SUMMARY A 21-year-old male patient was admitted to our hospital with a complaint of an incidentally discovered asymptomatic splenic space-occupying lesion for 2 wk.Abdominal computed tomography(CT)scan showed a circular low-density shadow in the hilum of the spleen.Contrast-enhanced CT revealed an aneurysm located in the hilum of the spleen before operation.Laparoscopic splenectomy was performed and postoperative pathology revealed the presence of SH.CONCLUSION Imaging studies are insufficient for the differential diagnosis of SH from other diseases,and laparoscopic splenectomy is a less invasive procedure and useful for the diagnostic purpose as well.展开更多
Splenic hamartoma(SH)is a rare benign tumor usually detected accidentally,which is composed of an aberrant mixture of normal splenic elements.Here,we report a case of 54-year-old man who presented with symptomatic mul...Splenic hamartoma(SH)is a rare benign tumor usually detected accidentally,which is composed of an aberrant mixture of normal splenic elements.Here,we report a case of 54-year-old man who presented with symptomatic multinodular SH and was admitted initially for thrombocytopenia and anemia.Physical examination revealed that the patients had an anemic appearance and palpable spleen,extending 10 cm below the costal margin.Preoperative ultrasound and computed tomography(CT)indicated splenomegaly with multinodular lesions.On enhanced CT scanning,during the arterial phase,the lesions demonstrated inhomogeneous enhancement,and in the portal phase the lesions were more hyperdense than the splenic parenchyma.The images were highly suggestive of a metastatic tumor.Splenectomy was performed 1 wk later.The tumor was eventually diagnosed as SH according to the morphological features and immunohistochemical detection,by which CD34 was positive in lining cells and some spindle cells,vimentin was positive in the tumor,factor-Ⅷ-related antigen was positive multifocally in lining cells,and smooth muscle actin was positive in some spindle cells.Thrombocytopenia and anemia were cured after splenectomy.展开更多
Non-neoplastic tumor-like lesions in the pancreas are uncommon.Here,we present a case of multiple solid pancreatic hamartomas in a 78-year-old Japanese woman.Her computed tomography revealed a pancreatic mass,measurin...Non-neoplastic tumor-like lesions in the pancreas are uncommon.Here,we present a case of multiple solid pancreatic hamartomas in a 78-year-old Japanese woman.Her computed tomography revealed a pancreatic mass,measuring 1.8 cm in maximum diameter.However,no symptoms were found.She was not an alcoholic and had no history of pancreatitis.The patient underwent a pancreatoduodenectomy,and three well-demarcated solid nodules measuring 1.7 cm,0.4 cm,and 0.3 cm in diameter were found in the pancreatic head.Microscopically,the lesions were composed of non-neoplastic,disarranged acinar cells and ducts embedded in a sclerotic stroma with elongated spindle cells that lacked discrete islets.The stromal spindle cells were immunoreactive for CD34 and CD117.The histological diagnosis was multiple solid hamartomas of the pancreas.There has been no recurrence 30 mo after surgery.So far,18 cases of pancreatic hamartoma have been reported in the English literature,including our case.Six out of these 18 cases seemed to fit the criteria of solid pancreatic hamartoma.Although the number of cases was limited,solid pancreatic hamartomas seem to be benign tumor-like lesions,which are found incidentally in healthy middle-aged adults,but occasionally involve the whole pancreas,resulting in a poor prognosis.Solid pancreatic hamartoma was sometimes associated with minor pancreatic abnormality,and multiple small lesions other than the main tumors were detected in a small number of cases.From these findings,one may speculate that solid pancreatic hamartoma could be the result of a malformation during the development of the pancreas.展开更多
Mesenchymal hamartomas of the liver(MHLs) in adults are rare and potentially premalignant lesions, which present as solid/cystic neoplasms. We report a rare case of orthotopic liver transplantation in a patient with a...Mesenchymal hamartomas of the liver(MHLs) in adults are rare and potentially premalignant lesions, which present as solid/cystic neoplasms. We report a rare case of orthotopic liver transplantation in a patient with a giant MHL. In 2013, a 34-year-old female sought medical advice after a 2-year history of progressive abdominal distention and respiratory distress. Physical examination revealed an extensive mass in the abdomen. Computed tomography(CT) of her abdomen revealed multiple liver cysts, with the diameter of largest cyst being 16 cm × 14 cm. The liver hilar structures were not clearly displayed. The adjacent organs were compressed and displaced. Initial laboratory tests, including biochemical investigations and coagulation profile, were unremarkable. Tumor markers, including levels of AFP, CEA and CA19-9, were within the normal ranges. The patient underwent orthotopic liver transplantation in November 2013, the liver being procured from a 40-year-old man after cardiac death following traumatic brain injury. Warm ischemic time was 7.5 min and cold ischemic time was 3 h. The recipient underwent classical orthotopic liver transplantation. The recipient operative procedure took 8.5 h, the anhepatic phase lasting for 1 h without the use of venovenous bypass. The immunosuppressive regimen includedintraoperative induction with basiliximab and high-dose methylprednisolone, and postoperative maintenance with tacrolimus, mycophenolate mofetil, and prednisone. The recipient's diseased liver weighed 21 kg(dry weight) and measured 41 cm × 32 cm × 31 cm. Histopathological examination confirmed the diagnosis of an MHL. The patient did not experience any acute rejection episode or other complication. All the laboratory tests returned to normal within one month after surgery. Three months after transplantation, the immunosuppressive therapy was reduced to tacrolimus monotherapy, and the T-tube was removed after cholangiography showed no abnormalities. Twelve months after transplantation, the patient remains well and is fulfilling all normal activities. Adult giant MHL is extremely rare. Symptoms, physical signs, laboratory results, and radiographic imaging are nonspecific and inconclusive. Surgical excision of the lesion is imperative to make a definite diagnosis and as a cure. Liver transplantation should be considered as an option in the treatment of a non-resectable MHL.展开更多
Splenic hamartoma is a rare benign malformation, composed of an anomalous mixture of normal splenic elements, often found incidentally while working up other complaints or at autopsy. A splenic mass was incidentally f...Splenic hamartoma is a rare benign malformation, composed of an anomalous mixture of normal splenic elements, often found incidentally while working up other complaints or at autopsy. A splenic mass was incidentally found while evaluating the effects of a traffic accident in a 63-year-old woman. Abdominal computed tomography revealed a well-defined splenic mass with rim enhancement. The patient underwent splenectomy. The resected spleen contained a well-defined mass lesion measuring 3.5 cm × 3.0 cm. Microscopic examination revealed disorganized slit-like vascular channels lined by plump endothelial cells without atypia. The cells lining the vascular channels were positive for CD8, CD31, CD34 and vimentin. Endothelial cells that are positive for CD8 are a key feature that differentiates hamartoma from other vascular lesions of the spleen. Although this tumor is very rare, it must be included in the differential diagnosis of splenic mass-forming lesions.展开更多
AIM： To evaluate the imaging findings of biliary hamartomas （yon Meyenburg complexes, VMCs） and discuss the differential diagnosis with other related diseases. METHODS： Imaging findings of biliary hamartomas on ul...AIM： To evaluate the imaging findings of biliary hamartomas （yon Meyenburg complexes, VMCs） and discuss the differential diagnosis with other related diseases. METHODS： Imaging findings of biliary hamartomas on ultrasonography （US）, computed tomography （C-T）, magnetic resonance imaging （MRI）, MR cholangiopancreatography （MRCP）and hepatobiliary scintigraphy were retrospectively analyzed in six patients. RESULTS： On ultrasound images, five of the six cases showed multiple small hyper- and hypo-echoic lesions with comet-tail echoes, especially when magnified by US with the usage of zoom function. In all the six cases, multiple tiny hypodense lesions less than 10 mm in diameter were revealed as scattered throughout the liver with no enhancement on CT. These tiny lesions were demonstrated to be hyper- and hypo-intensity on T2- and TI-weighed images, respectively, in three patients who underwent MRI examinations. MRCP was performed in two patients, and clearly showed multiple tiny irregular- and round-shaped hyper-intensity lesions. MRCP and hepatobiliary scintigraphy showed normal appearances of intra- and extra-hepatic bile ducts in two and one patients, respectively. CONCLUSION： Imaging modalities are useful in the diagnosis and differential diagnosis of VMCs. A correct diagnosis might be obtained when typical imaging findings are present even without a histological confirmation.展开更多
Splenic hamartoma is a rare benign tumor,and although minimally invasive surgery may be suitable for this condition,there have only been 2 previous reports of laparoscopic surgery.Here we report the third case of sple...Splenic hamartoma is a rare benign tumor,and although minimally invasive surgery may be suitable for this condition,there have only been 2 previous reports of laparoscopic surgery.Here we report the third case of splenic hamartoma managed by laparoscopic splenectomy.A 37-year-old male was incidentally diagnosed by abdominal ultrasonography with a hypoechoic mass measuring 2.5 cm × 2.4 cm in the spleen.Color Doppler sonography showed multiple flow signals within the mass and contrast-enhanced computed tomography revealed strong enhancement of the lesion.On T1-and T2-weighted magnetic resonance images,the splenic mass was demonstrated as isointense and hyperintense respectively.Although a malignant tumor could not be ruled out,a hand-assisted laparoscopic splenectomy was performed because the splenic mass was limited in size and had not invaded adjacent organs.The pathological diagnosis was splenic hamartoma.The postoperative course was uneventful and the patient was discharged by the seventh postoperative day.Although splenic hamartomas have some specific imaging features,more reports and analyses of these cases are required to increase the reliability of the diagnosis and management.Hand-assisted laparoscopic splenectomy may play a pivotal role in the postoperative diagnosis and management of this condition.展开更多
We present the gray-scale ultrasonography （GSUS）, power Doppler ultrasonography （PDUS）, abdominal computed tomography （CT）, and magnetic resonance imaging （MRI） findings for a case of splenic hamartoma in a 27...We present the gray-scale ultrasonography （GSUS）, power Doppler ultrasonography （PDUS）, abdominal computed tomography （CT）, and magnetic resonance imaging （MRI） findings for a case of splenic hamartoma in a 27-year-old man, showing a φ 50 mm homogeneous, iso- and hypo-echoic splenic mass with evidence of a small plural cystic lesion. This splenic hamartoma showed increased vascularity on power Doppler sonograms. PDUS showed multiple circular blood flow signals inside the mass （i.e. a basket pattern）, which was consistent with the small plural cystic lesion shown by GSUS. Spectral analysis also confirmed arterial and venous flow. CT scans showed that the mass had lowdensity relative to the normal spleen and MRI showed that the mass was isodense, relative to the normal spleen. Therefore, CT and MRI are not useful for the diagnosis of splenic hamartoma. Ultrasonography can be used to diagnose splenic hamartoma without administration of a contrast matedal and therefore is an indispensable method for the diagnosis of splenic hamartoma.展开更多
AIM: To investigate our clinical experience with the colonic manifestations of phosphatase and tensin homolog on chromosome ten (PTEN) hamartoma tumor syndrome (PHTS) and to perform a systematic literature review rega...AIM: To investigate our clinical experience with the colonic manifestations of phosphatase and tensin homolog on chromosome ten (PTEN) hamartoma tumor syndrome (PHTS) and to perform a systematic literature review regarding the same.展开更多
A solitary Peutz-Jeghers-type hamartomatous polyp in the duodenum is rare, and few reports have described its characteristic endoscopic features. We describe three cases of solitary Peutz-Jeghers-type hamartomatous po...A solitary Peutz-Jeghers-type hamartomatous polyp in the duodenum is rare, and few reports have described its characteristic endoscopic features. We describe three cases of solitary Peutz-Jeghers-type hamartomatous polyp and their endoscopic findings in detail. The polyp in all of our three cases showed an irregularly lobular or nodular surface, whereas adenomas often show a regularly nodular or granular surface. The color of the polyp was whitish in all of our cases. In the present cases, close observation by endoscopy revealed that the solitary Peutz-Jeghers-type hamartomatous polyps looked whitish because of the presence of diffusely scattered white spots on the surface of the polyps. Duodenal polyps that exhibit the aforementioned endoscopic characteristics may be diagnosed as Peutz-Jeghers-type hamartomatous polyps and treated by polypectomy because of the malignant potential.展开更多
Spontaneous rupture is a rare complication of splenic hamartoma. A review of the literature revealed only four such cases. To the best of our knowledge, this is the first report of spontaneous rupture of splenic hamar...Spontaneous rupture is a rare complication of splenic hamartoma. A review of the literature revealed only four such cases. To the best of our knowledge, this is the first report of spontaneous rupture of splenic hamartoma associated with liver cirrhosis and portal hypertension. A 53-year-old woman, who was followed up for aortic dissection and hepatitis C virus （HCV）-related liver cirrhosis, was referred with sudden left chest and shoulder pain. An abdominal ultrasound showed intraabdominal bleeding, and computed tomography indicated rupture of a splenic tumor. Emergent splenectomy was carried out. The postoperative course was uneventful, and the patient was discharged on the 13th postoperative day. Pathology revealed the tumor to be a ruptured splenic hamartoma. The non-tumorous splenic parenchyma revealed congestive changes. We consider that the presence of liver cirrhosis and portal hypertension are risk factors for spontaneous rupture of the splenic hamartoma.展开更多
: Hamartomas of the bile duct (von Meyenburg complex) are benign neoplasms of the liver, constituted histologically cystic dilatations of the bile duct, encompassed by fibrous stroma. We report a 42-year-old female pa...: Hamartomas of the bile duct (von Meyenburg complex) are benign neoplasms of the liver, constituted histologically cystic dilatations of the bile duct, encompassed by fibrous stroma. We report a 42-year-old female patient with symptomatic cholecystitis, whose gross and ultrasonic appearance suggestive of multiple liver metastases. Magnetic resonance imaging and liver biopsy are the gold standards for diagnosis of this rare hepatobiliary condition.展开更多
We present a case of a 72-year-old man with a common bile duct cancer, who was initially believed to have multiple liver metastases based on computed tomography findings, and in whom magnetic resonance cholangiography...We present a case of a 72-year-old man with a common bile duct cancer, who was initially believed to have multiple liver metastases based on computed tomography findings, and in whom magnetic resonance cholangiography （MRC） revealed a diagnosis of bile duct hamartomas. At exploration for pancreaticoduodenectomy, liver palpation revealed disseminated nodules at the surface of the liver. These nodules showed gray-white nodular lesions of about 0.5 cm in diameter scattered on the surface of both liver lobes, which were looked like multiple liver metastases from bile duct cancer. Frozen section of the liver biopsy disclosed multiple bile ducts with slightly dilated lumens embedded in the collagenous stroma characteristics of multiple bile duct hamartomas （BDHs）. Only two reports have described the MRC features of bile duct hamartomas. Of all imaging procedures, MRC provides the most relevant features for the imaging diagnosis of bile duct hamartomas.展开更多
Dear Editor,I＇m Dr. Gilda Cennamo from the Eye Clinic of Department of Neurosciences, Reproductive Sciences and Dentistry,University of Naples Federico II, Naples, Italy. I write to present four cases ofjuxtapapillar...Dear Editor,I＇m Dr. Gilda Cennamo from the Eye Clinic of Department of Neurosciences, Reproductive Sciences and Dentistry,University of Naples Federico II, Naples, Italy. I write to present four cases ofjuxtapapillary hamartoma evaluated with non-invasive optical coherence tomography-angiography （OCT-A）.展开更多
Objective:To examine the values of endoscopic ultrasonography (EUS) on diagnosis and treatment of esophageal hamartoma. Methods:We compared and analyzed various kinds of imaging examinations such as barium esophagram,...Objective:To examine the values of endoscopic ultrasonography (EUS) on diagnosis and treatment of esophageal hamartoma. Methods:We compared and analyzed various kinds of imaging examinations such as barium esophagram, con-trast-enhanced computed tomography (CT) and conventional gastroscopy in retrospectively reviewing the clinical data of an esophageal hamartoma patient seen in our clinic in the First Affiliated Hospital, School of Medicine, Zhejiang University, Hangzhou, China. Having received various imaging examinations, this patient was finally diagnosed with esophageal hamartoma and underwent gastroscopic resection of hamartoma with the diagnostic information obtained from EUS. The patient had been regularly followed up for 13 months after treatment. Results: Barium esophagram, CT and conventional gastroscopy detected the lesion, but were unable to distinguish it from common esophagopolypus and other submucosal lesions, and unable to determine etiopathogenisis. EUS detected the hamartoma and identified its internal structure, echo, exact size, depth of invasion, origin and the relationship between adjacent tissues and organs, differentiating the lesion from other submucosal tumors and clearly defining the diagnosis. EUS-guided fine needle aspiration (FNA) also helped to identify the etiological diagnosis. Conclusion: EUS was superior to other imaging means in diagnosis and treatment of hamartoma.展开更多
The safety and efficacy of retroperitoneoscopic microwave ablation (MWA) in the treatment of renal hamartoma were evaluated.From July 2007 to July 2009, a total of 16 cases of renal hamartoma were treated with retrope...The safety and efficacy of retroperitoneoscopic microwave ablation (MWA) in the treatment of renal hamartoma were evaluated.From July 2007 to July 2009, a total of 16 cases of renal hamartoma were treated with retroperitoneoscopic MWA.Peri-and post-operative findings were observed.Middle-term efficacy was assessed by contrast-enhanced computerized tomography (CT) in follow-up period.All patients received MWA of 1-5 points.The mean operative time was 85 min and the mean blood loss was 65 mL.During a median follow-up of 16 months, no evidence of disease recurrence was observed despite of incomplete ablation in 1 case.Retroperitoneoscopic MWA is a relatively simple procedure with less impact to renal function and less complication.The outcome of middle-term follow-up is satisfactory.Thus, retroperitoneoscopic MWA appears to be a safe and effective technique for renal hamartoma in selected patients.展开更多
基金Supported by Clinical Research Grant from Pusan National University Hospital in 2024.
文摘BACKGROUND Primary splenic lesions are rare and often detected incidentally through imaging,biopsy,or autopsy,typically without distinct clinical symptoms.Although imaging can help differentiate benign from malignant lesions,splenic hamartomas,and angiosarcomas may exhibit overlapping features,making diagnosis challenging.This report presents a case of splenic hamartoma suspected to be an angiosarcoma based on preoperative imaging.Splenic hamartomas that mimic angiosarcomas are exceedingly rare.CASE SUMMARY A 33-year-old male presented to the Department of Emergency with frank red blood hematemesis and a 1-week history of epigastric pain.On arrival,he was alert and hemodynamically stable.Contrast-enhanced abdominal computed tomography revealed splenomegaly with significant engorgement of the portal and splenic veins,along with a diffuse nodular splenic lesion measuring 8.2 cm×6.2 cm.Following esophageal varix ligation,abdominal magnetic resonance imaging demonstrated iso-to high-signal intensity within the splenic mass and multiple hypervascular lesions in the right hepatic lobe,raising suspicion for splenic an-giosarcoma with hepatic metastases.18F-fluorodeoxyglucose positron emission tomography-computed tomography showed diffusely mild increased metabolic activity in the spleen.The patient subsequently underwent splenectomy and liver biopsy.Histopathological examination revealed chronic inflammation in the liver,and the splenic lesion was confirmed to be a splenic hamartoma.The patient successfully returned to work and remains in good health.CONCLUSION This rare case of splenic hamartoma mimicking angiosarcoma highlights the importance of differential diagnosis in managing splenic tumors.
基金Supported by Jilin City Science and Technology Innovation Development Plan Project,China,No.20230406201Jilin Province Traditional Chinese Medicine Technology Project,China,No.2024159.
文摘BACKGROUND Breast hamartomas are rare benign breast tumors,with an incidence rate of 0.8%-4.8%.Further,the coexistence of hamartomas and carcinoma is also uncommon.Our case report presents a unique instance where invasive ductal carcinoma(IDC)and ductal carcinoma in situ were found both inside and outside a breast hamartoma.This is the second case reported in the literature.CASE SUMMARY A 51-year-old woman presented with a 6.0 cm breast tumor on mammography and ultrasound,with suspicious areas indicative of malignant transformation.Biopsy of the suspicious area confirmed IDC with intraductal carcinoma.Breast magnetic resonance imaging showed typical hamartoma changes with irregular areas of abnormal enhancement both inside and outside.A breast-conserving surgery was performed,and postoperative pathology confirmed mammary hamartoma,concurrent with IDC and intraductal carcinoma occurring both inside and outside the hamartoma.Subsequently,appropriate adjuvant therapy was initiated.Currently,the patient is in good condition.Breast cancer may be located both inside and outside the ipsilateral mammary hamartoma,which is difficult to detect preoperatively,especially when there is a focus of intraductal carcinoma,requiring accurate assessment of the tumor extent by modern imaging techniques.Early detection of the coexistence of cancer is clinically important as it can alter patient management.CONCLUSION This case emphasizes the importance of modern imaging techniques in accurately evaluating mammary hamartomas associated with malignancies prior to surgery.
文摘Splenic hamartomas(SHs)are uncommon,benign vascular lesions of unclear etiology and are mostly found incidentally on abdominal images,at surgery,or at autopsy.Since the first case description,in 1861,less than 50 pediatric SH cases have been reported in the literature.In this article,we have performed an analysis of all SH cases in children published in the literature to date and presented our case of an 8-year-old male with SH.These lesions in children were shown to cause symptoms more often than in the adult population.The observed SH sizes in children ranged from a few millimeters to 18 cm,and the symptomatic lesions were mostly larger or multiple.The most common clinical finding was splenomegaly.Signs of hypersplenism were present in children with a single SH larger than 4.5 cm(diameter range:4.5-18.0 cm)and in those with multiple hamartomas,ranging from a few millimeters to 5 cm.Eighty percent of patients with available laboratory findings had hematological abnormalities such as anemia,thrombocytopenia,or pancytopenia.Other symptoms and signs included abdominal pain,recurrent infections,fever,night sweats,lethargy,growth retardation,and weight loss.The use of multiple imaging modalities may suggest the preoperative diagnosis of a splenic mass in children and determine the therapeutic approach.However,the final diagnosis of SH relies on histopathological evaluation.Surgery,including total or partial splenectomy(PS),is the mainstay of SH management.Milickovic M et al.Splenomas in children WJCC https://www.wjgnet.com 1910 April 16,2024 Volume 12 Issue 11 Although total splenectomy carries a greater risk of overwhelming post-splenectomy infection than PS it has remained the most performed surgical procedure in children with SH.In the majority of pediatric patients with symptomatic SH,resolution of symptoms and resolution or improvement of cytopenias occurred after surgical treatment.
文摘Background: Hypothalamic hamartoma is a rare non-neoplastic lesion, typically identified in early childhood during investigations for precocious puberty and/or gelastic seizures. However, cases of diagnosis even earlier or during fetal development have been documented. Case Presentation: A newborn girl was diagnosed with hydrocephalus during pregnancy. An MRI revealed a large oval hypothalamic process, which suggested a hypothalamic hamartoma. At the age of 2, she experienced alternating crying and laughing seizures, followed by a decrease in visual acuity. Due to involvement of the optic pathways, surgery was not performed and she underwent antiepileptic medicines and gamma knife radiotherapy. At the age of 5 years and 3 months, she presented with breast development and laboratory tests confirmed central precocious puberty. Quarterly injections of GnRH agonists have since been administered with favorable results. Conclusion: Early-diagnosed hypothalamic hamartomas require close monitoring, by an experienced multidisciplinary, to promptly detect and treat potential complications, especially precocious puberty, and prevent any undesirable impact on final height.
文摘BACKGROUND Splenic hamartoma(SH)is a rare,benign vascular proliferation that is often found incidentally.It may be misdiagnosed as a splenic aneurysm or splenic malignancy.CASE SUMMARY A 21-year-old male patient was admitted to our hospital with a complaint of an incidentally discovered asymptomatic splenic space-occupying lesion for 2 wk.Abdominal computed tomography(CT)scan showed a circular low-density shadow in the hilum of the spleen.Contrast-enhanced CT revealed an aneurysm located in the hilum of the spleen before operation.Laparoscopic splenectomy was performed and postoperative pathology revealed the presence of SH.CONCLUSION Imaging studies are insufficient for the differential diagnosis of SH from other diseases,and laparoscopic splenectomy is a less invasive procedure and useful for the diagnostic purpose as well.
文摘Splenic hamartoma(SH)is a rare benign tumor usually detected accidentally,which is composed of an aberrant mixture of normal splenic elements.Here,we report a case of 54-year-old man who presented with symptomatic multinodular SH and was admitted initially for thrombocytopenia and anemia.Physical examination revealed that the patients had an anemic appearance and palpable spleen,extending 10 cm below the costal margin.Preoperative ultrasound and computed tomography(CT)indicated splenomegaly with multinodular lesions.On enhanced CT scanning,during the arterial phase,the lesions demonstrated inhomogeneous enhancement,and in the portal phase the lesions were more hyperdense than the splenic parenchyma.The images were highly suggestive of a metastatic tumor.Splenectomy was performed 1 wk later.The tumor was eventually diagnosed as SH according to the morphological features and immunohistochemical detection,by which CD34 was positive in lining cells and some spindle cells,vimentin was positive in the tumor,factor-Ⅷ-related antigen was positive multifocally in lining cells,and smooth muscle actin was positive in some spindle cells.Thrombocytopenia and anemia were cured after splenectomy.
文摘Non-neoplastic tumor-like lesions in the pancreas are uncommon.Here,we present a case of multiple solid pancreatic hamartomas in a 78-year-old Japanese woman.Her computed tomography revealed a pancreatic mass,measuring 1.8 cm in maximum diameter.However,no symptoms were found.She was not an alcoholic and had no history of pancreatitis.The patient underwent a pancreatoduodenectomy,and three well-demarcated solid nodules measuring 1.7 cm,0.4 cm,and 0.3 cm in diameter were found in the pancreatic head.Microscopically,the lesions were composed of non-neoplastic,disarranged acinar cells and ducts embedded in a sclerotic stroma with elongated spindle cells that lacked discrete islets.The stromal spindle cells were immunoreactive for CD34 and CD117.The histological diagnosis was multiple solid hamartomas of the pancreas.There has been no recurrence 30 mo after surgery.So far,18 cases of pancreatic hamartoma have been reported in the English literature,including our case.Six out of these 18 cases seemed to fit the criteria of solid pancreatic hamartoma.Although the number of cases was limited,solid pancreatic hamartomas seem to be benign tumor-like lesions,which are found incidentally in healthy middle-aged adults,but occasionally involve the whole pancreas,resulting in a poor prognosis.Solid pancreatic hamartoma was sometimes associated with minor pancreatic abnormality,and multiple small lesions other than the main tumors were detected in a small number of cases.From these findings,one may speculate that solid pancreatic hamartoma could be the result of a malformation during the development of the pancreas.
基金Supported by National Natural Science Foundation of China,No.81400680the National High Technology Research and Development Program of China,No.2012 AA021001
文摘Mesenchymal hamartomas of the liver(MHLs) in adults are rare and potentially premalignant lesions, which present as solid/cystic neoplasms. We report a rare case of orthotopic liver transplantation in a patient with a giant MHL. In 2013, a 34-year-old female sought medical advice after a 2-year history of progressive abdominal distention and respiratory distress. Physical examination revealed an extensive mass in the abdomen. Computed tomography(CT) of her abdomen revealed multiple liver cysts, with the diameter of largest cyst being 16 cm × 14 cm. The liver hilar structures were not clearly displayed. The adjacent organs were compressed and displaced. Initial laboratory tests, including biochemical investigations and coagulation profile, were unremarkable. Tumor markers, including levels of AFP, CEA and CA19-9, were within the normal ranges. The patient underwent orthotopic liver transplantation in November 2013, the liver being procured from a 40-year-old man after cardiac death following traumatic brain injury. Warm ischemic time was 7.5 min and cold ischemic time was 3 h. The recipient underwent classical orthotopic liver transplantation. The recipient operative procedure took 8.5 h, the anhepatic phase lasting for 1 h without the use of venovenous bypass. The immunosuppressive regimen includedintraoperative induction with basiliximab and high-dose methylprednisolone, and postoperative maintenance with tacrolimus, mycophenolate mofetil, and prednisone. The recipient's diseased liver weighed 21 kg(dry weight) and measured 41 cm × 32 cm × 31 cm. Histopathological examination confirmed the diagnosis of an MHL. The patient did not experience any acute rejection episode or other complication. All the laboratory tests returned to normal within one month after surgery. Three months after transplantation, the immunosuppressive therapy was reduced to tacrolimus monotherapy, and the T-tube was removed after cholangiography showed no abnormalities. Twelve months after transplantation, the patient remains well and is fulfilling all normal activities. Adult giant MHL is extremely rare. Symptoms, physical signs, laboratory results, and radiographic imaging are nonspecific and inconclusive. Surgical excision of the lesion is imperative to make a definite diagnosis and as a cure. Liver transplantation should be considered as an option in the treatment of a non-resectable MHL.
文摘Splenic hamartoma is a rare benign malformation, composed of an anomalous mixture of normal splenic elements, often found incidentally while working up other complaints or at autopsy. A splenic mass was incidentally found while evaluating the effects of a traffic accident in a 63-year-old woman. Abdominal computed tomography revealed a well-defined splenic mass with rim enhancement. The patient underwent splenectomy. The resected spleen contained a well-defined mass lesion measuring 3.5 cm × 3.0 cm. Microscopic examination revealed disorganized slit-like vascular channels lined by plump endothelial cells without atypia. The cells lining the vascular channels were positive for CD8, CD31, CD34 and vimentin. Endothelial cells that are positive for CD8 are a key feature that differentiates hamartoma from other vascular lesions of the spleen. Although this tumor is very rare, it must be included in the differential diagnosis of splenic mass-forming lesions.
文摘AIM： To evaluate the imaging findings of biliary hamartomas （yon Meyenburg complexes, VMCs） and discuss the differential diagnosis with other related diseases. METHODS： Imaging findings of biliary hamartomas on ultrasonography （US）, computed tomography （C-T）, magnetic resonance imaging （MRI）, MR cholangiopancreatography （MRCP）and hepatobiliary scintigraphy were retrospectively analyzed in six patients. RESULTS： On ultrasound images, five of the six cases showed multiple small hyper- and hypo-echoic lesions with comet-tail echoes, especially when magnified by US with the usage of zoom function. In all the six cases, multiple tiny hypodense lesions less than 10 mm in diameter were revealed as scattered throughout the liver with no enhancement on CT. These tiny lesions were demonstrated to be hyper- and hypo-intensity on T2- and TI-weighed images, respectively, in three patients who underwent MRI examinations. MRCP was performed in two patients, and clearly showed multiple tiny irregular- and round-shaped hyper-intensity lesions. MRCP and hepatobiliary scintigraphy showed normal appearances of intra- and extra-hepatic bile ducts in two and one patients, respectively. CONCLUSION： Imaging modalities are useful in the diagnosis and differential diagnosis of VMCs. A correct diagnosis might be obtained when typical imaging findings are present even without a histological confirmation.
文摘Splenic hamartoma is a rare benign tumor,and although minimally invasive surgery may be suitable for this condition,there have only been 2 previous reports of laparoscopic surgery.Here we report the third case of splenic hamartoma managed by laparoscopic splenectomy.A 37-year-old male was incidentally diagnosed by abdominal ultrasonography with a hypoechoic mass measuring 2.5 cm × 2.4 cm in the spleen.Color Doppler sonography showed multiple flow signals within the mass and contrast-enhanced computed tomography revealed strong enhancement of the lesion.On T1-and T2-weighted magnetic resonance images,the splenic mass was demonstrated as isointense and hyperintense respectively.Although a malignant tumor could not be ruled out,a hand-assisted laparoscopic splenectomy was performed because the splenic mass was limited in size and had not invaded adjacent organs.The pathological diagnosis was splenic hamartoma.The postoperative course was uneventful and the patient was discharged by the seventh postoperative day.Although splenic hamartomas have some specific imaging features,more reports and analyses of these cases are required to increase the reliability of the diagnosis and management.Hand-assisted laparoscopic splenectomy may play a pivotal role in the postoperative diagnosis and management of this condition.
文摘We present the gray-scale ultrasonography （GSUS）, power Doppler ultrasonography （PDUS）, abdominal computed tomography （CT）, and magnetic resonance imaging （MRI） findings for a case of splenic hamartoma in a 27-year-old man, showing a φ 50 mm homogeneous, iso- and hypo-echoic splenic mass with evidence of a small plural cystic lesion. This splenic hamartoma showed increased vascularity on power Doppler sonograms. PDUS showed multiple circular blood flow signals inside the mass （i.e. a basket pattern）, which was consistent with the small plural cystic lesion shown by GSUS. Spectral analysis also confirmed arterial and venous flow. CT scans showed that the mass had lowdensity relative to the normal spleen and MRI showed that the mass was isodense, relative to the normal spleen. Therefore, CT and MRI are not useful for the diagnosis of splenic hamartoma. Ultrasonography can be used to diagnose splenic hamartoma without administration of a contrast matedal and therefore is an indispensable method for the diagnosis of splenic hamartoma.
文摘AIM: To investigate our clinical experience with the colonic manifestations of phosphatase and tensin homolog on chromosome ten (PTEN) hamartoma tumor syndrome (PHTS) and to perform a systematic literature review regarding the same.
文摘A solitary Peutz-Jeghers-type hamartomatous polyp in the duodenum is rare, and few reports have described its characteristic endoscopic features. We describe three cases of solitary Peutz-Jeghers-type hamartomatous polyp and their endoscopic findings in detail. The polyp in all of our three cases showed an irregularly lobular or nodular surface, whereas adenomas often show a regularly nodular or granular surface. The color of the polyp was whitish in all of our cases. In the present cases, close observation by endoscopy revealed that the solitary Peutz-Jeghers-type hamartomatous polyps looked whitish because of the presence of diffusely scattered white spots on the surface of the polyps. Duodenal polyps that exhibit the aforementioned endoscopic characteristics may be diagnosed as Peutz-Jeghers-type hamartomatous polyps and treated by polypectomy because of the malignant potential.
文摘Spontaneous rupture is a rare complication of splenic hamartoma. A review of the literature revealed only four such cases. To the best of our knowledge, this is the first report of spontaneous rupture of splenic hamartoma associated with liver cirrhosis and portal hypertension. A 53-year-old woman, who was followed up for aortic dissection and hepatitis C virus （HCV）-related liver cirrhosis, was referred with sudden left chest and shoulder pain. An abdominal ultrasound showed intraabdominal bleeding, and computed tomography indicated rupture of a splenic tumor. Emergent splenectomy was carried out. The postoperative course was uneventful, and the patient was discharged on the 13th postoperative day. Pathology revealed the tumor to be a ruptured splenic hamartoma. The non-tumorous splenic parenchyma revealed congestive changes. We consider that the presence of liver cirrhosis and portal hypertension are risk factors for spontaneous rupture of the splenic hamartoma.
文摘: Hamartomas of the bile duct (von Meyenburg complex) are benign neoplasms of the liver, constituted histologically cystic dilatations of the bile duct, encompassed by fibrous stroma. We report a 42-year-old female patient with symptomatic cholecystitis, whose gross and ultrasonic appearance suggestive of multiple liver metastases. Magnetic resonance imaging and liver biopsy are the gold standards for diagnosis of this rare hepatobiliary condition.
文摘We present a case of a 72-year-old man with a common bile duct cancer, who was initially believed to have multiple liver metastases based on computed tomography findings, and in whom magnetic resonance cholangiography （MRC） revealed a diagnosis of bile duct hamartomas. At exploration for pancreaticoduodenectomy, liver palpation revealed disseminated nodules at the surface of the liver. These nodules showed gray-white nodular lesions of about 0.5 cm in diameter scattered on the surface of both liver lobes, which were looked like multiple liver metastases from bile duct cancer. Frozen section of the liver biopsy disclosed multiple bile ducts with slightly dilated lumens embedded in the collagenous stroma characteristics of multiple bile duct hamartomas （BDHs）. Only two reports have described the MRC features of bile duct hamartomas. Of all imaging procedures, MRC provides the most relevant features for the imaging diagnosis of bile duct hamartomas.
文摘Dear Editor,I＇m Dr. Gilda Cennamo from the Eye Clinic of Department of Neurosciences, Reproductive Sciences and Dentistry,University of Naples Federico II, Naples, Italy. I write to present four cases ofjuxtapapillary hamartoma evaluated with non-invasive optical coherence tomography-angiography （OCT-A）.
文摘Objective:To examine the values of endoscopic ultrasonography (EUS) on diagnosis and treatment of esophageal hamartoma. Methods:We compared and analyzed various kinds of imaging examinations such as barium esophagram, con-trast-enhanced computed tomography (CT) and conventional gastroscopy in retrospectively reviewing the clinical data of an esophageal hamartoma patient seen in our clinic in the First Affiliated Hospital, School of Medicine, Zhejiang University, Hangzhou, China. Having received various imaging examinations, this patient was finally diagnosed with esophageal hamartoma and underwent gastroscopic resection of hamartoma with the diagnostic information obtained from EUS. The patient had been regularly followed up for 13 months after treatment. Results: Barium esophagram, CT and conventional gastroscopy detected the lesion, but were unable to distinguish it from common esophagopolypus and other submucosal lesions, and unable to determine etiopathogenisis. EUS detected the hamartoma and identified its internal structure, echo, exact size, depth of invasion, origin and the relationship between adjacent tissues and organs, differentiating the lesion from other submucosal tumors and clearly defining the diagnosis. EUS-guided fine needle aspiration (FNA) also helped to identify the etiological diagnosis. Conclusion: EUS was superior to other imaging means in diagnosis and treatment of hamartoma.
文摘The safety and efficacy of retroperitoneoscopic microwave ablation (MWA) in the treatment of renal hamartoma were evaluated.From July 2007 to July 2009, a total of 16 cases of renal hamartoma were treated with retroperitoneoscopic MWA.Peri-and post-operative findings were observed.Middle-term efficacy was assessed by contrast-enhanced computerized tomography (CT) in follow-up period.All patients received MWA of 1-5 points.The mean operative time was 85 min and the mean blood loss was 65 mL.During a median follow-up of 16 months, no evidence of disease recurrence was observed despite of incomplete ablation in 1 case.Retroperitoneoscopic MWA is a relatively simple procedure with less impact to renal function and less complication.The outcome of middle-term follow-up is satisfactory.Thus, retroperitoneoscopic MWA appears to be a safe and effective technique for renal hamartoma in selected patients.
