Cholangiocarcinoma is a rare gastrointestinal malignancy that arises within the intrahepatic, perihilar, and/or extrahepatic bile ducts. Individuals with cystic fibrosis are at increased risk for gastrointestinal malignancies. The most common gastrointestinal malignancy in cystic fibrosis is colon cancer, but other gastrointestinal malignancies also occur at greater rates than the general population.We present a case of a rapidly progressive metastatic intrahepatic cholangiocarcinoma in an individual with cystic fibrosis who was 5 months postpartum, incidentally found while undergoing a lung transplantation evaluation.A heightened clinical awareness of gastrointestinal malignancies, beyond colon cancer, in individuals with cystic fibrosis is warranted. It remains unclear if pregnancy is an additional risk factor for gastrointestinal malignancies in cystic fibrosis. Individuals with cystic fibrosis (CF) are at higher risk for digestive tract cancers , 2. Cyst1) of 21% predicted and a 6-l home oxygen requirement, was undergoing evaluation for bilateral lung transplantation. Her medical history included recurrent respiratory infections with multi-drug resistant pathogens, pancreatic insufficiency, malnutrition requiring percutaneous gastrostomy feeds, atrioventricular nodal reentry tachycardia status post ablation, and the recent delivery of a healthy baby. She had no history of biliary tract stones, cirrhosis, hepatitis B, or hepatitis C. As part of the lung transplantation workup a CT scan of the chest, abdomen, and pelvis revealed a new peripherally enhancing 6.3 cm mass in the right lobe of her liver, and subsequent MRI suggested intrahepatic cholangiocarcinoma. At this time, the patient reported feeling a hard mass under her right ribs along with intermittent right upper quadrant pain and fullness, anorexia, weight loss, and daily subjective fevers for several weeks.A 26-year-old female with CF (genotype F508del/F508del), with a baseline forced expiratory volume in 1 s , GATA-3, a transcription factor important in the regulation of certain genes, and p40, a marker of squamous differentiation. Notably, the tumor was negative for estrogen receptor (ER), thyroid transcription factor 1 (TTF-1), cytokeratin 20 (CK20), and high risk human papillomavirus ribonucleic acid in situ hybridization (HR HPV RNA ISH), and therefore did not support tumor origin of breast or gynecologic, lung, lower gastrointestinal, or cervical or head/neck, respectively. Serum tumor markers demonstrated normal alpha-fetoprotein (AFP) and carcinoembryonic antigen (CEA) levels, but a markedly elevated carbohydrate antigen (CA)19–9 level of 5949.5 U/mL (0–36 U/mL), which can be elevated in pancreatic, gastric, hepatobiliary, and colonic malignancies. Given the tumor location, serum markers, and pathology findings, the tumor was thought to be most consistent with a primary cholangiocarcinoma.The patient underwent complete staging following hospital discharge, where radiation therapy with curative intent was recommended. Ultimately her poor performance status, chronic pulmonary disease, and risk of severe life-threatening infection influenced the decision not to pursue systemic chemotherapy or surgery as the initial treatment modality. However, prior to starting treatment, she developed pneumonia leading to acute respiratory failure requiring intubation. A second CT of the chest, abdomen, and pelvis  revealed the liver mass had grown to 13.6 × 10 cm and there were patchy pulmonary consolidations concerning for both pneumonia and metastatic disease. Positron emission tomography (PET) scan demonstrated uptake in the lungs bilaterally, mediastinal lymph nodes, right iliac, and right femur, consistent with metastatic disease. Given her critical condition, it was decided she was no longer a candidate for treatment of her cancer. She was transitioned to home hospice where she passed away approximately 10 weeks after the initial CT.Autopsy revealed a 20 × 17 × 23 cm tan mass in the right lobe of the liver  and fluid transport, impaired release and clearance of mucus which can lead to meconium ileus and distal intestinal obstructive syndrome (DIOS), pancreatic insufficiency, decreased intestinal lumen pH, increased intestinal stem cell pH, abnormal bacterial colonization, microbial dysbiosis, and impaired innate immune responses that lead to chronic inflammation . As a reHelicobacter pylori, and certain parasitic infections, obesity, medications, CF, and several other genetic conditions such as Lynch syndrome, multiple biliary papillomatosis, and BAP1 tumor predisposition syndrome [Cholangiocarcinoma refers to cancers of the bile duct that arise in the intrahepatic (< 10% of cases), perihilar, or distal (extrahepatic) biliary tree . Cholangsyndrome , 21, 22.syndrome . In addisyndrome –26.Other than CF, our patient did not have any known risk factors for cholangiocarcinoma that have been previously described in the literature. However, she was 5 months postpartum at the time of her cancer diagnosis. Although we cannot be certain as to whether the cholangiocarcinoma was present before her pregnancy, this is of particular interest as Qasrawi and colleagues reported a case of cholangiocarcinoma discovered during pregnancy and compared their case to 9 other similar cases in the literature where cholangiocarcinoma was diagnosed during pregnancy , 7. FurtIn conclusion, our case demonstrates a unique presentation of a rapidly progressing intrahepatic cholangiocarcinoma in an individual with cystic fibrosis who was 5 months postpartum undergoing lung transplantation workup. It remains unclear as to whether the presentation of her advanced disease was due to immunosuppression of pregnancy or delayed diagnosis because of the pregnancy itself. Our case, supported by the literature, suggests that a heightened clinical awareness for the increased risk of gastrointestinal cancers, beyond colon cancer, and in pregnancy, is warranted in CF.


Inflammatory or allergic sinonasal polyps are characterized by extensive vascular growth and ectasia with deposition of pseudoamyloid in 5% of cases. Angiomatous nasal polyp (ANP) is a relatively rare benign lesion, which may be misdiagnosed as a benign or malignant tumor. The characteristic pathological features of ANP are expanded angiogenesis, accumulation of extracellular amorphous eosinophilic substance, and atypical stromal cells. This report aimed to outline the histological differential diagnosis of ANP.Through a full histopathological examination, we studied biopsies and resected specimens from five patients who were diagnosed with ANPs, including one with facial deformity. Gross findings showed that tumors were firm in consistency, lobulated on the surface, and lined by partially ulcerated mucous membrane. Light microscopy showed clusters of widened, thin-walled blood vessels among congo red-negative eosinophilic substance with an area of necrosis and irregular stromal spindle cells. Presence of endothelial cell myofibroblasts were confirmed by electron microscopy and immunohistochemistry.This is a report of four cases which showed extreme examples of ANPs that was completely resected by endoscopic sinus surgery for all patients. A full histopathological examination is recommended to confirm the possible differential diagnoses for a better management plan. Inflammatory sinonasal polyps are assorted into main five variants: edematous, glandular, fibrous, cystic and angiomatous polyp. Clinically, patients may experience a gradual obstruction of the nasal cavity, loss of smell sensation, nasal discharge or epistaxis. Also, the mass could cause exophthalmoses, and visual disturbances. Angiomatous nasal polyps (ANPs) are relatively rare benign lesions recognized by extensive vascular growth and ectasia, with scant of inflammatory cells and abundant extracellular fibrin -4. CliniFurthermore, inflammatory nasal polyps could be misdiagnosed histologically with other tumors including papilloma, squamous cell carcinoma, nasal lymphoma, and other soft tissue neoplasms in the nasopharynx , 6. Few In this article, we aim to represent clinical, radiological and pathological features of ANP of the paranasal sinuses that resulted in bone erosion and expanded to the right nasal cavity and the ipsilateral pterygopalatine fossa. Case 1A 53 years old female with known history of hypertension presented with a chief complaint of left progressive nasal obstruction and nasal discharge for a duration of six months. She had no aggravating or alleviating factors and had no otological symptoms , postnasal drainage, epistaxis, facial numbness, diplopia and any impairment in her visual acuity.On clinical examination, the patient was generally stable. However, she had a grade two left nasal polyp, a (R/L) deviated nasal septum, and normal cranial nerve exam. There were no other remarkable findings on her ENT examinations. Her laboratory investigations were within normal limits. A CT was performed which showed left homogenous soft tissue mass in the nasal cavity extending to the left maxillary ostium Figure . SeptoplCase 2A 31-year-old female presented with bilateral nasal obstruction and nasal discharge for more than one year. These symptoms were progressive in their course and without aggravating and relieving factors were present. This was associated with headache and anosmia with no history of epistaxis, visual loss, diplopia or otological symptoms. The patient reported no fever, weight loss or change in appetite. The patient underwent functional endoscopic sinus surgery for bilateral anterior and posterior ethmoidectomy, bilateral maxillary antrostomy and bilateral sphenoidotomy seven years ago. Biopsy was obtained with no obvious histopathological findings.On clinical examination, the patient was conscious, oriented, and hemodynamically stable. By local examination, bilateral nasal polyp and hypertrophy inferior turbinate were noted. No visual loss or diplopia was present. Remaining ENT examinations were unremarkable. Her laboratory investigations were within normal limits. A CT scan showed opacification of all paranasal sinuses, without any bony erosion. Endoscopic Sinus Surgery was performed and polyp was sent for histopathology department. Follow up of this case after five years shows disease recurrence Figure .Case 3A 32-year-old male came with a chief complaint of bilateral nasal obstruction and nasal discharge for more than one year; these symptoms were progressive. No aggravating or alleviating factors were present. The condition was associated with headache. The patient reported no history of epistaxis, visual loss, diplopia or otological symptoms. However, the patient was known to have a history of allergy to dust. Two years ago, the patient had similar surgical intervention as case 2, and also with no obvious histopathological finding. On examination, bilateral nasal polyp were found. No visual loss or diplopia was reported and other ENT examinations were unremarkable. His laboratory investigations including full blood picture, coagulation profile, kidney function test, and hepatic panel were within normal limits. A CT scan showed partial opacification of ethmoid sinuses without any bony erosion  were within normal limits. A CT scan indicated a soft tissue mass in the left nasal cavity showing marked hypervascularity expanding the left maxillary ostium, extending anteriorly to the left check, and to the posterior naris on the left side including the left side of pterygoplatine fossa.Under general anesthesia, a biopsy was taken and sent as frozen section because of a high suspicion of malignancy. After the result of histopathology, the lesion was excised without any complications and had no index of recurrence throughout the follow-up time. In the operation, the tumor was restricted in the left nasal cavity and reached about 6 cm in diameter with a little pedicle that emerged via the middle meatus.Results of Light Microscopy The roofs of the lesions were ulcerated with areas lined by pseudostratified ciliated respiratory epithelium with squamous metaplasia. Thin-walled vessels were detected in almost all cases; they were irregular in shape and encircled by extensive Congo red negative eosinophilic extracellular material. Fibrin thrombi are also noted in some blood vessels lumina. The stroma was dispersed with irregular spindle cells, and the vascular spaces were covered by endothelial cells Figure . The atyImmunohistochemical StudiesEndothelial cells were demonstrated in the vascular-looking channels and papillary-like projections through immunoperoxidase stains for endothelial markers . Further, desmin and smooth muscle actin were positively stained in the normal sized walls and the cytoplasm of rare atypical and non-atypical stromal cells. However, in the widened and cavernous vascular spaces, they were negatively stained. In addition, cytokeratin stains were positive in the epithelial part only.ANPs are deemed infrequent lesions, representing only 4-5% of whole nasal polyps . BatsakiIn angiectatic polyps, presence of scattered atypical pleomorphic spindle cells (Myofibroblasts) in the stroma is frequent; however, they are less frequent in sinonasal polyp , 15. In Majority of the ANPs originate in the maxillary sinus and expand across the choana and into the nasopharynx , 16 ProgOn the other hand, Yfantis et al. mentioned that in certain cases, angiomatous nasal polyps may lead to massive bone erosion and/or nasal bleeding. The incidence of any of the aforementioned symptoms may consolidate the clinical doubt of the presence of malignant lesion . GeneralDai et al. and Yfantis et al. observed that the most noteworthy pathological characteristics of ANP were the great quantity of blood vessels, evidence of intravascular thrombosis, the massive necrosis of the lesion, and the extravasation of the blood composition into the nearby stroma , 4. The In summation, the characteristic pathological features of ANP are protracted vascular growth, collection of extracellular amorphous eosinophilic substance, and atypical stromal cells , 4. In oAngiomatous polyps are rare lesions which entail additional concerns and efforts for diagnosis and surgical intervention. Angiomatous nasal polyps are rapidly developing and aggressively expanding lesions that may turn malignant prior to surgical management. The clinical and imaging features of these lesions are not specific hence they usually boost the consideration of multiple neoplastic lesions in the differential diagnosis.


Leiomyosarcomas are rare malignant tumors which originate from smooth muscle cells and very seldom give rise to intracerebral metastases. Nearly all cases of intracranial metastases stem from leiomyosarcomas of the uterus. We present a 61-year-old Caucasian man who developed multiple intracranial and extracranial metastases from leiomyosarcoma of the right forearm, diagnosed and treated 9 years before the current presentation.The Caucasian patient presented to the emergency department due to a progressive hemiparesis on the left side. Magnetic resonance imaging scans of the neurocranium showed multiple intracerebral masses with perifocal edema. One of these was located in the right parietal lobe, corresponding to the hemiparesis. The patient underwent microsurgical complete resection of the parietal mass and was subsequently subjected to further radiotherapy. Histopathological studies revealed metastasis of the former leiomyosarcoma.Leiomyosarcomas represent a rare entity of mesenchymal tumors. Intracerebral metastasis of these tumors is even less frequent. This case shows the importance of long-term follow-up in patients with leiomyosarcoma. Leiomyosarcomas are rare mesenchymal tumors which originate from smooth muscle cells. These tumors occur in all age groups and account for 10% of all soft tissue sarcomas  polymerase 1 (PARP-1) inhibitors, which are already used in breast, ovarian, and prostate cancer, could establish a novel therapeutic pathway. Nevertheless, as for the aforementioned Akt signaling inhibition, further mechanistic evaluations of these signaling pathways are awaited.The management of intracranial sarcoma metastasis includes surgical resection, radiotherapy, stereotactic radiosurgery, and chemotherapy. Since complete resection of all intracranial metastases was not possible in this case, the patient underwent whole-brain irradiation after neurosurgery, but unfortunately did not agree to the proposed chemotherapy (doxorubicin and ifosfamide).Doxorubicin, which was introduced already in the 1970s, still remains the most effective chemotherapy for advanced sarcomas. Novel combination therapies have recently been explored in clinical trials. The combination of olaratumab, a platelet-derived growth factor receptor alpha (PDGFRα) antibody, with doxorubicin showed no additional benefit in the phase III ANNOUNCE trial .Pazopanib and regorafenib, which represent multikinase small-molecule inhibitors, are further promising agents. Here, the phase II REGOSARC trial showed enhanced progression-free survival in patients with advanced non-adipocytic sarcomas under regorafenib treatment, along with an acceptable safety profile .The prognosis of leiomyosarcoma depends on the histological grade, tumor size, and tumor depth. These factors apply to all leiomyosarcoma, regardless of uterine or non-uterine localization . Tumor sCerebral metastasis of a non-uterine leiomyosarcoma is an extremely rare disease. The treatment has to be based on a multidisciplinary, individualized decision, but the mainstay remains neurosurgical resection, adjuvant radiotherapy, and subsequent administration of doxorubicin.Additional file 1: Figure S1. Positron emission tomography-computed tomography imaging showing an increase in FDG uptake in the tumor (arrow) of the right forearm


Background: Burkitt lymphoma (BL) is a non-Hodgkin’s B-cell tumor that can be classified into three variants, based on clinical characteristics and epidemiology: endemic, human immunodeficiency-related and sporadic. Oral sporadic BL is quite an unusual entity, with the gastrointestinal trait being often the first site of appearance.Clinical finding: A 15-year-old patient that presented a symptomatic swelling of the right maxilla, unsuccessfully treated as a primary endodontic disease, displaying solid tissue on CT scan, “starry sky” pattern on oral biopsy, multifocal bone and lymph node uptake on PET.Diagnoses, interventions, and outcomes: A diagnosis of stage IV BL was formulated; Rituximab was then administered for three months according to Inter-B-NHL ritux 2010 protocol and CYM (cytarabine and methotrexate) chemotherapy. The patient was followed-up for three years, with no recurrence.Conclusion: It is important for general dental practitioners to suspect a malignancy in the differential diagnosis of unresponsive odontogenic infections in young healthy patients. Endemic BL, which was the first to be described as a “sarcoma” by Denis Burkitt in African children 60 years ago2, occurs mostly among six-year-old males of equatorial Africa and Papua New Guinea, mainly within the maxillofacial complex, with an estimated 50% of cases detected in jaws or facial bones. Sporadic BL is typically observed in Western countries, with a European incidence of 2.2 cases per million, affecting mostly young adult Caucasian males, frequently within the abdomen, particularly in the ileocecal trait1.Burkitt lymphoma (BL) is a mature, aggressive high-grade B-cell non-Hodgkin’s lymphoma, which occurs in three distinctive subtypes: endemic (African), human immunodeficiency-related and sporadic (nonendemic)The oral cavity is rarely the first site of onset. In this report, we describe the peculiar case of an IV-stage BL arisen as a maxillary swelling in a 15-year-old girl, misdiagnosed at first as endodontic disease.In November 2015, a healthy 15-year-old female was referred to our Department with chief complaint of dull pain on the permanent maxillary right second molar, on whom the general dental practitioner had already performed root canal treatment and administration of two grams of amoxicillin daily for one week, to no avail. Further questioning revealed an associated hypoesthesia of the right lower lip.Conventional oral examination showed an overall swelling of the gingiva surrounding the painful teeth and extending to the right palatal mucosa , whereasDue to the unreliability of OPT and the unresponsiveness to the combination of root canal and antibiotic treatments, a contrast-enhanced CT scan of the maxillo-facial district was urgently required, revealing solid tissue with high uptake in the right maxilla, with osteolysis and disruption of the floor of the right maxillary sinus .in situ hybridization (FISH) revealed IgH/Myc translocation in 70% of the nuclei, being negative for IgH/BCL2 and IgH/BCL6 translocation. CISH staining for EBV-encoded RNA (EBER) transcript was widely positive  chemotherapy, between December 2015 and March 2016.In May 2016, a PET/CT scan showed a complete remission of the disease; clinical oral examination showed the complete remission of gingiva-palatal enlargement. Since then, the currently 18-year-old patient appears to be in good health.13. To the best of our knowledge, this is the first detailed report of oral sBL in a teenage patient in Italy.Oral sporadic Burkitt lymphoma (sBL) is a rare clinical entity among children, with few case reports published to date18 show an infrequent involvement of the mouth as the first site, accounting for 3%15, 9.5%16, up to 16%18 of all sBL. Clinically, oral sBL acts as a fast growing, rapidly expanding tumor, which may cause dull, toothache-like pain, teeth malposition, occlusal precontact with subsequent difficulty in chewing and open-bite;6 in some cases sudden teeth loosening can be elicited11.Case-series available worldwide3, causal periodontal treatment11 or even teeth extractions10. Moreover, when tonsil is the primary site, dysphagia and obstructive sleep apnoea may occur12.Such behavior can easily be mistaken for an odontogenic disease, leading to the general dental practitioner (GDP) to administer antibiotics and perform unnecessary procedures, such as root canal treatments of the teeth closest to the swollen or painful area, as the presented case and a previous report have shown4, or can reveal an ill-defined isolated9 or multifocal11 radiolucency with loss of lamina dura and periodontal ligament13, sometimes displaying a worrisome “floating teeth” appearance7. Thus, cervicofacial CT is mandatory to properly assess the degree of disruption of the cortical bone and the status of the neighbouring naso-orbital areas11. On the other hand, an 18-FDG PET may be needed either to fulfil the diagnostic work-up when suspecting a widespread lymphoma10, or used as a monitoring tool, especially in the first 12 months after diagnosis, where relapse can be more frequently encountered12. Ultimately, an in-depth histologic analysis, comprehensive of immunophenotyping showing CD20 positive clones and FISH revealing of IgH/Myc translocation will be the key to unravel the differential diagnosis with a wide range of diseases which may share overlapping symptoms and clinico-radiological signs, such as osteomyelitis, benign odontogenic epithelial neoplasms, other types of lymphomas, Langerhans cell histiocytosis, Ewing’s sarcoma, osteosarcoma, chondrosarcoma, neurosarcoma and fibrosarcoma10Panoramic radiograph can be either unremarkable, as in our case and a similar report19.Being a rapidly growing tumor with a very high replicative activity, BL proves to be particularly sensitive to cytotoxic chemotherapeutic agents, in particular in a polychemotherapy regimen alongside rituximab, an anti-CD20 monoclonal antibody responsible of a further increase of the five-years survival rate, which can amount up to 90–100% or early stagesTherefore, since prognosis is closely related to the earliness of diagnosis, the specialists and GDPs should consider the possibility of a BL in case of rapid swelling of the jaws in young healthy individuals, especially if nonresponsive to traditional antibiotic therapy.An informed consent form was signed by the patient’s mother, in order to obtain permission for photo usage and for the use and publication of the young patient’s data.All data underlying the results are available as part of the article and no additional source data are required. This is a well written paper that presents a very interesting case report that concisely describes and addresses a rare clinicopathologic entity in the realm of lymphoma, in particular oral lymphomatous proliferation. The case details were presented clearly and concisely. The relevant literature and studies were nicely summarized and addressed. All in all, I support a decision "to accept".I confirm that I have read this submission and believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard. The Authors describe a case of Sporadic Burkitt lymphoma in an adolescent patient with an "non conventional" oral presentation. They well describe the imaging appearence, the morphology and immunophenotyping as well as molecular characteristics of this entity, highlinting the importance of the differential diagnosis in the oral pathology in young patients.I confirm that I have read this submission and believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard.


Intussusception in infants and children represents a relatively usual challenge for the pediatric surgeon. However, the incidence of lymphoma of the small intestine or inflammatory myofibroblastic tumor, acting as a lead point for invagination, are rather rare. We hereby present two cases of secondary intussusception, with the aforementioned lead points. Pediatric lymphoma represents almost 15% of all pediatric malignancies . NeverthHistory: an eight-year-old boy with prior uncomplicated medical history was admitted to our pediatric surgery emergency department complaining about abdominal pain. In particular, symptoms began 2 days prior to his admission to our faculty, having intermittent characteristics and located particularly to the mesogastrium. The patient had no other accompanying symptoms, such as fever, nausea or vomiting. He had a normal defecation during the day of his admission.Physical examination: physical examination revealed tenderness in the periumbilical area and right iliac fossa, with no palpable mass or distension. He also presented with vital signs within normal limits, according to his age. Laboratory exams at the emergency department resulted in leukocytosis with neutrophilic shift , with normal procalcitonin levels. An abdominal ultrasound examination and erect abdominal X-ray were not indicative for certain pathology. Due to his clinical condition, an admission to our department was proposed to his parents, for surgical investigation.Management: the patient was guided to the operating theatre and an exploratory laparoscopy was initiated. Inspection of the peritoneal cavity revealed a macroscopically inflammatory appendix, with serosal hyperaimia. A laparoscopic appendicectomy was performed. Furthermore, when the small intestine was inspected, an ileoileal intussusception was also noted, approximately 40cm from the ileoceacal valve . The young patient recovered easily after the surgery. Up to date, 2.5 years postoperatively, follow-up laboratory studies and Computed Tomography (CT) scan are normal.Intussusception represents one of the most frequent causes of acute bowel obstruction in infants and toddlers and the second most common cause of acute abdominal pain in infants and preschool children after constipation. As Gross stated in 1953: “there are few illnesses in which the clinical history and physical findings are more suggestive of the correct diagnosis” [The pathologic anatomy of each intussusception follows a certain path: the proximal invaginated bowel (intussusceptum) carries its mesentery into the distal recipient bowel (intussuscipiens). Then, mesenteric vessels are angulated and compressed between the layers of the intussusceptum, causing local oedema, venous compression, congestion and stasis, leading to the outpouring of mucus and blood from the engorged intussusceptum, the classic red currant jelly stool. If left unabated, intussusception leads to bowel necrosis, in most cases about 72 hours after the beginning of the invagination .Regarding the cause of intussusception, up to 95% of the cases are categorised in the so-called “idiopathic” type, which in fact represents an invagination of a thickened bowel wall lymphoid tissue (Peyer patches) acting as an actual (non-pathologic) lead point. The incidence of intussusception from a pathologic lead point in infants and children ranges from 1.5% to 12% . Most coHowever, secondary intussusception with a pathologic lead point of non-Hodgkin lymphoma (NHL) tumor is a rather rare condition , althougRegarding IMT, it is a neoplasm of myofibroblastic spindle cells with inflammatory infiltration. It mostly occurs between 2-16 years of age. Such lesions have been reported in a variety of organs including the colon, lung, bladder, spleen, breast, pancreas, liver, spermatic cord, prostate, peripheral nerves, soft tissue and orbit. Small bowel localizations are particularly rare  and usuaA clinical pediatric surgeon has to raise high suspicion in terms of some rather rare causes of secondary intussusception, especially regarding cases of benign or malignant tumors acting as a lead point, as such cases require different postoperative follow-up than usual.Ethical considerations: patients´ parents provided written informed consent for their children´s surgical operation and agreed with also written consent for the use of history, laboratory-radiological exams and images without identifying information.


An astrocytoma is a subclassification of glioma, with primary spinal manifestations accounting for less than 10% of all spinal cord tumors, with the majority encompassing low-grade features. It is even more uncommon for such lesions to demonstrate intracerebral metastasis. We report such an occurrence in a 39-year-old female who initially presented with an intramedullary and intradural mass from T10-L1, as well as secondary metastasis to the mesial right temporal lobe and cerebellum upon clinical follow-up. Surgical resection of the spine and subsequent temporal lobe biopsy confirmed high-grade glioma. Given the rarity and poor prognosis of spinal gliomas with cerebral metastasis, we also summarize all previously reported cases to date. We recommend that physicians maintain an index of suspicion for spinal gliomas in young patients with cord compression related symptoms outside the event of traumatic injury. Primary spinal cord tumors are uncommon, constituting approximately 2-10% of all Central Nervous System (CNS) tumors, with the overwhelming majority (70%) of such lesions being of low-malignant potential . DespiteWe present the case of a patient with a low-grade astrocytoma of the thoracic spinal cord who then developed additional cervical and thoracic spread, followed by high-grade transformation upon cerebral metastasis. We discuss the pertinent clinical, operative, and histopathological findings. We also performed a comprehensive review of the literature of all reported cases of primary spinal astrocytomas with brain parenchyma extension.A 38-year-old female presented with a three-month history of progressive low back pain radiating to bilateral lower extremities with paraparesis and radiating shock-like pain with Valsalva. In addition, she reported gait instability, falling episodes, and bilateral foot drop (left worse than right) with muscle laxity. She denied any bowel or bladder incontinence. Physical exam demonstrated gait instability, impaired toe walking, impaired heel walking, impaired tandem gait, and an antalgic gait. The bilateral upper extremity strength was 5/5. Lower extremity exam demonstrated 4/5 psoas strength bilaterally, 3/5 quadriceps strength bilaterally, 2/5 (left), and 1/5 (right) tibialis anterior strength, 2/5 (left) 1/5 (right) extensor hallucis longus, 3/5 gastrocnemius strength bilaterally. All upper extremity deep tendon reflexes were 2/4 bilaterally. Patellar and ankle reflexes were 4/4 bilaterally. There were no changes in sensation among upper and lower extremities bilaterally. Babinski and Hoffman signs were absent. Finger to nose coordination was unremarkable.Magnetic resonance imaging (MRI) demonstrated a large intradural intramedullary mass extending from T10-L1  were recorded. Baseline MEPs were abnormal, consistent with the preoperative presence of paraparesis on admission. A vertical midline incision was made overlying the thoracolumbar junction, and a complete laminectomy from T10-L1 was performed. The spinal cord was abnormal, demonstrating an enlarged component, consistent with an intramedullary tumor. The surface of the spinal cord appeared discolored, and a midline myelotomy was performed. A large, firm, rubbery, pale mass that encompassed the overwhelming majority of the spinal cord was encountered, internally debunked, and sent for frozen pathology, which confirmed a World Health Organization (WHO) grade II diffuse astrocytoma. The tumor demonstrated extensive spread, and several areas were encountered that did not demonstrate a definable plane. The myelotomy was extended from the bottom of T10 down to L1. Serial MEPs were performed throughout the bulking process, which eventually indicated the loss of distal right lower extremity MEP and possible left lower extremity MEP loss. Given the clear intraoperative findings that this invasive tumor was without clear margins, and the evidence of MEP change, the surgical resection remained as a subtotal resection. The wound was then irrigated with copious amounts of antibiotic irrigation and hemostasis obtained. The dura was then closed in a watertight fashion and copiously irrigated with 3 liters of pulsatile antibiotic irrigation. During the closure of the case, there was an improvement in the left lower extremity MEPs and a questionable slight improvement in the right distal lower extremity MEPs. Cerebrospinal fluid (CSF) cytology was negative.Following surgical resection, physical exam demonstrated 5/5 upper extremity muscle strength bilaterally. Lower extremity physical exam demonstrated 5/5 (left) and 4/5 (right) psoas strength, 5/5 (left) 3/5 (right) quadriceps strength, 0/5 dorsiflexion strength bilaterally, 1/5 (left) and 0/5 (right) extensor hallucis longus strength, and 0/5 (left) and 4/5 (right) gastrocnemius muscle strength. New-onset paresthesia was reported from L4 to distal right lower extremity, as well as from L1 to distal left lower extremity. Due to social considerations, the patient began a seven-week course of chemoradiation two months postoperatively. This included six cycles of adjuvant temozolomide. Over the next two months, the patient reported progressive lower extremity weakness, dizziness, fatigue, poor appetite, and depressed mood. Repeat MRI of the spine revealed a new expansile cord lesion with heterogeneous T2 signal within the thoracic spinal cord spanning from T1 to T5 Figure . In addiA stereotactic needle biopsy of the right temporal lobe was performed, with subsequent histopathology demonstrating WHO grade III anaplastic astrocytoma without microvascular proliferation or necrosis Figure . ModeratThe patient survived for only eight months post-diagnosis.Literature reviewThe PubMed database and all major neurosurgery journals were searched during June of 2020 using the keywords “glioma”, “astrocytoma”, "glioblastoma", “spine”, and “cerebral extension”, alone or in combination to obtain articles fitting the inclusion and exclusion criteria. The inclusion criteria were high-grade astrocytomas or glioblastomas involving the spine with secondary brain parenchyma metastasis.To date, 96 cases of primary spinal gliomas with secondary intraparenchymal manifestation have been reported since 1908, including this case  of metastatic origin. Primary spinal tumors are most often intramedullary and are rare even within the realm of CNS neoplasms, accounting for less than 10% of all primary CNS lesions . Among tWhile the isolated occurrence of these tumors is certainly uncommon, subsequent metastasis of these lesions into the intraparenchymal space is even rarer.A striking feature of our particular case was the supratentorial dissemination of the spinal lesion, which gave a progressive and multifocal picture. Of note, although it is certainly possible that the brain metastases are due to CSF dissemination and seeding during the initial surgery, it is also possible that the tumor disseminated postoperatively, given the aggressiveness and invasiveness seen with the H3K27M mutation profile.The H3K27M mutation is an important consideration regarding high-grade gliomas, as the prognosis of H3K27M gliomas remains poor, and even less favorable than Glioblastoma Multiforme (GBM), with few options in treatment. One clinical trial worth noting has demonstrated efficacy and an exceptional safety profile targeting gliomas with such mutations via a selective dopamine receptor D2/3 antagonist (ONC201), regardless of age or tumor location . Our assessment highlights several patterns regarding the presentation and treatment of these tumors. First, primary spinal high-grade gliomas most commonly occur in young males  with a mean age of 20 years old (95% CI 17.0 to 23.28). This is in stark contrast from primary intracranial gliomas, which have a mean age of diagnosis of 60.4, as well as the overall incidence of primary spinal cord tumors overall . Second,In sum, given the consistent constellation of symptoms portraying cord compression, and the unique age of presentation of high-grade spinal gliomas, we recommend providers maintain an index of suspicion for such patient presentations in practice when instances of traumatic injury as a potential cause are not apparent. Lastly, because secondary cranial metastasis is possible  we recommend routine brain imaging surveillance of all patients with primary high-grade gliomas.Primary high-grade gliomas of the spine with cerebral metastasis is a rare occurrence with a poor prognosis and most commonly presents in a unique patient population. Although surgical resection is commonly attempted, tumor recurrence and three-year mortality still remain close to 100%. Following our evaluation of the data, we recommend that providers maintain an elevated index of suspicion for spinal high-grade gliomas in young persons who present with cord compression related symptoms outside the realm of traumatic injury and that all patients with such tumors undergo aggressive surveillance imaging to monitor for secondary cranial spread.


Vascular malformation can present as nerve compression syndrome in middle age due to slow lesion growth, after first appearing in childhood. In a case of vascular malformation involving the peripheral nerve, surgical resection may cause uncontrollable hemorrhage and further nerve compression. Therefore, a more conservative approach is needed in such cases.A 48-year-old woman complained of pain and paresthesia of the fingers, and was diagnosed with a large vascular malformation expanding from the proximal forearm to the palm on MRI. Because of the size of the lesion and involvement of soft tissue, resection had a risk of major hemorrhage and incomplete removal. Thus, carpal tunnel release was performed as a less invasive procedure, and was effective for reducing pain while avoiding the risks associated with resection.Our case indicates that a vascular lesion is a possible cause of nerve compression in the carpal tunnel. Finger numbness and pain were caused by this nerve compression, but the lesion itself was not painful. The median nerve was separate from the venous structure, and the case was diagnosed as an extraneural type.Symptoms that develop in middle age due to an extraneural vascular malformation in the carpal tunnel can be treated successfully using carpal tunnel release, instead of more invasive resection of the lesion. In carpHere, we report a case in which vascular malformation presenting as median nerve neuropathy was diagnosed in middle age due to slow growth of the lesion. Resection had a risk of inadequate excision and subsequent persistent expansion, whereas carpal tunnel release had the advantage of improving the symptoms without affecting the lesion, as well as being a less invasive procedure. This case is reported in line with SCARE criteria .2The patient was a 48-year-old woman who had occasionally felt pain and swelling in her left forearm since childhood, but she had not visited a clinic because the pain resolved naturally. She had no medication history or surgical history, and no relevant family history. She had felt increasing pain and numbness in her left finger for about 1 month, and she was referred to our hospital after the pain increased further.Her forearm were broadly distended  and the Imaging findings showed no abnormalities on plain X-rays. MRI revealed hyperintensity on T2-weighted imaging and suggested that the vein had expanded from the proximal forearm to the palm . The sizPregabalin was given for pain, but there was no improvement. Resection of the vascular malformation was considered to be invasive and to have a risk of incomplete resection. Therefore, carpal tunnel release was performed as an alternative treatment. A skin incision was made from the distal forearm to the palm, and the flexor retinaculum was opened. The veins were present on the ulnar side of the median nerve  and were3This case shows that vascular malformation can present as a nerve compression syndrome in middle age, after appearing in childhood. A less invasive operative treatment of carpal tunnel release was effective for reducing pain. Vascular anomalies are divided into malformations and tumors such as hemangioma, which both occur in infancy and childhood. Hemangiomas display rapid early growth, and resection may be the only treatment that can control growth . SeveralThe presence of a tumor or a tumor-like lesion should be considered as a possible, but rare, cause of nerve compression syndrome. A review of 541 cases of nerve compression syndrome found only three caused by vascular tumors, including a case with an intraneural invasive vascular tumor that could not be removed, resulting in unchanged paresthesia . In a reIn our case, finger numbness and pain were caused by median nerve compression at the carpal tunnel. The lesion itself was not painful. MRI showed relatively large expansion of the lesion from the proximal forearm to the palm through the carpal tunnel. In the carpal tunnel, the median nerve was separate from the venous structure, and the condition was diagnosed as an extraneural type. Resection of the lesion may have resulted in incomplete excision with subsequent persistent expansion and uncontrollable hemorrhage. Therefore, we performed carpal tunnel release, which is less invasive and does not have these risks, and this procedure improved the symptoms. The lesion remained untouched, and long-term follow-up will be important to evaluate the need for additional interventions if there is further expansion.The symptoms of an intraneural vascular malformation may not be improved by carpal tunnel release. Furthermore, surgical excision may be most effective when the lesion is limited to the hand and upper extremity. Conservative treatment such as sclerotherapy and embolization can also be considered if surgery is inadequate, but these procedures also have a risk of nerve injury or ischemia because of the agent and blood loss .4In this case, vascular malformation in middle age presented as pain due to expansion of the lesion from the proximal forearm to the palm through the carpal tunnel. Less invasive treatment of carpal tunnel release was effective for reducing pain, as an alternative to resection of the lesion.None of the authors have a conflict of interest.This study did not receive any external funding.A case report does not require approval from the Ethics Committee in our hospital.Written informed consent was obtained from the patient for publication of this case report and accompanying images.Morisaki S and Fujiwara H wrote the manuscript and prepared the figures. Morisaki S was the main surgeon, and is a specialist in hand surgery. Tsuchida S and Oda R contributed to the conception and design of the study and critically revised the manuscript. All authors read and approved the final manuscript.Name of the registry: researchregistry5540.Shinsuke Morisaki.Not commissioned, externally peer-reviewed.


Hepatic toxocarosis is caused by the dog´s roundworm, Toxocara canis. Responsible for an eosinophilic inflammatory syndrome causing liver damage that can be detected on ultrasound, computed tomography and sometimes magnetic resonance imaging. We report the case of a nine-year-old child, living in countryside, with a notion of cohabitation with canids. He presented a digestive symptomatology revealed by abdominal pain, with a hemeosinophilia in the hemogram. The etiological assessment of hyper eosinophilia objectified a positive Toxocara canisserology. The imaging assessment in search of digestive visceral lesions, found multiple heterogeneous hypoechogenic areas, poorly defined, scattered in the liver. On the abdominal CT scan, its areas appear of unenhanced density and low density and better visible after injection of contrast product. This observation reveals that imagery, although not very specific, helps in the assessment of liver damage from digestive toxocarosis. Toxocara: it is a parasitic deadlock [Helminths and protozoa can determine various digestive manifestations, by the presence of the parasite in the digestive tract. He is the cause of the visceral larva migrans syndrome testifying to the migration of second stage nematode larvae through the tissue of the human viscera. Toxocarosis is a zoonosis corresponding to the infestation of humans by larvae of roundworms belonging to the genus deadlock -3. Cosmodeadlock . Two maideadlock . We repotoxocaraserology was positive with 43 μL of Toxocara canis IgG, with confirmation of the presence of anti-toxocara canis antibodies. An abdominal ultrasound (larva migransdigestive toxocara. The child was started on albendazol tablet therapy, but was non-observant.He is a nine-year-old patient, living in a rural area, with no particular pathological history. He consulted in hematology for subacute, poorly systemic abdominal pain of moderate intensity, without notion of digestive disorders, evolving for 15 days, in an apyretic context without alteration of the general condition. The physical examination noted hepatomegaly, with a slight helpless peri-umbilical pain without dermatological lesion or neurological disorders. The hemogram showed an increase in leukocytes at 34,400/μL, with increased eosinophilia at 23,000/μL, motivating the etiological research of the increase of eosinophils, through the realization of a thoracic radiography and an examination of the stools, not finding anomalies. Only the trasound , performtrasound ; allowedtoxocaraeggs. In adults, cases of hepatic toxocariasis have been recently reported and this tendency is so much to be attributed to the particular habit of adults to want to eat undercooked meat [Toxocaraanti-excretors/secretors [Toxocariasis is known to be a common disease in preschoolers because they play with canines, especially dogs, on clean grounds and are therefore more likely to ingest soil contaminated with ked meat -4, 6. Inked meat -4, 6. Whked meat . Biologiecretors . These ctoxocara on imaging reflect the granulomas or abscesses surrounding live or dead larvae and or eosinophilic inflammation of the liver parenchyma [Digestive lesions due to renchyma -4, 6-8. renchyma , 6-7. Thet al. in South Korea [Toxocara sp.uses anthelmintics [This description of the semiology on the CT scan of the hepatic lesions of toxocarosis, was found in our patient with the CT scan complement. Jae Houm Lim th Korea , performlmintics -8.Toxocara canis larva migrans syndrome; result in imaging, by the presence of multiple, ill-defined ranges of variable texture, depending on the type of imaging used, in relation to inflammatory to eosinophilic granulomas. It is therefore important that any radiologist masters these types of lesions so as not to ignore them.Liver damage due to


Endosalpingiosis in the lymph nodes of the mesocolon is very rare. We reported a case with appendiceal endometriosis who had endosalpingiosis in the lymph nodes of the mesocolon that was found during laparoscopic ileocecal resection.The patient was a 44-year-old woman who had visited a physician for fever, bloody stool, and abdominal pain 1 year earlier. She was diagnosed with ulcerative colitis on colonoscopy, and symptoms improved with oral treatment. A colonoscopy performed 2 months after diagnosis detected a hard, 20-mm submucosal tumor (SMT) in the cecum. On abdominal contrast CT, an intensely stained mass, including a low-density region, was observed in the cecum. A boring biopsy was performed after mucosal resection of the cecal SMT at our hospital, but diagnosis could not be made. Since the possibility of a malignant lesion could not be ruled out, laparoscopic ileocecal resection was performed. In the resected specimen, a 29 × 27 × 21-mm mass was present in the appendicular root. On histopathological examination, appendiceal endometriosis and endosalpingiosis in the lymph nodes around the ileocolic artery were observed. The postoperative course was favorable, and the patient was discharged 7 days after surgery.Differentiation of endosalpingiosis in lymph nodes in the mesocolon from lymph node metastasis of adenocarcinoma is important in patients with an abdominal mass. Endosalpingiosis is considered as ectopic benign epithelial lesion histologically similar to tubal epithelium, and many lesions are observed in either the peritoneum, great omentum, urinary bladder, or pelvic and para-aortic lymph nodes –3. When The patient was a 44-year-old woman who visited a physician for fever, bloody stool, and abdominal pain 1 year earlier. She was diagnosed with ulcerative colitis on colonoscopy, and symptoms improved with oral treatment. A colonoscopy performed 2 months after diagnosis detected a hard, 20-mm submucosal tumor (SMT) in the cecum, and the patient was referred to our hospital for additional examination.Her past medical history included polyostotic fibrous dysplasia that was followed for 2 years and ulcerative colitis treated orally for 1 year. There was no particular familial medical history, and her pregnancy/delivery history was 2 gravida 1 para.The findings of her first examination by us were as follows: height, 164 cm; body weight, 41 kg; body temperature, 36.5 °C; blood pressure, 102/64 mmHg; and pulse, 84/min. No jaundice or anemia was noted in the palpebral conjunctiva. The superficial lymph nodes were not palpable.Her blood chemistry showed no abnormal finding other than a mild increase in a tumor marker, CEA (≦ 3.0 ng/dl), to 3.4 ng/dl.On abdominal CT, an intensely stained mass with a 30-mm diameter, including a low-density region, was observed in the cecum. No lymph node swelling was noted around either the cecum or ileocolic artery Fig. .Fig. 1OSMT with a 20-mm diameter was noted in the cecum on colonoscopy. A boring biopsy was performed after resection of the mucosa using a snare, but a tumorous lesion was not observed  was maintained at about 1–2 ng/dL after surgery.Endosalpingiosis was initially used in 1930 by Sampson to describe the tubal epithelial lesions observed in scar tissue after salpingectomy , and it Unlike endometriosis, subjective symptoms are absent in many cases of endosalpingiosis because neither the growth or inflammation is consistent with the menstrual cycle. Fewer cases are reported by surgeons and gynecologists compared with the number of reports on endometriosis. Endosalpingiosis and endometriosis can be distinguished by normal H&E staining. The histological characteristic of endosalpingiosis is the presence of ciliated cells, non-ciliated pale secretory cells containing pale cytoplasm, and peg-shaped intercalated cells in which the cytoplasm, including the nucleus, is mostly present in the upper cellular region in gland ducts –3. In maIn our patient, the level of the tumor marker CEA was mildly elevated before surgery. Regarding endosalpingiosis, there has been no report indicating any association with CEA and any association between endometriosis and CEA is negative . The couSince endosalpingiosis is recognized as a 1–2-mm gray cystic or papillary lesion, its differentiation from a disseminated malignant tumor and metastasis is difficult. It is also difficult to differentiate it from metastasis of malignant tumor when it is present in a lymph node, as histological investigation is necessary. Dallenbach-Hellweg summarized and reported points of differentiation between malignant tumor and endosalpingiosis .Including our patient, cases with endosalpingiosis in mesentery lymph nodes are often complicated by intestinal endometriosis , suggestWe treated a patient with endosalpingiosis in the lymph nodes in the mesocolon. At present, many points remain unclear with regard to the details of endosalpingiosis. It is expected that surgeons will increasingly recognize endosalpingiosis and increase the number of case reports, which will enable close further investigation.


Hemangiomas are usually found in cutaneous or mucosal layers, less than 1% of hemangiomas develop in skeletal muscles. Intramuscular hemangioma (IH) in the head and neck areas is relatively infrequent, accounting for 15% of IH. Most of them are identified as a benign mass, and rapid changes in size or internal bleeding are rare.A 60-year-old female patient presented with a 2-week history of sudden onset posterior neck pain. There was no neurological deficit except limited neck motion due to pain. The palpable mass was noted on the paraspinal muscles of cervicothoracic junction, which was located midline to left side portion with tenderness.Magnetic resonance imaging demonstrated a round shaped, multi-lobulated, and well-defined mass lesion (4.1 × 2.6 × 0.9 cm) embedded from the inter-spinous space of T1-2 to the left paraspinal muscles. The lesion was iso-intense on T2-weighted images (WI), iso- to slightly low-intense on T1-WI, heterogeneous enhancement of intra- and peri-mass lesion on contrast-enhanced T1-WI. Vascular structures presented as signal voids were identified internally and around the mass lesion. Histological examination revealed a mixed-type hemangioma.The mass was removed completely including some of the surrounding muscles where boundaries were unclear between the mass and surrounding muscles with ligation of peritumoral vessels. Dark-brown colored blood was drained from the ruptured tumor capsule during the dissection. There was no bony invasion.The preoperative symptoms improved immediately after the operation. There is no residual or recurrence lesion by the 15-months follow-up.IH with hemorrhagic transformation in the head and neck is extremely rare. In the case of intramuscular tumors accompanied by a sudden onset of severe acute pain, we recommend considering a differential diagnosis of IH with hemorrhagic transformation. Complete resection of the tumor mass including surrounding muscles is required to prevent recurrence. IH are usually found as an incidental benign palpable mass, but accurate diagnosis before surgery is difficult due to non-specific symptoms, deep anatomical positions, and a low frequency of occurrence.,7Hemangiomas are the most common benign endothelioma in infancy, accounting for approximately 7% of soft tissue tumors and are usually found in cutaneous or mucosal layers.As most IH develop congenitally and are identified as an incidental benign mass, mass growth or hemorrhagic change is uncommon. We report a rare case of IH with hemorrhagic transformation arising from the paraspinal muscles of the posterior neck, around the cervicothoracic junction.2Patient has provided written informed consent for publication of this case report and any accompanying images.A 60-year-old female patient presented with a 2-week history of a sudden onset natured posterior neck pain around the cervicothoracic junction and aggravation over the last week. The patient had a history of lipoma removal on the back 2 years ago as well as hypertension and diabetes. On neurological examination, she demonstrated no definite neurological deficits except limited neck motion caused by pain. The palpable mass was noted on the area of paraspinal muscles, which was located midline to left side portion with tenderness. The mass had a fixed in nature and was 4.1 × 2.6 × 0.9 cm in size.Preoperative magnetic resonance imaging (MRI) demonstrated a round shaped, multi-lobulated, and well-defined mass lesion embedded from the inter-spinous space at the T1-2 level to the left paraspinal muscles. The lesion was iso-intense on T2-weighted images (WI), iso- to slightly low-intense on T1-WI, heterogeneous enhancement of intra- and peri-mass lesion on contrast-enhanced T1-WI. Vascular structures presented as signal voids were identified internally and around the mass lesion . There are thin-walled vascular spaces and hemorrhage.   without an invasion of surrounding nerve components. Thrills, bruits, pulsations, and skin color changes are uncommon unless tumor has invaded large vessels.IH was first reported by Liston in 1843. IH is a non-metastatic congenital benign tumor, and approximately 50% of cases remain undetected until related symptoms are present.MRI is a useful method for providing better detection and delineation of the extent of IH. MRI findings suggestive of IH are1)hyper-intensity on both T1- and T2-WI2)serpiginous pattern, septated-striated high signal channels and curvilinear areas of low intensity consistent with vascular spaces3)focal heterogenicities representing areas of thrombosis, fibrosis or calcification4),10 In these aspects, a retrospective review of preoperative MRI findings from our patient shows iso- to slight hyper-intensity on T1-WI, iso-intensity on T2-WI, and homogenous enhancement of the entire lobulated mass on contrast-enhanced T1-WI with intra- and peri-tumoral signal voids. We think that lower signal intensities on T1- and T2-WI compared to the typical MRI features of IH may be associated with subacute hemorrhage inside the mass. However, preoperative accurate diagnosis of IH with hemorrhagic transformation is difficult considering the low incidence of IH itself and accompanying hemorrhagic change.adjacent focal muscular atrophy. Capillary hemangiomas account for 50% of all IH and 68% of head and neck IH. Capillary hemangiomas grow faster than the other types, with a high recurrence rate of around 20%. Cavernous hemangiomas consist of a large number of thin-walled vascular channels lined by a single layer of endothelial cells, which are separated by fibrous connective tissue, whereas capillary hemangiomas have no fibrous septa and smaller vascular lumens.,12 Cavernous hemangiomas account for 26% of the head and neck IH with a recurrence rate of about 9%. Mixed-type hemangiomas account for only 5% of IH in the head and neck but have the highest recurrence rate of 28%. The IH of our case was identified as a mixed-type by histological examination, requiring a sufficient follow-up period for recurrence.Hemangiomas can be subdivided into capillary, cavernous, and mixed-type, depending on whether capillary and cavernous predominant. The recurrence may be related to the microscopically infiltrative pattern into the surrounding muscular tissue and minor arterial feeder vessels. Surgical margin and tumor size have been identified as independent factors predicting local recurrence, and incomplete resection can lead to an increasing rate of recurrence. The optimal treatment is complete resection including the muscles surrounding the tumor and controlling the feeding vessels to prevent recurrence. In a large-sized IH, preoperative angiography can be helpful to reduce intraoperative bleeding by the embolization of feeding arteries. In our patient, the tumor was removed completely including some of the surrounding muscles using the en bloc method with ligation of peritumoral vessels. MRI conducted after the 15-month follow-up period revealed no residual or recurrence lesionIH generally does not show natural regression and malignant change, but increased size and development of symptoms such as pain require surgical treatment. Local recurrence rate was reported at approximately 9% to 28% after surgical resection due to the infiltrative growth pattern. These processes are prompted by an increase in angiogenic factors such as vascular endothelial growth factors (VEGF) and matrix metalloproteinases (MMPs) and a decrease in anti-angiogenic factors.,20 Tumor growth is promoted by high blood estrogen levels during puberty, pregnancy, oral contraceptive use, and androgen treatment. Hemorrhagic transformation of a hemangioma is rare. There are some cases involving hemorrhage in hepatic and vertebral hemangiomas have been documented in the literature, but to date, none have been reported in IH. In the cases of hepatic hemangioma, voluntary rupture occurs in 1% to 4%. Most of them are giant hepatic hemangioma (6–25 cm) with a mortality rate of 75%. In our case, the size of the hemangioma was relatively small and there were no specific factors related with tumor growth.While the pathophysiology of hemangiomas is not clearly defined, it is believed that abnormal angiogenesis may be involved.4IH in the head and neck is uncommon, and hemorrhagic transformation is extremely rare. In the case of intramuscular tumors accompanied by a sudden onset of severe acute pain, we recommend considering a differential diagnosis of IH with hemorrhagic transformation. Complete resection of the tumor mass including surrounding muscles is required to prevent a recurrence.Conceptualization: Ikchan JeonData curation: Ikchan JeonFunding acquisition: Ikchan JeonMethodology: Dongwoo Yu, Ikchan Jeon, Joon Hyuk ChoiResources: Dongwoo Yu, Ikchan Jeon, Joon Hyuk ChoiSupervision: Ikchan JeonWriting – original draft: Dongwoo YuWriting – review & editing: Ikchan Jeon


EWSR1-CREB1.Primary pulmonary myxoid sarcoma (PPMS) is an extremely rare lung sarcoma that is characterized in most cases by recurrent balanced chromosomal translocation t leading to the oncogenic fusion gene EWSR1-CREB1 gene fusion in this tumor. During 38 months of follow-up, the patient was alive with no clinical or radiological evidence of recurrence or metastasis.We report a case of PPMS with molecular confirmation using fluorescence in situ hybridization (FISH) and DNA sequencing in a 45-year-old female patient. Computer tomography (CT) scanning revealed a peripheral circumscribed solid mass of 2.1 × 2 cm in the right lung superior lobe. Histologically, the tumor cells ranged from stellate, polygonal to chondrocyte-like or physaliferous-like, forming reticular network of delicate lace-like cellular strands and cords in abundant myxoid stroma. The tumor cell immunophenotype was positive for vimentin, EMA and negative for CK-pan, TTF-1, CAM5.2, S-100, calponin, SMA, desmin, ALK, CD31 and CD34. Molecular analysis demonstrated PPMS is a rare low-grade sarcoma with distinct histological and genetic features. We add another case to the literature of this rare tumor and report for the first time occurrence of chondrocyte-like and physaliferous-like tumor cells in this tumor, thus enriching its morphologic and cytologic spectrum. EWSR1-CREB1 fusion in most cases [EWSR1-CREB1 gene fusion. We report another case of PPMS harboring the EWSR1-CREB1 gene fusion confirmed by molecular method, with review of the literature.Primary pulmonary sarcomas are extremely rare with prevalence of about 0.2% . They cost cases .. To thest cases , among wA 45-year-old woman was referred to our hospital for physical checkup. She was asymptomatic with no pulmonary obstructive symptoms or pneumonia. The patient was a non-user of alcohol and tobacco products. General physical and laboratory examination was unremarkable. Chest computed tomography (CT) revealed a 2.1 × 2 cm peripheral solid mass in the right lung superior lobe featuring moderate heterogeneous enhancement , and specific gene fusion transcripts by reverse transcription-polymerase chain reaction (RT-PCR). Primers for the RT-PCR were located in exon 7 of EWSR1 (5’TCCTACAGCCAAGCTCCAAGTC3’) and in exon 7 of CREB1 (5’GTACCCCATCGGTACCATTGT3’). FISH showed a clear separation of red and green signals within a single tumor cell, demonstrating the presence of a EWSR1 gene rearrangement  to the 3′ region of CREB1 (exon 7)  are reviewed and summarized in Table EWSR1-CREB1 gene rearranged low grade myxoid sarcoma of the pulmonary artery [EWSR1-CREB1 gene fusion, one showed EWSR1 rearrangement but not fusion with CREB1 (case 16) [ESWR1-CREB1 gene fusion. This makes a 75% positive rate for EWSR1-CREB1 fusion, 79% for EWSR1 rearrangement, and a negative rate of 21% for ESWR1-CREB1 fusion or ESWR1 rearrangement in the 24 cases where genetic study was performed in the reviewed series of PPMS. These findings are broadly similar to the previously reported EWSR1-CREB1 gene fusion rate of 70% [EWSR1 and/or CREB1 gene rearrangement rate of 89% [EWSR1-CREB1 fusion gene in 7 PPMS, with the fusion loci located in exon 7 of EWSR1 and exon 7 of CREB1 in 6 of the 7 cases. One other case showed fusion loci in exon 7 of EWSR1 and exon 8 of CREB1. There was no histological difference in the tumors bearing the different gene fusions. Furthermore, one case in this series with no EWSR1-CREB1 fusion died from brain metastasis, while another case bearing the EWSR1-CREB1 fusion of the same series had kidney metastasis but was alive and well after 3 years [EWSR1-CREB1 fusion developed metastasis to the contralateral lung, but remained disease free 72 months after removal of the lung metastasis. It appears that PPMS with EWSR1-CREB1 fusion fares better than those without EWSR1-CREB1 fusion. However, whether presence of EWSR1-CREB1 fusion connotates better prognosis in PPMS requires further study.Nicholson et al. first described a rare primary pulmonary myxoid sarcoma in 1999 as a novel low-grade malignant myxoid endobronchial neoplasm . FurtherR1-CREB1 , which hR1-CREB1 . To dateR1-CREB1 –11,with 1 fusion , 3, 5–10EWSR1-NR4A3、TAF15-NR4A3 or TFG-NR4A3 gene fusion [EWSR1-CREB1 fusion gene, allowing distinction from EMC. Differentiation between PPMS and angiomatoid fibrous histiocytoma (AFH), which may occur in the lung [EWSR1-CREB1 fusion gene, they differ in morphology. PPMS is composed of cords and clusters of spindle, stellate, ovoid, and in our case chondrocyte-like and physaliferous-like tumor cells arranged in a reticular pattern within prominent alcian blue positive myxoid stroma. In contrast, AFH comprises sheets and islands of spindle to epithelioid cells with bland ovoid vesicular nuclei and abundant eosinophilic cytoplasm within loose stroma. PPMS has more abundant myxoid stroma and lacks the prominent peripheral cuff of lymphocytes usually present in AFH. Moreover, desmin is expressed in 50% of AFH, which is not present in PPMS. Furthermore, ALK expression is common in AFH, which is negative in PPMS. Finally, AFH may show other fusion genes, such as EWSR1-ATF1 and FUS-ATF1, both of which are absent in PPMS. PPMS should be also be distinguished from myoepithelial tumors, which can also arise in the lung [EWSR1 rearrangements. Myoepithelial tumors are immunohistochemically positive for CK, p63, SMA, calponin and S-100 protein, which are negative in PPMS. Pulmonary microcystic fibromyxoma (PMF) are composed of bland spindle to stellate cells with uniform nuclei widely spaced within fibromyxoid stroma which is alcian blue positive and hyaluronidase sensitive [EWSR1-CREB1 gene fusion, distinct from PPMS. Another differential diagnosis is inflammatory myofibroblastic tumor (IMT) which may possess myxoid stroma and inflammatory infiltrates. The stellate tumor cells in IMT are immunophenotypically positive for SMA, desmin and ALK, which are negative in PPMS. Since the physaliferous-like tumor cells in PPMS exhibit cytoplasmic bubbles, they can potentially be confused with lipoblasts. PPMS thus needs to be differentiated from low grade primary or metastatic myxoid liposarcoma [DDIT3 gene, which is not present in PPMS. The various differential diagnoses and their features are summarized in Table PPMS should be differentiated from extraskeletal myxoid chondrosarcoma (EMC), which can also arise in the lung , 14. Hise fusion , and maythe lung , is morethe lung  with endensitive . Howeverosarcoma . Myxoid EWSR1 rearrangement [EWSR1-CREB1 gene fusion [EWSR1-CREB1 fusion or wild type EWSR1 may portend poor clinical outcome [Although most patients treated by adequate surgery had no local recurrences, three patients developed metastasis within a follow up period from 4 months to 15 years. The metastatic sites included the contralateral lung , the lefangement  or EWSR1e fusion , 7 may h outcome , 11.EWSR1-CREB1 fusion gene. Our case adds to the literature of this rare tumor and enriches its morphologic and cytologic spectrum with occurrence of chondrocyte-like and physaliferous-like tumor cells. Early diagnosis of PPMS requires high index of suspicion in pulmonary myxoid tumors and is based on clinical features, histological and molecular studies. The clinical behavior of this tumor is still uncertain due to its rarity and limited follow up of reported cases. There are no reliable histological or clinical features to predict prognosis and outcome of this tumor.PPMS is a rare low-grade sarcoma that occurs mostly in middle age women with most harboring the characteristic


Adult intussusception (AI) is both a challenging and rare diagnosis, with predisposing factors including malignancy, surgery and infection to name a few. Transient jejunal intussusception is a subset of AI which is usually diagnosed radiologically, with diagnostic laparoscopy utilised to determine whether a malignant cause is identifiable and subsequently treatable.We present the case of a previously healthy 36-year-old male diagnosed with transient jejunal intussusception on computed tomography after presenting with abdominal pain. Blood tests on admission were normal apart from polycythaemia. His only significant history was that of chronic anabolic steroid use. He had a subsequent normal gastroscopy and colonoscopy with diagnostic laparoscopy demonstrating thickening of the small bowel. Histopathological analysis of the intraoperative specimen was normal. The patient improved and was discharged with no further complications.This case highlights the potential association between anabolic steroid use resulting in polycythaemia, and AI or transient jejunal intussusception, along with further validating a conservative approach in the management of AI in patients deemed to be low risk of malignancy on pre-operative evaluation. Reported frequently in children, intussusception is a rare presentation in adults, with an incidence of 2–3 per 1000000 and a predominance in females . The preTransient jejunal intussusception is a rare pathological condition and is usually a diagnosis of exclusion, with risk factors including Crohn’s disease, celiac disease, inflammation and adhesions . Compute2A 36-year-old male presented to the emergency department of a metropolitan hospital complaining of abdominal pain, described as intermittent and crampy. He denied previous episodes of similar pain in the past. He denied any previous medical history with nil previous abdominal surgery reported. On further history he admitted to the chronic use of oxandrolone, an androgen and anabolic steroid. On examination, he was haemodynamically stable, with periumbilical tenderness detected on abdominal palpation. Examination was otherwise remarkable.Blood samples were obtained from the patient which revealed an elevated haemoglobin at 164 g/L and haematocrit of 50 %. CT scan of the patient’s abdomen demonstrated mild diffuse small bowel wall thickening, with two separate areas of apparent short segment jejunal intussusception without evidence of obstruction . After rHistopathological assessment revealed normal villous architecture and inflammatory cells within normal limits. No evidence of dysplasia or malignancy was reported.With the apparent resolution of the small bowl intussusception and abdominal pain, the patient was discharged home. He was advised on the cessation of regular anabolic steroid use and on routine follow-up, the patient reported significant improvement in abdominal pain with no further presenting episodes.3Intussusception if characterised as the invagination of a proximal segment (intussusceptum) into an adjacent distal segment of bowel (intussuscipiens). This presented case highlights the clinical phenomenon of transient jejunal intussusception in a previously healthy male. Transient intussusceptions have not been well reported in the literature, and its pathophysiology is poorly understood. Steroid-induced polycythemia can cause hyperviscosity and consequent mechanical vascular obstruction. These physiologic changes put patients with a history of chronic anabolic steroid use at an increased risk for ischemic events . It has The clinical presentation of intussusception is variable but generally marked by abdominal pain and signs of bowel obstruction. Some patients may also present with nausea, vomiting, bleeding, changes in bowel movements, constipation or bloating . PersistGiven the non-specific clinical presentation and the vast differential, choice of imaging modality is critical to arriving at a timely diagnosis. CT scan remains the gold standard to obtain a diagnosis and elucidate a possible aetiology, with a reported sensitivity and specificity of 80 % and 100 % respectively ,21. The en-bloc resection in intussusception is based on a theoretical risk of venous embolization of potential malignanct cells on manipulation of the affected segment of bowel, and the risk of bowel perforation resulting in peritoneal seeding of malignant cells. There is a definitive lack of evidence to support this theory. A recent meta-analysis reported small bowel malignancy being the cause of intussusception in 22.5 % and 36.9 % of enteric and ileocolic intussusceptions respectively, thus supporting the approach for laparoscopic reduction and subsequent resection [In general, intraoperative management of AI is mostly done by en bloc resection without attempts at reduction if there are any doubts regarding a benign aetiology preoperatively on history, examination and radiological assessment  . While sesection . The autesection .4Transient jejunal intussusception, a rare form of AI, is usually idiopathic in nature and diagnosed on radiological analysis. This report highlights a potential relation of transient jejunal intussusception to chronic anabolic steroid use. In addition to this, this report provides further evidence for a conservative approach in the management of AI in patients with clinical and radiological features which are contrary to the diagnosis of malignancy.The authors declare no conflicts of interest in the production of this case report.This case report had no sponsors.This case report is exempt from ethical approval by our institution.Informed consent was obtained from the patient for publication of this case report.Dr Marie Shella De Robles and Dr Robert O’Neill were involved in the writing of the presented case report. Associate Professor Christopher Young and Dr Marie Shella De Robles were involved in the clinical care of the patient.This is not a ‘first in humans’ report, so it is not in need of registration.Associate Professor Christopher Young.Not commissioned, externally peer-reviewed.


Chloromas are an atypical cellular infiltrate of immature granulocytic cells that can occur specially in patients with acute myelogenous leukemia (AML), but can be present in nonleukemic patients. Its clinical course will be dependent on its size and location, from asymptomatic to simulating a malignant gastrointestinal neoplasia. Definitive diagnosis is made upon an immunoprofile that is similar to that present in the blasts and precursor cells of acute myeloid leukemia. Endoscopic and CT images are variable being only part of the protocol panel. Treatment is the same as to AML, but surgery and radiation must be used in order to maintain low relapse and better overall survival. Chloromas, granulocytic sarcoma or myeloid sarcoma (MS) is an extramedullary solid tumor of immature granulocytic cells, known to occur commonly in patients with acute myelogenous leukemia (AML), affecting 2.5%-9.1% of cases, but it can be present in nonleukemic patients .Described more than two centuries ago by Burns, clinical and pathological diagnosis of chloroma is still highly challenging . Any atyWe present the case of an intestinal chloroma.A 22-year-old female patient, with no significant medical history, was referred to our hospital because of growing bilateral cervical masses, odinophagy, disphony, unquantified fever, and nocturnal diaphoresis for 40 days. Three days later, petechiae were noted on her limbs, with minor transvaginal bleeding. An out of hospital bone marrow aspiration reported AML.At physical examination, the only finding was bilateral cervical lymphadenopathy, being painful on the left side. The pulse rate was at 100 bpm, and there were no significant alterations with the rest of the vital signs. Laboratories evidenced anemia with 7 g/dL hemoglobin (range 12-15.5 g/dL), leucopenic with 0.65 x 10⁹/L of leukocytes (range 4.5-11 x 10⁹/L), profound neutropenia with 0 neutrophils , thrombocytopenic with platelets 22,000/µL .Hyper-CVAD was initiated. She presented fever of 38.5°, cultures were taken as well as antibiotics initiated. She had poor evolution with persisting fever and the need of a platelet concentrate transfusion. As cultures were negative, a thoracoabdominal tomography was done with findings of inflammatory process in peritonsyllar tissue, concentric thickening of 6 mm in ascending colon and slight increase in the density of pericolic fat Figures -2, neutr Some 10 days postadmission, she had gastrointestinal bleeding that caused persistent hypovolemic shock (with need of six blood transfusions in 24 h). Urgent surgical intervention was performed with findings of three intraluminal masses, two at 60 cm of ileocecal valve and one in the cecum; performing right hemicolectomy and ileal resection with terminal ileostomy with no complications.She had an insidious evolution in the ICU. Some 24 h after first surgical intervention, secondary to persistent mixed hypovolemic and septic shock, a thoracoabdominal tomography is made where free abdominal fluid is evidenced. She is reintervened in the operation room, with surgical drainage of 1000 cc of citrine fluid and no other significant findings. Hours later, she died in ICU.Surgical pathology results revealed acute colitis with microperforations, lymphatic nodes with presence of blasts, without evidence of malignancy.In histopathological sections we observe infiltration of the entire colonic wall by cells of a wide cytoplasm and evident nucleoli Figure , which aChloromas display predominantly green color, due to the presence of MPO, as described earlier 1853) by King 3 by King, and finClinical presentation is varied and it is highly dependent on tumor size and location. Tumors located in gastrointestinal tract can manifest in several ways like:· Stomach: Epigastralgy, hemorrage, perforation.· Small intestine: Recurrent abdominal pain, subocclusive episodes, chronic diarrhoea.· Large intestine: Simulated colorectal cancer.Endoscopic aspects includes polipoid tumors, exofitic masses, stenosis, and ulcerations. This is why precise diagnosis is difficult .The CT features of granulocytic sarcoma of the bowel are variable. Lesions appear as an intraluminal or exophytic polypoid mass, as bowel wall thickening, or as a combination of these manifestations. The enhancement patterns of these lesions are variable. Therefore, leukemic lesions of the bowel itself cannot be differentiated at CT from lymphoma or other neoplastic conditions or inflammatory bowel diseases .In chloroma, several markers have been employed for identification of myeloid origin of tumor infiltration. MPO and chloroacetate immunostaining with antibodies are important for the identification of chloroma, bearing in mind that precursor cells of AML and myeloblasts in chloroma have similar antigen profile .With conventional light microscopy, differential diagnosis of the pathological specimen may be difficult, particularly in the case of lymphoblastic, Burkitt’s, or diffuse large B-cell lymphoma or even a nonhematological malignancy .As this leads to a completely different treatment and prognosis, immune-phenotyping is mandatory. In 92 patients, Pileri et al. demonstrated that the most commonly expressed marker was CD68/KP1 (100%), followed by MPO (83.6%), CD117 (80.4%), CD99 (54.4%), CD68/PG-M1 (51.0%), and CD (43.4%) [Antic et al. suggested that the minimum panel of immunohistochemical markers should include MPO, CD68, CD43, lysozyme, CD33, CD34, and CD117 in order to establish a definitive diagnosis [There are no consensus guidelines or protocol regarding chloroma treatment. This will depend on several factors including: location of disease, whether it is a relapse or an initial diagnosis, age of patient, and performance status. Current treatment regimen in patients with chloroma or isolated chloroma is conventional AML chemotherapeutic protocols with or without radiotherapy. Surgery can be considered before systemic treatment when rapid debulking, symptomatic relief is needed or to confirm diagnosis. Hematopoietic stem cell transplantation (HSCT) should also be considered in relapsed or refractory patients or even as a consolidation treatment .Although relatively uncommon, chloroma should be suspected in every patient with myeloid neoplasia history with added symptoms. Even though the clinical manifestations are variable, image should always be part of the diagnostic arsenal in order to establish therapeutic regimen that must include surgical, radiation, and chemotherapy. Treatment guidelines based on prospective studies of this rare manifestation are required to determine the best combination of actual therapies, as early detection and treatment will lead to a better overall patient's survival and quality of life.


The fact that some patients with Monro foraminal stenosis may be asymptomatic increases the possibility of underdiagnosis, so we consider it a condition that radiologists should be aware of, mainly taking into account the fact that the diagnosis of this entity is usually radiologic3 and the potential complications associated with treatment.Bilateral Adult Idiopathic Oclussion of Foramen of Monro is a rare entity, with less than 22 cases published in the literature so far, all of them symptomatic. A 42-year-old female with clinical history of hypothyroidism and symptoms of right temporomandibular joint (TMJ) dysfunction was scheduled for a temporomandibular MRI scan, in which an enlargement of lateral ventricles was incidentally observed. She was then referred to the Neurology Department, where no signs of intracranial hypertension were found, nor history of headache, vomiting, dizziness, incontinence or gait disturbance, and a whole brain MRI study for hydrocephalus assessment was requested.2T weighted sequences, and there were no significant brain herniations, except a subtle herniation of the precuneus, probable associated with focal tentorial hypoplasia , inflammatory (ventriculitis), tumoral lesions  or tumor-like lesions and cysts (e.g. neurocysticercosis or colloid cyst) should be excluded, and the administration of i.v. contrast is usually necessary.Enlargement of the lateral ventricles with slit-like third ventricle must raise the suspicion of idiopathic foramina of Monro stenosis. Nevertheless, secondary obstructive cause at or near the foramina should be firstly ruled out, since they are more frequent: hemorrhagic (2 depending on the unilateral or bilateral nature of the occlusion and the presence or absence of membranes. The most frequent symptoms found in these patients are headache and those associated with intracranial hypertension,1 none of them present in our case. We consider that patients with less severe or compensated stenosis and few or no symptoms are at risk of being underdiagnosed, mainly if radiologists don’t report adequately this disease when these patients have a brain CT or MRI done for other reasons. In order to avoid unnecessary invasive treatments, neurologists and neurosurgeons should also be aware of the existence of these asymptomatic cases, taking into account the possibility of complications associated to endoscopic approach, such as lesion of the fornix.Foramen of Monro stenosis is a rare entity, with only a few cases published so far, all of them symptomatic. Four different types of stenosis have been described,When a Monro foramina stenosis is suspected, MRI with i.v. contrast administration and three-dimensional high resolution sequences should be the technique of choice, as it allows a correct diagnosis and a better visualisation of potential membranes, and gives guidance for future surgical or endoscopical procedures.Adult idiopathic bilateral stenosis of the Foramina of Monro is a rare cause of enlargement of the lateral ventricles.Stenosis of the Foramina of Monro should be considered when a biventricular hydrocephalus with slit-like third ventricle is found, even in asymptomatic patients.Radiological diagnosis of stenosis is done by exclusion after causes of obstruction, such as tumors or cysts, have been ruled out.MRI with i.v. contrast agent administration and high resolution three-dimensional sequences should be the technique of choice for its diagnosis, to exclude inflammation or tumor, and to try to assess the presence of potential membranes as well as guide the endoscopic treatment, when indicated.


This study presents a rare case of pseudoaneurysm mimicking a tumor on the back, with no history of fever, trauma, or surgical intervention. In which no identifiable symptoms or warning signs were present. This pseudoaneurysm arises from intercostal arteries and segmental arteries. Both of which, to the limit of our knowledge, have not been discussed before.This study was done using chart and literature review. We present a case of a 46-year-old man with a known case of left-sided intracranial hemorrhage due to hypertension and an old cerebrovascular accident. The patient has a positive history of hypertension, which could have attributed to the pseudoaneurysm; however, he has no history of vascular disease otherwise. The patient reports of back swelling and intermittent back pain for the past 3 years. On MRI, the mass showed a pulsating pattern around it. It also showed a layering effect because of different wall thicknesses and enhanced patterns, and the enhancement ratio was increased. In addition, it showed flow artifacts with T1 hyperintense areas because of associated thrombus and blood products. These changes noted on the MRI prompted the team to do a color Doppler study to confirm the presence of an aneurysm and if present, to do a CT angiography. The color Doppler showed a turbulent flow, that is, there was a bidirectional pulsatile flow which further confirms the presence of a pseudoaneurysm. Spine CT with contrast showed a right paraspinal lesion at the T9-T11 level. It had contrast enhancement and flow inside, consistent with a partially thrombosed aneurysm. The CT also showed evidence of bone remodeling in the adjacent thoracic vertebrae. The patient opted for spinal vascular emobilization and vascular sheath removal. The right and left intercostal arteries were selected at the level of left and right T4, left T8, bilateral T9, and bilateral T10.Differentiating between pseudoaneurysms and osteogenic tumors is essential to target later investigations accordingly. In addition, if pseudoaneurysms are left untreated, they could cause bony erosions of the vertebra, which lead to compression fractures. They can further compress the adjacent neurovasculature, which worsens the morbidity. The mechanism and causation of pseudoaneurysm is not exactly known. However, it has been proposed that they result from a gradual growth that occurs because of constant arterial pressure and the development of a reactive fibrous capsule.3 Moreover, they usually occur after a penetrating injury, blunt trauma, or an endovascular procedure.Aneurysms are localized swellings in the walls of an artery, usually because of a weak spot. They can be true, with all three vessel walls, or pseudoaneurysms, where an opening in one of the vessel walls allows leakage of blood through its wall.3Differentiating between pseudoaneurysms and osteogenic tumors clinically is challenging because they share similar signs and symptoms. They also share a similar presentation on a CT scan because they both appear as a lesion or a mass. Moreover, CT scans show bony erosions that further support the overlap between them and osteogenic tumors Figure . This ov4 Pseudoaneurysms in the spine can occur because of an iatrogenic, traumatic, neoplastic, infective, vasculitic, or inflammatory etiology. They can also occur in several sites, such as knees and elbows.4 The site of the pseudoaneurysm usually suggests the underlying cause .Pseudoaneurysms represent a pulsating encapsulated hematoma in contact with the lumen of a ruptured vessel. They usually occur after a disruption in the arterial wall continuity, which leads to blood dissecting into the surrounding soft tissues without including the arterial wall in the aneurismal sac.This study presents a rare case of pseudoaneurysm mimicking a spinal tumor, with no history of fever, trauma, or surgical intervention. This pseudoaneurysm has risen idiopathically from intercostal arteries and segmental arteries, both of which, to the limit of our knowledge, have not been described before.We present a case of a 46-year-old man living in a remote rural area with a history of an old left-sided intracranial hemorrhage. The patient had lingering neurologic deficit from his previous stroke and presented with a report of intermittent back pain for the past 3 years. The pain first started as pressure-like in nature, 2 of 10 in intensity, not radiating, and occurring once every 2 to 4 weeks. Two weeks before presenting at our institution, the patient experienced gradual increase in his pain symptoms, the pain was continuous and pressure-like in nature (6 to 7 of 10), and radiating to the upper and lower spine without limb involvement; however, it was controlled with simple analgesia. One week later, the patient sought medical treatment in an outside facility where tramadol was needed to control the pain (dosages unknown). The patient denied any limb weakness, radicular pain or numbness, and urinary or stool incontinence at the time. The patient was referred to our institution's spine unit for assessment. His referral included an MRI (without contrast) and a medical report suggesting an osteogenic tumor.On examination, the patient was vitally stable. He had a normal gait; however, the patient could not walk in a tandem gait or on tiptoes/heels alone. It was noted that the pain increases with axial rotation of the abdomen and lateral bending. Bilateral lower limb examination showed normal motor function and reflexes. Sensory examination revealed hypoesthesia to pinprick and intact vibration sense in the left lower until midthigh, all of which were present since his previous brain hemorrhage. The examination was otherwise normal.The plan was to control the pain, monitor distal neurovascular status, rule out brucellosis by a brucella titer, and repeat erythrocytes sedimentation rate and C-reactive protein tests, in addition to doing an acid-fast bacilli test and culture for tuberculosis.2).However, a secondary review of his MRI by a senior musculoskeletal radiologist leaned toward a possible vascular etiology  study Figure  was recoNephrology consultation was made because the patient was expected to undergo multiple contrast-enhanced studies and/or treatment modalities. After proper optimization and clearance, we proceeded with CT angiogram that showed a right paraspinal mass at the T9-T11 level Figure . It had The procedure was discussed, along with its indication, alternatives, and potential complications, and informed consent was obtained. Using a standard technique, right femoral artery access was obtained. An aortic angiogram was performed, which showed the posterior intercostal arteries at the levels of T8, T9, and T10. A large aneurysm was noted, arising from the midpart of the right T10 segmental artery with a narrow neck, pointing toward the spinal column with an estimated sac of 3 to 4 cm. A tributary to the anterior spinal artery coming from the left T10 segmental artery was also noted. Embolization of the pseudoaneurysm was performed by using Onyx 18  Figure . PostembPostprocedure, the patient was stable and discharged the next day. His pain completely resolved postembolization. Patient was pain free due to the pressure relief that was pressing on his vertebra in association with the pain medications he was discharged on; the patient was discharged on tramadol, acetaminophen, omeprazole, and nifedipine. He was seen in clinic 14 days later with no new symptoms or reports. Follow-up imaging was planned for his 6-week visit, but he was lost to follow up.56789101112 Liu et al recently published a literature review of iatrogenic vascular injuries postspinal surgery. They included 20 case reports totaling 26 patients, of which, 12 patients sustained a pseudoaneurysm during their clinical course. Cappuzzo et al recently published a case report with a literature review pertaining to a delayed traumatic case resulting in pseudoaneurysms in the spine. In their report, they discuss six studies with 47 patients in total. Most of the cases were because of penetrating trauma (60%).1213141516 There are several other reports in the literature describing posttraumatic spinal pseudoaneurysms with most patients manifesting their symptoms within 1 year.17181920Pseudoaneurysms occurring in the spine are thought to be a rare event but more reports are being published recently describing multiple etiologies. Iatrogenic and posttraumatic causes seem to be the more common types reported in the literature.4 We found two cases in the literature where a pseudoaneurysm was initially thought to be a soft-tissue tumor using imaging studies. Both case reports discussed a pseudoaneurysm occurring in the distal femur. Bregowda et al presented two patients with a rather short clinical history of pain and swelling (6 and 3 months). Both cases in the report were initially diagnosed as osteogenic sarcoma of the femur. Both cases underwent a comprehensive multidisciplinary review, and a vascular etiology was confirmed before any interventions.3 Albert et al paint a similar picture with their report, initially pursing a diagnosis of soft-tissue sarcoma because of MRI findings. However, contrary to the previous report, the patient was eventually taken for an open soft-tissue biopsy which was complicated by a massive hemorrhage. A repair was ultimately done during the same surgery, and the patient did not suffer any lasting consequences. No cases were found in the literature describing a similar presentation in the spine. However, the reports discussed above offer a cautionary reminder of the possible detrimental outcomes of misdiagnosing pseudoaneurysms.Cases of pseudoaneurysms mimicking tumors in their presentation remain exceedingly rare with a missed diagnosis presenting a possible devastating outcome.The symptoms noted in the patient described in this report were initially thought to be due to a tumor or an infectious process. Contrast imaging was not preferred because of his marginal kidney function initially. However, Doppler US was the key diagnostic modality that swayed the diagnostic process in the right direction. His history of vascular events, although unclear because of a lack of proper documentation, was also a factor that assisted in pursuing the correct diagnosis. Unfortunately, our patient was lost to follow up after his initial visit to the clinic; however, his pain and swelling resolved on day one postprocedure with no neurologic deficits.This report, similar to others in the literature, further emphasizes the difficulty in diagnosing pseudoaneurysms and the need for a multidisciplinary approach in such cases. A vascular cause of spinal masses must be ruled out if imaging is not able to diagnose with certainty because of its high morbidity and possible mortality.