Dunbar syndrome is a rare anatomical abnormality characterized by the extrinsic compression of the celiac trunk by the median arcuate ligament (MAL). Though it is rarely misdiagnosed, the clinical diagnosis may be difficult, especially after complex visceral surgery such as esophagectomy.A 62-year-old male patient with a squamous cell carcinoma of the distal esophagus, placed under trimodal treatment  presented with abdominal pain refractory to analgesics, anti-spasmodic, opioids, and neuronal celiac plexus ablation in the late post-operative period. He was diagnosed with extrinsic celiac trunk compression based on abdominal angiotomography findings. Retrospectively, similar images were found in conventional abdominal tomography at pre-operative staging, but this time, the patient had only dysphagia. After surgical treatment of MAL, the patient had total relief of pain and symptoms.Abdominal pain after complex surgical procedures is very frequent and its investigation is mandatory, even more after refractory clinical management. Dunbar syndrome is related to ambiguous abdominal pain. It is uncommon and its diagnosis with angiotomography is accessible.Vascular disorders should be investigated in cases of abdominal pain after complex surgical procedures. In lateLow abdominal implantation of the median arcuate ligament (MAL) is a rare condition. When this is associated with celiac trunk (CT) compression and abdominal pain, it characterizes the Dunbar Syndrome .Asymptomatic patients with low implantation of MAL may have changes in vascular dynamics after esophagectomy, due to the ligation of vascular trunks and may evolve with ischemic symptoms.This case has been reported in line with the SCARE  criteria2A 62-year-old male patient with progressive dysphagia and weight loss was diagnosed with distal squamous cell carcinoma of the esophagus staged as T3N2 pre-operatively. The CT tomography revealed a discreet compression though he was asymptomatic.He was placed on trimodal therapy . On follow-up, he developed postprandial dysphagia, vomiting, and epigastric pain. He was initially treated with prokinetics and proton pump inhibitors, but his symptoms were not relief.A complete medical examination was performed, including upper endoscopy and esophagogram. However, abdominal computed tomography suggested compression of the CT by the arcuate ligament of the diaphragm, while other results were normal. Angiotomography A and B aIn this case, Doppler ultrasound (US) detected the color aliasing in the proximal lumen of the celiac trunk during the expiratory phase with a significant increase of the peak systolic velocity (249.7 cm/s), 2 times greater than the velocity seen on inspiration (124.8 cm/s) A and B. The postoperative course was uneventful and follow-up imaging, including angiotomography and Doppler ultrasound, showed decompression of the CT , the thin, and people between 18 and 30 years.The pathophysiology of the disease consists of extrinsic CT compression by the abnormally low-implanted MAL. Compression worsens with expiration as the diaphragm moves caudally, resulting in worsening of CT stenosis. This compression results in reduced CT blood flow, visceral ischemia, and postprandial abdominal pain .Overstimulation of the celiac ganglion is also believed to result in chronic pain in these patients. Some patients may experience changes in bowel habits secondary to midgut ischemia due to the diversion of collateral blood flow from the superior mesenteric artery to the CT .Similar to the present case, asymptomatic patients with evidence of celiac trunk compression have achieved homeostasis via collateral arterial branches .We performed esophagectomy using a gastric tube reconstruction with ligation of the left gastric artery and the left gastroepiploic artery. After this procedure, the branches responsible for the vascular supply of the constructed gastric reservoir were; the right gastroepiploic artery a branch of the gastroduodenal artery, and the right gastric artery, a branch of the hepatic artery itself, which is a direct branch of CT . Thus, aWhen symptomatic, MALS usually causes postprandial epigastric pain (80%), nausea (9.7%), weight loss (48%), diarrhea 7.5%), and an epigastric murmur may be present on auscultation [%, and anOwing to nonspecific symptoms, the diagnosis usually made by excluding other conditions. The medical examination of these patients includes abdominal ultrasonography with Doppler study of mesenteric vessels and the CT, abdominal computed tomography with vascular study (angiotomography) or angiography .Patients with a history of esophagectomy with postoperative dyspepsia should initially be suspected for post-surgical complications such as gastroduodenal reflux or pyloric stenosis or delayed gastric tube emptying . The recIn the past, angiography was considered the gold standard for the diagnosis of the condition. In the sagittal section, it reveals focal stenosis of the CT associated with post-stenotic dilation and increased collateral vascularization from the superior mesenteric artery .Currently, thin-cut multiple-channel angiotomography associated with three-dimensional reconstruction has become the best method for obtaining high-resolution images of the aorta and its branches. CT angiography, especially during expiration, has a high accuracy in identifying the syndrome. Moreover, this method also allows visualization not only of the stenosed vessel but also of the underlying median arcuate ligament and adjacent tissues. Angiotomography is also important to exclude the presence of calcifications in the CT which is an important cause of arterial stenosis .Treatment options include video laparoscopy or laparotomy with sectioning of the MAL and celiac plexus fibers, as well as with the percutaneous transluminal angioplasty approach. Persistent vessel deformity or pressure gradient after the decompression procedure would be indications of approach with vascular reconstruction. The persistence of symptoms due to incomplete CT release or restenosis has been successfully treated by angioplasty with endovascular stenting within three months.4Vascular disorders should be investigated in cases of refractory abdominal pain after complex surgical procedures.The authors declare no conflict of interest.The authors received no specific funding for this work.Ethical approval exemption was given for this study.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Flavio Roberto Takeda: Conceptualization, writing, formal analysis.George Darce: writing – review and editing.Lucas Sobrado: writing – review and editing.Luisa Faria: writing – review and editing radiological images.Francisco Tustumi: writing – review and editing.Rubens Sallum: writing and Supervision.Manoel de Souza Rocha: writing – review and editing radiological images.Ulysses Ribeiro: writing and Supervision.Ivan Cecconello: Supervision.Not applicable.Flávio Roberto Takeda.Not commissioned, externally peer-reviewed.


Acute intramural hematoma in colon is a rare presentation following trauma. There are reports in literature of acute colonic hematoma following trauma, warfarin intake and in patient with coagulation disorders. In traumatic acute colonic intramural hematoma, most of the reported cases were managed surgically. Very few cases were successfully managed conservatively. We present a case of 28-year-old male who presented to the surgical emergency after two days of road traffic accident. After relevant investigations, he was found to have intramural hematoma of ascending colon, which was managed conservatively. Acute colonic intramural hematoma is a rare case and only a few cases had been reported in the past. The common causes of acute intramural hematoma are trauma, warfarin, and coagulation disorders . Blunt aA 28-year-old gentleman presented with an alleged history of a road traffic accident (RTA) two days before the presentation. He presented with injury over the right side of the abdomen and had continuous pricking type of pain in the right lower quadrant. He had vomiting and a few episodes of loose stools each day. He had no complaints of generalized abdominal distension, hematemesis, or melena. His vitals were stable. On abdominal examination, there was a tender palpable mass in the right iliac fossa. The digital rectal examination was normal. Chest X-ray erect didn’t show any free air under the diaphragm. Emergency ultrasonography (USG) showed minimal free fluid suggestive of hemoperitoneum. Contrast-enhanced computed tomography (CECT) of the abdomen was showing a small subdiaphragmatic air pocket suggestive of pneumoperitoneum Figure . Also, tAcute colonic intramural hematoma rarely occurs following trauma to the abdomen. The first case of intestinal intramural hematoma was reported in 1855 by Thompson. Hughes et al. reported about 109 cases of post-traumatic acute intramural hematoma in 1977, in which only 4% of cases were colonic intramural hematoma . Cases wThe pathophysiology for traumatic acute intramural hematoma was the shearing of the bowel wall layers due to decelerating forces or crushing forces, which causes rupture of terminal vessels entering into the bowel wall from mesentery, causing intramural hematoma . The bowIntramural hematoma can lead to acute and chronic complications. The acute complications are severe hemodynamic compromise due to blood loss by free rupture of hematoma into the peritoneum, gangrene of the involved bowel, intestinal obstruction, and intussusception . The latConservative management can be considered if the patient is hemodynamically stable with no generalized peritoneal signs ,9. But, Traumatic acute colonic intramural hematoma is a rare presentation following blunt trauma abdomen, and treatment should be individualized according to the patient. Conservative management can be considered for patients who are hemodynamically stable with no generalized peritoneal signs. Contained colonic perforation can be managed conservatively in the absence of generalized peritonitis.


Gall bladder cancer (GBC) is the fifth most common malignancy in the gastrointestinal system and the most common malignancy of the biliary system. GBC is a very aggressive malignancy having a 5 year survival rate of 19%. Giant Gall Bladder (GGB) is an uncommon condition that can result from cholelithiasis or chronic cholecystitis and rarely with malignancy.A 65 year old lady presented with vague abdominal pain for 12 years and right abdominal lump of size 20 × 8 cms was found on examination. CT scan showed a circumferentially irregularly thickened wall (2.5 cm) of gall bladder indicative of malignancy. Per-operatively a GB of size 24 × 9 cm was noted and patient underwent radical cholecystectomy. It was surprise to find such a giant malignant GB with preserved planes. Histopathology, it was well differentiated adenocarcinoma of gall bladder of Stage II (T2a N0 M0).It is known that mucocoele of GB can attain large size, however chronic cholecystitis will lead to a shrunken gall bladder rather than an enlarged one. A malignant GB of such size and resectable is rare without any lymph node involvement or liver infiltration. Few cases of giant benign gall bladder have been reported in literature, however this appears to be the largest resectable gall bladder carcinoma reported till date as per indexed literature.Giant GB is an uncommon finding. They are mostly benign, however malignant cases can occur. Radiological findings may suggest features of malignancy and define extent of disease. Prognosis depends on stage of disease and resectability, irrespective of size. Gall Bladder Cancer (GBC) is the most common biliary tree malignancy worldwide accounting for 80–95% of malignancies and the fifth most common malignancy of gastro intestinal system . The incA 65 year old lady presented with a progressive swelling in right side of abdomen for 8 months. She had vague right sided abdominal discomfort and heaviness for the past 12 years. There was no history of jaundice, vomiting or abdominal distension. Her bowel and bladder habits were unremarkable. Her past medical history, menstrual history was also unremarkable. On examination she was anicteric, vitals stable and with a Karnofsky performance scale of 80. Her BMI was 21. There was fullness of right side abdomen on inspection and further examination revealed a non-tender, firm lump of approximately 20 × 8 cms extending from right hypochondrium to right iliac fossa, whose upper and lower margins were not palpable. There was slight side to side mobility. Liver was not enlarged, but the lump was contiguous with the liver, no nodules were palpable on the liver surface and no sign of ascites was found.Ultrasonogram of abdomen revealed a massively distended gall bladder with multiple gall stones, largest of size 4 cm and circumferentially irregularly thickened wall. Computerized Tomography of the abdomen (performed 2 months before in another institution) showed a hugely distended gall bladder of size 23 × 8 cm with markedly thickened walls with multiple neovascularization along the neck of gall bladder suggestive of neoplastic etiology. Figs. , 2 ThereAs the radiological features were indicative of malignancy, patient was planned for radical cholecystectomy. At the initial diagnostic laparoscopy, no metastatic deposits seen on liver, peritoneum, omentum and no free fluid was found in the peritoneal cavity. It was followed by open radical cholecystectomy with wedge resection of liver and regional lymphadenectomy. Intra operatively a GB of size 24 × 9cm was noted Figs. , 6. It wGBC accounts for 1.2% of all global cancer diagnoses, but 1.7% of all cancer deaths . AccordiIn the US, the average survival rates for Stage II, III and IV are 49, 24 and 8 months respectively, however we do not have sufficient data in India . The cliIt is important to differentiate at an early stage which eventually is likely to have better prognosis. Ultrasound (USG), computed tomography (CT), and magnetic resonance imaging (MRI) have improved the possibility of differentiating and choosing the correct treatment. Mass occupying lesion may be present in 40–65% of patients with GBC at initial detection. GBC may present as focal or diffuse asymmetric wall thickening, which can be detected by imaging techniques like contrast-enhanced CT and MRI . The feaAlthough there are no clear-cut definitions, gallbladders of size > 14 cm and volume ≥ 1.5 L have been regarded as Giant gall bladders (GGB) –19. AmonSurgery is the mainstay of treatment of GBC which essentially is radical cholecystectomy with resection of 3 cm of liver parenchymal segments IVb and V along with regional lymphadenectomy. A minimum of 6 retrieved lymph nodes are necessary for adequate staging, indicating a thorough lymphadenectomy. Regional lymphadenectomy improves survival in T1b to T3 GBC . DiagnosGiant GB is an uncommon finding. They are mostly benign, however malignant cases can occur. Giant malignant GB of the size 24 × 9 cm, that too resectable is a rare finding. The features of malignancy and extent of disease must be identified in radiological scans. GBCs have a better prognosis if diagnosed and treated early as in Stage I or II. Radical cholecystectomy is the standard treatment for GBC. The staging may not correlate always with the size of gallbladder whereas prognosis depends on stage of disease and resectability, irrespective of size.


Pineal dysgerminomas are sporadic pediatric intracranial tumors that usually grow as midline lesions around the third ventricle, most frequently the pineal gland and the pituitary regions of the brain. The severity of symptoms is dependent on the location of the lesion and can present with increased intracranial symptoms. We report a 20-year-old man who presented with new-onset headaches over the past month that would wake him from his sleep at night. The headaches, however, resolved completely one week prior to his first neurological evaluation. A thorough neurological examination was normal. A careful review of the literature does not show a case of a pineal tumor presenting with spontaneous regression of intracranial pressure, and therefore we would like to raise awareness among clinicians about this potential course. A delay in obtaining imaging could have been life-threatening; thus, we recommend a high index of suspicion when patients present with recent symptoms suggesting increased intracranial pressure. Our patient had an excellent outcome two years after his presentation, with appropriate management including drainage of the cerebrospinal fluid, chemotherapy, and radiotherapy. A germinoma is an intracranial germ cell tumor (iGCT). The incidence of germ cell tumors (GCTs) varies significantly according to geographical location. In North America and Europe, they account for 0.4-3.4% of all pediatric central nervous system (CNS) tumors, whereas Japan and other Asian countries report that CNS GCTs account for up to 11% of all pediatric brain tumors . There iMost commonly, germinomas grow as midline lesions around the third ventricle, especially the pineal gland and the neurohypophyseal region of the brain. In females, suprasellar tumors are more common (75%), whereas in males, pineal region tumors are more prevalent (70%). Only a small percentage of patients (5%) present with dual intracranial germinoma, which is the involvement of both regions  ,4. FurthThe symptoms depend on the location of the mass, which is dependent on the mass's etiology . Pineal If the pineal lesion produces pressure on the reflex nuclei of the quadrigeminal plate, patients may develop Parinaud's syndrome, known as dorsal midbrain syndrome, which leads to difficulty in upward vertical gaze (sunsetting eyes), blepharospasm, mydriasis, and impaired ocular convergence. Other symptoms associated with pineal GCTs include ataxia, behavioral changes, and a decline in academic performance ,9. SpontWe report a case of a 20-year-old man with no significant medical history, except for a remote history of a concussion from which he recovered completely. He had a one-month history of new-onset headaches, which had resolved one week before his initial evaluation. His headaches were frequent, woke him up in the middle of the night, were more prominent in the left frontotemporal region, were non-radiating with a pressure sensation, and had a pain intensity of 10/10, especially when bending forward. The headaches were associated with photophobia, nausea, and vomiting. The headaches were not triggered by any specific food or preceded by visual aura. They did not get worse with other Valsalva maneuvers such as coughing. They improved when he lay down and worsened every time he sat up abruptly, sometimes causing dizziness, which he described as lightheadedness. He usually slept in a supine position. There was no history of recent trauma, sinus disease, fever, or toxic exposure. Acetaminophen or ibuprofen had been taken multiple times with no significant improvement. The patient had had a complete resolution of his symptoms one week prior to the time of his neurological evaluation. His family, however, wanted to make sure he did not have a major illness before he returned to college.General physical examination was unremarkable. Vital signs were normal. He appeared well-developed and well-nourished, and he was in no apparent distress. He had no dysmorphic features or neurocutaneous stigmata. His neck was supple. On neurological examination, he was alert, awake, and attentive. His naming, repetition, and immediate recall were intact. Short-term memory and delayed recall were normal. He had no problem stating the months of the year backward. His backward digit span was normal. He had no right-to-left confusion. He was able to do 100 - 7, five times without a problem. His speech was fluent. He was able to read, write, and copy a geometric figure without difficulty. His cranial nerve examination showed no deficits. His pupils were equal, round, and reactive to light. Extraocular movements were intact. There was no facial asymmetry. Fundus examination showed no papilledema. There was normal facial sensation and normal hearing to rubbing of the fingers on both ears. His palate elevated symmetrically and his tongue was in the midline without fasciculations. He had normal neck strength.On motor examination, he had normal muscle tone and muscle bulk, and his strength was full and symmetric. His deep tendon reflexes were 2+ throughout. Babinski's sign was negative. He did not present clonus. He did not have sensory deficits. He had no ataxia or dysmetria. Romberg's test was negative. He exhibited no abnormal movements. He was able to do toe-walking, heel walking, and tandem gait. He was able to run with no posturing, and his running was appropriate for age. There were no gait disturbances.Brain magnetic resonance imaging (MRI) with and without contrast was obtained, which showed a predominantly T2 hypointense mass involving the pineal region, causing a mass effect on the cerebral aqueduct and the third ventricle, resulting in obstructive hydrocephalus Figure . The difLaboratory workup included tumor markers such as HGC (human chorionic gonadotropin) and AFP , which were within the normal range. Lumbar puncture (LP) was performed and cytology was negative for malignant cells.A third ventricular fenestration was performed to decompress the hydrocephalus Figure . The bioSpace-occupying lesions involving the pineal region produce signs and symptoms related to the mass effect on the adjacent tissues  or invasTo understand this unusual presentation, we must focus on the importance of the pineal gland anatomy. The pineal gland is a small structure, variable in size of about 5 mm, located in the midline, above the tentorium, and below the splenium of the corpus callosum . It is cWe speculate that the improvement in our patient’s symptoms was due to increased pressure inside the third ventricle that was able to open the aqueduct of Sylvius; alternatively, increased compliance of the third ventricle and nearby tissues is possible. Finally, we also contemplated the possibility of a positional change as the tumor increased in size, caused by gravity, but this is not consistent with the patient's initial symptoms. We suspect his symptoms eventually would have returned, but it is not possible to predict how long that would have taken or what would have been his clinical presentation. Most concerning is the possibility of an acute hydrocephalus due to sudden obstruction of the third ventricle, culminating in significant disability or death at the time of the recurrence.Sato et al. described an 11-year-old boy who presented with polyuria and headache. Head CT and brain MRI revealed tumors in the suprasellar and pineal regions with obstructive hydrocephalus. A repeat brain MRI demonstrated shrinkage of the tumors, resection was deferred, and the patient was discharged with serial MRI for follow-up. The tumor continued to regress for three weeks; however, the patient was readmitted due to a tumor regrowth . The resAccording to Zhang et al., spontaneous regression of iGCTs is a rare phenomenon. Only 10 cases have been reported comprising one female and nine males, with a mean age of 22.1 ± 10.3 years (range:12-43 years). Among those cases, nine were diagnosed as germinoma, and four cases showed regression followed by regrowth . Zhang eThe diagnosis of iGCTs is best determined by brain MRI, which is the preferred imaging study due to its high sensitivity. It is the most accurate method to identify the tumor and delineate its relationship to adjacent structures, allowing true pineal masses to be distinguished from parapineal masses . Awa et Our patient's brain MRI clearly showed a hypointense mass involving the pineal region; although there was no thick edema, it caused a mass effect on the cerebral aqueduct and third ventricle. At the presentation in the office, even without symptoms, the clinical course prompted imaging and the findings demanded an emergent intervention. The sizeable clinical judgment and intuition prompted the imaging examination and identification of treatable pathology, which improved the clinical prognosis of our patient.To our knowledge, our patient is the first-ever reported case in the literature with complete resolution of all clinical symptoms of increased intracranial pressure before any therapies were instituted and normal neurological examination due to a pineal dysgerminoma. While we hypothesize that our patient's symptom resolution may have been due to the increased pressure in the third ventricle or increased compliance of the ventricle and adjacent structures, it is not entirely clear to us why it happened and further research on the dynamics of the CSF might be helpful in the future. Clinicians, however, must be aware of this potentially misleading course and consider appropriate imaging in the setting of new-onset headaches, even if increased intracranial pressure symptoms have resolved, as this might be a life-saving decision.


Hemorrhagic vestibular schwannoma (HVS) consisting of acute intratumoral and subarachnoid hemorrhage presents with acute nausea, vomiting, facial numbness, headache, loss of consciousness, and significant functional impairment of the facial and vestibulocochlear nerves. The current case is of a 31-year-old man who was presented with acute left lateral suboccipital headache, vomiting, ataxia, and loss of consciousness. Brain CT revealed a large iso-intense lesion with internal hematoma at the left cerebellopontine angle in association with internal acoustic canal dilation. In addition, MRI confirmed a 32 x 25 x 26 mm vestibular schwannoma (VS) with 20 x 15 x 5 mm intratumoral hematoma. The patient had undergone left lateral suboccipital craniotomy and microscopic tumor resection. Pathological findings revealed that his lesions were VS. The average incidence of HVS is around 2.15 cases per year worldwide. Therefore, HVS incidence in proportion to VS is very low and consequently rare. Vestibular schwannoma (VS), also called acoustic neuroma, is a benign tumor of the vestibulocochlear nerve that has an incidence of 11-13 cases per million every year. It arises from Schwann cells at the Obersteiner-Redlich zone [Hemorrhagic VS (HVS) is a rare phenomenon. Only limited cases are reported and there has been no definite estimation about HVS incidence until now. When significant HVS occurs, patients may experience acute neurological defects . ConsideThe treatment is based on surgery, and radiation therapy (RT) and observation are other choices of treatment. There are three standard surgical approaches: retromastoid suboccipital (retrosigmoid), translabyrinthine, and middle fossa approach (MFA). Although surgery is the treatment of choice for VS, there are many different RT techniques including single-session stereotactic radiosurgery (SRS), fractionated conventional RT, fractionated stereotactic RT (FSRT), and proton therapy -10.Herein, we report a case of a patient who was admitted with HVS. Some similar cases have also been reviewed as literature review.All procedures performed in studies involving human participants were in accordance with the ethical standards (code: IR.UMSHA.REC.1399.388) and the 1964 Helsinki Declaration and its later amendments or comparable ethical standards. Of note, informed consent was obtained from the patient in order to use his information for the case presentation. A 31-year-old shepherd man was referred to our center due to loss of consciousness. He was able to communicate and explained his experiences of a sudden severe headache in the left lateral suboccipital (retroauricular) region, as well as left hemi-facial numbness, nausea, and vomiting. After a while, he developed disequilibrium and loss of consciousness instantly after walking a few steps. Neurological examination showed he had facial paresis (House-Brackmann grade II), vertigo, and ataxia, whereas other examinations were normal.His past medical history revealed he had a history of appendectomy 30 months before current admission. He had also developed deep vein thrombosis and pulmonary emboli 18 months ago and had used aspirin after thrombolytic therapy. Moreover, he had a history of surgical removal of lung hydatid cyst 12 months ago as well as a history of headache for three years, which was intractable to any treatment. His prothrombin time (PT), partial thromboplastin time (PTT), and international normalized ratio (INR) were normal, and no abnormal findings were reported in lab results.Brain CT revealed extra-axial lesion at the left cerebellopontine angle (CPA) region with an intralesion hematoma that expanded tumor volume Figure . The lefIn the MRI, 32 x 25 x 26 mm VS at the left CPA with extension to IAC was seen. The lesion was hypo- and hyperintense on T1-weighted image (T1WI) and T2-weighted image (T2WI), respectively Figures -1D and eThe patient was operated at semi-lateral supine position with left lateral suboccipital craniotomy and microscopic retrosigmoid approach. Intratumoral hematoma was seen during tumor resection. The tumor was totally resected, and the facial nerve was preserved. The patient’s postoperation course was uneventful and without any new neurological deficit Figures , 2B.In the hematoxylin and eosin (H & E) staining, hypercellular (Antoni A) and hypocellular (Antoni B) areas are seen. Dense Antoni A areas consisting of interlacing bundles of spindle cells with oval nuclei, eosinophilic cytoplasm, and indistinct cytoplasmic borders are also seen. Hypocellular dense Antoni B areas are composed of haphazardly arranged spindle cells in loose myxoid collagen fibers. Blood vessels with hyalinized walls were visible.The VS is an uncommon intracranial tumor and frequently presents with chronic hearing loss, headache, tinnitus, disequilibrium, and facial numbness, whereas acute overt hemorrhage including subarachnoid hemorrhage (SAH) and ITH as its first presentation is quite rare .Of all intracranial hemorrhages, 1-11% belong to hemorrhagic brain tumors. SAH that arises from brain tumors accounts for 0.4% of all cases of SAH. Furthermore, 1.7- 10% of brain tumors cause intracerebral hemorrhage . ITH genAccording to Mathkour et al.’s literature review, there were only 48 cases of VS secondary to ITH, whereas in accordance with our review of the literature Table , there wBased on the reported cases, the average incidence of HVS worldwide is approximately only 2.15 cases per year; therefore, HVS is a quite rare entity among VS tumors.Some studies have reported hemorrhagic cystic VS (CVS) and therefore it can be considered as a risk factor for HVS Table . AlthougIt is assumed that anticoagulant therapy could be a potential risk factor for HVS. In accordance with other reports, our case had a history of anticoagulant (aspirin) therapy ,16.Our patient had high physical activities and he was a shepherd. The patients reported by others had a history of pregnancy, farming, heavy weight lifting, strenuous exercise, and hypertension -20. As aMicro-ITH may happen more commonly in VS, but clinically significant hemorrhage has a very rare occurrence. HVS risk factors consist of huge tumor size, cystic development, history of anticoagulant therapy, and strenuous activities.


Large intracardiac bronchogenic cysts are rare mediastinal masses. However, they must always be considered in the differential diagnosis of heart failure with abnormal chest X-ray.We present a 60-year-old female patient with de novo atrial fibrillation, heart failure and a very large intrapericardial mass. The patient underwent successful surgical resection, with pathological findings confirming a bronchogenic cyst.Large bronchogenic cysts located intrapericardially are very rare. However, they should be included in the differential diagnosis of patients presenting with atrial fibrillation and heart failure with abnormal radiologic studies. A bronchogenic cyst is a remnant of the primitive gut tube occurring as a consequence of abnormal embryonic development in the tracheobronchial tree between the 5th and 16th gestational week .2 and is classified based on location as parenchymal or mediastinal, the latter being 86% of the cases. Intrapericardial and intracardiac locations are not common in the literature [It is a rare congenital defect, with an incidence of 1/50000 people , represeA 66-year-old female patient, with no previous cardiovascular diseases, presented to the emergency room (ER) with fatigue and severe chest pain that worsened with exertion. The patient had the symptoms for a few months, but because of increased pain and dyspnea she decided to consult. Upon arrival, the electrocardiogram showed a de novo atrial fibrillation with a rapid ventricular response. The resulting chest X-ray revealed a poorly defined retrocardiac opacity suggestive of a giant mass Fig. . The traThe pericardial masses were further studied with a contrasted CT scan that showed a 10 cm compressive mass in the posteromedial mediastinum very suggestive of a pericardial cyst. A cardiac magnetic resonance image (MRI) defined a 96 × 88 × 77 mm mass in its transverse, anteroposterior and cephalo-caudal diameters, respectively. The mass was located in the superior pericardial ridge, in contact anteriorly with the aorta and pulmonary artery, laterally with the superior vena cava, posteriorly with the vertebral bodies, and inferiorly with the left atrium roof producing partial left atrial compression. The mass had a homogeneous high signal intensity in T2, without any changes in the opposite phase, and did not highlight with intravenous contrast [Fig. After considering different surgical approaches , the latter was chosen based on the CT scan and MRI results. Severe pericardial adhesions did not allow access to the mass, requiring conventional aortic and bi-caval extracorporeal circulation. After careful dissection, the 8 × 6 cm cyst was exposed. The cyst was firmly attached to the left atrial wall including the right superior and inferior pulmonary veins, superior vena cava and right pulmonary artery Fig. . SurgeryAfter a 4-day intensive care unit (ICU) stay, the patient was transferred to the step down unit. A post-operative transthoracic echocardiogram showed a LVEF of 55% and no pericardial effusions. Clinically there was no atrial fibrillation nor any signs of congestive heart failure. She was discharged 3 days later. The macroscopic pathological report described an 8x6cm mass with a 0.3 cm wall thickness of fibroelastic consistency. The histological findings suggested a benign cystic lesion made up of fibromuscular wall and mature cartilage, outlined by ciliated columnar epithelium, consistent with a bronchogenic cyst. Patient follow-up at 1, 3, and 6-months later reported a Short From 36 (SF-36) quality of life scale of 95% .On her first year out-patient follow-up she was asymptomatic on New York Heart Association class I. The check-up CT scan on this consult was completely normal.2 [Table Bronchogenic cysts are rare congenital malformations dependent on the anterior intestine . This ma2 Table .Table 1Often asymptomatic, 19% of cases are diagnosed as incidental findings on chest x-rays , 3. TypiA CT-scan is used to identify location, size, shape, and its relationship with adjacent structures, unfortunately it is effective in only 10–40% of patients . Other sThe definitive diagnosis of a bronchogenic cyst is histopathological with the surgical resection . TypicalThe best treatment is surgical resection in symptomatic patients in order to avoid late complications such as infection, rupture or malignity , 5. ProgAlthough numerous bronchogenic cyst reports have been published, this is the first one with such a large sized intrapericardial and retrocardiac location. In patients presenting with heart failure, de-novo atrial fibrillation and abnormal chest X-ray, a mediastinal mass cannot be ruled out and should be considered within the differential diagnosis. The chest CT scan and cardiac MRI are excellent complementary images; however the definitive diagnosis is a histopathological examination of the cyst’s surgical extraction.


We describe a rare case of posterior semicircular canal (PSC) fibrosis following acute labyrinthine ischemia in the territory supplied by the common cochlear artery (CCA) and review the relevant literature. A 71-year-old man with multiple vascular risk factors presented 12 days after the onset of acute vertigo and profound left-sided hearing loss. Right-beating spontaneous nystagmus with downbeat components elicited by mastoid vibrations and headshaking was detected. The video head impulse test (vHIT) revealed an isolated hypofunction of the left PSC, whereas vestibular evoked myogenic potentials (VEMPs) showed ipsilateral saccular loss. The clinical presentation and instrumental picture were consistent with acute ischemia in the territory supplied by left CCA. Compared to previous imaging, a new MRI of the brain with 3D-FIESTA sequences highlighted a filling defect in the left PSC, consistent with fibrosis. Hearing function exhibited mild improvement after steroid therapy and hyperbaric oxygen sessions, whereas vHIT abnormalities persisted over time. To the best of our knowledge, this is the only case in the literature reporting a filling defect on MRI, consistent with semicircular canal fibrosis following acute labyrinthine ischemia. Moreover, PSC fibrosis was related with poor functional outcome. We therefore suggest using balanced steady-state gradient-echo sequences a few weeks following an acute lesion of inner ear sensors to detect signal loss within membranous labyrinth consistent with post-ischemic fibrosis. Besides addressing the underlying etiology, signal loss might also offer clues on the functional behavior of the involved sensor over time. In cases of acute loss of inner ear function, a careful bedside examination supplemented by instrumental assessments, including vHIT and VEMPs, of vestibular receptors and afferents may be completed by MRI with balanced steady-state gradient-echo sequences at a later time to confirm the diagnosis and address both etiology and functional outcome. Inner ear fibrosis and ossification result from fibrous tissue deposits and new bone formation, respectively, in labyrinthine structures. These conditions represent subsequent final steps of several inner ear pathologies, including infections, genetic and autoimmune diseases, trauma, and ischemia . Animal Magnetic resonance imaging (MRI) with balanced steady-state gradient-echo sequences has demonstrated higher sensitivity in detecting signs of early fibrosis compared to high-resolution CT (HRCT) scans . In partThe development of modern tools for vestibular testing, such as video head impulse test (vHIT) and vestibular evoked myogenic potentials (VEMPs), has allowed fast measurements of the activity of each semicircular canal (SC) and otolith receptors/afferents in clinical settings , 9. ThanHere we describe the onset of posterior semicircular canal (PSC) fibrosis on MRI in a patient with a clinical presentation and instrumental findings consistent with acute stroke in the territory supplied by the common cochlear artery (CCA). We also review the relevant literature.A 71-year-old man was admitted to our institution for evaluation of sudden onset of left-sided hearing loss (HL), vertigo, and severe unsteadiness persisting for over 12 days. His clinical history was consistent with arterial hypertension, atrial fibrillation , and myelodysplastic syndrome (regular hematologic follow-ups). He reported a transient ischemic attack 4 years earlier but denied any previous inner ear symptoms. Corresponding brain MRI images were reviewed, showing signs of periventricular leukoaraiosis and normal posterior fossa structures, including normally fluid-filled SCs on FIESTA sequences .via headphones. The recording system used an EMG-based biofeedback monitoring method to minimize variations in muscle contractions and VEMPs amplitudes. Each stimulus was retested to assess reproducibility of responses, and the stimulus intensity was reduced by 10 dB until the threshold for each side was reached. Although no cervical responses could be detected on the pathologic side consistent with left-sided isolated saccular loss, symmetrical potentials were evoked for ocular testing  and oral treatment with 48 mg/day betahistine for 2 weeks, in accordance with recommendations available in the literature . SimultaDue to related cardiovascular risk factors, a new brain MRI was also scheduled in the following days. Acute brainstem infarction in diffusion-weighted and T2-weighted fluid-attenuated inversion recovery (FLAIR) images were excluded, whereas widened cerebrospinal-fluid spaces on atrophic basis and increased areas of leukoaraiosis compared with previous neuroimaging were detected . 3D-FIESAt 3-week evaluation with video Frenzel goggles, slight spontaneous right-beating nystagmus enhanced by mastoid vibrations and headshaking could still be detected. Hearing evaluation with pure-tone audiometry showed a moderate to severe down-sloping sensorineural HL on the left side, consistent with mild recovery of cochlear function . NeverthAlthough vHIT showed further reduction in left-sided PSC activity , the patWritten informed consent was obtained from the patient for the publication of this case report, including all data and images.The inner ear is supplied by the internal auditory artery, which branches from the anterior-inferior cerebellar artery (AICA) and divides into two main terminal branches: the anterior vestibular artery and CCA. Whereas, the first mostly supplies the utricle and the horizontal and superior SCs, the latter mainly serves the cochlear turns, saccule, and PSC , 12 Fig.In recent years, thanks to the introduction in clinical practice of fast modern devices such as vHIT and VEMPs, which provide a precise assessment for each SC and the otolith end organs in the high-frequency domain, instrumental testing has demonstrated its pivotal role in the topographical diagnosis of selective dysfunction of inner ear structures, including in the acute stage , 9. NeveSteady-state gradient-echo MRI such as FIESTA and CISS sequences offer a high signal-to-noise ratio that allows submillimeter imaging and high-quality multiplanar reconstructions. These images are widely used when a high-contrast evaluation of the fluid-filled spaces on the membranous labyrinth is required to check for cochlear patency and inner ear malformations in patients scheduled for cochlear implant surgery , 23. TheTo the best of our knowledge, this is the first report showing newly formed inner ear fibrosis based on MRI findings before and after a CCA ischemic lesion. Besides clinical and instrumental findings supporting this etiopathogenetic hypothesis, labyrinthine ischemia is also consistent with the high cardiovascular risk factors exhibited by the patient. Additional MRI data, including pronounced periventricular leukoaraiosis and wide cerebrospinal fluid spaces, are also in accordance with atrophic sequelae following chronic hypoxic insult to the brain tissue. The aforementioned findings strengthen the assumed mechanism underlying the presenting scenario involving ischemic mechanisms. It might be postulated that the lack of concurrent cochlear and saccular abnormalities on imaging may reflect possible variable susceptibility to steroid and oxygen therapy among different hair cells or different inter-individual patterns of venous drainage among inferior labyrinthine structures, making PSC more vulnerable to hypoxia in this case . The asyTemporal bone HRCT scans performed almost 2 months after symptom onset showed normally patent inner ear structures, excluding progression of damage toward ossification. Nevertheless, due to the short-term follow-up, we cannot exclude a possible late onset of PSC ossification in our patient.Since balanced steady-state gradient echo sequences represent a reliable tool to detect inner ear fibrosis irrespective of the patients' age , 30, it Though this report likely represents a unique case with obvious limitations related to single-case reports without histopathological support, it might be assumed that post-ischemic fibrosis of inner ear receptors could not represent an exceptional finding, particularly when we consider that steady-state gradient-echo MRI sequences are not routinely included in the imaging protocol adopted for acute HL and vertigo. Therefore, in the case of acute cochleovestibular symptoms where a careful clinical/instrumental assessment orients the diagnosis toward a peripheral lesion, along with recommending a global vestibular assessment with vHIT and VEMPs, we would also suggest postponing neuroimaging and routinely using the 3D-FIESTA MRI sequences to detect possible signs of post-ischemic fibrosis. This finding may be extremely helpful especially in those cases where underlying etiopathological mechanisms are not adequately addressed by clinical and instrumental assessments, which was not the case for our patient.The patient described in this report exhibited on MRI an unusual filling defect consistent with fibrosis in the PSC following acute labyrinthine ischemia. This finding seems also to be related to poor functional outcome for the affected end organ. Therefore, it may be reasonable to suggest using balanced steady-state gradient-echo sequences a few weeks following an acute lesion of the inner ear sensors, as they might detect signal loss within the membranous labyrinth suggesting post-ischemic fibrosis. This finding, besides addressing the diagnosis in cases where the underlying etiopathological mechanisms are unclear, might hypothetically provide clues on the functional behavior of the involved inner ear sensors over time. However, additional reports reproducing similar findings with longer follow-up are needed before any conclusion can be reached on the clinical value of filling defects within inner ear on MRI in patients with acute cochleovestibular symptoms.The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.Written informed consent was obtained from the patient for the publication of any potentially identifiable images or data included in this article.AC, EP, and AB: conceptualization, investigation, data acquisition and interpretation, and original draft preparation. AC: images and artwork. CS, MB, CB, SD, FC, MF, and FL: investigation, data collection, and original draft preparation. SM and PM: substantial role in data interpretation and manuscript revision. GB and AG: supervision and manuscript review. All authors approved the final version of the manuscript.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


Collision tumors are two independent, distinct tumors occupying the same anatomical space. This case presents a pituitary adenoma-craniopharyngioma collision tumor presenting with hemianopsia. A 60-year-old with a past history of a nonsecretory pituitary adenoma presented with progressive headaches, bitemporal hemianopsia, and nausea. Previously, in 2008, his adenoma was effectively treated with nasal septal flap and transsphenoidal pituitary resection. A magnetic resonance imaging (MRI) was ordered for concern of recurrence, given his history and neurologic complaints. The MRI revealed a suprasellar mass extending into the third ventricle with displacement of the hypothalamus and optic chiasm. Laboratory testing revealed no indicators of endocrinopathy. The neurosurgical and otolaryngologic teams were elected to perform tumor resection given the ongoing symptoms. An image-guided transsphenoidal tumor resection with abdominal fat graft harvest and septal mucosal flap CSF leak repair was performed. Histopathological examination revealed two tumor components within the resection including an adamantinomatous craniopharyngioma and recurrent pituitary adenoma. Collision tumors are defined as two independent, histologically distinct tumors that occupy the same anatomical space. Suprasellar collision tumors, specifically pituitary gland tumors within the sella turcica, display a remarkable variety of malignancy. The coexistence of a pituitary adenoma with craniopharyngioma is an exceptional clinical finding. A pituitary adenoma is the most common intracranial sellar mass, comprising 10–15% of all intracranial tumors, while craniopharyngiomas account for 1.0–4.0% of intracranial tumors , 3. ThisA 60-year-old left-handed male with a past medical history of a pituitary adenoma (2.5 cm × 2.0 cm), type-2 diabetes mellitus, hypertension, and hyperlipidemia presented to his optometrist with complaint of nonremitting visual deficit. On further examination, the optometrist diagnosed the patient with a bitemporal visual field deficit. Due to the patient's previous history, the optometrist recommended follow-up with his primary care provider (PCP) for further investigation.A week later, he presented to his PCP with the continued complaint of progressively worsening headaches and visual changes outlined above. The patient reported a four-week-history of daily, visual disturbance with associated headache, imbalance, and nonintractable nausea. Of note, his past surgical history was significant for nasal septal flap and transsphenoidal pituitary resection in 2008 for primary pituitary adenoma. He failed to receive postoperative imaging surveillance after this procedure as he was lost to follow-up.2 was 93%. Due to previous history, lack of surveillance, and progressive symptoms, brain imaging was obtained. An MRI was ordered by the primary care provider. An MRI without gadolinium revealed a heterogeneous 3 cm suprasellar mass extending into the third ventricle, with superior displacement of the hypothalamus and optic chiasm , luteinizing hormone (LH), follicle-stimulating hormone (FSH), thyroid release hormone (TRH), cortisol, and T4. Thereafter, the patient received a CT scan prior to surgery revealing the tumor invading into the third ventricle .Image-guided endonasal transsphenoidal anterior skull base tumor resection with abdominal fat graft harvest and septal mucosal flap CSF leak repair was performed. Subsequent histopathological examination of the surgical specimen revealed two tumor components within the resection: adamantinomatous craniopharyngioma and hypercellular recurrent pituitary adenoma . The pitPostoperatively, the patient developed diabetes insipidus overnight, which required two doses of oral administration (PO) of desmopressin (DDAVP). The only visual complaint was diplopia. Otherwise, there were no other abnormalities. The next day, the patient was cleared to work with physical therapy and occupational therapy. That same day, he again met criteria for DI and received a third dose of DDAVP. Due to this, the patient was started on 2 micrograms (mcg) DDAVP PO BID, and his follow-up sodium was 135 mEq/L. After this, his sodium level warranted to decrease his DDAVP dose to 1 mcg PO BID. On the patient's fourth postoperative day, his sodium levels were in the normal range. He was discharged with a repeat sodium the following week which showed to be in normal ranges. No other endocrinological abnormalities were noted postoperatively. The patient was scheduled for serial follow-ups in the next upcoming months and was informed about the possible symptoms caused by developing sodium imbalances.Collision tumors are a rare subset of tumors, especially when located in the sella turcica. To date, literature review yields only fifteen previously documented cases of collision tumors of pituitary adenoma with coexisting craniopharyngioma. Both pituitary adenomas and craniopharyngiomas are benign tumors that arise from the adenohypophysis. Pituitary adenomas are the most common causes of sellar masses, which typically arise in the third decade of life . CraniopThere are two known theories regarding the presence of craniopharyngiomas. Embryonal craniopharyngiomas are generally consistent with the adamantinomatous (AD) subtype of craniopharyngiomas, which are more commonly seen in the pediatric population. The other subtype is metaplastic papillary craniopharyngiomas, which are primarily seen in the adult population. This case differs from the current paradigm as this patient's histology revealed an adamantinomatous subtype of craniopharyngioma instead of a metaplastic papillary craniopharyngioma, which would be expected based on this patient's age .The embryonal model describes the more common subset, which is found predominantly in infants and young adolescents. This paradigm conjects that this tumor originates from defects in embryogenesis, where remnants of Rathke's pouch fail to involute, and residual tissue remains in the sella turcica, resulting in the formation of a craniopharyngioma . Given iIn contrast, the metaplastic model of craniopharyngioma origin is suggested to be a result of a metaplastic process of the anterior pituitary. Specifically, the cells of the pars tuberalis undergo a metaplastic process that allows squamous cells to proliferate and develop into a papillary craniopharyngioma . This paDiagnosis and treatment of pituitary collision tumors are frequently challenging because of the limited space for operation and biopsy. Additionally, the predilection of these tumors to adhere to surrounding structures and ability to recur make surgical intervention complicated. Given the small potential space of the sella, the tumor frequently extends into the pituitary stalk, hypothalamus, third ventricle, cerebellopontine angle, and subtemporal area . In thisSome debate whether surgical resection of a recurring pituitary tumor via the transsphenoidal approach is appropriate. Literature suggests that craniotomy is the superior approach for large complex pituitary tumors versus endoscopic transsphenoidal resection in reducing the risk of tumor recurrence . IdeologDue to the limited literature on endoscopic skull base approaches, it is also questioned whether endoscopic transsphenoidal approaches are superior to microscopic approaches. Both produce relatively similar outcomes and complications, yet the endoscopic procedure does allow better visualization of the tumor and surrounding structures . It alsoDespite better outcomes with transsphenoidal surgery, many cases of pituitary tumors have displayed recurrence of their primary tumor. This patient initially presented with a diagnosis of pituitary adenoma and progressed to have recurrence of this tumor compounded with the addition of a craniopharyngioma, leading to the diagnosis of pituitary adenoma-craniopharyngioma.Interestingly, other cases have also described tumor recurrence as a pathway to collision tumor . This brThe presence of a collision tumor in the pituitary region including a pituitary adenoma and craniopharyngioma is an exceptional clinical finding. Diagnosis is difficult, as CT scan and MRI have difficulty in distinguishing between two distinct tumors and other mimicking pathologies. Definitive diagnosis requires histopathological examination demonstrating the presence of 2 distinctly different tissues. Early identification with clinical suspicion and diagnosis can prevent invasive treatment via craniotomy and allow for more efficacious endonasal transsphenoidal approach for tumor resection.


Peritoneal recurrence of hepatocellular carcinoma (HCC) after hepatectomy occurs rarely, accounting for less than 1% of all recurrences. Reported causes of such dissemination include a history of rupture of the original HCC, needle biopsy or puncture treatment, and surgical procedures. There is no consensus on the optimal treatment strategy for peritoneal dissemination. There have been few reports on assisting resection of peritoneal dissemination by using indocyanine green (ICG) fluorescence.A 57-year-old man underwent posterior sectionectomy for HCC. Six months later, computed tomography revealed multiple nodules suspected of indicating peritoneal dissemination. Various preoperative imaging studies demonstrated only four nodules, the doubling time of the tumors being rapid at 22 days. The nodules were therefore resected. ICG (0.5 mg/kg) was injected intravenously 2 days before the procedure, enabling identification of the nodules by their brightness in the operative field under near-infrared lighting. A total of eight lesions were detected during the procedure and resected, some of which had not been identified by preoperative imaging studies. We diagnosed peritoneal dissemination of HCC based on the pathological findings and their similarity to those of the original HCC. We concluded that the recurrences were likely attributable to exposure of the tumor to the serosa at the time of the original operation.Although ICG fluorescence is useful for identifying peritoneal dissemination of HCC, attention should be paid to the difficulty in detecting deep lesions and occurrence of false positives. Although hepatocellular carcinoma (HCC) is the most common primary malignant tumor of the liver, peritoneal recurrence after hepatectomy is rare, accounting for less than 1% of all recurrences , needle A 57-year-old man underwent posterior sectionectomy for a hepatic tumor of maximum diameter 10 cm that protruded from segment 6 into the retroperitoneum Figs. . Histolo9 /L with normal differential counts and a C-reactive protein concentration of 0.5 mg/L. Concentrations of hepatobiliary enzymes were within normal limits, whereas those of alpha-fetoprotein (AFP) and protein induced by vitamin K absence or antagonist-II (PIVKA-II) were high at 1,480 ng/mL and 970 mAU/mL, respectively; these had tended to increase since the original operation. CT and magnetic resonance imaging (MRI) showed only four nodules in the abdominal cavity, the doubling times of which were rapid at 22 days. No other metastases were detected by these imaging studies.After 6 months with watchful waiting, follow-up computed tomography (CT) revealed multiple nodules suspected of indicating peritoneal dissemination Fig. . The patOn the basis of these findings, we diagnosed peritoneal dissemination of HCC after hepatectomy. Because of their rapid growth and apparent resectability, surgical resection was carried out. ICG 0.5 mg/kg) was injected intravenously 2 days before the procedure and the peritoneal nodules identified by their brightness in the operative field using an ICG near-infrared fluorescence imaging system . A total of eight lesions were thus identified and resected, including some that had not been identified by preoperative imaging studies Fig. . However mg/kg waThe postoperative course was smooth and the patient was discharged soon after surgery. Two months later, follow-up CT revealed further peritoneal dissemination that had not been detected by imaging studies performed preoperatively or intraoperative investigation. For these reasons, the patient was commenced on lenvatinib. Thereafter, the patient’s condition was stable and no new lesions were identified in the subsequent 10 months, during which there was a progressive decrease in concentrations of tumor markers.Peritoneal recurrence of HCC is rare. According to the report of the 20th nationwide follow-up survey of primary liver cancer in Japan , only 35The treatment strategy for recurrences after hepatectomy is basically the same as the initial strategy; however, no consensus on treatment of peritoneal dissemination has yet been established. Although peritoneal dissemination of other gastrointestinal cancers is generally not resectable because of its frequent association with ascites, peritoneal dissemination of HCC characteristically takes the form of isolated or localized nodular lesion. Various treatments, including intraperitoneal hyperthermia or chemotherapy , systemiReliable means of intraoperative identification of extrahepatic lesions are needed to achieve complete tumor clearance. The usefulness of ICG fluorescence in surgery has been widely discussed, including in the hepatobiliary-pancreatic fields, in which identification of tumors during laparoscopic hepatic resection –20, idenHow effectively lesions can be detected using ICG fluorescence, however, depends on their depth: fluorescence is not visible when a lesion is deeply located. Several studies have reported that tumors located more than about 5–10 mm from the organ surface are not visualized by this method , 24, 25.We have herein reported using ICG fluorescence to facilitate resection of peritoneal metastases of HCC after hepatectomy, a procedure that has been reported infrequently. Although peritoneal dissemination of HCC has a poor prognosis, selected patients may benefit from complete excision of peritoneal lesions. Assisting surgery by intraoperative ICG fluorescence may be useful in preventing residual tumors. However, it should be noted that all metastatic lesions may not be detected and false positives can occur.


We present the autopsy findings and differential diagnosis in a 42year old male who presented with fever and rapidly progressive respiratory symptoms like breathlessness, nonproductive cough and right sided chest pain. Initial imaging workup done at our hospital revealed a large unilateral tumor with tracheal shift. While being evaluated patient developed facial puffiness, tachypnea suggestive of superior vena cava obstruction. Antemortem biopsy of lung mass was attempted twice and that suggested malignant lesion. Unfortunately, the individual had a rapid downhill course following admission. Post mortem examination was conducted that on opening the thoracic cavity revealed total replacement of right lung tissue by a necrotic growth which was deeply adherent to the rib cage. The contralateral lung as well as all other visceral organs were unremarkable grossly. Histopathology confirmed primary Ewing sarcoma of the lung. We hereby, report a rare case of primary lung Ewing sarcoma diagnosed at autopsy. Ewing sarcoma/primitive neuroectodermal tumor(ES/PNET) is a rapidly-progressing tumor grouped under the category of small round blue cell sarcomas.ES/PNET is characterized by the presence of undifferentiated small cell phenotype with a wide variety of morphological differentials that includes rhabdomyosarcoma, monophasic synovial sarcoma, non-Hodgkin lymphoma, neuroblastoma. The diagnosis of the ES/PNET involving primarily the lung is challenging and requires the differential with other pulmonary malignancies sharing similar histological features. Therefore, the diagnosis requires a combination triad of morphology, supported by immunohistochemistry and cytogenetics, as these tumors are characterized by distinct genetic translocation t (11:22) forming EWSR1-ETS fusion chimeric transcription.Herein, we report the clinicopathological correlation in a 42-year-old man, nonsmoker with no known comorbidities who presented with fever, progressively increasing breathlessness, chest pain, and developed superior vena cava (SVC) syndrome to which he succumbed. On post-mortem examination, he was found to have a massive pulmonary mass, which had almost completely replaced the right lung parenchyma.A 42-year-old previously healthy male was referred to our tertiary center complaining of high-grade fever accompanied by chills over the last 20 days. He also complained of progressively increasing breathlessness, nonproductive cough, and right-sided chest pain. There was no history of weight loss or abdominal pain. He denied smoking tobacco. On arrival at our center, he was found to be febrile and dyspneic. His pulse was 120/min, respiratory rate 28/min, and SP02 was 95% at room air. The systemic examination of the chest revealed decreased breath sounds in the right lower zone and coarse crackles in the right infrascapular region. No peripheral or deep chain lymphadenopathy was detected on physical or imaging examinations. The hematological workup showed a leukocyte count of 18,000/µl  with 80% polymorphs. Liver and renal function tests were within normal limits. The serum lactate dehydrogenase (LDH) was 1650 U/L . The chest X-ray revealed a heterogeneous opacity in the right pulmonary lower lobe and pleural effusion, which showed to be hemorrhagic after diagnostic thoracocentesis A. The coA new CT-guided biopsy was attempted and showed atypical cells consistent with an underlying malignant lesion A. The thMeanwhile, the patient’s clinical condition deteriorated, he became tachypneic and developed puffiness of the face and altered sensorium and a clinical diagnosis of superior vena cava obstruction was made. He was kept on an antibiotic regimen with Piperacillin-Tazobactam, corticosteroids and controlled ventilation. Bedside 2D echocardiogram failed to evidence a pericardial tamponade, but the mass was seen compressing the right atrium and superior vena cava. On the eighth day of hospitalization, his general condition suddenly worsened, and he expired. Immediately. the autopsy was performed with the working diagnosis of a right lung mass with SVC syndrome, probably due to malignant mesothelioma or a high-grade sarcoma.External examination revealed proptosis of eyes and midline shift of trachea to the left. On the opening of the chest cavity, the right hemithorax was replaced by a massive hemorrhagic and necrotic tumor mass, which was adherent to the chest wall. In comparison, on the left side, there were 25 ml of straw-colored pleural fluid noted with pleural sheen A. The riThe liver was enlarged and weighed 1800 g (RR:1500-1800 g). The external surface was smooth and glistening. No nodularity was noted. The cut surface showed a nutmeg appearance suggestive of passive venous congestion. No mass lesion was identified.The spleen was enlarged, and weighed 230 g (RR:150-200 g). External and cut surface showed congestion. Both kidneys, pancreas, stomach, intestines, heart and brain were externally and on cut sections unremarkable.Histological examination of the lung depicted tumor cells in cohesive sheets, having a peritheliomatous arrangement amidst predominantly necrotic areas. The tumor cells were round to oval, had coarsely stippled chromatin with scant to mildly eosinophilic cytoplasm. Nucleoli were inconspicuous, and mitosis was brisk. Numerous apoptotic bodies were also noted 2C.IHC reactions showed the tumor cells to be diffusely positive for Vimentin (strong cytoplasmic positivity), CD99 (strong membranous positivity), and FLI 1(strong nuclear positivity). The tumor cells were diffusely negative for calretinin, WT1, D240, synaptophysin, CD56, TTF1, Tdt, S-100, desmin, myogenin, BCL-2, LCA, and EMA. On Periodic Schiff stain (PAS), the cytoplasm of tumor cells showed strong magenta cytoplasmic positivity AD. SectiNo tumor deposits were seen. Sections from the right and left lobe of the liver showed features of passive venous congestion. No tumor deposits were noted. Sections from the spleen showed expansion of red pulp with attenuation of the white pulp. Final diagnosis was primary Ewing sarcoma of right lung causing SVC syndrome.In 1918, Dr. Arthur Purdy Stout described an undifferentiated tumor arising from the ulnar nerve. He called it PNET because of the presence of rosettes. In 1921 James Ewing, an eminent American Pathologist, reported a tumor affecting long bones, and it was named after him as Ewing sarcoma (ES). Subsequently, these two lesions were described as different entities affecting various sites. However, by 1975, the distinction between ES and PNET began to blur, and it was realized that these two tumors represented two ends of the same histological spectrum and were grouped under the category of Ewing’s family of tumors (EFTs).EWSR1 (22q12) gene with one of the members of the ETS family of transcription factors: the FLI1 gene (11q24) in 85% of cases and the ERG gene (21q22) in 5-10% of cases. Less frequently, EWSR1 is fused with FEV (2q36), ETV1 (7p21), or ETV4 .,The EFT’s are characterized genetically by specific chromosomal translocations resulting in a fusion of the The ES/PNET arising primarily in the lungs is extremely rare, and a high index of suspicion is required to clinch the early diagnosis and initiate timely treatment. Also, the rarity of primary ES/PNET of lung necessitates a detailed clinical and radiological examination to rule out metastasis from an extrapulmonary site.ES cells are positive for vimentin, CD99/MIC 2, and FLI1, as was seen in our case. Though CD99 is highly sensitive for ES, its specificity is low, and can also be seen in rhabdomyosarcoma, synovial sarcoma, and lymphoblastic lymphoma. Antibody against FLI1 is considered to be specific for ES/PNET.Another differential to be considered, that was lower in our list was malignant melanoma, a known mimicker for various malignant entities. Hence, it is imperative to employ the full IHC panel and negate other tumors before arriving at the definitive opinion. Demonstration of t   by fluorescent in situ hybridization and/or reverse transcription-polymerase chain reaction(RT-PCR) is a definitive investigation option.ES/PNET is an aggressive neoplasm, which is potentially lethal if not intervened timely. The deceased was referred to our hospital at a very advanced stage of the disease and had a rapid downfall in our hospital, intriguingly with a concise time of symptomatic disease. The therapeutic approach requires a combination of surgical resection, followed by chemotherapy and radiotherapy to achieve disease-free survival. Presently newer modalities of targeted immunotherapy against EWS-FLI1 chimeric fusion transcript are being explored.


An invading mass in a postmenopausal female is usually suspicious of being cancerous. Even though, endometriosis should always be included in the differential since its surgical management involves significant morbidity and even mortality. An invading mass in a postmenopausal female is usually suspicious of being cancerous. Even though, endometriosis should always be included in the differential since its surgical management involves significant morbidity and even mortality. The prevalence of endometriosis is around 6%‐10%.259‐year‐old woman presented to the emergency department complaining of acute rectal bleeding while denying previous occurrences. The only significant past medical history was essential hypertension. The patient's surgical history included the following: total abdominal hysterectomy 20 years prior due to a fibroid uterus and a left oophorectomy due to a large cyst. Physical examination was insignificant and stool occult blood test returned as positive. The patient had a CT of the abdomen and pelvic with contrast demonstrating a 7 × 6 × 5 cm pelvic mass surrounding and invading the sigmoid colon. MRI was additionally ordered confirming the CT findings Figure . It coul3Deep endometriosis invading the bowel possesses a tremendous challenge and dilemma for the gynecologist. This is usually due to the high morbidity which involves surgical management. Intestinal involvement of deep invasive endometriosis is about 8%‐12% out of all endometriosis cases.4Postmenopausal invasive endometriosis is a rare finding but should be considered in the differential of suspected pelvic malignancy in this group.None declared.Dr. Ariel Polonsky (corresponding author). Dr. Polonsky was responsible for managing the case and all its postoperative complications. He participated in literature review and initial manuscript writing. Dr. Lance Bruck (co‐author). Dr. Bruck was responsible for paper editing and review in addition to image selection.Written informed consent was obtained from the patient for publication of this case report and accompanying images.


Clear cell meningioma (CCM) is one of the rarest but most aggressive forms of meningioma, with a tendency to occur at a high recurrence rate. Intraspinal CCM, especially the nondura-based type, is even rarer than the intracranial CCM.We report a case of a 45-year-old woman who presented with a 1-month history of episodic pain in the lower back and in both thighs in the front side. Femoral nerve stretch tests were positive on both sides. Magnetic resonance imaging (MRI) demonstrated an intradural tumor at the L3 level, which was isointense on T1- and T2-weighted images (WI) and homogeneously enhanced on gadolinium-contrast T1 WI.The space-occupying lesion was pathologically confirmed as CCM.During surgery, we found that the tumor adhered to a nerve root, without dural attachment. The nerve root was partially removed to achieve complete resection.The pain disappeared after the operation. The 1 year follow-up MRI revealed no evidence of tumor recurrence or metastasis.Nondura-based intraspinal CCM is easier to completely remove, and such complete removal should be achieved during the first operation. Although the recurrence rate of this particular type of meningioma appears to be lower than that of other types, close clinical and radiological follow-up is necessary. After its first description, CCM has been reported in the cerebellopontine angle, spinal/intradural  locations, and supratentorial locations, with the lumbar region and cerebellopontine angle being the most common locations.Clear cell meningioma (CCM), which was first described by Scheithauer,3 CCM differs from other varieties of meningioma in terms of its aggressive nature, high recurrence rate, and tendency to metastasize. According to the literature, the recurrence rate is 63.3% for intracranial CCM,6 and 40% for intraspinal CCM.,7,8 These clinical characteristics led the World Health Organization (WHO) to change the classification of CCM from grade I to grade II.Histologically, CCM is composed of clear, glycogen-rich, polygonal cells, and the abundant glycogen explains the name “clear cell.”Intraspinal CCM, especially the nondura-based type, is even rarer than the intracranial CCM. Herein, we report a case of intraspinal CCM without dural attachment and review the related literature. Our aim was to investigate the clinical characteristics, imaging features, treatment strategy, prognosis, and prognostic predictors of nondura-based intraspinal CCM. To the best of our knowledge, the number of patients included in our review is by far the largest in the literature.22.1A 45-year-old woman presented with a 1-month history of episodic pain in her lower back and legs. Her pain was aggravated while changing posture, and it relieved after rest. The patient was treated only with medication (Loxoprofen) and the pain was partially relieved. No significant past trauma or medical or family history was noted. On physical examination, the pain was identified to occur in the lower back and in the front side of both thighs, without numbness. Femoral nerve stretch tests were positive on both sides, and worse on the left. The straight leg raising test was negative, and no positive results were found in muscle strength, sensation, deep tendon reflexes, Babinski sign, bladder function, or anal sphincter function.2.2Lumbar spine magnetic resonance imaging (MRI) showed a well-demarcated, intradural oval lesion at the L3 level Fig. . The les2.3Under a small laminectomy and durotomy at the L3 level, we found a yellowish-pink, oval, well-encapsulated mass, which adhered to a nerve root without dural attachment Fig. . Careful2.4The pathological results showed that the tumor was composed of polygonal cells with a clear glycogen-rich cytoplasm Fig. . Immunoh2.5The pain disappeared after the operation. No neurological deficits such as numbness, weakness, or sphincter dysfunction were found. The patient was very satisfied with the results of the operation and was discharged 7 days after surgery. At follow-up 1 month after surgery, the patient had involuntary twitching of the right thigh muscle, but the frequency was not high and did not affect her quality of life. The involuntary muscle twitching disappeared almost completely 10 months after surgery. One-year follow-up MRI (whole neuraxis) revealed no evidence of tumor recurrence or metastasis Fig. .Written informed consent was obtained from the patient for the publication of the case and all accompanying images, and the study design was approved by the ethics committee of authors’ institute.3We present a case of intraspinal clear cell meningioma without dural attachment. To put the case into the context of current knowledge, and to attempt to draw some general conclusions about the prognosis and the treatment of this rare condition, we performed a review of similar cases reported in the literature. PubMed and Embase were searched with the keywords “nondura” and “intraspinal clear cell meningioma.” The references cited in the articles were also searched manually and carefully reviewed to identify potential additional studies. Detailed information about cases of nondura-based intraspinal CCM is shown in Table 3.1Most spinal meningiomas have a dural attachment site, while nondura-based intraspinal CCMs are very rare. According to our review, 23 cases of nondura-based intraspinal CCM have been reported Table . Patient This type of intraspinal CCM is more common in the lumbar segments and is closely related to the cauda equina. In some cases, as in the one we reported, nerve root attachment could be clearly seen. The image-based diagnosis was often schwannoma or meningioma. Because of the lack of specificity in the imaging features, the gold standard for the diagnosis of nondura-based intraspinal CCM remains the pathological examination.The MRI features of nondura-based intraspinal CCM are similar to those of other types of meningioma: isointense on T1-WI, isointense or hyperintense on T2-WI, and strongly enhanced on gadolinium-contrast T1-WI.Regarding recurrence, 4 studies did not report follow-up and recurrence information and 4 patients experienced recurrence at various follow-up times; hence, the recurrence rate of nondura-based intraspinal CCM was 21.1% (4/19). The mean follow-up time was 19.3 months  in 4 cases with recurrence and 26.4 months  in 15 cases without recurrence. Although the extent of recurrence might be underestimated because the follow-up time was relatively short, the 21.1% recurrence rate was much lower than that previously reported , which is an interesting observation worthy of further analysis and discussion.3.2,24,27 As for specific surgical techniques, Kawasaki et al suggested complete tumor removal and avoiding destroying the tumor capsule so as to reduce the chance of dissemination of the tumor cells during surgery. According to Nakajima et al, CCM should be removed totally together with the surrounding meninges (arachnoid and dura mater). If the tumor is tightly attached to the cauda equina (one of the most common areas of CCM), or the cauda equina is embedded within the tumor, then it is necessary to remove the pial membrane of the cauda equina, or even the cauda equina nerve root itself, to achieve complete resection.It is generally believed that the extent of resection is the most important predictive factor of CCM prognosis, and total resection should be performed whenever possible to reduce the risk of recurrence. that adjuvant radiotherapy could be beneficial in patients with a high Ki-67 index to reduce the recurrence rate. Colen et al argued that radiation therapy is recommended in CCM, even when total resection has been achieved. However, patient 8 in our review underwent a second radical resection and radiotherapy after the tumor returned but the tumor returned again 20 months later. On the other hand, considering the lack of conclusive evidence and the potential harm inflicted by radiotherapy to the spinal cord (radiation-induced myelopathy) or other organs, especially in pediatric patients, we do not recommend adjunct radiotherapy for patients with CCM with total resection.Radiotherapy can be used as an adjunct therapy in patients whose tumor is difficult to remove completely such as some specific sites of intracranial CCM. Moreover, it has been suggested found that the mean Ki-67 index of recurrent patients  was appreciably higher than that of non-recurrent patients , leading the authors to conclude that the Ki-67 index significantly differs between recurrent and non-recurrent CCM. However, cases of recurrence with a low Ki-67 index have been reported in the literature as well as cases with a high Ki-67 index without recurrence.,14,27 Although there are few studies on Ki-67, it may have a predictive effect on CCM prognosis and is thus worthy of further research.There is no consensus on whether the Ki-67 index can be used as a prognostic index for CCM. After reviewing 13 patients with CCM, Zorludemir et al,31 having, in particular, a strong predictive value for tumor recurrence (P < .0001). Regarding the relationship between PR-negativity and CCM prognosis, more data and research are necessary to reach a conclusion. Tong-tong et al suggested that CCM with anaplastic features represents an aggressive behavior that should be classified as WHO grade III. The extensive infiltrative growth pattern of CCM might hinder the achievement of complete microscopic resection. Whether there is a relationship between anaplastic features and CCM recurrence is unknown, and this still remains to be clarified because of the scarcity of relevant data.Other factors analyzed in the literature as prognostic predictors of CCM include PR-negativity and histological features. Previous analyses demonstrated that PR-negativity can be regarded as a prognostic factor in conventional meningioma,3.3The patients in our review were divided into 2 groups according to the degree of tumor resection: group A  and group B . Recurrence occurred in 1 case in group A (1/12) and 3 cases in group B (3/7). It appears that the more thoroughly the tumor is removed, the lower the recurrence rate. When considering our review of nondura-based intraspinal CCM as a whole, we found that the recurrence rate of this CCM type was 21.1% (4/19), lower than other types of CCM. A possible reason for this difference is that surgical removal of this CCM type is more complete than the surgical removal of other types of CCM. Relevant supporting arguments are as follows: the anatomy of the spinal canal is relatively simple, allowing for a clearer surgical visualization. In our review, 78% (18/23) of the nondura-based intraspinal CCMs were located in the lumbar spine, making surgery relatively easier. In this series of cases, all tumors were attached to the nerve root rather than to the dura mater. This made it easier to remove the tumor completely without destroying its capsule. In some cases of this series, surgeons had to remove the nerve root that was tightly attached to the tumor, just as in our case. In such a situation, the tumor was removed together with the pial membrane of the nerve root, and the resection was therefore more thorough. Overall, nondura-based intraspinal CCM cases are easier to completely resect. Therefore, we believe that not only the extent of tumor resection but also the surgical techniques  are the predictive factors of nondura-based intraspinal CCM prognosis.None of the patients underwent radiotherapy after the first surgical resection, a finding that is consistent with most physicians’ opinion that radiotherapy is not recommended after total resection. Few studies are available to date regarding the predictive value of Ki-67, PR-negativity, and histological features. For example, in our review, specific Ki-67 values were reported for 9 patients only and for none of the 4 patients with recurrence. Therefore, further studies are needed to analyze the role of these factors.3.4Regarding the treatment strategies for nondura-based intraspinal CCM, the case we reported and those we reviewed lead us to the following recommendations: first, thanks to the anatomical features of the spinal canal, nondura-based intraspinal CCM should be totally removed during the first surgery whenever possible, and radiotherapy is not recommended after total resection, whether in children or in adults. Second, because of the potential aggressiveness and high recurrence rate of CCMs, we recommend close postoperative clinical and neuroradiological follow-up (with neuroimaging of the whole neuraxis every 6 months). Third, radiotherapy should be performed in cases of subtotal resection or of recurrence.4Because of the rarity of the tumor, together with the short follow-up times, it is difficult to assess the recurrence rate of this tumor and to draw persuasive conclusions.Therefore, we recommend that future studies on this rare tumor contain detailed information, including relevant clinical data before, during, and after surgery as well as imaging, pathological, immunological, and even genetic data. In this way, we will be able to achieve a better understanding of CCM.5Nondura-based CCM should be considered in patients with suspected meningioma in the lumbar spine. Nondura-based intraspinal CCM is easier to remove completely, and complete removal should be achieved during the first operation, even if it requires the sacrifice of a portion of the nerve root. Although the recurrence rate of this particular type of meningioma appears to be lower than that of other types, close clinical and radiological follow-up is recommended.Conceptualization: Xiaolei Zhang, Guihuai Wang.Data curation: Xiaolei Zhang, Sheng Dong, Youtu Wu, Huifang Zhang.Formal analysis: Youtu Wu.Methodology: Sheng Dong.Supervision: James Jin Wang, Guihuai Wang.Writing – original draft: Xiaolei Zhang, Peihai Zhang.Writing – review & editing: Xiaolei Zhang, James Jin Wang, Guihuai Wang.


It shows less than 1 % of all soft tissue hemangiomas. In the head and neck area, it occurs usually in the masseter muscle followed by temporalis and sternocleidomastoid muscles.we present a case of 25-year-old male patient with a chief complaint of slowly growing facial swelling in the left zygomatic area. Clinical, imaging and histopathological evaluation lead to the diagnosis of intramuscular hemangioma in the zygomaticus major muscle.IMH in the zygomaticus muscle is very rare; hence, the clinical diagnosis of IMH is challenging. Different diagnostic procedures can be used such as CT and MRI. In addition, the ideal therapy for esthetic disfiguring IMH in the head are is the complete surgical excision of the lesion. Through the review of literature and to our knowledge this case is the first report of intramuscular hemangioma in the zygomaticus muscle.IMHs are rare in the head and neck area and must be considered in differential diagnosis of isolated muscle mass in this region. It commonly found in infants in the first year of life and then the lesion disappears with age . Hemangi2https://www.researchregistry.com/register-now#user-researchregistry/.A 25-year-old male patient referred to with no significant medical history presented with a chief complaint of slowly growing facial swelling in the left zygomatic area which had been gradually increasing in size for the last eight months. On clinical examination , a smootIn the present case, the patient was provided with information regarding the condition, possible treatment options and associated outcomes. Moreover, on consenting for surgical therapy under general anesthesia, the patient underwent complete excision of the intra-zygomaticus major muscle mass via an extended subciliary approach a. The su3Intramuscular hemangioma (IMH) was first reported in 1843 by Liston. IMH is comparatively rare benign vascular tumor of the skeletal muscles. It shows less than 1% of all soft tissue hemangiomas. IMH usually occurs in the large muscles of the trunk and extremities. In the head and neck area, it occurs usually in the masseter muscle followed by temporalis and sternocleidomastoid muscles . IntramuThe etiology of IMH is unknown, although trauma, abnormal embryonic tissue development, hormonal changes are considered predisposing factors in the growth of the IMH by stimulating more blood flow in the mass ,4. InitiTreatment of the IMH should be customised based on the patient age, lesion site, accessibility, extent, growth rate, and esthetic considerations. All conservative treatments such as systemic corticosteroids, embolization, radiation, and sclerotherapy have high recurrence rate. Due to the close approximation of the lesion to a vital surrounding structure (the eye), the surgeon was against other conservative therapy options. Moreover, sclerotherapy causes sclerosis and fibrosis of the lesion and this material may pass through the inferior orbital artery and cause eye damage. Embolization was not recommended because the lesion as shown in the MRI did not have a feeder vessel.The optimal treatment for small hemangioma in adults is the complete surgical excision of the lesion with a rim of normal surrounding muscle to minimize the risk of recurrence . Capilla4To sum up, intramuscular hemangiomas are rare in the head and neck area and should to be considered in differential diagnosis of isolated muscle mass in this region. Professional clinical examination, imaging interpretation and histopathological evaluation of the completely excised tumor gives an accurate final diagnosis of the intramuscular hemangiomas.The authors declare that they have no conflicts of interest.


Telangiectatic osteosarcoma is a rare variant of osteosarcoma and can be easily misdiagnosed as aneurysmal bone cyst. We report an atypical case of iliac telangiectatic osteosarcoma in a young healthy female, who presents with painful slow growing expansile lytic septate lesion in the left hemipelvis, which is initially treated as aneurysmal bone cyst. The diagnosis of aneurysmal bone cyst is made after histopathological examination of core needle biopsy. Her condition became unstable and massive bleeding is noted at the lesion site after sclerosant injection. She undergoes emergency hemipelvectomy and eventually the biopsy turns up to be telangiectatic osteosarcoma. Our case highlights that core needle biopsy is not useful in making diagnosis for iliac telangiectatic carcinoma. Hence, an open biopsy should be carried out in our case.This case also emphasises on careful evaluation for malignancy which is mandatory because bleeding from pelvis after an unsuitable treatment can be grave, to the extent that major amputation hemipelvectomy is an option. Even though telangiectatic osteosarcoma has the same prognosis and treatment with conventional osteosarcoma, the outcome of delayed treatment for telangiectatic osteosarcoma is not good due to the dilemma in establishing an early correct diagnosis. Delay in the definite treatment for TOS carries a poor outcome. We reported an uncommon case of iliac TOS in an otherwise healthy 18-year-old female, which was initially treated as ABC. She had to undergo an urgent lifesaving hemipelvectomy after her general condition became unstable due to massive bleeding from the sclerosant injection site of left pelvis. The diagnosis of TOS was established through histopathologic examinations (HPE) following hemipelvectomy.Telangiectatic osteosarcoma (TOS) of ilium is very rare. Pelvic TOS forms only 3.1% of all TOS1. The radiographic and histological features of Aneurysmal bone cyst (ABC) is often indistinguishable from TOS, which poses as a challenge to establish an early diagnosisA healthy 18-year-old Malay female presented with slow growing locally painful swelling at left hip for three months. The pain was aggravated by movement, worse at night, and was relieved by taking painkiller. She walked with a limping gait. The swelling gradually enlarged for three months until she was completely bed ridden before being transferred to our hospital. Her appetite was reduced where she lost 5kg of weight within the three months. She denied any recent trauma, fever, family history of cancer and other joint pain or swelling.Our examination revealed a diffuse swelling at the left iliac region, which was tender, warm, hard in consistency, not mobile and not fluctuant. Her left hip range of movement was limited due to pain. Neurovascular assessment of the left lower limb was normal. Patient was afebrile throughout her illness. All blood investigation was normal except an increase in the level of lactate dehydrogenase (733 U/L).Plain radiograph of pelvis  showed aPercutaneous sclerosing injection of the left iliac lesion  was done2. TOS commonly occurs in metaphysis of long bones. It frequently involves femur, followed by tibia and humerus2. Iliac TOS is unique because of its rarity. The incidence of telangiectatic osteosarcoma in pelvis only 3.1%1.Even though osteosarcoma is the most common primary malignant bone tumour, TOS is a very rare variant, accounting for only 2 to 12% of all osteosarcoma2. Thus, the diagnosis should be established through careful histological examination1. However, HPE is a major dilemma because core needle biopsy in our case is not helpful for us in differentiating ABC with TOS. The accuracy of core needle biopsy for TOS is very low. Yuan et al reported core needle biopsies done by surgeons showing a lower success rate in TOS (55.6%), but a higher success rate (95.4%) in conventional osteosarcoma3. The accuracy of TOS is low because some parts of TOS are resembling ABC features in HPE. In addition, obtaining tissue through core needle biopsy to diagnose TOS in a large area of pelvic lesion is just like ‘finding a needle in a haystack’. Therefore, an open biopsy should be carried out in our case.Our patient with iliac TOS posed a challenge to us in making a diagnosis due to its uncommon location and its resemblance to ABC. TOS is often indistinguishable from ABC on radiographMassive bleeding from left pelvis after sclerosant therapy is a major dilemma to us in managing this patient. Limb salvage surgery was not suitable for our patient as her unstable condition does not permit for long operative time. Furthermore, deep vein thrombosis carries high risk of embolism. Thus, we opted for emergency hemipelvectomy as a life-saving procedure4, and we were able to secure the iliac vein to prevent pulmonary embolism. However, hemipelvectomy was a very tough decision as previously she was an active young lady.5. However, the prognosis in our patient with iliac TOS was not favourable as we encountered difficulties in establishing an early correct diagnosis. Early diagnosis in TOS with prompt treatment was imperative for disease-free survival2.The prognosis of TOS was initially thought to be poor, but it is no longer true as current literature has shown that TOS and conventional osteosarcoma share the same treatment and prognosis with the survival rate of 60% at 10 yearsIn conclusion, iliac TOS is often misdiagnosed as ABC. Therefore, awareness of this uncommon condition and high index of suspicion is important to establish early correct diagnosis. Open biopsy should be performed for iliac telangiectatic osteosarcoma if core biopsy result is doubtful.