Biliary sludge is an extremely viscous sediment, consisting essentially of calcium bilirubinate granules and cholesterol crystals, which, due to its high viscosity, has poor and slow movement, leading to a mass-like configuration called tumefactive biliary sludge. Tumefactive sludge was first described with the advent of ultrasonography in the 1970s and is an uncommon intraluminal lesion of the gallbladder (GB).The differential diagnoses for an echogenic mass in the GB lumen include GB carcinoma, tumefactive sludge, and gangrenous cholecystitis. Ultrasonography is the election method for the screening of GB diseases, with diagnostic accuracy exceeding 90%. The point-of-care ultrasound (POCUS) has shown a major improvement in the evaluation of hepatobiliary diseases. POCUS allows the detection of GB wall thickness, pericholestatic fluid, sonographic Murphy's sign, and dilatation of the common bile duct. The authors present a case of abdominal pain caused by the presence of tumefactive sludge in the GB, in which POCUS helped establish the diagnosis and therapeutic guidance. Abdominal pain is one of the main causes leading people to the emergency department and has a variable clinical outcome . RecentlTumefactive sludge translates the precipitation of solid particles from bile and has a global estimated incidence of 2% with higher numbers often associated with pregnancy, patients with rapid weight loss, critically ill patients under parenteral nutrition, drugs such as Ceftriaxone and Octreotide and those with sickle disease or other causes of hemolysis . TumefacA 79-year-old female, partially dependent on daily life activities (55 points on the Barthel Index), and a personal history of heart failure with preserved ejection fraction and class II on the New York Hearth Association scale, hypertension, atrial fibrillation, diabetes, status post-ischemic stroke with secondary vascular epilepsy, chronic obstructive pulmonary disease, obesity, dyslipidemia, and chronic gastritis, presented to the emergency department with colicky acute abdominal pain in the right hypochondrium starting approximately 48h prior to admission. The pain was described as 8/10 in intensity, no aggravating factors were noted but the pain gets better with analgesics. The patient also mentioned having anorexia and asthenia, while denying fever, vomiting, or other gastrointestinal symptoms.On physical examination, the patient was hemodynamically stable, with mild signs of dehydration, afebrile, with a bulbous and distended abdomen, with positive murphy sign, and without clinical signs of peritonitis.9 /L) with neutrophilia (74.5%) and increased C-reactive protein (147.6 mg/L). There was also mild hypernatremia on the ionogram  with no other relevant changes. Liver function tests were unremarkable.Blood analyses showed leukocytosis  in the ER and revealed a moderately distended GB, with regular unthickened walls, containing a low-level echoes polypoid mass in its dependent portion (round and lobulated) without acoustic shadowing Figures , 2. GiveThe patient underwent thoraco-abdomino-pelvic computed tomography (CT scan) to better characterize the mass identified by POCUS and to exclude another potential infectious focus. The CT scan confirmed the presence of a mass‑like configuration with a pseudotumoral appearance, compatible with tumefactive biliary sludge Figure . The patGB and bile duct diseases are common in clinical practice and the most common pathology is gallstones . AnnuallTo improve decision-making capacity at the patient's bedside and optimize patient care, the use of POCUS in the emergency department has shown increasing importance, particularly in the hepatobiliary field ,8. ThrouThe biliary sludge is an extremely viscous sediment, consisting essentially of calcium bilirubinate granules and cholesterol crystals, which appear in ultrasound-like low-level echoes that lay in the dependent portion of the GB without acoustic shadowing ,3,6. DueThe clinical situations associated with a higher incidence of biliary sludge are pregnancy, rapid weight loss, hemolysis, prolonged fasting, especially in critically ill patients, and the use of total parenteral nutrition ,3,5,10. Medical therapy is limited. However, the use of drugs namely ursodeoxycholic acid, cholecystokinin, and stimulation of the GB have shown some benefits ,10. In tThe echographic image of biliary sludge can be easily mistaken by a neoplastic pathology or a large calculus of the GB, making its recognition important considering its benign evolution and potential spontaneous resolution. When an echogenic mass in the GB is identified, tumefactive sludge must be considered. When these lesions are discovered, the examination and ultrasound must be repeated out of the acute phase.The vesicular POCUS plays a key role in the evaluation of patients with right upper quadrant abdominal pain, given it can be decisive in the immediate therapeutic decision . The technical mastery of abdominal POCUS in the ER is essential to define priorities in acute situations.


We present the case of a 47-year-old man with left exophthalmus. MRI showed a left intraorbital intraconal cavernous malformation, located in the superoesternal quadrant and medially displacing the optic nerve. An endoscopic transpalpebral approach was performed and total removal was achieved after dissection of the lesion from the optic nerve and other orbital structures. Pathology confirmed the diagnosis of cavernous malformation. The patient was discharged neurologically intact on the second postoperative day free of complications. Follow-up MRI demonstrated radical resection of the cavernoma and resolution of the exophthalmus with an excellent esthetic result.https://youtu.be/o1a1tneZ6qk.The video can be found here:  This video represents the surgical management of an intraorbital intraconal cavernous angioma, located laterally to the optic nerve, in the left side.A 47-year-old man presented with a 6-month history or progressive left exophthalmus. An MRI was performed, showing a spotting enhancing lesion mainly located in the superoexternal quadrant of the orbit, medially displacing the optic nerve, compatible for a cavernous angioma.Many surgical approaches are possible for orbital lesions, mainly depending on their location, each one with proper advantages and limits .For lesions located anteriorly to the bulb, inferior or superior transconjunctival approaches may be adopted . However, for deeper lesions other approaches are preferred, as the transcranial ones, which allow to enter into the orbit after its unroofing, such as for omolateral frontotemporal or frontotemporozygomatic or bifrontal .The main limits of these routes are the brain retraction, required to expose the orbit roof, which could lead to possible postoperative complications, such as cerebral edema, stroke, seizures, and brain injury . Moreover, for tumors located in the inferior quadrants these approaches may require also a long corridor inside the orbit, with possible manipulation of the optic nerve or of its vascular supply .The endoscopic endonasal approach may combine the advantages of not needing any brain retraction with a direct minimally invasive extracranial exposure of the lamina papiracea . However, this approach is limited laterally by the optic nerve, which cannot be crossed to avoid permanent injury .Therefore, for tumors located lateral to this structure the alternatives to transcranial approach are represented by lateral orbitomy or other transfacial approaches, which are limited by the risk of not esthetic scars and which could require a careful reconstruction of the lateral wall of the orbit to avoid a poor cosmetic result for the patients . A promising option for such tumors, which has been recently proposed, is represented by the transpalpebral approach. It gives a direct and straightforward access to lateral quadrants of the orbit through a centimetric skin incision in the eyelid, which has not esthetic consequences . Moreover, it allows to resect the lesion with the standard bimanual microsurgical technique with minimal manipulation of the orbital structures .Our surgery was performed under general anesthesia with orotracheal intubation. Patient was positioned in simple supine position with head in a median position. A 2-cm linear incision was performed following a palpebral crease, starting from lateral canthus. The orbicularis oculi muscle was identified and dissected to start to identify the superoexternal orbital rim.Periobita was dissected and retracted from this bone plane, which partially drilled out to increase the working space. The roof of the orbit was identified to orientate the surgeon.Afterwards, periorbita was dissected and cut through a round scalpel and opened with scissors. With a hook this incision was enlarged and the orbital fat starts to come out.Through a blunt dissection the intraorbital structures were progressively identified, mainly the lateral rectus muscles. During this phase it is important to avoid any manipulation of the ventral side of the muscles to avoid damages to neural motor fibers.A large is mass was progressively identified medially to the lateral rectus muscle. The lesion was dissected from surrounding fat and other orbital tissue and isolated putting cottonoids at its sides. More tenacious fibers covering the lesion may be cut, after recheck of the position of the other anatomical structures.During these dissection maneuvers, the dark-colored mass appeared in the surgical field. Its gross morphology was compatible with the preoperative suspect of cavernous angioma. Continuing with the blunt dissection of the lesions with cottonoids at its sides to avoid dangerous blind maneuvers and adopting sharp instruments only under the visual control of the surgeon on the front of the lesion, its anterior surface was now exposed and partially coagulated to shrink the mass.With the same technique, it is possible to complete the dissection of the lesion, starting from its lateral side and then moving on the medial one, avoiding any direct traction or hazardous maneuver on the optic nerve, which was covered by cottonoids and not exposed.Finally, the posterior portion of the cavernoma was resected with no direct manipulation of the lateral rectus muscle, which was anatomically and functionally spared, adopting the usual technique of dissection. Then, the tumor was delivered out of the orbit through the skin incision en bloc. Hemostasis was performed also adopting Floasel as direct hemostatic agent. A little piece of abdominal fat is placed to compensate the minimal lateral orbit wall drilling to avoid the development of enophthalmus.Gross appearance of the mass was of a brownish lesion of about 2.5 cm of length. At histological examination, large, irregularly shaped and sized vascular channels surrounded by thick walls were observed without evidence of atypia. Endothelial cells showed positivity for antibody anti-CD 31, and they were negative for antibody anti-D2-40, confirming the diagnosis of cavernous angioma.Patient was discharged intact at second postoperative day and MRI showed the complete resection of the tumor with resolution of exophthalmus. Cosmetic result was satisfactory with no evident scar on the superior eyelid.


The synchronicity of situs inversus totalis (SIT) and infantile hypertrophic pyloric stenosis (IHPS) is rare. We have reported a case of this concurrency in a 3‐day‐old newborn with classic symptoms. Even non‐bilious vomiting can signify a severe underlying condition in newborns and demands careful evaluation.  The vomiting was progressive, usually followed breastfeeding, and was not accompanied by abdominal distention or defecation problems. Findings on ultrasonography revealed that the pyloric canal is 22 mm in length and 4 mm in width. No other abdominal anomalies were reported. An incidental finding was the mirror displacement of the abdominal viscera. In clinical examination, the “olive sign” was absent. Preoperative echocardiography and upper gastrointestinal (UGI) barium contrast series were requested for preoperative preparation, demonstrating dextrocardia Figure . Given tSince IHPS does not usually present this early in life, an upper gastrointestinal series was also performed to rule out other possible causes. No evidence of malrotation or midgut volvulus was seen. Barium enema did not reveal evidence of intestinal atresia.Fluid resuscitation was continued for 24 hours to normalize acid–base disturbances, electrolytes, and bicarbonate levels. We underwent abdominal exploration through a transverse left‐leaning supraumbilical incision after stabilization. No anomaly except SIT and a thickened pylorus on the left side were detected. The thickness zone was visible in the pyloric region, compatible with the ultrasonography report Figure . After c3Situs inversus viscerum occurs when intra‐abdominal organs are mirrored inside the abdomen. This rare phenomenon has a prevalence rate of 0.1—0.6 per 10,000 live births.Pyloric muscle progressively gets hypertrophied and obstructs gastric emptying. Since sometimes the emesis is infrequent, it may be misdiagnosed as gastroesophageal reflux disease (GERD). If left untreated, there is a risk of death due to dehydration, malnutrition, and hypochloraemic, hypokalaemic metabolic alkalosis. The typical examination finding is palpation of the “olive” in the epigastrium, the thickened pylorus. However, it is not uniformly present, and its recognition is hampered by the examiner's experience, presence of gastric distention, and an unsettled infant. For these reasons, ultrasonography is the investigation of choice with a sensitivity and specificity of 100% in experienced hands.4Vomiting in the newborn is severe, even if it is non‐bilious and needs careful evaluation. Although IHPS is a disorder of infants generally aged three weeks to three months, it may rarely present earlier. Very rarely, as in our patient, SIT can be associated with gastrointestinal disorders such as IHPS.Khashayar Atqiaee and Ali Tabatabaey involved in planning. Khashayar Atqiaee and Ali Samady Khanghah involved in conduct. Khashayar Atqiaee involved in reporting. Ali Samady Khanghah involved in data acquisition. Ali Samady Khanghah involved in data interpretation. Ali Tabatabaey involved in manuscript preparation, editing, and review.The authors received no funds to conduct the study. It was self‐funded.The authors declare no competing interests relating to this original work.We confirm that all named authors have read and approved the manuscript. The protection of intellectual property associated with this manuscript has been our consideration.The authors confirmed that they had gotten all proper patient written consent formats. The parents have given their consent for their images and other clinical information to be reported in the form. We informed The parents about the confidentiality of the names and initials, and efforts would be made to hide their identity.


Mesiodens are the most common supernumerary teeth and are detected incidentally during routine radiographic examination, so late diagnosis complications are very common. The dentist must make a timely diagnosis and thus avoid clinical complications. Despite advances in knowledge of dental morphogenesis and differentiation, the etiology of mesiodens remains unclear. Therefore, several theories have been postulated to explain how and why they develop. It was described in the literature that heredity could play an important role in the appearance of supernumerary teeth, with a higher rate of appearance in relatives of those affected. This article reports three cases, a mother and two children, who present mesiodens, which shows that supernumerary teeth may involve a genetic factor. In addition, a literature review was carried out to assess the importance of the genetic factor as a possible cause of mesiodens. The relevance and implications of timely diagnosis in clinical practice to avoid manifestations of clinical complications are discussed. Therefore, the identification of the genetic risk factors responsible for the formation of supernumerary teeth is essential for developing a screening tool to determine an individual’s genetic risk. Supernumerary teeth are a developmental anomaly that is defined as “any tooth or odontogenic structure that forms from tooth buds more than the usual number for any given region of the dental arch” ,2. They In general, an asymptomatic, impacted mesiodens is detected incidentally during routine radiographic examination, because only 25% of maxillary anterior supernumerary teeth erupt ,10. TherPanoramic, maxillary occlusal, and periapical radiographs are indicated to aid in the diagnosis of mesiodens ,8. A panTooth development is an intricate mechanism involving multiple factors, both genetic and environmental, and to fully understand abnormalities of tooth development we must understand the mechanisms and explanations that have been given for these processes over time. There are several theories about the etiology of supernumerary teeth; however, their origin is unknown to date ,10,11,12Many publications focus on the prevalence or treatment of mesiodens supernumerary teeth; however, the etiology of the condition is still unknown. Therefore, the objective of this article is to document three cases of mesiodens, a mother and her children, showing that supernumerary teeth may involve a genetic factor. In addition, a literature review is carried out to assess the importance of the genetic factor as a possible cause of mesiodens. The relevance and implications of timely diagnosis in clinical practice are discussed.The three cases below differ in the diagnosis of mesiodens according to the time of dental consultation. Authorization to publish the cases was obtained through informed consent and ethical authorization from the Scientific Ethics Committee of the Universidad de La Frontera.In 2017, he attended dental care at the Teaching Dental Clinic of the Faculty of Dentistry of the Universidad de La Frontera, Temuco, Chile, as a 9-year-old male patient. The reason for the consultation was dental caries. He did not present any syndrome, systemic disease, or medication; he was a shy but cooperative patient who responded favorably to behavioral management techniques.In the intraoral clinical examination, the patient presented mixed dentition with multiple cavitated and non-cavitated, active carious lesions. In soft tissue, there was an increase in volume in the hard palate between the upper central incisors (1.1 and 2.1), of hard consistency, painless, with years of evolution according to the mother’s report.Cone-beam computed tomography was requested for the maxilla . The radIn September 2021, the mother of the patient reported in case 1 attended for dental care; she was a 49-year-old female patient with no underlying pathology. Intraoral examination showed partial edentulism, multiple active, cavitated caries, root remains, and discoloration in tooth 2.1, which responded negatively to thermal and percussion tests, with active, cavitated caries on the mesial–palatal–buccal surface. Periapical radiography was requested to plan endodontic treatment in tooth 2.1  and panoIn October 2021, a 22-year-old woman presented for dental care. Patient had no underlying pathology. There were no signs of any syndrome. On intraoral examination, the patient presented normal, complete permanent dentition, with carious lesions on teeth 1.2 and 1.4. The intraoral soft tissues were healthy, and she practiced good oral hygiene. In the anamnesis, the patient reported a clinical history of the extraction of mesiodens diagnosed by routine radiographic examination 11 years ago. In addition, she reported a history of mesiodens in her mother (case report 2) and her brother (case report 1) Despite advances in knowledge and differentiation of dental morphogenesis, the etiology of mesiodens remains unclear ,10,11,12The databases searched in this literature review were PubMed/MEDLINE, Web of Science, Science Direct, EMBASE, and SCOPUS, using keywords Mesiodens AND Familial Occurrence AND Impacted Teeth AND Etiology, and included primary studies that reported the presence of non-syndromic maxillary mesiodens in subjects without relevant medical history from the same family. The data were extracted from reports of selected cases, and information considered relevant for the analysis are shown in Twelve studies supported the presence of non-syndromic maxillary mesiodens in subjects without relevant medical history from the same family ,22,23,24Kawashima et al. (2006) reported that if either parent exhibited a supernumerary tooth, then the child had a 5989-fold increased risk of developing a supernumerary tooth . Two repThe literature is abundant on the occurrence of mesiodens in siblings of different ages. The literature search revealed four reports of mesiodens in two siblings ,18,19,21It was reported in the literature that mesiodens are about twice as common in men as in women ,7. This Supernumerary teeth are detected incidentally during radiographic examination since mesiodens rarely erupt within the permanent dentition about 25%), so late diagnosis complications are very common . It is a%, so latThe timing of removal of any unerupted mesiodens is controversial in the literature . ImmediaThis report of three family cases with the presence of mesiodens indicates and corroborates that heredity is involved in the etiology of supernumerary teeth. In general, the detection of mesiodens after the presence of complications can be pretty obvious and late, either due to the patient’s complaint or in the clinical and radiographic examination. Therefore, early and timely detection is a tool that will allow complications to be avoided. This study encourages dentists to identify the presence of mesiodens early through the clinical history of family history of mesiodens, in routine radiographic examination, and during clinical examination. This will allow us to carry out an early approach in the case of patients who do not report signs or clinical symptoms. In addition, it will avoid complications and allow treatment to be planned according to the location and associated anatomical structures of each case.


Acute proptosis is a very rare condition presenting to the emergency department. As there are very few case reports of patients with acute onset proptosis, it is important to report each new case. This case report is of a 38-year-old lady who presented to our emergency department with a headache for three days, altered sensorium for eight hours, and acute proptosis of the left eye for 40 minutes. She was diagnosed to have a venous hemorrhagic infarct in the left parietal-occipital-temporal region with thrombosis of the left transverse and sigmoid sinuses. To the best of our knowledge, there is no documented case report or study which featured acute proptosis as a clinical sign in a patient with venous hemorrhagic infarct or where acute proptosis was associated with thrombosis of a cerebral venous sinus other than cavernous sinus. This study shows that acute proptosis can be a presenting sign even in venous hemorrhagic infarct and acute proptosis can be associated with cerebral sinus venous thrombosis even without the involvement of cavernous sinus. So although rare, venous hemorrhagic infarct and cerebral venous sinus thrombosis irrespective of the venous sinus involved should be considered in any patient presenting to the emergency department with acute onset proptosis. Proptosis, also known as exophthalmos, is the protrusion of the eyeball from the orbit . Acute pCase reports of acute proptosis are very few. Thus each new case is essential. This case report is of a 38-year-old lady who presented to our emergency department with a headache for three days, altered sensorium for eight hours, and acute proptosis for 40 minutes. She was diagnosed with a venous hemorrhagic infarct in the left parietal-temporal-occipital region with thrombosis of left transverse and sigmoid sinuses, mass effect in the form of midline shift to the right side, an intraventricular extension of hemorrhage onto the left lateral ventricle.To the best of our knowledge, no documented case report or study featured acute proptosis as a clinical sign in a patient with venous hemorrhagic infarct or where acute proptosis was associated with thrombosis of a cerebral venous sinus other than cavernous sinus. This case report shows that acute proptosis can be a presenting sign even in venous hemorrhagic infarct, and acute proptosis can be associated with cerebral sinus venous thrombosis even without the involvement of the cavernous sinus.A 38-year-old lady presented to the emergency department with a headache for three days, altered sensorium for eight hours, and proptosis of the left eye for 40 minutes. The headache started around three days back. It was gradual in onset, persistent with medium severity. The patient was taken to a local clinic, and medicines were prescribed, but the headache persisted. Around eight hours back, at around noon, she developed altered sensorium and multiple episodes of vomiting. She was taken to a local hospital where a computerized tomography (CT) scan of the brain was taken which was normal. Then she was referred to our hospital, which is a tertiary care center. According to the relatives, around 40 minutes back, the patient developed sudden proptosis of the left eye, which is progressing.On physical examination, vitals were as follows: pulse rate: 82 per minute, blood pressure: 140/80 mmHg, respiratory rate: 20 per minute, temperature: 97.5°F, and oxygen saturation: 99% on room air. On neurological examination, her Glasgow coma scale (GCS) score was 11. She opened her eyes to voice (eye response score - three); she was speaking inappropriate discernible words to pain stimulus ; and she was localizing to pain with her left hand (motor response score - five). Her right pupil was 3 mm reactive to light, and her left pupil was 5 mm nonreactive to light. She was moving all her limbs, but there was right-sided weakness compared to the left. Exact power could not be assessed due to altered sensorium. Babinski’s sign was positive on the right side. On auscultation, her lungs were clear and had equal air entry bilaterally. Her heart rate was regular with normal S1 and S2, and there were no murmurs, rubs, or gallops. The abdomen was soft, non-tender, and non-distended, with normal bowel sounds.A CT brain scan with contrast study was taken, which reported venous hemorrhagic infarct in the left parietal-temporal-occipital region with thrombosis of left transverse and sigmoid sinuses, mass effect in the form of midline shift to the right side, an intraventricular extension of hemorrhage onto the left lateral ventricle shown in Figures The patient was treated in the emergency department with prophylactic antiepileptics, antacids, low-molecular-weight heparin, mannitol, dexamethasone, and intravenous (IV) fluid normal saline. The patient was advised to get admitted to the neurosurgery department. However, due to financial reasons, they requested a referral to a government hospital and were referred, and the patient was lost to follow-up.Past history: no history of trauma, surgery, oral or IV drug use, recreational drug usage, immunosuppression, hypertension, coronary artery disease, cerebrovascular accident, cancer, coagulopathy, or use of anticoagulants.There was no family history of cerebrovascular accident, hypertension, diabetes mellitus, or coronary artery disease. The patient was a non-alcoholic, non-smoker.Acute proptosis is a rare condition presenting to the emergency department. It is mainly seen in trauma patients with retrobulbar hemorrhage. There are very few case reports of patients with acute proptosis associated with cortico-cavernous fistula, trauma patients with extradural hemorrhage, retrobulbar hemorrhage, subperiosteal hemorrhage, isolated rectus muscle hematoma, intracerebral hemorrhage following thrombolysis, fever with orbital cellulitis and subperiosteal hemorrhage that have been reported -9. Thus This case report is of a 38-year-old lady who presented to our emergency department with a headache for three days, altered sensorium for eight hours, and acute proptosis for 40 minutes. She was diagnosed with a venous hemorrhagic infarct in the left parieto-occipitotemporal region with thrombosis of the left transverse and sigmoid sinuses. Following an extensive search, we could not find a documented case report or study which featured acute proptosis as a clinical sign in a patient with venous hemorrhagic infarct or where acute proptosis was associated with thrombosis of a cerebral venous sinus other than cavernous sinus. This study shows that acute proptosis can be a presenting sign even in venous hemorrhagic infarct, and acute proptosis can be associated with cerebral sinus venous thrombosis without cavernous sinus involvement.Studies and case reports of acute proptosis are very few. Most case reports are of patients with retrobulbar hemorrhage presenting to the emergency department following trauma and those with carotico-cavernous fistula. According to some case reports, acute proptosis may also occur in patients with cavernous sinus thrombosis and trauma patients with extradural hemorrhage, retrobulbar hemorrhage, subperiosteal hemorrhage, and isolated rectus muscle hematoma. There are also isolated case reports of patients with acute proptosis with retrobulbar hemorrhage and intracerebral hemorrhage following thrombolysis and fever with orbital cellulitis and subperiosteal hemorrhage -9. AccorCerebral venous infarction is a rare type of stroke. It is most commonly caused by the thrombosis of the cerebral venous sinuses or the cerebral veins. When the infarct occurs in an uncommon area, cerebral venous infarction should be considered among the differential diagnoses. Cerebral venous infarction is also commonly associated with venous hemorrhage in about half of the patients with cerebral venous infarction, and this association has a poorer prognosis . The incThe risk factors of thrombosis are hypercoagulability, injury to the vessel wall, and alterations in blood flow, which are together described as Virchow’s triad. Deficiency of protein C, protein S or antithrombin, pregnancy, puerperium, and use of estrogen-containing contraceptive pills are conditions leading to hypercoagulability. Chronic inflammatory diseases like inflammatory bowel diseases, lupus, Behcet’s disease, etc., are conditions where the vessel wall is affected. These conditions are associated with cortical venous sinus thrombosis. Hematological disorders like polycythemia vera, paroxysmal nocturnal hemoglobinuria, infections and medical procedures involving ear, nose, throat, paranasal sinuses, sickle cell anemias, homocysteinemia, dehydration, COVID-19, iron deficiency anemia in children, malignancies are other conditions associated with cortical venous sinus thrombosis .The most common symptom associated with cortical venous sinus thrombosis is a headache which can vary in severity from mild to severe. Other commonly seen symptoms are motor weakness due to infarction, ocular signs and symptoms, seizures, and altered sensorium. Concerning ocular findings in cortical venous sinus thrombosis, papilledema is the most common. Sixth nerve palsy, diplopia, vision loss, and proptosis are other ocular signs and symptoms associated with cortical venous sinus thrombosis. As cortical venous sinus thrombosis is commonly associated with infarction and hemorrhage and has a poor prognosis, it has to be rapidly diagnosed, and treatment should be started rapidly. A CT scan or a magnetic resonance imaging (MRI) scan of the brain can diagnose cortical venous sinus thrombosis. Anticoagulation should be started as soon as cortical venous sinus thrombosis is diagnosed. It is important to note that venous hemorrhage associated with cortical venous sinus thrombosis is not a contraindication for anticoagulation therapy ,14.Acute proptosis is usually seen in trauma with retrobulbar hemorrhage and cortico-cavernous fistula, but in the case of our patient who presented to the emergency department, it was a venous hemorrhagic infarct. This was a dilemma of diagnosis. So clinically along with other differential diagnoses, venous hemorrhagic infarct should also be ruled out. Also, it is uncommon for proptosis to be associated with cortical venous sinuses other than cavernous sinuses according to studies. Our patient had thrombosis of the left transverse and sigmoid sinuses, which shows that proptosis can occur acutely even without the cavernous sinus being involved. Though the case report has no treatment details or follow-up, this case adds to the limited medical literature on acute onset proptosis.


Cloacal malformation (CM) is a severe, complex, and extremely rare category of anorectal and urogenital tract malformations. Prenatal diagnosis is illusory and vague; therefore, magnetic resonance imaging (MRI) is the most effective test point toward an accurate diagnosis. Thus, careful investigation and evaluation are mandatory since they could be associated with syndromes and other anomalies, including urogenital tract, vertebral, and cord abnormalities.Despite the severity and complexity of the deformity, CM cases are curable, not desperate, and can have an excellent prognosis with great surgical correction. However, managing persistent cloaca necessitates a careful assessment because corrective surgeries require inclusive surgical planning, multidisciplinary, expert, and highly specialized medical center. In surgically repaired malformations, fecal and urinary incontinence has been a major issue, which was resolved when Dr. Pena Alberto suggested safer dissection and less harmful techniques for neurovascular structures and great functional corrected anomaly to ensure fertility and less incontinence. For improved results and prognosis on quality of life, patients should be scheduled for extended bowel training along with the clinical evaluation follow-up. In this article, we present a case successfully treated with the posterior sagittal approach, Pena operation, and anorecto-vagino-urethroplasty with feminizing clitoroplasty and highlight the value and impact of prenatal evaluation, diagnosis, and management. The rarity of the case and excellent results, including fair to normal bowel and urinary control, prompted us to report it and assert the significance of assessment, surgical management and technique, challenges, postoperative bowel training, and clinical investigation and examination. Cloacal malformation (CM) is an extremely rare disease with an incidence of ~0.002% -3. It isCloaca is found transiently during embryological evolution between the fourth and seventh weeks of intrauterine life when the urogenital sinus structures and gastrointestinal tracts emerge from the same origin, the endoderm. All genitourinary organs originate from the mesoderm, except for urogenital sinus structures, genital swellings, and adrenal medulla, which emerge from the endoderm.The common chamber of the urogenital and anorectal tracts separates into hindgut and urogenital sinus. Tourneux fold and Rathke’s plicae fuse to form the urorectal septum, which divides the common chamber into the urogenital sinus and anorectal canal. Impairment and unsuccessful separation and subdivision will result in persistent cloaca malformation.If not promptly and properly treated, the malformations are formidable, severe, and fatal. CMs are considered the most immense technical challenge to repair anatomical defects in anorectal and urogenital congenital malformations.Surgical management is a challenging, advanced, and distinct mode of surgery, with the ultimate goal of ensuring well reconstructed functional and controlled fecal, urine, sexual organs, and future fertility. Since the posterior sagittal approach was proposed in 1980, surgical outcomes have significantly improved, and it is currently preferred over the abdominal approach. The initial treatment and definitive reconstruction of CM are the most technically challenging clinical treatment options .Dr. Alberto Pena proposed the posterior sagittal technique in 1980, and in 1853, Amussat is credited with being the first individual to sew the rectum with the perineum. Hence, we present a case managed by one session posterior sagittal anorectoplasty, vagino-urethroplasty, and feminizing clitoroplasty, with fair to normal bowel and urinary control and excellent anatomical repair shape.A one-year-old girl with a known case of persistent cloaca was referred to our department of urology for definitive reconstructive posterior sagittal anorecto-vagino-urethroplasty (PSARVUP). Physical examination revealed an enlarged clitoris with a single perineal opening, while preoperative abdomino-pelvis ultrasound revealed urinary tract anomalies. The patient came with a colostomy  done on the second day of life.The procedure was done at the age of one year old, with an endoscopic evaluation cystoscopy , genitography , and the insertion of a urinary catheter, followed by vaginoscopy and the insertion of a vaginal catheter Figure .After putting the patient in a prone position, the posterior sagittal incision was made. The anal sphincter was identified by nerve stimulation, posterior sagittal anorectoplasty with total urinary mobilization was performed, and the Pena operation, wherein the urethra and vagina were separated as one structure (both the urethra and the vagina were mobilized together as a single unit), was conducted Figure . The anoAfter shifting the patient to a supine position, vaginal and urethral walls were dissected, and vaginoplasty with urethroplasty was performed. Feminizing clitoroplasty was also performed Figure .The operative time was 160 min. Postoperative and recovery were uneventful and the patient was doing well and discharged after one week. She was scheduled for a long-term follow-up to assess her bowel and urinary control. On the second week, postoperative visit and examination revealed patent vaginal, anal, and urethral openings.The post-operative outcomes were examined based on the following objective measures after six months and during the follow-up appointment: rectal and urinary continence, ability to evacuate the rectum and bladder spontaneously, preservation of renal integrity, and adequacy of vagina and sexual function. Our patient had no problems with his capacity to urinate or evacuate her rectum, no constipation, just infrequent episodes of diarrhea, and normal kidney function tests. However, we will need to examine our patient again at the age of four years to adequately re-evaluate and assess her urinary and rectal continence.Persistent cloaca malformation is assessed as the extreme type of anorectal malformations (ARMs). “Persistent cloaca” is a severe malformation affecting females, wherein the urinary, genital, and alimentary tracts share a single conduit .Cloaca malformation is characterized by the passage of meconium, stool, and urine through a single perineal opening. Prenatal diagnosis is intractable and tricky, and the ultrasound findings are vague and nonspecific; therefore, the MRI is preferred and highly recommended for diagnosis since it is more informative and accurate.Bischoff et al. reported the frequency of abnormal prenatal ultrasound findings in patients with persistent cloaca as follows: abdominal cystic mass (41.1%), hydronephrosis (37.9%), oligohydramnios (24.2%), distended bowel (20%), ascites (15.8%), two-vessel cord (14.7%), dilated bladder (14.7%), dilated ureter (14.7%), polyhydramnios (10.5%), echogenic bowel (8.4%), multicystic kidneys (8.4%), hydrops fetalis (7.4%), hydrocolpos (4.2%), absent kidney (3.2%), abnormal spine (3.2%), and anorectal atresia (3.2%) .When cloaca malformation is not treated properly and promptly, it can be lethal and dismal. It could be isolated or may be associated with other systems’ malformations and syndromes. Because the complications are severe and lethal, immediate and careful management is essential and lifesaving.Because a fetus can die in the uterus from high-grade urinary obstruction , and a nSurgical correction of anatomical defects is always challenging as it is highly technical to reattain excellent functional anatomical repair, with the main target of ensuring fair bowel and urine control, fair sexual function, and fertility and renal function.Persistent cloaca is a potentially devastating disease, necessitating multiple rounds of corrective surgery. Even with the most advanced treatments, there can be significant urological and gynecological sequelae, including incontinence and infertility . AdvanceTo avoid severe and life-threatening complications of obstruction, colostomy should be considered first; however, the reconstructive repair surgery is usually postponed until the child is between the ages of six and 18 months.Following the recommendation of the posterior sagittal approach, which promotes a clear vision of the anatomical defect, safe dissection, mobilization, and thus less muscle complex injury than in the abdominal approach, and less fecal, urine incontinence, which are the major complications encountered and been for a long time, a dilemma of repaired congenital malformations. The prevalence of fecal and urinary incontinence following posterior sagittal anorectoplasty for anorectal malformation has been widely observed and assessed, with many variations; according to a study by Ghorbanpoor et al., good fecal continence was observed in 91.3% of patients with low type anorectal malformation compared to 72.8% of patients with high type anorectal malformation; however, the difference was not significant . FurtherReconstructive surgery for the cloaca and anorectal malformations was significantly changed by PSARVUP or TUM approach, which is preferable, better, and safer than the abdominal approach for the cloaca and anorectal malformations. As the prognosis on bowl, urinary control, sexual function is better with the Pena technique. This approach facilitates a clear vision and is more obvious in the dissection, resulting in reduced neurovascular injury, the main issue of anorectal malformations. Fecal and urinary incontinence significantly decreased and guaranteed normal renal function and fertility. An experienced and skilled multidisciplinary team is essential; collaboration with specialists and mental health support is also highly recommended.


Esophageal leiomyoma is the most common benign submucosal mesenchymal tumor. Esophageal intraepithelial neoplasia includes low-grade and high-grade intraepithelial neoplasia. The coexistence of epithelial lesions and the subepithelial lesion is rare. We recorded a case of esophageal low-grade intraepithelial neoplasia (LGIN) overlying multiple esophageal leiomyomas and followed with a review of the literature.A 49-year-old female patient came for the treatment of esophageal lesions. The submucosal eminences were observed in the right posterior wall and the left anterior wall of the esophagus by Esophagogastroduodenoscopy (EGD). Additionally, we noticed the mucosa of the right wall with brown background color and the dilated, tortuous vessels by narrow-band imaging (NBI). Then we ensured that the submucosal lesions originated from the esophageal mucosal muscle by endoscopic ultrasonography (EUS) and enhanced CT. Subsequently, the submucosal eminence of the right posterior wall and the overlying mucosal lesion were removed together by endoscopic submucosal dissection (ESD). Postoperative pathological diagnosed esophageal submucosal leiomyoma with focal LGIN. Review EGD showed white scars on the right wall of the upper esophagus three months later, while pathological biopsy showed slight squamous epithelial hyperplasia in the left wall. We decided that the left submucosal lesion can be resected at a selective-time operation, and we continue to follow up as planned.The case of intraepithelial neoplasia overlying the submucosal tumor is rare. Either missed diagnosis or overdiagnosis should be avoided through EGD and pathological biopsy. The coexistence of epithelial lesion and subepithelial tumor is rare. There were few case reports about the coexistence of esophageal leiomyoma and esophageal severe dysplasia –3 and eaA 49-year-old female patient came to our hospital for the treatment of esophageal high-grade intraepithelial neoplasia overlying subepithelial tumor, which was confirmed at another hospital 4 months ago Table 1.Physical examination findings were typically normal. Serological examination showed no obvious abnormality.Esophagogastroduodenoscopy (EGD) demonstrated submucosal eminences in the right posterior wall and the left anterior wall of the esophagus, 18–25 cm away from the incisor teeth, with the widest diameter of about 1.5 cm. Multiple redness and shallow depressions were discovered on the right posterior wall surface additionally (We definite the origin and character of the lesion by endoscopic ultrasonography (EUS). We found that esophageal epithelial lesions are clearly stratified, while subepithelial lesion displayed as multiple uniform hypoechoic, irregularly shaped, and clear-boundary masses, which originated from the esophageal mucosal muscle layer and protruded to the cavity inside and outside. The largest one had an ultrasonic diameter of about 9.9×6.9 mm  and the overlying mucosal lesion were removed together successfully by ESD without intraoperative complications such as bleeding and perforation, while the submucosal eminence of the left wall was not treated temporarily  introduced the concept of intraepithelial neoplasia to diagnose precancerous lesions and early cancers of the gastrointestinal tract; LGIN is equivalent to mild and moderate dysplasia, and high-grade intraepithelial neoplasia (HGIN) is equivalent to severe dysplasia and carcinoma for LGIN , and Ki-for LGIN .en bloc resection and is appropriate for both epithelial and subepithelial lesions  for the publication of any potentially identifiable images or data included in this article.WP, JW, CL, YH, JY conceived and designed the study, and were responsible for the final decision to submit for publication. All authors were involved in the development, review, and approval of the manuscript. All authors contributed to the article and approved the submitted version.This research is funded by Sichuan Medical Association Digestive Endoscopy Special Committee (Jiexiang) Special Research Project (No: 2021XHNJ35)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.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


Sacral giant cell tumors are a rare cause of low back pain and may be challenging to identify via routine clinical examination and radiography. A 47-year-old woman presented to a chiropractor with a one-month history of worsening low back pain with radiation to the posterior thighs, worsened with ambulation, and used a cane to walk. She previously saw an orthopedic surgeon and was diagnosed with lumbar spondylosis, having tried anti-inflammatory medications, exercises, and acupuncture without success. The chiropractor ordered lumbar magnetic resonance imaging which revealed an aggressive sacral lesion and referred the patient to an oncologist. The oncologist performed positron emission tomography/computed tomography and biopsy, confirming a sacral giant cell tumor. A surgical team recommended tumor resection, lumbosacral fusion, radiotherapy, and zoledronic acid infusion. Sacral giant cell tumors are rare and may be challenging to identify via routine radiography. These tumors are an important differential to consider for patients with unexplained lumbosacral symptoms unresponsive to care. Giant cell tumors (GCTs) of bone are locally aggressive primary tumors that rarely undergo malignant transformation ,2. GCTs Most patients with GCT are diagnosed between the ages of 20 and 40 , and theSacral GCTs are challenging to diagnose as they are often clinically silent early on and may not be identified until they reach a large size . GCTs maAs chiropractors often treat low back pain, it is possible that patients with undiagnosed sacral GCT could present to these providers. However, a search of PubMed, Google Scholar, and the Index to Chiropractic Literature on December 16, 2022, using the search terms “chiropractor” and “giant cell tumor,” and hand-searching of a review paper  revealedConsidering that sacral GCTs may cause low back pain and are difficult to diagnose, we present the case of a woman with sacral GCT which was initially missed via radiography and then identified after presenting to a chiropractor who ordered magnetic resonance imaging (MRI).A 47-year-old woman presented to a chiropractor with an exacerbation of frequent pain at the lumbosacral and coccygeal regions, with radiation to the proximal posterior thighs bilaterally, rated 8/10 on the numeric pain rating scale. The patient reported a two-year history of mild, localized low back pain, which had gradually worsened over the past four weeks without a specific injury. She noted being unable to sit, stand, or walk for over 15 minutes, and recently began using a cane to help with ambulation. She noted the pain could be aggravated by coughing or sneezing and lying supine, and her sleep quality was accordingly poor. She endorsed fatigue but denied having any bowel or bladder disturbances, nausea, vomiting, or recent change in weight. She was a non-smoker and did not drink alcohol.She initially tried over-the-counter nonsteroidal anti-inflammatory drugs (NSAIDs) which did not relieve her symptoms. She then consulted with an orthopedic surgeon who ordered lumbar radiography. The radiography report mentioned degenerative changes but did not mention any suspicious findings in the sacral region Figure . The ortOn physical examination by the chiropractor, the patient reported tenderness along the lumbar paraspinal musculature. Palpation revealed motion restriction and tenderness at the T7-T8 and T12-L1 levels, as well as with pressure over the upper sacrum. The examination also revealed hypertonicity at the iliopsoas, rectus femoris, and erector spinae bilaterally. The patient’s lumbar spine range of motion was restricted with an increase in pain at 15° extension . Straight leg raising did not reproduce her symptoms. Her neurological evaluation revealed 4/5 strength  of hip abduction bilaterally and her Achilles muscle stretch reflexes were 1+ bilaterally. Sensation to light touch was intact.The chiropractor’s initial differential diagnosis was (1) lumbosacral radiculopathy based on the lumbosacral pain with radiation and weakness, followed by (2) coccydynia, based on positional tenderness at the sacral region. However, considering the patient’s worsening clinical picture and lack of relief with previous treatments, the chiropractor ordered a lumbar MRI on the day of the first consultation, which was performed two days later. Initially, prior to receiving the MRI results, the chiropractor performed a trial of manual lumbar spinal manipulation, avoiding pressure over the sacrum given the tenderness in this region. This treatment was well-tolerated and provided the patient with transient relief.The patient’s lumbar MRI revealed an irregular bone lesion infiltrating the upper sacrum from S1-S3, measuring 6.4 × 4.7 cm on the sagittal view, in addition to evidence of previously identified degenerative changes Figure . AccordiThe oncologist ordered positron emission tomography/computed tomography (PET/CT) as a screening test for possible metastasis and further characterize the sacral lesion, as well as CT-guided biopsy. Fluorodeoxyglucose whole-body PET/CT revealed a destructive, hypermetabolic sacral bone lesion with encasement of the sacral canal at the S1-S3 levels, and involvement of the presacral soft tissue (Figure CT-guided biopsy was performed using a left posterior approach, targeting the S1-S3 levels. The CT scan revealed a large lytic sacral mass with cortical thinning, a non-sclerotic border, and no signs of pathologic fracture Figure . MacroscHistopathology of the mass revealed an indolent giant cell-rich bone lesion, consisting of abundant osteoclast-like multinucleated giant cells associated with bland-looking spindly fibroblast-like mononuclear cells, young capillaries, and focal stromal hemorrhage, findings typical for GCT Figure . There wBased on the conclusive findings of the imaging, biopsy, and laboratory testing, the oncologist referred the patient to a spine surgery team. After discussion with the surgical team, the patient consented to an en-bloc resection of the sacral tumor with a lumbosacral fusion with instrumentation, followed by radiotherapy and zoledronic acid infusion. Details regarding the patient’s surgery and postoperative course were unavailable from the patient and treating spine surgery department upon request. Written informed consent was obtained from the patient to publish her case and any accompanying images.This case illustrates, to our knowledge, the first patient with sacral GCT identified by a chiropractor. This case reinforces that sacral GCTs are challenging to identify, and because of this, such patients may seek a chiropractic provider due to having symptoms of low back pain that appear musculoskeletal in origin.Providers should be aware of the optimal diagnostic algorithm for sacral GCT given the pitfalls in their diagnosis. For example, symptoms mimic more common forms of low back pain, there may be no overt radiographic features , and neuWhen clinical features are suggestive of a primary sacral tumor such as sacral GCT, CT and MRI may be helpful to assess the extent of the tumor ,2. ScintThere is currently no consensus on the treatment of sacral GCTs . Non-surSurgical options likewise carry significant risks due to the location and potential large size of sacral GCTs. In particular, spinal/pelvic instability, nerve root injury, and risk of infection are potential drawbacks ,6. HowevThis case highlights a woman with low back pain caused by sacral GCT which was initially missed by radiography and then identified by a chiropractor who ordered MRI. As GCTs of the sacrum may cause chronic low back pain and can be challenging to visualize with plain radiography, patients could present to chiropractors unaware that they have a sacral tumor. Sacral GCTs should be diagnosed and characterized with advanced imaging and biopsy to be differentiated from other primary sacral tumors. As there is no consensus strategy on the treatment of sacral GCTs, patients may make an informed decision regarding treatment in conjunction with their surgical and oncologic providers’ recommendations.


Although hemangioma is a common benign tumor of childhood, hemangioma of the genitalia is rarely seen. Multiple treatment options are available for the management of ulcerated hemangiomas but deciding on surgery to treat an ulcerated genital hemangioma is usually difficult due to the lack of guidelines addressing this entity. Here we report a case of an ulcerated hemangioma of the scrotum, that was managed by surgical excision in a four-month-old boy, and a review of related literature. Infantile hemangioma (IH) is the most common tumor of childhood. IHs in certain locations on the skin can be associated with unique medical concerns . UlceratA four-month-old male patient was presented to our emergency department pursuing further management for an ulcerated lesion over an already known hemangioma on the anterior surface of the right hemi-scrotum. The patient was a full-term boy with negative antenatal history and a normal spontaneous vaginal delivery.The patient was initially managed in another healthcare facility; his hemangioma was diagnosed soon after birth, and a pediatrician followed its natural history of uncomplicated rapid growth. However, two months before his presentation, the hemangioma started to ulcerate without discharge, and it was associated with discomfort and mild pain during urination and defecation.General examination was unremarkable and the baseline blood investigations were within normal range. Local physical examination showed a normal circumcised penis and a hemangioma measuring 4 x 4.5 cm, occupying the anterior surface of the right hemi-scrotum totally. Moreover, extensive ulceration covering almost the entire hemangioma surface was also observed. The ulcer was 5 mm deep, indurated with punched-out edges. The floor of the ulcer was a mixture of granulation tissue and slough. No bleeding or discharge was noted Figure . The hemA color Doppler ultrasonography scan of the scrotum showed that the right scrotum was occupied by a heterogeneous hypervascular lesion Figure . A normaA pediatric urologist, a dermatologist, and a general pediatrician assessed the patient. Possible management options including medical treatment and surgical excision were explained to the parents. After a discussion with the parents, a decision for surgical excision was made.Under general anesthesia and caudal block, skin limits of the lesion were marked. A circumferential and tapered incision on healthy scrotal skin surrounding the lesion was made, followed by progressive dissection of the scrotal layers; the pedicle of the feeding vessel was identified and ligated. The hemangioma was entirely excised and the specimen was sent for histopathologic examination Figures -5.We were able to restore the scrotal defect that remained after the excision of the hemangioma with primary closure Figure .The patient, who tolerated the procedure well, was admitted for postoperative observation. He was discharged home in a good condition, 24-hours after the surgery. The histopathology exam confirmed the diagnosis of IH.Six weeks postoperatively, the patient was brought for a follow-up visit. He was doing fine with healed wounds and no complaints. Right orchidopexy after a six-month interval was planned with regular periodic clinic visits.IH is the most common benign tumor in infancy with an estimated incidence of 5% . They vaDiagnosis of IHs usually depends on physical findings. Routine imaging is not recommended for localized hemangioma. However, when the diagnosis is uncertain, or there is a concern of associated structural abnormalities, magnetic resonance imaging (MRI) and ultrasound evaluation may be considered ,3,4,6,7.Hemangiomas can be classified based on their depth , anatomical pattern  , or riskHigh-risk lesions include hemangiomas that may cause permanent scarring and disfigurement, hepatic or airway hemangiomas, and hemangiomas with the potential for functional impairment, ulceration, or associated underlying abnormalities. These hemangiomas should prompt concern, emphasizing increased attention and consideration of early treatment ,3-6. UlcEarly treatment is indicated in approximately 10% of IH cases . Early iUlceration is usually an indication of intervention. Currently, available treatment options include wound care, medical therapy, laser therapy, and surgery . HoweverLarge and ulcerated genital hemangiomas are quite a rare entity, and often pose diagnostic and treatment challenges for the treating surgeon. Various treatment options are described in the literature, but there is no clear consensus on its management due to its rarity ,13. GiveIn our case, no cross-sectional imaging investigation was recommended due to the localized nature of the lesion. Ultrasonography scan was used only to assess the characteristics of the undescended testis. For our patient, both conservative (wound care and propranolol) and surgical management were offered to the parents. The possible lengthy conservative management course, the possible need for future orchiopexy, and the concern of testicular damage were also explained. The decision for surgical excision was made following parental preference.Despite the presence of recent guidelines addressing the management of IHs, developing a rationale for interventions can be challenging. With the dearth of clinical evidence existing about the sequelae of ulcerated scrotal hemangioma, surgical excision is supported by the ambiguity about the effect of these lesions on testicular function and fertility. Until the capability to predict the prospective consequences of scrotal hemangioma is improved, surgical excision of these lesions could be a safe option. Furthermore, parental preferences, after a thorough discussion, might guide planning therapy and follow-up.


Surgical management of post‐esophagojejunostomy aortoesophageal fistula (AEF) has been scarcely reported, but is universally fatal. This report described a case of AEF after total gastrectomy with Roux‐en‐Y esophagojejunostomy and adjuvant chemoradiotherapy for gastric cardiac cancer. A three‐stage hybrid approach was used to successfully manage this complication. First, thoracic endovascular aortic repair curbed bleeding. Second, radical fistula resection eradicated infected areas and adjacent structures. Third, esophageal reconstruction using an ileocolonic conduit restored gastrointestinal continuity. This strategy could be safely feasible for managing post‐esophagojejunostomy AEF. This report described a case of aortoesophageal fistula (AEF) after total gastrectomy with Roux‐en‐Y esophagojejunostomy and adjuvant chemoradiotherapy for gastric cardiac cancer. A three‐stage hybrid‐approach was used to successfully manage this complication, as follows: (1) bridging thoracic endovascular aortic repair controls bleeding and stabilizes hemodynamics; (2) radical fistula resection eradicates infectious source and fragile structures; (3) esophageal reconstruction using an ileo‐colon conduit restores gastrointestinal continuity. This strategy could be safe and feasible for managing post‐esophagojejunostomy AEF. Aortoesophageal fistula (AEF) is rare, but life‐threatening. The management of secondary AEF is even more challenging because severe adhesions caused by previous operation. The optimal strategy remains controversial. We report a case of post‐esophagojejunostomy AEF, which was successfully managed via a three‐stage approach.In January 2019, a 67‐year‐old man with gastric cardiac cancer underwent total gastrectomy and Roux‐en‐Y esophagojejunostomy via a circular staple technique in a hospital. The pathological result showed signet‐ring cell carcinoma, pT3N3aM0, stage IIIB, with positive proximal margins; hence, adjuvant chemoradiotherapy was performed.After 4 months of surveillance, he was admitted to the emergency department of our hospital for intermittent bloody stool; panendoscopy showed ulceration near anastomosis without evidence of local recurrence. Massive hematemesis with hemorrhagic shock occurred 9 months after primary surgery, and panendoscopy revealed one marginal ulcer with spurting bleeding over the esophagojejunal anastomosis. Urgent aortic angiography confirmed the AEF. Thoracic endovascular aortic repair (TEVAR) with a stent graft helped control bleeding Figure .After aortic stenting, the patient's hemodynamic status was stable and there was no bleeding. He was kept at nil per os under parenteral nutrition support for ~2 weeks with the hope that the esophageal defect would heal spontaneously. However, a spiking fever was noticed after resuming oral intake; moreover, panendoscopy revealed a deteriorated AEF with an exposed aortic stent Figure . After mOn gross examination, the AEF had a 1‐cm defect in diameter and presented at the esophagojejunal anastomosis line Figure . The patAt the 1‐year follow‐up, the patient underwent esophageal reconstruction using an ileo‐colon conduit Figure . First, AEF is rare, but life‐threatening and often diagnosed by autopsy.3 also reported a patient with aortogastric fistula 46 days after esophagectomy and intrathoracic esophagogastric anastomosis. The fistula was successfully obliterated via primary closure twice and sequential TEVAR.5 died 11 days after esophagogastrectomy, and the diagnosis of AEF was confirmed on autopsy. In our case, there was a 9‐month interval between primary surgery and the development of AEF, and the patient also underwent adjuvant radiotherapy, which would deteriorate fistula formation and hinder subsequent surgical procedures. This is the first report on concomitant transverse colon resection with radical AEF resection; moreover, we demonstrated the feasibility of using an ileocolonic conduit even after segmental transverse colectomy. Two years after esophagogastrectomy for cardiac cancer, the patient was alive and without recurrence.Only a few post‐esophagojejunostomy AEFs have been reported. Molina‐Navaro et al.Conservative therapies cause most patients with AEF to die of infectious complications or recurrent bleeding; surgical management remains the only chance of long‐term survival.This is a case of post‐esophagojejunostomy AEF who survived more than 2 years. A three‐stage hybrid strategy is safe and feasible to manage fatal secondary AEF, as follows: (1) bridging TEVAR controls bleeding, (2) radical fistula resection eradicates infectious source and fragile structures, and (3) esophageal reconstruction restores gastrointestinal continuity. Most importantly, multidisciplinary approach is the key for success.The authors declare no conflict of interest.


A 30-year-old nulligravid woman with a history of polycystic ovarian syndrome presented for evaluation of left adnexal mass. The patient was referred to the gynecologic oncology clinic after endorsing signs of abdominal pain for a month and the pelvis ultrasound demonstrated hypoechoic solid mass in the left ovary. Magnetic resonance imaging with T1- and T2-weighted images demonstrated progressive centripetal “filling-in” of the mass suggesting a unique variation of malignant ovarian mass, similar to what is seen in hepatic hemangioma. Upon resection of the ovarian mass, pathology reported that the mass was filled with numerous small blood vessels with single later of endothelial cells confirming the diagnosis of ovarian hemangioma, capillary-type—a rare finding. Ovarian hemangiomas (OH) are a rare gynecologic finding, with less than 60 reported cases in the literature In this case report, we present a 30-year-old female with a history of polycystic ovary syndrome (PCOS) who underwent a diagnostic laparoscopy due to inconclusive left adnexal mass findings on ultrasound (US) and magnetic resonance imaging (MRI). Radiology review suggested a unique presentation of the left adnexal mass with MRI findings similar to that of a hepatic hemangioma. Specifically, a progressive centripetal enhancement seen on post-contrast imaging in a typical liver hemangioma which is also seen in the MRI images of this patient's left ovary. Thus, we report an incidental finding of an OH which mimics a presentation of a hepatic hemangioma highlighting the unique radiologic characteristics of this pathology in the ovary2). TSH and HgbA1c were within normal limit. Tumor markers were CA-125 is 12 units/mL and HE4 is 31 pmol/l.A 30-year-old nulligravid woman with a history of PCOS presented to the clinic for evaluation of adnexal mass. The patient was referred by her primary care physician due to increasing left sided abdominal pain for the past month. During the visit, the patient complained of mild pain in that location but denied any fever, chills, chest pain, and shortness of breath. Of note, the patient does not have a family history of breast, cervical, and colon cancer. Despite her PCOS, the patient reported regular menstrual cycle and an unremarkable gynecologic history. Otherwise, the patient had unremarkable physical exam limited slightly by her BMI  and cavernous type (large blood vessels) The ovary is a well vascularized organ of the female reproductive system. However, vascular tumors of the ovary—ovarian hemangiomas (OH)—are very rare with less than 60 cases reported in the literature. In fact, most cases of OH are incidental findings during surgery or autopsy The etiology of OH remains unknown. However, OH can or has been described as a complication of stromal luteinization, stromal hyperplasia, and pseudo-Meigs’ syndrome Given the vascular nature of an ovarian hemangioma, pelvic arteriovenous malformation (AVM) must also be considered in the differential diagnosis of patients with adnexal mass without other signs of malignancy. AVM are aberrant connections between arteries and veins and can be found anywhere in the body. AVM can be asymptomatic, or when multiple shunts are created within an AVM, its size enlarges, making the vessels weak and leak, which then acutely bleed. Depending on the AVM—its size and flow characteristics—treatment can be sclerotherapy or embolization, obviating the need for surgery Our patient had a clinical presentation of persistent localized left abdominal pain that triggered the pelvic US imaging of her primary care provider. This led to the findings of an adnexal mass. Additional MR imaging confirmed a left adnexal mass that was uniquely described similar to that of a liver hemangioma. Given the lack of family history of cancer and other symptoms such as ascites, weight loss, and high CA-125 levels, the clinical suspicion of ovarian cancer remained low, however, not ruled out. The American College of Obstetricians and Gynecologists recommendation is to surgically resect this tumor and send for pathology for definitive diagnosis and correlated treatment The pathology of the adnexal mass correlates with the US and MR radiographic findings which allowed us to definitively diagnose the patient with an OH. Grossly, the specimen consisted of an ovary with attached fallopian tube weighing 24 g. The ovary measured 4.0 × 3.5 × 2.5 cm, and the outer surface was tan-gray and smooth to focally cerebriform. Sectioning revealed a 2 cm in diameter round well-circumscribed red-brown rubbery to spongy nodule comprising 50% of the ovary. The remaining ovarian tissue was unremarkable and included few small cystic follicles . The falMicroscopic examination showed a vascular lesion composed of numerous small blood vessels lined by a single layer of endothelial cells and containing red blood cells  and 8. TOH vary in characteristics from anatomical and pathological presentations. Size ranging from 5 mm to 24 cm in width have been found. These vascular tumors are usually unilateral, but there have been some bilateral cases reported From a radiological standpoint, the preoperative transvaginal US indicated solid mass in the left ovary. In the setting of PCOS, endometrioma was included in the differential diagnosis. However, there was convincing evidence with our MRI images that this adnexal mass carries a unique characteristic. Here, the T1 imaging revealed that the mass is being filled in with contrast which alludes to the vasculature nature of the tumor. Further, the T1-weighted MR images demonstrated a hypointense mass with markedly increased intensity on the T1-weighted images from 30 to 120 seconds. When compared to cavernous hemangioma found in the liver—a neoplasm that has a long transverse relaxation time compared with normal liver parenchyma—the patient's MR images were suggestive of radiologic diagnosis of an OH Ovarian hemangioma is a rare diagnosis that is commonly discovered as an incidental finding during surgery or autopsy. Although there is variance in both age and clinical presentations, these benign tumors should be considered as a possible pathology of any adnexal mass. MR imaging of ovarian hemangioma is comparable to that of a hepatic hemangioma. This radiological similarity can be leveraged by clinicians in including this rare finding in their differential diagnosis and properly treat this vascular lesion.We obtained both verbal and written informed consent from the patient that her case will be used for publication.


Endometriosis of the rectus abdominis muscle is an extremely rare form of extrapelvic localization of the disease. It is usually iatrogenic and develops after caesarean section or gynecological surgery. Preoperative diagnosis is very difficult and a challenge for gynecologists and surgeons; thus, the diagnosis is histological. The treatment of choice consists of wide local excision of the lesion on healthy margins. We cite a case of isolated endometriosis in the rectus abdominis muscles in a 46-year-old patient with a previous caesarean section, the diagnosis of which was made randomly when performing abdominal total hysterectomy for the treatment of chronic pelvic pain. Histological examination of the surgical specimen confirmed the diagnosis. Simultaneously, the surgical specimen of the uterus and ovaries was free of endometriosis. Postoperatively, the patient mentioned discharge of her symptoms. No further therapeutic intervention was deemed necessary, as it was considered that a complete resection of the endometrial tissue implantation from the muscles of abdominal wall was performed. The present case report lay emphasis on the significant difficulties involved in the preoperative diagnosis of endometriosis of the rectus abdominis muscle. Concurrently, it is pointed out that, despite its rarity, individual extrapelvic endometriosis located in the rectus abdominis muscle should be included among other pathological entities in the differential diagnosis of chronic pelvic pain in women of reproductive age, who gave birth by caesarean section or underwent gynecological surgery with abdominal or laparoscopic access. It was  century . Endomet century  and is m century . It is u century . In thisPatient information: a 46-year-old reproductive patient, with a medical history of one caesarean section and a known presence of multiple and small uterine fibroids, visited the outpatient gynecological practice for pain in the lower abdomen for about ten years. The personal medical history of the patient was free. No problems were found from the urinary or gastrointestinal tract, to which chronic pelvic pain could be attributed. In addition to performing appendectomy at a young age, our patient had not undergone any other surgery.Clinical findings: the patient described the onset of pain about 7 months after performing a caesarean section with a Pfannenstiel incision. Gradually, she reported a progressive deterioration of her condition. The pain described by the patient, initially as a feeling of heaviness in the lower abdomen, over the last one to two years has become of increasing intensity. Sometimes the pain may have been more intense during the days of menstruation, but usually the patient described non-intermittent pain in the lower abdomen of approximately the same intensity every day. The clinical examination did not reveal any palpable mass or other type of damage to the abdominal wall.Diagnostic assessment: the findings from ultrasound scan, computerized tomography and magnetic resonance imaging of the abdomen were compatible with the presence of uterine fibroids. The levels of cancer antigen 125 in the blood serum were within the normal range.Therapeutic intervention: based on preoperative evaluation, chronic pelvic pain was attributed to the presence of uterine fibroids. After informing the patient and her relatives in more detail regarding the therapeutic approach of the disease, it was decided to perform abdominal total hysterectomy. Intraoperatively, in the rectus abdominis muscles and slightly above the level of the pubic symphysis, infiltration of the muscular wall was found by a hard-textured flat mass, in length 4-5 cm, the surface of which was solidly adhering to the parietal peritoneum. A wide resection of the lesion was performed, including bilaterally a widespread part of the wall of the rectus abdominis muscles and the matted peritoneum (ritoneum . The plaritoneum . HistoloFollow-up and outcomes: postoperatively, the patient mentioned relief of her symptoms. No further therapeutic pharmaceutical intervention was recommended, as it was considered that the endometriotic foci was completely resected from the abdominal wall.Informed consent: this was sought and obtained from the patient. Anonymity was maintained for confidentiality.Abdominal wall endometriosis is one of the rarest extrapelvic forms of endometrial disease. It is usually iatrogenic and develops after caesarean section or open or laparoscopy surgeries for the treatment of gynecological diseases . The lesDespite the great development that has been achieved in recent years in imaging techniques, preoperative diagnosis is difficult and a challenge for gynecologists and surgeons. The diagnosis is usually made late and accidentally during gynecological surgery or postoperatively. Diagnosis is confirmed after histological examination of the surgical preparation . The cycUltrasound is a first line tool in diagnostics approach to extrapelvic endometriosis. With abdominal ultrasound can be detected, intramuscular endometriosis in the anterior abdominal wall, the characteristics of which may vary from a completely solid mass or a mix echogenic mass with solid and cystic elements . Also, tThe treatment of endometriosis of the rectus abdominis muscle requires surgery. The treatment of choice is wide local resection of the lesion on sound boundaries. The local exclusion of the lesion that can be done under ultrasound monitoring  is usualPreoperative diagnosis of extrapelvic endometriosis with individual localization in abdominal muscles is a challenge in daily clinical practice. Despite the rarity of endometrial lesion in the muscular abdominal wall, the significant increase of the percentage of caesarean sections observed in recent years is necessary the inclusion of this rare extrapelvic form of endometriosis in the differential diagnosis between all the painful masses of the abdominal wall, thus allowing the early diagnosis and avoidance unnecessary diagnostic and therapeutic interventions. Early diagnosis and selection of the most appropriate therapeutic manipulations are judged currently necessary in order to minimize the risk of recurrence and to avoid the possibility of malignant recurrence of the disease.


Extraskeletal soft tissue chondroma (STC) is a rare benign tumor. Soft-tissue chondromas rarely occur in the oral cavity. In this study, we aimed to confirm a slow-growing tongue mass using magnetic resonance imaging.A 60-year-old woman presented with a painful, slow-growing tongue mass that had persisted for 17 years. Intraoral examination revealed a pedunculated mass covered with mucosa on the right side of her tongue.CT and MRI revealed a lobulated heterogeneously enhancing mass without calcification. Compared with previous images obtained 17 years prior, the mass presented slow growth, more prominent enhancement, and lobulated contour. Histopathological examination confirmed the presence of STC.Excision of the mass surrounding normal tissue was performed under general anesthesia.During 1-year follow-up period, no recurrence was observed.In this study, STC lesions were slow-growing, and changed from weakly homogeneous enhancement and clean margins to markedly heterogeneous enhancement and lobulated margins over time. Other locations include the dura, larynx, skin, and fallopian tubes. STCs rarely occur in the oral cavity. The subsites where these tumors have been reported include the tongue, cheek, hard and soft palate, masseter muscle, and masticatory space.Soft tissue chondromas (STCs) are rare, benign tumors that occur in extraosseous and extrasynovial locations.STCs usually present as asymptomatic, slow-growing, well-defined nodules that expand into surrounding tissues. They are predominantly composed of adult-type hyaline cartilage, devoid of other differentiated elements, except for osseous, fibrous, and/or myxoid stroma.The tongue is the most common site of occurrence of oral STCs. Oral STC presents as an elastic semicircular nodule with a diameter of approximately 5 mm on the dorsum of the tongue. Forty-five cases of fibrochondroma of the tongue have been reported in literature.However, there have been few studies on the imaging findings of chondromas in the oral cavity. Therefore, in this study, slow-growing chondrocytes were identified using computed tomography (CT) and magnetic resonance imaging (MRI), and the changes in radiological findings as they grew were investigated.2A 60-year-old woman presented with complaints of a slightly painful, slow-growing, 17-year standing lesion. Intraoral examination revealed a pedunculated multilobulated mass covered with mucosa on the right side of the tongue.Radiologically, pre-contrast CT revealed a well-delineated homogeneous soft-tissue mass. Contrast-enhanced CT showed a peripheral enhancing mass with an internal heterogeneous enhancing portion, and the size was measured to be approximately 3.0 cm × 2.0 cm × 1.6 cm. The soft tissue mass was separated from the bone, but there was no chondroid matrix calcification , which are typically punctate, curvilinear, or ring-like. On MRI, tumors usually have a multilobulated appearance with low-to-intermediate signal intensity on T1-weighted images, high signal intensity on T2-weighted images, and peripheral or septal contrast enhancement. Chondroid matrix calcifications may be evident on MRI as areas of low signal intensity, but these types of calcifications are easily recognized on CT.–12 Compared to the MRI image taken 17 years prior, as the size of the lesion increased, there was a change from weakly homogeneous enhancement and clean margins to markedly heterogeneous enhancement and lobulated margins. When we correlated the change in heterogeneous enhancement with pathological findings, it was considered that the sclerotic and fibrotic areas, which are the backgrounds of these nodules, were enhanced, not the multiple nodular areas composed of mature chondrocytes. The distinction between these areas became clear as the size of the lesions increased over time, resulting in markedly heterogeneous enhancement. And the long diameter of the lesion has increased in size from 1 cm to 3 cm over 17 years, and it is thought that it is a slow growing mass, which is considered to have shown as a lobulated margin. Low-grade chondrosarcomas may present variable enhancement patterns and margins, but soft tissue masses with heterogeneous enhancement and lobulated margins are common imaging features in many cases. It can be seen that as the size of tumors increases, low-grade chondrosarcomas have radiologic characteristics.Distinguishing between chondromas and LGC is a common challenge, as the lesions are both histologically and radiographically very similar. Diagnosis is difficult because cartilaginous neoplasms have different histological patterns, ranging from benign chondroid tumors to malignant undifferentiated neoplasms. The MRI features of mesenchymal chondrosarcoma are variable, but the tumor often appears as a lobulated soft tissue mass with low signal intensity on T1-weighted images, variable and heterogeneous signal intensity on T2-weighted images, and complex heterogeneous enhancement after intravenous administration of gadolinium-based contrast material. Chondroid matrix calcifications may be evident on MRI as curvilinear or stippled areas with low signal intensity. Plain radiography and CT are particularly helpful in identifying these calcifications.Radiologically, minor salivary gland tumors, STCs, and LGC were considered differential diagnoses. In our case, it was difficult to make a diagnosis because ring and arc calcifications, which are characteristic findings of chondromas, are not visible. Since the size of a well-defined lesion is less than five–six centimeters and surrounding bone destruction is not clear, it was considered benign rather than malignancy.Surgical excision is the treatment of choice for this condition. Ten to fifteen percent of tumors may recur locally after surgical excision.4STC is a benign cartilage-forming tumor that occurs in the extraarticular soft tissues. Clinically, these tumors present as slow-growing, non-tender masses that are sometimes painful and not attached to the bone. Radiologically, these tumors show well-defined, heterogeneously enhancing masses. Similar to our case, as the size of the tumor increased, it showed LGC characteristics. As the diagnosis of oral STCs may be difficult, radiologists should always keep a differential diagnosis in mind.This work was supported by a National Research Foundation of Korea (NRF) grant funded by the Korean government (MSIT) (No. 2021-0080).Conceptualization: Yoonsoo Park.Supervision: Ha Youn Kim, Joo Heon Kim.Writing - original draft: Yoonsoo Park.Writing - review & editing: Dong-Sik Jang, Ha Youn Kim, In-Kyu Yu.


FOXO1 fusion in a tumor is one of the most important prognostic factors in rhabdomyosarcoma. Most histologically defined alveolar rhabdomyosarcomas bear a FOXO1 fusion. We discuss a case that was initially thought to be a rhabdomyosarcoma but was later discovered to have an RREB1-MRTFB fusion. This fusion has never been reported in rhabdomyosarcoma but typically characterizes ectomesenchymal chondromyxoid tumor (ECT), a neoplasm with typically rather benign behavior. In this article, the authors discussed whether this patient’s aggressive presentation represents a variation of ECT or an unusual case of rhabdomyosarcoma.The presence of a  Rhabdomyosarcoma is the most common soft tissue sarcoma in children and represents about half of pediatric soft tissue sarcomas; however, it is rare in adults ,2. SincePAX-FOXO1 fusion-positive or fusion-negative rhabdomyosarcoma [FOXO1 fusion. Spindle cell/sclerosing rhabdomyosarcoma histology is generally associated with an MYOD1 mutation, and a VGLL2-NCOA2 fusion can be seen in a congenital subtype [FOXO1 fusion-positive tumors include either a PAX3-FOXO1 or a PAX7-FOXO1 fusion, and the presence of a FOXO1 fusion is an important marker of tumor biology. Fusion-positive alveolar rhabdomyosarcoma is generally worse than the so-called fusion-negative alveolar rhabdomyosarcoma or embryonal rhabdomyosarcoma, although fusion-negative alveolar rhabdomyosarcoma is commonly approached in a way similar to embryonal rhabdomyosarcoma [One of the most important discriminators in rhabdomyosarcoma is whether the tumor is a osarcoma . Most al subtype ,8. In coosarcoma ,10.RREB1-MRTFB fusion. This fusion has never been reported in rhabdomyosarcoma but typically characterizes ectomesenchymal chondromyxoid tumor (ECT), a recently described mesenchymal tumor with a typically benign clinical course [In this article, we discuss a case initially which was classified as rhabdomyosarcoma, mostly due to its apparent myogenic differentiation, but the subsequent mutational analysis found an l course . WhetherA 26-year-old man with no significant past history presented with hemoptysis one day prior to admission. He had a one-week history of increasing sore throat and voice change and had been spitting up blood throughout the day of admission. His family history was unremarkable. He had no history of smoking, alcohol use, or recreational drug use. On examination, he had signs of hypovolemic shock with hypotension (BP 60/26), altered mental status, elevated lactate (5.8 mmol/L), and acute renal failure with a creatinine of 1.32 mg/dL. He was treated with two liters of IV fluid, two units of packed red cells, and 1 gram of tranexamic acid IV in the emergency department. Tranexamic acid inhibits the conversion of plasminogen to plasmin, thereby exerting an anti-fibrinolytic effect. Clinically, he stopped spitting up blood after stabilization. After hemodynamic stabilization, he underwent a direct laryngoscopic examination; this revealed a smooth, soft, reddish mass on the right side of the oropharyngeal tongue. No active bleeding was seen at the time of the laryngoscopy. A pre-chemotherapy CT scan of the neck demonstrated a heterogeneously enhancing mass measuring 4.9 cm x 3.9 cm x 5.1 cm at the base of the tongue and lingual tonsils extending from the tip of the soft palate down to the epiglottic vallecula , as shown in Figure max: 7.3) tumor arising from the tongue base, filling in the vallecula and abutting the suprahyoid epiglottis with no distant metastasis. There was intense fludeoxyglucose (FDG) uptake in symmetric nasopharyngeal adenoidal thickening and slightly enlarged FDG-avid bilateral level 2 lymph nodes.Immunohistochemical staining for Ki-67, a cellular proliferation marker, was moderately increased at 10%. Positron emission tomography-computed tomography (PET-CT) revealed an exophytic, hypermetabolic (SUVFOXO1 fusion was detected by fluorescence in situ hybridization (FISH) assay in the resection specimen.The patient received chemotherapy with cyclophosphamide, doxorubicin, and vincristine (CAV) alternating with ifosfamide and etoposide (IMV) for five cycles. Restaging MRI showed a residual, heterogeneous mass of approximately 2 cm x 1.3 cm in size, which was centered in the tongue base with no lymph node or distant metastasis , and an RREB1-MRTFB fusion.The patient received postoperative proton radiation therapy (3600 cGy in 20 fractions) and two cycles of CAV/IMV. Follow-up MRI showed no evidence of disease with the most recent follow-up at two years after completion of the treatment. The tissue was later sent for next-generation sequencing (NGS) using the Sema4 platform , which confirmed no FOXO1-negative rhabdomyosarcoma and was treated for that condition. The patient presented with acute onset of voice changes and massive hemoptysis leading to hypovolemic shock. Later, the genetic analysis demonstrated an RREB1-MRTFB fusion. The majority of alveolar rhabdomyosarcoma cases are associated with a characteristic chromosomal translocation involving a fusion between PAX3 or PAX7 with FOXO1, which is associated with more aggressive behavior and poorer prognosis [PAX-FOXO1 fusion is viewed as a distinct biological subtype. The tumor biology and outcome are similar between fusion-negative alveolar rhabdomyosarcoma and embryonal rhabdomyosarcoma; however, they are significantly different from fusion-positive alveolar rhabdomyosarcoma [CTNNB1, FBXW7, and BCOR, as well as gain-of-function mutation of genes in the RAS pathway, including RAS, PIK3CA, and FGFR4 [We describe the case of a patient with a very aggressive clinical course that was felt to be a rognosis . Embryonosarcoma . A comprnd FGFR4 . No mutaRREB1 with the C-terminal transcriptional activation domain and leucine zipper-like domain from MRTFB [RREB1-MRTFB fusion has never been reported in rhabdomyosarcoma but has been identified in 90% of ECT, a mesenchymal tumor with generally benign behavior which is found almost exclusively on the anterior dorsal tongue and stains positively for smooth muscle actin, desmin, and myogenin [RREB1-MRTFB fusion with no tongue involvement in a 53-year-old man [The predicted fusion protein found in this neoplasm contains the N-terminal zinc finger domain of om MRTFB . The RREmyogenin . ECT wasmyogenin ,15. Sieg-old man , and sub-old man . A recen-old man . A recen-old man . In thisGiven the tumor location in our case and the clinical presentation, it is possible that this patient’s tumor may be a biologic variant of ECT with more aggressive biology than classic ECT, based on its apparent rate of growth. Alternatively, this case may represent an unusual variant of rhabdomyosarcoma.RREB1-MRTFB fusion in rhabdomyosarcoma. The location of the tumor and presentation could represent an atypical rhabdomyosarcoma or a biologic variant of a different mesenchymal tumor, ECT. This case highlights the importance of NGS in fusion-negative rhabdomyosarcoma, especially in the head and neck. We suggest that this genetic fusion should be investigated in rhabdomyosarcoma with no PAX-FOXO1 fusion especially if they are located in the oral cavity and tongue area to aid in the diagnosis and better clarify the biology of this disease.This is the first report on


A 43-year-old man with a short history, incomplete hemisymptomatic and cranial nerve IX and XII lesions, and an exophytic, unilateral  ring-like medullary lesion shown on MRI.Why not surgical exploration, if personal, even published, expertise in brainstem surgery and anatomical and neurophysiological knowledge about safe entry zones is practiced and documented convincingly in a video? The navigated resection of a circumscribed firmer tumor appears more like a lower-grade glioma than the finally diagnosed GBM—locally very rare in adults—avoiding additional neurological deficits. Intraoperative MRI could offer another potential for resection control.It remains open how far “only biopsy” prior to tailored radio-chemotherapy—as a conventional procedure—would have changed the early and long-term outcomes.


Only 5% of all cases of intussusceptions occur in adults. Although it is known to occur frequently due to inflammatory bowel disease, postoperative adhesions, or neoplastic masses, inflammatory fibroid polyps (IFP), which are rare lesions of the gastrointestinal tract, may present this clinical picture. In rare cases of intussusception due to IFP, clinical suspicion should be kept in the foreground and mind in the differential diagnosis. In this article, the purpose was to present the clinical, radiological, and pathological findings and the treatment of obstruction caused by this rare lesion. Intussusception is the condition that appears when the proximal bowel loop is invaginated into the distal intestinal loop lumen. Its prevalence is only 5% in all invagination cases in adults. There is often an underlying organic lesion . It ofte9/L) in the blood cell count, and other blood parameters were normal. There were one/two air-fluid levels in the abdominal X-ray, the non-contrast abdominal tomography performed for further examination revealed an appearance that was compatible with ileocolic invagination of the hypodense appearance of the intestinal loop in the peripheral area and with mesenteric fatty tissue in the center , leukocytes were 12.84 (normal: 3.8-10 × 10A filling defect in the lumen was noted in the proximal soft tissue density. A minimal dilatation with a 3-cm diameter was detected in the small intestinal loops. When findings consistent with invagination and ileus were detected, urgent surgery was planned for the patient. It was observed during the exploration that the segment located 10 cm distal to the ileocecal valve was invaginated into the ascending colon along with the ileocecal valve. Dilatation had developed because of obstruction proximal to the invagination. When a solid mass was detected in the ascending colon, the patient underwent right hemicolectomy and end-to-side anastomosis Figure .The patient was discharged on the 7th postoperative day with full recovery. The distal 50 cm ileal segment was examined in the colonoscopic examination in the postoperative 4th month, and no pathological finding was found. The specimen that was extracted macroscopically consisted of a 9-cm-long small intestine and 23-cm-long colon resection material. When the material was opened, an 11x5x4 cm polypoid lesion was found, which originated from the ileum and which invaginated the colon lumen with the ileocecal valve. It was observed on the section surface that this area consisted of a 4.5-cm diameter yellow-white colored nodular solid lesion distally and there was an invaginating small intestine segment. It was observed in the microscopic examination of the samples that were taken from the lesion that the surface was covered with focal ulcerated small intestinal mucosa. In the submucosal area, inflammatory cells rich in eosinophils, vessels, spindle, and stroma that consisted of stellate cells were observed. Atypia, mitosis, and necrosis were not observed. In the immunohistochemical examination, positive immunoreactivity was detected with CD34, and negative immunoreactivity with CD117, S-100, Desmin, Dog-1 in spindle cells (Figures One of the rare causes of intestinal obstruction is invagination. IFPs are one of the rarest causes of invagination in adults. IFPs were first reported in 1949 as “gastric submucosal granuloma with eosinophilic infiltration” . AlthougIFPs can be observed in many parts of the gastrointestinal tract. The most common area is the antrum, which is followed by the small intestines. Colorectal IFPs are seen between 4 and 7% . Ileum-lIFP is an asymptomatic lesion, and polyps in the small intestine or colon give symptoms with signs of invagination or obstruction. Nausea, vomiting, diarrhea, bloody stools, and tenesmus may also accompany these clinical conditions . The ageThe first tool used in the diagnosis of invagination was the standing direct abdominal radiography; however, it is generally insufficient to make a specific diagnosis. Double-contrast imaging methods are more useful in diagnosis. Colonoscopy can also be used for diagnosis in colonic IFPs, but in the literature, it is reported that invagination develops over IFP after colonoscopy , 8. The Obstruction due to inflammatory fibroid polyp is a rare phenomenon. The underlying cause may be benign, malignant, or iatrogenic. High clinical suspicion is required to make this rare diagnosis in patients presenting with classical ileus. It is important to reveal this lesion, which has no potential for malignancy, with appropriate imaging and diagnostic techniques, to perform appropriate treatment methods, and to examine the postoperative specimen for possible additional diagnoses with appropriate pathological methods.