Juvenile ossifying fibroma is a rare benign destructive lesion of nose and paranasal sinuses. It occurs in the craniofacial bones of children below 15 years of age. It is usually discovered in the mandible and maxilla and rarely in the paranasal sinuses.We present a case of a 15-year-old girl with extensive right ethmoid sinus juvenile ossifying fibroma with intracranial and orbital involvement that was managed endoscopically at our center followed by lateral rhinotomy and frontal craniotomy with reconstruction due to the aggressive nature and recurrence of the disease. Patient was followed up post operatively for 3 years and was found free of symptoms ever since.In this case report we present our experience in managing this aggressive recurrent disease of juvenile ossifying fibroma which requires multiple endoscopic and open surgical procedures. Such pathology needs a close and long follow up due to the aggressive nature of this disease. •Juvenile ossifying fibroma is a rare benign destructive lesion of nose and paranasal sinuses.•Occurs in the craniofacial bones of children below 15 years. It is usually discovered in the mandible, maxilla and rarely in the paranasal sinuses.•Most of the cases are asymptomatic and present lately when complications occur. Few cases involving the ethmoid sinus were reported.•The main-stay management is debulking the lesion by preserving adjacent vital structures like the orbit.•Long term follow-up is recommended due to high recurrence rate. Patients with JPOF were found to be usually older than patients with JTOF. During treatment, the recurrence rate was higher in the case of JOF treatment by curettage and enucleation only and less when enucleation was followed by curettage/osteotomy The purpose of this study was to report a rare case of aggressive juvenile ossifying fibroma of the ethmoid sinus with orbital and intracranial extension and to review the published data on similar cases. The work has been reported in line with the SCARE criteria 22.1A 15-year-old girl presented to King Fahad specialist hospital, Dammam, complaining of progressive right-sided eye proptosis associated with right-sided nasal obstruction. The proptosis developed gradually over the course of three months; however, the vision remained normal. Upon discovery, the patient was admitted, and pediatric, ophthalmology, otolaryngology, neurosurgery opinion was sought. When questioned, the patient denied experiencing any discomfort, headaches, changes in vision, or diplopia. There was no weight loss nor loss of appetite recorded. On inspection, no signs of allergic rhinitis or sinusitis were found.After ocular examination, the patient showed right-sided eye proptosis, which is common in cases of ethmoidal involvement. For further examination, nasal endoscopy was performed. The endoscopy revealed several nasal polyps on the right side, as well as a deviated nasal septum to the left due to the strain created by the polyps.2.2A CT scan of the nose and paranasal sinuses revealed a large, calcified mass based on the right orbital plate; the mass is expansile with heterogeneous density and a ground-glass appearance, and it is correlated with the remodeling of the surrounding bony margins. With the obliteration of the right ostiomeatal complex, the mass has spread to the right ethmoid cavity. Orbital proptosis is present , Fig. 2.2.3The patient underwent multiple operations, including endoscopic debulking of the lesion followed by lateral rhinotomy, and finally, frontal craniotomy with reconstruction as a combined procedure with the neurosurgery team . Post-op3In this report, we presented a case of aggressive juvenile active ossifying fibroma of the ethmoid sinus with orbital and intracranial extension. The case had to undergo multiple operations, including endoscopic debulking of the lesion followed by lateral rhinotomy, and finally, frontal craniotomy with reconstruction as a combined procedure with the neurosurgery team.Surgical excision of the lesion is crucial because of its aggressive development and high rate of recurrence. Therefore, also, post-operative follow-up and inspection for recurrence are mandatory A previous case report of a psammomatoid ossifying fibroma (JPOF) in an 8-year-old boy reported recurrence within a year after treatment, even with surgical enucleation and curettage JOF can also extend to the brain and the eye, as presented in our report. Another report with similar findings was of a 12-year-old boy who had a JPOF arising from paranasal sinuses and which in turn extended to the anterior skull base and the orbit. The diagnosis of similar cases, however, is a bit tricky given the similarity between them and other types of ossifying fibroma or meningioma On radiological examination, lesions appear variable depending on the stage and the degree of calcification. JOF has three stages in radiographs; the first stage appears as a well-defined radiolucency without any sign of internal calcification, the second stage includes spots of radio-opaque material within the radiolucency. The third and the last stage is presented as a complete radio-opaque lesion In addition to this case, nasal polyps were also noted in previous cases of JOF, leading to nasal obstruction, which is a common symptom of JOF when it affects the sinuses Despite its being benign, JOF tends to cause more serious damage and erosion, in addition to adherence to soft tissue in comparison with malignant lesions such as RMS or chondrosarcoma 4Juvenile ossifying fibroma is a rare entity of benign fibro-osseous lesions. Most of the cases are asymptomatic and present lately when complications occur. Few cases involving the ethmoid sinus were reported. The main-stay management is debulking the lesion by preserving adjacent vital structures like the orbit. Long-term follow-up is recommended due to its high recurrence rate. In this report, we presented a case of aggressive juvenile ossifying fibroma of the ethmoid sinus with orbital and intracranial extension. The patient had no recurrence after surgical interference.The data used to support the findings of this study are included within the article. Also, they are available from the corresponding author upon request.Written informed consent was obtained from the patient's next of kin for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Not commissioned, externally peer-reviewed.Ethical approval by IRB for case reports is not needed in this institution.This work was not supported by any forms of funding.Ali Almomen.This is not the first in man study.Dalia Al Arfaj: data collection and analysis, writing the original manuscript draft.Haifa Alenzi:, review and editing of the manuscript.Musab Bakri: data analysis.Ali Al Momen: study concept, data analysis, and final approval of manuscript.The authors declare that there is no conflict of interest regarding the publication of this paper.


Mesenteric and omental cysts can be seen at any age, with one in three cases seen in patients under 15 years old. These cysts represent one in 20,000 pediatric admissions. Here we share the case of a five-year-old female patient at a health center in a developing country, with the purpose of contributing to documentation in the region.  Mesenteric and omental cysts are cystic abdominal swellings present with painless, often uniform enlargement of the abdomen. They can occur at any age, but one in three cases is seen in patients younger than 15 years old .The reported incidence of mesenteric cysts is one in 140,000 hospital admissions and one in 20,000 pediatric admissions -5. AboutAn otherwise healthy five-year-old female patient presented to the Emergency Department with a history of crampy abdominal pain and vomiting for 12 hours of duration. The patient did not have any relevant personal or family history. On physical examination, a distended abdomen and remarkable tenderness to palpation were noticed. An abdominal ultrasound revealed a cyst in the abdominal cavity, which needed a computerized axial tomography  and computed tomography (CT). If the diagnosis is unclear and the physician is trying to decide between a conservative approach and a surgical intervention, these imaging modalities can help distinguish mesenteric and omental cysts from other intra-abdominal cysts. Abdominal CT helps to characterize the size more precisely, delimits the extent of the mass, shows the relationship with other abdominal organs, and helps detect calcium deposits, as well as can help detect compression of the adjacent structures and whether there is a retroperitoneal extension ,14.Given the difficulty in distinguishing between these lesions on ultrasound, most patients will need a laparoscopy to confirm the diagnosis. This can occur in the event of cyst torsion, as it is likely to mimic a condition requiring urgent exploration . In cases where the patient is not a good candidate for surgery, additional imaging with CT or MRI can help limit the differential diagnosis ,3,15. ThMesenteric cysts can be found anywhere in the mesentery of the small intestine, large intestine, or retroperitoneum. They are most frequent along the mesentery of the small intestine (60%), followed by the large intestine (24%) and the retroperitoneum 15%) [5% [1].The treatment of omental and mesenteric cysts depends on their presentation. These presentations range from an incidental radiographic or intraoperative finding to a serious and life-threatening finding secondary to volvulus or intestinal obstruction. The diagnosis is usually made in the setting of acute abdominal pain, and treatment is usually surgical resection ,4,5.In cases where the cyst affects a significant part of the intestine or extends to a large part of the retroperitoneum, partial resection with marsupialization and the use of sclerotherapy agents have been described. The goal of the intervention is to eliminate the source of the symptoms, preferably by complete excision. Due to the high recurrence rate of drainage alone, the cyst lining must be sclerosed with agents such as 10% glucose or ethyl alcohol .Approximately 40% of cysts are discovered incidentally at laparotomy for other conditions and must be removed if the outcome of the primary surgery is not negatively affected. Cysts that are discovered on abdominal pain imaging may be the cause of the symptom and should be removed as soon as possible .Observation alone can result in twisting of the cyst, intestinal obstruction, volvulus, or bleeding in the cyst that can mimic an acute abdomen. Moreover, cysts can get infected or might rupture. Although rare, a malignant transformation has been reported; therefore, it is not advisable to keep a cyst unresected .Complete excision of the cyst is curative. However, because of the multiloculated nature of some of the cysts, intraoperative rupture can lead to incomplete resection and thus recurrence. Some small cysts may also be missed, which could become enlarged and cause a recurrence. The recurrence rate is generally low (0-15%) and is even lower after complete surgical resection ,4.In pediatric patients, mesenteric cysts can present with abdominal pain, vomiting, and bloating, as seen in this case. Ultrasound is commonly used due to availability and non-invasiveness, but abdominal CT delimits the extent of the mass more precisely. It is not normally associated with malignancy in children and the goal of the intervention is to improve the symptoms. Complete excision of the cyst can be curative with a low recurrence rate.


A patient, recently diagnosed with non-Hodgkin lymphoma, presented with acute tetraplegia after surgical cervical lymph node biopsy. MRI of the cervical spine demonstrated an epidural space-occupying lesion with compressive myelopathy. While epidural hematoma was the tentative diagnosis, intra-operatively non-Hodgkin lymphoma was found. Several factors may have accounted for the inaccurate interpretation of the MRI: the acute clinical presentation appearing shortly after surgery, the non-specific signal intensities of (hyper-) acute hematomas, the lack of contrast-enhanced images, and the absence of the FDG-avid spinal mass in the PET/CT-report. Without radiological features of invasiveness and contrast-enhanced images, careful interpretation is mandatory for space-occupying epidural lesions.Caution is needed when interpreting an epidural space-occupying lesion in the absence of contrast-enhanced images. Non-Hodgkin lymphoma is the most common hematological malignancy worldwide, comprising 3% of all cancer diagnosis . LymphomA 60-year-old woman with general malaise and dyspnea presented at the emergency department. Clinical history and physical examination were unremarkable. Computed tomography (CT) scan of the thorax and abdomen revealed extensive mediastinal and retroperitoneal mass lesions. Positron emission tomography – CT (PET-CT) scan showed fluorodeoxyglucose (FDG) avid masses throughout the body consistent with lymphoma. Excisional cervical lymph node biopsy was performed under general anaesthesia, and pathological analysis confirmed aggressive B-cell non-Hodgkin lymphoma.Unfortunately, the patient developed acute tetraplegia the first post-operative day. Magnetic resonance imaging (MRI) of the cervical spine revealed a space-occupying lesion in the posterior epidural space  with fusIn retrospect, the FDG-PET-CT scan already showed FDG-avid disease in the cervical spinal canal consistent with intraspinal extradural lymphoma, which was inconspicuous on the cross-sectional anatomical information provided by the CT part of the examination .Secondary central nervous system (CNS) lymphoma indicates CNS spread of  lymphoma originating elsewhere. Secondary spinal lymphomas are more frequent than primary lymphomas . Intrasp2Several clinical, radiological and technical features of the epidural lymphoma in this patient may have contributed to spinal lesion being mistaken for an hematoma initially.First, the (hyper-)acute symptomatology of spinal cord compression might rather have been suggestive of a rapidly growing expansile process, such as a hematoma. It is unclear to which degree peri-operative manipulation of the neck  could have triggered the acuteness of this clinical presentation.Second, the radiological features of the lymphoma were erroneously interpreted as a hematoma. As for hematomas, the lymphoma in this patient strictly respected the anatomical boundaries of the epidural space, with normal (bony) posterior elements and no extension in either the intradural or interspinous spaces. In addition, the signal characteristics of an epidural lymphoma may resemble those observed in an hyperacute or acute hematoma on both T1- and T2 turbo spin echo-sequences, before the spontaneously bright T1-signal of methaemoglobin emerges in the subacute stage .Third, contrast enhanced MRI was not performed because the clinical history was inappropriately taken into consideration. Homogeneous contrast enhancement as expected in lymphoma would have made this a more straightforward diagnosis, in particular in the clinical setting of systemic lymphoma .Last, despite avid FDG-uptake, involvement of the spinal canal was overlooked on PET-CT performed days before the onset of paraplegia.In conclusion, lymphoma should be considered in case of an epidural space-occupying lesion presenting with spinal cord compression. Since the majority of spinal lymphomas represent secondary disease, close correlation with clinical history is essential. Lack of invasive imaging features should not withhold the radiologist from gadolinium administration in this clinical setting.


Background: Cerebral cavernous malformations (CCMs) are a major type of cerebrovascular lesions of proven genetic origin that occur in either sporadic (sCCM) or familial (fCCM) forms, the latter being inherited as an autosomal dominant condition linked to loss-of-function mutations in three known CCM genes. In contrast to fCCMs, sCCMs are rarely linked to mutations in CCM genes and are instead commonly and peculiarly associated with developmental venous anomalies (DVAs), suggesting distinct origins and common pathogenic mechanisms. Case report: A hemorrhagic sCCM in the right frontal lobe of the brain was surgically excised from a symptomatic 3 year old patient, preserving intact and pervious the associated DVA. MRI follow-up examination performed periodically up to 15 years after neurosurgery intervention demonstrated complete removal of the CCM lesion and no residual or relapse signs. However, 18 years after surgery, the patient experienced acute episodes of paresthesia due to a distant recurrence of a new hemorrhagic CCM lesion located within the same area as the previous one. A new surgical intervention was, therefore, necessary, which was again limited to the CCM without affecting the pre-existing DVA. Subsequent follow-up examination by contrast-enhanced MRI evidenced a persistent pattern of signal-intensity abnormalities in the bed of the DVA, including hyperintense gliotic areas, suggesting chronic inflammatory conditions. Conclusions: This case report highlights the possibility of long-term distant recurrence of hemorrhagic sCCMs associated with a DVA, suggesting that such recurrence is secondary to focal sterile inflammatory conditions generated by the DVA. KRIT1/CCM1, CCM2 and PDCD10/CCM3, which have been implicated in a plethora of essential physiological functions, including modulation of cadherin- and integrin-mediated cell adhesion, actin cytoskeleton dynamics, and signal transduction involved in the maintenance of endothelial barrier stability [Cerebral cavernous malformations (CCMs), also known as cavernomas or cavernous angiomas, are vascular malformations characterized by clusters of abnormally dilated capillary channels lined by a thin and leaky endothelial cell layer predisposed to rupture ,2. They tability . Howevertability ,9. Convetability ,12, suggtability , while ptability ,15,16,17DVAs are the most common cerebrovascular malformations, accounting for up to 60% of the total and being present in approximately 3% of the general population ,19. TheyCCMs can either remain asymptomatic or manifest across a wide range of different clinical symptoms, including seizure (30% of cases), intracranial hemorrhage (ICH) (26% of cases), and focal neurological deficits (16% of cases) , with anThe pathophysiology of CCM disease remains to be fully elucidated, as there is still some controversy regarding the multiple mechanisms proposed so far. Among others, recent genetic analyses of surgically excised lesions from sCCM patients and distinct studies in conditional knockout mouse models have clearly demonstrated that homozygous loss-of-function mutations in CCM genes are neither necessary nor sufficient to cause the development of CCM lesions, thereby limiting the two-hit model proposed previously ,43 and sHerein, we report a case of long-term distant recurrence of a sporadic CCM lesion in the proximity of a voluminous DVA draining in the superior sagittal sinus (SSS), and provide contextualized support to the intriguing hypothesis that such recurrence is secondary to local sterile oxy-inflammatory conditions generated by the DVA.Our case report concerns a patient affected by the sporadic form of CCM disease, who underwent two neurosurgical interventions, 18 years apart, for the excision of an original and a recurrent symptomatic CCM lesion adjacent to the same pre-existing DVA. Specifically, in the year 2000, at the age of 3, the patient underwent neurosurgery following the diagnosis of intracerebral bleeding caused by a hemorrhagic CCM lesion located in the right frontal lobe of the brain and associated with voluminous DVA draining in the superior sagittal sinus (SSS) . In accoThree years later, in June 2018, the patient experienced an acute onset of critical episodes of paresthesia in the left lower limb, with ascending progression to the left arm and face. A brain CT scan was, therefore, performed, which revealed a right frontal parasagittal hemorrhagic lesion. Diagnostic completion with MRI showed the presence of a vascular lesion, highly compatible with CCM imaging features, located within the hemorrhagic focus and close to the pre-existing known DVA A.Neurosurgery was then performed through the same approach used for the first intervention. Briefly, in both the first and the second resections, a right parasagittal craniotomy was performed, without exposing the superior sagittal sinus for safety reason. After dural opening, en bloc resection was achieved in both cases.During the second neurosurgical intervention, a 1 cm large mulberry-like CCM lesion was detected along the medial hematoma wall next to the falx. The mass was surgically removed en bloc and sent for histological examination, which confirmed the diagnosis of a newly formed CCM lesion. The DVA was preserved intact for the same reasons as the previous surgical intervention. Postoperative MRI examination demonstrated complete removal of the CCM lesion B. The paThis case report demonstrated the recurrence of a sporadic cerebral cavernous malformation (CCM) in the vicinity of a voluminous developmental venous anomaly (DVA) 18 years after neurosurgical excision of the first CCM lesion, and it provided preliminary neuroimaging data suggesting that such recurrence is secondary to focal oxy-inflammatory conditions generated by the pre-existing DVA. Further circumstantial evidence that supports this possibility is provided and discussed in detail below.CCM1/2/3), known to be responsible for at least 80% of fCCM cases [PIK3CA and MAP3K3, thereby suggesting a distinct genetic origin [CCMs are abnormally dilated and angiographically occult capillary sinusoids, whose natural history investigation was effectively possible only after the advent of magnetic resonance imaging (MRI), which eventually became the optimal standard tool for their clinical diagnosis and follow-up, as well as for a better knowledge of their peculiar clinical and epidemiological features ,57,58. ICM cases , are rarc origin ,11,12. Ac origin . Taken tAmong the potential pathogenic mechanisms that may underlie the development of both sCCM and fCCM lesions and modulate the severity of major associated symptoms, such as focal intracerebral hemorrhage (ICH) and neurological deficits, there is an increased sensitivity of key NVU components, including endothelial cells and astrocytes, to focal acute or chronic oxidative stress and inflammatory (oxy-inflammatory) conditions ,45,50. SDVAs are congenital abnormalities of cerebral venous drainage, consisting of multiple small medullary veins that join to drain into a larger collector vein, which then drains superficially into a dural sinus, or less often into the deep venous system. Additionally known as cerebral venous angiomas or cerebral venous malformations, DVAs are the most common vascular malformations that occur in the CNS, and they are easily identified by contrast-enhanced brain MRI or susceptibility weighted imaging (SWI) sequences, assuming a characteristic “caput medusae” appearance . Normal Accumulated evidence unequivocally indicates that structural and hemodynamic conditions within the DVA are causative factors for the de novo formation of sCCM lesions ,15,37,94Overall, on the basis of the compelling evidence described above, it is possible to propose that sterile inflammatory responses induced in the brain parenchyma by changes in angioarchitectural and hemodynamic features of a pre-existing DVA may represent a novel plausible etiological mechanism for the long-term distant recurrence of a DVA-related hemorrhagic sCCM lesion identified in our study. Indeed, in addition to correlating with the age-dependent increase in the prevalence of sCCMs associated with DVAs , such a While specific molecular and pathological analyses are required to confirm the occurrence of DVA-associated sterile inflammatory responses, our observational study provides proof of concept of the presence of neuroimaging signal-intensity abnormalities attributable to neuroinflammatory events, including point-like contrast enhancements and hyperintense gliotic areas in the brain parenchyma surrounding a distant recurrent sCCM lesion associated to a pre-existing large DVA. Accordingly, despite several limitations, our case report paves the way for future comprehensive investigations in a large number of DVA-associated sCCM cases through advanced imaging approaches more suitable for studying brain inflammation, including advanced PET guided neuroinflammation imaging with specific tracers ,97,98,99


Esophageal squamous papilloma (ESP) is a rare benign tumor. ESP is generally detected incidentally during esophagogastroduodenoscopy, which is usually performed to investigate dyspepsia. We present three cases of this rare endoscopic finding. While two of our patients were asymptomatic, one had dysphagia, and the lesions were excised. Endoscopists should be able to make the differential diagnosis of papilloma detected in the esophagus and have knowledge about these lesions as they may carry malignant potential. Excision of papillomas is recommended for definitive diagnosis and treatment. Esophageal squamous papilloma (ESP) is an uncommon epithelial tumor, and it is caught incidentally during an upper endoscopy in most cases. According to the endoscopic series, the prevalence of ESP is between 0.01% and 0.45% and is more common in middle-aged males. Large lesions may present with dysphagia, but ESP is usually asymptomatic. ESP is mainly detected in the middle to lower esophagus. The characteristic endoscopic figure of ESP is a single small, whitish, wart-like lesion grown from the mucosa. However, histopathological examination is needed for a definitive diagnosis [The pathogenesis of ESP has not been completely clarified. Gastroesophageal reflux disease (GERD), esophagitis, mucosal irritation, and human papillomavirus (HPV) infection are identified as risk factors. HPV positivity has been reported between 0% and 87.5% in ESPs. Though ESPs are thought to be benign, some publications also report malignant potential -4. We reCase 1A 27-year-old female was admitted to the otolaryngology clinic with a history of globus sensation lasting for six months. The patient had a history of allergic rhinitis and was using nasal steroids, and there was no physical examination finding to explain her symptoms. The patient was referred to general surgery for upper gastrointestinal endoscopy. She had an intermittent feeling of difficulty swallowing and nausea. The patient underwent an esophagogastroduodenoscopy and detected a nonulcerated, polypoid, approximately 4 mm in size mucosal lesion in the middle thoracic esophagus. The lesion was excised by biopsy forceps. Pathology reported squamous esophageal mucosa with papillary proliferation, consistent with benign squamous cell papilloma. After excision, the patient's symptoms resolved.Case 2Helicobacter pylori positivity. He was treated with antibiotics and an acid-reducing proton pump inhibitor.A 36-year-old male patient was admitted to our outpatient clinic with complaints of dyspepsia. He had a history of essential hypertension and irregular antacid use. Esophagogastroduodenoscopy (EGD) showed small papillomas in the upper and middle thoracic esophagus Figure  and antrCase 3A 48-year-old female patient with hypothyroidism and a history of peptic ulcer treatment six months ago was evaluated by endoscopy for upper abdominal pain and burning sensation in the stomach. EGD showed a papilloma in the middle thoracic esophagus Figure , Panel AESP is a rare benign tumor. ESPs are usually asymptomatic but may present with multiple clinical manifestations such as dysphagia, anemia, and hematemesis. It is generally detected incidentally in EGD, which is usually performed to investigate dyspepsia ,6. AlthoESPs are usually seen as solitary lesions smaller than 5 mm, but they can be large and multiple ,8. Our cEPS etiology is not fully determined. Gastroesophageal reflux disease, mucosal damage, esophagitis, HPV infection, alcohol, and tobacco are risk factors for ESP ,6,8. TheAlthough ESPs are primarily benign tumors, some publications reveal their relationship with squamous cell carcinoma (SCC). The malignant potential is high in patients with multiple lesions (papillomatosis) or a huge lesion. Publications state that it is challenging to identify malignant areas within ESP or papillomatosis in routine endoscopy and biopsies, especially in papillomatosis of the esophagus. There is no consensus on endoscopic surveillance. The standard recommendation for an evaluation of ESP is a biopsy. Several suggested endoscopic treatments for squamous papillomas include forceps biopsy resection, cautery, photodynamic therapies, radiofrequency ablation, and mucosectomy for larger lesions. Esophagectomy can be performed in patients unsuitable for endoscopic follow-up and treatment  ,2,6,8.ESP is a rare benign tumor. ESPs are usually asymptomatic but may present with dysphagia. Endoscopists should be able to make the differential diagnosis of papilloma detected in the esophagus and have knowledge about these lesions as they may carry malignant potential. Excision of papillomas is recommended for definitive diagnosis and treatment.


Intraoral osseous choristoma represents a benign lesion of growing ectopic bone in the soft tissues of the oral cavity. It is considered as rare entity while fewer than 100 cases have ever been reported worldwide. Nevertheless, the pathogenetic mechanism that drives this abnormal ossification still remains controversial. In the present study a case of lingual osseous choristoma in a 50-year old male is presented. The patient was treated with surgical excision and no recurrence was observed. In addition epidemiology, clinical presentation, and pathogenesis are reviewed, serving as a reminder of this rare pathology. Key words:Osseous choristoma, osteoma, tongue. Thoseristoma” . Their eristoma” .We present a case of a lingual osseous choristoma and review epidemiology, clinical presentation, and pathogenesis of oral osseous choristomas.A 50 year-old man presented for evaluation of a mass on the tongue that he noticed more than a year ago, while brushing his teeth. He reported slow enlargement during the last 4 months, causing a “lump in the throat” sensation, without dysphagia, odynophagia, or bleeding. His medical history was noncontributory; he smoked 20 cigarettes per day. No history of intraoral trauma was elicited.Clinical examination showed a pedunculated, dome-shaped tumor on the distal dorsal tongue, located and close to the midline. It had pale color, smooth surface, hard consistency and measured approximately 8 mm Fig. . The resMicroscopic examination of 5μm thick sections showed that the tumor was composed of dense, lamellar bone, with small and irregular marrow spaces containing loose fibrous connective tissue. It was covered by normal-appearing stratified squamous epithelium Fig. . The diaHealing was uneventful and there is no evidence of recurrence 12 months after excision.The case presented herein had a microscopic appearance diagnostic of an oral osseous choristoma. The tongue is the most common location, with 74 cases including the present one reported so far, followed by the buccal mucosa with 15 cases ,7; one cLingual osseous choristoma shows a strong female predilection, with the female/male ratio being approximately 2.75:1 . The patClinical differential diagnosis of oral osseous choristomas includes mostly benign tumors or tumor-like lesions i.e., fibroma, salivary gland tumors, lipoma, neural tumors, as well as soft tissue cysts ,9,12. DeConsidering pathogenesis, developmental and reactive origin have been proposed. Tongue is formed by the fusion of its posterior third, deriving from the third branchial arch, with the anterior two-thirds, deriving form the first branchial arch. Those branchial arches give rise to several normal osseous structures such as incus, malleus, styloid process, and the hyoid bone, through endochondral ossification ,5. AccorSurgical excision of intraoral osseous choristomas remains the standard treatment, with laser/CO2 excision suggested as an alternative procedure ,14, but


Azygos vein aneurysms (AVAs) are extremely rare. The majority of patients have no obvious clinical symptoms, so they are found by physical examination or by chance. There is limited clinical treatment experience that can be referred to, and there are no clear guidelines or research evidence standardizing the surgical and interventional therapy. Here, we report a patient with idiopathic AVA whose three-dimensional reconstruction of the tumor was completed before surgery. On the basis of three-dimensional reconstruction, single-port thoracoscopic resection of the AVA was successfully completed and reported for the first time. The previously reported cases are summarized to provide guidance for the diagnosis and treatment of patients with AVAs.A 56-year-old man was transferred to our hospital due to “dysphagia”. The diagnosis of AVA was made after enhanced computed tomography, gastroscopy, fiberoptic bronchoscopy, and three-dimensional reconstruction. Congenital weakness or degenerative changes causes the vein walls to be extremely thin that the AVA had the risk of ruptur. Furthermore, the patient had symptoms of dysphagia, he received single-port thoracoscopic surgery. After the operation, his dysphagia disappeared. The postoperative pathology confirmed hemangioma. The patient was discharged 3 days after surgery without any complications.AVAs are rare. Preoperative three-dimensional reconstruction can greatly help surgeons clarify the disease diagnosis, formulate the surgical plan, avoid damage to the surrounding vital organs, and reduce intraoperative bleeding. Thoracoscopic surgery to remove AVAs is difficult and has a high risk of bleeding, while more minimally invasive single-port thoracoscopic surgery is also safe and effective for the treatment of AVAs. Azygos vein aneurysms (AVAs) are rare . In radiA 56-year-old man complained of dysphagia. He was referred to our hospital without special treatment because of a mediastinal mass on chest computed tomography (CT). The patient had no relevant medical history. He denied any falls or other trauma. Body temperature was 37.0 °C, heart rate was 78 beats/min, blood pressure was 127/74 mmHg, and respiratory rate was 20 breaths/min. Breath sounds were normal. No moist or dry rales. No heart murmurs or muffled heart tones. Contrast-enhanced CT revealed a mediastinal mass (4.2 × 3.7 × 2.6 cm) located at the tracheal bifurcation. It has smooth borders with no obvious nodules. No other aneurysmal malformations were detected. The esophagus was obviously compressed on the corresponding plane. At the same time, contrast-enhanced CT suggests unremarkable enhancement in the arterial phase. It showed delayed enhancement with a CT value of approximately 118 HU in the venous phase  to respect the AVA under double-lumen tracheal intubation 3 days after hospitalization. The patient was in the left lateral decubitus position during surgery. The intraoperative exploration results were consistent with the three-dimensional reconstruction. The AVA was located in the azygos vein arch, and there was no abnormal pulmonary artery or pulmonary vein connected with it Fig. A. The AVAVA is a rare disease. AVA is generally defined as a diameter of the AV exceeding 2.5 times the normal diameter. According to its morphology, it is currently divided into fusiform AVAs, with the overall expansion of the AV, and saccular AVAs, with local expansion of the azygos wall . AccordiCurrently, there are no guidelines on the optimal treatment of AVAs. In a previous report, conservative observation combined with oral anticoagulation was successful in asymptomatic AVA patients. Ko et al.  reportedAt the same time, interventional therapy is a relatively new option for AVA. There are case reports of successful implantation of covered stents within the aneurysm , 25, 35,In conclusion, some patients may have symptoms such as dysphagia and chest tightness due to tumor compression of the esophagus and trachea. Preoperative three-dimensional reconstruction greatly helps surgeons clarify the disease diagnoses, formulate surgical plans, avoid damage to the surrounding vital organs, and reduce intraoperative bleed. We proved that minimally invasive single-port thoracoscopic surgery is also safe and effective for the treatment of AVA.


Retrocaval ureter is a rare, congenital anomaly of the inferior vena cava. Due to an abnormal process in embryogenesis, the ureter descends posterior to the inferior vena cava. Although the anomaly can be found in pediatric patients, it most commonly manifests symptoms between the third and fourth decade of life that are typically due to ureteric obstruction, such as hydronephrosis. Retrocaval ureter can be asymptomatic and may be the reason for unreported cases and low incidence in the world. In this case, we reviewed the record of a patient with the diagnosis of retrocaval ureter incidentally found during admission for a lower gastrointestinal bleed. Circumcaval ureter, more commonly known as retrocaval ureter, is a rare congenital anomaly with fewer than 200 cases being reported since first described in 1893 [In this case report, we present a patient who arrived at the emergency department (ED) for dark stools for two days, and a retrocaval ureter was seen on computed tomography (CT) of the abdomen and pelvis as an incidental finding. Although the patient had mild hydronephrosis secondary to his retrocaval ureter, he remained asymptomatic.History of presentationA 55-year-old African American male with a history of hypertension and chronic alcohol use disorder complained of painless, burgundy-colored bowel movements for three days associated with chronic, intermittent lower back pain. He denied difficulty initiating urination, hesitancy, or urgency despite his enlarged prostate. He denied any medication use besides occasional aspirin. He had an extensive alcohol intake history. On physical examination, the patient was hypertensive (171/103), afebrile, had a regular heart rate, and was in no acute distress. His abdomen was soft, nontender, and nondistended with normal bowel sounds.InvestigationsThe patient’s complete blood count (CBC), chemistry panel (CMP), and liver function tests were relatively unremarkable, but he was found to have a positive fecal occult blood test and microscopic hematuria. His ethanol level was less than 10 mg/dL. His electrocardiogram on arrival revealed normal sinus rhythm with a heart rate of 73 beats/minute. Management in the ED included 1 L bolus of normal saline gastrointestinal prophylaxis and a CT of the abdomen and pelvis with intravenous (IV) contrast.CT of the abdomen and pelvis with IV contrast revealed moderate proximal right hydroureteronephrosis likely as a result of compression of the proximal right ureter from the IVC, more commonly known as a retrocaval ureter. There was also a nonobstructive 8 mm calculus in the inferior pole of the right kidney Figure  and a noManagementThe patient was primarily admitted to the hospital for managing his lower gastrointestinal bleed, but further intervention was required due to the incidental findings of a retrocaval ureter anomaly causing hydroureteronephrosis. The repeated CBC and CMP over the next few days remained unremarkable. A colonoscopy revealed internal hemorrhoids and severe diverticulosis with active bleeding of the diverticulum, which was controlled by applying two hemostatic clips. Per urology, definitive reconstructive surgery and stone removal can be performed simultaneously by a reconstructive urologist in the outpatient setting. He was discharged on tamsulosin to manage his new diagnosis of retrocaval ureter.There are two classifications of retrocaval ureters, namely, type one and type two. In type one, the ureter travels posterior to the IVC at the level of the third lumbar vertebra and presents as an S- or a fish-hook-shaped deformity presenting with subsequent moderate-to-severe hydronephrosis. Type two is less common, and it is depicted by the proximal ureter traveling posterior to the IVC at the same level of the renal pelvis forming a smooth, sickle-shaped curve -5. HydroThe clinical presentation of a symptomatic patient is most commonly described as obstructive uropathy symptoms, such as right flank pain, recurrent urinary tract infections, nephrolithiasis, and hematuria . On a reThere are multiple modalities in which imaging can be used to diagnose a retrocaval ureter. The method of choice for diagnosing retrocaval ureter is CT urography because it is the most efficacious and least invasive . It provAsymptomatic patients with mild-to-moderate hydronephrosis can be managed without surgical intervention; however, symptomatic patients with severe hydronephrosis and with worsening kidney function require surgical treatment . There aRetroperitoneal laparoscopic ureteroplasty in symptomatic patients with retrocaval ureter is recommended as the first choice in corrective surgery due to its safety and effectiveness, faster recovery, decrease in blood loss, shortened hospital visits, and decreased postoperative pain . A case This case highlights a unique presentation of clinically asymptomatic, moderate hydronephrosis due to retrocaval ureter incidentally found on CT imaging. Preoperative diagnosis is best established through a CT scan. The standard of care to correct this anomaly is minimally invasive laparoscopic surgery; however, asymptomatic patients should be managed conservatively. Retrocaval ureter is a rare anomaly, but the number of cases reported is slightly increasing. The cause for this is most likely multifactorial, which could include improvement in radiographic imaging, increased access to healthcare, or simply an increased number of reports by physicians. Nevertheless, it is important to consider retrocaval ureter in a patient with asymptomatic hydronephrosis, despite a low index of suspicion for this disease.


Intestinal arteriovenous malformation is an abnormal connection between arteries and veins that bypasses the capillary system and may be a cause of significant lower gastrointestinal bleeding. On endoscopy, arteriovenous malformations are usually flat or elevated, bright red lesions. Overall, rectal localization of arteriovenous malformations is rare. The same may be said about polypoid shape arteriovenous malformations. Herein, we present a case of a large rectal polypoid arteriovenous malformations.Clinical, diagnostic, and treatment modalities of the patient were reviewed. Pre- and post-operative parameters were collected and analyzed. The clinical English literature is also reviewed and discussedA 60-year-old female patient was admitted to our emergency department for rectorrhagia and anemia. Rectoscopy revealed a polypoid lesion in the rectum and the biopsy showed fibrosis, necrosis areas, and hyperplastic glands. A total body contrast-enhanced computed tomography (CT) was performed revealing a parietal pseudonodular thickening with concentric growth and contrast enhancement, extending for about 53 mm. The mass wasn't removed endoscopically due to concentric growth, sessile implant, and submucosal nature. The patient underwent an uneventful laparoscopic anterior rectal resection. The postoperative hospitalization was free of complications. Histology showed the presence of a polypoid AVM composed of dilated arteries, veins, capillaries, and lymphatics, engaging the submucosa, muscularis, and subserosa layer.After a review of the current English literature, we found only one case of rectal polypoid AVM. The scarcity of documented cases encumbers optimal diagnostic and treatment approaches. Intestinal Arteriovenous malformation (AVM) is a cause of lower gastrointestinal bleeding alongside diverticula, colon neoplasia, and internal hemorrhoids . AVM is A sixty-year-old female patient was admitted to our emergency department for rectorrhagia and anemia. Her medical history was significant for chronic atrial fibrillation treated with rivaroxaban and propafenone. On arrival, she was hemodynamically stable, and the level of hemoglobin was 9.6 g/dl. Other laboratory tests were not remarkable. Digital rectal exploration was performed showing circumferential hemorrhoids without bleeding. For further investigation, rectoscopy was accomplished and revealed an intraluminal, submucosal, polypoid lesion in the rectum. The mass was biopsied, and the histology showed fibrosis, necrosis areas, and hyperplastic glands. The patient was admitted to General Surgery Department to complete the clinical assessment.A total body contrast-enhanced computed tomography (CT) was performed revealing a parietal pseudonodular thickening with concentric growth and contrast enhancement, extending for about 53 mm . RoundedGastrointestinal tract AVMs are a rare disease and are reported to account for 0.8%–3% of all cases of intestinal bleeding . VariousAVMs are extremely rare in people under the age of 50 . The patMoore et al. reviewed and classified intestinal AVMs based on angiographic characteristics, localization, patient age, and family history (Moore et al. classification, because of distinct clinical and endoscopic characteristics and different management strategies ( history . The aut history , 11. Somrategies . In factDiagnosis of intestinal AVMs is based on colonoscopy, contrast-enhanced CT, and MRI. In cases of active bleeding, accurate localization of the source of gastrointestinal hemorrhage is an important factor in determining appropriate management. Polypoid rectal AVM, due to its submucosal localization and rarity, presents a diagnostical challenge. This, as in our case, may lead to misdiagnosis and subsequent suboptimal treatment or intra- and post-operative complications.Various AVM treatment possibilities were suggested. Generally speaking, AVMs can be treated with endoscopic hemostasis and resection, intravascular embolization, and surgery , 12. MinOptimal, evidence-based management of AVMs is challenging due to the paucity of literature on the subject, even more so, the management of rectal polypoid AVMs, of which only one case is reported to date. Small polypoid AVM lesions can be safely treated endoscopically. Surgical resection may be indicated for larger AVMs. Due to the scarcity of documented cases, no strong recommendations may be given on the treatment modality. Reporting other cases may aid in the guidance of therapeutic approach.


Hemangiopericytomas (HPCs) invading the cavernous sinus (CS) are extremely rare invasive tumors that have a great propensity for local recurrence. To date, only eight cases have been reported in the literature. Owing to the abundant vascular supply of HPCs, intracavernous bleeding and important blood vessels and nerves passing through the CS, it is very difficult and challenging for neurosurgeons to completely resect HPCs. Here, we report two cases of HPCs invading the CS and introduce their clinical manifestations, imaging findings, surgical approaches and histopathological features in detail. We have implemented the surgery by the endoscopic transpterygoid transcavernous approach (ETPTCa) for the two patients, and one patient has undergone gross total resection (GTR) and another has undergone subtotal resection (STR) and postoperative stereotactic radiosurgery (SRS). The ETPTCa may serve as a viable option to facilitate HPCs resection. Radiotherapy is helpful in prolonging progression-free survival (PFS) following STR of the tumor. Intracranial HPCs are rare mesenchymal tumors that originate from pericytes around the capillaries and constitute less than 1% of intracranial tumors , while iA 63-year-old female complaining of ptosis of the right eye was admitted. Positive neurological signs included partial inferior visual defects and limited inward, upward and downward ocular movement. A preoperative cranial CT scan showed a slightly hyperintense lesion in the right CS . Cranialvia an endoscopic endonasal transpterygoid approach to resect the intracavernous lesion in a four-handed and binostril manner under general anesthesia  showed that the tumor cells of HPCs with round or egg-shaped nuclei and inconspicuous nucleoli had consistent sizes, and the boundary between the tumor cells was unclear . The mitA 23-year-old female complaining of intermittent headache and numbness of the left face was admitted. Physical examination showed that the left masseter muscle was slightly atrophied. Preoperative head CT revealed a slightly hyperintense lesion in the left CS and middle cranial fossa . Cranialvia the left pterional interfascial approach, made an arc incision with a length of 20 cm, cut the skin, subcutaneous tissue and galea aponeurotica layer by layer, freed the temporal muscle and removed the bone flap. The dura tension was high, and the temporal encephalocele was apparent, so we carefully released cerebrospinal fluid from the sylvian fissure to create a surgical corridor. After lifting the temporal lobe, the tumor was found in the middle cranial fossa base, which was grayish white, hard, and tightly attached to the trigeminal ganglion; the tumor was resected piece by piece. After subtotal tumor resection, complete hemostasis was achieved, and the dura defect was repaired with artificial meninges. Intraoperative blood loss was 6,000 ml, and twelve red blood cell units, 1,600 ml fresh plasma and twenty cryoprecipitate units were infused into the patient. The patients' headache disappeared, and numbness of the left face was not relieved. The patient showed no new symptoms of neurological deficits after the operation and underwent SRS forty days after the operation.We performed the surgery by an endoscopic endonasal transpterygoid combined with a pterional approach to resect a lesion invading the cavernous and middle cranial fossa. When the optical canal, lateral opticocarotid recess, vidian nerve, foramen lacerum, pterygopalatine fossa contents, anterior wall of the CS and dura mater of the middle cranial fossa were fully exposed based on the procedures of case 1, the dura mater of the middle cranial fossa and the capsule of the tumor were sectioned. The tumor was solid and hard and showed abundant blood supply and obvious bleeding, which invaded into the CS and adhered firmly to the ICA and trigeminal nerve. The intracavernous tumor was subtotally resected because of severe bleeding of the CS and the tumor. After successful hemostasis, the dural defect was repaired with artificial meninges, and the skull base was covered by a nasal septum-base mucosal flap. Then, we performed the craniotomy Histopathological examination (H&E) showed that HPCs were composed of closely packed cells with thin-wall fissure-like vessels, and staghorn-like vessel lacunes were observed; the nuclei were ovoid or long-spindle-shaped with inconspicuous nucleoli . The mitHPCs of the central nervous system are rare malignancies that have a high propensity for local recurrence and distant metastasis –9. They A definite preoperative diagnosis of HPCs is important for surgeons to select the optimal surgical method to carry out the operation. Nevertheless, it is difficult to distinguish HPCs from meningiomas, especially an angiomatous meningioma, because of their similar radiological features. The HPCs are generally irregular or lobulated with obvious intratumoural flow voids and no calcification and may be related to osteolytic destruction of adjacent bone based on MRI. Cystic changes and necrosis are common in HPCs. In the present two cases, the tumors were all irregular on MRI; the tumor of case 2 showed obvious intratumoral flow voids, cystic changes and necrosis. Some studies have shown that susceptibility-weighted imaging (SWI) and diffusion-weighted imaging (DWI) are helpful for the differential diagnosis of HPC and AM; HPCs have higher normalized apparent diffusion coefficients (nADC) and degrees of intratumoral susceptibility signal intensity (ITSS) than meningiomas , 21. FurGTR of intracranial HPCs is difficult and pose challenges for neurosurgeons because of severe intraoperative bleeding. Many case reports show that for large or giant HPCs, preoperative embolization can remarkably reduce blood loss during surgery and achieve GTR of these tumors , 22, 23.via ETPTCa combined with the pterional approach. The GTR of HPCs is highly important for patients to improve OS and PFS, which is supported by many studies  can be observed at 360° under endoscopy. In previous literature reports, seven of the eight patients with intracavernous HPCs underwent craniotomy, and one underwent an endoscopic endonasal approach; GTR was achieved in 3 patients, STR in 3, near subtotal resection in 1 and partial resection in 1. In our two cases, GTR was achieved in one case by ETPTCa, and STR was achieved in one case  studies , 24–26.  studies . Fritchi studies .Of the 8 cases, 7 were confirmed as HPCs (WHO grade II) by histopathology and immunohistochemistry. There was only one case diagnosed as anaplastic HPCs (WHO grade III) in which an 11-year-old male complaining of headache and diplopia died due to septicemia two days after craniotomy. The diagnostic criteria of anaplastic HPCs included 5 or more mitoses per 10 high power fields, necrosis and at least 2 of the following features: hemorrhage, high cellularity and moderate to high nuclear atypia .For giant HPCs invading the CS and middle and posterior cranial fossa, even though combined approaches were used for tumor excision, it is still difficult to achieve complete tumor resection. Adjuvant radiation therapy (RT) to prevent tumor recurrence is beneficial for patients with residual tumors. Some studies indicated that adjuvant RT could significantly prolong PFS after the initial operation , 26, 30 HPCs invading the CS are extremely rare and there have been eight cases reported in the literature thus far. Surgical resection was the most effective and optimal way to treat HPCs, and ETPTCa was feasible and more promising for tumors in the sellar and parasellar regions. RT or GKS significantly improved PFS after STR of the tumor.


Gastrointestinal stromal tumors are rare gastrointestinal tract growths associated with high rates of malignant transformation. Most cases are asymptomatic and can be identified by computed tomography scan. We present the case of a 50-year-old male with melena and fatigue. Endoscopy showed an ulcerated submucosal tumor diagnosed as a gastrointestinal stromal tumor after surgical resection; it did not present with metastasis and was successfully treated surgically without relapse. Gastrointestinal stromal tumors (GISTs) are rare gastrointestinal tract growths originating from the interstitial cells of Cajal. Most cases are asymptomatic and identified incidentally by endoscopy or computed tomography (CT) scan. They have great potential for malignant transformation and account for 1%-2% of gastrointestinal neoplasms; unfortunately, chemotherapy and radiation are ineffective, so surgical resection has always been the mainstream treatment. As the knowledge regarding mutations associated with these tumors increases, therapy has evolved. The selective tyrosine kinase receptors inhibitor (TKI), imatinib mesylate, is used as an adjuvant or neoadjuvant therapy that improves the risk of local recurrence and metastasis. However, due to tumor resistance, sunitinib and regorafenib are effective second-line TKIs [Our patient is a 50-year-old man who presented to the outpatient clinic complaining of a one-month history of black stools, nausea, fatigue, and dyspnea on exertion. His past medical history was remarkable for hemorrhoids, and he had never had a colonoscopy or upper endoscopy. Upon interrogation, he denied abdominal pain. His family history was negative for stomach or colon cancer. He had no record of smoking, alcohol, or drug abuse.On physical examination, the patient had a severely pale appearance; the abdomen was soft and non-tender, and bowel sounds were normal. Rectal examination revealed an empty ampulla with fecal residues. His initial hemoglobin was 5.2 g/dL, and he received four doses of intravenous iron supplementation. Repeat blood work showed hemoglobin levels of 4.5 g/dL, which prompted the medical team to perform an endoscopy that showed an ulcerated submucosal tumor (IIb Forrest). A CT scan of the abdomen reported stomach-thickened walls of approximately 33 mm at the antrum level and a large pylorus Figures , 2. The After the gastric tumor was laparoscopically resected and sent for histopathological evaluation, pathology determined a GIST measuring 5 cm x 10 cm, with a mitotic index of 5 mitosis/high power fields (HPF). Of note, the patient did not receive imatinib, and his six-month follow-up CT scan of the abdomen showed no signs of a remaining mass or metastasis.Our patient with a history of hemorrhoids presented with melena, fatigue, nausea, dyspnea on exertion, and hemoglobin of 5.2 g/dL. Even though rare, bleeding internal hemorrhoids can lead to iron deficiency anemia and necessitate transfusion. The patient received four doses of intravenous iron supplementation. The repeated hemoglobin levels were 4.5 g/dL, and a subsequent endoscopy showed an ulcerated submucosal tumor (IIb Forrest) as the probable cause of his anemia.Submucosal lesions are growths in the gastrointestinal tract covered with normal mucosa and may also be ulcerated or inflamed. Gastric submucosal tumors are usually incidentally found during routine health examinations without any symptoms, but sometimes, abdominal pain, palpable abdominal mass, and weight loss may present. The most prevalent are GIST, leiomyoma, neuroendocrine tumors, lipomas, granular cell tumors, heterotopic pancreas, Brunner's gland hamartoma, and lymphangiomas. By the site of origin, leiomyomas most frequently appear in the esophagus, lipomas in the right colon, and granular cell tumors in the lower and middle esophagus. Leiomyomas are pale, firm, rubbery masses characterized by a bland spindle cell population arranged in fascicles and whorls, with few mitoses, and immunohistochemistry (IHC) that is negative for CD34 and CD117. Schwannomas are benign gray masses characterized by spindle cells, lymphocytes, and a nodular lymphoid cuff, and they are positive for S100 and vimentin on IHC.Granular cell tumors are benign neurogenic tumors that appear as sheets of uniform histiocyte-like cells with abundant, eosinophilic, periodic acid-Schiff-reaction-positive cytoplasm containing lysosomal granules and small vesicular nuclei, and they are positive for S100 and neuron-specific enolase -5.Our patient's final diagnosis was GISTs, which are neoplasms that are found along the gastrointestinal tract and located in the stomach more than 50% of the time. These tumors can go from a few millimeters to more than 20 cm in diameter, with a typical average of 5-8 cm; they are gray and well circumscribed but not encapsulated and may show signs of hemorrhage, calcification, ulceration, necrosis ,3,6,7.GISTs originate from the known peacemaker's gastrointestinal tract cells, the Cajal cells, which express two protein tyrosine kinase receptors in the muscularis propria: KIT and PDGFRA. A mutation in the c-kit gene causes the activation and gain of function of the enzymatic activity of the KIT type II transmembrane tyrosine kinase receptor affecting the intracellular signal transductions, which regulates proliferation, differentiation, and anti-apoptotic signaling. This causes a ligand-independent tyrosine kinase activity, resulting in uncontrolled cell proliferation and altered cell growth. The mutation in the PDGFRA receptor causes similar effects. Most GISTs are sporadic (97%), while the remaining percentage includes familial GIST syndromes such as neurofibromatosis and Carney-Stratakis syndrome .GISTs represent about 1% of all primary malignant tumors and are the most common mesenchymal neoplasms of the gastrointestinal tract. The worldwide annual incidence in 2021 is approximately seven to 15 cases per million people, with 600 new patients diagnosed annually in the United States. The median age of the diagnosis is between 50 and 70 years old, with no predilection for either gender or known predisposing risk factors .GISTs are usually asymptomatic until they reach a size of about 6 cm. These large tumors are often vascular and can present with pain or GI bleeding toward the gastrointestinal lumen or abdominal cavity. Less common presentations include nausea, pleuritic chest pain, pelvic pain, and small bowel obstruction. Approximately 20% of patients have metastasis when they are diagnosed. The most common sites of metastasis are the liver, abdominal cavity, and lymph nodes .An echoendoscopy-guided biopsy is the technique of choice for pathological diagnosis, with CT-guided percutaneous biopsy as the second choice. Three histology types, namely fusiform, epithelioid, and mixed morphology, are described. CD117 protein is the most specific marker for GISTs on IHC .The treatment of choice for localized GISTs is complete surgical resection with negative margins. GISTs rarely involve the regional lymph nodes, so extensive lymph node exploration or wide lymphadenectomy is rarely indicated. Small tumors/gastric GISTs (<5 cm) are managed laparoscopically, and minimally invasive techniques are submucosal tunneling endoscopic resection, endoscopic full-thickness resection, and laparoscopic, cooperative endoscopic surgery ,3,11-13.Tumor size less than 5 cm in diameter, a complete surgical resection without tumor rupture, and low histologic grade of tumor are significant favorable prognostic factors. The recurrence rate following resection is 35% on average, and it has been reported that only 10% of these patients are disease-free on long-term follow-ups .Seven cases of GIST presenting with moderate and mild anemia have been reported -22 in pePatients with GIST can present with progressive anemia. Some concomitant diseases like hemorrhoids can be attributed as the cause of anemia, and GIST or other tumors can be missed. Other gastrointestinal disorders can be ruled out with the help of upper and lower endoscopies and a CT scan of the abdomen and pelvis. Patients with an ulcerated subepithelial mass, such as a GIST, causing bleeding should undergo surgical resection of the tumor to prevent rebleeding. Most GIST patients benefit from targeted molecular therapies. Even though they can be successfully treated with surgery, follow-up is mandatory due to the risk of recurrence.


This study reports a case of hepatoblastoma with onset at 30-weeks' gestation and rapid growth rate. The postnatal enhanced CT confirmed an intrahepatic mass with a size of 8.5 cm × 6.6 cm and a clear boundary accompanied by uneven enhancement, displacement, and narrow lumen of the hepatic vein due to compression. The alpha-fetoprotein (AFP) at birth was 1,002,632 ng/ml . A diagnosis of congenital hepatoblastoma was established based on the imaging and laboratory outcomes. The infant received chemotherapy of Cisplatin-5 fluorouracil-Vincristine (C5V) on the fourth day after birth. After four courses of C5V, a complete tumor resection was performed, and the postoperative pathology was consistent with mixed epithelial and mesenchymal hepatoblastoma. Four more courses of C5V and one course of C5VD (C5V plus doxorubicin) followed the surgery. Infectious diarrhea and acute kidney injury (stage I) occurred during chemotherapy, which recovered after anti-infection and symptomatic treatment. The patient is currently 2 years old and still in complete remission. In this case, the onset of hepatoblastoma was early, and the tumor grew rapidly, resulting in an obvious compression effect. Chemotherapy was started early after birth, and the curative effect was satisfactory, suggesting that the hepatoblastoma based on clinical diagnosis with rapid tumor progression and severe dysfunction of surrounding organs caused by compression should undergo chemotherapy as soon as possible if a pathological diagnosis cannot be obtained temporarily, which also plays an important role in improving the complete resection rate of intraoperative tumor and reducing the recurrence rate of postoperative tumor. Hepatoblastoma is the most common primary malignant liver tumor among children and is an embryo-derived tumor. Among children up to 5 years old, the incidence of hepatoblastoma is 1.2–1.5 per million children per year , 4. PreoA 32-year-old primiparous pregnant woman's routine prenatal ultrasound at 30-weeks' gestation revealed a 1.8 cm × 1.8 cm × 1.5 cm medium-to-high echoic mass with a clear boundary surrounded by a low echoic dark halo in the fetus' parenchyma of the right lobe of the liver. Inside the mass, a few strips of blood flow signals could be detected . A fetalA female infant was delivered by cesarean section at 39 + 3-weeks' gestation with a birth weight of 3,040 g and Apgar scores of 10 for the 1st and 5th min. The infant suffered from poor appetite, frequent vomiting, yellow skin, and abdominal distension. On physical examination, the liver could be palpated 10 cm below the ribs; firm consistency, blunt edges, and a smooth surface were observed. Laboratory tests showed alanine aminotransferase (ALT) 380 IU/L, aspartate aminotransferase (AST) 315I U/L, total bilirubin (TBIL) 251 umol/L, direct bilirubin (DBIL) 165 umol/L, and fibrinogen (FIB) 1.2g/L; alpha-fetoprotein (AFP) was significantly increased to 1,002,632 ng/ml . An abdominal ultrasound on day 1 showed a mass of about 8.1 cm × 7.2 cm with mixed echoes, a clear margin, and detectable calcification inside. It was mainly located in the right anterior lobe. An abdominal enhanced CT and a three-dimensional model on day 2 showed a large round mass (8.5 cm × 6.6 cm), a clear boundary, and uneven enhancement . The hepDue to the large size and rapid growth of the tumor, the compression on the surrounding organs led to liver and coagulation dysfunction and feeding difficulties. Additionally, the tumor surface tension was so high that the risk of tumor rupture during biopsy was increased significantly. After full communication with the surgeon, it was clear that there were too many temporary contraindications for puncture and operation. After the parents understood the condition and situation of the infant, they all hoped to start chemotherapy as soon as possible to reduce the load of the tumor. Preoperative chemotherapy with Cisplatin-5fluorouracil-Vincristine (C5V) was initiated on day four. After four courses of chemotherapy, the tumor was reduced to 5.8 cm × 5.3 cm, and the AFP had declined to 2,516 ng/ml . During chemotherapy, infectious diarrhea and transient acute kidney injury (type I) occurred on the third day of the first chemotherapy remission. The highest serum creatinine was 1.55 mg/dl, urinary microalbumin 193 mg/L , urinary transferrin 9.48 mg/L , and urine α1 microglobulin 94.7 mg/L , indicating early kidney injury. These readings returned to normal after anti-infection and symptomatic treatment. Complete resection of the tumor in the right lobe and part of the left lobe of the liver was performed 3 months after birth. The tumor size was about 5.5 cm × 5 cm × 4 cm, and no intravascular tumor thrombus was found.Postoperative pathology showed mixed epithelial and mesenchymal hepatoblastoma. The epithelial component was the fetal type which differentiated well. After surgery, four courses of C5V were continued, followed by one course of C5VD (C5V plus doxorubicin). There was no organ injury during chemotherapy, and no cardiotoxicity, hepatotoxicity, or neurotoxicity was detected. In terms of blood, platelet levels remained normal, and occasionally, granulocytopenia or anemia could be recovered. Abdominal enhanced CT re-examination showed that the tumor had disappeared completely. Postoperative chemotherapy was continued until 6 months after birth. After that, the patient had repeated abdominal ultrasounds and abdominal enhanced CT re-examinations at 6 months and 1 year old, and no evidence of recurrence was found. The patient's AFP gradually decreased to 18–32 ng/ml . She is now 2 years old and is still receiving follow-up examinations .Congenital liver occupation is a rare occurrence, with hemangioma being the most common, followed by hepatoblastoma and mesenchymal hamartoma . HepatobIt has been reported that only 1 out of 42 congenital hepatoblastoma cases was diagnosed as intrauterine . DelayedPrognostic factors for hepatoblastoma include the tumor's PRETEXT stage, Evans stage, AFP level at diagnosis, and pathological classification. They are also associated with early detection and timely chemotherapy and surgical treatment before distant metastasis, occurrence of respiratory and heart failure and tumor rupture due to compression effects –16. To iThe neonatal period is a period of fast growth. The development of fetuses' organ structures and functions is so different that the pharmacokinetics and pharmacodynamics for chemotherapy drugs are unstable. Additionally, the infants' tolerance to chemotherapy drugs and the forward impact on development make chemotherapy in newborns difficult . Four inIn recent years, the combination of surgery and chemotherapy is recommended internationally for treatment of hepatoblastoma; this treatment can significantly improve the negative rate of tumor resection margin and improve the prognosis. Congenital hepatoblastoma has the characteristics of early onset, rapid progression, easy metastasis, and risk of tumor rupture and compression effect. Preoperative chemotherapy should be implemented early, once the diagnosis is made. Sometimes, if biopsy is not available for clinically diagnosed hepatoblastoma, which progresses rapidly, squeezes other organs severely, and has a high risk of rupture, we can also start chemotherapy to reduce the harm caused by the tumor and create opportunities for biopsy and surgery. Although the structure and function of neonatal organs grow and develop rapidly, resulting in the instability of pharmacokinetics of chemotherapy drugs, reports in recent years state that chemotherapy can be tolerated in the short and long terms when the general condition of infants is stable. The infant in our case report had a stable general condition, although infectious diarrhea and acute kidney injury (stage I) occurred during chemotherapy but returned to normal after anti-infection and symptomatic treatment. Occasionally, we detected neutropenia or deficiency anemia, but all of which were recoverable. We followed up this infant for 2 years, and no tumor recurrence or functional impairment of systems and organs was observed.The original contributions presented in the study are included in the article/supplementary material, further inquiries can be directed to the corresponding author/s.ZY wrote the manuscript. WB and LL collected the data and followed up the case. SL and YH supervised the management and follow-up of the case. All authors revised and approved the final version of the manuscript and agreed to be accountable for the content of the article.This research was supported by Beijing Municipal Science & Technology Commission (Z191100006619049).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.


Solitary fibrous tumour of the pleura (SFT) is rare neoplasms and consist less than 5% of the primary tumours of the pleura. In the English literature, very few cases of giant solitary fibrous tumours have been described. We report a clinical case of an intrathoracic giant SFT of the pleura in a 62‐year‐old female patient. Additionally, we reviewed the clinical, imaging and histopathological features, the therapeutic management and the clinical course of giant SFTs published in the English literature. For this, we conducted a comprehensive electronic search at the PubMed using the key words giant, huge, big and enormous. Solitary fibrous tumour of the pleura (SFT) is rare neoplasms and consist less than 5% of the primary tumours of the pleura, with very few cases published in the English literature. We report a clinical case of an intrathoracic giant SFT of the pleura in a 62‐year‐old female patient. Additionally, we provide a review of the clinical, imaging and histopathological features, the therapeutic management and the clinical course of giant SFTs published in the English literature. Solitary fibrous tumour of the pleura (SFT) is a rare neoplasm accounting for less than 5% of primary pleural tumours and the incidence is approximately at 2.8 cases/100,000 per year.We report a clinical case of an intrathoracic giant SFT of the pleura in a 62‐year‐old female patient. Additionally, we reviewed the clinical, imaging and histopathological features, the therapeutic management and the clinical course of giant SFTs published in the English literature. For this, we conducted a comprehensive electronic search at the PubMed using the key words giant, huge, big and enormous.2 = 91% on room air) and low‐glucose levels (45 mg/dl). Chest x‐ray revealed opacification of the left middle and lower pulmonary fields  at the last week. Physical examination of the chest revealed absent breath sounds on the entire surface of the left hemithorax. Standard laboratory results showed elevated C‐Reactive Protein levels (6.5 μg/dl), hypoxemia  patients suffered from refractory hypoglycemia (Doege–Potter syndrome). The low‐glucose levels observed in our patient resolved immediately after surgery and could be attributed to that syndrome. In 13% (n = 29) of the cases, a pleural effusion accompanying the giant SFT was noted. It accompanied both malignant and benign tumours and, in all cases, cytological examination of the pleural fluid was negative for malignant cells. Histologically, approximately 25% (n = 59) of the giant SFTs were found to be malignant with six of those patients presenting distant metastasis. In the literature, different histological features have been described which are suggestive of malignancy. These features include increased cellularity, increased mitotic activity (≥4/10 high‐power fields [HPF] or >2 mitoses/2 mm2), nuclear pleomorphism and tumour necrosis.n = 8) of the giant SFTs, while post‐operative radiotherapy was performed in the 5% (n = 11) of the patients . Ten malignant giant SFTs recurred. Four of these patients underwent repeat surgical resection and were disease‐free at last follow up. The other six patients were treated with chemotherapy/radiotherapy, but two of them died within 9 months. Relapse was also noted in six benign giant SFTs. During a 20‐year period, one of those patients experienced three relapses, each treated with repeat surgical resection. The rest of the patients with giant SFTs were alive at last follow‐up.Until now, there have been described in the literature approximately 220 cases of SFTs with maximal diameter > 10 cm . The mean age of the patients being 57 years (range 15–83 years), both men (46%) and women (54%) are affected, while most of the tumours (52%) were located on the left hemithorax. Sixteen patients (7%) were asymptomatic at presentation. Dyspnea was the most frequent symptom (46%), followed by chest pain (21%) and cough (20%). Fever has been documented in about 4% of the cases. Digital clubbing and hypertrophic pulmonary osteoarthropathy  seemed to affect up to 5% of patients with giant SFTs, with the remission of the syndrome observed immediately after the removal of the tumour, while 17% (In conclusion, we here report a case of middle‐aged woman with a giant SFT of the left hemithorax treated with surgical resection through median sternotomy. A comprehensive review of the English literature on SFTs revealed that these tumours may occur in both sexes, they are most often benign and they are treated with surgical resection, most commonly conducted through thoracotomy. Disease re‐occurrence and death are uncommon.Study conception and design: Theofani Rimpa, Ioannis Kalomenidis. Data collection: Kalliopi Athanassiadi, Christina Vourlakou, Ioannis Chrysovergis, Zacharias Diakonikolaou. Analysis and interpretation of results: Theofani Rimpa, Kostantinos Eleftheriou. Draft manuscript preparation: Theofani Rimpa, Paraskevi Katsaounou, Ioannis Kalomenidis.None declared.The authors declare that appropriate written informed consent was obtained for the publication of this manuscript and accompanying images.


Sinonasal lesions have a wide range of presentations and etiology. Vascular lesions of the nose and sinuses usually present as epistaxis. Bleeding nasal lesions are a challenge as they very often present in the emergency. Since biopsy from a bleeding lesion is difficult and may lead to catastrophe, the diagnosis mainly depends on the radiology. Computerized tomography is the mainstay of sinonasal radiology followed by magnetic resonance imaging, whenever required. However, computerized tomography and magnetic resonance imaging would give an accurate picture of the extent of the disease, its usefulness decreases when a proper diagnosis is required. However, radiology would be helpful in differentiating between inflammatory, benign and malignant lesion.An 85 year-old male patient presented to the ENT outpatient department with a history of right sided nasal obstruction of 2 months duration and intermittent right sided epistaxis for a 1 month duration. The bleeding episodes were mild and did not require any consultation or intervention as they stopped spontaneously. He was a known hypertensive on medication. There were no other comorbidities. Nasal endoscopy of the patient revealed a pinkish bleeding mass in the posterior part of the right nasal cavity and nasopharynx A and B. A 22 year-old male presented to our outpatient department with gradually progressing nasal obstruction of the left side for 2 months and swelling over the nasal dorsum and nasofacial groove for 1 month. He also complained of a lumpy feel in the throat and increased watering from the left eye. He gave a history of intermittent oral and nasal bleeding which was trivial and did not require any intervention. On examination, his left inferior and middle turbinates were enlarged and the mass was going posteriorly into the nasopharynx and oropharynx A and B. A 61 year-old male patient presented to the outpatient department with a history of recurrent early morning nasal bleeding, typically on bending down. He was hypertensive and had coronary artery disease for which he was on anti-hypertensives and antiplatelets in the form of aspirin and clopidogrel. A diagnostic nasal endoscopy was done which showed bleeding medial to the middle turbinate. A contrast-enhanced computerized tomography was done to look for any vascular malformation. The radiology revealed an osteolytic lesion involving the floor of the sphenoid sinus and the clivus. It showed homogenous post contrast enhancement involving the upper clivus without involving the brain and paranasal sinus A and B. Sinonasal diseases are very common ranging from inflammatory conditions to malignancies. Since the region is surrounded by bone, computerized tomography remains the first line of investigation, which is further complimented by magnetic resonance imaging. Histopathological examination is required for the final confirmatory diagnosis as most of the tumors have nonspecific radiological features. But radiology is helpful in differentiating the benign and malignant tumors. Features like bone remodeling and thickening are features of benign lesions, whereas bone destruction, ill-defined margins, and the presence of lymphadenopathy are features of malignancy.Our first patient had a vascular lesion in the nasal cavity, which looked benign. The computerized tomography revealed a post contrast heterogeneously enhancing mass without any bony destruction. The lesion was diagnosed as hemangioma due to benign nature and intense post contrast enhancement without any local bone invasion or destruction.Our second patient had a huge lesion completely filling the entire left nasal cavity. Both the inferior and middle turbinate was involved with the disease and the tumor extending into the oropharynx. The contrast enhanced computerized tomography showed moderately enhancing infiltrating lesion in the left nasal cavity with the destruction of the medial wall of maxillary sinus and inferior and middle turbinate. In sinonasal imaging, generally bone destruction suggests malignancy.Our third patient had a vascular lesion in the clivus. Clival tumors are a rare diagnosis and they usually present with cranial nerve palsies. Clival chordoma and chondrosarcoma are the most common tumors of clivus comprising only 0.1%–0.2% of the overall skull base tumors. Metastasis to clivus is rarer and metastasis in the absence of primary tumor is extremely rare.Clival chordoma presents as a centrally located, well-circumscribed, expansile soft-tissue mass associated with extensive lytic bone destruction. The main tumor is usually hyperattenuating with intratumoral calcifications from either destruction or calcification.Diagnosis of sinonasal lesions from the radiology may be accurate in the majority of situations. However, in rare situations, the radiological and histopathological diagnosis may be entirely different. Hence, it is always better to discuss the other possibilities at least under broad terms like benign or malignant to the patient before proceeding with surgical excision so that the patient gets prepared for unexpected outcomes.The authors declare no conflicts of interest.


The proximity of the uterus and the cervix to the urinary tract predisposes the latter to injury during obstetrical and gynaecological surgical procedures. Following a difficult surgical procedure on the lower uterine segment and or adnexa, urinary tract injury should be excluded.A booked 39-year-old G3P2 lady who suffered an ischaemic stroke in the index pregnancy had a caesarean delivery at 39 weeks of gestation and sustained an extensive tear that extended inferiorly on the left lateral aspect of the uterus and this resulted in postpartum haemorrhage. Following the repair of the tear, uterine artery ligation was performed to achieve haemostasis.Postoperatively, conventional ultrasonography which was performed to exclude ureteric injury suggested left hydronephrosis and a preliminary report of computerized tomography (CT) showed the same finding. The patient subsequently had left ureteric stenting. The final report of the CT scan was delayed but showed a simple left renal cyst and no hydronephrosis.Renal cyst is a differential diagnosis of hydronephrosis. Delayed availability of the final result of medical investigations jeopardises patients' safety. A preliminary imaging report is prone to error and its use to determine the indication for an invasive procedure should be limited to emergencies. One of the measures to control postpartum haemorrhage (PPH) is uterine artery ligation.In the case presented, a woman living with HIV had a stroke during pregnancy. She had caesarean delivery at term and developed PPH which was controlled with uterine artery ligation. Postoperatively, ultrasonography and preliminary report of computerized tomography (CT) intravenous pyelogram (IVP) misdiagnosed renal cyst as hydronephrosis and the patient had stenting of left ureter before the final CT report with the correct diagnosis was available. This report discusses the following lessons: (i) delay in availability of medical investigation report which frequently occurs in low-resource settings, (ii) succinct technique of uterine artery ligation and how to prevent and evaluate inadvertent ligation of the ureter, and (iii) adjunct obstetric concerns. This article is the first reported case of renal cyst mimicking hydronephrosis in the context of PPH amenable to uterine artery ligation.2) with a normal obstetric ultrasound and blood pressure of 121/80 mmHg during the first antenatal clinic visit. She suffered ischaemic stroke at 20 gestational weeks and developed slurred speech and left hemiparesis with limping gait (power on the left limbs was 4/5).In details, a 39-year-old G3P2 lady commenced antenatal care in a private hospital. She was a woman living with HIV and had been on treatment since 2012 with a CD4 count of 399 cells/µl. The viral load was lower than detectable. The patient was obese , abdominopelvic ultrasonography was requested based on the local protocol (unpublished) to exclude intra-operative ureteric injury. The ultrasonography was performed on day 2 postpartum and showed features of left hydronephrosis. The renal function test was normal. CT IVP was requested on day 3 postpartum. The facility for CT IVP was unavailable at the base hospital and the imaging was performed in another health facility on day 4 postpartum due to transport challenges. The preliminary report of the CT IVP suggested features of hydronephrosis. Following a 24-hour delay without receiving the final CT IVP report, retrograde ureterogram was performed on day 5 postpartum and was normal. At the same time, a stent was inserted in the left ureter because of the possibility that the ureter could be kinked. The final report of CT IVP was obtained 12 hours after the stent had been inserted and showed simple left renal cortical cyst (Bosniak I lesion) The index case report shows that the scope of routine antenatal ultrasonography in the early pregnancy is limitedFurthermore, the delay in receiving the final CT IVP report was a major concern. This is a challenge associated with some medical investigations requested in many low-resource settings. The delay may be caused by reasons such as lack of onsite capacity, power outage, lack and faulty equipment, delay in payment of medical services by patients,To perform uterine artery ligation, the bladder is mobilized inferiorlyclinicians should consider renal cyst as a differential diagnosis of hydronephrosis and exercise caution when deciding on a surgical intervention based on preliminary imaging report. Assessment of renal calyces during antenatal ultrasonography, where possible, may prevent misdiagnosis of a pre-existing renal lesion as a complication of childbirth.


Methotrexate-related lymphoproliferative disorder (MTX-LPD) is a rare but serious complication that occurs in patients treated with methotrexate (MTX); although MTX-LPD has been reported recently, the incidence in the colon is very low. A 79-year-old woman who had been receiving MTX for 15 years came to our hospital complaining of postprandial abdominal pain and nausea. Computed tomography scan showed the dilation of the small bowel and a tumor in the cecum. In addition, numerous nodular lesions were seen in the peritoneum. Ileal-transverse colon bypass surgery was performed for small bowel obstruction. Histopathological findings of both the cecum and the peritoneal nodules revealed the diagnosis of MTX-LPD. We report MTX-LPD occurring in the colon; it is important to consider MTX-LPD when intestinal symptoms occur during MTX therapy. Methotrexate (MTX) has generally been the first-line drug for RA treatment since its adoption in the 1980s; MTX has benefited a significant number of RA patients.et al. found the development of lymphoma in RA patients treated with MTX. Since then, after similar cases were reported, the condition became known as MTX-LPD.The first association between MTX and lymphoma was reported in 1991 when Ellman About 50% of MTX-LPD occurs in extralymphanode sites such as the lungs, skin and oropharynx, but it is uncommon to occur in the colon. About half of the MTX-LPD patients improve with the discontinuation of MTX.Here, we report a rare case of small bowel obstruction because of MTX-LPD, in which an ileal-transverse colon bypass was performed.A 79-year-old woman, who had been taking MTX 4.0 mg/week for rheumatoid arthritis since she was 64 years old, came to our hospital complaining of vomiting repeatedly. Physical examination revealed no tenderness in the abdomen, but the abdomen was distended and a mass was observed in the right lower abdomen. Contrast-enhanced computed tomography revealed a mass lesion with contrast effect in the cecum and multiple peritoneal tumors  and 2.Colonoscopy revealed an irregular ulcerative lesion in the cecum, invading the Bauhin valve .Ileal-transverse colon bypass was performed because of the obstruction of the small intestine.The patient underwent surgery after removal of the disseminated nodule for additional histopathological examination.The postoperative course was unremarkable.Immunohistochemistry showed CD5(+) and CD20(+) in both the ulcerative lesion in the cecum and the peritoneal disseminated nodule, and a diagnosis of diffuse large B cell lymphoma (DLCBL) was made.Since the patient had been taking MTX, he was diagnosed with methotrexate-associated lymphoproliferative disorders (MTX-LPD) and MTX was discontinued.The patient was transferred to another hospital for chemotherapy.et al. [MTX is known as an anticancer drug and immunosuppressive drug and is widely used as a treatment for rheumatoid arthritis in particular, and reports of MTX-LPD as a side effect of MTX have been increasing since Ellman et al.  reportedClassification of Tumors of Hematopoietic and Lymphoid Tissues, Fourth Edition, similar to immunodeficiency-related LPD, including posttransplant LPD and human immunodeficiency-related LPD [Methotrexate-related lymphoproliferative disorder (MTX-LPD) is classified as ‘other iatrogenic immunodeficiency-related LPD’ in the World Health Organization’s ated LPD . MTX cauThe etiology is still unknown, but histology has shown that it is primarily of B-cell origin, suggesting an association with EBV.Pathologically, DLBCL is the most common, accounting for 35–60% of MTX-LPD cases, followed by Hodgkin lymphoma in 12–25% of cases [Extralymphatic lesions are seen in about half of the cases, but the most common sites other than the lymph nodes are the lungs, skin and oropharynx, and cases affecting the large intestine are extremely rare .Risk factors for the development of MTX-LPD include taking more than 8 mg/week , and RA Diagnostic criteria for MTX-LPD have not established yet. The Japan College of Rheumatology recommends that the possibility of MTX-LPD be considered when RA patients on MTX therapy present with systemic symptoms such as indeterminate fever, general malaise and weight loss, or with laboratory abnormalities such as hepatosplenomegaly, abnormal white blood cell differential counts, decreased platelet counts, anemia and high lac-acid dehydrogenase . PatientThe first option for treatment is the discontinuation of MTX. Previous studies have reported that almost half of the MTX-LPD cases show regression with MTX discontinuation alone , 7.et al. [Inui et al.  reportedFor these patients who do not respond to MTX withdrawal, conventional chemotherapy for lymphoma should be considered .In this case, the patient was found as a colorectal mass and was suspected to have peritoneal dissemination.It was accompanied by small bowel obstruction, and bypass surgery was performed. The histopathological diagnosis of the disseminated nodule was also a result of DLBCL.It is important to keep in mind the possibility of MTX-LPD when a patient on MTX medication presents with gastrointestinal lesions.Although MTX-LPD is rare, it has been increasing in recent years and care should be taken when treating it with MTX.Treatment should include the discontinuation of MTX, which improves about half of the patients, and chemotherapy if there is no improvement after 8 or more weeks of discontinuation.Patients being treated with MTX should be suspected of involvement if they present with lymphoma.


The ectopic pancreas is a benign subepithelial tumor (SET) mostly found incidentally in the stomach and duodenum. Here, we present computed tomography (CT) scans and endoscopic ultrasound (EUS) images from a 71-year-old Taiwanese man newly diagnosed with colonic adenocarcinoma. CT examination revealed a mural nodule in the proximal jejunum, with good enhancement after IV contrast medium administration. Push enteroscopy was performed to localize the lesion and evaluate its nature, and a 1 cm subepithelial lesion was found. The lesion appeared hyperechoic within the submucosal layer of the bowel wall on endoscopic ultrasound examination. A tattoo was performed, and the lesion was removed during the resection of colon cancer. The histopathology confirmed the presence of pancreatic tissue inside. As far as we know, this is the first description in the literature of an endoscopic ultrasound finding of a jejunal ectopic pancreas. A 71-year-old man with underlying hypertension and hepatitis B. He visited our gastrointestinal outpatient ward due to stool occult blood testing positive in a regular health examination. A colonoscopy revealed a fungating mass about 2 cm in size at the ascending colon. A biopsy confirms the diagnosis of colonic adenocarcinoma. In addition, an abdominal computed tomography (CT) was arranged, which reported no lymph node or distant metastatic lesions. However, a small mural nodule was found , arrow iTo evaluate the nature of the lesion pre-operatively, we performed push enteroscopy, and a 1 cm oval lesion was found in the proximal jejunum , arrow.To further characterize the lesion, a mini-probe ultrasound was performed, which revealed a homogeneous-echogenic lesion involving the submucosal layer with an indistinct margin , arrows.The endoscopy biopsy was negative for malignancy. Given the lesion’s feasibility and undetermined nature, the patient chose to have the jejunal tumor treated at the same time as his colon cancer via laparoscopic surgery.The resected jejunum measured 5 cm in length and 5.5 cm in circumference. There was a tumor lesion measuring 1.5 × 1 × 0.7 cm on the mucosal surface. The histopathology revealed the presence of pancreatic tissue in the submucosal layer of the jejunum. According to Heinrich’s classification [The ectopic pancreas, also called the heterotopic or aberrant pancreas, is defined as pancreatic tissue that is anatomically separate from the central gland. Most of the ectopic pancreas was asymptomatic and presented as a small subepithelial tumor (SET) in the gastric antrum with a small central umbilication ,3,4. TheIn addition to cross-sectional imaging, an endoscopic ultrasound (EUS) is the best tool to characterize the features  of the subepithelial lesion . The ectIt is tough to diagnose an incidentally detected ectopic pancreas in the jejunum. Therefore, we present a complete image study for this rare jejunal subepithelial lesion. It is the first reported EUS image of the jejunal ectopic pancreas in the literature.


We report the case that a patient with Neurofibromatosis type 1 experienced bowel intussusception and adhesive intestinal obstruction. Bowel intussusception was considered to be due to long intestinal tube and multiple intraabdominal lesions including gastrointestinal stromal tumors (GISTs). A 63‐year‐old man with neurofibromatosis type 1 (NF1) presented to the emergency department with a loss of appetite and vomiting for 3 days. He had abdominal pain 3 days prior to presentation, which started after consumption of partially cooked fish. Although the pain subsided, he was unable to eat. The patient had no other medical history, including previous abdominal surgery. His vital signs were normal. Abdominal examination revealed minor distention and no tenderness. Several neurofibromas and café au lait macules were observed. Computed tomography (CT) revealed an obstruction in the small intestine, suggesting an adhesion or cord‐like structure, which changed the caliber of the small intestine Figure . Bowel rNF1 is a neurocutaneous disorder that causes 25% of all gastrointestinal tract lesions.In this case, the adhesion and cord‐like structure between the greater omentum and mesentery caused the obstruction. The insertion of the long tube may have induced bowel intussusception.The authors declare no conflict of interests for this article.Obtained.