Meningiomas are tumors that originate from the meningeal or dural cover of the brain and are the most common primary benign brain tumors. Currently, the accepted management of these tumors is attempted total surgical excision when technically possible and associated with an acceptable risk. However, radiation therapy has been shown to provide excellent local control when used either as an adjunct to surgery or as a primary treatment. We present a case report of a 46-year-old female patient with an unresectable left foramen magnum meningioma resulting in headaches, neck pain, and swallowing difficulty. The patient underwent CyberKnife  radiosurgery to a dose of 3,000 cGy in five fractions in March 2011. The patient tolerated treatment without complications and has remained clinically well with no ongoing cranial nerve deficits as of the last examination in late 2019. This demonstrates the excellent local control obtained when using radiosurgery as both a surgical adjunct and a primary treatment for meningiomas. Meningiomas are tumors that originate from the meningeal or dural cover of the brain. Meningiomas are the most common benign primary brain tumor, accounting for approximately 25% of all intracranial neoplasms, which increased to 40% if autopsy data are included . The vasA 46-year-old female patient presented with a one-year history of progressive left occipital headaches and neck pain, with a six-month history of difficulty swallowing and a thickening sensation in her tongue. She also reported some difficulties with speech and a heightened sensitivity in the left V1/V2 trigeminal distributions, with referred otalgia. MRI completed in July 2010 revealed a mass in the left foramen magnum, isointense on T1 and T2 and enhancing with gadolinium, compressing the left side of the medulla and upper cervical cord, and extending through the left occipital bone and hypoglossal canal to the left jugular foramen region and left carotid sheath. The patient was initially seen at a more remote neurosurgery center, following which the patient was referred for a multidisciplinary assessment at a tertiary care center with expertise in skull base surgery. The tumor was deemed inoperable due to high morbidity associated with the location and extent of the tumor. The patient was referred to our center for consideration of CyberKnife .On examination in January 2011, she was found to have increased sensitivity in the V1/V2 distributions and impaired mobility of the tongue resulting in dysarthria. A pre-procedure swallowing study revealed no oral or pharyngeal swallowing difficulties; however, the patient subjectively had some difficulty swallowing both solids and liquids.In March 2011, the patient underwent CyberKnife radiosurgery to a dose of 3,000 cGy in five fractions. Treatment was delivered using CyberKnife robotic stereotactic radiosurgery (SRS) with real-time tumor tracking (Figure The patient tolerated treatment without complications and no pre-medication was required. Nine months post-radiosurgery, the patient had improvement in pain and no longer required opioids around the clock. Examination revealed relatively normal cranial nerve examination with good swallowing and gag reflex. Facial sensation was within normal limits.The patient remained clinically well and as of the most recent examination in 2019, she continues to remain well with no cranial nerve deficits, a slightly deviated tongue, no swallowing issues, and normal gait (Figure Meningiomas may present with a wide array of symptoms dependent upon their size, location, and associated presence of vasogenic edema. Foramen magnum meningiomas represent a common histological tumor in a rare and eloquent location . Surgery3 enhancing meningioma. The most recent surveillance MRI in April 2019 showed a residual enhancing volume of 10 cm3, which is equivalent to 30% volumetric involution.Pre-treatment volumetric data from 2011 showed a 14.2 cmConsidering the complex anatomy of the foramen magnum region and the encasement of anatomical structures by the tumor, surgical resection would result in extensive morbidity and result in subtotal resection. Radiosurgery is a well-established alternative to treat unresectable lesions. Given the mass effect of the tumor over the brainstem, it was elected to proceed with fractionated radiosurgery in order to respect the dose constraints for the brainstem.The patient tolerated the treatments without issue and she started to improve clinically and radiographically within nine months post-treatment. She is currently eight years post-treatment continuing to present gradual and progressive clinical improvement and radiological regression of the residual enhancing lesion.The dramatic presentation and learning points pertaining to this case include the compression of the medulla and upper cervical cord, with tumor extending through the left occipital bone and hypoglossal canal to the left jugular foramen region and left carotid sheath. Tumor regression with symptom improvement and long-term local control were obtained with fractioned CyberKnife radiosurgery.


Teaching point: Clival chordoma is a rare find in children, presenting as a locally destructive, T2 hyperintense, and strongly enhancing mass. Figure 1, arrow), without any signal suppression on the fluid-attenuated inversion recovery (FLAIR) sequence. There was a strong heterogenous contrast enhancement . Computed tomography (CT) showed a sharply delineated soft-tissue mass, with neither calcification nor bone tissue . No other osteolytic bone lesion was detected. Based on imaging findings, the diagnosis of chordoma was proposed. The lesion was resected surgically and the diagnosis of chordoma was confirmed on histology. Postoperatively, the patient underwent several sessions of radiation therapy and is currently under follow-up, free of relapse.An eleven-year-old girl was referred to a pediatrician because of a persistent headache. Physical examination was normal. Magnetic resonance imaging (MRI) of the brain showed a 2 cm sharply delineated mass in the caudal part of the clivus, at the height of the basion, with low signal-intensity on T1-weighted imaging. On T2-weighted imaging, the lesion appeared heterogeneously hyperintense . The tumor is mainly locally destructive, and distant metastases are relatively rare. The symptoms of a clival chordoma include diplopia and headache. The tumor presents as a lobulated, gray mass, with possible areas of calcification and hemorrhage. On CT it appears as a sharply defined, expansile, soft tissue mass, with extensive lytic bone destruction. On MRI the tumor is typically hyperintense to the cerebrospinal fluid on a T2-weighted sequence and may have low-signal fibrous septations. On a T1-weighted sequence, the tumor appears heterogeneously hypo- to isointense compared to the bone marrow, showing mild to intense enhancement after administration of contrast. Differential diagnosis should be made with benign notochordal cell tumors, chondrosarcomas, and Langerhans cell histiocytosis. Benign notochordal cell tumors usually have no contrast enhancement. The majority of chondrosarcomas are located off the midline, have a lytic aspect with calcifications, and show a serpiginous enhancement. Langerhans cell histiocytosis appears as lytic punched out lesions, typically in the skull, long bones, and mandible and shows a diffuse contrast enhancement. A chordoma is usually treated by surgical resection with adjuvant radiation therapy. After successful treatment, recurrence is not uncommon, especially along the operative tract. The five-year overall survival rate of pediatric chordomas in the literature varies between 60–80%. While the prognosis in children is better than in adults, patients younger than 5 years old generally present with very aggressive chordomas and poor outcomes [


PMS2 gene. She developed an early stage adenocarcinoma of the colon at the age of 14. Surveillance MRI of the brain at age 18 resulted in the detection of an asymptomatic brain cancer. On resection, this was diagnosed as an anaplastic astrocytoma. Due to emerging literature suggesting benefit of immunotherapy in this patient population, she was treated with adjuvant dual immune checkpoint inhibition, avoiding radiation. The patient remains stable with no evidence of progression 20 months after resection. The patient’s clinical course, as well as the rational for considering adjuvant immunotherapy in patients with CMMRD are discussed in this report.Constitutional mismatch repair deficiency (CMMRD) is a rare autosomal recessive hereditary cancer syndrome due to biallelic germline mutation involving one of the four DNA mismatch repair genes. Here we present a case of a young female with CMMRD, homozygous for the c.2002A>G mutation in the  MLH1, MSH2, MSH6, or PMS2) [Constitutional mismatch repair deficiency (CMMRD), first described in 1999 [or PMS2) . The MMRor PMS2) ,5. CMMRDor PMS2) . The mosor PMS2) ,8.PMS2, with the mean age of first malignancy of 10 years, compared to 7.5 and 8 years for patients with biallelic loss of MSH6 and MLH1/MSH2, respectively [PMS2 mutation is also associated with a greater incidence of brain tumors, colorectal cancer (CRC), and endometrial carcinomas compared to biallelic mutations in MLH1, MSH2, and MSH6 which have a proportionally higher rate of hematologic malignancies [The first malignancy in CMMRD is usually diagnosed within the first decade of life . Brain tectively ,11. Bialgnancies ,13. Varignancies ,13. Wholgnancies  and patignancies  and survgnancies .IDH-mutant anaplastic astrocytoma (AA). To our knowledge, this is the first published case of a CMMRD associated IDH-mutant AA treated with adjuvant nivolumab and ipilimumab after resection, without adjuvant radiation.As CMMRD is rare, with less than 200 cases reported in the literature, surveillance guidelines and optimal cancer treatment options are still evolving ,9,12. CMPMS2 gene. This variant is classified as likely deleterious and has been described as a founder mutation in the Inuit population of northern Quebec [Here we present a case of a 19-year-old Inuit female who comes from a family known to have CMMRD following the diagnosis of gastric cancer in a sibling at age 11. Our patient underwent molecular testing at age 10, which confirmed she was homozygous for the c.2002A>G mutation in the n Quebec . She wasPMS2 in both neoplastic epithelial and normal mesenchymal elements, a finding specific for CMMRD [The patient subsequently began surveillance gastroscopies and colonoscopies at age 11. At age 14, she was diagnosed with a pT2N0 moderately differentiated adenocarcinoma of the colon, treated with total colectomy. MMR immunohistochemistry showed loss of expression of or CMMRD . Since 2or CMMRD ,11,12,13IDH1 R132H mutant, WHO grade 3, with immunohistochemistry results suggestive of biallelic PMS2 mutation .On first surveillance brain MRI, at age 18, she was found to have a 2.6 cm mass-like area of FLAIR hyperintensity in the right frontal lobe . She undmutation . ImmunohAn MRI brain 12 weeks after surgical resection demonstrated a slight increase of the non-enhancing region deep to the resection cavity, suggestive of possible residual disease. There was new increased signal intensity on FLAIR along the superior and deep margin of the resection cavity. Because of the rarity of this condition, her case was discussed in a multidisciplinary fashion with experts across Canada. Emerging literature suggests that high-grade brain malignancies in children with CMMRD can demonstrate objective responses and prolonged survival following immune checkpoint inhibition (ICI) ,20. As sAfter cycle 1 of nivolumab and ipilimumab, she developed grade 1, clinically asymptomatic hyperthyroidism . She was assessed by endocrinology, and the differential diagnosis was postpartum thyroiditis versus thyroid dysfunction secondary to immunotherapy . Immunotherapy continued with close monitoring of clinical symptoms and bloodwork with gradual improvement in her thyroid function tests. After cycle 4, she developed headache, decreased appetite, and fatigue. Bloodwork confirmed grade 2 hypothyroidism  and grade 2 adrenal insufficiency (random cortisol 38 nmol/L). MRI showed immune-related hypophysitis. Maintenance nivolumab was held and she was treated with levothyroxine, as well as a tapering course of prednisone followed by replacement hydrocortisone. Hospitalization was not required. No other immune-related adverse events were experienced. A brain MRI demonstrated stable post-treatment changes prior to the re-initiation of maintenance nivolumab. Her MRI continues to show stability with no evidence of progression 20 months post-resection and 8 months post-hypophysitis.IDH1 mutant MMR-deficient HGG have been documented to have a median OS of 15 months with conventional chemo-irradiation based approaches [Gliomas are classified and graded based on histologic and molecular features. Grade 3 (AA) and 4 (glioblastoma) gliomas are classified as HGG. Standard treatment is dependent on these characteristics, with AA being treated with concurrent radiation and temozolomide followed by adjuvant temozolomide . Pediatrproaches .A recent report from an international consortium demonstrated that early detection of cancers using systematic surveillance can significantly improve survival in patients with CMMRD, including those with malignant brain tumors . As CMMRIn many areas of oncology, ICI have provided promise and dramatically altered the landscape in cancer treatments and outcomes, predominantly in tumors with high mutation burden, microsatellite-instability and/or an immune microenvironment ,29; howeThe KEYNOTE 158 study evaluated pembrolizumab in tumors with high mutational burden (≥10 mutations/Mb) and found a 29% response rate, resulting in Food & Drug Administration (FDA) approval of pembrolizumab for this indication in June 2020 . BecauseTo our knowledge, we present the first case of a patient with a CMMRD associated HGG, detected at an asymptomatic stage using systematic surveillance, and treated using adjuvant nivolumab and ipilimumab following surgical resection. She is alive 20 months following her diagnosis. The prolonged survival following ICI is remarkable for a rapidly fatal and aggressive cancer. The additional opportunity to omit radiotherapy, thereby eliminating its late sequelae, may offer a new paradigm for management of these patients in the future.


Mesenteric cysts are uncommon benign tumors with atypical clinical presentations. An acute presentation may not necessarily warrant emergency surgery, and planned surgery can achieve excellent results if the patient is hemodynamically stable at presentation. Mesenteric cysts are uncommon benign tumors with atypical clinical presentations. An acute presentation may not necessarily warrant emergency surgery, and planned surgery can achieve excellent results if the patient is hemodynamically stable at presentation. This report describes the case of a patient who presented acutely. The patient was initially managed conservatively and later surgically. Open laparotomy was performed; histology revealed a benign hemorrhagic cyst of the mesentery.Mesenteric cysts are uncommon benign intra‐abdominal tumors that occur across the life course, with a reported incidence of 1/100 000 in adults and 1/20 000 in children and a female‐to‐male ratio of 2:1.Complete surgical excision, by either open or laparoscopic approach, ensures the lowest risk of recurrence. This report highlights the case of a 47‐year‐old patient who presented acutely but was successfully managed semi‐electively.22.1A 47‐year‐old farm worker presented to our hospital with a 1‐year history of progressive abdominal distension that was initially painless but became painful 3 days prior to admission. At the time of presentation, he reported falling on his abdomen while working with cattle in the field 3 days earlier. He also reported a history of difficulty in breathing while lying supine. However, there was no history of cough. The patient had normal bowel and bladder habits.2.2Physical examination revealed the patient to be in fair general condition with no wasting. He was not febrile, anemic, or jaundiced. There was great abdominal distension with no clear delineation of any abdominal masses. Other systemic examinations were unremarkable, with no clinically significant findings.2.3The initial blood work‐up included complete blood count, liver function, renal function, and serum electrolytes, all of which were within normal ranges. The patient was hepatitis B surface antigen‐negative and sero‐negative for human immunodeficiency virus.Imaging investigations included an abdominopelvic ultrasound scan, which revealed a cystic mass occupying much of the abdominal cavity, with no clear site of origin. A contrast‐enhanced computed tomography (CT) scan of the abdomen showed a cystic mass extending from the lower edge of the pancreatic tail to the pelvic brim, with anterior and posterior extension almost to the kidneys. The cyst measured approximately 15.4 cm X 27.4 cm (Figure The above features suggested the presence of a large mesenteric cyst, with a differential diagnosis of a large pancreatic pseudocyst. On the basis of the presentation and imaging findings, the patient was prepared for exploratory laparotomy within 1 week.2.4Initial management included analgesia and broad‐spectrum antibiotics for symptom relief and for treatment of a suspected infected cyst. Because of the suspicion of infection, exploratory surgery was delayed. After 1 week, the decision was made to perform exploratory laparotomy. When the abdomen was opened, the cyst was observed to occupy much of the abdominal cavity Figure . The cysClose inspection revealed involvement of the mesentery of the ileum, with the appendix firmly adhered to the cyst wall. No adhesions were found between the cyst wall and other intra‐abdominal structures. The cyst was carefully dissected from the mesentery of the ileum, sparing the blood supply. Complete enucleation of the cyst was accomplished without the need for bowel resection. However, appendectomy was performed because the appendix was firmly adhered to the cyst wall Figures  and 5.The final specimen sent for histopathology was a thick‐walled disk‐shaped cystic mass measuring 20 × 18 × 40 cm. The immediate postoperative period was uneventful, and the patient was discharged on the third day after the surgery.2.5By the end of the first week postsurgery, there was complete healing of the surgical incision with no wound infection. Histology confirmed a benign hemorrhagic cyst of the mesentery, with the appendix attached to the wall of the cyst. Subsequent reviews were conducted every 2 weeks for 1 month and then at 3 months and again at 6 months. Follow‐up abdominal ultrasound scans revealed no suspicious intra‐abdominal lesions.3Mesenteric cysts are rare intra‐abdominal tumors with varied presentations ranging from incidental discovery during routine imaging investigations to diagnosis following complications such as bowel obstruction.The patient described in this report presented with a long‐standing history of progressive abdominal distension, 3 days of abdominal pain, and a recent fall on the abdomen. The abdominal pain may have been caused by probable hemorrhage into the cyst resulting from a trivial trauma to the abdomen. It is also possible that the abdominal pain was caused by infected cyst fluid, but this is unlikely because the patient was afebrile and because his complete blood count was within the normal range.The etiology of cysts of this type is unclear. Some theories suggest a benign proliferation of ectopic mesenteric tissue that fails to communicate with the core lymphatic system.Imaging such as ultrasound scan, CT scan, and magnetic resonance imaging are the investigation modalities of choice. In the case presented here, a CT scan was sufficient to have a high index of suspicion of mesenteric cyst, although a huge pancreatic pseudocyst was considered as a differential diagnosis.Approaches to the management of mesenteric cysts vary in the available literature. Surgical excision, either by laparotomy or laparoscopy, is the preferred approach.Emergency surgery has inherent risks of complications such as bowel perforation, damage to surrounding structures, wound infection, and intra‐abdominal abscess. In the case presented here, performing the surgery semi‐electively avoided these complications associated with emergency surgery. This approach also ensured that the surgical team had time to prepare mentally and in terms of the necessary skills because this was the first case of its kind to be handled in our facility. However, such an approach is only possible if the patient is stable.4Mesenteric cysts presenting in acute settings can initially be managed conservatively, followed by a planned elective surgical excision of the cyst, preferably by laparotomy. However, the decision to delay surgery should be considered on an individual basis. Overall, the outcome from this approach is excellent if the proper decision is made, as our case clearly demonstrates.None declared.AO: was a major contributor in writing the manuscript and was the lead surgeon in the management of the patient. HA and VM: participated in the literature review and in revising the manuscript. FO and EN: were involved in critically revising the manuscript for important intellectual content. All authors: read and approved the final manuscript.Written informed consent was obtained from the patient for publication of this report and any accompanying images. A copy of the consent form is available for review by the Editor‐in‐Chief of this journal.


This study aimed to present the case of a patient in whom a chopstick, which had been in the duodenum for 10 years, was finally removed by endoscopy. This case was reported because of the long-time retention and noninvasive removal by endoscopy without sedation or complication.A 30-year-old male patient with intermittent upper abdominal pain.During upper-gastrointestinal (GI) endoscopy, a long-strip foreign body (Fb) was seen in the descending part of the duodenum. An upper-GI barium examination was performed, which revealed a linear Fb in the duodenum cavity. The Fb was >10-cm long. Combined with his history , the diagnosis of Fb in the duodenum was confirmed.The Fb was removed from the duodenum cavity by upper-GI endoscopy successfully.The patient was discharged after the removal of the Fb.Endoscopic removal and nonoperative management might be feasible in carefully selected patients with a long and old Fb in the duodenum without the need for anesthesia or surgery as well as no occurrence of complications and laceration. A careful physical examination should be done to detect signs of perforation, such as subcutaneous emphysema or severe abdominal pain, as well as nausea and vomiting. Computed tomography (CT) scanning is superior to plain radiography and identifies the Fb in 70% to 100% of patients.Foreign body (Fb) ingestion is a common emergency. Patients who purposely swallow a true object are typically younger, more often male, and commonly associated with psychiatric illness or drug abuse. To date, only a few cases have been reported on the removal of large Fbs retained in the duodenum using upper-GI endoscopy. A case of long-term retention of a pencil in the upper-GI tract was reported by Lianjun et al. After 5 years of swallowing the pencil, the patient developed abdominal pain, as well as nausea and vomiting, when the body position changed. The diagnosis of perforation by imaging examination was definite, and surgical treatment was the final choice. The present case was reported because of the long-time retention and noninvasive removal by endoscopy without sedation or complication.Fb in the duodenum is more specific than that in other parts of the upper-gastrointestinal (upper-GI) tract. Li et al, in their series of 1088 cases, noted that only 0.04% of ingested Fbs were lodged in the duodenum. No epidemiological data were available on the retention time of Fbs in the upper-GI tract, but the time of endoscopic intervention was dictated by the perceived risks of aspiration or perforation; those without evidence of high-grade obstruction or acute distress could be handled less urgently.In the past, retained Fbs were usually removed by open laparotomy. Actually, the majority (80%–90%) of Fbs pass spontaneously; only 20% of Fb ingestions require endoscopic removal while surgery is necessary for 1% of them.The patient has provided informed consent for the publication of the case.2A 30-year-old male patient with intermittent upper-abdominal pain consulted the department of spleen and stomach in Shaanxi Traditional Chinese Medicine Hospital. During upper-GI endoscopy, a long-strip Fb was observed in the descending part of the duodenum Fig. . No obvi Even so, the success rate of removal of Fb >10 cm was still very low at their endoscopy center. The removal of the chopstick was performed in 2 steps in the present case: to avoid sticking, the procedure was started with the black circle  to pass through the physiological bending part. The angle between the second and third parts of the duodenum is so quite small and the smooth muscle structure of the duodenum wall results in inferior lateral and longitudinal elasticity, allowing movement only in the form of peristalsis.The common complications of Fb in the GI tract, such as mucosal laceration, bleeding, pyrexia, and perforation, were fortunately not present, which was the most important reason for the 10-year retention of the Fb in this case. The duodenum had an oblique diameter of only 7.3 to 10.4 cm.Imaging examination played a key role in the choice of Fb treatment. Some scholars suggested that for patients with Fb impaction for >24 hours or with an endoscopic indication for more difficult treatment and higher risk, preoperative CT examination needed to be performed. It could not only clearly identify but also assess the risk. In addition, barium examination is also a good choice. In this case, an Fb >10-cm long was found in the second part of the duodenum; the corresponding duodenum was stiff, with no sign of barium leakage. Moreover, the patient showed no obvious signs of peritonitis or melena. Therefore, a diagnosis of perforation was not considered. Endoscopic intervention is the criterion standard technique used for the removal of fb from GI tract, and postoperative complications of surgical treatment included wound infection, pneumonia, and formation of biliary fistula, but in some cases, fb ingestion is followed by life-threatening complications, for which surgical intervention is essential.The risk of an Fb in the digestive tract is related to its shape and size, as the mortality and the risk of perforation increase with the size of these objects, leading to peritonitis, abscess formation, inflammatory mass formation, obstruction, fistulae, and hemorrhage.4To sum up, Fb ingestion should be kept in mind in a patient admitted with abdominal pain. Clinical suspicion and patient history may help to make the diagnosis clear or avoid unnecessary surgery. Moreover, a flexible endoscope is the diagnostic and therapeutic method of choice for fb in the duodenum with strong operability, less trauma, and fewer complications.Conceptualization: Tao Yu, Ya-nan Guo.Data curation: Fang Li, Fu Huang.Formal analysis: Tao Yu.Investigation: Fang Li, Fu Huang.Resources: Tao Yu.Software: Ya-nan Guo.Validation: Tao Yu.Visualization: Tao Yu.Writing – original draft: Ya-nan Guo,Fu Huang, Fang Li, Tao Yu.Writing – review & editing: Ya-nan Guo, Fang Li, Tao Yu.


One of the purposes of echocardiography is to determine the nature of a space-occupying lesion. The conventional transthoracic echocardiogram (TTE) is the preferred method for the diagnosis of cardiac space-occupying lesions as it can reveal the baseline information. For patients with poor conditions, however, TTE cannot clearly display the boundary, it has a limited role in determining the nature of the lesions.A 47-year-old woman presented with intermittent fever for 7 days and chest distress/shortness of breath for 5 days.In our current case, we inferred the nature of space-occupying lesions in the left atrium more accurately using transesophageal echocardiography (TEE) than TTE, which may offer diagnostic evidence for surgical treatment.The patient underwent surgical resection of the left atrial tumor and reconstruction of the left atrial wall. However, the patient's posterior lobe of the mitral valve was infiltrated by tumor, which was difficult to completely remove.Echocardiography was performed 3 months after surgery and the tumor recurred in the posterior lobe of the mitral valve. Although almost all tumors have been removed by surgery, the average survival time is often less than 1 year, as it is difficult to completely remove and easy to relapse with poor prognosis.Transesophageal echocardiography (TEE) plays a relatively more important role in the determination and differential diagnosis of cardiac space-occupying lesions The conventional transthoracic TTE is the preferred method for the diagnosis of cardiac space-occupying lesions as it can reveal the baseline information, such as location, size and morphology. For patients with poor conditions, however, TTE cannot clearly display the boundary, root and pedicles of the tumor and its relationships with surrounding tissues; in addition, it has a limited role in determining the nature of the lesions. Transesophageal echocardiography(TEE) can provide clearer images and clarify the nature of space-occupying lesions, which has a great advantage over TTE.,3 In our current case, we inferred the nature of space-occupying lesions more accurately using TEE than TTE, which may offer diagnostic evidence for surgical treatment.Space-occupying lesions of the heart mainly include benign and malignant tumors, thrombus and abscesses, although hematomas have also been reported. Surgical resection is the mainstay treatment for benign tumors, whereas surgery, chemotherapy, and/or radiotherapy can be chosen for the treatment of malignant tumors depending on their pathological type and disease status.22.1A 47-year-old woman presented with intermittent fever for 7 days and shortness of breath for 5 days.2.2She was generally healthy but had a history of hypertension for 20 years. She denied any history of diabetes, hepatitis, or infectious disease . She had no history of allergy, blood transfusion, trauma, or surgery. The relevant physical examination (PE) results showed no abnormality.33.19/L; neutrophil ratio, 84.3%; NT-proBNP’, 1284 pg/ml (reference range: 0–125 pg/ml); procalcitonin (PCT), 0.07 ng/ml (reference range: 0–0.05 ng/ml); tumor marker CA-125, 173.8 U/ml (reference range: 0–49 U/ml); and neuron-specific enolase (NSE), 27.45 ng/ml (reference range: 0–16 ng/ml).D-dimer was 2.36 mg/L (reference range: 0–0.55 mg/L); white blood cell count, 12.3 × 103.2Routine TTE revealed 2 dense-echo masses  in the left atrium. The masses showed irregular shapes, and markedly heterogeneous internal echoes. The masses had poor mobility and were tightly attached to the posterior mitral valve, accompanied by pericardial effusion  of the tumor. However, echocardiography is a subjective technique and is highly dependent on the experience of the ultrasonographist.Primary cardiac malignant tumors are rare, with an incidence of only 0.001% to 0.28%.,9 However, TTE often fails to make a correct diagnosis for cardiac tumors if the patient has poor body shape and inappropriate position or if the lesion is behind a rib. In our center, we once misdiagnosed a mass in the right atrium of an elderly patient as a myxoma by using TTE; later, the patient underwent TEE, which not only showed a lesion in the right atrium but also revealed multiple similar hyper-dense masses at the right atrial appendage wall and the superior vena cava entrance via multi-angle scanning. The final diagnosis by TEE was thrombosis. The lesion markedly shrank after thrombolytic therapy.Currently, cardiac space-occupying lesions are usually discovered first by TTE, which can distinguish thrombosis from tumors. For tumors with typical ultrasound characteristics, two-dimensional ultrasound is sufficient to make a diagnosis.Compared with the conventional TTE, TEE has the following advantages:1.TEE can more clearly display small structures such as tumor pedicles and small thrombi, which enables a definite diagnosis in combination with other imaging features and medical histories.2.As the TEE probe is closely attached to the left atrium with a higher frequency, TEE can clearly show the boundary between the space-occupying lesion and the surrounding tissues. A clear boundary between a lesion and its surrounding tissue is an important factor for distinguishing benign tumors from malignant ones.3.,10 In a few patients, the crista terminalis protrudes from the cardiac cavity, which may be easily misdiagnosed as a space-occupying lesion on TTE; in contrast, TEE can reveal the connection of the base of this structure with the pectinate muscle of the right atrium, thus ruling out the diagnosis of a space-occupying lesion.TEE is helpful in differentiating normal from abnormal structures. For example, located in front of the entrance of the superior and inferior vena cava, the crista terminalis of the right atrium is a fibrous muscular bridge that separates the primitive atrium from the sinus venarum cavarum. It has different morphologies depending on the degree of degradation.4. These advantages enable TEE to make relatively more accurate diagnosis of the space-occupying lesions with typical sonographic changes.Compared with TTE, TEE can display the internal echoes of the tumor more clearly due to the high frequency and high resolution of its probe.In our current case, the nature of the mass was more accurately identified by TEE, which provided the surgeon with adequate preoperative information and assisted in formulating a reasonable surgical plan. Surgical pathology also showed that the mass had a poorly-demarcated boundary with its surrounding tissues; in particular, the tumor had infiltrated the posterior leaflet of the mitral valve and was difficult to be completely removed. Literature has shown that although surgical resection is the primary treatment for spindle cell sarcomas of the left atrium, these tumors are difficult to be completely removed and can easily recur. The prognosis is poor, with an average survival of less than 1 year.In summary, although the incidence of cardiac malignancies is low, more accurate diagnosis and timely treatment are of great clinical importance. The TEE probe is placed very close to the left atrium and not affected by unfavorable factors such as ribs, thoracic gas, and obesity. In addition, as the TEE probe has a higher frequency than the TTE probe, it can more clearly display the tumor pedicle, small thrombus, thickened crista terminalis, and other structures that TTE does not easily show clearly. TEE can also demarcate the boundary between a space-occupying lesion and its adjacent tissues more clearly. Therefore, TEE is relative more superior over TTE in terms of diagnostic accuracy and can assist in formulating a surgical plan, and has higher value for diagnosis of malignant tumors.Sun YY, Chen X and Jing B acquired case information; Su ML designed research; Wang XY, Wu Y and Song Y acquired imaging examination data; and Sun YY and Zhang GM wrote the paper.Conceptualization: Yuan-Yuan Sun, Guo-Ming Zhang, Xu Chen, Maolong Su.Data curation: Yu Song, Maolong Su.Formal analysis: Xin-yu Wang, Guo-Ming Zhang, Yuan Wu, Maolong Su.Funding acquisition: Guo-Ming Zhang.Investigation: Xin-yu Wang, Guo-Ming Zhang, Bo Jing, Maolong Su.Methodology: Yuan-Yuan Sun, Xin-yu Wang, Xu Chen, Maolong Su.Resources: Yuan-Yuan Sun, Xin-yu Wang, Bo Jing, Yuan Wu, Maolong Su.Software: Guo-Ming Zhang, Yu Song.Validation: Xin-yu Wang, Xu Chen, Maolong Su.Visualization: Guo-Ming Zhang, Maolong Su.Writing – original draft: Yuan-Yuan Sun, Guo-Ming Zhang.Writing – review & editing: Yuan-Yuan Sun, Guo-Ming Zhang, Maolong Su.


Upper gastrointestinal bleeding (UGIB) in patients suffering from hepatocellular carcinoma (HCC) is usually due to oesophageal or gastric varices secondary to portal hypertension. Very rarely, HCC can directly invade into the stomach or duodenum resulting in UGIB.A 62-year-old man presented to the emergency department for haematemesis and tarry stool. He was a hepatitis B carrier and had received open radiofrequency ablation and wedge resection for HCC previously. Urgent endoscopy and contrast computed tomography confirmed recurrent HCC invading into the duodenum. The patient received radical resection and remained disease free for 7 years after the operation.Direct invasion into gastrointestinal tract by HCC is rare. Different modalities of treatment have been reported in the literature with variable success. En-bloc resection should be considered if surgically feasible in order to achieve good haemostasis and possible long term survival.Upper gastrointestinal bleeding is a rare presentation of hepatocellular carcinoma and long term survival can be achieved by curative surgery. Upper gastrointestinal bleeding (UGIB) in these patients are usually due to oesophageal or gastric varices secondary to portal hypertension. Very rarely, HCC can directly invade into the stomach or duodenum causing UGIB. Here we report a case of a recurrent HCC presented with haematemesis and tarry stool. The patient received radical resection of the tumour and remained disease free 7 years after the operation. The work has been reported in line with the SCARE criteria .2A 62-year-old man who was known to be a hepatitis B carrier and had a history of treated HCC presented as emergency with hematemesis and tarry stool. For the history of HCC, he has received open radiofrequency ablation and cholecystectomy for a segment V tumour seven years prior to the current presentation. Lamivudine was started post operatively after assessment by hepatologist and seroconversion of HBeAg was achieved one year later. Open wedge resection for two small recurrent tumours at segment VI was performed two years later. For the current admission, urgent upper endoscopy revealed a 1 cm irregular ulcer at posterior wall of first part of duodenum with adherent clot . AdrenalPatient was offered laparotomy for resection of recurrent HCC. During operation, a 2 cm tumour was found arising from inferior part of segment V of liver directly invading into first and second part of duodenum. Wedge resection of the tumour together with a cuff of involved duodenum was performed . The duo3HCC is well known to be a vascular tumour and has the tendency to invade to adjacent vascular structures like the portal vein, the hepatic vein or even the inferior vena cava . Direct UGIB due to direct HCC invasion of the gastrointestinal tract can be difficult to manage as the involved mucosa have malignant infiltration. Usual endoscopic haemostatic measures can provide temporary effect but rebleeding is likely. Successful endoscopic treatment with ethanol injection has been reported . People The predisposing factors for gastrointestinal tract involvement were large liver lesions >5 cm), subcapsular location and exophytic growth pattern [ cm, subcSuccessful resection combined with gastrectomy or duodenectomy have been reported in the literature ,12. Surg4Upper gastrointestinal bleeding is a rare presentation of hepatocellular carcinoma and long term survival can be achieved by curative surgery.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.All authors report no conflicts of interest.All authors report no source of funding in this study.The submitted study is not a research study.Written consent has been obtained from the patient.Study concept and design: Liu, Lee.Data acquisition: Liu, Lo, Lee.Data analysis and interpretation: N/A.Drafting of the manuscript: Liu, Lee.Critical revision of the manuscript for important intellectual content: Lo, Chong, Lai.Statistical analysis: N/A.Obtained funding: N/A.Administrative, technical, or material support: N/AStudy supervision: Lee, Chong, Lai.The submitted study is not a research study.Liu.Not commissioned, externally peer-reviewed.


Duplex ureter, an embryological developmental anomaly, can lead to intra-operative injuries, even by surgeons with a stronghold on normal ureteric anatomy. We describe the first case of an ectopic ureter transected during vaginal hysterectomy performed for pelvic organ proplase, due to its abnormally low implantation into the bladder, worsened by cystocoele. The injury was recognised, and the duplex ureter was diagnosed with cystoscopy and retrograde pyelography. A post-operative computed tomography urogram allowed us to map the exact course. In this case, there was injury to the ectopic, non-functional ureter, thus averting any further intervention. However, lower urinary tract injuries are serious complications with high morbidity, especially during delayed diagnosis. Knowledge of the ureter variants, meticulous tracing of the course, and use of post-operative cystoscopy could reduce these complications, particularly in face of increasing minimally invasive approaches. Iatrogenic ureteric injury is a significant intra-operative concern for gynaecologists, owing to its proximity to the uterine arteries and cervix, retroperitoneal pelvic course, and at the infundibulopelvic ligament . The oveA 65-year-old woman, post-menopausal since 20 years, had the complaint of a mass descending through the vagina since the past 30 years, which had gradually worsened with time. She also complained of stress urinary incontinence (SUI) and increased frequency of micturition since three years and had taken multiple courses of antibiotic therapy. The mass required manual reduction and splinting during micturition. She had no history of vaginal discharge or post-menopausal bleeding. She had five term vaginal deliveries which were uneventful. She had no history of chronic cough, constipation, or heavy weight lifting.On examination, she was found to have third degree cervical descent, 3+ cystocoele, 1+ rectocoele, no enterocoele, and a keratinised area of congestion over the posterior lip of the cervix, POPQ (pelvic organ prolapse quantification) stage III with cervix as the leading point [Vaginal hysterectomy with pelvic floor repair was planned. The procedure began uneventfully, and the hysterectomy specimen was successfully removed. It was at the initiation of anterior colporrhaphy (cystocoele repair) that a steady drainage of purulent material was observed through the open vault, the origin of which was found to be a cylindrical structure at the vault margin. Upon passing a paediatric Foley’s catheter through it, 20 mL of pus was drained.Cystoscopy and retrograde pyelography were performed to delineate and confirm the suspected ureteric transection. On cystoscopy, the right ureteric opening was seen normally, with normal efflux. Two left ureteric openings were seen. A guide wire inserted through the inferior opening just proximal to the external sphincter seemed to pass into the pelvic cavity and not the kidney, and no efflux was visible from this orifice Figure .Bladder capacity appeared normal. Retrograde pyelography was done from the cut end of the ureter which delineated an apparently blind proximal end, with no contrast in the pelvicalyceal system Figure .A ureteric access catheter was placed in the cut end of the presumed left ectopic ureter and fixed. The vault was closed; anterior colporrhaphy with Kelly’s stitch and posterior colpoperineorrhaphy were completed. The patient had an uneventful post-operative period. A contrast computed tomography (CT) urogram was obtained on the second post-operative day to confirm and map the ureteric system Figure .We concluded that the upper kidney pole and the ectopic ureter (which was transected) were rudimentary and no active intervention was necessary Figure .The patient was discharged with urethral catheter on post-operative day 4 to promote epithelialisation of the ectopic ureter which was inserting into the bladder. She was doing well on follow-up at two weeks, and the catheter was removed. There were no residual or new urinary symptoms. This case demonstrates the potential risk of iatrogenic injury to anomalously located ureters. While we identified the injury intra-operatively and happened to transect the non-functioning ureter, there is always the possibility of damaging the functional ureter and delay in diagnosis. The need for laparotomy was averted in our case as we correctly assumed the rudimentary status of the cut ureter; nevertheless, it is often required. The presence of pyoureter, which led to recognition of injury, may have been due to vesicoureteric reflux and chronic infection, which was worsened by pelvic organ prolapse. The transection of this blind-ending ureter may theoretically reduce the subsequent incidence of urinary tract infection (UTI).Duplication of ureter is one of the most common renal developmental anomalies, occurring in one in 500 people, and may be complete or incomplete . On the This case is, to the best of our knowledge, the first to describe an intra-operatively recognised injury to an ectopic ureter during vaginal hysterectomy. Dalzell et al. had described a case of duplex ureter damaged during laparoscopic hysterectomy which lead to ureterovaginal fistula six weeks after surgery . NotablyDuplicated ureteric systems may cause unanticipated intra-operative injuries. Intra-operative cystoscopy is an excellent way to diagnose lower urinary tract injury, whether used selectively or universally. The mapping of a complicated ureteric system may require multiple diagnostic modalities like cystoscopy, retrograde pyelography, and CT urogram.


Abdominal wall endometriosis (AWE) arising from a surgical scar of cesarean delivery or other abdominopelvic operation, is a rare event with an incidence of 0.03 to 2% , 2. MaliWe herein report a case of a 43-year-old woman with clear cell adenocarcinoma arising from malignant transformation of AWE 19 years after cesarean section. She was initially hospitalized for abdominal wall mass 3 months ago. The 10 × 5 cm of mass was palpated in the right lower abdomen. The mass was firm, tenacious, indolent to palpation, and had poor mobility. Abdominal needle biopsy was performed, and the histopathological results confirmed adenocarcinoma. Cancer antigen (CA) 125 level before the surgery was 26.20 IU/ml.The patient was then sent for whole-body Fluorine-18 FDG positron emission tomography magnetic resonance (18F-FDG PET/MR) assessment for preoperative staging. A hybrid 3.0 T PET/MR scanner  was used. Default clinical MRI sequences including T1w and T2w were scanned. 18F-FDG PET/MR imaging revealed a 9.4 × 4.6 × 4.8 cm solid, heterogenic mass in the abdominal wall on its exterior part, partially involving the right rectus abdominal muscle, with significantly increased FDG uptake (SUVmax = 9.61). Several enlarged retroperitoneal lymph nodes and left inguinal lymph nodes with high FDG uptake were also found in PET/MR-fused images (SUVmax = 4.25). There was no abnormal 18F-FDG uptake in the uterus and bilateral ovaries.An extensive resection with bilateral appendectomy of uterus, pelvic lymph node cleaning, and reconstruction of the abdominal wall was performed. During the operation, a mass of 10 × 5 × 5 cm was found in the lower right rectus abdominis and between the peritoneum. The mass was firmly attached to the rectus abdominis, and it was difficult to separate it from healthy tissue. Multiple enlargement and fused lymph nodes were found in the bilateral iliac blood vessels and around the retroperitoneum. No obvious mass was found in the uterus and bilateral ovaries, and no effusion/infiltration was found in the abdominal and pelvic cavity.Histopathological diagnosis was a stage IIIc clear cell adenocarcinoma originated in ectopic endometrial tissue, and 51 lymph nodes out of 69 ones was positive. Chemotherapy with taxol and carboplatin was then initiated.


Myxofibrosarcoma (MFS) is a soft tissue sarcoma that commonly occurs in late adult life. It is mainly located in the subcutaneous soft tissues of extremities characterized by a high recurrence rate at the original site. MFS of the head and neck is rare, while it occurs in the maxilla and mandible is extremely rare.We report a case of MFS of the mandible in a 51-year-old female who presented with a painless gingival swelling and mobile, super-erupted right mandibular second and third molars. Panoramic x-ray and maxillofacial CT revealed an ill-defined radiolucent lesion surrounding the mandibular molars giving a teeth-floating-in-air appearance. Histopathological examination showed scattered spindle and stellate cells with mild atypia distributed in the myxoid stroma. Only a few mitotic figures were identified and no area of tissue necrosis was found. The characteristic thin-walled and curvilinear vasculature were prominent. Immunohistochemistry analysis revealed the tumor cells being positive for vimentin and vascular CD31. CK, S-100, P63, HHF-35 stains were negative. The labeling index of Ki-67 was about 30%. Based on the histopathological and immunohistochemical examinations, the diagnosis of a low-grade MFS was established. This patient underwent a radical segmental excision with a 2-cm margin, supraomohyoid neck dissection and immediate reconstruction of the mandibular continuity defect with a fibular osteocutaneous free flap. This patient has been followed for 20 months to date and has remained disease free.This report describes a rare case of MFS of the mandible. Recognizing the histopathological features of MFS and applying the appropriate immunohistochemical examinations are crucial in establishing the correct diagnosis. Our case may provide diagnosis and treatment experiences of MFS occurs in the mandible. Myxofibrosarcoma (MFS) is a fibroblast-derived sarcoma, which accounts for approximately 5–10% of all soft tissue malignant tumors . The meaThis is a 51-year-old female who presented with a three-month history of an enlarging gingival mass of the right mandible. Head and neck evaluation revealed no facial asymmetry, cervical lymphadenopathy nor trismus. She denied pain, sensory alterations or bleeding associated with the lesion. The right mandibular second and third molars were super-erupted, grossly mobile, but not painful to palpation. The exophytic gingival lesion was covered with a yellowish pseudomembrane Fig. a, b.FigOn admission, imaging studies including panoramic radiograph and maxillofacial computed tomography (CT) without contrast were obtained. An ill-defined radiolucent lesion involving the right mandibular angle was noted. The lesion involved the entire bucco-lingual width of the right mandibular angle with extension through the lingual cortex. The right mandibular second and third molars were both super-erupted with a teeth-floating-in-air appearance  was first reported in the literature in 1951 . In 1996As a soft tissue sarcoma, MFS mainly occurs in the dermis or subcutaneous tissue, and occasionally occurs in the subfascial or intramuscular tissue . The incLike most soft tissue sarcomas, the pathogenesis of MFS is still unclear. It is suspected that multiple factors contribute to the development of MFS. Current research mainly focuses on the genetic characteristics of soft tissue sarcomas. It is found that MFS has a highly complex karyotype and shows gains and losses of numerous chromosomes or chromosome regions. Therefore, it is speculated that the pathogenesis of MFS may be related to chromosome abnormalities .The clinical courses of MFS vary significantly and no specific clinical features can be identified. It often presents as a painless, slow-growing tumor. Low-grade MFS often demonstrates expansive growth, while high-grade MFS often shows local invasion or compression of the surrounding anatomical structures. High-grade MFS involving the airway or the major vasculature of the head and neck can lead to life-threatening complications.The tumor in this case caused aggressive destruction of the right mandible, but without significant cortical expansion leading to facial asymmetry. The patient did not experience sensory alteration with the tumor involving the mandibular canal. The predominant symptom was the local swelling of the overlying gingiva. The referring dentist initially suspected inflammatory lesion of the oral mucosa, so radiographic examinations were not performed. The pathology report of the initial gingival biopsy suggested pyogenic granuloma. It wasn’t until the patient presented to our department  for treatment, the necessary diagnostic radiographic examinations were performed. The radiographic findings prompted us to perform incisional biopsies of the gingival mass and mandibular lesion. The surgical pathology report supported the diagnosis of low-grade MFS of the mandible and chronic suppurative inflammation of the overlying gingiva.MFS is associated with a relatively high rate of local recurrence or distant metastasis. Accurate preoperative diagnosis and proper treatment strategies are critical in managing patients with MFS. Preoperative imaging examinations including ultrasound, CT, and MRI can be used to determine the anatomical extent of the lesion involvement. Histopathologic examinations are considered the gold standard in establishing the diagnosis of MFS.MFS is more likely to arise superficially in the subcutaneous tissue or dermis, than in deep soft tissues, such as the sub-fascial or intramuscular tissues. On gross examination, the superficial MFS tend to present as multinodular lesions along the epidermis or dermis and demonstrate variability in the gelatinous cut surfaces. The deep tissue MFS, on the other hand, often presents as a solitary mass with a grayish, fish-meat appearance with infiltrative borders.Although the microscopic morphologies of MFS vary, there are some common histopathological features of MFS. These commonalities include varying degrees of myxoid  matrixes composed of spindle to stellate cells that demonstrate nuclear atypia and pleomorphism, and curvilinear vasculature. According to the spectrum of cellularity, cytologic atypia, and mitotic activity, MFS is categorized into low, intermediate and high-grade.In low-grade MFS, the tumor shows characteristics such as hypocellularity and prominent myxoid stroma on histology. Tumor cells are scattered and non-cohesive. They often present as plump spindle or stellate shaped cells with ill-defined and slightly eosinophilic cytoplasm and hyperchromatic nuclei arranged disorderly or in bundles. Mitotic figures are uncommon and tumor necrosis cannot be seen. In some cases, hyperchromatic multi-nuclear tumor cells can be seen. Occasionally, vacuolar and acidic mucous-filled pseudolipoblasts can also be seen, which are positive on AB-PAS stain. The essence of pseudoadipocyte is fibroblast, and there are no fat droplets in the cytoplasm. The characteristic thin-walled and curvilinear vasculature are prominent. In a few cases, the vasculature is clustered or branched. A variable number of inflammatory cells and tumor cells can be seen around the blood vessels , 28.High-grade MFS is either partially or predominantly composed of solid sheets and cellular fascicles of spindle and pleomorphic tumor cells with marked nuclear and cytologic pleomorphism. Bizarre and multinucleated tumor giant cells with abundant eosinophilic cytoplasm and irregularly shaped nuclei are common. Mitotic figures, atypical cells, hemorrhage and coagulative necrosis are also common. Features such as prominent myxoid matrix and numerous curvilinear vessels can be found focally. Mucous degeneration and rich, slender capillary networks can be seen in the tumor stroma , 28.The histopathological features of intermediate-grade MFS are between the low and high-grade MFS. Intermediate-grade MFS is more cellular and pleomorphic than the low-graded tumors. It lacks the extensive solid/sheet-like areas, pronounced cellular pleomorphism, and necrosis found in high-grade MFS. Mitoses are more commonly found than in low-grade tumors. Curvilinear vasculature is often prominent , 28.In immunohistochemistry, the tumor cells of MFS are positive for vimentin. MSA and α-SMA can be focally positive, which indicate myofibroblast differentiation. CD31 is positive in tumor vasculature. S-100, desmin, caldesmon, keratin and histocyte markers such as CD68, Mac387, XIIIa are negative. Ki-67 is partially positive. Ki-67 has a positive correlation with tumor recurrence and can be used as an indicator of recurrence , 28.Based on the clinical and radiographic findings described in this case report, the differential diagnoses include low-grade malignant fibromyxoid sarcoma, myxoid liposarcoma, myxoma, Nodular fasciitis, Spindle cell lipoma, and nerve sheath myxoma , 28. HisAlthough there are some variations in management of MFS, the primary treatment approach is radical surgical resection including a wide margin of adjacent disease-free tissue. Preoperative maxillofacial CT is necessary to determine the anatomical extent of the tumor and involvement of the surrounding tissue. It is generally believed that resection of at least 2 cm around the tumor is needed to ensure a negative surgical margin and reduce the chance of local recurrence and distant metastasis . For theIn this case report, this patient underwent radical hard and soft tissue resection with right hemimandibulectomy, disarticulation of the right mandibular condyle. The large mandibular defect was reconstructed with a fibular osteocutaneous free flap. Due to the consideration that MFS can metastasize to the adjacent cervical lymph nodes, supraomohyoid neck dissection was performed on the right side. The final surgical pathology report showed no cervical lymph node involvement, postoperative radiotherapy and chemotherapy were not prescribed.Based on the current literature, the local recurrence rate for MFS does not correlate well with its histopathological grade. Most cases of MFS are low-grade lesions, and the short-term prognosis of this subtype is generally good. The local recurrence rate for a low-grade MFS is similar to that of a high-grade MFS. Low-grade MFS, however, can transform into a high-grade MFS after recurrence . The locDistant metastasis is rarely seen in low-grade MFS. The rate of metastasis can be as high as 20 to 35% in intermediate and high-grade MFS. Distant metastases usually occur in the lungs and bones. The 5-year overall survival rate for MFS is between 61 and 77% . MetastaIn conclusion, besides odontogenic tumors, soft tissue malignancies such as MFS can occur in the mandible and cause the radiographic findings described in this case report. Recognizing the histopathological features of MFS and applying the appropriate immunohistochemical examinations are crucial in establishing the correct diagnosis. Wide Surgical excision with at least 2-cm margin of adjacent normal tissue and adjuvant postoperative radiochemotherapy are the primary treatment recommendations in managing patients with MFS. Long-term follow up for tumor surveillance is advisable in all cases. Many clinical questions such as the pathogenesis of the disease, tumor origin, efficacy of the adjuvant chemoradiotherapy remain unanswered. Further studies are necessary in improving our understanding of this malignancy which may help in providing better outcomes for our patients.


Choriocarcinoma is an aggressive malignancy of trophoblastic tissue, typically of gestational etiology. Sporadic, nongestational cases are rarely found outside of the gonads. There are only 31 cases of primary choriocarcinoma of the colon reported in the literature. As a consequence of their rarity and aggressive nature, timely diagnosis and effective treatment have proved challenging, and prognosis is very poor. For that reason, we present a rare case with prolonged survival in the youngest reported patient .A 26-year-old Caucasian woman presented with abdominal cramping and rectal and vaginal bleeding. Elevated serum human chorionic gonadotropin and an 8-cm right-sided mass seen on ultrasound suggested ectopic pregnancy. The patient was treated with methotrexate; however, her symptoms persisted, and her human chorionic gonadotropin levels continued to rise. Further workup showed a large mass of the sigmoid colon with multiple hepatic lesions suggestive of metastases. Preliminary pathology showed adenocarcinoma. Despite surgical resection and initiation of FOLFOX chemotherapy , the patient had significant clinical deterioration, and her human chorionic gonadotropin increased exponentially. Further pathological review showed two distinct phenotypes: adenocarcinoma merging with choriocarcinoma. The result of evaluation of the metastatic lesions was also positive for choriocarcinoma. Treatment was promptly changed to a choriocarcinoma-targeting chemotherapy regimen of EMA/CO , resulting in rapid and dramatic response. The patient had mild progression after 1 year and was switched back to FOLFOX with bevacizumab. After five cycles, scans showed further progression, and the patient was started on third-line therapy with FOLFIRI  and bevacizumab. Eighteen months after her diagnosis, the patient was alive and maintaining an overall response.Our patient achieved a marked response and prolonged survival. Although a comprehensive review of the literature showed that survival with these tumors has improved over the past 10 years, prognosis remains poor. Currently, there is no established algorithm for the management of these rare tumors, but both the literature and our patient’s case indicate that a choriocarcinoma-targeted regimen is critical for survival. Further evaluation of these rare tumors is warranted in order to identify pathological patterns that may help in the diagnosis, management, and survival of these malignancies. Nongestational choriocarcinoma is a rare malignancy of trophoblastic tissue. These tumors most often arise in the uterus or gonads, but they have also been observed to arise from extragenital sites, typically in midline structures such as the retroperitoneum, mediastinum, and pineal gland. Even rarer is their occurrence in parenchymal organs such as the liver, lungs, or gastrointestinal tract. Within the gastrointestinal tract, these malignancies are most commonly found in the stomach. Extragonadal, nongestational choriocarcinomas of the bowel are extremely rare, with only 31 cases reported in literature , –31, 1–31n = 23); yet, 76% of metastases with reported histology were solely choriocarcinoma. In our patient’s case, there were features of choriocarcinoma and adenocarcinoma in both the primary and metastatic lesions. However, it was noted by pathology that the cellular architecture became poorer and less differentiated as the tumor extended and spread, with the metastatic liver lesion more closely resembling choriocarcinoma than the adenocarcinoma of its primary source. This histological pattern is consistent with other reported cases and congruent with Pick’s [in situ. In cases where there is solely choriocarcinoma without any adenomatous component identified in the primary lesion, it is believed that trophoblastic metaplasia has occurred to the extent that the more aggressive choriocarcinoma has essentially overtaken the adenomatous tumor, resulting in complete tumor dedifferentiation [Of the cases reported, primary colon lesions were predominantly biphasic tumors, comprised of adenocarcinoma and choriocarcinoma , or BEP  have most commonly been used. The literature suggests that the presence of choriocarcinoma has the greatest impact on prognosis, even more than delay in diagnosis, metastatic disease burden, or the coexistence of adenocarcinoma . Review However, it is important to recognize the heterogeneity of these tumors. Although the presence of choriocarcinoma is observed to be the predominant factor influencing prognosis, these tumors are noted to be more aggressive than traditional gestational trophoblastic neoplasias . In our Our patient was a woman with primary choriocarcinoma of the sigmoid colon. She is the youngest reported patient with this malignancy and achieved longer-than-average survival secondary to radical surgical resection, a single dose of FOLFOX, prolonged EMA/CO therapy, and multiple cycles of FOLFOX with bevacizumab, followed by FOLFIRI with bevacizumab. The patient is maintaining her overall response and remained alive 18 months after her initial diagnosis.Colorectal choriocarcinoma is an extremely rare malignancy with a very poor prognosis. It disproportionately affects younger patients and has no established standard treatment. Given that timely diagnosis has proved challenging and its aggressive course, one should maintain a high index of suspicion in younger patients presenting with symptoms of colon malignancy or in patients with elevated β-hCG for whom gestation cannot be confirmed. Further investigation of molecular, genetic, and immunohistochemical characteristics of these tumors is encouraged because identification may lead to more timely and accurate diagnoses, development of a standard treatment regimen, and prolonged survival in this unique group of patients.


Here, we report a case of haemangioma on middle mediastinum accompanied by unilateral pleural effusion, which was initially suspected to be lung cancer and pleurisy. During annual check‐up, chest radiography of a 30‐year‐old female showed homogeneous opacity in the left lower pulmonary field. Excision was performed, and the mass was pathologically diagnosed as benign mediastinal vascular tumour with exudative pleural effusion. To our knowledge, this presentation occurs in <0.5% of tumours of the mediastinum, and furthermore, the presence of pleural fluid is extremely rare, and the underlying mechanism is unknown. Although mediastinal haemangioma is hard to diagnose without surgery, we should include it in the differential diagnosis of a tumour with unilateral pleural effusion. Here, we report a case of haemangioma on middle mediastinum accompanied by unilateral pleural effusion, which was initially suspected to be lung cancer and pleurisy. Although mediastinal haemangioma is hard to diagnose without surgery, we should include it in the differential diagnosis of a tumour with unilateral pleural effusion. Mediastinal haemangioma is a very rare neoplasm, present in <0.5% of mediastinal masses. Here, we report a rare case of a mediastinal mass accompanied by unilateral pleural effusion impossible to be diagnosed without excision.A 30‐year‐old non‐smoker female was referred to our outpatient clinic with left‐sided limited pleural effusion on X‐ray radiography, detected at an annual health check‐up. She had a past medical history of endometriosis, ovarian cyst, and fibroadenomas of the left breast, which were all treated two years ago by polypectomy of the uterine cervix and hormone therapy. For the past eight years, she has been travelling abroad once a month for business, from east Asia to Europe, and had been to a coal mine in Indonesia for more than 15 times. She had been followed up since the last eight years, with no definitive diagnosis; moreover, she had no cough, sputum, or breathing difficulty.At our clinic, the patient appeared generally good and conscious, with a body temperature of 37.1°C, blood pressure of 108/70 mmHg, and heart rate of 70 beats/min. Her respiratory rate was 16 cycles/min and oxygen saturation was 96% on room air. Cardiopulmonary examination revealed diminished breath sounds on the left lower lung lobe and dullness on percussion was also noted. All other findings on physical examination were unremarkable. Laboratory tests showed normal blood levels , C‐reactive protein (<0.04 mg/dL), interleukin‐2 receptor (293 U/mL), and tumour markers (carcinoembryonic antigen (CEA): 0.5 ng/mL, stage‐specific embryonic antigen‐1; (SSEA‐1) sialyl Lewis X‐i antigen (SLX): 34.9 U/mL, carbohydrate antigen 19‐9 (CA 19‐9): 7.9 U/mL).Chest radiography showed a homogeneous opacity in the left lower pulmonary field. On high‐resolution computed tomography (HRCT), multiple mass lesions on the medial side of the left lower lobe appeared to be scattered, which are supposed to be pleural in origin. They showed strong and non‐uniform contrast enhancement, with the largest one being 27 × 15 × 33 mm, which appeared to be nourished by the musculophrenic branch of the left internal thoracic artery  was performed and indicated no 18F‐fluorodeoxyglucose (FDG) uptake, and the pleural effusion had increased Fig. . After aPleural effusions are a common medical problem with over 50 recognized causes . MediastAppropriate written informed consent was obtained for publication of this case report and accompanying images.


Whereas the uterus masculinus has been reported to communicate with the urethra in men and horses, no communication between the uterus masculinus and urethra has been identified in dogs.Paraprostatic cysts are large structures that develop between the prostate gland and urinary bladder, usually in older, intact dogs. Their incidence is reported to be 1.1–5.3% in dogs with prostatic disease. The aetiology of paraprostatic cysts is not fully understood, but they are believed to develop from the uterus masculinus was made on the basis of the findings of abdominal ultrasonography and histopathology of tissues obtained during an exploratory laparotomy. In addition, a Sertoli cell tumour affecting both testes was diagnosed following scrotal castration. The cystic uterus masculinus was completely resected, after which the tenesmus and bloody discharge resolved. Thus, cystic uterus masculinus should be considered as a differential diagnosis for a paraprostatic cyst when such a lesion develops as part of the feminising effect of a Sertoli cell tumour.An entire male dog was presented with a bloody discharge from its penis and tenesmus of 5 days’ duration. A diagnosis of cystic uterus masculinus should be considered as a differential diagnosis for tenesmus and penile discharge, and for structures resembling paraprostatic cysts. This case report confirms that a uterus masculinus can communicate with the urethra in dogs, as in other species, and demonstrates endocrine responsiveness, manifesting as epithelial and glandular metaplasia and mucus production, with the potential for subsequent infection.Cystic  They usian tube , 9, 10. ian tube , 5, 6.The treatment of paraprostatic cyst in dogs may involve drainage, resection or marsupialization , 10–12.A 12-year-old entire male Basset Hound presented with a 1 week history of tenesmus, bloody penile discharge and anorexia, and polyuria and polydipsia of 2 days’ duration. General examination revealed mild dehydration and a high rectal temperature (39.5 °C). Digital rectal examination and caudal abdominal palpation revealed an enlarged, symmetrical and painful mass that extended from the cranial hypogastrium to the pelvic cavity, just cranial to the prostate. The prostate was smooth, non-painful and of a normal size. Scrotal palpation revealed diffusely swollen scrotal tissue and vaginal tunics, and small, solid, non-painful testes. There was a mucopurulent discharge from the external orifice of the urethra.Haematology revealed mild non-regenerative anaemia, leucocytosis and neutrophilia with left shift. A serum biochemistry panel yielded results consistent with dehydration: slight increases in alanine aminotransferase , alkaline phosphatase  and hyperglobulinaemia  were noted. Subsequent endocrine evaluation revealed a low serum testosterone concentration  and high concentrations of oestradiol  and progesterone .Escherichia coli.Abdominal ultrasonography revealed a hypoechoic hollow lesion that was 10 cm in length and up to 4 cm wide, which was adjacent to the prostate and extended cranially, beyond the level of the apex of the urinary bladder Fig. a. Cytoloducti deferentia lay lateral to the cyst. The cyst was ligated and fully resected, and the abdominal cavity was closed in routine fashion. The testes were removed via a scrotal approach and then scrotal ablation was performed. Both testes were firm and small (2 cm in diameter and 3.5 cm in length). A 10 day course of oral amoxicillin/clavulanic acid  was then administered and post-operative analgesia was provided using meloxicam .Exploratory laparotomy revealed the presence of a single, large paraprostatic cyst that was located dorsal to the neck of the urinary bladder, arose from the prostate, extended cranially into the abdominal cavity, and had two horn-like blind-ended tubes that were suspended from the genital fold Fig. a–b. The The tenesmus and penile discharge resolved immediately following the surgery and examinations conducted 10, 30, and 60 days later were unremarkable. Ultrasonography performed 60 days after surgery revealed normal hypogastric and pelvic anatomy.uterus masculinus was made . However, the previous studies mainly reported uterus masculinus as an incidental finding during abdominal ultrasonographic examination for other problems [uterus masculinus in a vestigial state, i.e., without a completely developed lumen or communication with the urethra.In addition, this case presented with bilateral Sertoli cell tumour. Similar to other reported cases , 21, 22,problems , 26. Theuterus masculinus in the present case was similar to that described in the previous studies [uterus masculinus hydrometra in a miniature schnauzer and six other dogs reported similar ultrasonographic findings [The ultrasonographic appearance of the  studies , 26, witfindings , 23.uterus masculinus in the present case showed the presence of simple columnar epithelium and glandular structures, which is typical of the epithelial lining of the female uterus. Previous reports of the histopathology of uteri masculini have described various types of epithelial linings, including pseudostratified columnar, cuboidal and squamous epithelium. This variability is likely to be related to the fact that each uterus masculinus developed under different circumstances. The present case also had high circulating concentrations of oestrogen and progesterone, which likely had direct influences on the development of the uterus masculinus. High circulating oestrogen concentrations typically accompany testicular Sertoli cell tumours [uterus masculinus communicated with the urethral lumen and had a comparable bacterial flora to that of pyometra likely explains the development of a bacterial infection in the present case.Histopathology of the  tumours , 27. The tumours . Finallyuterus masculinus, following ligation to close its connection with the prostate and urethra, given its infected content.The preferred surgical therapy for a paraprostatic cyst includes exploratory laparotomy with cyst drainage, debulking and either omentalisation or marsupialisation , 12, 25.The case report described here has similarities with the persistent müllerian duct syndrome which is a rare from of pseudohermaphroditism of dogs which is is characterized by XY chromosomal constitution, testes, and feminization of the internal or external genitalia feminized. Commonly these individuals are cryptorchids . Howeveruterus masculinus as a differential diagnosis for tenesmus and penile discharge and for structures resembling a paraprostatic cyst. Furthermore, we propose the unification of the terminology regarding uterus masculinus, such that the term “true” uterus masculinus should be used only if communication with the urethra is present, and “cystic” uterus masculinus should be used for cases in which there is no communication with the urethra and columnar glandular epithelium is present. In our opinion, the term “cyst” should be reserved only for an enclosed hollow cavity lined with epithelium and containing a liquid or semi-solid substance [In conclusion, the findings presented in this report suggest the importance of considering a ubstance , and shoubstance , 21–23.


Neuroblastoma is the most common childhood malignancy arising from the sympathetic neuroblast cells. The most common sites of origin are the adrenal glands and paravertebral regions. However, the involvement of the heart is a rare occurrence in adolescents. Here, we report a case of a 12-year-old male child who was misdiagnosed as a case of cardiac myxoma on initial presentation. Following surgical resection and histological examination, neuroblastoma was revealed. This case report highlights the differential diagnosis for the cardiac mass in an adolescent with an unknown primary origin and also the importance of tissue histopathology for the diagnosis and management of neuroblastoma. Neuroblastoma (NB), a tumor originating from neural crest cells that are designated for the development of postganglionic sympathetic neurons, encompasses the most common malignancy among extracranial solid childhood tumors. The total burden of neuroblastoma is one in 8,000 live births, which constitutes 6%-10% among all childhood tumors and causing 12-15% mortalities related to malignancies in childhood . By cont2) of 92% while breathing ambient air. The cardiopulmonary examination was unremarkable, with no murmurs or audible extra heart sounds. All other physical examinations and laboratory findings were unremarkable. After initial stabilization, baseline investigations were performed. Laboratory studies were pertinent for normocytic normochromic anemia with hemoglobin of 11 g/dL. Blood culture showed no growth of organisms. All other values within complete blood count and complete metabolic panel were unremarkable.A 12-year-old male presented to the Emergency Department with complaints of shortness of breath associated with a persistent cough, weight loss, and intermittent low-grade fever for the past few months. The patient had no significant past or family medical history. On physical examination, the pale-looking adolescent male had a temperature of 38.4 degree Celsius, blood pressure of 111/59 mmHg, heart rate of 117 beats/minute, respiratory rate of 30 breaths/minute, and oxygen saturation  attached to the lateral free wall of the left atrium, suspicious for a neoplastic process. Figures . The masThe cardiology team was consulted to assess the neoplastic lesion and based on the chest X-ray and echocardiography findings, the mass was suggestive of left atrial myxoma. Cardiovascular surgery was consulted for the resection of the left atrial myxoma. The patient underwent cardiac surgery five days after the initial diagnosis for mass removal. Operative findings revealed that the mass was found to be involving the lower left pulmonary vein and extending inside it. Histopathological report of surgical specimen describes the mass as reddish rubbery tissue measuring 45 mm in greatest diameter with small round blue cell tumor with occasional rosettes, rejecting the hypothesis of a cardiac myxoma and instead suggesting a diagnosis of “neuroblastoma.” Chest, abdominal, and pelvis CT scans did not detect any other mass or lesion. Since extensive imaging failed to show any primary site, a diagnosis of metastatic neuroblastoma with an unknown primary site was concluded.Neuroblastoma is undifferentiated tumors that arise from the neural crest cells designated for the development of postganglionic sympathetic neurons. It is the most prevalent malignancy among extracranial solid childhood tumors. Based on its embryological origin and anatomical locations, it is usually found in the adrenal gland and around sympathetic chains. While the majority of them are located in the abdomen, 50% of them originate from the medulla of the adrenal gland, and the rest may emerge in the thorax (20%), neck (5%), or pelvic region (5%). Also, approximately 1% of cases have an unknown primary site ,4.The median age at diagnosis is around 17.3 months; however, cases have been reported in the pediatric population of older age . PrognosThe initial presentation in young children includes high temperature (25%), abdominal pain (22%), abdominal mass 19%), and bone pain (19%) [%, and boPrimary cardiac tumors are unusual, with an incidence rate of less than 0.1%. The most common primary neoplasm is myxoma in adults, whereas rhabdomyomas occur frequently in children. Myxoma has a predilection for the left atrium, and the initial presentation includes non-specific and variable symptoms such as fever, weight loss, and anorexia. However, secondary tumors are more common than primary tumors, and metastatic dissemination to the heart is still rare. In the presented case, a 12-year-old male child, who presented with constitutional symptoms as the initial presentation with a diagnostic challenge, was misdiagnosed with left atrial myxoma. It is difficult to differentiate this case clinically from other cardiac tumors, but histological findings of small, round, blue cells with occasional rosettes confirmed the diagnosis. Once the diagnosis had been made as neuroblastoma, the next challenge was to determine the origin of the occult tumor using imaging modalities and immunohistochemistry. Our patient’s imaging studies, which included CT, US of the abdomen, and bone scan, did not detect a measurable primary tumor. However, a chest X-ray revealed multiple lesions in the lungs, suggesting the possibility of seeding to the left atrium through a hematogenous route.Regardless, cardiac neuroblastoma is extremely rare and is usually a result of the intracavitary cardiac extension of neuroblastoma, making it vulnerable to be missed . Until nAccording to the International Neuroblastoma Staging System (INSS), cardiac involvement is considered a stage 4 disease . ManagemAs of now, in the literature, there are very limited data regarding metastasis of neuroblastoma to the left atrium with no known primary site, and there is a high probability that it can be neglected as a potential metastatic site by physicians. Analyzing the unusual spread will assist oncologists in understanding the disparities in the clinical behavior of neuroblastoma in older children. A multidisciplinary team of pediatric oncologists, surgeons, and radiation oncologists is important for the most effective outcome for patients.


Necrotizing Sialometaplasia (NS) is a rare, benign, self-limited, inflammatory and necrotizing reaction of the salivary glandular tissue. Due to the clinical picture (a painful ulcer with well-defined edges), and histopathological characteristics  NS can mimic a malignant lesion. The correct diagnosis is important because NS shows a spontaneous resolution and therefore no further treatment is needed. We report a very unusual case of spontaneous and recurrent NS located on the anterior tonsillar pillar in a 43-year old man, which clinically mimics a malignant lesion. The clinician should be aware to include NS in the differential diagnosis of ulcers in soft palate and tonsillar pillars. Key words:Minor salivary gland, necrotizing sialometaplasia, oral ulcer, squamous cell carcinoma, tonsillar pillar. Necrotizing sialometaplasia (NS) is a rare , benign,A very uncommon case of NS due to its location (anterior tonsillar pillar), history of recurrence and not-known predisposing factor, which clinically mimics a malignant lesion, is presented.A forty-three-year-old male was referred by a private physician to a teaching Oral Medicine Clinic, because he had “a wound on the palate that does not heal”. The medical antecedents of the patient were not relevant for the case. At anamnesis, the patient reported that two months ago, he noticed a small ulcer located in the posterior area of the soft palate painful while swallowing. The ulcer grew in a continuous way, decreasing painful symptoms. The patient referred that 2 years ago, he suffered two similar lesions in the same area of the current ulcer, with an interval of appearance within three months approximately, in both times remitted in approximately two weeks. He denied any kind of traumatism or the use of recreative drugs.At intraoral clinical examination, an ulcer of approximately 3.5 cm in diameter located at the anterior tonsillar pillar was observed. The ulcer was irregular in shape with elevated, erythematous, slightly indurated and poorly defined edges. The bottom of the ulcer was white-yellowish in color with fibrin Fig. A. At palMicroscopically, the sample was covered by a stratified squamous epithelium, which showed a loss of continuity compatible with an ulcer. Underlaying the epithelium, salivary gland tissue was observed. The sero-mucous salivary acini retained its lobular architecture; however, the presence of coagulative necrosis was identified Fig. A. A seveAt subsequential 7 day-postsurgical follow-up appointment, the patient showed a noticeable process of healing of the surgical area Fig. B. He didThe present report describes an uncommon case of spontaneous and presumably recurrent tonsillar NS. To our knowledge, in the scientific literature only three cases of NS have been reported at tonsils: two cases at tonsillar pilli, and one case at tonsillar fossa ,5. The pNS is generally associated with a predisposing or triggering factor; however, spontaneous lesions have been reported ,9. In thIt has been reported that NS does not show recurrence . To our Clinically and histopathologically NS can mimic a malignancy, principally oral squamous cell carcinoma and mucoepidermoid carcinoma. Clinically, NS is characterized by a superficial ulcer with or without indurated edges . MalignaOthers clinical differential diagnoses included syphilitic gum , traumatThe present case of NS constitutes an unique case due to its location, absence of predisposing factors and its apparent capacity of recurrence. It also shows that a benign lesion (NS) located in an uncommon site constitutes a diagnostic challenge. Because NS can mimic clinically and histopathologically a malignant neoplasm, the clinician must be alert to the presence of a sudden ulcer in the tonsillar region and perform a meticulous examination to avoid misdiagnosis and unnecessary invasive treatments.


Exserohilum species are among the rare ones, as only a few cases have ever been reported a few times in the literature. The objective of this report is to highlight this unusual fungal type causing unilateral AFS in Saudi Arabia.Allergic fungal sinusitis (AFS) is a result of an inflammatory reaction to fungi in the nasal and paranasal sinuses. Although the causative agents of AFS vary, We present a case of AFS who initially presented at the age of 15 years, and was operated on in 2015 by another health care provider. He presented again in 2019 complaining of intermittent loss of smell and greenish nasal discharge, mainly from the right side for 3 months, which was associated with right-sided nasal obstruction. Based on the history and physical examination, a diagnosis of refractory chronic rhinosinusitis was made. The patient underwent functional endoscopic sinus surgery (FESS) of the paranasal sinuses and polypectomy. Histopathology of the samples taken during the surgery showed Exserohilum specie. The diagnosis of AFS was confirmed using Bent and Kuhn’s criteria.Allergic fungal sinusitis is believed to be an allergic reaction caused by a broad spectrum of species, the majority being Aspergillus in India and Saudi Arabia. Exserohilum species are one of the rare organisms causing AFS in our region.Exserohilum AFS in an immunocompetent pediatric patient.Most cases of AFS in our region have been reported to be caused by Aspergillus. Here we report a case of unilateral  The patient presented to the Otolaryngology clinic at our institution  in 2019, complaining of intermittent smell loss and greenish nasal discharge mainly from the right side with acute intermittent nasal obstruction. Upon physical examination, smell diskettes test showed a result of 3/8 (anosmia), with an intact subjective retronasal smell . Rhinoscopy showed second grade polyps based on Meltzer Clinical Scoring System in the right nasal cavity and a polypoid middle turbinate in the left cavity Exserohilum species. Histopathology also reported the presence of eosinophilic infiltrate at the subepithelial and epithelial surfaces as well as the presence of fungal elements within the eosinophilic mucin .Written informed consent has been obtained from the patient, submitted and approved by the local IRB committee.Arwa Al muslat: First author, writing and editing – original draft, data collection and finalized the manuscript.Basmah Alghmdi: First author, writing and editing – original draft, data collection and finalized the manuscript.Abdullah J. Alshehri: Writing – review, editing and finalized the manuscript for submission.Rakan Alhaidy: Participated in writing the discussion and literature review.Muhammad A. Dabbabo: Contributed at managing the case, revised the manuscript.Naif H. Alotaibi: The primary physician – treating and following up the patient, writing – supervision, critical revision of article and final approval for submission.All authors approved the final version of the manuscript.King Faisal Specialist hospital and Research Center, Research ethics committee. RAC #5166461.Dr. Naif H. Alotaibi MD, Associated professor, College of Medicine, Alfaisal University, Department of Otolaryngology, King Faisal Specialist Hospital and Research Center, Riyadh, Saudi Arabia.Not commissioned, externally peer-reviewed.


Malignant ileocolonic fistulas have seldom been documented as complications of a primary gastrointestinal lymphoma (PGIL) such as aggressive diffuse large B cell lymphoma (DLBCL). These fistulas are frequently misdiagnosed due to the nonspecific clinical presentation. Currently, there is no standardized treatment approach, although a couple have been suggested with varying outcomes. We describe a case of DLBCL complicated with a malignant ileocolonic fistula in a 55-year-old male with a favorable outcome after surgery and chemotherapy. This article was previously presented as a meeting abstract at the Annual American College of Gastroenterology (ACG) 2020 virtual Scientific Meeting on October 23, 2020.Primary gastrointestinal lymphoma (PGIL) is a rare malignancy that proves to be a diagnostic challenge due to the nonspecific clinical presentation and may easily be misdiagnosed as Crohn’s disease or intestinal tuberculosis ,2. DiffuA 55-year-old male presented to the emergency room complaining of three months of watery, non-bloody, non-melanotic diarrhea with generalized abdominal pain for three weeks and nausea with constant gurgling “washing machine” sounds. He denied any vomiting, fever, chills, anorexia, or weight loss. His medical history was significant for hypothyroidism and Hodgkin's lymphoma 25 years ago, which required a staging laparotomy with splenectomy and was treated with mantle field radiation therapy for a cure. The physical exam, including abdominal examination, was unremarkable. Of note, his labs revealed elevated C-reactive protein (CRP) 15.3mg/dL and leukocytosis of 14.3K/uL, but were otherwise within normal limits. Non-contrast CT scan of the abdomen and pelvis showed a focal aneurysmal dilation of a loop of small bowel in the mid-lower abdomen with several enlarged mesenteric lymph nodes, and a fluid-filled sigmoid colon . Doxorubicin was avoided in view of the patient's ventricular ejection fraction (EF) of 40-45%. Repeat PET/CT scan after the sixth chemotherapy cycle showed decreased size of the lymph nodes with resolution of their hypermetabolic state and no acute bowel pathology or metastatic disease. The patient was declared in remission with a satisfying clinical outcome. Malignant ileo-colonic fistula is occasionally associated with adenocarcinoma of the cervix, ovaries and colon ,8. In 18Small bowel lymphoma is reported as the cause of less than one percent of all gastrointestinal malignancies, and in the absence of prior chemotherapy, surgical resection, or recent radiotherapy, the development of fistula is extraordinarily rare . Nine prThe formation of a malignant fistula is a chronic process that occurs over an extended interval . The lymThe nonspecific symptoms of GI fistula delay diagnosis, with the most common presenting symptoms being abdominal pain (84%), weight loss (81%), and diarrhea (39%). Twenty-three percent of patients present with small bowel perforation . The aneAlthough PET/CT is instrumental in detecting PGIL, defining the degree of metastasis, stage, and subsequent treatment response, our patient required a colonoscopy to confirm the ileocolonic fistula suspected on the abdominal CT imaging. There is currently no unified consensus on the optimal treatment strategy for intestinal lymphoma fistulas . Zhuang Ileo-colonic fistula due to intestinal lymphoma is a rare occurrence. Due to nonspecific clinical presentation, a high index of suspicion is required to establish the diagnosis of ileo-colonic fistula. Besides, we recognize colonoscopy's role in confirming the ileo-colonic fistula diagnosis if the diagnosis is not clear on CT imaging. Currently, there is no consensus on a standardized treatment approach, but we recognize the approach of en bloc surgical resection of the tumor followed by standard adjuvant chemotherapy as the best treatment approach to ileo-colonic fistulas. However, we admit that more data is required to demonstrate the most effective approach to PGIL with fistula. We hope this case serves to assist in early detection and guiding treatment to pursue the optimal clinical outcomes for patients with PGIL complicated by fistula.