Gastrointestinal schwannomas are benign, slow-growing and usually asymptomatic tumors. In some cases bleeding, epigastric pain and palpable mass may be occurring. Preoperative diagnosis is challenging due to the difficulty of differentiation from other submucosal tumors. Diagnosis is most often provided through the histology report.In this study we report two cases of gastric tumors with the suspicion of a GIST preoperatively but histologically confirmed to be gastric schwannomas. Two patients of our study gave to us their written consent for publication. Research work has been reported with the PROCESS criteria.Surgical resection should be considered the mainstay of treatment in patients with gastric schwannomas. Possible complications such as bleeding or pyloric stenosis can be presented. The size and location of the tumor, as well as its relation to the surrounding organs, are essential factors in determining the type of resection.Gastric schwannomas are usually presented us submucosal mass. Preoperative diagnosis is challenging due to the difficulty of differentiation from other submucosal tumors. Endoscopic Esophagogastroduodenoscopy with biopsy and endoscopic ultrasound is essential to determine the nature of these lesions. Resection of the lesion in healthy borders is the treatment of choice. Patho-logical examination usually revealed positive S-100 protein and negative CD34, CD117, Actin and desmin strains. Among t254-year old female with a history of hypertension and gallbladder polyp was admitted to our department for the evaluation of a gastric lesion that was detected incidentally during ultrasonography scan of the upper abdomen A. Esopha377-years old female with a history of cholelithiasis presented with epigastric pain persisting for the last six months. CT scanning revealed a homogeneous round mass measuring 5 cm, arising from the posterior wall of the stomach A. Submuc4Schwannomas, also known as neurilemmomas or neurinomas, are benign neurogenic tumors, originating from Schwann cells, which generally form the sheath around the axons of the peripheral nerves. Although schwannomas can develop anywhere along the peripheral course of the nerve, they most commonly occur in the head and neck but rarely in the GI tract, where, they arise from the nerve plexus of the gut wall  ,2. The sSchwannomas account 0.2% of all gastric tumors, 6.3% of gastric mesenchymal tumors, and 4% of all benign tumors of the stomach ,3. Most 5Schwannomas are benign submucosal lesions, originating from Schwan cells. They most commonly occur in the head and neck but rarely they arise from nerve plexus of GI wall (Auerbach and Meissner plexus). Stomach is the most common site followed by the colon. Preoperative diagnosis is challenging due to the difficulty of differentiation from other submucosal tumors. The most common presenting symptom is abdominal pain or discomfort, followed by upper GI bleeding. CT, MRI, and EUS of upper gastrointestinal, were usually without specific findings and the definitive diagnosis of gastric schwannomas is determined on final pathology report. Resection of the lesion in healthy borders is the treatment of choice. Immuno-histochemical studies show universal positivity for S100 protein and frequent negative for CD117, DOG-1, CD34, muscle actin and desmin strains.No conflict of interest.This study is own funding.We have approval from bioethical committee of Aristotle University of Thessaloniki Greece.Written informed consent was obtained from the patient for publications 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.Daniel Paramythiotis for study design.Anestis Karakatsanis writing the paper.Diamantoula Pagkou, Niki Mantha and Sofia Lypiridou data collection.Petros Bangeas writing the paper and data analysis.Antonis Michalopoulos Study concept.researchregistry4205.Daniel Paramythiotis.Not commissioned, externally peer reviewed.


A primordial odontogenic tumor (POT) is a rare, benign, mixed epithelial and mesenchymal odontogenic tumor that has been included as a new entity in the latest World Health Organization (WHO) classification (2017). POT consists of dental papilla-like myxoid connective tissue covered with a delicate membrane of ameloblastic epithelium. Only 15 cases have been documented worldwide, and here, we report the sixteenth case and the first one of South Korea.An asymptomatic lesion was discovered as an incidental radiographic finding in a 10-year-old boy. The patient had no complaints about the lesion. Cone-beam computerized tomograms revealed a round cavity with a defined cortical border measuring approximately 5 × 5 × 5 mm in size. The lesion was a POT. The patient was treated with enucleation. The tumor showed no recurrence for one year.This is the first report of POT in South Korea using the novel diagnosis of POT after it was recognized and defined in the latest WHO classification. This novel diagnosis will be useful for pathologists and clinicians in diagnosing and differentiating this new and rare disease from other odontogenic tumors. A primordial odontogenic tumor (POT) is a new entity classified as a benign, mixed odontogenic tumor in the fourth edition of the World Health Organization (WHO) classification of Head and Neck Tumors in 2017 [To date, most cases of POT were found as well-defined unilocular or multilocular radiolucent lesions adjacent to the crown of an unerupted tooth. Patients showed asymptomatic bone swelling, producing root resorption, and buccal or lingual cortical expansion. Macroscopically, the tumor is a pale, slippery, solid nodule that tends to be encapsulated , 3. HistUntil now, only 15 cases have been documented worldwide . To bettA 10-year-old healthy boy visited the Department of Pediatric Dentistry, Chonnam National University in July 2018 to complete root canal therapy. An asymptomatic lesion was discovered incidentally in a conventional panoramic X-ray. There was no history of trauma to the area and he had no complaints about the lesion. There were no abnormal findings in either the physical examination or laboratory data.Cone-beam computerized tomograms depicted a round cavity with a defined cortical border measuring approximately 5 × 5 × 5 mm in size, mesiolingual to the root of tooth 34 Fig. a. A panoThe diagnostic hypotheses from radiology were simple bone cyst, periapical cemental dysplasia, and paradental cyst because of its location and radiologic features. A tumor enucleation was performed, and a whitish, firm, myxoid connective tissue was transferred for pathological analysis. Based on the histopathological study results, POT was confirmed as a definitive diagnosis. There were no adverse events neither signs of recurrence after surgery during a one-year follow-up.The gross specimen showed a 5 × 5 × 5-mm-sized well-defined and slippery white nodule of pale, translucent, firm, myxoid connective tissue. Histologically, the periphery of the tumor was enveloped by a delicate membrane of ameloblastic epithelium, which is a single layer of columnar epithelium exhibiting typical “reverse nuclear polarization,” i.e., displacement of nuclei away from the basement membrane and vacuolated cytoplasm at the bottom part. Most of the tumor was composed of loose and myxoid fibrous tissue, including spindle cells Fig. a, b. In Upon immunohistochemistry analysis, the epithelial component demonstrated strong positivity for cytokeratin 19 only in columnar cells, which was consistent with previously reported cases. Vimentin was also positive throughout the tumor tissue; specifically, strongly positive in the epithelial layers and moderately positive in mesenchymal tumor cells. In addition, alpha smooth muscle actin (α-SMA) and S100 protein were negative in the ectomesenchymal cells. Ki67 expression was lower than 2% Fig. .Fig. 3HPOT is a new neoplastic entity, classified as a benign, mixed epithelial and mesenchymal odontogenic tumor in the fourth edition of the head and neck WHO blue book in 2017 [Until now, there had been only 15 cases of POT reported in the literature. The previously reported cases share similar clinical and radiological findings, which are shown in Table The present case showed a rare location with a small tumor, whereas clinical, radiological, and pathologic findings are similar to those of the previous reported cases. Interestingly, the location of this tumor was near the root of the tooth, whereas previously reported cases presented as a pericoronal location in close association with unerupted teeth. To verify a relationship between the patient’s dentition stage and the location of the tumor, we summarized the location of all POTs and listed the information in a schematic diagram. We then attempted to classify the location of POT by three types based on the previous literature Fig. : Type A,In histological findings, our case was enveloped by a single layer of columnar epithelium exhibiting typical “reverse nuclear polarization”, which is known as ameloblastic epithelium. In some areas, the cord-like or islands of the enfolded epithelium possessed stellate reticulum between the columnar cells. Calcification was found in the peripheral portion of the myxoid connective tissue, which represents an association between the tumor and adjacent tooth. The pathological characteristics in our case are conclusive enough to make a diagnosis , 8.According to Mosqueda-Taylor et al. , who firAmong the 16 cases (including this case), only 11 cases have been analyzed immunohistochemically Table . SpecifiWe herein report the first case of POT in South Korea since it was newly categorized in the latest WHO classification in 2017. As there are only 16 documented cases, including this report, in English literature worldwide, more clinical, pathological, and radiographical information is needed to further understand the disease entity. The size of the POT reported in our article is smaller than previously reported cases. In most of other previously reported POT cases, the tumor showed an apparent pericoronal position with unerupted tooth , 8, 11.


The congenital simple hamartoma of the retinal pigment epithelium is a benign lesion and previous observations with noninvasive imaging have detected potential photoreceptor abnormalities and retinal function interplay.A 35-year-old woman was found to have an asymptomatic, solitary, circumscribed, pigmented lesion in her left eye. The patient underwent ophthalmic examination including multimodal evaluation with fluorescein angiography, near-infrared reflectance scanning laser ophthalmoscopy, blue autofluorescence, enhanced-depth imaging spectralis B-scan optical coherence tomography (EDI-SBOCT), en face OCT angiography (OCT-A) and microperimetry plus adaptive optics imaging. Ophthalmoscopic examination revealed a juxtafoveolar pigmented lesion with feeding retinal arteriole, consistent with congenital simple hamartoma of RPE. There was no macular edema, exudation, hemorrhage, traction or subretinal fluid. Multimodal imaging of the mass using fluorescein angiography revealed intra-lesion late staining, near-infrared reflectance imaging demonstrated intrinsic hyperreflectivity, short-wavelength autofluorescence and red-free filter photography revealed blocked signal, and SBOCT showed abrupt shadowing. On OCT-A, an exclusive ring-shaped vascular circuit with increased foveal avascular zone was noted. Adaptive optics revealed cell density arrangement and retinal sensitivity correlations on microperimetry.These findings suggest that this hamartomatous lesion might cause specific cellular changes that impact retinal sensitivity response and potentially result from vasculature malnourishment to the outer retinal layers. Congenital simple hamartoma of the retinal pigment epithelium (CSHRPE) is a rare benign pigmented lesion. Few reports have been published in the literature and most authorities presume this to be a congenital disorder . This tuMost current multimodal imaging techniques are insufficient for evaluation of CSHRPE partly due to the presence of heavy pigmentation within the mass. The SBOCT shadowing effect is a direct result of heavy pigmentation and has failed to demonstrate the internal structure of this tumor , 5. DesiRecently, Arjmand et al.  and ZolaHerein, we assess CSHRPE with multimodal imaging to further evaluate retinal microvasculature, retinal sensitivity and photoreceptor arrangement.A 35-year-old white woman was found on routine evaluation to have a solitary, circumscribed, nodular, and heavily pigmented retinal lesion in her left eye (OS). Visual acuity was 20/20 in the right eye (OD) and 20/25 in OS. The anterior segment and intraocular pressure were normal in both eyes, as was fundus examination of the OD. Fundus examination OS Fig. a showed Near-infrared reflectance imaging Fig. c demonstThe images were captured with the standard macula protocol with a resolution of 2 mm × 2 mm from the Avanti (Optovue) system . Hereby,Microperimetry showed an abnormal macular integrity index of 72.9 OS and normal macular integrity index of 9.5 OD,  [2), and number of neighboring cells  using the AO software images to create color scale graphic representation [2, respectively. Intercellular spacing was 11.98  demonstrated an irregular reflective cell mosaic that became discontinuous with enlarged hyperreflective and hyporeflective polygonal shapes at the site of the lesion (Fig. entation . MeasureTumors of the RPE are uncommon. They are classified into diagnostic groups including CSHRPE, congenital hypertrophy of the RPE, combined hamartoma of the retina and RPE, congenital albinotic and amelanotic spots of the RPE, torpedo maculopathy, and congenital RPE dysgenesis . It has In agreement to Gass  descriptAccording to the literature, surrounding mild retinal traction can be associated to CSHRPE in 80% cases . In one For the first time in the literature according to a Medline search, the present report confirms the presence of cones inner/outer segments in a CSHRPE using adaptive optics analysis Fig. E1, F1; nIn an effort to explore the potential retinal function at the site of this pigmented lesion and surrounding margin, we used microperimetry to determinate the sensitivity response Fig. a, b. TheStrengths of this study include the systematic qualitative multiple correlated imaging assessment from advanced technology using multimodal devices to better understand the architectural changes of CSHRPE. Limitations of this study include the ones usually related to the case report nature of the manuscript. Small number with limited variability of data. Limitations of the imaging techniques such as cone density measurements as commented above.In summary, OCT-A, adaptive optics, and microperimetry findings provide novel data that could predict functional visual outcome of this tumor. Future research multimodal imaging of this rare condition is warranted to better understand the pathophysiology and visual impact of this condition.


Acute upper digestive tract hemorrhage most often arises from gastric and esophageal vessels located in the mucosa or the submucosa. Rupture in the upper gastrointestinal tract is a classical but uncommon complication of arterial  aneurysms. Splenic artery aneurysm usually ruptures in the peritoneum, unless it is associated with a disease eroding the gastrointestinal wall. We present and describe the management of the rare occurrence of an intragastric rupture of a splenic aneurysm associated with a pancreatic cancer. This article was originally published without the text content, which was added shortly after. The publisher apologizes for this omission.Acute upper digestive tract hemorrhage (UDTH) is a life-threatening condition with multiple causes, mostly arising from the rupture of vessels in the esophageal or gastric submucosa or mucosa. Patients with UDTH are amenable to endoscopic diagnosis. Then, depending on the cause and the patient’s status, the treatment may include endoscopic surgery or endovascular procedures. When endoscopy fails to contribute to the diagnosis, intravenous contrast-enhanced computed tomography (CT) may help in identifying unsuspected causative lesions and hence facilitate surgical or endovascular treatment. We present an uncommon case of UDTH, where bleeding arose from the intragastric rupture of a splenic artery aneurysm (SAA) after erosion by a pancreatic cancer.A 61-year-old male was admitted to our hospital for unconsciousness followed by melena. On admission, he remained hemodynamically stable with a hemoglobin level at 12 g/dl . The patient underwent an upper endoscopy which revealed a stomach full of fresh blood. Neither esophageal varices nor ulcer were found. The patient rapidly progressed to hemodynamic instability, requiring intubation and polytransfusion. He was then taken to the angiography suite. The global aortic angiogram demonstrated the existence of a saccular aneurysm of 2.5 cm in diameter on the distal third of a splenic artery which also showed irregular contours Figure . HoweverIn the wake, a CT scan was performed. A distal pancreatic tumor abutting the posterior gastric wall was demonstrated. We speculate it could be responsible for the splenic artery erosion, with the development and secondary rupture of a false aneurysm in the gastric cavity Figure . The intSix days later, the patient presented a slow decrease of his blood hemoglobin (7.4 g/dl). A new contrast-enhanced CT scan revealed a new retrogastric blood extravasation originating from the distal third of the splenic artery Figure . A superThe main causes for severe UDTHs remain gastroduodenal ulcers and esophageal varices in the context of portal hypertension . In rare234567811Splenic artery aneurysms (SAAs) represent about one fifth of splanchnorenal aneurysms . They ar25The false aneurysm or pseudoaneurysm occurs in the case of trauma of the artery and has no own artery wall. It results from a direct trauma or, in the case of acute pancreatitis, for example, from a parietal digestion by pancreatic enzymes 8. Most o25Our case reports the association of a pancreatic cancer with SAA which ruptured in the stomach. To the best of our knowledge, this is the first such case reported in the medical literature. It is virtually impossible to know whether a true SAA has preceded the neoplasm or if the cancer has induced the formation of a false aneurysm, but the anfractuosity of the splenic artery on angiography makes us rather advocate for the second hypothesis.SAA rupture in general happens in one or two stages with, in the latter case, a hemorrhage which is first limited to the omental bursa and then extends to the whole peritoneal cavity 8. SAAs dThe ideal endovascular treatment consists in the deployment of a stent graft in the splenic artery at the level of the aneurysm, which may be prevented in some cases by the vessel tortuosity. Another method consists in the placement of a noncovered stent and secondary filling of the aneurysmal sac through the prosthesis. In case of acute emergency, the splenic artery must be embolized in taking care, mainly in the case of proximal SAA and if it is technically feasible, to proceed upstream and downstream the aneurysmal sac (or “sandwich” method) to avoid rebleeding by reperfusion due to collateral blood supply . In the Intragastric rupture of an SAA is a possible but rare etiology of UDTH, especially after gastric erosion by a pancreas cancer, as in the case we have presented. A correct identification of this potentially lethal entity is within the reach of modern CT scanners; it enables a relevant orientation of patients to a well-targeted endovascular or surgical procedure.The authors declare that they have no competing interests.


Minimally invasive techniques are generally applied for patients suspected of having benign fibroids if medical treatment is insufficient. On the other hand, sometimes some occult carcinomas of uterus like leiomyosarcomas may be reported for the patients' applied morcellation. This condition is rare but outcomes are clinically significant. Fragmentation of occult sarcoma in the abdominal cavity without isolation bag results in widespread and poor survival. In this article, we report a case of 37-year-old woman suffering from pain due to unexpected leiomyosarcoma. Laparoscopic myomectomy was performed with power morcellation in an isolated bag. Although isolation bag is generally reported to be preventive, recurrence of sarcoma was seen at 5th month of follow-up. Even though morcellation within a bag seems to block wide spreading, dispersion of tumor cannot be stopped and more investigations have to be done. Uterine sarcomas comprise 3% of uterine malignancies and leiomyosarcoma (LMS) accounts one-third of all uterine sarcomas . LMS is We report a case of 37-year-old young women with presumed benign leiomyoma treated with laparoscopic myomectomy with morcellation within an isolated morcellation bag reported as leiomyosarcoma at paraffin section .3, hgb was 12 g/dl, htc was 35%, plt was 282000/mm3, and no unusual characteristics was found in complete urinalysis. Ultrasonography revealed a mass consistent with a degenerated myoma measuring 77x82mm, of which subserous component was greater in the posterior wall and endometrial thickness was 7-8 mm, which was concordant with the cycle. The left ovary of the patient was normal, but since the right ovary could not be fully evaluated, computerized tomography (CT) was requested for possible adnexal pathologies. CT demonstrated a hypodense nodular lesion with a diameter of 75 mm that extended to the right adnexal region and MRI with contrast was performed on the patient. MRI revealed a mass with a hypovascular appearance following a heterogeneous and hypointense IV contrast material with a diameter of 8 mm, which appeared to displace the posterior cervix.37-year-old woman presented to the emergency room with headache. Bilateral pelvic sensitivity and a mass painful to touch in a plastic texture that filled the Douglas' pouch were detected during the examination; the mass was evaluated in favor of leiomyoma. ß-hcg was negative, wbc was 9800/mmIt is seen that myoma included by central necrosis depleted by cervix in the posterior part of the uterus. Also the development of necrosis was seen and myomatosis was evaluated in favor of degeneration .In addition, since the patient's pain regressed spontaneously during the follow-up, surgical operation was postponed to perform in elective conditions. After making necessary preparations for the operation, laparoscopic myomectomy was performed. Myoma was not considered suspicious apart from being degenerated. It was removed by morcellating in an isolated bag Figures .No complication or hemorrhage occurred during surgery. As there was not any problem observed during the post-op follow-up, the patient was discharged from the hospital on condition that the pathology report was brought on the second day of post-op. The subsequent report revealed marked cellularity and pleomorphism, extensive necrosis, mitosis >5/10BBA, KI67 proliferation index >%50, weak positivity for p53, and LMS with no detectable lymphovascular invasion.The patient was informed about the pathology report and interned again to perform transabdominal hysterectomy and bilateral salphingoopherectomy. The pathology report revealed “atypical pleomorphic fusiform cell proliferation area” measuring 3x2 mm in a focal area within the cavity consistent with residual LMS when evaluated together with laparoscopic myomectomy material.st surgery, positron emission tomographic scan (PET-CT) was performed and fluoro-2-deoxyglucose (FDG) uptake is noted in the area above rectosigmoid mesentery towards the left abdominal wall in pelvic floor (To follow-up the patient was referred to radiotherapy and medical oncologists. After 3 months from her 1ic floor .rd operation was performed. A mass about 4-5 cm of size in defined localization was excised; there were not any other mass and lymphadenopathy detected during the surgery. The pathology result was high grade malign mesenchymal tumor with high cellularity, extensive necrosis, mitosis 40/10 BBA, and moderate atypia. There was no tumor in surgical margin. She is now being followed up by the gynecologic oncologist with appropriate medical treatment.She was referred to gynecologic oncologist and 3Myomectomy is a surgical approach for uterine fibroids for young patients unresponsive to medical treatment . UterineThis soft tissue sarcoma is rare but aggressive and highly resistant malignancy with poor survival. This can be potentiated by power morcellation due to dispersion of tumor . As provAlthough this type of cancer is rarely seen, morcellating it within an isolated bag is important to prevent the worsening of its already adverse consequences. There was no known risk factor involved in this case; however, it is worth noting that any leiomyoma could have a potential risk for cancer. The patient must be informed in detail about this cancer risk even all the risk factors are excluded.Morcellation within a bag seems to prevent dispersion but even in a benign suggestive case, like ours, dispersion could not be prevented and that again reminds us about myomectomy procedure's role contributing to dispersion before putting the tumor into bag. We assume that only wide dispersion may be prevented with morcellation within a bag as in our case. Using bag may not be preventive due to inoculation during myomectomy via bleeding or seeding with local transmission by touching malign mass.


Glandular odontogenic cyst (GOC) is an uncommon cyst of the jaw. Less than 200 cases are reported in the literature, and only 22 cases are associated with an unerupted tooth (dentigerous relationship). Although it is an asymptomatic lesion, it can be destructive and has high recurrence rates. The diagnosis can be especially challenging due to the lack of distinct diagnostic clinic-radiological criteria and nonspecific microscopic features, mimicking benign and malignant lesions. Conservative surgical treatment has been the choice for most surgeons, but marginal or partial jaw resection has been reported. This report describes a rare case of GOC in a dentigerous relationship, which was treated with enucleation and peripheral osteotomy. Glandular odontogenic cysts (GOCs) are uncommon jawbone cysts of odontogenic origin which were firstly described in 1987 by Padayachee and Van Wyk  in 1988 To the best of our knowledge, there are 196 GOCs in the English literature –14. ClinRadiographically, it presents as a uni- or multilocular cystic lesion, with well-defined margins, though some lesions exhibit scalloped borders. Other findings include loss of cortical integrity, root resorption, and association with unerupted teeth . Some caMicroscopically, the diagnosis of GOC can be challenging, given the rarity of the lesion and the fact that the differential diagnosis includes benign and malignant lesions, such as botryoid cysts, surgical ciliated cysts, radicular or dentigerous cysts with metaplastic changes, and low-grade mucoepidermoid carcinoma (MEC) , 16, 17.Several treatment modalities have been indicated for the GOCs, including conservative approaches, such as enucleation with or without curettage, marsupialization, peripheral ostectomy and chemical cauterization with Carnoy's solution, and marginal resection/partial jaw resection . This reA 36-year-old male, with no medical history, was referred to the Clinical Hospital of the Federal University of Goiás, Goiânia, Goiás, Brazil, for evaluation of an asymptomatic radiolucent lesion in the posterior mandible region. Cone-beam computed tomography (CBCT) scan showed a well-defined unilocular radiolucency associated with an impacted right third molar, extending to the distal root of the second molar, measuring 17 × 12.5 mm . IntraorThe histopathological examination revealed cyst wall lining by nonkeratinized stratified squamous epithelium with varied thickness . Duct-liGlycogen-rich and mucin-secreting cells were highlighted by periodic acid-Schiff (PAS), periodic acid-Schiff diastase (PAS-D) Figures , and mucOur study reports an uncommon case of GOC associated with an unerupted third molar mimicking a dentigerous cyst. This characteristic was defined by Fowler et al.  as a “deClinical diagnosis of GOC is challenging. The differential diagnosis includes radicular and dentigerous cysts, odontogenic keratocysts, and ameloblastoma. Although Krishnamurthy et al.  suggest The histopathological diagnosis of GOC also remains a challenge. Microscopic features include focal epithelial thickening, epithelial plaques, and glycogen-rich epithelial cells, which are also observed in botryoid and lateral periodontal cysts. The presence of ciliated epithelium and duct-like spaces with mucous cells and eosinophilic cuboidal cells located in the epithelial surface support the diagnosis of GOC , 31. AccDue to the overlapping of histological features with others lesions, Fowler et al.  suggesteMinor surgical procedures, such as enucleation with or without curettage and peripheral ostectomy, are the most common treatment modalities reported in the literature , 32. In This report describes an uncommon case of GOC-DR mimicking other lesions in the oral cavity. These lesions tend to most commonly affect the posterior mandible and younger male patients.


Renal cell carcinoma (RCC) metastasizing to the heart with inferior vena cava (IVC) involvement is well-documented. However, its metastasis to the right heart without venous involvement is very rare. To the left atrium, metastasis is even rarer with only a few cases reported in medical literature. Herein, we report a case of a 56-year-old man who presented to our department for the treatment of a right renal mass and a right adrenal mass discovered on a follow-up plain computed tomography (CT) 13 years after left laparoscopic radical nephrectomy. During the workup, a transthoracic echocardiography (TTE) revealed a left atrial mass with a suspicion of a myxoma. This finding prompted a cardiac surgery consult which proposed a surgical removal of the mass. Intraoperatively, the tumor was found to invade the coronary sinus as well. The entire tumor was successfully removed and surgical repair of the unroofed coronary sinus was performed. Pathological examination of the tumor along with immunohistochemistry–showing positivity for CAIX, CD10, Vimentin, and PAX-8–pointed to a diagnosis of metastatic clear cell RCC. Eight months postoperatively, he was free of any symptom. In conclusion, RCC metastasizing to the left atrium is extremely rare. A comprehensive search revealed only nine reports in the literature. We report, to our knowledge, the first case of RCC metastasizing to the left atrium with concomitant invasion of coronary sinus. Surgical resection combined with unroofed coronary sinus repair allowed a complete removal of the tumor. In patients with a history of RCC, a metastasis should be thought of when a left atrial mass is present. Renal cell carcinoma (RCC) is an aggressive malignancy representing 2–3% of all cancers, and it is the most lethal of all urologic cancers. In 2018, there were approximately 136,500 new cases of renal cancer and 54,700 kidney-cancer-related deaths in Europe .Work-up: An abdominal and pelvic non-contrast CT revealed a 36 × 31-mm, ellipsoid, and hypodense mass in the external branch of the right adrenal region . It was During the patient evaluation phase, a non-contract CT of the chest revealed multiple nodules in both lungs, the largest one being located in the middle lobe of the right lung . ComparePrior to any other intervention, a cardiac surgery consult was sought, suggesting surgical removal of the mass due to the risk of it to detach. The patient was then transferred to cardiac surgery department where a repeat TTE showed a 27.7 × 16-mm isoechoic mass attached to the posterior leaflet annulus of the mitral valve in the enlarged left atrium . While tThrough a median sternotomy, cardiopulmonary bypass (CPB) was established by cannulating the ascending aorta and the superior and inferior vena cava. The plan was a hypothermic CPB with cold cardioplegia. After and through right atriotomy, an incision was made in the fossa ovalis, exposing a yellow mass close to the posterior leaflet of the mitral valve. It was a mass with a broad base rather than a pedicle. When the part of the tumor protruding inwards was entirely removed, the remaining part of the tumor was found to be in immediate vicinity of the coronary sinus. Via a longitudinal incision in coronary sinus, the tumor was entirely removed with its intact pseudocapsule. An exploration of the left atrium and the opening of pulmonary veins did not reveal any residual tumor. An artificial pericardial patch was applied to the incised part of the coronary sinus and the latter was successfully repaired without causing any leakage. Pericardial patch was also used to close the fossa ovalis and the right atriotomy was closed. Post-operative TEE did not reveal any residual tumor or abnormality in coronary sinus drainage. Two specimens of 2.7 × 1.5 × 1.5 cm and 2 × 1.7 × 0.7 cm were obtained . They weRCC is an aggressive disease accounting for 3% of all human malignancies and it is the most lethal of all the urologic cancers. In 2018, there were ~136,500 new cases of renal cancer and 54,700 kidney-cancer-related deaths in Europe  AND ] AND . The search was updated till February 24th, 2019 without any language restriction to detect LA metastases from RCC without IVC involvement. A hand search in Google scholar was also performed. To date, only 9 cases are available in the literature . The preDue to the risk of sudden death, embolism, and intraoperative or perioperative events in every patient with an intracardiac mass , it is wCardiac metastases from RCC are thought to occur via two mechanisms: The venous hematogenous spread through renal vein to the right heart and the lymphatic spread via carinal lymph nodes and parasternal lymph vessels. It has been reported that hematogenous spread is the most commonly involved pathway in metastasis to the right heart while the lymphatic spread is frequently involved in metastasis to the left heart . Left heRCC has a characteristic feature of a high propensity to venous invasion. On review of literature, except the present case, all the previously reported cases of LA metastasis from RCC revealed pulmonary veins involvement. Apart from the report by Miyamoto et al.  where a There is no established standard management for cardiac metastases from RCC. That said, metastasectomy is still playing an important role in RCC treatment and it has been shown to improve both overall survival and cancer-specific survival . MetastaCurrently, there is limited evidence regarding the efficacy and safety of immune checkpoint inhibitors in patients with intracardiac metastases. To date, only one report by Ansari et al.  describeIn conclusion, RCC metastasizing to the left atrium is extremely rare with only 9 reports in the literature. We report the 10th case of LA metastasis of RCC occurring 13 years post radical nephrectomy. This case highlights the need for a metastatic RCC to be considered when a left atrial mass is present in a patient with a history of renal cancer. To our knowledge, this is the first case of RCC metastasizing to the left atrium with concomitant invasion of coronary sinus. Surgical resection combined with unroofed coronary sinus repair allowed a successful removal of the entire tumor. In the absence of standard therapy for such cardiac metastasis, surgical removal appears to be both a diagnostic and therapeutic tool, preventing the risk of heart failure and sudden death. In surgically unresectable cases, conservative therapy with molecular targeted therapy (either tyrosine kinase inhibitors or mTOR inhibitors) constitutes a treatment option. Despite the limited evidence regarding the efficacy and safety of immune checkpoint inhibitors for patients with intracardiac RCC metastases, these agents may improve outcomes in this subpopulation.All datasets generated for this study are included in the manuscript/supplementary files.A written informed consent to publish the report and associated medical images was obtained from the patient.GN and QW collected and analyzed the patient's clinical data and designed the research. GN performed the review of literature and drafted the manuscript. QW, FT, SJ, WS, and LZ revised the manuscript. WS and SJ supervised the report and the publication process. All authors have read and approved the final version of the manuscript.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


This case presentation discusses an extremely rare diagnosis presenting with common symptoms, attributed to influenza, which were not investigated further when clear cardiac symptoms developed. The patient initially presented with symptoms consistent with influenza, but when orthopnea and dyspnea on exertion developed, these cardiac symptoms continued to be attributed to a postviral syndrome and were not further evaluated. Premature closure bias contributed to a delay in diagnosing a rare cardiac condition. The diagnostic momentum, or the continuing of a diagnostic label, occurred across multiple providers and settings. This case demonstrates the risk of premature closure and diagnostic momentum and reminds clinicians to reframe the differential diagnosis as more information on history or physical exam becomes available. A cardiac sarcoma is a rare malignant cardiac tumor that can present with nonspecific symptoms (similar to most cancers) in combination with clinical signs of heart failure due to the mass effect of the tumor. Timely diagnosis is crucial to prevent further obstruction as the tumor grows. If the tumor is recognized in the earlier stages, complications including heart failure, arrhythmias possibly leading to sudden cardiac death, and ischemia secondary to obstruction of coronary arteries can be avoided. Additionally, early detection can allow treatment before the cancer can metastasize.A 50-year-old Asian male with a past medical history of supraventricular tachycardia and obstructive sleep apnea on CPAP at night presented with one month of intermittent flu-like symptoms, orthopnea, and dyspnea on exertion. At the onset of these symptoms, he presented to a walk-in clinic and was diagnosed with influenza. He was treated symptomatically and noted improvement, but one week later he had a recurrence of symptoms while playing volleyball. From that time on, he noticed dyspnea on exertion, continued malaise, fevers, and diffuse joint pains so he presented multiple times to outpatient providers. He received doxycycline without improvement, and follow-up testing showed a mild leukocytosis, negative EBV, and an unremarkable chest X-ray. He was diagnosed with lingering postviral symptoms from influenza. He ultimately presented as a walk-in patient to the cardiology clinic when he started having chest tightness, palpitations, and his dyspnea progressed to occurring at rest, relieved only with a tripod position.EKG on presentation  showed rA cardiac sarcoma is a primary malignant cardiac tumor. These are exceedingly rare, with the incidence of primary neoplasms of the heart found at autopsy being 0.017%–0.056% . Of thesThe epidemiology indicates the rarity of this case, but the importance of recognizing this presentation in an outpatient setting before it progresses should not be lost. Beyond adding to the differential diagnoses of a patient presenting with progressive dyspnea on exertion, this case reinforces the importance of reframing the diagnosis when seeing a patient multiple times within a short timeframe. This patient was seen four times within a month by a combination of outpatient providers. Premature diagnostic closure bias creates difficulties for busy practitioners to reassess a case and develop a new differential when the previous diagnosis no longer applies. Our patient had a viral-like illness at the onset, but persistent symptoms of dyspnea on exertion and orthopnea should prompt a cardiac workup. An EKG showing right axis deviation and ST-T wave changes may have alerted practitioners that further workup is needed, and an echocardiogram or CT angiogram would have discovered the rare underlying diagnosis and allowed for expedited diagnosis and treatment.


The rectovaginal septum is a rare location for gastrointestinal stromal tumors (GIST) to occur. When such is the case, the question arises as to whether the lesion, which is morphologically and immunophenotypically identical to its gastrointestinal counterpart, should be referred to as an extragastrointestinal stromal tumor (EGIST). A 77-year-old, gravida 4, para 4004 post-menopausal female with an unremarkable gynecologic history presented with brown vaginal discharge. On examination, a 4 to 5-cm nodule was palpated along the rectovaginal septum. Ultrasound revealed a 4.8-cm circumscribed, solid mass with internal blood flow located posterior and inferior to the cervix. At laparoscopy, the uterus and adnexae were deemed to be normal for age, without gross pathologic abnormalities. The nodule was resected in an enucleation procedure; subsequent histopathologic examination revealed a low-grade, spindled cell neoplasm with diffuse immunoreactivity for CD117 (cKit) and DOG1, diagnostic of GIST. Further molecular testing elucidated a mutation in exon 9 of the Kit gene. A decision was made by the patient for close observation; there is no clinical or radiographic evidence of recurrence one year after initial diagnosis. Gastrointestinal stromal tumors (GIST), as the most common mesenchymal tumor of the gastrointestinal (GI) tract, typically originate from the tubal wall and extend intraluminally towards the mucosa, outwardly towards the serosal surface, or even in both directions -3. They A 77-year-old, gravida 4, para 4004, post-menopausal female presented with brown vaginal discharge. Her gynecologic history had been essentially unremarkable, with negative Pap Tests, and negative testing for high-risk human Papillomavirus. Physical examination revealed atrophic external genitalia. A mobile, 4 to 5-cm nodule was palpated along the rectovaginal septum, with a muscle-like consistency and texture. Pelvic ultrasound showed a uterus and ovaries with measurements as expected for a post-menopausal state. In addition to a 2.5-cm calcified uterine fibroid, a 4.8-cm solid mass with internal blood flow was identified posterior and inferior to the cervix Figure . 2; a rare focus of necrosis was observed  -19. Whil2 versus >5 per 5 mm2) [2 would estimate the risk of progressive disease of approximately 8.5%; if the lesion was believed to originate from the rectum, the closest gastrointestinal anatomical site [As remote recurrences of GISTs may be encountered years after excision, most regard this tumor as having at least some potential for metastasis . Guidelir 5 mm2) . In thisIt is estimated that approximately 85% of GISTs demonstrate mutations in the KIT gene, while approximately 10% harbor PDGFRA gene mutations; the remaining may show mutations in both genes . Those wAlthough GISTs rarely arise in the rectovaginal septum, it should be considered in the differential diagnosis of a spindled cell lesion when encountered in such location. The absence of a direct mural connection to the tubular intestinal tract raises the possibility that such lesion should be referred to as "extragastrointestinal stromal tumor", or EGIST.


Abdominal localization of cyst lymphangioma is rare. The splenic involvement is exceptional.A 63-year-old woman, who was followed for martial anemia evolving associated with abdominal pain in the past 12 months. On clinical examination, she had pallor conjunctival mucosa, with a normal abdominal and lymph node examination. The abdominal ultrasound showed multiple splenic cysts. The abdominal CT scan showed a normal-sized spleen with multiples hypodense cystic lesions. At the laparotomy exploration a multinodular spleen was found which measured 18 cm*15 cm*6 cm. The histological exam results showed concluded to a splenic cavernous lymphangioma without malignity signs. The follow-up after 12 months was normal.splenic lymphangioma is rare and benign tumor. Total splenectomy under laparotomy or laparoscopy is the appropriate treatment. The hea2A 63-year-old woman, with hypertension for 12 years and under treatment (amlodipine and bisoprolol), who was followed for martial anemia evolving associated with abdominal pain, in the past 12 months, with oral iron treatment. Her pain was moderated and localized in the left upper quadrant without any radiation.On clinical examination, she had normal vitals, pallor conjunctival mucosa, with a normal abdominal and lymph node examination. Her blood count revealed an anemia with 9,9 g/dl of hemoglobin.The abdominal ultrasound showed multiple splenic cysts without ganglionic hypertrophy. The abdominal CT scan with intravenous contrast showed a normal-sized spleen with multiples hypodense cystic lesions without enhancement and no enlarged lymph nodes .Picture At the laparotomy exploration a multinodular spleen was found which measured 18 cm*15 cm*6 cm. The abdominal viscera were macroscopically healthy. A total splenectomy was done, with a simple post-operative period. She received pneumococcal, meningococcal and Hemophilus vaccination after the surgery.The histological exam results showed a regular lymphoid splenic parenchyma with multiple cystic formations of variable diameter sometimes bordered by flattened endothelial cells. These cells are filled by serosities with lymphocytes and or red blood cells. There is fibrosis and calcification. It concluded to a splenic cavernous lymphangioma without malignity signs. The follow-up after 12 months was normal.3Lymphangiomas are rare benign tumor of the lymphatic vessels, diagnosed the most in children and young adult . In 60% The diagnosis is improved by medical imaging including ultrasound, CT scan and MRI . Ultraso-Parasitic cyst with the dermoid, squamous or transitional epithelium,-Cystic lymphangioma with a lymphatic endothelium-Traumatic cyst without any coating .The differential diagnosis of splenic lymphangioma is broad including hemangioma, splenic infarction, septic embolism, lymphoma or metastasis . There aIn case of any doubt, a fine-needle aspiration cytology guided by ultrasound is performed. However, the incidence of false negatives varies between 0,06 % and 2% with a hemorrhagic risk ,22.The treatment of splenic lymphangioma depend of the lesion size and the presence of complications . The treThe surgery is performed without delays except in case of surinfection or other contraindications . The ratThe histological exam of the operative specimen allows to confirm the lymphangioma diagnosis. It shows cystic formation with septa constituted with a connective stroma with lymphoid tissue, striated muscle and lining with a lymphatic endothelium (positive factor D2-40) . These r4Lymphangiomas are rare benign tumor of the spleen that are especially rare in adults. Splenic localization is incidental occurring in left upper quadrant pain exploration and rarely during a complication. The diagnosis is improved by the ultrasound and CT scan. But the histological exam allows to confirm the splenic lymphangioma diagnosis. The treatment is the total splenectomy under laparotomy or laparoscopy. The conservative medical treatment has not yet shown its effectiveness.This manuscript was written according to the rules of the SCARE .The authors declare no conflict of interest.The authors declare they have received no funding for the preparation of this documentThe ethical committee of the hospital gave the agreement to report this case.Written informed consent was obtained from the patient for publication of this case report and accompanying images.Thiam Ousmane, Faye Papa Mamadou these authors participated in the making and correction of this document. all authors agreed with the publication of the document.Researchregistr4759.Papa Mamadou FAYE.Not commissioned, externally peer-reviewed.


Desmoid tumours are rare benign neoplasms with strong tendency to local invasion and recurrence. They usually rise from muscles and aponeurosis and could be challenging for medical treatment. We here report the case of a patient requiring the excision of part of the abdominal rectus muscle and an abdominal wall reconstruction.A 38 years old women referred to our institution complaining of a painful lump in the anterior abdominal wall. She underwent ultrasonography (US), magnetic resonance imaging (MRI) and a computed tomography (CT) biopsy who showed the presence of a desmoid tumour arising in the right abdominal rectus muscle. She underwent surgical excision of the mass and an abdominal wall reconstruction according to the posterior component separation technique, with a large polypropylene mesh reinforcement. The post-operative period was uneventful and at 6 months she shows no clinical nor radiological recurrence.Desmoid tumours are rare neoplasms rising in many anatomical spaces and requiring challenging decisions. A radical resection with free margins remains the principal determinant of outcome. Non-surgical approaches and adjuvant therapy may be useful for patients with unresectable lesions.The optimal treatment strategy for desmoid tumours still remains unclear. A multidisciplinary approach is always the best strategy to establish the correct treatment. These lesions are also known as deep fibromatoses or aggressive fibromatoses and they have a strong tendency to invade and recur locally after resection, with an intermediate biological behaviour between benign fibrous tumours and fibrosarcomas. Desmoid tumours could be extra-abdominal , abdominal, intraabdominal, multiple, multiple familial and associated with FAP or Gardner's syndrome. They represent the 0,03% of all neoplasms and 3% of all soft tissue tumours . These nAccording to the SCARE criteria , we pres2A 38 years old woman referred to her general practitioner in August 2018 for a painful mass in the right anterolateral abdomen. On physical examination the mass was tough and fixed to the abdominal wall. In September 2018 an ultrasound examination was performed and demonstrated an oval solid mass of heterogeneous echogenicity in the context of right rectus abdominis muscles, with irregular margins and modestly vascularized  . In Octo2). A posterior-component separation technique was performed, extending the dissections towards the lumbar and ileo-psoas muscles to allow the closure of the midline. A 40 × 60 cm mesh obtained suturing two 20 × 30 cm polypropylene meshes  [In January 2019 an “en-bloc” resection of the mass was performed including the peritoneum and the right rectus muscle , Fig. 3,, Italy) ,18 was pMacroscopically the tumour was solid, with a firm texture. On the cut surface it appeared drawn, stretchy and greyish, resembling scar tissue. The histological diagnosis was of a desmoid tumour and every margin was free. The post-operative period was uneventful and the patient was discharged on the tenth post-operative day. At six months follow-up after surgery the patient is healthy, shows complete remission without any other treatment and MRI showed no local recurrence nor incisional hernias.3Desmoid tumours are benign deep fibromatoses that originate from fascia and muscular aponeuroses, especially the rectus and internal oblique muscle and occasionally cross the midline. They have an infiltrating growth pattern and less commonly originate from the external oblique and the transversalis muscle or fascia. They are rare tumours occurring in 3.7 cases per million individuals every year. They have been correlated with female gender, FAP and occasionally with abdominal trauma .Plenty of modern imaging method are used for the diagnosis. In ultrasonography desmoids have variable echogenicity, with smooth, well-defined margins. In CT scan the lesions are generally characterized by high attenuation (relative to muscle) and have either well or ill-defined margins. Even a CT scoring system has been developed, characterizing specimens according to the presence of desmoid precursor lesions (mesenteric fibrosis) and true desmoid tumours. This has provided further evidence for a stepwise progression in desmoid development. The degree of enhancement after the intravenous administration of contrast medium is variable. In MRI scans desmoid tumours have a low signal intensity in T1-weighted images (relative to muscle) and variable signal intensity on T2-weighted images (high in our case). Furthermore, MRI scans can indicate how the tumours will probably behave, with a bright signal indicating a high-water content, which has been correlated with rapid growth. Sometimes low T2 signal intensity bands are characteristic and represent foci of high concentrations of collagen deposition. However, the definitive diagnosis must be established with a histopathological analysis with diffuse cell infiltration of the adjacent tissue structures. The immunohistochemical response for actin may be partially positive and immunohistochemical muscle cell markers may distinguish desmoid tumours from fibrosarcoma . In our The local invasive nature and high rates of recurrence of this type of lesion, force surgeons to manage with difficult choices and require complicated treatments. There are several possibilities as surgical resection, radiotherapy, hormonal therapy (tamoxifen or toremifene) and use of non-steroidal anti-inflammatory drugs . We could say that chemotherapy is usually reserved for patients with significant symptoms that do not respond to milder approaches such as NSAIDs and tamoxifen . AbdominRadiation therapy has been used predominantly for the extra-abdominal desmoid tumours treatment and has resulted in an improvement of local control by reducing local recurrence rates. External beam irradiation or brachytherapy may be used alone, especially in patients with unresectable lesions, even if they are correlated with high failure rates. They may also be used either as new-adjuvant or adjuvant therapies following incomplete surgical resection ,14.The recurrence rate is 20%–77% depending on location, extent and completeness of initial resection. Abdominal wall desmoid tumours have a significantly lower recurrence rate (20–30%) and usually becomes evident within six months after excision or in connection with subsequent gestations or deliveries. Metastatic disease has not been reported with desmoid tumour . In womeRadical tumour resection with free margins has typically been recommended as the first-line therapy; however, an increasing number of surgeons are using “wait and see” strategies because an optimal management strategy has not been defined. Even the UK guidelines, referred to the recent European Consensus, stated that watchful waiting should be the standard first-line option ,19.There are several studies and case reports describing surgical approaches for resecting desmoid tumours when present in the abdominal wall but the best surgical strategy is controversial and it is dependent on the patient's background and the surgeon's preference ,3,13,17.4The optimal treatment strategy for desmoid tumours still remains unclear. Surgery is the primary treatment and a radical resection with free margins remains the principal determinant of outcome ,15,19. NInternational System of Units (SI).The authors declare that no sources of funding have been requested for this research.The authors declare that this study is exempt from ethical approval.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.The authors declare that no registration is needed for this work.Mr Bruno Scotto is the Guarantor for the work.Not commissioned, externally peer-reviewed.Francesca Ascari: Data curation, Writing - original draft. Silvia Segattini: Visualization, Investigation. Michele Varoli: Supervision. Massimo Beghi: Software, Validation. Simone Muratori: Conceptualization, Methodology, Software. Bruno Scotto: Writing - review & editing, Supervision. Marco Gasperoni: Data curation, Software, Validation.The authors declare that have no conflicts of interest.


Dedifferentiated liposarcomas most commonly arise in the retroperitoneum, accounting for 10% of liposarcomas. Heterologous differentiation occurs in 5-10% of dedifferentiated liposarcomas; however, divergent osteosarcomatous differentiation is rare. We report a rare case of initial presentation of dedifferentiated liposarcoma with osteosarcomatous component as a colonic mass in a 72-year-old man. The tumor is mainly composed of bony trabeculae with intervening highly atypical cells and adjacent high-grade mesenchymal nonlipogenic tumor, as well as areas of well-differentiated liposarcoma. Immunohistochemical studies showed diffuse positivity for SATB2 in the atypical cells and fluorescence in situ hybridization revealed high-level amplification of MDM2 gene, supporting the diagnosis of well-differentiated and dedifferentiated liposarcoma with heterologous osteosarcomatous differentiation. Liposarcomas are the most common malignant mesenchymal neoplasms, accounting for about 20% of all sarcomas . DediffeA 72-year-old male presented with a history of weakness and constipation, for which he underwent colonoscopy, revealing a submucosal mass in the right colon, compressing on the lumen extrinsically. The mucosa overlying the mass was intact and colonoscopy showed an adjacent 12 mm polyp, which turned out to be a tubular adenoma. He had history of removal of previous tubular adenomas on prior colonoscopies. Computed tomography (CT) imaging showed a 7.0 cm lobulated mass arising from mid-ascending colon with exophytic growth and foci of calcifications. CT findings were consistent with a mesenchymal tumor and carcinoma was less likely. No fatty component was reported . SubsequGross examination of the right hemicolectomy specimen demonstrated a firm and calcified intramural mass in the posterior wall of ascending colon, extending into the mesocolic fat, without involvement of the mucosa. Microscopically, distinct histological patterns were identified, with the majority of the tumor being composed of a central ossified core surrounded by a minor component of a high-grade mesenchymal nonlipogenic tumor and adipose tissue. The central bone forming component showed areas of osteoid matrix with highly atypical cells resembling a high-grade osteosarcoma as well as areas of more mature appearing woven bone trabeculae with intervening mildly atypical spindle cells similar to a low-grade osteosarcoma (LGOS). A thin rim of a high-grade sarcoma composed of atypical spindled and pleomorphic cells was identified in the periphery of the bony mass. In addition, small foci of mature-appearing adipose tissue containing rare atypical hyperchromatic stromal cells were detected adjacent to the high-grade sarcoma and at the retroperitoneal resection margin, raising consideration of a WDL Figures  and 3. TOn immunohistochemical (IHC) studies, SATB2 was diffusely positive in the atypical cells between bony trabeculae and in high-grade sarcomatous areas without evident bone formation. High and low molecular weight keratin (pancytokeratin), CD117, DOG-1, desmin, SMA, and S-100 stains were negative. Fluorescence in situ hybridization (FISH) revealed high-level amplification of MDM2-gene, supporting the diagnosis of retroperitoneal WDL with DDL showing heterologous osteosarcomatous differentiation and forming a mass involving the ascending colon.The term dedifferentiated liposarcoma (DDL) was first introduced by Evans in 1979 to define the morphological progression from a well-differentiated liposarcoma to a nonlipogenic sarcoma. DDL commonly occurs in the retroperitoneum showing a variable histological appearance, most frequently a high-grade sarcoma which is much less aggressive than other types of high‐grade pleomorphic sarcomas . A minorPresentation of a heavily ossified deep visceral mass is an unusual finding, which proposes a differential diagnosis of heterologous osseous differentiation in a mesenchymal or epithelial neoplasm or rarely extraskeletal osteosarcoma. In our case, the initial presentation of the patient with constipation, colon mass, and concurrent and previous tubular adenomas raised a clinical concern for colorectal carcinoma. Imaging findings, however, were consistent with a mesenchymal tumor of the colon with no fatty component. When present, imaging findings of a fatty component could be helpful in diagnosis of a liposarcoma. Although rare, osseous differentiation in soft tissue sarcomas and colorectal carcinomas have been reported –8. In thEvaluation of MDM2 and CDK4 expressions by IHC and FISH have been increasingly used for distinction of WDL/DDL from other sarcomas . HoweverIn summary, DDL with osteosarcomatous dedifferentiation is a rare entity. Our case illustrates an unusual initial presentation of a DDL with osteosarcomatous components as a colonic mass. The microscopic recognition of WDL was the key finding in diagnosis of this case.


MRI plays a critical role in the evaluation of osteomyelitis. However, MRIfindings of osteomyelitis are not entirely specific and may mimic infiltrativetumors. We describe a case of extraosseous extruded medullary fat with a tinytranscortical tract caused by acute osteomyelitis, diagnosed by MRI andconfirmed with intraoperative findings and pathology. Identification of extrudedmedullary fat is a specific MRI finding that aids in the differentiation ofacute osteomyelitis from infiltrative tumor thereby preventing unnecessarybiopsy and facilitating prompt diagnosis and initiation of appropriatetreatment. A 62-year-old male with a history of diabetes mellitus and rheumatoid arthritis,treated with leflunomide and rituximab, presented with bacteremia and acute onsetright knee pain. Physical examination revealed right knee and proximal tibiatenderness and erythema. Initial laboratory work-up revealed gram-positive coccibacteremia, elevated C-reactive protein, and right knee joint aspiration with noorganisms seen on gram stain.1 imaging revealed smallfoci of extraosseous macroscopic fat tracking from a tiny cortical defect in theanterior tibial cortex (Imaging of the right knee, tibia, and fibula was preformed to evaluate for a sourceof pain. Radiographs were normal .MRI witl cortex .vs infiltrative or hematological malignancy vssystemic marrow process. The identification of a transcortical tract with expulsionof marrow fat to the extraosseous soft tissues, however, is considered a highlyspecific finding in acute osteomyelitis effectively narrowing the differential to asingle diagnosis and preventing an unnecessary biopsy to exclude malignancy.Given the diffuse though well-defined heterogeneous signal abnormalities asymmetricwithin the tibial shaft, initial diagnostic consideration included osteomyelitisEmpiric vancomycin was subsequently narrowed to cefazolin following blood culturesensitivities revealing methicillin-sensitive Staphylococcus aureus. With thedefinitive imaging diagnosis of acute osteomyelitis, continued positive bloodcultures, and progressive symptomatology despite appropriate antibiotic treatment,the patient was referred to The Limb Restoration Program for multidisciplinaryevaluation. The multidisciplinary program group decision was for urgent surgicalirrigation, debridement, and saucerization of the proximal tibial metadiaphysis asthe patient’s immunocompromised state limited his ability to clear theinfection with systemic therapy alone. Intraoperative findings were notable formacroscopic extraosseous fat and an adjacent osteomyelitic tibial sinus track . Deep soPrior to hospital discharge, the patient’s blood cultures revealed no growthand a peripherally inserted central catheter was placed to complete a 6-week courseof cefazolin. Upon outpatient follow-up after completing 2 weeks of cefazolin, thepatient was found to have a healing wound site and no systemic signs or symptoms ofinfection.1,2 Radiographic misdiagnosis of osteomyelitis is especially notable in thepediatric population where it is estimated that half of subacute osteomyelitis casesare misdiagnosed as tumor.3 The imaging feature of extraosseous marrow fat in the region of marrow signalabnormality is a highly specific finding in acute and aggressive osteomyelitis andmay assist in the differentiating of these entities by imaging alone.4Osteomyelitis, infectious inflammation of the bone marrow, is a cause of significantmorbidity and mortality. Although MRI is the most sensitive imaging modality fordiagnosing osteomyelitis, imaging features can be confusing often mimickingaggressive infiltrative tumor. Overlapping clinical features of osteomyelitis andskeletal neoplasm, including focal pain, fever, and elevated inflammatory markers,further complicate this diagnostic dilemma. Osteomyelitis has been previouslydescribed masquerading as Ewing sarcoma, chondrosarcoma, and other skeletal neoplasms.4,5 Interestingly, foci of extraosseous fat are predominately a feature of acuteosteomyelitis and not identified in subacute osteomyelitis patients.4 This observation is in keeping with the pathophysiology of aggressive acuteosteomyelitis resulting in extrusion of medullary fat as opposed to a subacute,slowly progressive process.Understanding of the pathophysiology of osteomyelitis facilitates the interpretationof extraosseous fat signal when there is clinical concern for osteomyelitis. Acutesuperative response from medullary bacterial proliferation can cause an acuteincrease in intramedullary pressure, ultimately leading to a rupture of the cortexand expulsion of medullary fat into the adjacent soft tissues.4,6,7 To our knowledge, this is the only reported case confirmed with both bonebiopsy and intraoperative findings documenting a cortical sinus tract withmacroscopic extraosseous fat. Extraosseous fat fluid levels have been elegantlydepicted on fat-suppression MRI sequences and previously described as a specificsign for acute osteomyelitis in patients presenting with a soft tissue infection.6,7 In the absence of trauma, the extraosseous fat fluid level on MRI has beenproposed as a specific sign for osteomyelitis with cortical breach even when thecortical breech was not seen.7 In contrast to acute osteomyelitis, the penumbra sign consisting ofhyperintense signal interposed between low signal abscess cavity and marrow has beenpreviously described as highly specific for chronic, subacute, or acute on chronic osteomyelitis.8There are few publications documenting the imaging finding of extraosseous fat as aspecific feature of acute osteomyelitis.Extraosseous marrow fat is a specific imaging feature of acute aggressiveosteomyelitis assisting in the differentiation between osteomyelitis andinfiltrative tumor, thereby preventing unnecessary biopsy for diagnosis andfacilitating prompt treatment.In the absence of trauma, extraosseous marrow fat in the setting of softtissue infection is indicative of acute osteomyelitis. This may beparticularly helpful when the cortical defect is not seen due to wideinterslice gaps.Extraosseous medullary fat aids in determining aggressiveness of acuteosteomyelitis, as it requires extensive, rapid marrow necrosis in order forthe macroscopic fat to accumulate in extraosseous soft tissues.


The urachus is the remnant of the cloaca and allantois, and attaches the umbilicus to the bladder dome. Urachal anomalies are symptomatically identified during childhood. It rarely occurs in adults, making diagnosis difficult. We present and discuss the case of an infected patent urachus in a 30-year-old male. Ultrasound and CT scan play an important role in the diagnosis of this pathology.The urachus is a midline tubular structure that extends upward from the anterior dome of the bladder towards the umbilicus. The urachus involutes before birth, remaining as a fibrous band with no known function.–3. A urinalysis was within normal ranges.A 30-year-old male presented with history of lower abdominal pain and periumbilical erythema, which had been persisting for 15 days, with nausea, vomiting, fever and umbilical discharge for the past 8 days. Physical examination revealed a temperature of 38.7°C. His abdomen was soft and there was umbilical discharge with erythema and a tender umbilical mass. Laboratory tests revealed leucocytosis of 22,000 mmUltrasonography revealed the presence of a heterogeneous mass of size 7.2 × 3.2 cm in the midline extending from the anterior and superior wall of the urinary bladder to the umbilicus with presence of gas . Colour 1 This structure closes by the third trimester of gestation or after birth, forming the median umbilical ligament.3 The urachus lies in the space of Retzius, between transversalis fascia anteriorly and the peritoneum posteriorly. Histologically, it is composed of all the three bladder layers, the innermost layer being lined with transitional epithelium in 70% and columnar epithelium in 30% of cases.4The urachus is a vestigial structure that develops from two embryological structures: the cloaca  and the allantois (derived from yolk sac). During the fourth and fifth months of gestation, the foetal bladder descends into the pelvis and the allantois lumen is obliterated to form the urachus.1 These anomalies are twice as common in males, and the most common presentation is the patent urachus in about 50% of cases.5 The patent urachus usually presents during the neonatal period with urinary discharge from the umbilicus, and are extremely rare in adults. An umbilical–urachal sinus, vesicourachal diverticulum or urachal cyst is generally asymptomatic and can reopen in association with pathological conditions.2The incomplete involution of urachus results in four types of abnormalities: patent urachus, umbilical–urachal sinus, vesicourachal diverticulum and urachal cyst .1 These 6 showed that children are more likely to have an infected urachal cyst, while adults are more likely to have an infected urachal sinus.6 Infection may spread through the blood, lymphatic or directly from the bladder. The cultured organisms include Escherichia coli, Enterococcus faecium, Klebsiella pneumoniae, Proteus, Streptococcus viridians and Fusobacterium.7Luchtman et al3The clinical manifestations of patent urachal pathologies are umbilical discharge, erythema and mass within the umbilicus. The abdominal pain can mimic other conditions such as appendicitis or Meckel’s diverticulum. The differential diagnoses include abscess, umbilical hernia, urachal carcinoma and other tumours.8 described the urachus as an elliptical, hypoechoic structure, with variable size in the middle of the anterosuperior surface of the urinary bladder.8 A CT scan can accurately confirm the type of urachal anomaly. However, differentiating between an infection and carcinoma of the urachus can be difficult; calcification occurs in 50–70% of urachal carcinomas and this data can help in the differential diagnosis.1Urachal anomalies can be accurately demonstrated by both a CT scan and an ultrasound; other diagnostic studies include i.v. pyelography, cystography, cystoscopy and MR. Ultrasound is usually enough to diagnose an infected urachal remnant. Cacciarelli et alThe treatment of an infected urachus should include antibiotics combined with drainage of the abscess, followed by total excision of the remnant. This treatment seems to be the most effective option for avoiding the future development of malignant disease.The presence of mass in the midline, umbilical discharge and abdominal pain are useful in clinical suspicion of urachal anomalies.The imaging studies confirm the diagnosis, help define the anatomical relationships of the lesions and are useful in differentiating between benign and malignant disease.An understanding of the embryological failure, associated imaging findings and complications are important for early diagnosis.Informed consent has been provided by the patient.


Angiolymphoid hyperplasia with eosinophilia is a rare nodular skin tumor characterized by eosinophilic invasion and vascular proliferation. Previous reports suggested that irritation and inflammation are the causative factors of this disease. Most cases of angiolymphoid hyperplasia with eosinophilia occur around the auricle, forehead, and scalp; the hand is rarely affected. Moreover, the tumor seldom presents as multiple nodules.A 67-year-old Japanese woman presented with a complaint of skin masses on her left thumb and index finger, which had gradually grown in size over the past few months. A biopsy was performed confirming a diagnosis of angiolymphoid hyperplasia with eosinophilia. The aponeurosis on her index finger was resected and tissue was reconstructed using a reverse palmar digital island flap harvested from the base of her index finger. The thumb lesion was also resected and covered with collagen-based artificial skin. Gradual progression of skin epithelialization followed by healing was noted 2 months after the surgery.Angiolymphoid hyperplasia with eosinophilia is a rare tumor; it is seldom seen in the hands. It is generally treated by surgical resection. It is important to resect a sufficiently large area of the tissue due to the possibility of relapse in some cases. Furthermore, appropriate reconstruction is mandatory after wide margin tumor resection. Angiolymphoid hyperplasia with eosinophilia (ALHE) was first reported by Wells and Whimster in 1969 , thus, nHere we present a rare case of ALHE of the hand in a woman.A 67-year-old Japanese woman presented with complaints of a mass of skin on her left thumb and index finger that had been gradually increasing in size over the preceding few months. Her general condition was good . No abnormal breath or heart sounds were heard during auscultation. An abdominal examination revealed no tenderness, rigidity, or rebound, and her bowel sounds appeared normal. Neurological abnormalities such as absent or brisk deep tendon reflexes, muscle weakness, and hyperesthesia were not observed. In addition, no edema was present. According to the laboratory data, glycated hemoglobin (HbA1c) was high (8.9–9.4 over the previous 6 months); however, no abnormalities were noted in the other parameters. Furthermore, no abnormalities or signs of infection were observed in her urine analysis  for diabetes mellitus, hypertension, and allergic rhinitis, respectively. She had a history of smoking 10 cigarettes a day over the past 20 years. She did not drink alcohol, and there was no history of ALHE or other allergic diseases in her family. She was a housewife and was not involved in any specific occupation.Several skin masses, each approximately 10 mm in diameter, were observed at the base and dorsum of her left thumb and on the volar side of her index finger  than the reference value. Magnetic resonance imaging revealed that the tumor was confined to the skin and had not extended to the thumb joint  . Moreoveet al., ALHE is not a vascular lesion, but a type of CD4-positive T cell lymphoma [ALHE does not develop complications during conservative therapy, and no cases of malignant transformation have been reported so far . Yet, ALlymphoma –15. AccoIn some cases the outcomes of administration of local and systemic corticosteroids to patients with AHLE were unclear. Furthermore, laser treatment has also been provided with unsatisfactory results , 17. ThePathologically, ALHE involves vascular damage characterized by the presence of an unstable basement membrane and arteriovenous shunts , 19. TheALHE is a rare, benign skin tumor characterized by the presence of vascular proliferation and eosinophil invasion, and is often treated conservatively. However, when accompanied by symptoms such as pain, surgical resection of the tumor is required. In such cases, removal of a sufficiently large area of resection is important to ensure a favorable outcome. In addition, adequate reconstruction is necessary to guarantee post-surgical functions.


Gastric glomus tumor (GGT) is a rare gastrointestinal tumor and its preoperative imaging features are significant to make a correct diagnosis, while the assessment of the pathological and immunohistochemical characteristics of the specimen are the main methods used for its diagnosis. This study introduces the clinical uniqueness, endoscopic ultrasonography, radiology, histology and immunohistochemistry results of a patient with GGT to discuss the imaging and clinico-pathological features, diagnosis and differential diagnosis of GGT.The patient expressed a complaint concerning an “intermittent abdominal pain for 4 months”.The patient was diagnosed with gastric stromal tumor according to the clinical manifestations and imaging examination before the operation. The pathological examination of an intra-operative frozen sample confirmed the benign nature of the tumor, while post-operative immunohistochemistry results indicate the presence of a GGT. The postoperative histology revealed a tumor tissue composed of irregular blood vessels and glomus cells of same size with interstitial hyaline and mucoid degeneration. Immunohistochemical staining showed positivity for SMA (+), vimentin (3+), CD 34 (vascular +), and Factor VIII (vascular +).The tumor was completely removed by surgery.The patient recovered well, and was discharged from the hospital. Five months after the operation, a normal gastric mucosa was observed by gastroscopic examination.Most of the GGTs are benign lesions, surgical resection is the preferred treatment and they result in a good prognosis. However, malignant GGT should be treated as soon as possible because of its metastatic potential and recurrence. Adjuvant radiotherapy or chemotherapy might be useful after operation. With the development of various diagnostic techniques, reports on GGT are rising. They accounts for approximately 1% of gastric mesenchymal tumors.,4 Most GGTs are benign, only few cases are malignant. GGT and gastric submucosal tumors such as gastrointestinal stromal tumors (GISTs) and others have similar clinical manifestations and imaging features. Therefore, GGT diagnosis mainly relies on postoperative immunohistochemical findings. Endoscopy and imaging examinations have little significance in making a preoperative diagnosis. This article mainly reports a case of GGT and explores the endoscopic, radiological and pathological features, clinical symptoms and the diagnosis and differential diagnosis of GGT through the interpretation of the relevant literature.Glomus tumor is a mesenchymal tumor that originates from the neuromuscular arterial canal or vascular lumen. It can develop in any part of the body, mainly occurring in the surrounding soft tissues and distal limbs.2A 62-year-old male patient was admitted to hospital on August 31st, 2017 due to an “intermittent abdominal pain for 4 months". The patient did not show any clear cause that justified the abdominal pain, no history of nausea, vomiting, hematemesis, melena, and no clear recent change in body weight. The patient was diagnosed with gastric tumor at a local hospital while having gastroscopic examination. During hospitalization, physical examination, blood, urine, stool, liver, and kidney function tests were examined and was found to be in a normal range, with normal levels of blood coagulation factors and tumor markers. Preoperative diagnosis of gastric stromal tumor was made on the basis of the results of endoscopic ultrasonography (EUS) , vimentin (3+), CD 34 (vascular +), Factor VIII (vascular +), Ki-67 <5%, ckp (−), CD 117 (−), and CD 31 (−).4The patient recovered well, the symptoms disappeared completely, and 5 months after operation, the gastroscopic examination showed normal gastric mucosa.55.1 Paroxysmal pain is the main clinical symptom. This tumor rarely occurs in the gastrointestinal tract, nasal cavity, lung and other visceral organs. The currently known GGT was first diagnosed in 1948. However, it was described in 2 other patients in 1951.,6 Since it is mainly located in the muscular layer of the gastric wall, a layer having scarce nerve supply, the clinical symptoms are atypical. The most common clinical symptoms include abdominal pain and discomfort, loss of appetite, gastrointestinal bleeding and ulcers with or without nausea and vomiting. Although most GGTs are benign, reports of the malignant form are rare, but cannot be ignored. Kirsch Baum et al first reported a malignant GGT in a 40-year-old male in 1939.Glomus tumor is a benign mesenchymal tumor composed of glomus cells arranged in a way that resembles the structure of an organ. WHO classification of soft tissue tumors (2002) placed it into the group of perivascular cell tumors, frequently found in the surrounding soft tissues and distal limb subcutaneous tissues.5.2,10 In the abdominal enhanced MDCT examination, GGTs are usually located in the gastric antrum. In the arterial phase, GGT is often manifested as an inhomogeneous enhancement of the high-density shadow. In the portal venous phase, the tumor is continuously and uniformly enhanced. However, the delayed phase is a submucosal lesion characterized by the presence of the same density of the liver. The CT value and enhancement curves are consistent with the inferior vena cava, portal vein or descending aorta signs of vascular disease. Histologically, the tumor usually has a defined boundary, composed of prominent circular glomus cells, accompanied by aneurysmal dilatation. Immunohistochemistry shows positive vimentin and SMA, and negative S-100, CD 34, CD 117, desmin, CD 56, synaptophysin, chromogranin, neuron specific enolase and cytokeratin.MDCT, endoscopic and other imaging examinations often misdiagnose it as GIST or leiomyoma. Its endoscopic performance is often related to its growth pattern. If the GGT grows into the gastric cavity, then it is often shown as a spherical submucosal protuberant lesion with smooth edges in the gastric antrum under endoscopy. However, if it grows outside the lumen of the stomach, common endoscopy can hardly find it. In this case, EUS or abdominal MDCT is more appropriate. EUS reveals a hypoechoic region in the third or fourth layer of the gastric wall, which cannot be distinguished from other gastric submucosal tumors.5.3 2- EUS, which shows a heterogeneous hypoechoic area that cannot be distinguished from other possible lesions such as GIST; 3- GGT by abdominal MDCT usually reveals it in the gastric antrum. Arterial phase scan often shows the inhomogeneous enhancement of the high-density shadow. Coronal portal phase CT image shows a sustained uniform enhancement of the tumor. Delayed phase scan shows a submucosal lesion characterized by the presence of the same density of the liver. Despite the presence of certain characteristics, it is still difficult to distinguish GGT with other features. 4- At present, some authors believe that endoscopic ultrasound-guided fine needle aspiration biopsy can diagnose the GGT before operation, but still a lot of misdiagnoses are made. The main reason is that the endoscopic ultrasound-guided needle is not one hundred percent accurate to evaluate the lesion tissue, and the tumor bleeding may affect the judgment.,10 Under these difficulties in making a correct diagnosis, only the postoperative pathological and immunohistochemical diagnosis of GGT is still recognized as the gold standard. Although almost all glomus tumors are benign, some malignant cases have been reported, thus the diagnostic criteria have been proposed: 1- Deeper location, in the lower part of the fascia or in the solid organs, with a tumor diameter of more than 2 cm; 2- presence of atypical mitoses; 3- presence of nuclear pleomorphism, moderate to high nuclear grade, mitotic figures (5/50 HPF).,13 However, although the average size of GGT is 2 cm, a lot of benign GGT are larger than 2 cm. There are also reports of very low mitosis (1-3/50 HPF) in GGT metastasis.,13 Therefore, the diagnostic standards described above for malignant tumors should be further evaluated to conclude that they can be valid standards in the GGT diagnosis.GGT mainly occurs in the mucosa or muscular layer of the gastric antrum, rarely occurs in the gastric fundus or body. There are several diagnostic examinations, although not perfect enough for making an accurate diagnosis: 1- Endoscopic examination, which commonly shows only a spherical submucosal protuberant lesion with a smooth edge in the gastric antrum, thus, without a specific diagnostic significance;5.4GGT can be misdiagnosed with other gastric submucosal tumors originating from the mesenchyme, such as GIST, carcinoid tumors, neurilemomas and hemangiomas of the stomach and some other rare tumors. The gastric submucosal tumor belongs to non-epithelial mesenchymal tumors, mainly originating from the submucosa of the stomach or gastric muscular layer, usually covered with mucosa, and account for approximately 0.1% to 1% of the gastrointestinal tumors. The most common diagnostic modality used for gastric submucosal tumors are EUS and MDCT. EUS can be used to determine the origin of the tumor, which is generally seen as a hypoechoic tumor located in the third or fourth layer. Endoscopic ultrasound-guided biopsy is an accepted method for the diagnosis of submucosal lesions, while MDCT is used to determine tumor characteristics by contrast enhancement. The MDCT arterial phase scan of GGT results in images different from the ones resulting from a gastric stromal tumor. The degree of enhancement is significantly higher than that of gastric stromal tumor. GIST shows a lack of dilated capillaries in the pathological examination, and immunohistochemistry shows positive CD117, CD 34 and DOG-1 in tumor cells.,16 However, the results of GGT immunohistochemistry usually reveal positive vimentin, SMA, and negative CD 34, CD 117, DOG-1.GIST is the most common gastric submucosal tumor, while many submucosal tumors are confused with GIST. The MDCT arterial phase scan in GIST shows a small or smooth structure on the edge of the tumor, which is characterized by a strong heterogeneity in the arterial phase, often with cystic degeneration, necrosis and hemorrhage.Gastric carcinoids can occur in any part from the cardia to the pylorus, and invade the gastric layers  or adjacent tissues. It is always accompanied by lymph node invasion and distant metastasis, but GGT is usually a single benign tumor occurring in the gastric antrum, with no metastasis or invasion of surrounding tissues. Carcinoids usually have a specific structure of nuclear staining and positive neuroendocrine markers that can be used to distinguish between gastric carcinoid and GGT.Gastric hemangiomas are not true tumors; they belong to the vascular malformation, thus, CT features visible inside the tumor vein stone, can allow the differentiation from GGT.Gastric schwannoma mainly occurs in the body of the stomach, followed by a localization in the gastric fundus, while it is rarely localized in the gastric antrum. CT scan shows inhomogeneous low density, and cystic degeneration of the tumor. However, GGT mainly occurs in the gastric antrum, and CT scan shows no cystic degeneration.Paragangliomas occur in the retroperitoneum, while rarely occur in the stomach. Immunohistochemical staining shows positive synaptophysin, chromogranin A, and S-100 protein. Thus, it can be distinguished from GGT.Another rare tumor, the hemangiopericytoma, should also be taken into account. The histological characteristics consist of spindle cells with elongated nuclei, and the immunohistochemical staining usually results in a negative SMA, while glomus tumor appears as circular endothelial cells with the regular elliptic or circular nucleus.5.5Most of the GGTs are benign, can be cured by surgery, and with the development of various endoscopic techniques, lesions can be treated by endoscopic resections. Our patient's tumor was completely removed by surgery. No recurrence or metastasis was observed after 6 months of follow-up.6,14,20,21 The existing auxiliary examinations, such as the imaging examination and endoscopic ultrasound-guided fine needle aspiration have an important significance in the diagnosis and treatment of GGT. However, its final and correct diagnosis depends on the postoperative pathological and immunohistochemical analysis. Laparoscopic wedge resection of the stomach is considered as the first-line treatment. Clinical treatment should be based on the specific location and size of the tumor. In addition, a literature report shows that GGT can be treated with EUS; however, GGT contains abundant blood vessels, with high risk of intra-operative bleeding, and many primary hospitals in our country are unaware of this endoscopic treatment technique, thus, surgical treatment is preferred. Due to the lack of long-term follow-up, there have been no reports of patients’ prognosis and recurrence after surgery. Most of the GGT are benign lesions, can be cured by surgical resection and have a good prognosis. Several other case reports of malignant GGT are also available,–25 accompanied by certain characteristics of recurrence and metastasis. Thus, it is necessary for the patients carrying a potential malignancy to undergo surgical resection as soon as possible, with adjuvant therapies that might be useful. However, there is not any definitive diagnostic criterion for malignant GGT, thus, a large number of observations and studies are still needed to obtain definitive diagnostic evidence.GGT is rare, and previous studies indicate that it is more likely occurring in the gastric antrum of adults of any age with a significant female predominance, rarely reported in other parts of the stomach.Conceptualization: Xingcheng Wang.Data curation: Xingcheng Wang, Shahbaz Hanif, Binsheng Wang, Chen Chai.Formal analysis: Xingcheng Wang, Shahbaz Hanif, Binsheng Wang, Chen Chai.Project administration: Chen Chai.Resources: Xingcheng Wang.Software: Xingcheng Wang.Supervision: Xingcheng Wang.Validation: Xingcheng Wang.Visualization: Xingcheng Wang.Writing – original draft: Xingcheng Wang.Writing – review & editing: Xingcheng Wang.Xingcheng Wang orcid: 0000-0002-6980-9241.


Actinomyces israelii, a commensal of the bronchial and gastrointestinal tracts, is responsible for the majority of actinomycostic infections in humans. Actinomycosis has widely varying clinical presentations ranging from asymptomatic states to infiltrative mass lesions that mimic malignant abdominopelvic disease. Described as one of the most misdiagnosed diseases, actinomycosis poses challenges to accurate preoperative diagnosis. A 67-year-old woman with no significant medical history presented with features of acute intestinal obstruction. Computed tomography revealed a terminal ileal stricture causing intestinal obstruction and a right ovarian mass lesion. On laparotomy, a granular mass (2×2 cm) at the base of the mesentery and a right ovarian hard nodular growth (3×3 cm) were found that were connected by a dense fibrotic band, causing ileal obstruction with a transitional zone that was 10 cm proximal to the ileocecal junction. The mesenteric granular mass was excised together with the dense fibrotic band, and a right salpingo-oophorectomy was also undertaken. On postoperative histopathological examination, band formations by dense inflammatory tissue with neutrophilic infiltration were observed; moreover, there were sulfur granules that showed a positive reaction on Periodic Acid Schiff staining. The resected ovarian parenchyma showed infiltration by bacterial colonies with Splendore-Hoeppli phenomenon and evoked dense neutrophilic infiltration. The postoperative period was uneventful, and the patient was placed on penicillin therapy for a year. Abdominopelvic actinomycosis should constitute part of the differential diagnosis when evaluating mass lesions, especially in elderly women with a history of intrauterine device (IUD) use. Actinomycosis is a rare disease caused by the gram-positive anaerobic bacteria Actinomyces israelii, a commensal microrganism of the alimentary, respiratory, and urogenital systems. The sites most commonly affected by actinomycosis are cervical (50–65%) and abdominal 20%), followed by the chest 15%) [5% [1]. D0%, folloA 67-year-old woman presented to the surgical emergency department with complaints of diffuse, colicky abdominal pain, abdominal distension, obstipation, and multiple episodes of bilious vomiting for six days. Moreover, the patient had a history of recent weight loss and loss of appetite. There was no history of vaginal discharge or IUD insertion or any significant past medical or surgical conditions. On examination, the patient was conscious, oriented, and had tachycardia with normal blood pressure. The abdomen was distended, with diffuse tenderness and guarding. On auscultation, bowel sounds were exaggerated. Abdominal X-ray showed multiple air-fluid levels with loops of distended small bowel. Contrast-enhanced computed tomography (CT) revealed a terminal ileal stricture close to the ileocecal junction together with proximal dilated, and distal collapsed, bowel loops, suggestive of intestinal obstruction. After optimal hemodynamic resuscitation, the patient underwent exploratory laparotomy under general anesthesia. Intraoperatively, we found a granular mass (2×2 cm) at the base of the mesentery and the right ovary with a hard nodular growth mimicking a malignancy (3×3 cm) Figure .A dense fibrotic band extended between the two masses, causing ileal obstruction, and a transitional zone was present 10 cm proximal to the ileocecal junction Figure .Both the mesenteric granular mass and the dense fibrotic band were excised, a right salphingo-oophorectomy was conducted, and resected specimens were sent for histopathological examination. On microscopic examination, the Actinomyces species was identified in the evaluated specimens, and a final diagnosis of abdominopelvic actinomycosis was made Figures -3E.Actinomyces israelii is a saprophyte occurring commonly in the oral cavity, female urogenital system, and alimentary tract. Various predisposing factors could disrupt the normal barrier mechanism, leading to pathogenicity by commensals. The predisposing factors most often encountered include trauma, surgery, endoscopic procedures, chronic inflammatory processes, and immunocompromised status secondary to malignancy or diabetes mellitus or following a renal transplant .Despite the widespread use of IUDs, which can lead to pelvic infections, abdominal/pelvic actinomycosis is a rare occurrence. Pelvic actinomycosis has been associated with IUD use in 80% of cases and is more frequent in those with an IUD usage duration of more than four years .Clinical symptoms are often nonspecific, and the condition manifests with acute, subacute, or chronic presentations. Patients can present with acute abdominal pain, vomiting, constipation, and obstipation, as in the present case. Subacute conditions may present with symptoms of malabsorption, vague abdominal pain, loss of appetite, and weight loss. In chronic infection, actinomycosis may present with mass formations that mimic malignancy and strictures or fibrotic bands that lead to intestinal obstruction and perforation . Some paAbdominopelvic actinomycosis is difficult to diagnose preoperatively. Routine blood biochemistry and hematology rarely give a clue to this diagnosis. Ultrasonography may be used as an initial imaging technique that can exclude other common conditions. A plain abdominal radiograph may show features of intestinal obstruction and perforation that can aid symptomatic diagnosis. On contrast enhanced CT, bowel wall thickening and a homogenous enhancement pattern are detected more often. Some patients can have abdominal or pelvic masses that, often, may appear as a heterogeneous enhancement .Abdominal actinomycosis can be included in the differential diagnosis when a CT scan shows bowel wall thickening, strictures, and a mass. This is especially noteworthy when there are non-specific signs in women with a history of IUD use . VariousIn the present case, CT showed stricture formation proximal to the ileocecal junction with dilation of proximal bowel loops and collapse of distal loops, which necessitated emergency surgery. In most cases, preoperative diagnosis is difficult as in the present case.The final diagnosis was aided by histopathological examination of the surgical specimen. On examination, sulfur granules may be present, although it is not a pathognomic feature ; the graThe primary establishment of a diagnosis of abdominopelvic actinomycosis is difficult. Clinical presentation is frequently variable, and presenting features may mimic a malignancy. Radiological imaging can reveal mass lesions that may be associated with a fluid collection, but it is difficult to distinguish among Crohn’s disease, tuberculosis, pelvic inflammatory disease, diverticulitis, and malignancy; abdominopelvic actinomycosis should be included as a differential diagnosis when evaluating these conditions. Despite diffuse disease, antibiotics either alone or in combination with surgery can cure the disease.