Lung cancer still remains one of the most common cancers throughout the world, especially in smokers. Adenocarcinoma is now the predominant histological type in many western countries. The etiology of adenocarcinoma is unknown, but evidence suggests that atypical adenomatous hyperplasia (AAH) may act as a precursor lesion. Here we present two case reports of patients diagnosed with AAH on biopsy, highlighting 1) available treatment strategies and 2) AAH’s progression to adenocarcinoma. A review of AAH is warranted as little literature is currently available regarding its treatment strategies, especially in light of its role as a precursor to adenocarcinoma.In this review, we will address the following topics:1. What is the pathophysiology of AAH?2. What is the natural history of AAH and its risk of malignant transformation?3. When is surgery recommended?4. What is the role of stereotactic body radiotherapy (SBRT) in the rare patient who refuses surgery? Lung cancer is one of the most common malignancies in the world, especially in smokers. Pulmonary adenocarcinoma is the most common cell type, though the exact etiology of adenocarcinoma is unknown. Pulmonary atypical adenomatous hyperplasia (AAH) may act as a precursor lesion to adenocarcinoma . Some paA number of different terms have been used in the literature to refer to AAH. These include but are not limited to the following: atypical alveolar epithelial hyperplasia , atypicaSome studies have suggested that the grade of atypia in AAH is less than that of adenocarcinoma. However, the definitive histologic criteria for diagnosis of AAH remain to be defined. In fact, the distinction between AAH with severe atypia and well-differentiated bronchioloalveolar carcinoma (BAC) can be very difficult on the basis of histopathology alone. The diagnosis is frequently made using a combination of radiographic and clinical findings. The natural history of AAH is poorly understood. Many clinicians regard AAH as a precursor lesion of BAC ,7, whileHere, we present two patients' cases that 1) demonstrate the different options for AAH treatment and 2) demonstrate AAH acting as a precursor lesion to adenocarcinoma. In addition, this manuscript aims to discuss the epidemiology and recent advances in the understanding of AAH, which may help diagnostic pathologists recognize AAH. This review further discusses studies that have used various ancillary methodologies such as morphometry, immunohistochemistry, cytogenetic methods, and molecular analyses.A 65-year-old man with a 60-pack-year smoking history presented with a right middle lobe ground glass opaque (GGO) nodule which measured 1.9 cm. One year later, the nodule progressed in size to 2.8 cm Figure . CT scanIn another case, a 73-year-old female with a long smoking history was referred to thoracic surgery for a slow-growing nodule in her left upper lobe. A CT scan showed a 2.3 x 1.3 x 0.9 cm GGO nodule in the anterior left upper lobe Figure . At thisUpon presentation to thoracic surgery, the patient continued to smoke. She denied any symptoms of cough, weight loss, or change in appetite. Given the location and concerns for potential malignancy within the previously identified atypical adenomatous hyperplasia, the patient was consented for lobectomy. The patient underwent an uneventful left upper lobectomy and lymph node dissection. Final pathology showed two foci of invasive moderately differentiated adenocarcinoma, acinar pattern Figure -3D. MultTogether these cases demonstrate the varying opinions on clinical management of AAH and to date, there is no empiric treatment for AAH. Traditional treatment options include surgical resection or observation with serial CT scans. The role of SBRT is unknown due to lack of data on the use of SBRT in treating AAH. In addition, the latter case demonstrates that AAH can act as precursor lesion of adenocarcinoma.Adenocarcinoma is now the predominant histologic type of lung cancer in many western countries. Clearly, an understanding of possible precursor lesions for this increasingly frequent lung cancer would help establish a diagnostic classification, as well as guide management strategies. Many investigators have suggested that AAH is a precursor lesion of adenocarcinoma . The WorEpidemiology and pathologyPopulation-based epidemiologic studies addressing the frequency of AAH have not been reported. However, there are some studies that provide some data on the frequency of AAH in hospital-based populations. Miller examined 247 cases of surgically resected lung carcinomas by sectioning all areas of the lung transversely at 1.0- to 1.5-cm intervals . They foOther means to assess AAH have been used, such as morphometry, immunohistochemistry, and molecular studies; though, some morphometric, molecular analyses and immunohistochemical studies are not always available to many practicing pathologists. Therefore, diagnosis of AAH will likely continue to rest on subjective histologic criteria. Some authors suggested the following guidelines for the diagnosis of AAH: prominent cuboidal to low columnar alveolar epithelial cells with different degrees of atypia, less than that seen in the more common adenocarcinoma; irregularly bordered focal proliferations of atypical cells spreading along the pre-existing alveoli; increased cell size and nuclear-cytoplasmic ratio with hyperchromasia and prominent nucleoli; intercellular attachment of atypical cells generally intact with occasional existence of empty-looking spaces between them without high cellularity.On the other hand, any precursor lesion of a cancer should fulfill certain criteria . First, Guidelines for screening, follow-up, and interventionThe current recommendation of low-dose CT screening for patients at risk will undoubtedly pick up more pulmonary lesions; some of these lesions eventually will be AAH. Current guidelines for lung cancer screening from the US Preventative Services Task Force recommend annual screening with low-dose CT scan for adults 55-80 years of age who have at least a 30-pack year smoking history. The patient must be a current smoker or have quit within the last 15 years. Screening ends if the patient has not smoked for 15 years, develops a life limiting illness, or becomes unable (or unwilling) to undergo curative surgery . HoweverAAH is nearly indistinguishable from focal inflammation and BAC, with each appearing as ground glass opacities (GGO) . RegardiManagement of pulmonary AAH varies, with a conservative approach recommended by Ritter . This coThe diagnostic guidelines for AAH for the pathologist can be controversial. Similarly, managing this condition once diagnosed is also difficult, and there are no definitive guidelines. Previous authors have discussed treatment options ranging from conservative observation to surgical resection. Some studies show observation is safe with compliant patients who show up for routine CT surveillance at appropriate frequency . The surgical series showed that AAH lesions were removed with a variety of procedures ranging from wedge resection, segmentectomy, to lobectomy .Currently, there is very little data supporting the use of stereotactic body radiation therapy (SBRT) for pulmonary AAH. On the rare occasion of patients with pulmonary AAH, which carries a 10-15% chance of harboring malignancy, SBRT may be justified in the non-compliant patients that may be lost to follow-up.Is AAH a pre-malignant lesion? The current data suggests that AAH plays a role in the development of pulmonary adenocarcinoma, particularly in the development of BAC. Further work is needed to establish widely acceptable histopathologic criteria for the diagnosis of AAH. In addition, further study is warranted on the role of SBRT for patients with AAH who deny surgical intervention. The intriguing nature of pulmonary AAH in the development of pulmonary adenocarcinoma deserves further study.


This case demonstrates angiodysplasia as an impending source of occult gastrointestinal bleeding.•According to most authors histological diagnosis of angiodysplasia is based on the presence of mucosal vascular ecstasies with associated prominently dilated capillaries penetrating the muscularis mucosae. In this case these criteria are satisfied; however presence of large, thick walled vessels in submucosa is not described in the classic case of angiodysplasia.•In this case report: I am discussing this case and making a comparison with the ususal presentation of angiodysplasia, how to diagnose obsure bleeding.•Role of the radiological as well as triphasic CT scan finding to diagnose the angiodysplasia of the small intestine.•Incidine of the angiodysplasia in the small intestine. Angiodysplasia is the second most common vascular abnormality of the GI tract after diverticulosis and is the second principal cause of lower GI bleeding over 60 years.A 68-year-old male patient presented to our institute in the outpatient clinic with pallor of gradual onset and progressive course since 6 months. Detailed history revealed hematochezia, sometimes melena. Physical examination revealed signs of iron deficiency anemia which was confirmed by laboratory results and hemoccult positive stool. The patient refused the colonoscopy. A bdominal CT with contrast was then performed which revealed an evidence of abnormal subtle mural contrast flocculation within the small bowel loop with luminal extravasations of contrast at small segment of distal ileum few centimeters from ileocaecal valve with mildly dilated and early filling of its drained vein, this abnormal contrast extravasations and flocculation increased through dynamic study. In addition, evidence of contrast mural flocculation within the left lateral wall of distal few centimeters of rectum was also observed. This abnormal mural contrast flocculation within the wall of solitary segment terminal ileum and distal rectum suggest the evidence of venous ectasia or angiodysplasia. Surgical resection was done with end to end anastomosis of the small intestine without any complication in the postoperative progress.This case also emphasizes the critical significance of a mutual multidisciplinary approach to occult gastrointestinal bleeding. The management of angiodysplasia is considered as multidisciplinary team work that would consist of expertise from different departments as in endoscopy, angiography, and gastrointestinal surgery. It is c2A written consent was taken from patient for publication of the presenting case. A 68-year-old male patient presented to our institute with pallor of gradual onset and progressive course since 6 months. Detailed history revealed hematochezia, sometimes melena. Physical examination revealed signs of iron deficiency anemia which was confirmed by laboratory results and hemoccult positive stool. The patient refused the colonoscopy. A bdominal CT with contrast was then performed and the patient was subjected to preprocedure preparation for 3 days before examination in polyethylene glycol as a bowel-cleansing was administered and low-fiber diet was taken. Additionally, a water-soluble iodinated contrast medium was administered orally for fecal and fluid tagging. CT images were then obtained which revealed the followings: evidence of abnormal subtle mural contrast flocculation within the small bowel loop with luminal extravasations of contrast at small segment of distal ileum 30 cm from ileocaecal valve with mildly dilated and early filling of its drained vein, this abnormal contrast extravasations and flocculation increased through dynamic study. In addition, evidence of contrast mural flocculation within the left lateral wall of distal few centimeters of rectum was also observed . This ab3Angiodysplasias are a frequent cause of gastrointestinal bleeding with a diverse clinical manifestations ranging from symptoms and signs of iron deficiency anemia to occult bleeding and even life-threatening bleeding . This caAccording to most authors histological diagnosis of angiodysplasia is based on the presence of mucosal vascular ecstasies with associated prominently dilated capillaries penetrating the muscularis mucosae. In this case these criteria are satisfied; however presence of large, thick walled vessels in submucosa is not described in the classic case of angiodysplasia. The differential diagnosis of angiodysplasia includes other varieties of telangectasias, arteriovascular malformations and haemangiomas. Mucosal lymphangiectasia superficially resembles angiodysplasia but the vessels do not contain blood and there is no associated submucosal venous ectasia ,15.Angiodysplasia is often segmental or even multifocal, and therefore a careful inspection of the whole bowel should be carried out during the instance of exploration. For lesions found in the right colon which is considered the most common site a right colectomy is highly recommended . There aMedical therapies including long-acting somatostatin analogues have been shown to diminish the number of re-bleeding on patients with intractable small bowel angiodysplasia . Therapy4This case also emphasizes the critical significance of a mutual multidisciplinary approach to occult gastrointestinal bleeding. The management of angiodysplasia is considered as multidisciplinary team work that would consist of expertise from different departments as in endoscopy, angiography, and gastrointestinal surgery. In addition, council from all these departments should be present at the time of operation when surgery is recommended to optimize the time of finding the difficult lesions and wholly enhanced the chance for achievement.No source of funding and it is my personal effort.The case report is approved from the ethical committee in the college of medicine – Al Baha university.The fully informed written consent is taken from patient.I did as a single author the study concept, data collection, data analysis and interpretation, and I wrote the paper.N/A.Dr Thamer Alghamdi.Not commissioned, externally peer-reviewed.No financial and personal relationships with other people or organisations that could inappropriately influence their work.


Intrascrotal lesions are common findings with a majority occurring in paratesticular tissue. Fibrous pseudotumors are rare, benign lesions of the testicular tunics and present with mass lesion(s) in the scrotum. Preoperative clinical and radiological diagnosis is challenging. We report a case of a 34-year-old man who presented with a 3-year history of left testicular swelling and was advised left radical orchidectomy by another surgeon. Physical examination revealed a firm, nontender mass attached to the lower pole of the testis. Testicular tumor markers were all negative, and ultrasound scan showed a relatively hypoechoic lesion closely associated with the left testis and suspicious for neoplastic process. The patient underwent a testicular sparing surgery. An intraoperative frozen section biopsy confirmed the lesion to be benign and this was reported on permanent section to be fibrous pseudotumor of the tunica albuginea. We also present the clinical, sonographic, and histopathological findings of this condition along with the literature review. Intrascrotal lesions are common findings in the male population with a majority occurring in paratesticular tissue. Contrary to testicular lesions which are 95% malignant, most paratesticular lesions are benign , 2. FibrComprising only 6% of paratesticular tumors, these lesions pose a diagnostic and therapeutic dilemma due to their confusion with malignant lesions .Preoperative diagnosis of fibrous pseudotumors is challenging both clinically and radiologically . Many ofWe report case of a young man who came for a second opinion at our clinic for left scrotal swelling for which he was advised a left radical orchidectomy by another surgeon. Surgeons should be aware of this rare condition and must do a careful evaluation to prevent unnecessary removal of the testis.A 34-year-old man, father of 2 kids, presented to urology outpatient clinic complaining of swelling in the left testis for the last 3 years. The swelling gradually increased in size and was not associated with any pain or fever. The patient also denied prior history of any trauma, infections, or scrotal surgeries. The past medical and surgical history was otherwise unremarkable.Physical examination revealed a circumcised penis with bilateral developed hemiscrotum and normally descended testes. The right testis was normal, and the left testis had a firm, nontender 2.5 × 2.0 cm smooth mass attached near its lower pole with an associated lax hydrocele.N = 208–378), alpha feto protein (αFP) 4.0 IU/ml (N ≤ 6.7), and beta human chorionic gonadotropin (βHCG) <2.0 mIU/ml (N < 10). An ultrasound scan was done which showed a 23.4 mm × 22.6 mm well-circumscribed lesion closely associated with the left testis. On color Doppler, no significant vascularity was observed  275 IU/l , the lesions have intermediate-to-low signal intensity on T1W sequence (similar to that of the testis) whereas they have low signal intensity on T2W images . There iOn gross appearance, they are well circumscribed and white-tan or yellowish in color and can be bulging or whorled on the cut section . On micrThe treatment of choice for paratesticular fibrous pseudotumors is surgical excision. Intraoperative frozen section biopsy is recommended when the testis is involved with the tumor  and can


A 23-year-old woman presented with a 1 year of slow-growing soft tissue nodule on the volar aspect of the base of the left index finger . GradualWhat is the differential diagnosis of a hand mass?What is fibroma of the tendon sheath?What is the clinical presentation of fibroma of the tendon sheath?What is the appropriate treatment?,Certain tumors are more likely to be located in the soft tissue in the hand than in other parts of the body. All forms of soft tissue lesions must be considered in the differential diagnosis. This includes lipoma, lipofibromatous hamartoma, epidermal cyst, neuroma, angiomyolipoma, nonskeletal chondroma, synovial sarcoma, ganglion cyst, Dupuytren disease, fibromatosis, and tenosynovial giant cell tumor. The clinical features of tenosynovial giant cell tumor are particularly similar to those of fibroma of the tendon sheath (FTS). Both are likely to occur in the fingers with attachment to tendon sheath, possess similar magnetic resonance imaging (MRI) signals, and have firm, well-circumscribed, multilobulated gray-white appearance1Fibroma of the tendon sheath is an uncommon, benign, slow-growing, solitary, neoplastic condition consisting of well-circumscribed, lobulated fibrocartilaginous tissue attached to tendon or tendon sheath. It typically develops in the hands, wrists, and fingers but can also occur, albeit rarely, in the lower extremities. The volar aspect of the thumb, index, and middle fingers is the commonest site of origin. FTS commonly presents within the third to fifth decades of life, with a predominance in males over females (M:F ratio of 3:1).The clinical presentation of FTS often occurs years after its formation as a painless, slowly growing mass that may irritate the surrounding tissues by compression.-About one-third of the cases present with tenderness and mild pain due to compression of the nerves underlying FTS.5Less than 10% of patients have reported a history of trauma.Treatment consists of local excision with preservation of important anatomical structures. The excision of the tumor is difficult at times because of its adherence to tendinous structures, and it is aimed to relieve symptoms but preserve function . Approxi


Cystic pancreatic neuroendocrine tumor accounts for 13-17% of pancreatic neuroendocrine tumors and 9-10% of resected cystic tumors. This unusual variant can make clinical diagnosis particularly challenging. A 28-year-old female patient presented with abdominal pain and dyspepsia. General and physical examination was unremarkable. On computed tomography scan a 7.5×5.4×5.2cm thick-walled cystic lesion was noted in the tail region of the pancreas in the stomach bed (A). On the basis of the above findings, the radiologist reported it as suggestive of solid pseudopapillary neoplasm and advised pathological correlation. No abdominal lymphadenopathy was noted. The patient underwent laparoscopic removal of a pancreatic cyst. Intraoperatively, a large cyst was seen located posterior to the stomach in retroperitoneum and was intimately related to the tail of the pancreas. The cyst could be enucleated. On histopathological examination, the sections from the cyst wall showed collagen-rich fibrous tissue in which were embedded small clusters and nests of round cells (B). The cells were monotonous, small to medium in size with amphophilic cytoplasm with indistinct cell borders. The nucleus showed typical stippled nuclear chromatin and no mitoses were noted (C). There was no capsular invasion. Neuroendocrine nature of these round cells was confirmed by performing immunohistochemistry with synaptophysin and chromogranin A (C). Ki67 stained <3% of the cells. These findings corroborated the benign nature of the disease indicating a very good prognosis. Postoperative recovery of the patient was uneventful


Clinical manifestations of liver transplantation complications can be subtle and non-specific. Medical imaging, mainly Doppler ultrasound, plays an important role to detect and grade these. Colour Doppler ultrasound exams are routinely performed at 24–48 h, on the 7th day, the first and third month after transplantation. MDCT and MR images are acquired based on the Doppler ultrasound (DUS) findings, even in the absence of abnormal liver function. As vascular complications appear early after surgery, DUS should be performed by experience personnel. Diagnostic angiography is seldom performed. This pictorial review illustrates the key imaging findings of vascular complications in patients with liver transplantation: hepatic artery complications , portal vein abnormalities (such as occlusion and stenosis) and hepatic veins and/or inferior vena cava flow changes (Budd-Chiari syndrome). Highlight the importance of early detection of vascular complications after liver transplantationDescribe key colour Doppler ultrasound findings as the initial imaging test.Review the CT and MR imaging findings of the arterial and venous complications.Currently, liver transplantation is the first-line treatment for patients with terminal liver disease, both acute and chronic.In recent years, living donor liver transplant has been introduced, especially in children. It reduces the waiting period for a deceased donor transplant and also the ischemic period of the transplanted organ . In thisVascular complications after transplantation are infrequent. Their reported incidence is close to 7% for cadaveric donor liver transplantation and around 13% for living donor liver transplantation . UnfortuColour Doppler ultrasound (CDUS) is the most appropriate imaging test, allowing the early evaluation of the patient, even within the recovery unit after surgery, and also precisely assessing the graft vessels patency . When CDTraditionally, surgery has been the first-line treatment of complications, although, nowadays, endovascular treatments have been positioned as first options, limiting surgery to those cases where interventional radiology is limited or failed . AbdominPostoperative CDUS at 24/48 h and day 7 after surgery Fig. . These eContrast-enhanced CT images are obtained when a vascular lesion is observed on CDUS images or when the liver function is impaired. Occasionally, MR is performed if contraindications to contrast-enhanced CT are present.T-tube cholangiography images with direct opacification are also performed on day 4 and the third month to assess the biliary tree.Regarding vascular imaging evaluation, the following protocol is used in our institution, where more than 100 cadaveric liver transplants are performed every year:In recent years, hepatic intra-operative ultrasonography has emerged as a new technique. It allows real-time evaluation of the anastomosis and so immediate treatment before abdominal closure .Usual posttransplantation findings include right-sided pleural effusion, ascites, perihepatic hematoma and periportal oedema. All of them should resolve in the first weeks after surgery .Vascular and biliary anastomoses should be assessed by CDUS. Radiologists should have a clear knowledge of the individual patient postoperative anatomy, since anastomoses are the locations where complications occur most frequently. Moreover, it is important to be aware of the anatomic variants, both in the donor and in the recipient .Currently, the most common surgical technique is orthotopic liver transplantation, where the graft is placed in the right upper quadrant, at the anatomical liver location, after removal of the native liver . Four anThe most frequently affected anastomosis is at the hepatic artery . This anRelated to their onset, hepatic artery complications can be defined as early (within the first month) or late (later than 1 month) complications. Early complications are the most important for patient prognosis because they are associated with graft loss and a high mortality rate.Artery thrombosis is the most serious complication of orthotopic liver transplantation, occurring approximately in 4 to 12% of cases . ThromboPulsed and power CDUS has a high sensitivity and specificity to detect the thrombus and grade the degree of stenosis and flow properties, not only at the hepatic artery but also at the intrahepatic branches. Parameters to be considered are hepatic artery diameter, hepatic artery peak systolic velocity and hepatic artery resistance index. Intraoperative ultrasound examination has high sensitivity and specificity for early detection of these findings . Proper When the blood flow is not identified by CDUS, some different conditions should be considered such as slow flow secondary to vasospasm or low cardiac output. Also, many of these patients have a poor sonographic window due to the surgical dressing materials. Use of a contrast agent  will improve the US diagnostic performance in these cases with a near perfect accuracy .CT angiography is the best technique to further evaluate difficult cases due to its high accuracy, short examination time and facility to be performed with poor patient condition . MRI hasIf thrombosis is suspected, a diagnostic arteriography will confirm the diagnosis and allow the best treatment decision. Some endovascular treatments are available to these patients, such as intraarterial thrombolysis (IAT), percutaneous transluminal angioplasty (PTA) and stent placement . If treaThe most frequently affected location is the anastomosis (2 to 13% of patients) . TherefoStenosis may progress to thrombosis. So, stenosis and thrombosis are two entities of the same spectrum of vascular complications of liver transplantation.Stenosis can lead to splenic steal artery syndrome .It usually occurs in the first 3 months (median time to diagnosis 90 to 120 days)  25], bu, bu25], Doppler ultrasound is the most useful technique to show this complication, as explained for thrombosis.Prestenotic segment shows elevation of resistance index (more than 0.8) and low flow.Stenotic segment displays a very high flow rate and aliasing artefact, due to the turbulent flow. Blood systolic peak is more than 200 cm/s.Poststenotic segment presents a low resistance index (less than 0.5) and a parvus et tardus morphology of the spectral curves with long systolic acceleration time (more than 0.08 s). It has also a turbulent flow.It shows a characteristic pattern, depending on the segment studied and its relationship with stenosis. Power Doppler is also useful to quantify blood flow and study spectral curves .PrestenoIt is important to remember that, in the first 3 days after liver transplantation, an increased resistance index of the hepatic artery (greater than 0.8) is found in approximately 50% of the patients .If found, it should be monitored until it has normalised, typically in the fourth day after the transplantation .Although the severity of the described findings correlates with the degree of the stenosis, ultrasound does not allow proper quantification of this: the technique of choice is CT angiography . In addiMRI angiography is a limited technique because of a relatively high false-positive rate .Hepatic artery stenosis requires early treatment. First, an angiography and an angioplasty should be made. If this procedure fails, surgery is required. Once again, retransplantation has a better outcome, but is preferred as a second-line treatment, to use when endovascular therapy does not work .Pseudoaneurysm of the hepatic artery and its branches presents the same features as in other parts of the body, differently to other vascular complications of liver transplantation. In this case, it can affect any of the branches, not only the site of the anastomosis.It is a rare complication, with an incidence of 2.5% of the cases of liver transplantation, according to the retrospective study having the largest sample. It has no bias for any of the indications of liver transplantation .In ultrasound, pseudoaneurysms present as a hypoechoic structure with turbulent blood flow within as observed by colour Doppler. Typically, due to swirl formed by the inlet and blood outlet, it is possible to observe the yin-yang sign .If CT angiography is performed, it characteristically presents an arterial enhancement equal to the other arterial vessels, with an equal wash out in the later stages.They are classified according to their location: extrahepatic and intrahepatic.The most common site is the arterial anastomosis. They can occur spontaneously or as a complication of treatment of a preexisting stenosis. Bacterial or fungal infection isolated from the peritoneal fluid or from the pseudoaneurysm wall can be present in up to 81% of cases, according to series .Characteristically, it is a complication of percutaneous liver biopsy, the usual cause, but also may be secondary to bile duct infections . In caseIn both cases, treatment consists of coil embolisation and stent placement to prevent inflow to the pseudoaneurysm. If the results of this treatment are not satisfactory, surgical resection can be performed .Hepatic infarction is very rare in normal patients, since the liver is a richly vascularised organ with blood from different circuits: the hepatic artery and the portal vein. Inside the liver, there are numerous anastomotic vessels and collateral branches.However, in liver recipient patients, anastomoses are stopped, so hepatic infarction is much more common. It is usually associated with arterial occlusion (85% of cases), and, rarely with portal vein occlusion . Bile duIschaemia and liver infarction can be consequences of all three described alterations of the hepatic artery: thrombosis, stenosis and pseudoaneurysm.Portal vein complications are infrequent. They affect less than 2% of liver transplantations .The most common surgical technique is to directly anastomose the portal vein of the donor with that of the recipient.However, sometimes this is not possible, because there is a portal thrombus which prevents direct anastomosis. In such cases, it is necessary to remove the segment occupied by the thrombus and perform a bypass. Typically, the donor iliac vein is used to make the bypass .Complications of the portal vein usually affect the anastomosis, so it is important to know its location.The most common complications of the portal vein will be explained.It is a rare complication. It occurs most often in the extrahepatic portal vein, at the site of the anastomosis. It can be proved by the absence of Doppler flow on ultrasound examination or a filling defect in the contrast-enhanced CT scan or MRI.Some studies show that contrast-enhanced ultrasound is a promising technique being able to identify cases of thrombosis missed by other imaging techniques .If thrombosis is seen in the first 72 h after surgery, surgical revision of the anastomosis should be made. Later, the treatment is done with percutaneous thrombolysis, angioplasty, stenting or, if unable to perform the treatment with these less invasive techniques, surgery.Doppler ultrasound, there is typically an increase in portal blood flow velocity at the point of the anastomosis (greater than 125 cm/s) or three times higher at the site of the stenosis in comparison with the prestenotic segment . As explAn aliasing artefact can also appear in the stenotic segment because of turbulent flow. This can be also a normal finding in the early postoperative period. So, in the case of turbulent flow, anastomosis should be evaluated in future controls, for comparison .CT or MR angiography can observe and quantify the degree of stenosis.The site most frequently involved is the anastomosis. The characteristic findings in each imaging technique are as follows:Stenosis should be carefully differentiated from a physiological mild reduction in vessel calibre at the anastomotic site. This finding in more appreciable if there is a size discrepancy between the donor and recipient portal veins. In this case, the focal narrowing is a normal finding and it is not related to stenosis. Knowledge of preoperative anatomy and assessment of the graft will help to make the differential diagnosis. Nevertheless, this finding should be followed-up, because it predisposes to the development of a stenosis . TreatmeAlthough much more common in the case of arterial complications, it may also occur as a result of stenosis or portal vein thrombosis.Inferior vena cava complications are in frequent. They affect less than 2% of liver transplantations . CoronalUsually, they occur because of the surgical technique or a hypercoagulable state. Diagnosis and treatment are similar to that of portal vein thrombosis.Just as in the other vessels, the most common site is the anastomosis. It is also possible that extrinsic compression stenosis occurs, secondary to oedema of the graft or fluid collections, hematomas or abscesses. The diagnostic and therapeutic techniques are similar to those described in the portal vein.These are rare complications. As in the rest of the vessels, characteristic complications are thrombosis (Budd-Chiari syndrome) and stenosis.In the case of living donor transplantation, knowledge of the surgical anatomy is very important. Usually, the right hepatic vein is preserved in the right lobe of the graft, but the middle hepatic vein is usually left for the safety of the donor. Sometimes, the middle hepatic vein is also included in the graft, and therefore, it should be assessed in the postoperative control .Early detection of vascular complications of liver transplantation is essential in establishing effective treatment: this determines the effectiveness of transplantation and patient mortality and morbidity.Doppler ultrasound is the initial imaging test. If it does not allow definitive diagnosis, other techniques  are indicated.


Müllerian duct anomalies represent a group of congenital malformations that result from failure to complete bilateral paramesonephric duct elongation, fusion, canalization, or septal resorption. These anomalies are rare in the general population with a bicornuate or didelphys uterus being among the common ones. Bicornuate uterine malformations are of clinical significance due to their adverse reproductive outcomes. Metroplasty has been shown to improve reproductive outcomes of bicornuate uterine malformations. We document a case of bicornuate uterus that was managed with Strassman metroplasty and a subsequent successful pregnancy outcome. A Black African lady was seen with a history of six prior miscarriages. Her diagnostic workup revealed a bicornuate uterus for which she had a Strassman metroplasty performed. She later conceived and was followed up to term with a successful live birth. Strassman metroplasty is a rare procedure in Sub-Saharan Africa and this case seeks to add to the body of knowledge on surgical management of Müllerian duct anomalies specifically bicornuate uterus in this region. This case report aims to increase the awareness of Müllerian duct abnormalities specifically bicornuate uterus in cases of recurrent miscarriages and highlight the diagnostic strategies to investigate and to demonstrate management options in low resource settings. Müllerian duct anomalies represent a group of congenital malformations that result from failure to complete bilateral paramesonephric duct elongation, fusion, canalization, or septal resorption . These aThe prevalence of these malformations in general population is approximately 5%. It is 5–10% in those suffering from recurrent miscarriages and greater than one in four in women with late pregnancy losses and preterm deliveries . Among tBicornuate uterus is caused by incomplete fusion of the Müllerian ducts. It is characterized by two separate but communicating endometrial cavities and a single uterine cervix. Failed fusion may extend to the cervix resulting in a complete bicornuate uterus or may be partial, causing a milder abnormality. Pregnancy outcomes in patients with these anomalies are not favorable. Previous reports have demonstrated that those with bicornuate uterus have higher rates of miscarriages and preterm delivery  . The cliUterine malformations are rare occurrences worldwide and surgical management of these cases is not well documented in Sub-Saharan Africa perhaps due to challenges in diagnosis or treatment. We present a case of a patient with a bicornuate uterus that was surgically managed with open Strassman metroplasty with a successful subsequent pregnancy.A 35-year-old para 0+6 Black African lady presented with a history of five first and one second trimester recurrent pregnancy losses. In addition, she had a nine-year history of irregular heavy bleeding associated with dysmenorrhea.Her menarche was at 17 years of age with regular painful cycles that lasted 10 days. She was not on any contraceptive method and did not report any dyspareunia or urinary symptoms. Her first miscarriage occurred 11 years and was surgically managed by dilatation and curettage. Subsequently she noted changes in her menstrual cycle. Her menstrual cycle became irregular with a heavy flow for 10 days associated with severe dysmenorrhea and bowel symptoms of bloating and diarrhea. She used tranexamic acid one gram three times a day during her menses for the heavy prolonged periods and Mefenamic Acid 500 milligrams three times a day for the dysmenorrhea with reported relief of the symptoms.The physical examination was unremarkable except for mild tenderness in the suprapubic region; she had grossly normal external genitalia and normal looking cervix.  FSH: 4.5 IU/m (3.1-7.9 IU/L)  LH: 10 IU/L (1-18 IU/L)Her initial hormonal profile was as follows: She had a recent pap smear one year ago that was normal.Transabdominal and transvaginal (TVS) scans had shown separate right and left cornu with multiple cysts in the peripheral ovarian parenchyma features suggestive of bicornuate uterus and polycystic ovaries . She alsHer past medical history was not significant. She was recently divorced, a condition she attributed to her history of several miscarriages in the context of an African cultural expectation of siring children.She was subsequently scheduled her for hysteroscopy and diagnostic laparoscopy. She also gave consent for possible open Strassmans metroplasty.On hysteroscopy, a normal looking endometrium was visualized with a right sided tubal ostia and a small tubal ostia on the left that did not appear patent . LaparosShe had an unremarkable follow-up after surgery with no complications reported.A HSG 6 months after surgery revealed unification of the uterine horns but both tubes were not delineated.Patient was lost to follow-up but two years later she presented having conceived spontaneously and a TVS revealed a single intrauterine pregnancy at 13 weeks. Her antenatal workup was as highlighted in She was subsequently followed up at the antenatal clinic and had no concerns till 33 weeks when she presented with a history of lower abdominal pain worsening over the past few weeks and right sided fundal tenderness pain score of seven out of ten on the pain assessment scale. The main concern at the time of assessment was possible uterine rupture. She did not have any features suggestive of bowel obstruction as she was passing stool and bowel sounds were present. She had a normal NST and an obstetric ultrasound that was done showed a single intrauterine pregnancy with normal biophysical profile and no features of placental abruption or slower uterine segment site dehiscence. She had no other investigations done at this time. The pain was significant enough to warrant an admission for which she subsequently received antenatal steroids for lung maturity and opioid analgesia for pain relief. However, the pain did not completely resolve and she underwent a caesarean delivery at 33 weeks and 5 days due to persistent lower abdominal pain. The outcome was a live male infant with a birth weight of 1950 grams and APGAR of 9, 10, 10. There was no evidence of uterine rupture although she had venous congestion and massive varicose veins bilaterally at the cornual areas. The previous metroplasty scar was intact.She had a resolution of the abdominal pain postoperatively and had an unremarkable postoperative recovery. She was discharged on the fifth postoperative day.Müllerian duct anomalies represent a group of congenital malformations that result from failure to complete bilateral duct elongation, fusion, canalization, or septal resorption .Various classification schemes for female reproductive tract anomalies exist, but the most common classification was proposed by Buttram and Gibbons (1979) and adapted by the American Society for Reproductive Medicine (ASRM) . Within this system, six groups are elucidated: segmental Müllerian hypoplasia or agenesis (Group I), unicornuate uterus (Group II), uterine didelphys (Group III), bicornuate uterus (Group IV), septate uterus (Group V), arcuate uterus (Group VI), and diethylstilbestrol related anomalies (Group VII) . The casAmong the Müllerian duct anomalies, bicornuate and didelphic uteri are common forms, with the prevalence of 25 and 11%. (WT1), paired box gene 2 (Pax2), WNT2, pre-B-cell leukemia transcription factor 1 (PBX1), and homeobox (HOX) genes [There are no specific risk factors highlighted in literature that predispose to these abnormalities. Uterine congenital anomalies have a heterogeneous genetic basis, with implications of Wilms tumour 1 geneX) genes . In the Most cases are diagnosed during evaluation for obstetric or gynecologic conditions, but in the absence of symptoms, most anomalies remain undiagnosed . In thisThe question of whether Müllerian anomalies are significantly more often combined with endometriosis is a controversially discussed problem. Some publications described this association in patients with obstructive but not nonobstructive Müllerian anomalies or controls without Müllerian anomalies. It is well known that obstructive Müllerian anomalies are significantly more often associated with endometriosis, a disease with an adverse effect on fertility. The underlying pathophysiological mechanism could be the increased risk of retrograde menstruation , 8. RadiSurgical intervention through Strassman metroplasty provides an important decrease in the percentage of fetal loss (8-12%) compared to patients without surgical treatment (70-96%) . ConventThe actual benefit of metroplasty for a bicornuate uterus, however, has not been tested in a controlled clinical trial and metroplasty for now should be reserved for women in whom recurrent pregnancy loss occurs with no other identifiable cause .In other centers, laparoscopic and hysteroscopy metroplasty has been shown to be a safe procedure and with all the additional benefits of minimally invasive surgery and it is a viable alternative to conventional open abdominal metroplasty . SignifiAll patients with Müllerian agenesis should be offered counseling and encouraged to connect with peer support groups where available. The psychological effect of the diagnosis of Müllerian agenesis cannot be underestimated. Many patients experience anxiety and depression, question their female identity, and grieve their infertility. These patients struggle with how to share their conditions with family members, peers, and romantic partners. Several cultural issues also come into play in the African setting with stigma directed to women who have had miscarriages and those with challenges getting children. This is an aspect of care that should be considered in cultural contexts similar to this case.There is a paucity of data demonstrating reproductive success after metroplasty in cases of bicornuate uterus in Sub-Saharan Africa. In Kenya, not much work on management of Müllerian anomalies has been reported or published in recent years. This case report aims to increase the awareness of Müllerian duct abnormalities more specifically bicornuate uterus in cases of recurrent miscarriages, highlighting the diagnostic strategies to investigate and to demonstrate management options in low resource settings.


Wilms tumor(nephroblastoma) is the most common renal tumor in childhood.The most frequent anomalies and syndromes associated with this tumor involve the genitourinary tract. The occurrence of Wilms tumor with renal ectopia is rare.When present,Wilms tumor is usually found in association with other forms of renal ectopia like horseshoe kidneys or crossed fused renal ectopia. However, reports of the occurrence of Wilms tumor in unilateral pelvic renal ectopia is uncommon. We report a case of Wilms tumor in a pelvic left kidney.A 10 year old girl presented to the out-patient department with a one year history of lower abdominal mass, pain and hematuria. Examination revealed a mass in the suprapubic region. Radiological investigations showed a tumor in a left pelvic kidney. She was managed with preoperative chemotherapy, surgery and adjuvant chemotherapy. Histopathologic examination of the nephrectomy specimen confirmed the diagnosis of nephroblastoma.The occurrence of Wilms tumor in association with renal ectopia is rare. There are few reports of tumors arising in unilateral pelvic ectopic kidneys. A multi-modal treatment approach involving the use of chemotherapy, surgery and radiotherapy (for high-risk tumors) in the management of Wilms tumor gives good outcome.Tumors arising from a pelvic kidney should be considered as part of the differential diagnosis of lower abdominal masses in children. The majWe present a rare case of nephroblastoma associated with a left ectopic kidney in a 10-year-old child successfully treated with preoperative chemotherapy followed by surgery and adjuvant chemotherapy. This work has been reported in accordance with the SCARE criteria .2A 10 year old girl presented at the out-patient department with a one year history of progressively increasing lower abdominal mass with associated episodes of frank haematuria. She complained of dull aching pain which developed a few weeks prior to presentation. There were no other gastrointestinal or genitourinary symptoms. The child had experienced weight loss in the preceding few months before presentation.No history of facial or leg swelling, jaundice, cough, fever or bone pains.On examination, she was not pale, afebrile, and was well hydrated. Her weight was 22 kg. Pulse was 110beats/minute, respiratory rate 22cycles/minute and blood pressure 110/80 mmHg.Abdominal examination revealed a globular shaped, non-tender suprapubic mass extending slightly to the left iliac fossa measuring about 16 cm × 12 cm. It was firm, could get above and below it, and was slightly mobile. The liver and spleen were not palpably enlarged and the right kidney was not ballotable. Digital rectal examination was unremarkable. A presumptive diagnosis of suspected nephroblastoma in a pelvic kidney to rule out an ovarian teratoma or endodermal sinus tumor was made. Abdominopelvic ultrasonography revealed an empty left renal fossa with a mass in the pelvic lying left kidney. The liver was normal. Serum alpha Fetoproteinand liver transaminases were normal but total protein(51 g/l) and albumin(26 g/l) were low.The hematocrit was 30 % and serum electrolytes, urea and creatinine were within normal limits. Urinalysis confirmed hematuria (++). Chest radiograph ruled out pulmonary metastasis.CT IVU showed empty left renal bed with an enlarged, malrotated left kidney in the pelvis, superior to the bladder dome. It demonstrated a huge, 9.5 × 10 cm hypodense mass in the mid-portion of the pelvic kidney. There was prompt excretion of contrast from the upper and lower pole calyces. No para-aortic lymph node enlargement was seen. A definitive diagnosis of nephroblastoma in a left pelvic renal ectopia was made. She was commenced on neo-adjuvant chemotherapy for 6weeks and subsequently scheduled for open  nephrectomy. Intraoperative findings include a tumor involving the left pelvic kidney situated above the dome of the bladder . The bloHistopathologic examination showed a 10 × 8.5 × 7 cm nephrectomy specimen weighing 150 g with an intact capsule and a 10 cm long ureter . Cut sur3Pelvic kidney is a rare form of renal ectopia that occurs with an estimated incidence of 1 in 2200 to 1 in 3000 autopsies. There is a slight preponderance on the left side . Renal eWilms tumor is the most common renal tumor in childhood, with survival rates over 85 % in high-income regions of the world . HoweverThere have been few reports written on malignancies in a pelvic kidney . Even moThe administration of neo-adjuvant chemotherapy reduced the tumor size by more than 30 % in the index patient. She was successfully managed with a combination of surgery and chemotherapy.In conclusion, tumors arising from a pelvic kidney should be considered as part of the differential diagnosis of lower abdominal masses in children.No source of funding for this study.The study is exempt from ethical approval in our institution.Full informed consent was obtained from the parents of the patient.Adewale O Oyinloye: Conceptualization and design of paper.Adewale O Oyinloye, Samuel Wabada and Auwal M Abubakar; Manuscript drafting and review.Lateef O Oyebanji, Chrsitopher Rikin: Case summary, study design and image acquisition.All authors read and approved the final manuscript.All authors fully participated in the care of the patient.Not applicable.Adewale O Oyinloye stands as the guarantor for this study.Not commissioned, externally peer-reviewed.All authors have no conflict of interest to declare.


NeuroEndocrine Neoplasms (NENs) are rare and can originate from any epithelial organ. We describe a very rare case of retroperitoneal metastasis from a non-functioning neuroendocrine tumor of the ethmoid region and focus on the various issues related to NENs, from their nosological classification up to the most recent acquisitions in the diagnostic and therapeutic field.A woman presented with a mass in the right retroperitoneal area that infiltrated liver and kidney. The anamnestic data showed a previous undifferentiated small cell tumor of the left ethmoid-nasal-orbital region. The mass was removed surgically and the definitive histological examination revealed a non-functioning undifferentiated malignant small cell neoplasm.NENs represent a chapter of oncology whose systematization remains difficult. The lack of hormonal syndrome in Biologically Inactive Neuroendocrine Tumors (BINTs) may delay the diagnosis. Clinical manifestations relate to the size and location of the neoplasm. Small cell NeuroEndocrine Carcinoma (NEC) of the ethmoid-nose-orbital region is an extremely rare occurrence and generally the most frequent metastatic localization is represented by the larynx.This case highlights the contradictions typical of NENs and only new scientific acquisitions in the histopathological field can help us in the future. Surgery remains the only possibility of treatment and diagnosis of large masses of inactive NENs. This is the only case in the world of metastasis from NEC of the nose-orbital region in the retroperitoneum. They re2At our institute, a 58-year-old female patient in good health came to our attention. Access to our department was determined by a persistent abdominal pain localized in the right lumbar region. The collection of anamnestic data revealed a small cell type NEC, discovered two years before, with positivity to Synaptophysin and Neuron Specific Enolase (NSE) but not to Chromogranin A, of the left ethmoid-nasal-orbital region, treated with chemo- and radiotherapy cycles. In our department the patient underwent a total body Computed Tomography (CT) scan  which shThe whole mass weighed 1200 g, and was 17 × 13 × 12 cm in size, linked to all removed structures , Fig. 55The final histological examination showed a NEC composed of small cells with immunohistochemical characters that were Synaptophysin-positive, NSE positive, but chromogranine negative .Fig. 6DeThe post-operative course was satisfactory and the patient was released from the hospital on the 24th day in good condition and entrusted to oncologists for treatment of the case. The patient had a positive follow-up at 6 months and 12 months.3The definition of neuro-endocrine cells has been changing over the years owing to improved knowledge and acquisition of modern research techniques. From the proposal put forward by Pearse in the 1960s and the introduction of the Amine Precursor Uptake Decarboxylation (APUD) system, which led to a common embryological histogenesis of these cells that showed their derivation from the neural crest, to the studies by Le Dourein, which highlighted that this was in part untrue, since the endocrine cells of the digestive tract, pancreas and lung are of endodermal origin . NENs ar1)NET G1 (mitotic count <2/10 HPF and/or ≤2% Ki-67 index);2)NET G2 (mitotic count 2–20/10 HPF and/or 3–20% Ki-67 index);3)NEC (mitotic count >20/10 HPF and/or >20% Ki-67 index);4)Mixed AdenoNeuroEndocrine Carcinoma (MANEC), where at least 30% of either component must be identified to qualify for this definition;5)Hyperplastic and preneoplastic lesions.In 2010, WHO released a new classification of these neoplasms. Based on their mitotic rate  and Ki-67 labeling index)  Table 1Table 1 NThis classification is highly relevant to the prognosis and treatment of choice. The diagnosis of NEN is therefore morphofunctional and based essentially on microscopic features and immunohistochemical data: positive to Synaptophysin, to NSE and to Chromogranin. It is to be noted that well-differentiated NENs are intensely positive to the latter marker. This is true only in 10–15% of NENs in small cells are . The stu4NENs represent a chapter of oncology whose systematization remains difficult. Classification, study and research on these neoplasms entails complex knowledge involving all branches of medical science. The rarity of most NENs necessitates multicenter studies that allow histopathological classification. The non-active forms usually have a paucisintomatic symptomatology that is revealed only when the tumors occupy space. However, they are very aggressive neoplasms, especially in their poorly differentiated form. The diagnostic procedure includes ultrasonography, CT scan, immunoscintigraphy and PET. Immunohistochemical findings, and even those of genetic mutations, should not be overlooked for prognostic purposes. From a therapeutic point of view, surgery plays a fundamental role: it must be radical, with extensive lymphectomies and aggression of any metastases. The discovery of new biomarkers may open up new possibilities for classification, diagnosis and type of treatment. Ultimately, the clinical case presented herein highlights all the contradictions and problems typical of NETs and only new scientific acquisitions in the histopathological field will open new and more comforting scenarios in biological and therapeutic behavior.This study did not receive any funding.The study not require ethical approval.Written informed consent was obtained from the patient.Study concept or design: Saverio Cerasari, Carlo CataldiData collection: Carlo CataldiData interpretation: Saverio Cerasari, Carlo CataldiLiterature review: Saverio Cerasari, Carlo Cataldi, Gaetano PoillucciDrafting the paper: Saverio CerasariEditing of the paper: Saverio Cerasari, Gaetano PoillucciRevision: Pierluigi Marini, Carlo CataldiThis study does not require registration.Carlo CataldiNot commissioned, externally peer-reviewed.The authors declare that they have no financial conflict of interest related to this paper.


A myxoma is a neoplasm comprised of mesenchymal connective tissue. Myxomas of the upper extremity, and particularly of the hand, are rare. We present a case of a hypothenar myxoma causing ulnar neuropathy in a patient with a history of acute inflammatory demyelinating process. Treatment and management of myxoma may vary depending on whether the process is malignant or benign; thus, histologic diagnosis is critical to determining treatment. The purpose of this article is to review the pathophysiology and clinical features of myxomas, and to provide recommendations for evaluating and treating individuals with extremity masses of unclear clinical diagnosis. A 55-year-old right-hand-dominant male with a history of acute inflammatory demyelinating process and a 3-month history of a left volar wrist and hypothenar soft tissue mass presented for evaluation. The patient was admitted to the hospital with generalized weakness, ataxia, and multiple sensory deficits, including complete lack of sensation in the left small finger and ulnar side of his hand. Although his sensory deficits were initially thought to relate to his generalized demyelinating disorder, on examination, the ulnar deficit was thought to be related to the mass in his hand. The mass was soft, nontender, and did not limit range of motion. Two-point sensation was absent in the ulnar nerve distribution distal to the wrist, and grip strength, while not measured objectively, was decreased compared with the contralateral side. There was no history of trauma, previous surgery, or other masses, and radiographs of the left upper extremity were normal. A lipoma was the suspected initial diagnosis. Magnetic resonance angiography of the left hand showed a soft-tissue mass of unclear etiology Fig. .Operative excision was performed under general anesthesia. A longitudinally oriented ulnar-sided skin incision was made over the mass, with dissection through the palmar fascia. The ulnar nerve and artery were dissected free from the mass, and Guyon’s canal was released. The mass was noted to be arising from beneath the hypothenar musculature. Grossly, it was tan-white, rubbery, and lobulated Fig. . Patholo2 Stout’s 1948 definition refers to myxoma as, “a neoplasm composed of stellate cells set in a loose mucoid stroma through which course very delicate reticulin fibers in various directions.”3 While the histologic criteria of myxoma are well-established, the etiology remains unclear.1 Although the majority of myxomas occur in the heart, extracardiac myxomas do occur, and may be found either subcutaneously or embedded within muscle; intramuscular myxomas are generally found in the thigh, lower leg, or gluteal region, with an incidence of approximately 0.1 per 100,000.7Soft-tissue myxoma was first described in 1863 by Virchow as a benign tumor similar to mucous tissue of the umbilical cord.6 Myxomas have been reported to occur in the fingertip, phalanges, and metacarpal and palmar regions.6 Benign myxomas of the upper limb are classified according to the tissue of origin, with subungual myxomas occurring most commonly, and intraosseous and intramuscular myxomas occurring most rarely.8Myxomas of the hand are rare and may be confused with other benign and malignant neoplasms on clinical examination or after imaging.We report a case in which the patient’s history of an acute demyelinating disease made diagnosis more challenging. Without knowledge of the hypothenar myxoma, the symptoms of hand numbness and weakness had been attributed to acute inflammatory demyelinating process. However, after a thorough hand examination, discovery of the mass and patient’s ulnar neuropathy supported the decision to excise the symptomatic soft-tissue mass.1 However, myxoma may be confused with other types of lesions, and other etiologies such as sarcoma, cysts, and infectious processes must not be ruled out until histopathologic information can confirm the diagnosis.1Magnetic resonance imaging allows for characterization of soft-tissue lesions of the hand and should be used when a myxoma is suspected to aid ascertainment of whether the mass arises from a nerve sheath or has an intramuscular course.6 Malignant myxomas  display higher vascularity, infiltration, and more variation in thickness when silver-stained than their benign counterparts.9 If a biopsy is performed, a myxoma should be excised, regardless of malignancy or presence/absence of symptoms.6 Some authors report obtaining a diagnosis using an intraoperative frozen section.4 Recurrence is generally rare and may be associated with incomplete resection or malignancy.9 Malignant myxomas are capable of metastasizing9; thus, subsequent treatment is different than local excision of a benign myxoma, often requiring more radical excision and, in some cases, amputation.9Myxomas are most frequently benign, though they may infiltrate surrounding tissues and structures.Myxomas of the upper extremity, particularly the hand, are rare. Although they are generally benign, malignant transformation has been described and should not be overlooked in a differential diagnosis. Resection is recommended, with close attention to whether the mass involves and/or arises from neurovascular structures.The authors greatly appreciate pathologists Michael L. Schwalbe, MD, and Thomas Warner, MD.


Bacterial biofilms can be calcified. Granulomas or cystic lesions are the most commonly found entities in endodontics. Surprisingly, this case report presents a rare radiopaque image, in a fan shape, of a calculus-like deposit in the periapical region of the maxillary left central incisor.A 34-year-old male, with a history of trauma, presented with apical periodontitis associated with an uncommon image, similar to a calculus-like deposit adhered to the apical region of the maxillary left central incisor. Nonsurgical endodontic intervention was performed, followed by apicoectomy and histopathological analysis of the collected material. The results of the biopsy were not compatible with a cyst or granuloma but showed fibrous connective tissue with calcified areas.Correct diagnosis in endodontics is possible with a well-conducted anamnesis, complementary imaging exams and, in some cases, histopathological analysis. The periapical calculus-like deposit, associated with a periapical radiolucent lesion, was a result of the body's fight for healing, producing unusual radiopacity.The presence of the calculus-like deposit in a fan shape at the root surface represented dystrophic calcification as a manifestation of the attempt to heal. In the present case, apicoectomy and tissue biopsy for histological evaluation were fundamental for the correct diagnosis. •Root canal is critical area for biofilm development.•Extraradicular biofilm on the surface of the root may cause persistent periapical lesions.•Uncommon radiopaque image association with endodontic periapical lesions was related.•Persistent periapical lesions associate with extraradicular biofilm represents a challenge for Endodontic treatment.•Only surgical resection was capable to repair the injury. These rSeveral studies have demonstrated the formation of a biofilm from bacterial invasion in the extraradicular area, adherent to the cementum around the root apex . BacteriHistorically, clinical examination data associated with the radiographic presentation were central to the diagnosis of apical periodontitis . NeverthThe aim of the present study was to report a case of apical periodontitis associated with an unusual, fan-shaped, radiopaque image of the periapical region of the maxillary left central incisor in a 34-year-old male. This curious endodontic calculus-like deposit resulted from an intense inflammatory response, leading to the formation of granulation tissue with calcified areas, which demonstrates the attempt of the organism to repair itself ,9.2In accordance with the principles laid down in the Declaration of Helsinki, this case report integrates a project research previously approved by Ethics Committee (CAAE: 01958818.4.0000.5137). This work is reported in line with the SCARE criteria .A Brazilian patient  reported to the endodontic clinic of our institute, complaining about the presence of a swelling in the region of the maxillary anterior teeth. During the interview, he reported a car accident 13 years ago. The patient did not remember the dental procedures that had been performed. The patient was a strong and healthy young man, with no history of alcoholism and smoking. No signs and symptoms of chronic diseases. Your parents are alive and healthy.On clinical examination, the presence of a labial swelling was observed in the apical region of the maxillary left central incisor. The referred element presented no significant periodontal probing depth, and endodontic access was sealed with a resin composite A–D. In aPeriapical radiographic examination showed an extensive radiolucent area associated with the apex of the maxillary left central incisor, extending to the lateral incisor A. The prThe CBCT images B showed Endodontic retreatment of tooth #21 was performed in multiple sessions. Initially, the patient was administered anesthesia by an infiltration technique with 2% lidocaine with epinephrine, 1:200,000 , followed by isolation with a rubber dam and access to the root canal system of the central left maxillary incisor. A cotton ball and two cone fragments of absorbent paper for intracanal medication were removed from the interior canal E. The puAfter 7 days of nonsurgical endodontic treatment, the patient was referred to a maxillofacial surgeon with extensive experience for surgical intervention because of the extent of the bone loss and its proximity to noble areas. After surgical access, we carried out enucleation of the lesion, which was still attached to the end of the resected root H. All su3Granulomas or cystic lesions are the most commonly found entities in endodontics ,20. The CBCT, already well established in the literature, is a fundamental tool for the diagnosis and treatment planning . In the A similar study in the literature describes another case of unusual radiopacity associated with a periapical radiolucent lesion on the root surface of the maxillary left central incisor, previously treated endodontically . PatholoSurprisingly, this case presents an unusual connective tissue with dystrophic calcified areas, as an intense response of the body in an attempt to heal. Another study of a series of cases has emphasized that periapical surgery should be considered the last resort in endodontics to effectively deal with periapical lesions that result from pulp necrosis. The study showed that different extraradicular infections were associated with persistent exuding symptoms, leading to endodontic failure, and were successfully treated only by periradicular surgery ,18. UnfoTherefore, confirmation of the final diagnosis was provided by histopathological examination of tissues, impractical in the case of nonsurgical endodontic treatment . This ca4This study demonstrates a case of apical periodontitis associated with a deposit similar to periapical calculus that represents an unusual response of the organism in repairing the damages caused by the presence of extraradicular biofilm and, consequently, the intense inflammatory response. The surgical approach was essential for the diagnosis and appropriate treatment of this occurrence.Written informed consent was obtained for publication of this case report.Not commissioned externally peer reviewed.In accordance with the principles laid down in the Declaration of Helsinki, this case report integrates a project research previously approved Ethics Committee (CAAE: 01958818.4.0000.5137).The author affirm that did not receive any sources of funding for your research.All authors contributed significantly and in agreement with the content of the article. Tonelli, Toubes and Silveira collected all datas and photographs to draft the manuscript. Toubes realized the endodontic treatment of the canal. Oliveira realized the surgery. Tonelli, Nunes, Silveira, and Duarte wrote the manuscript for submission. All authors presented substantial contributions to the article and participated of correction and final approval of the version to be submitted.The author declare none conflict of interest statement.The authors declare that studies like this have already been performed/reported in the literature.Dr. Frank Ferreira Silveira.


We present a case of a desmoid tumor recurrence in a patient with a history of a resected desmoid tumor of the right neck area with free surgical margins six months earlier. The neoplasm was found to invade the parapharyngeal space, and wide excision was performed including most of the sternocleidomastoid muscle (SCM), the thrombosed internal jugular vein (IJV), and the infiltrated spinal accessory nerve (SAN). The histopathologic findings displayed free microscopic margins, with close margins at the site of the parapharyngeal space extension. After 3 months, there was no sign of tumor recurrence. After 6 months, local tumor recurrence was identified on clinical examination and imaging. The decision of the Oncology Board was further treatment with radiotherapy (RT). Response to treatment was satisfactory, and the patient was on close follow-up for twelve months. Desmoid tumors are very rare benign neoplasms of mesenchymal origin with negligible mortality but high morbidity, due to their high recurrence rates, local tissue infiltration, and unpredictable disease course and response to treatment. No universally acceptable treatment protocols have been introduced to date. Appropriate patient counseling and close follow-up are warranted in all cases. Desmoid tumors, also known as “well-differentiated nonmetastasizing fibrosarcoma,” “Grade I fibrosarcoma,” and “aggressive fibromatosis,” are very rare benign neoplasms of mesenchymal origin . They arA 22-year-old Caucasian female, nulligravida, presented to our institute  complaining of a slowly growing painful mass at the right lateral neck. Past medical history included a resected desmoid tumor with free surgical margins from the same region six months ago in another center . No otheMRI revealed an enhancing mass at the cephalic third of the SCM, in close contact with the right IJV, with no signs of vessel infiltration . No pathGiven her past medical history, imaging findings, and clinical presentation, the patient was scheduled two months later for surgical excision of the tumor recurrence under general anesthesia. Access to the surgical field was via an oblique right lateral neck incision. The neoplasm was found to originate from the upper portion of the SCM, extending to the parapharyngeal space, and infiltrating the SAN . A wide The patient was discharged on the second postoperative day in good condition. The range of right upper extremity extension was limited, and the patient was referred for physiotherapy.β-Catenin nuclear staining was also present  without atypical mitoses. Immunohistochemically, neoplastic cells stained vimentin , smooth  present . The indOn the 3-month postoperative follow-up, no clinical signs of tumor recurrence were noted, and the right upper extremity mobility was satisfactory, indicating successful anastomosis of the SAN. Unfortunately, on the 6-month follow-up, local pain and swelling indicated new tumor recurrence which was confirmed by MRI. The case of our patient was brought to the Oncology Board. Unanimously, it was agreed that a wait-and-see policy was not a sound option and some kind of treatment should be administered since the tumor recurred within six months. Based on imaging findings, excellent performance status of the patient, and our experience as a tertiary referral center on head and neck surgery, excision of the new tumor recurrence was considered as the primary treatment modality. RT was also recommended, but given its long-term side-effects and the proximity of neural structures to the tumor bed, it was considered as an alternative option. Cytotoxic agents, tyrosine kinase inhibitors, and hormonal therapy were considered but discarded, given the reproductive age of our patient and the absence of contraindications for surgery or RT. The patient declined the option of further surgery but elected for RT and received 54 Gy of external beam RT over six weeks. Treatment was well tolerated. After the completion of RT, complete clinical remission of the disease was noted. No evidence of recurrence has been detected on clinical evaluation and imaging studies after twelve months of follow-up.According to the location of occurrence, desmoid tumors are classified into intra-abdominal, abdominal, and extra-abdominal types . About 3Desmoid tumors present as circumscribed, firm overgrowths of fibrous tissue arising from virtually any area with a connective tissue component, showing propensity to infiltrate surrounding tissues and occasionally encase blood vessels and nerves, without apparent deep invasion . HistoloNo universally acceptable therapeutic protocols for extra-abdominal desmoid tumors have been introduced to date. Most information regarding this rare entity originates from retrospective studies and sporadic case reports. It should be noted that while the vast majority of patients are adults, it does not spare the pediatric population . As suchSurgical excision of the disease has long been the mainstay of treatment. Tumors of the head and neck pose considerable challenges for the surgeon due to close proximity to vital structures. In addition, treatment-induced morbidity and unsatisfactory local control rates, along with spontaneous regression observed occasionally, have put aggressive surgical treatment into question. Finally, there are no established prognostic factors that can be employed for decision-making on treatment modality .Significance of surgical margin status for this neoplasm is a matter of debate. When technically feasible, complete tumor resection with negative microscopic margins is the optimal treatment choice. The importance of microscopic positive margin status with regard to local recurrence rates is still unclear; the presence of microscopic disease has not been shown to affect long-term disease-free survival , 13. Morβ-catenin, and APC locus gene mutations have been implicated with tumor pathogenesis and disease prognosis [Further research is required to decrypt the molecular mechanisms underlying desmoid tumor genesis and progression. Acquiring such knowledge may lead to better patient selection, superior treatment, and optimal assessment of prognosis of each case. According to the current literature, CTNNB1 , rognosis , but theMultiple conservative treatment modalities have been recommended and used in different settings. Radiotherapy has been applied primarily for inoperable or inaccessible disease. In addition, it has been used as adjuvant treatment in cases with positive surgical margins and a higher risk of local recurrence. Noncytotoxic regimens like nonsteroidal anti-inflammatory drugs (NSAIDs) and/or tamoxifen have been employed as first-line medical treatment. Cytotoxic agents including anthracycline-containing regimens, doxorubicin, vinblastine, methotrexate, and hydroxyurea have also been employed in cases where surgery and/or radiotherapy are contraindicated, producing variable but, in general, satisfactory results. Response rates to tyrosine kinase inhibitors (imatinib) administration have been shown to be subpar, and this choice should be reserved for cases that have failed other treatment options , 14, 15.Desmofibromatosis of the head and neck has negligible mortality but high morbidity, due to high recurrence rates and unpredictable biological behavior and response to treatment. A high level of suspicion, appropriate patient counseling, and close follow-up are warranted in all cases .


Herlyn-Werner-Wunderlich (HWW) syndrome is a rare congenital urogenital defect. It is detected by unilateral low vaginal obstruction, uterus didelphys, and ipsilateral kidney agenesis. It usually becomes apparent with pain, dysmenorrhea, and presence of a vaginal or pelvic mass. Purulent vaginal discharge may also happen rarely because of infective complications of the obstructed hemivagina. In this report, we describe a post-pubertal case with acute abdominal pain. The patient was a 13-yr-old girl who was referred to us with acute abdominal pain one year after the onset of her menarche. In the pelvic examination, we detected hematocolpos. Abdominopelvic-computed tomography scan confirmed the presence of mullerian duct anomalies with uterus didelphys. This case of HWW syndrome along with pyocolpus was managed by vaginal septum resection, drainage of pus, and salpingectomy. The symptoms of HWW syndrome should be monitored in early puberty to prevent more complications. Herlyn-Werner-Wunderlich (HWW) syndrome is a rare congenital obstructive mullerian anomaly with 0.1-3.8% prevalence in women. It is detected by hemilateral vaginal obstruction, uterus didelphys, and ipsilateral kidney agenesis . It is caused by lateral non-fusion of the mullerian ducts in the one-sided obstructed vagina . Usually patients are asymptomatic before puberty; however, the symptoms become present gradually because of the acute congestion of blood in the vagina after menarche (4). This syndrome is characterized by a triad of uterus didelphys, ipsilateral kidney agenesis, vaginal septum, and hemilateral vaginal obstruction (5). Uterus didelphys is class 3 anomaly of the American Fertility Society classification (5). Most patients are adolescent girls with pelvic pain and mass after menarche (1). In a few cases, pyohematocolpos, pyosalpinx, and peritonitis may appear because of the ascending infection in the obstructed hemivagina (6). In the pelvic examination, hematocolpos is seen with a visible bulge in the vagina (6). Although the ultrasound imaging is the first choice for detection, magnetic resonance imaging is suggested for complementary and definitive diagnosis (4). Laparotomy and laparoscopy are used in acute cases only (1).In this report, we described an unusual case of a young girl with HWW syndrome with pyocolpus, peritonitis, and endometriosis that was successfully treated by resectioning vaginal septum, salpingectomy, and pus drainage.Our patient was a 13-yr-old girl who was referred to our hospital because of acute abdominal pain. She had a fever for two weeks before being referred to us. Her menarche had begun one year before her referral. She had suffered from severe dysmenorrhea until then, and her last menstrual cycle had been two days before coming to the hospital. Her abdominal pain had increased during her last menstruation. The patient had a fever, chills, abdominal pain, and nausea at the time of admission to the hospital. She did not have a vaginal discharge with bad odor and had no particular problems in her medical history. Generalized abdominal tenderness was diagnosed in the physical examination, but mild vaginal bleeding and right lateral vaginal wall bulging were found in her vaginal and transrectal examinations. She underwent laboratory tests including leukocytosis , platelet , hemoglobin (10.7), C-reactive protein (3+), normal liver and kidney function tests, and normal coagulate profile tests. Her abdominal ultrasound images showed uterus didelphys with a mixed-pattern cystic mass measuring 72 We decided to do abdominal and pelvic computed tomography scan to evaluate the cystic mass which was reported in abdominal ultrasound imaging. We had planned to do the surgery in an appropriate condition. This meant with patient's intestinal preparation after evaluating computed tomography results, and in the presence of an expert general surgeon. However, a few hours after the admission, we had to do an emergency surgery on the same night because she had a high fever, increased ESR and CRP, and a possible risk of septic shock. We administered many antibiotics before the emergency surgery because of the acute abdominal pain.During the first surgery, we did a physical examination while she was under anesthesia. The hymen was normal and intact. But a buldge was found in the right side of her vagina during the rectoabdominal examination. This was confirmed by vaginal examination with a speculum. The right longitudinal septum was revealed with a closed end. At first, we cut the closed end of the hemilateral obstruction of the vagina and then the vaginal septum was resected with an electrocautery device. Afterward, a 200-cc pus was drained out, and this sample was sent to a laboratory for culture and antibiogram.The patient's abdomen was opened with a midline incision. There was a severe adhesion of the intestine and omentum to the abdominal wall. Since no intestinal preparation had been done and there was a high risk of intestinal rupture, the general surgeon decided that it would be better to end this surgery and prepare the patient for another abdominal surgery in better conditions.Abdominopelvic-computed tomography scan (with and without contrast which was done after the first surgery) confirmed the ultrasound imaging findings of uterus didelphys with multi-cystic mass with severe development in the right adnexa. A remarkable right hematocolpus hematosalpynx was seen that was due to longitudinal vaginal septum and hemivaginal obstruction. The right kidney was not observed . Her problem was detected as HWW syndrome infected with pyocolpos.We did the second laparotomy in less than 48 hr after the first surgery with the same incision in the presence of an expert surgeon. Our patient had severe intestinal adhesion and perforated appendix fistulated to the right fallopian tube beside distal small intestinal obstruction. We saw pelvic inflammatory mass beside endometriosis. We did an appendectomy, right salpingectomy, entrolysis, and abscess drainage. Cefepim (Maxipim) and cloxacillin  were administrated intravenously as specific infectious disease drugs until seven days after the surgery according to the specimen culture. We ordered continuous low-dosage contraceptive drugs for preventing the progression of endometriosis. Our patient was discharged from the hospital after seven days with a good condition.After the completion of treatment, we asked the patient for the permission to publish her anomaly as a case report. She accepted and gave us an oral consent.The female genital system has two mullerian ducts fused in the midline with urogenital sinus (6). Failure of resorption during this fusion leads to an obstructive anomaly of mullerian system . A rare example of this obstructive anomaly is the HWW syndrome . This syndrome was explained by Herlyn-Werner in 1971 for the first time and it was completed by Wunderlich in 1976 (8). In this syndrome, obstructed hemivagina is the main reason for blood accumulation in the uterus and retrograde bleeding in the abdomen through fallopian tubes that can cause pelvic pain and mass, hematometra, hematocolpus, and finally endometriosis. The symptoms at the time of being referred to the hospital depends on the degree of the anomaly. It consists of dysmenorrhea, urinary incontinence, infertility, and acute abdominal pain due to endometriosis .Dongan and colleagues classified the HWW syndrome into two types: complete or incomplete vaginal septum (4). Tong and colleagues stated that the age of pelvic endometriosis onset is earlier than the age of finding endometriosis. Approximately, in 88.89% of HWW syndrome cases, the patients are affected by endometriosis (9). They are usually diagnosed in the pubertal or post-pubertal period, but Angotti and colleagues reported six cases of this problem in girls younger than five years old (8). The resectioning of the vaginal septum in obstructive hemivagina should be done through an abdominal or transvaginal approach (8).In our case after confirming the HWW syndrome with the computed tomographic scan, we did a vaginal septum resection. Our case was complicated because of pyosalpinx. So, investigative laparotomy was done. A two-step surgery is recommended in similar cases by pyocolpus for a complete vaginal septum resection (6). Sometimes, the closure of an opening will occur at the end of one year (4). Doing a complete vaginal septum resection is necessary for assuring the future fertility, while uterus didelphys has no effect on reducing the infertility. So, fertility prognoses remain good after surgery in uncomplicated cases .This report described a case of the unusual type of HWW syndrome with fever and acute abdominal pain. The main complaint of our patient was only severe dysmenorrhea. Cyclic abdominal pain, abdominal mass, dysmenorrhea, with or without urinary disorders, can lead to this problem in adolescence. So, the symptoms should be emphasized in early puberty to prevent more complications. Well-timed removal of the vaginal septum can improve fertility and endometriosis. Also, it decreases pyosalpinx, acute abdominal pain, and further surgeries.A limitation of our work was doing the surgery twice. If the patient was in a better condition, we could have done the surgery same as many other cases with opening the obstruction of vagina and laparoscopy. However, because of the different situation of the studied case and that she had a acute abdominal pain that could be a sign of infection, we tried to avoid sepsis with doing the surgery in two steps.Although dysmenorrhea is among girls in puberty, it is advised to follow-up acute dysmenorrhea soon to avoid further medical consequences.The authors report no conflicts of interest.


The vas deferens and spermatic vessels entering the inguinal canal through the internal inguinal ring is thought to exclude an intra-abdominal testis. We present a case of high bilateral intra-abdominal testes on a 46,XY boy despite the vas deferens and good-sized vessels passing through the deep rings.Data were collected from clinical records, radiology , and endocrine blood tests.This case underlines the importance of following the pathway of embryological descent of the testis cranially as well as caudally during diagnostic laparoscopy, to avoid missing this rare anatomical variant. Laparoscopic findings in case of impalpable undescended testes have been well described in the literature. When both the vas and vessels enter the internal inguinal ring, an exploration is generally indicated. However, in the absence of blind-ending vessels in the proximity of the internal inguinal ring, laparoscopic exploration must continue in a cranial direction to locate a possible high intra-abdominal testis.We present a unique case in which vas and vessels were entering the internal inguinal ring but the testes were found to be intra-abdominal. The diagnosis turned out to be a long bilateral testicular–epididymal dissociation (TED).A newborn, term baby had been referred to the local urology department for severe penoscrotal hypospadias and bilateral impalpable testes. The genetic and endocrine investigations confirmed a 46,XY karyotype as well as a normal response to Hcg (human chorionic gonadotropin) stimulation. Diagnostic laparoscopy at 1 year of age demonstrated the vas deferens and vessels entering the internal inguinal rings bilaterally. Groin exploration revealed what was considered to be atrophic testicular nubbins, which were left in situ at parental request. Hypospadias correction was performed separately. Throughout childhood, the endocrine status revealed normal hormonal levels .Spontaneous puberty and inability to locate palpable gonads triggered further investigations. Endocrine profiling at this point showed normal testosterone levels sustained by high gonadotropins. The anti-Müllerian hormone (AMH) and Inhibin-B levels were at the lower end of the reference range, suggesting functioning testicular tissue and3.The discrepancy between good hormonal response at a repeat Hcg test and the ongoing impalpable nature of the gonads led to the question of possibly missed intra-abdominal testes. Ultrasound scan failed to detect any gonads; however, subsequent abdominal magnetic resonance imaging (MRI) suggested intra-abdominal testes close to the lower pole of the respective kidneys . Familyrete testeslater.rete testesand the mesonephric tubules share a common lumen.Testis and epididymis follow differing embryological pathways: the former originates from the urogenital crest, whereas the latter derives from the mesonephric duct. The SRY (Sex-determining Region of Y-chromosome) gene expression guides the undifferentiated sex cords to transform into seminiferous cords first andThe classifications proposed by Turek et al and Windholz focused on features of aberrant fusion but lacked a qualitative description of the epididymis in its different portions in case of fusion defects. The epididymis can, in fact, be also maldeveloped. Simon included in their classification qualitative aspects of the fusion defect. They categorized cases of head attachment achieved by elongated efferent ducts (type b), as well as direct testis-vas deferens fusion with absent epididymis (type c), absent epididymal body (type d), and absent epididymal head and body (type e).Laparoscopy is considered the gold standard for the assessment of intra-abdominal testes. Once both vas and vessels are seen entering the internal inguinal ring, an inguinal exploration is generally advised. On the contrary, if both vas and vessels are blind ending, an inguinal exploration should be theoretically unnecessary.As the appearance of the nubbins was initially strongly indicative of gonadal remnants, we have also retrospectively postulated the polyorchidism variant. This is more common on the left side and can be associated with cryptorchidism.A two-stage Fowler–Stephen procedure has been offered for the left testicle as TEDs do not correlate with significant histological gonadal abnormalities; therefore, orchidopexy remains an option if the testicle is eligible for it.The incidence of testicular–epididymal fusion defects is high for undescended intra-abdominal testes, and, at laparoscopy, vessels deriving from an aberrant fusion attempt might be seen. Those vessels, if examined at the level of the internal inguinal ring only, can be indistinguishable from normal retroperitoneal spermatic artery and veins. If only a nubbin is found at exploration and the preoperative Hcg test is positive, suggesting functioning testicular tissue, a careful search along the pathway of testicular descent, with colonic mobilization, looking for high intra-abdominal gonads, is therefore strongly recommended to avoid missing the anatomical variant presented.


A solitary fibrous tumour (SFT) is a rare spindle-cell neoplasm of mesenchymal origin usually located in the pleura. It has been recently described as occurring in various head and neck sites, including the oral cavity. The purpose of this article is to report a case of SFT originating in the buccal space and extending into the infratemporal space treated by means of transoral approach surgery. A 25-year-old female patient reported to the Department of Oral and Maxillofacial Surgery, Universitas Airlangga Hospital, chiefly complaining of a painless lump in the left cheek which had been present for nine months. The diagnosis was arrived at on the basis of a combination of clinical investigation, imaging studies and histopathological examination (biopsy). The surgical approach involved transoral incision through the buccal mucosa. An SFT of buccal space may extend to nearby structures producing the anatomical challenge of removal through a transoral approach. Excisional biopsy involving a transoral approach is, nevertheless, considered appropriate because it produces an attractive aesthetic appearance, reduces morbidity from nerve/ vascular/ gland injury and promotes more effective healing. Key words:Solitary fibrous tumor, buccal space, infratemporal space, transoral approach. A solitary fibrous tumour (SFT) is an uncommon spindle-cell neoplasm of mesenchymal origin. An increasing incidence of the tumor has been found in the extrapleural sites, including the peritoneum, mediastinum, orbit, infratemporal fossa parapharyngeal space, upper airways and nose, salivary and thyroid glands and oral cavity. As documented in the literature, SFT accounts for 3% of all cases occurring in the oral region. When SFT develops in oral cavity it appears like a painless swelling, some of them may give rise to compression symptoms and with differential diagnosis lipomas, pleomorphic adenomas, schwannomas, fibrous histiocytomas, benign glandular tumors and dermoid cyst. Members of both sexes are equally affected, with patients being predominantly middle-aged or elderly. Symptoms depend on the site and depth of the tumour, but tend to be mostly asymptomatic slow growing masses and shows a high rate of positivity for CD34 on immunohistochemical staining -8.The case reported here is one of an SFT in the left buccal space extending into the infratemporal region. The depth at which the tumor was located between the spaces rendered both distinguishing it from other soft tissue tumor and its removal challenging. An initial diagnosis was established through clinical examination, imaging and incisional biopsy. Definitive treatment ultimately consisted of excisional biopsy incorporating a transoral approach due to the benefits of an attractive aesthetic appearance, reduced morbidity from nerve, vascular and gland injury in addition to enhanced healing.Case ReportA 25-year old female reported to the Oral and Maxillofacial Surgery Department, Universitas Airlangga Hospital, Surabaya presenting the primary symptom of a painless lump in the left cheek she had first detected nine months earlier. The individual concerned did not complain of toothache or numbness and had no previous history of ill-health. Further oral examination revealed asymmetry of the left-hand side of the face without accompanying redness Fig. A. PalpatAn initial orthopantomogram showed no destruction of the mandible and maxilla, but a post-extraction socket existed in the 36 region due to the patient having undergone a tooth extraction two months before Fig. B. UltrasA head and neck CT scan with contrast detected a developing soft tissue tumor 2.69x3.73x4.0 cm in size in the left buccal space Fig. D resultiA fine-needle aspiration biopsy was performed by puncturing an area from the extraoral to the intraoral site. Microscopic examination showed a hypocellular area consisting of a broad distribution of erythrocytes with a number of inflammatory cell histiocytes and neutrophils, but no signs of malignancy. An incisional biopsy was also performed through an intraoral incision on the left buccal which revealed tissue with wide fibroblast proliferation, extremely similar to a fibroma.Following the securing of patient consent, an excisional biopsy using an transoral approach was performed under general anesthesia. First, the left buccal was palpated to localize the mass area which was subsequently marked with methylene blue. The incision site was also marked in the middle of the mass. Prior to incision, 1:200.000 of pehacain was injected as a vasoconstrictor to reduce oozing during surgery  which can be traumatized if incision occurs in their proximity. This can result in facial paralysis which not only constitutes a severe cosmetic deformity but can, in addition, have significant functional ramifications . The othAccess to the mass was effected through an incision in the buccal mucosa Fig. B, with cLong-term follow up is mandatory for SFT patients because, in certain cases, clinical behavior does not correlate with histopathologic appearance. A slight but statistically significant increased risk of local disease recurrence was found in extrathoracic SFT -9.