Hepatic epithelioid hemangioendothelioma (HEH) is extremely rare, occurring in 1 to 2 per 100,000, with chemotherapy options not well defined. Our case involved a 49-year-old female who had hepatic masses and metastasis to the lungs with a liver biopsy revealing HEH. After developing a rash from sorafenib, thalidomide was started with the progression of disease stabilized. Resection is only an option in 10% of the cases; therefore, chemotherapy is the only line of treatment. Newer chemotherapy alternatives are targeting angiogenesis via the vascular endothelial growth factor. Thalidomide was first used as an antiemetic, but, sadly, soon linked to phocomelia birth defects. Given the mechanism of action against angiogenesis, thalidomide has a valid role in vascular tumors. In conclusion, the use of thalidomide as chemotherapy is novel and promising, especially in the setting of a rare vascular liver tumor such as HEH. Vascular in origin, hepatic epithelioid hemangioendothelioma (HEH) is an extremely rare diagnosis; therefore, treatment for this malignancy has not been well defined. Much of the data on HEH are in case studies and small case series with treatment usually confined to only a few options. Given the vascular nature of this cancer, thalidomide has been used with some degree of success. In the mid-twentieth century, thalidomide was used for morning sickness in pregnant women, and their babies developed well-documented phocomelia birth defects.From those horrific malformations, continued investigation into the explanation of thalidomide involvement has led to a better understanding of its harmful effects on angiogenesis. With this discovery, the careful use of the medication has been employed as chemotherapy in cancers such as HEH. In this case report, we present a female diagnosed with metastatic HEH who has been successfully treated with thalidomide chemotherapy.We present a 49-year-old Hispanic female originally seen in the ER for atypical chest pain needing cardiac evaluation. Patient presented months back with chest pain that started early the morning of admission, intermittent in nature, worsened with deep inspiration, and radiated to her back. No abdominal pain was noted, but she did complain of associated nausea, diaphoresis, fatigue, and dizziness. She also reported 30-pound weight loss in the last month with no change in appetite discussed. Past medical history was only significant for type 2 diabetes mellitus. Family history was significant for coronary artery disease in her father and stomach cancer in her mother. No pertinent surgical history was given. She denied any tobacco, alcohol, and illicit drug use.Full cardiac evaluation, including EKG, cardiac marker testing, and chest X-ray, was negative. Upon further review of her chart, 3 years prior to this presentation, a computed tomography (CT) of the abdomen and pelvis was ordered by an urgent care physician secondary to abdominal pain. It showed multiple hypodense masses in the right and left lobe of the liver. The largest one showed a lobulated margin measuring 6.7 cm × 5.5 cm. The masses did not show typical characteristics of hemangioma or simple hepatic cyst. Also noted were fatty liver changes. There was no older study for comparison at that time. A short term repeat CT scan was recommended but she was lost to follow-up. During this admission, CT abdomen with intravenous contrast showed hepatic masses, largest being 12 cm × 5.5 cm, and development of small pulmonary nodules worrisome for metastasis . Small lInitial gastrointestinal evaluation revealed normal bilirubin, PT, PTT, and INR. Hepatitis B and hepatitis C were both negative. She did exhibit elevated alkaline phosphatase of 311 IU/L and elevated cancer markers, including CEA of 6.0 ng/mL, CA 19-9 of 74.8 units/mL, and CA 125 of 36.3 units/mL. Ultrasound guided liver biopsy was elected at this time and pathology was consistent with HEH . OncologThe term epithelioid hemangioma was coined by Weiss and Enzinger in 1982 to describe a distinct entity, a soft tissue vascular tumor of endothelial origin with a clinical course intermediate between benign hemangioma and malignant angiosarcoma . Both muIn regard to treatment, resection is preferable with a 75% 5-year survival rate. However, resection is usually only an option in 10% of the cases . TherefoIn 1957, thalidomide was first released in West Germany. It was intended as a hypnotic and sedative medication but soon found favor working against nausea in morning sickness during pregnancy. Common side effects include somnolence, edema, hypotension, myelosuppression, peripheral neuritis, interstitial lung disease, and pneumonia. Sadly, thalidomide was also discovered to have a high association with birth defects such as phocomelia, a malformation of limbs. The phocomelia was severe with formation of the limbs appearing to be stunted and underdeveloped. 10,000 children were thought to be affected before it was taken off the market. One proposed teratogenic mechanism includes halting of angiogenesis by inhibition of fibroblastic growth factor (bFGF) and/or VEGF, which has been verified with in vivo studies . Thalidoα, various interleukins  production, and NF-κB and COX-2 activity. It modulates the production of IFN-γ and enhances the production of IL-2, IL-4, and IL-5 by immune cells. It also increases lymphocyte count, costimulates T cells, and modulates natural killer cell cytotoxicity [Other than its effects on vascular growth, thalidomide also inhibits tumor necrosis factor (TNF)-toxicity . Due to In conclusion, both the diagnosis of HEH and the use of thalidomide in a vascular mediated tumor are extremely rare. Given the mechanism of action against angiogenesis, thalidomide does show some benefit in treatment for such cancers. Furthermore, given the direction of cell receptor and/or growth factor guided chemotherapy, thalidomide may hold promising future derivatives that can better target malignant hepatic hemangiomas and other malignancies.


Severe acute pancreatitis often leads to pancreatic and peripancreatic collections but, rarely, it can lead to collections at sites remote from the pancreas. Three male patients presented with abdominal pain and inguinoscrotal swelling. They were initially misdiagnosed with obstructed inguinal hernia, epididymo-orchitis and hydrocele, respectively. Later, their diagnosis of acute pancreatitis was revealed on laparotomy in one patient and on computed tomography (CT) in the remaining two patients. All these cases had extensive peripancreatic necrosis and paracolic collections tracking along the psoas muscle, downwards towards the pelvis. These collections were initially managed by percutaneous drainage and saline irrigation as a part of the ‘step-up’ approach. Two of these patients required open necrosectomy, while all required incision and drainage of inguinoscrotal collections. All the patients were discharged in satisfactory condition. Inguinoscrotal swelling is unusual as a first presentation of acute pancreatitis. A high index of suspicion, with careful study of patient's history and examination along with CT, may provide an accurate diagnosis. Local drainage may be required to control sepsis and also provide an egress route for intra-abdominal collections. Reports of pancreatic collections, presenting at sites remote from the pancreas, are available in the literature , 2. The A 35-year-old male presented to a peripheral hospital with acute-onset epigastric pain radiating to the back, associated with bilious vomiting and left inguinal swelling for the previous 7 days. The patient was misdiagnosed as having an obstructed inguinal hernia and inguinal exploration was carried out. The contents of the left inguinal canal were inflamed, with haemorrhagic fluid in the inguinal canal seen to be arising from the deep inguinal ring. The patient was then converted to midline laparotomy. At exploratory laparotomy, there was fat saponification of the greater omentum and peritoneum, suggestive of acute pancreatitis. The abdomen was closed and the patient was referred to our institute. The patient's clinical history revealed that he was chronically alcoholic and had a ‘binge’ of alcohol 15 days previously, after which he had developed abdominal pain. The swelling in the inguinal region had developed later when the pain worsened. The patient was evaluated with a contrast-enhanced computed tomography (CECT) of the abdomen and pelvis. There was pancreatic necrosis with fluid collection in the peripancreatic region, left pararenal space, extending to the pelvis along the left psoas muscle. There was inflammatory fat stranding in the region of the left spermatic cord and left scrotum with oedema and bilateral pleural effusion A. The paRight retroperitoneal drainage of the peripancreatic- and right pararenal space necrosis and collections was done through a flank incision 24 days after the attack of pancreatitis. Repeat CECT abdomen was done after 25 days of surgical intervention which revealed undrained collections in the pelvic retroperitoenum and, in view of persistent high-grade fever, the patient were re-explored on the next day. Midline laparotomy revealed peripancreatic necrosis tracking along the right pass to the pelvis, pre-vesical space, right lateral pelvic wall and right inguinal space. Debridement and wide drainage was done. Subsequently, the patient recovered well and was discharged in a satisfactory condition after the second surgery.A 48-year-old male presented with acute-onset upper abdominal pain radiating to the back for the previous 5 days. He also had abdominal distension, nausea, vomiting and a painful swelling in the scrotum. On examination, there was tenderness in the epigastrium and right lumbar region. The scrotum was swollen and tender, with inflammatory changes of the skin suggestive of acute epididymo-orchitis. A CECT of the abdomen and pelvis carried out on admission revealed a dilated main pancreatic duct with stranding and fluid collection around the uncinate process extending to the right pararenal space, right paracolic gutter and pelvis with fat stranding of the root of mesentery, retroperitoneum and scrotum . The patin situ. On subsequent follow-up, the scrotal drain and the pigtail catheters were removed.A 28 year old male presented with acute abdominal pain radiating to the back and breathlessness after an alcohol binge to an outside hospital 2 days after the onset. He was diagnosed with severe acute pancreatitis by CECT done at that hospital. On post pain day 8, he was referred to our Institute. He was initially managed conservatively. He developed a painful swelling in the left scrotum 10 days after onset of acute pancreatitis. USG of the scrotum showed a fluid collection in the scrotal sac with multiple septations and echogenic content. Both testes and the epididymis were normal. The fluid obtained by aspiration showed haemorrhagic fluid, had no pus cells and was sterile on culture with high amylase. Scrotal support and anti-inflammatory agents were given. After 4 weeks of conservative management, the patient continued to have a high-grade fever. A CECT showed large peripancreatic and left paracolic collections, which were drained by pigtail as part of the step-up approach A and 3B.Severe acute pancreatitis manifesting as inguinoscrotal swelling is a rare presentation, causing a diagnostic dilemma , 4. The tunica vaginalis, is generally spared from the inflammation.Most commonly, inflammatory fluid and necrosis extends into the anterior pararenal space and retromesenteric plane. The fluid and inflammation may spread posteriorly to involve the retrorenal plane, and laterally into the lateroconal plane, or spread inferiorly in the combined interfascial plane to reach the pelvic retroperitoneum or superiorly along the diaphragm to enter the mediastinum. These fascial planes provide a weak barrier to spreading inflammation . The pelet al. proposed a severity classification system for acute severe pancreatitis [Based on the extent of spread of peripancreatic fluid in retroperitoneal interfascial planes, Ishikawa reatitis . The graThe initial management of patients suffering from acute pancreatitis with an inguinoscrotal collection is similar to those without it. It involves fluid resuscitation, nutritional support, prophylactic antibiotics and intensive care in case of organ failure. In previously reported cases, this supportive care was successful in resolution of inguinoscrotal collections , 4. If tThe present study supports our viewpoint that cases of inguinoscrotal swellings in severe acute pancreatitis may have variable extents of inflammation and therefore a different clinical course. Management thus has to be tailored according to the extent of spread of retroperitoneal inflammation. The value of serial computed tomography cannot be over-emphasized in this scenario. In addition, depending upon the severity of necrosis in the mesocolon, the vascularity of the affected colon may also be jeopardized, as seen in one of our cases in this series, which required sub-total colectomy.To summarize, inguinoscrotal swelling as a first presentation of acute pancreatitis is unusual. Scrotal collections appearing in the course of acute pancreatitis may contribute to continued sepsis. A high index of suspicion with a careful study of patient's history and examination along with computed tomography of the abdomen and pelvis may provide an accurate diagnosis and extent of disease. Local drainage may help in the resolution of sepsis, both by drainage of the collection and also by providing an egress route in patients undergoing pigtail catheter irrigation of a pancreatic collection. There may still be undrained collections in other dependent areas, which require separate management for a successful outcome.Conflict of interest statement: none declared.


Keratocystic odontogenic tumors are benign neoplasms of odontogenic origin with a potential for aggressive and infiltrative behavior. Many different treatments for this type of lesion have been reported. However, no common consensus has emerged to date regarding the most effective therapeutic approach. Cases of maxillary sinus giant keratocystic odontogenic tumors completely excised by enucleation or marsupialization via endoscopic sinus surgery are extremely rare, and, to the best of our knowledge, only one case has been described in the literature since 2005.We report a case of a 24-year-old Italian man who came to our department with maxillary sinus region swelling, pain and left nasal obstruction. A massive keratocystic odontogenic tumor involving the right maxillary sinus and causing focal erosions of the bony walls was diagnosed. The keratocystic odontogenic tumor was removed as much as possible by a transnasal approach using endoscopic sinus surgery, which produced optimal surgical and prognostic outcomes. Follow-up is reported for an 8-year period.Conservative management in this case demonstrated good therapeutic efficacy with a low risk of recurrence. For injuries involving the maxillary sinus, the possibility of decompression or marsupialization by endoscopic sinus surgery should always be considered because it demonstrated the potential to lead to excellent results even after 8 years of follow-up in our patient. To our knowledge, no case report has described follow-up longer than 8 years for a maxillary sinus keratocystic odontogenic tumor treated with endoscopic sinus surgery. The or (KCOT) ,6. The nr (KCOT) ,5,7.en bloc resection, have been reported. However, no common consensus regarding the most effective therapeutic approach has emerged to date [Many different treatments for this type of lesion, ranging from simple curettage to highly invasive  to date ,2.Although several cases of odontogenic parakeratotic cysts have been reported in the literature, only a few were related to the new WHO classification ,3. In adIn this case report, we present our experience in treating a patient with a KCOT removed by means of an ESS approach. There was no recurrence during 8 years of follow-up.A 24-year-old Italian man came to our department complaining of a 3-month history of left maxillary sinus region swelling with left nasal obstruction and associated mild facial pain. He had a history of repeated cycles of antibiotic therapy without any symptomatic improvement.At the clinical examination, tumefaction was found to involve the body and ascending ramus of the maxillary sinus with intact overlying skin. No epistaxis, peri-orbital proptosis, cranial nerve palsies or paresthesias were evident. Fiber-optic rhinoscopy showed that the middle nasal meatus was partially occupied by a reddish mass that was not bleeding, together with strongly hyperemic surrounding mucosa. Erosion of the medial walls of the maxillary sinus could be seen. The right nasal cavity was apparently normal.A computed tomography (CT) scan of the paranasal sinuses showed irregularly dense solid tissue that entirely occupied the right maxillary sinus, with massive erosion of the anterior and posterior walls of the sinus. Inferiorly, the mass encased the dental roots of the corresponding right dental arch, and its extension into the nasal cavity created induced compression and deflection of the nasal septum. No enhancement after contrast administration was evident Figure .To remove the mass, ESS was performed. After surgical volume reduction using radiofrequency of the inferior turbinate, the maxillary ostium was enlarged and the cystic lesion was opened and decompressed. Marsupialization was performed, and the cystic wall was also removed as much as possible.The histological examination revealed the presence of a cyst wall characterized by a few cell layers of squamous parakeratotic epithelium. In some areas, the epithelium was dehiscent with granulation tissue. Lymphocytes and plasma cells could be seen Figure . Such hiThe post-operative evaluation revealed no recurrence of the disease, with steady improvement in the patient’s clinical condition. Owing to the high tendency of KCOTs to recur, we opted for a long radiological follow-up with CT performed annually for the first 5 years and every 3 years thereafter.At the patient’s 8-year follow-up examination, a post-operative CT scan showed no KCOT recurrence in the right maxillary sinus. Scarce pathologic tissue could be seen in the alveolar recess of the right maxillary sinus (Figure et al. [In 2005, the new histological classification of odontogenic tumors was introduced by the WHO, which re-classified the OKC as a benign intra-osseous neoplasia, the so-called keratocystic odontogenic tumor . Originaet al.  reportedWith regard to symptoms, several researchers have reported that 50% to 90% of KCOTs are symptomatic at the time of diagnosis . The chiKCOT is characterized by very aggressive local behavior with a high rate of recurrence, ranging from 3% to 60% ,8-10. ItKCOTs may appear as small, round or ovoid, multi-lobulated lesions with well-defined margins and a radiolucent appearance on CT scans . Signs oet al. [et al. [In a series of 32 surgically treated KCOTs, Kuroyanagi et al.  observedet al. -11. Recuet al. . WoolgarThe appropriate treatment of KCOT lesions is still debatable -3. Most Recently, the surgical risk, morbidity and relatively high rate of recurrence even after the use of aggressive surgical techniques have resulted in increased use of conservative treatment -3,9,12. et al. [Pogrel et al.  describeet al. [It should be noted that if KCOTs exist in the maxilla, which is adjacent to the nasal cavity, the ESS via the transnasal approach should be considered suitable for marsupialization . Ohki etet al.  are the et al. .Our case report confirms the data reported in the literature to date regarding the clinical and histological features of odontogenic parakeratotic cysts. Our conservative management of our patient demonstrated good therapeutic efficacy with a low risk of recurrence. For injuries involving the maxillary sinus, the possibility of decompression or marsupialization by ESS should always be considered and has the potential to produce excellent results even after several years of follow-up.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.ESS: Endoscopic sinus surgery; KCOT: Keratocystic odontogenic tumor; OKC: Odontogenic keratocyst; WHO: World Health Organization.The authors declare that they have no competing interests.MD acquired data. GI and MZ acquired and interpreted the data and wrote the manuscript. KM analyzed and interpreted the patient data and wrote the manuscript. GM was a major contributor in writing the manuscript, analyzed interpreted the patient data. All authors read and approved the final manuscript.


Characterizing ovarian masses enables patients with malignancy to be appropriately triaged for treatment by subspecialist gynecological oncologists, which has been shown to optimize care and improve survival. Furthermore, correctly classifying benign masses facilitates the selection of patients with ovarian pathology that may either not require intervention, or be suitable for minimal access surgery if intervention is required. However, predicting whether a mass is benign or malignant is not the only clinically relevant information that we need to know before deciding on appropriate treatment. Knowing the specific histology of a mass is becoming of increasing importance as management options become more tailored to the individual patient. For example predicting a mucinous borderline tumor gives the opportunity for fertility sparing surgery, and will highlight the need for further gastrointestinal assessment. For benign disease, predicting the presence of an endometrioma and possible deeply infiltrating endometriosis is important when considering both who should perform and the extent of surgery. An examiner’s subjective assessment of the morphological and vascular features of a mass using ultrasonography has been shown to be highly effective for predicting whether a mass is benign or malignant. Many masses also have features that enable a reliable diagnosis of the specific pathology of a particular mass to be made. In this narrative review we aim to describe the typical morphological features seen on ultrasound of different adnexal masses and illustrate these by showing representative ultrasound images. The characterization of ovarian masses and distinguishing between benign and malignant pathology is important both to decrease unnecessary anxiety and enable decisions regarding optimal treatment. Benign pathology may be best treated conservatively or in a general gynecology unit using a minimal access approach. Conversely, suspected malignant masses should be referred to specialized units for further management. Thus prior knowledge of the nature of ovarian masses is essential not only for the patient but in order to organize clinical services in terms of planning, costs and overall management .Transvaginal ultrasonography (TVS) is the most commonly employed imaging modality for the assessment of adnexal masses, and a number of prediction models have been created to maximize its predictive capability. In many countries the risk of malignancy index (RMI)  which coFor the purposes of this review, the term ‘pattern recognition’ refers to the subjective evaluation of adnexal masses using grey-scale and power/color Doppler ultrasonography ,12. In tThese findings suggest that with adequate training and knowledge of the common features associated with particular pathologies, ultrasound examiners should be able to reliably diagnose and differentiate between certain specific types of adnexal pathology. It is important to remember that when evaluating women with an adnexal mass, ultrasound characteristics need to be correlated with the clinical history, as well as signs and symptoms before arriving at a diagnosis. This review describes only the features that may be found using ultrasound that may be used to predict common specific types of adnexal pathology.They are usually unilocular and thin walled with anechoic contents . They raThese are formed following the rupture of a mature Graafian follicle. They are thick walled hyperechoic cysts that typically demonstrate peripheral circumferential blood flow, sometimes known as the ‘ring of fire’ . Some cyPeritoneal pseudocysts, are collections of peritoneal fluid trapped in adhesions usually caused by previous pelvic surgery, pelvic inflammatory disease or endometriosis. They usually occur in premenopausal women, because of the presence of functional ovaries that release small amounts of fluid into the peritoneal cavity –18. TheyPseudocysts appear mainly as multilocular cysts, with a high number of septa that are adherent to the ovarian surface. Septa are most frequently complete and thin –18 Fig.. In contThe ipsilateral ovary is visible in almost all cases . It can Paraovarian cysts arise in the broad ligament between the ovary and the fallopian tube. They account for 5–20% of adnexal masses ,20. The Their mean diameter is usually <5 cm with no evidence of any follicles or significant vascularity. In almost all cases, it is possible to visualize the ipsilateral normal ovary, and to detect movement of the cyst in the opposite direction to the ovary when the area is pushed with the vaginal probe - the ‘split sign’. This may help to differentiate between a paraovarian and ovarian cyst when the ipsilateral ovary is not clearly visible .A normal Fallopian tube is rarely visible during an ultrasound examination. Hydrosalpinges have typical diagnostic features on ultrasound with anechoic contents and incomplete septae  Fig. 6)Fig. 6). Acute salpingitis typically appears like a pear-shaped unilocular mass with anechoic or low-level content, characterized by thickening of the wall (>5 mm) and the presence of incomplete septae . In tranIn chronic salpingitis the tube appears as an elongated fluid-filled mass, with incomplete septae, but the thickening of the wall is no longer visible. It is characterized by the typical sonographic ‘beads on a string’ sign, due to 2–3 mm sized hyperechoic structures on the tubal wall, seen on transverse section ,22–24.A tubo-ovarian complex represents the involvement of ovarian tissue in the inflammatory process. Normal ovarian parenchyma is visible, but it is usually seen separate from tubal structures ,22–24 F.In a tubo-ovarian abscess, ovarian tissue is no longer visible; the lesion may be unilocular, solid or multilocular-solid with mixed or ground-glass echogenicity. On the basis of the ultrasound features, these have to be differentiated from endometriomas or hemorrhagic cysts ,22–24. IThese appear as smooth, thin walled, anechoic, fluid-filled structures. They are bilateral in 15% of cases and their mean size is 5–8 cm . Some coMucinous cysts are classically thin walled, large and unilateral. They consist of internal thin-walled locules containing mucin which appears as fluid with low level echogenicity  Fig. 11Fig. 11. et al described the presence of variable echogenicity among different tumor locules as an ultrasound feature of multilocular mucinous cystadenomas . It is this ‘ground glass’ feature that is the most typical feature ,31–33 F.Endometriomas may also have atypical features, and frequently debris within the cyst may give the impression that it is a unilocular-solid lesion with solid papillary projections. In postmenopausal women the appearances of an atypical endometrioma should be examined very carefully as there is a significant risk of malignancy in such lesions in this age group ,32 Fig..During pregnancy endometriomas can change their appearance secondary to decidualization. The features may become quite alarming, with solid vascular projections into the cyst cavity. When no pre-existing scan of the ovary is documented it is difficult in these cases not to suspect malignancy , althougThese are benign tumors of stromal origin. Fibromas originate from spindle cells producing collagen and can be associated with ascites or Meig’s syndrome. Fibrothecomas originate from both spindle and theca cells and may produce a small amount of estrogens ,37.Their characteristic sonographic appearance is of a round or oval solid tumor, with regular margins. They may have stripy acoustic shadows, but these are present in just a small percentage of cases ,36,37 F. FibromaStruma ovarii is a rare subtype of mature teratoma characterized by the presence of ectopic thyroid tissue. They account for <5% of mature teratomas . AlthougBrenner tumors also arise from the ovarian stroma but are benign in 99% of cases. Their diagnosis is often an incidental finding in women between the fifth and the seventh decade of life. They are usually small and often coexist with serous or mucinous cystadenomas . They arStage 1 primary invasive ovarian epithelial cancers share similar ultrasound characteristics to borderline tumors, but they differ significantly from the appearances of later stage disease  Fig. 22Fig. 22. Later stage primary ovarian tumors are usually multilocular with a high proportion of solid tissue and are frequently associated with ascites as well as metastatic disease to the peritoneum, omentum and elsewhere in the abdomen and pelvis . They arThe presence of papillary projections within a cyst has been used as a discriminatory factor for serous borderline tumors . HoweverSerous and mucinous endocervical type BOTs are usually unilocular solid tumors with a high number of vascular papillary projections within the cyst. Mucinous intestinal type BOT are more often very large, unilateral, multilocular tumors with a high number of locules encased by thick, hyperechoic tissue with no evidence of solid components –26. TheyOvarian metastasis from breast, gastric, and uterine cancers as well as lymphomas appear as solid tumors on ultrasound examination  and 28. The vascularity of metastatic tumors is characterized by the presence of a ‘lead vessel’ - a single large vessel penetrating from the periphery to the central part of the lesion . FurtherPredicting the specific histopathology of an adnexal mass is important as it may lead to surgery being avoided or being less invasive in some cases whilst ensuring appropriate referral to a gynecological oncology surgeon in the case of malignancy. In general there is an intense focus on excluding malignancy when the characterization of ovarian pathology is considered. However the field has moved on, both in terms of tailoring treatment to individual patients and with what we know about the features of different types of ovarian pathology. In this review we hope we have illustrated some of the pathognomonic features of some of the more commonly found adnexal masses in clinical practice. By improving the specific classification of masses we hope that management decisions in relation to such pathology will become more patient specific and lead to improved outcomes.


Malignant schwannoma is an extremely rare tumor and the risk of malignant schwannoma increases in patients with von Recklinghausen's disease. Recently, we encountered a case of solitary malignant schwannoma in the choana and posterior nasal septum. Malignant schwannoma has not been previously reported in these locations. A 53-year-old man, who was immunologically healthy and showed no abnormal dermatological lesions, presented with a polypoid mass in the right nasal cavity and underwent endoscopic mass excision. The mass originated from the choana and the posterior portion of the right nasal septum. This mass was confirmed as a malignant schwannoma on histological examination and immunohistochemical staining. After endoscopic excision, postoperative adjuvant radiotherapy was administered, and there was no recurrence at 1 year after treatment. This case suggests that a solitary malignant schwannoma should be considered in the differential diagnosis of a mass in the posterior nasal cavity. Malignant schwannomas represent approximately 10% of soft tissue sarcomas and are rarely found in the head and neck region. The most common locations are the extremities, trunk, chest, and retroperitoneum . MalignaA 53-year-old man presented with nasal obstruction and frequent epistaxis that had persisted for 3 months. He had well-controlled hypertension and diabetes mellitus. No abnormal dermatological spots were found on the body and the serological test for human immunodeficiency virus was negative. Endoscopic examination revealed a polypoid mass extending from the midportion of the right inferior turbinate to the choana. Images of paranasal sinus computed tomography (PNS CT) revealed a 2.5 cm long and heterogeneously enhanced mass filling the right nasal cavity with extension to the right choana and nasopharynx and some erosion of the septal bone Figures . ParanasIn the present case, the patient did not have acquired immune deficiency syndrome or any dermatologic abnormalities such as café-au-lait spots that are suggestive of von Recklinghausen's disease. Therefore, this case is the first reported case of a solitary malignant schwannoma confined to the choana and posterior portion of the nasal septum. The diagnosis of malignant schwannoma is confirmed by hematoxylin and eosin staining as well as immunohistochemical staining with specific markers that are expressed in Schwann cells. Histologically, malignant schwannoma has a high cellularity with spindle-shaped cells arranged in bundles, palisades, or whirls. Spindle-shaped cells arranged in sweeping fascicles have a moderate amount of cytoplasm and large multilobulated nuclei. In addition, dense cellular areas alternate with hypocellular and myxoid zones , 12. SchImaging features of malignant schwannoma are generally nonspecific. Benign schwannomas are usually well encapsulated, whereas malignant schwannomas may show extensive infiltration. Bone destruction can occur secondary to pressure erosion; therefore, such a finding does not necessarily indicate malignancy . IntraleTreatment modalities for malignant schwannoma consist of surgical excision, systemic chemotherapy, or radiotherapy. Complete surgical excision with a safety margin is the mainstay treatment in patients with localized diseases. The role of radiotherapy and chemotherapy remains controversial; however, radiotherapy has been indicated in cases in which the tumor cannot be completely resected . In the From our review of the literature, it is the first report of malignant schwannoma arising from septum extending to the posterior choana. Although the mass was excised using a piecemeal technique, it is likely that this patient may have a favorable prognosis because the tumor was solitary and confined to the choana and nasal septum and was not associated with von Recklinghausen's disease.


Diverticular perforation is a common complication of diverticulitis and can lead to the creation of abscesses. The presence of such abscesses on the abdominal wall is rare and can lead to misdiagnosis. We present the case of a patient with abdominal pain and the formation of a large left lumbar abscess due to perforation of a diverticulum of the left colon and our surgical treatment of choice with favorable results. Diverticular disease affects mainly patients over forty years of age especially in the western countries, with left colon representing the most common site of presentation . Most paActually, diverticulum perforation may, usually, lead to abscess formation because of the inflammation of the nearby structures through tissue continuity. However, the presence of an abscess on the abdominal wall due to such perforation is rare. We present the rare case of a patient with a large perforated left lumbar abscess and vague abdominal pain that was diagnosed as diverticular perforation and was treated surgically, following the existing guidelines .μL with 74.5% neutrophils, Hb 11.2 gr/dL, and Ht 34.3%. His vital signs were between normal ranges. From his medical history he revealed two episodes of acute diverticulitis during the past five years , for which he followed conservative treatment. At the time of admission, he was not on immunosuppressant or any other medication.A 50-year-old male patient was admitted at the Emergency Department of our clinic complaining about mild diffuse abdominal pain, with no specific location of tenderness. This pain presented the previous day, along with fever up to 38.3°C. He did not mention any other symptoms, such as nausea, appetite loss, or bowel dysfunction. Prior to our examination, the patient was referred to the Internal Medicine Emergency Department, where he was scheduled to have a blood test and an X-ray examination of the abdomen . His labDuring the clinical examination, a mild diffuse sensitivity during palpation of the left abdominal area was observed, without any specific signs of abdominal pain. However, the patient's inspection revealed an abscess in the left lumbar area, which, according to the patient, was present for about three months and tended to reduce in size after defecation. The abscess seemed uncomplicated, not too large, and manageable through drainage under local anesthesia on bedside. Therefore, it was incised, but almost two liters of pus-like fluid was drained . Even thDue to these imaging findings in combination with the patient's condition, we decided to perform an exploratory laparotomy, during which a perforated diverticulum of the left colon was revealed. This perforation led to the inflammation of the posterior abdominal wall through tissue continuity and resulted in the formation of the lumbar abscess. However, there were no signs of generalized peritonitis, as the fascia was not infected. The inflamed segment of the colon was excised at healthy margins and an end-to-end anastomosis was performed Figures . The posDiverticulitis is staged according to the classic Hinchey classification as pericolic abscess or phlegmon (Hinchey I), pelvic, abdominal, or retroperitoneal abscess (Hinchey II), generalized purulent peritonitis (Hinchey III), and generalized fecal peritonitis (Hinchey IV). Many other classifications have been reported also  . The usuIn our case, the perforation appeared as a left lumbar area abscess of three-month duration, for which the patient did not seek treatment before, as he did not present any other symptoms. Apart from our case, only two other cases have been described clearly as an abscess in the lumbar area following diverticulum perforation in the literature  11, 12], 1211, 1The management of acute diverticulitis includes fluid and antibiotic support, percutaneous drain abscess drainage or resection, and 1- or 2-stage anastomosis, depending on each individual case . The firMany studies have shown improved results through primary anastomosis, depending on the surgeon's experience , while oAbdominal CT is the imaging examination of choice, as it sets the definite diagnosis, shows the extent of abdominal inflammation, and excludes any other causes of acute abdominal pain . In our The diagnosis of a diverticular perforation, especially when it appears with nonspecific symptoms as a lumbar area abscess, may be difficult to establish and is often misinterpreted. A detailed clinical history must be taken and the patient should be examined thoroughly. Imaging findings such as CT can help in the differential diagnosis and lead to the precise diagnosis and to early surgical treatment.


Osteoid osteoma is a bone-producing lesion frequently localized in long bones. The most common presenting symptom is pain with nocturnal exacerbation, responsive to nonsteroidal anti-inflammatory drugs . MinimalSeven patients affected by osteoid osteoma were consecutively recruited for MRgFUS . The diagnosis of osteoid osteoma was based on typical clinical and imaging findings (CT and MRI) . Pain and function were scored before and after MRgFUS , and any intra- and post-procedure complication or adverse effect was recorded. Initial follow-up included clinical assessment at discharge, and every month.2). This condition did not allow a correct positioning of the patient to find the optimal ultrasound beam window. Six males, ageing 18-46 years old  and presenting with pain scoring 5 to 10 (7.1±2.1) underwent MRgFUS . All osteoid osteomas were located at femur , and in one case the treatment was proposed for recurrence after being treated by radiofrequency. All treatments were performed under spinal anesthesia. The day after the procedure VAS score was 2.3±1.6 (0-5) (p<0.001), and dropped to 0 in all patients from the 1-month check onward. Table In one patient the lesion could not be approached due to the high body mass index (33 Kg/mMRgFUS of osteoid osteoma is an extremely promising treatment. Our first experience confirms its safety and efficacy. Additional imaging evaluation and an extended follow-up will be able to support preliminary evidence and to examine the biological evolution of the bone lesion.


Basal encephaloceles are rare clinical entities. Intrasphenoidal encephalocele (ISE) is a rare form of basal encephalocele. The clinical presentation is often subtle and may remain undetected. Only a limited number of cases of ISE have been reported, mostly in middle-aged patients who presented with symptoms. Clinical diagnosis and management remain a challenge. Our case reports ISE as an incidental finding in a very elderly male patient.We present a case of ISE discovered as an incidental finding in a 99-year-old male who presented to the emergency department (ED) after a fall from bed. The encephalocele was discovered in the evaluation of the computed tomography (CT) scan of the head.This is an incidental manifestation of a rare disease. It demonstrates that these encephaloceles may not manifest clinically or symptomatically as other reports have suggested. Basal encephaloceles are rare clinical entities. The clinical presentation is often subtle and may remain undetected. We present a case of intrasphenoidal encephalocele discovered as an incidental finding in a 99-year-old male who presented to the emergency department (ED) after a fall from bed. The encephalocele was discovered in the evaluation of computed tomography (CT) scan of the head. This is an incidental manifestation of a rare disease. It demonstrates that these encephaloceles may not manifest clinically or symptomatically as other reports have suggested.A 99-year-old male presented to the ED after he fell from his bed. History was obtained from the home attendant, who accompanied the patient, as the patient was non-communicative because of severe dementia. As per the home attendant, the patient fell from his bed from a height of approximately 4 feet. The attendant noticed a swelling on the forehead of the patient. The patient remained dazed for a while but there was no vomiting or abnormal movement. His past medical history was significant for Alzheimer’s dementia, hypertension, diabetes mellitus, and benign prostatic hyperplasia.In the ED, he was in no apparent distress and was at his usual baseline mental status. His vital signs were as follows; temperature 36.5°C (97.8°F), heart rate 82 beats per minute, respiratory rate 20 breaths per minute and blood pressure 125/60 mmHg. On physical examination, a soft tissue hematoma was noted on the forehead. His pupils were equal and reactive, and extraocular movements were intact. A linear 2-cm laceration was noted on his right periorbital region. There was no hemotympanum. There was no neck tenderness. His chest was clear with bilateral breath sounds. His abdomen was soft and non-tender. There was no swelling or tenderness over his extremities. The remainder of the physical examination was unremarkable.A CT scan of the head and facial bones was obtained. The images demonstrated severe dilation of the ventricles and sulci suggestive of cerebral and cerebellar cortical atrophy. Also noted was a new right parietal white matter hypodense (ischemic) lesion and brain material within the right side of the sphenoid sinus suggestive of an intrasphenoidal encephalocele Figures  and 2.Intrasphenoidal encephalocele [ISE] is a rare form of basal encephalocele. Only a limited number of cases of ISE have been reported; among these, most were reported in middle-aged patients who presented with findings of cerebrospinal fluid rhinorrhea  and moreEncephaloceles are divided into two types: anterior and posterior. Posterior encephaloceles account for three-fourths of those described and anterior for only about 1.5% of all encephaloceles . On the Though intrasphenoidal encephaloceles are classified as a type of transphenoidal encephalocele, they should be considered a completely separate entity . IntraspThe clinical presentations of ISE are caused by the involvement of the structures surrounding the sinus . The mosSurgical management of basal encephaloceles should be preceded by careful evaluation and establishing the location of the defect . ExtracrSince intrasphenoidal encephaloceles are such rare clinical entities, their diagnosis and management remain a challenge. Our case report demonstrates a completely asymptomatic presentation, which further emphasizes the necessity for radiological diagnosis.ED: Emergency department; ISE: Intrasphenoidal encephalocele.The authors declared that they have no competing interests.MW contributed to the concept, design and revision for important intellectual content. RU carried out the acquisition of data and drafting of the manuscript. HA contributed to the radiological findings and related intellectual content. SA gave final approval of the version to be published. All authors read and approved the final manuscript.


Uterine sarcomas spread via lymphatic and hematogenous dissemination, direct extension, or transtubal transport. Distant metastasis often involves the lungs. Ovarian metastasis is uncommon. Here we present an unusual case of a large, high-grade uLMS with metastatic disease internal to both ovaries without capsular involvement or other abdominal diseases, and discovered in a patient with distant metastases to the lungs, suggesting likely hematogenous dissemination of uLMS to the ovaries in this case. Knowledge of usual uLMS metastases may influence surgical management in select cases. Uterine leiomyosarcoma (uLMS) is an aggressive malignancy accounting for 1-2% of uterine malignancies and one-third of uterine sarcomas. The diagnosis of uLMS is often made incidentally during or after hysterectomy or myomectomy for presumed benign leiomyomas. Histopathologic diagnosis is determined by the presence of abundant mitoses, coagulative tumor cell necrosis, hypercellularity, and cellular atypia. Thirty percent of uLMS cases present with extrauterine disease [Treatment of uLMS includes total abdominal hysterectomy (TAH) with or without bilateral salpingo-oophrectomy (BSO) . AdjuvanA 53-year-old postmenopausal woman, gravid 4 para 3, presented to the emergency department with one week of left lower quadrant abdominal pain. She denied weight loss, fever, chills, shortness of breath, bloating, or any changes in bowel or bladder function. On physical exam, she demonstrated mild abdominal distention and mild, diffuse abdominal tenderness without guarding or rebound. Computed tomography (CT) of the chest, abdomen, and pelvis revealed bilateral adnexal masses (11.7 × 6.9 cm on the left and 6.4 × 7.7 cm on the right), an 8.9 × 8.4 cm heterogeneous lesion occupying the uterus, pelvic ascites, and extensive bilateral pulmonary nodules . Tumor mThe patient underwent TAH/BSO. Lymphadenectomy was omitted as preoperative CT imaging and intraoperative palpation did not suggest lymphatic involvement. Final pathology demonstrated a high-grade 8.5 cm fundal uLMS and bilateral ovarian metastases. Histology included hypercellularity with tumor necrosis, cellular atypia, and mitotic figures . ImmunohGiven imaging evidence of pulmonary metastases, the patient was considered stage IVB and received adjuvant docetaxel and gemcitabine. Interval CT imaging showed decreased size and number of pulmonary nodules without evidence of disease progression, suggesting good clinical response. She remains asymptomatic and without evidence of disease progression 15 months after diagnosis.Uterine sarcomas theoretically may metastasize via direct invasion, transtubal transport, or lymphatic or hematogenous dissemination. Recently Tirumani et al. found metastasis in 81% of patients with uLMS, most commonly involving the lungs (74%), peritoneum (41%), bones 33%), or liver (27%). Ovarian metastasis is less frequent. Seven cases of uLMS with ovarian metastasis  and six 3%, or liuLMS often spreads hematogenously because uLMS originates within the richly vascular myometrium and frequently presents with distant metastases. uLMS has been reported in a variety of distant locales, including the brain, gastrointestinal tract, and heart, typically with coexisting pulmonary metastases . PulmonaUterine malignancies that spread lymphatically often cause regional lymphadenopathy before distant metastasis, a pattern well recognized in epithelial-derived endometrial cancers. Malignant cells traveling via lymphatic networks in endometrial cancers have been mapped to pelvic and para-aortic lymph nodes, including external iliac, iliac vessel bifurcation, and aortic bifurcation nodes . LymphatAlvarado Gay and Vega Silva described a case of uLMS presenting as a 10 cm right-sided uterine mass extending into the ipsilateral fallopian tube and ovary, involving the ovarian serosa, and suggesting direct extension . AnotherIt is important to also consider the possibility of synchronous development of multiple primaries. Approximately 1-2% of women with a gynecologic cancer will have two simultaneous gynecologic malignancies, the most common combination being simultaneous endometrial and ovarian malignancies . LeiomyoHere we present an unusual case of a large, high-grade uLMS with metastatic disease internal to both ovaries without capsular involvement or other abdominal diseases and discovered in a patient with distant metastases to the lungs, suggesting likely hematogenous dissemination of uLMS to the ovaries in this case. Blood leaving the uterus as venous return coalesces into the uterine venous plexus and then it can enter the ovarian vein, a conduit to the ovarian venous plexus . Tumors Given that the incidence of ovarian metastasis due to uLMS is approximately 2–5%, premenopausal women hoping to avoid surgically induced early menopause may consider ovarian preservation , 4. NCCN


An increased number of adrenal tumors are now diagnosed due to the increased number of abdominal CT scans being performed. We present the first case of malignant lymphoma combined with clinically “silent” pheochromocytoma in the same adrenal gland. An abdominal CT scan demonstrates unilateral adrenal lesion which suggests pheochromocytoma or adrenal carcinoma. Laboratory examinations revealed a slight increase of 24-h urine vanillylmandelic acid and 24-h urinary methanephrine excretion. Histological examination revealed two intermingled tumor cell proliferations—diffuse B cell lymphoma and pheochromocytoma.Unexpected coexistence of catecholamine-producing tumor with the other adrenal lesion can lead to serious complications of diagnosis and treatment. The adequate preparation for surgery can protect patient from threatening catecholamine crisis. Lymphoma may occasionally involve the adrenal glands, but primary adrenal lymphoma (PAL) is very rare . AutopsyCombined adrenal neoplasms are also rare entities. We report herein, to the best of our knowledge, the first case of collision tumor composed of a primary adrenal lymphoma and a clinically “silent” pheochromocytoma.A 57-year-old man was admitted to the emergency department of Medical University of Gdansk with right abdominal pain suggesting cholelithiasis. There was a history of nausea, fatigue, and weight loss of 10 kg in the last 3 months. The patient had no other medical history.Primary physical examination revealed a normal blood pressure of 130/70 mmHg, pulse of 90/min, and no fever. The abdominal examination revealed a painful mass in the right side without hepatosplenomegaly. Neither signs of hypercortisolism nor lymphadenopathy were found.A contrast enhanced computer tomography (CT) was performed and showed a 13.2 × 11 cm solid right adrenal mass. The mass was homogenous with high Housfield score (51 HU) in basic condition, and contrast washout rate was 30 % after 10 min. No other abdominal lesions or lymphadenopathy were detected. Initial laboratory findings revealed a normal complete blood count, but erythrocyte sedimentation rate (ESR) was 74 mm/h , C-reactive protein (CRP) was 48 mg/L , and lactate dehydrogenase (LDH) was 240 IU/L . Hormonal laboratory tests showed a 24-h urine vanillylmandelic acid (VMA) excretion of 10.2 mg/24 h  and a 24-h urinary methanephrine excretion of 395 μg/24 h . Serum androstendione was slightly elevated to 4.58 ng/mL   and were not suggestive of lymphoma. A slight increase of catecholamine level with the absence of clinical symptoms of PHEO inclined us to small doses of alpha-blockers only—doxazosin 4 mg/24 h per os to control paroxysmal blood pressure rise during the surgery. Consequently, the patient underwent open right adrenalectomy. The mass was found to press the neighboring organs and vessels without evident infiltration. The tumor resection with right nephrectomy was performed.Macroscopically gray creamy tumor measuring up to 7 cm in greatest dimension was found in the right adrenal gland infiltrating periadrenal adipose tissue and the cortical part of the kidney. Renal parenchyma infiltration measured up to 3.5 cm.Gross lesion found in a section of the gland consisted of two parts: gray creamy tumor which destroyed the adrenal gland and the yellowish-brown spherical focus 1.8 cm in diameter which adheres to the adrenal mass. Adrenal tumor infiltrates periadrenal adipose tissue and the cortical part of the kidney  with the following immunophenotype: CD20(+), CD45(+), CD3(−), Ki67-50 %, CD5(−), CD10(−), BCL6(−), MUM1(−), and cyclin D1(−) , doxorubicin (50 mg/m2), vincristine (1.4 mg iv), and prednisolone (100 mg po).Before the therapy began, whole body CT scans and bone marrow aspiration biopsy was obtained. The absence of any lymphadenopathy and no bone marrow involvement ruled out a secondary lymphoma. The patient received chemotherapy: cyclophosphamide  and potassium level of 4.93 mmol/L . Due to the symptoms of hypotension, hyponatremia, and neoplasm enlargement of the left adrenal gland, adrenal insufficiency was suspected. The baseline serum cortisol was 149 nmol/L , and serum ACTH was elevated to 102 pg/mL , which indicated a primary adrenal insufficiency  and involvement of other organs are poor prognostic indicators , 3, 5. UPAL should be kept in mind in the differential diagnosis of patients with unilateral adrenal masses without nodal involvement in the absence of other malignancies , 13, 14.To the best of our knowledge, PAL and PHEO coexistence in one adrenal gland has never been described.Written informed consent was obtained from the patients for publication of this report and any accompanying images.


Perivascular epithelioid cell tumors of gastrointestinal tract (GI PEComas) are exceedingly rare, with only a limited number of published reports worldwide. Given the scarcity of GI PEComas and their relatively short follow-up periods, our current knowledge of their biologic behavior, molecular genetic alterations, diagnostic criteria, and prognostic factors continues to be very limited.We present 2 cases of GI PEComas, one of which showed an aggressive histologic behavior that underwent multiple combined chemotherapies. We also review the available English-language medical literature on GI PEComas-not otherwise specified (PEComas-NOS) and discuss their clinicopathological and molecular genetic features.Pathologic analyses including histomorphologic, immunohistochemical, and ultrastructural studies were performed to evaluate the clinicopathological features of GI PEComas, their diagnosis, and differential diagnosis. Immunohistochemistry, semiquantitative reverse transcriptase polymerase chain reaction, and DNA sequencing assays were carried out to detect the potential molecular genetic alterations in our casesPSF-TFE3 gene fusion in one of our cases. Furthermore, in this case microphthalmia-associated transcription factor and its downstream genes were found to exhibit elevated transcript levels.Microscopically, the tumors showed distinctive histologic features of PEComas-NOS, including fascicular or nested architecture, epithelioid or spindled cell type, and clear to eosinophilic cytoplasm. The tumor cells were immunohistochemically positive for melanocytic markers. Molecular pathological assays confirmed a Knowledge about the molecular genetic alterations in GI PEComas is still limited and warrants further study. The PECs also exhibit a distinct immunophenotype with a coexpression of melanocytic and myogenic markers, such as HMB45, Melan-A, MiTF, smooth muscle actin (SMA), and calponin.2 The PEComa family includes angiomyolipoma (AML), clear cell “sugar” tumor of the lung (CCST), lymphangioleiomyomatosis (LAM), clear cell myomelanocytic tumor of the falciform ligament/ligamentum teres (CCMMT), and unusual clear cell tumors in other locations. PEComas have been reported in various anatomic sites, with a marked female predominance. Due to their relative rarity, the diagnostic criteria, optimal treatment strategies, and prognostic factors for PEComas have not yet been confirmed at this time. We report 2 cases of PEComas arising in the gastrointestinal tract, including the clinicopathological features and potential molecular genetic alterations of this rare tumor.Perivascular epithelioid cell tumors (PEComas) are a family of rare mesenchymal neoplasms histologically and immunohistochemically characterized by perivascular epithelioid cell (PEC) differentiation.3 The patient did not have a medical history of gastrointestinal tumors, inflammatory bowel disease, or tuberous sclerosis complex. Her family history was unremarkable. Physical examination revealed a large mass at the right lower abdomen. All blood and biochemical tests were within the normal ranges, apart from a hemoglobin reading of 85 mg/dL and a C-reactive protein (CRP) reading of 15 mg/dL. An intravenous contrast-enhanced computed tomography (CT) scan showed an ill-defined multilocular soft tissue tumor measuring 13 cm × 8 cm × 7 cm in the pelvis and lower abdomen . In addition, granulocyte colony-stimulating factor  was given at a dose of 5 pg/kg/day subcutaneously from day 5 to day 12 of each cycle. Follow-up CT scans were performed every 6 months after chemotherapy. The patient was alive and well with no signs of recurrence or metastasis for 28 months of follow-up.A 29-year-old Chinese woman was admitted to our hospital because of gradual onset of abdominal pain, nausea, vomiting and weight loss for 6 months.9 cells/L with 87.3% neutrophils and a hemoglobin reading of 100 mg/dL. Serum levels of CA19-9, CEA, AFP, and CA125 were within normal limits. The abdominal CT scan and ultrasonography revealed ileocecal intussusception with a tumor in the ileum. An enteroscopy displayed a 2-cm diameter, polypoid, submucosal tumor in the terminal ileum  cancer or inflammatory bowel disease. Laboratory investigations showed a white blood cell (WBC) count of 11.6 × 10The tumor tissues were fixed in 10% phosphate buffered saline formalin solution and embedded in paraffin. Tissue sections were stained with hematoxylin and eosin for microscopic examination. Immunohistochemical stainings were performed with the primary antibodies listed in Table 2 Spirit TWIN electron microscope .The tumor samples were collected and fixed in 4% phosphate-buffered solution of glutaraldehyde and osmium tetroxide. Ultrathin sections were cut for ultrastructural evaluation using a Tecnai GA reverse transcription-polymerase chain reaction (RT-PCR) assay was performed to detect related gene fusions. Total RNA was isolated from the tumor tissues using TRIzol reagent  according to the manufacturer's instructions. The first strand of cDNA was obtained using RevertAid First Strand cDNA Synthesis Kit . PCR was performed with rTaq polymerase . The PCR primers and cycling conditions were performed as described in Table On gross examination, an ill-defined tumor measuring 13.5 × 7.5 × 7.0 cm was found at the terminal ileum about 12 cm from ileocecal valve Figure A. The neGrossly, a 2.2-cm diameter, polypoid, submucosal tumor was located in the terminal ileum protruding into the lumen. On cut section, the tumor was well-circumscribed but not encapsulated Figure . HistoloThe above pathologic findings in our cases confirmed a final diagnosis of GI PEComas-NOS.Melanosomes and premelanosomes were identified in the cytoplasm of both cases, using electron microscopy Figure .PSF-TFE3, NonO-TFE3, PRCC-TFE3, and EWS-ATF-1.4 A distinct band of 186 bp in length (lane 2) was amplified in case 1, representing the PSF-TFE3 fusion fragment  of GI PEComas-NOS have currently been reported in the English language medical literature ,37 rectum (n = 7),24 and stomach (n = 4).36 Of the patients, 43 were females and 27 were males, with a ratio of 2:1. The age at diagnosis ranged from 5.5 to 71 years. Until now, all reported cases of GI PEComas-NOS were sporadic, and only 1 patient was associated with tuberous sclerosis syndrome. Given the rarity of GI PEComas-NOS and their relatively short follow-up periods, our current knowledge of their biologic behavior, natural history, criteria for malignancy, and prognostic factors is limited.According to the World Health Organization Classification of tumors, PEComas are defined as a family of rare mesenchymal neoplasms histologically and immunohistochemically characterized by PEC differentiation.36 The proposed histologic features indicative of malignancy or high risk for aggressive clinical behavior in GI PEComas-NOS by Folpe et al38 include infiltrative growth pattern, tumor size (>5 cm), high nuclear grade, tumor necrosis, high mitotic activity (>1/50 HPF), and lymphovascular invasion. A recent case series study of 35 GI PEComas has shown that malignant behavior was statistically significantly associated with marked nuclear atypia, diffuse pleomorphism, and mitoses ≥2/10 HPF, but not with tumor necrosis.36 Optimal treatment strategies for GI PEComas-NOS have not yet been well established. Currently, surgical resection with a wide margin seems to be the mainstay of treatment. The benefit of adjuvant chemotherapy,21 radiation, and immunotherapy8 has not yet been established. In case 1, the tumor showed features of malignancy in the form of large size (13.5 × 7.5 × 7.0 cm), surrounding tissue invasion, necrosis, and high mitotic activity (3∼5/50 HPF). Given the high risk of tumor aggressive behavior, the patient was treated with multiple combined chemotherapies after surgery on the basis of nonrhabdomyosarcoma soft tissue sarcoma protocol (COG-ARST0332). She was alive and well with no signs of recurrence or metastasis for 28 months of follow-up. However, the benefit of adjuvant chemotherapy is still an area of controversy that requires more evidence-based studies.From recent clinical data, it appears that GI PEComas-NOS exhibit a spectrum of biologic behavior from benign to malignant. The majority of reported GI PEComas-NOS were considered to be benign or to have uncertain malignant potential, whereas only 22 cases (22/70) exhibited definite malignant behavior, with local recurrence in 4 cases, metastasis in 19 cases, and tumor-related death in 7 cases.TSC1 (9q34) or TSC2 (16p13.3) genes, whereas only one of the reported cases of GI PEComas-NOS showed an association with TSC until now. However, Cyclin D1 overexpression has been detected by immunohistochemistry in 5 cases of PEComas-NOS that were either malignant or had an uncertain malignant potential.39 The role of Cyclin D1 in the pathogenesis and progression of PEComas is becoming an area of interest. However, no Cyclin D1 immunoreactivity was evident in the 2 current cases. In recent years, it has been reported that a distinctive subset of PEComas harbors TFE3 gene fusion.40 Tanaka et al11 reported the first case of GI PEComa-NOS with a PSF-TFE3 gene fusion. In this study, we have confirmed the PSF-TFE3 gene fusion in another GI PEComa-NOS (case 1). In addition, MiTF and its downstream genes including TYR, CDK2, TBX2, and C-MET were detected in elevated transcript levels. TFE3 and MiTF belong to the MiTF/TFE transcription factor family, which is believed to be involved in pivotal developmental and cellular processes in various cell types. In different human tissues, the ratio of expression of the MiTF/TFE family members is found to be unique.41 Translocations of these genes are implicated in the MiT translocation subgroup of renal cell carcinomas.43 Recently, TFE3 has also been found to be an efficient regulator in melanocyte differentiation and pigment production under specific pathological conditions in vitro.44 Also, results from several recent clinical studies support the hypothesis that TFE3 can substitute for MiTF in a subset of MiTF-negative PEComas.38 As previously proposed by others,46 the subset of tumors harboring TFE3 gene fusions or exhibiting TFE3 immunoreactivity share distinctive clinicopathological features including relatively young age, nested/alveolar architecture, epithelioid cells with eosinophilic cytoplasm, and negative immunoreactivity for MiTF or muscular markers. Some studies also confirmed that overexpression of the TFE3 fusion protein is necessary for proliferation, migration, invasion potential, and long-term survival of UOK-145 cell lines.48 These findings suggest that TFE3 may play an important role in the tumorigenesis, and warrants further study.Knowledge about the molecular genetic alterations in PEComas is still limited. Some cases of AML, CCST, LAM, and CCMMT have been reported to be associated with tuberous sclerosis complex (TSC), a genetic disease caused by heterozygous mutations in the PSF-TFE3 gene fusion and to exhibit upregulation of MiTF and its downstream genes. Although the contribution of TFE3 to the pathogenesis and progression of PEComas-NOS remains poorly understood, the assessment of the TFE3 gene status may be necessary for an accurate diagnosis and prognosis of PEComas-NOS.In summary, we report 2 cases of PEComas-NOS arising in the GI tract, one of which was confirmed to harbor a


Odontogenic fibroma (OF) is a rare benign odontogenic neoplasm that is most commonly found in the mandibular/premolar region of female patients in the second to fourth decades of life. Well-defined radiolucent lesions that may induce root resorption are normally observed. Rare variants of OF have been described in the prior literature, including references to: 1) giant cell lesions, 2) amyloid-like protein deposition, and 3) ossifying variants. Immunohistochemistry can contribute to understanding the biological behavior and the pathogenesis of OF. Therefore, this case report aimed to describe a new case of ossifying OF and discuss the histopathology and immunohistochemical features. Key words:Odontogenic fibroma, jaw tumors, ossifying variant. Odontogenic fibroma (OF) is a rare benign odontogenic neoplasm. Clinically, OF is most common in the mandibular/premolar region of female patients in the second to fourth decades of life and is frequently found as radiolucent lesions that may induce root resorption .Histopathologically, OF is characterized by variable amounts of inactive-looking odontogenic epithelium embedded in a mature ﬁbrous connective tissue stroma . AlthougThe cytokeratin (CK) expression may well be a useful tool to conﬁrm the diagnosis of OF in epithelium-poor cases and to reveal the main types expressed in the OF when compared to other odontogenic tumors . Rare vaTherefore the present study is aimed to describe a rare case of ossifying odontogenic fibroma and discuss the histopathology and immunohistochemical features of this rare variant of OF.A 43-year-old woman with noncontributory medical, social, and cultural records was referred to the Oral and Maxillofacial Surgery Service, Department of Dentistry, Pontifícia Universidade Católica de Minas Gerais in Belo Horizonte, Brazil, to evaluate a nodule in her mouth. The patient reported that the lesion had been presenting a slow progressive growth for 3 years. Through clinical examination, a swelling of the left paranasal area with asymmetry could be observed extraorally. Intraorally, a swelling of the left maxilla associated with teeth 21, 22, and 25 could be identified. Teeth 23 and 24 were not presented. The lesion was painless, uneventful, well-defined, firm consistency, covered by normal color and texture of mucosa, and measured 32 x 30 mm. The cone-beam computed tomography (CBCT) of the maxilla showed a well-defined mixed hyperdense/hypodense lesion (Fig. OF is a very rare benign odontogenic tumor. According to the prior literature, OF represents 0 to 5.5% of all odontogenic tumors . AlthougTopographically, OF more commonly affects the premolar region of the mandible and the anterior region of the maxilla ,6. CliniConsidering only cases of ossifying OF in prior literature, three cases were more frequently reported in female patients in the fourth decade of life ,8.Conventional radiographs, fan-beam computed tomography or CBCT of OF predominantly shows unilocular or multilocular radiolucent images with well-defined margins ,10. IkesSpecific radiographic features for OF have yet to be clarified; however, severe effects on the surrounding bone trabeculae are often seen, even if the borders are well-defined in radiographic images. Diagnosis of OF should be complicated by several reasons, such as the diversity of radiographic and histopathological characteristics . Moreoveet al. (et al. (Histologically, OF is defined as an odontogenic tumor consisting of a moderately cellular fibrogenic proliferation with mono-morphic fibroblastic cells and a thin mature collagen admixed with epithelial cords and islands of odontogenic origin . Accordiet al. , the immet al. . The remet al. . Differeet al. . In the  (et al. .Considering the histological differential diagnosis, this lesion showed the typical fibroosseous microscopic features of an ossifying fibroma. This marked feature probably induces a misdiagnosis of a fibroosseous lesion after incisional biopsy. Therefore, immunohistochemical studies should be performed to clarify the diagnosis when the odontogenic epithelium has been associated with ossifications. The OF is considered a rare lesion, but information suggests that it can also be considered a distinct lesion. It is important to highlight that a review of the prior literature demonstrated only four cases of ossifying variants of OF, including this present case report. Therefore, it is important to note that new case reports regarding OF with ossifications are important in an attempt to better define this rare OF variant and describe its clinical, radiographic, and histological features, as well as its biological behavior.


Introduction. Both hepatocellular carcinoma (HCC) presenting during pregnancy and HCC presenting with obstructive jaundice due to a tumor cast in the biliary tract are very rare. The management of these patients remains challenging. Presentation of Case. A 23-year-old lady presented with obstructive jaundice at 38 weeks of gestation. Investigations showed HCC with a biliary tumor thrombus. She received percutaneous transhepatic biliary drainage (PTBD) and caesarean section. Right hepatectomy, extrahepatic bile duct resection, and left hepaticojejunostomy were carried out when the jaundice improved. The postoperative course was uneventful. She was discharged home on postoperative day 10. Histopathology showed HCC with a tumor thrombus in the bile duct. The surgical margins were clear. One year after surgery, the mother was disease-free and the baby was well. Conclusion. With proper management, curative treatment is possible in a pregnant patient who presented with obstructive jaundice due to a biliary tumor thrombus from HCC. Although the worldwide annual incidence of hepatocellular carcinoma (HCC) in women is 5.5/100,000–8/100,000 , HCC durHCC presenting as painless, progressive obstructive jaundice secondary to a biliary tumor thrombus is rare. It is important to diagnose the underlying cause of jaundice in these patients as this affects the management –15.We herein report a pregnant lady who presented with obstructive jaundice due to a biliary tumor thrombus from HCC.γ-glutamyl transpeptidase (GGT), 249 IU/L; and preoperative indocyanine green retention rate at 15 min (ICG-R15), 16.8%. She underwent laparotomy, right hepatectomy, extrahepatic bile duct resection, and left hepaticojejunostomy. Liver parenchymal transection was performed with an ultrasonic dissector and coagulative diathermy under vascular inflow control. At operation, there was a HCC in the right posterior sector of the liver, with multiple adjacent small satellite tumors. A tumor thrombus was found in the posterior and anterior sectorial ducts, the common hepatic duct, and the common bile duct (+), antibody hepatitis B e-antigen (Anti-Hbe)(+), hepatitis B core antibody (Anti-Hbc)(+), total, direct, and indirect bilirubin levels 183.2 umol/L, 112.1 umol/L, and 71.1 umol/L, respectively, and alpha-fetoprotein (AFP) level 23726 ng/mL. Both ultrasound (USG) and magnetic resonance imaging (MRI) showed a tumor at the right posterior sector of the liver, 114 mm × 108 mm, with adjacent satellite tumors. A tumor thrombus was seen in the right posterior sectorial duct, extending through the right hepatic duct into the common bile duct. The liver was cirrhotic. The diagnosis was HCC with a biliary tumor thrombus. Caesarean section was carried out and a normal male infant with body weight of 3.1 kg was delivered. The patient's jaundice increased and she received percutaneous transhepatic biliary drainage (PTBD) 2 weeks after delivery. The jaundice then improved gradually. At 38 days postpartum, computed tomography (CT) was repeated (Figures HCC presenting during pregnancy is extremely rare. Thus, the experience in its management and in its outcome is not clear. Pregnancy may have an adverse effect on the prognosis of hormonal dependent tumors, although this is still a subject of controversy for HCC. One explanation for the poorer prognosis for HCC points to the elevated levels of sex hormones . In our The most common presentation of HCC is right upper quadrant discomfort or pain. Jaundice occurs in 5–44% of patients –15. JaunWith an adequate preoperative assessment and management strategy, curative treatment is possible in a patient with extensive HCC during pregnancy.


Hemangiomas are benign vascular tumors. They are the prerogative of the liver and skin. And genitourinary localizations are rare and have only been rarely reported in the prostat, bladder, ureter or the perineum. In the light of published cases, urethral hemangiomas are mostly found in males. Few cases of hemangioma in the female urethra were reported. We report a cavernous hemangioma of the urethra in a 61 years old patient who presented bleeding from the urethra and micturition disorders. Physical examination revealed a tumor 3 cm x 2 polyploid arising from the terminal urethra . We performed surgical resection of the tumor, along with bladder drainage. The postoperative course was simple. We update through a review of the literature aspects of the diagnostic and therapeutic care of the urethral hemangioma. Hemangiomas are benign vascular tumors. They are the prerogative of the liver and skin. Genitourinary localizations are rare and have only been rarely reported in the prostat, bladder, ureter or the perineum . We repoA 61 years old woman with no particular medical history consulted for urethral mass causing dysuria and intermittent urethral bleeding since 2 months. Physical examination revealed a vascularized tumor which depended on the lower commissure of the urethral meatus of approximately 3 x 2 cm . The bioHemangiomas are benign vascular tumors that can occur at any age but it seems to be a male predominance . They arSimple transurethral excision of small urethral hemangiomas may be an effective treatment especially when laser facilities are not available. The cornerstone of the right management of urethral hemangiomas is still careful preoperative definition of the extension of the lesion.


Dear Editor:Endometriosis is defined as the presence of endometrial-like tissue outside the uterine cavity. Of all the cases of endometriosis, intestinal endometriosis accounting for about 10 %, the rectum and sigmoid colon are the most vulnerable position of the intestinal tract.For doctors, the diagnosis of colorectal endometriosis is difficult, especially the differential diagnosis between this disease and other diseases such as malignancies, irritable bowel syndrome, inflammatory bowel disease, ileal Crohn’s disease, and so on, due to its unspecific symptoms. Today, a large number of these cases are found accidentally at surgery and confirmed by pathology. Here, we describe a case which has not a definite preoperative diagnosis, but the histopathological examination of the resected specimen showed ileocecal endometriosis infiltrating the external muscular layer and two of the nine lymph nodes.A 47-year-old woman was referred to our hospital because of dyschezia, abdominal pain, abdominal distension, and mild nausea, associated with constipation. During 5 years before admission, the patient had episodes of abdominal pain especially at the lower abdominal and around the navel without obvious inducement. Abdominal pain can be relieved after defecation. Passing feces is tough for her and it occurs once each 2 to 3 days. The feces, shape is thinner than normal, with mucus on the surface, sometimes with few blood.’s hometown hospital 4 months before admission, at about 11–16 cm from the anal margin, revealed a stenosis with intact and a little thickened mucosa. Histopathological examination of the sigmoid from the colonoscopy showed chronic inflammation of mucosa.A colonoscopy performed in the patientTwo months earlier, the symptoms of dyschezia, abdominal pain, and abdominal distension became worse and much more frequent. However, the patient experienced no obvious improvement after antispasmodic and analgesic drugs were administered in her hometown hospital. The patient got married at the age of 26 years and had one normal labor at the age of 29 years, regular menses and no history of dyspareunia. Her last menstrual period was 1 week before and it was unremarkable. She had no past history of hypertension, diabetes, coronary heart disease, tuberculosis, or other infectious diseases. Besides, she had no operation history, radiation exposure history, or a poisonous chemical contact history. She also denied smoking and has occasional alcohol use. Her father and mother were healthy without reporting previous history of such disease. Her brother and her son were also healthy.The abdomen looked flat with no intestinal type or peristalsis wave. Palpation revealed mild lower abdominal tenderness and no abdominal masses or enlarged lymph nodes. The bowel sounds were slightly increased. Rectal examination showed no blood or distinct mass. On the gynecological examination, her vulva, vagina, and cervix appeared to be normal. Her uterus had normal size and was anteverted.Routine stool test was unnormal with occult blood active. Routine blood test, coagulation parameters, contagious parameters, routine urine test, liver function tests, renal function tests, AFP, CEA, CA199, and CA724 were all within normal range, whereas CA125 was twice as much the normal level.An ultrasound colonoscopy underwent in our hospital after admission showed an impassable stenosis, covered with rough, proliferous, anabrotic, and a little hematose mucous membrane, 15 cm from the anus, and the scope could not pass through the lesion. The ultrasound scan revealed obviously thickened intestinal wall, low echo in wide range, and mucosa with uncleared level. The pathobiology again confirmed mucosal chronic inflammation with lymphoid hyperplasia.A contrast-enhanced abdominal computed tomography scan revealed a swelling of the sigmoid wall and stenosis, and the uterine density was showed being not uniform. Concerning the duration, symptoms of incomplete intestinal obstruction, colonoscopy, and pathology reports, we thought the possibility of inflammatory bowel disease was large and could not completely rule out the possibility of tumor. In the last 2 weeks, symptoms of dyschezia became more serious; defecation was more difficult for her—it occurred once each 3–5 days, and abdominal pain, abdominal distension, vomit, nausea were worse than before, accompanied by hemafecia.A laparoscopy performed 3 days later revealed obviously a sigmoid mass near the peritoneal fold, 4 × 3 cm in size; adhesion between the neck of the uterus and rectum; and adhesion between the left ovary, fallopian tube, and the surroundings. No other organs were invaded. We resected the sigmoid at 12 cm above the tumor and 4 cm below the mass accompanied by lymph nodes in groups 241, 242, and 251 by the standard of carcinoma of the colon. Histopathological examination showed ileocecal endometriosis affecting the external muscular layer and two of the nine lymph nodes. The postoperative course was uneventful; the patient recovered well and left the hospital 8 days later. She was on regular follow-up and suffered no recurrence of the same symptoms one and half years later.Endometriosis is characterized as pelvic or extrapelvic, and the gastrointestinal tract is the most common site of the extrapelvic implantation sites. Compared to ileum, appendix, transverse colon, and cecum endometriosis usually arise in the rectosigmoid in 80 % of these cases.The etiology of endometriosis is still elusive and among the theories explaining the pathogenesis of endometriosis, the most widely accepted theory is Sampson’s “retrograde menstruation” theory: endometrial tissue regresses through the fallopian tubes, then implants and grows on the serosal surface of extra-uterine organs.Gastrointestinal tract endometriosis usually takes the form of asymptomatic small serosal implants. Influenced by hormone during menstruation, these implants may proliferate and bleed cyclically, bringing about non-specific symptoms like vomiting, diarrhea, constipation, pelvic pain, pencil-like stool, cyclical hematochezia, frequent urination, pain during defecation, dyspareunia, infertility, and abdominal mass. Forty percent of the patients present symptoms in a cyclic manner, which are usually related with menses. In our patient, symptoms relapsed irregularly and were not related with menses. Endometriosis infiltrating the muscularis propria and after cyclic proliferation, sloughing, and bleeding, inflammation and fibrosis appear and grow into the lumen, leading to obstruction. Invasive bowel endometriosis can present as bowel obstruction in an acute, chronic, or intermittent manner. The true incidence of endometriosis causing bowel obstruction is unknown, although complete obstruction of the bowel lumen occurs in less than 1 % of cases.Bowel symptoms can present a confusing clinical picture and masquerade a wide spectrum of disease processes, including irritable bowel syndrome, infectious disease, ischemic colitis, inflammatory bowel disease, ileocolonic intussusception, appendicitis, and malignancy.Physical examination cannot often provide sufficient clues for the diagnosis; hence, a timely and accurate preoperative diagnosis is often delayed. Imaging studies, such as abdominal CT, contrast-enhanced abdominal CT, magnetic resonance imaging (MRI), endoscopic ultrasound, and transvaginal sonography, are widely applied for detecting alterations in the intestinal wall. Among these ones, MRI appears to be the most sensitive technique for colorectal endometriosis with a positive predictive value of approximately 77.5–92.6 %. CT is not the primary imaging modality for evaluation of bowel endometriosis. Transvaginal ultrasound has a reported sensitivity of 91 % and specificity of 98 %. Endoscopy usually provide no valuable results for a definitive pathologic diagnosis because of the intact mucosa due to the reason that endometriosis involves the deep layers of the bowel wall in general. But in our practical work, we still use endoscopy routinely for differential diagnosis, whether it is endometriosis or malignant lesions or other diseases.Additionally, most cases are found accidentally at surgery and the gold standard for the diagnosis is laparoscopy or laparotomy, by which we can evaluate both the genital and intestinal tracts more completely and accurately.Treatment options comprise hormonal and surgical options. But we should choose the appropriate method according to patient’s age, fertility plan, the stage of the disease, and complications of the disease. Hormonal therapy can be considered when the disease has no symptoms of obstruction and we can apply hormones like danazol, gonadotrophin-releasing hormone (GnRH) analogs, and high-dose progestins. But these medicines are not recommended for patients who desire to become pregnant.Surgery is the choice of treatment for intestinal endometriosis when there are symptoms as intestinal obstruction, bleeding, severe pain, and if cancer cannot be excluded. Nowadays, we use laparoscopy to resect the intestinal lesions completely to prevent the tumor growth more often than laparotomy unless there are complicated adhesions.


There are several causes of peri-implant edema, pain, and swelling around implants after orthopedic fixation device application for fracture repair. The most common and well-known reason is infection, however, granulomas associated with foreign body reactions are rarely seen. In this report we present a case of a granulomatous reaction mimicking a hydatid cyst and sarcoma. We emphasize the importance of differential diagnosis in triggering appropriate management of the patient. Our case was unusual; to the best of our knowledge no similar clinical or pathological findings have been reported in current literature.A 56-year-old Turkish man who had been treated for a right femoral fracture via a plate fixation 10 years prior underwent an operation to treat pain and swelling around the plate. A hydatid cyst-like mass was observed during surgery, but subsequent examination revealed that no hydatid cyst was present; both malignancy and infection were also absent.Although infection is generally the first possibility that should be considered in a patient complaining of pain and swelling in the vicinity of an implant, malignancies, hydatid cysts, and  foreign body reactions should also be kept in mind as differential diagnoses. A soft-tissue reaction around a previously implanted plate should be managed carefully. Pre-operative radiological assessment, and biopsy to allow pathological and microbiological examination, should be considered in all suspected cases. Staphylococcus spp. are the most common infectious organisms associated with orthopedic implant surgery. Furthermore, hydatid cysts, especially in patients with predisposing factors, may cause implant infections[Echinococcus granulosus is usually responsible for the more commonly encountered hydatid diseases and/or infections[There can be several causes of peri-implant edema, pain, and swelling around the implant after orthopedic fixation device application for fracture repair. The most common and well-known reason is infection however, granulomas associated with foreign body reactions are rare. Swelling and pain may develop around the implant in patients with sarcomas. fections. Echinocfections. Althougfections.Sarcoma development due to implants can take several forms, but the most common are malignant fibrous histiocytoma and osteosarcoma. In addition, there has been one report of a peripheral nerve-sheath tumor and two reports of lymphoma resulting from implants. These tStaphylococcus (Staphylococcus epidermidis), which was determined to be due to contamination. Tests for a potential infection were taken. His test results showed a sedimentation rate of 107mm/h, CRP of 1.38mg/dl, a white blood cell count of 7.8×103/mm3, and eosinophils of 2.4. He was diagnosed with bursitis of the trochanter major, and removal of the bursitis and an implant operation were planned. The excised mass started from the lateral aspect of the distal femur (next to the implant) and spread postero-superiorly to the level of the greater trochanter, which was 15×12×6cm in diameter. The mass was filled with a 1×1cm cystic structure that appeared to be comprised of hydatid cyst vesicles  level was 3.7mg/dl and his eosinophil concentration was 12.4%. A biopsy specimen was taken from the area of swelling because of suspected infection. The biopsy specimen was considered to involve greater trochanter bursitis. However, because of increasing swelling and pain during the three months following the biopsy, he was re-evaluated in our clinic. A 25×12cm mass was present on the proximal anterolateral aspect of the right thigh, starting at the greater trochanter and spreading both distally and posteriorly. There was no warmth or redness associated with the mass. A diagnosis of bursitis of the greater trochanter was made due to the swelling, which was particularly severe on the greater trochanter and the screw heads on the proximal greater trochanter. To identify the causative organism, the mass was punctured again. The culture grew methicillin-sensitive, coagulase-negative Staphylococcus spp. are the most common causative organisms of infections that develop after orthopedic implant surgery[et al.[Staphylococcus spp. were detected in 43 out of 50 (86%) orthopedic implants. Moreover, Staphylococcus aureus was the only detected microorganism in 34 patients (68%). In our case, the pre-operative radiographs were normal, there were no signs of a loosening of the implant, and the bone plate attachment was quite firm. The intra-operative cultures were sterile. The large number of snowflake-like cysts, 1×1cm in size and demonstrating a cystic structure, were thought to be hydatid cysts as our patient was known to have a dog.Although implants are biologically inactive materials, they may cause soft-tissue reactions. The mos surgery. In a st[et al., StaphylThe hydatid cyst, which contained an outer fibrous macroscopic laminar layer and an inner germinal layer, had a so-called two-layered cyst wall containing a clear liquid. The germinal membrane contained small cysts and protoscoleces. Hydatid-related skin, brain, pancreaet al.[The most serious pathology that might trigger peri-implant edema and swelling after the use of orthopedic implants and prostheses is malignancy. Various animal studies have examined the biological effects of implants. Beryllium, cadmium, chromium, cobalt, iron, nickel, selenium, zinc, and titanium carcinogenicity studies have been conducted to demonstrate these effects-14. Howeet al. studied et al.. A stainAlthough infection is generally the first possibility that should be considered in patients with pain and swelling in the vicinity of a prior implant, malignancies, hydatid cysts, and  foreign body reactions should be kept in mind as differential diagnoses. No detailed investigation, such as magnetic resonance imaging, had previously been performed in our patient. Punctures were performed twice before the existence of greater trochanter bursitis was assumed, but the specimens were not sent for histopathological examination. This constitutes improper management of our patient. Insufficient pre-operative workup in such a case can result in an erroneous diagnosis, such as a hydatid cyst or a sarcoma. Therefore, we had planned to perform a wide resection following a possible diagnosis of malignancy or a hydatid cyst.In conclusion, the current literature does not report on any case exhibiting similar clinical and pathological findings to the best of our knowledge. A soft-tissue reaction around a previously implanted plate should be managed carefully. Our case is important because, despite the similarity between a macroscopic hydatid cyst and a sarcoma, they are only microscopically distinguishable from a soft-tissue reaction.Written informed consent was obtained from the patient for publication of this manuscript and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.CRP: C-reactive protein; CT: Computerized tomography.The authors declare that they have no competing interests.AS, KB, SS and HHC contributed to the conception and design of the study, carried out the literature research, manuscript preparation and manuscript review. SO performed the histological examination of the mass, and was a contributor in writing the manuscript. MSS was involved with the case and writing of the manuscript, general management of the patient and revised the manuscript for important intellectual content. All authors read and approved the final manuscript.


Epididymal cyst is a benign mass in the scrotum that is relatively common in adults but it is rare in children. In routine experience the treatment of such cysts is conservative. Torsion of these cysts is extremely rare and the diagnosis is made by exploration of the scrotum. Our patient was a 14-year-old boy who has been referred to hospital with scrotal pain followed by a minor trauma 3 days ago. Exploration of the scrotum to rule out testicular rupture was performed and a large black cyst connected to the head of the epididymis with 720-degree rotation was found. The cyst was resected and pathologic examination revealed an acquired epididymal cyst (spermatocele). The patient has normal physical exam after 3 months' follow-up. Epididymal cyst is a benign mass that is relatively common in adults but it is rare in children and its prevalence in children is 5–20% . When itThe patient was a 14-year-old boy who has been referred to hospital with erythema, swelling, and pain in left hemiscrotum. The patient had a history of minor trauma to scrotum 3 days ago. He had no fever or urinary symptoms. Physical examination demonstrated tenderness and swelling of the left hemiscrotum. The margins of left testis were vague but the right testis was normal. Urine analysis was unremarkable. Color Doppler Ultrasonography showed a heterogenic area in the upper pole of the left testis. Based on history and physical examination, patient was transferred to operating room with the diagnosis of testicular trauma. After exploration of tunica vaginalis, a large black cystic mass (4 × 4 × 3 cm) connected to the head of epididymis which was twisted for 720 degrees was seen. This cyst has a pedicle on the head of epididymis . As obviAcute scrotum is a urologic emergency. The etiologies are including testicular torsion, epididymorchitis, torsion of testis appendix, trauma, and hernia. In cases in which ultrasonography could not rule out the diagnosis of testicular torsion or rupture, exploration of scrotum is indicated. Treatment of epididymal cysts is conservative unless in case of huge size of the cyst or patient preference .Acute scrotum in these cysts  is extremely rare –4. As thAs cases of testicular torsion in association with epididymal cyst have been reported , it is iEpididymal cysts may twist as the testis torsion and may cause symptoms exactly similar to testicular torsion. Torsion of epididymal cysts should be considered in differential diagnosis of acute scrotum.