Borderline Brenner tumor of the ovary is a rare entity characterized by papillary structures with a fibro-vascular core, covered by a transitional epithelium, and by the absence of stromal infiltration. It is associated, by definition, with a benign component of Brenner tumor.We report a case of a 68-year-old woman, with a right ovarian mass, whose morphology and immuno-profile were consistent with the diagnosis of a borderline Brenner tumor. Immunohistochemistry carried out on selected markers may help to formulate the diagnosis, more than the molecular analyses.The online version of this article (doi:10.1186/s13048-014-0101-7) contains supplementary material, which is available to authorized users. Transitional cell tumors of the ovary, described for the first time by Brenner in 1907, are rare neoplasms and account for about 2% of all ovarian tumors.Transitional cell tumors, in turn, include two distinct clinic-pathological subtypes: Transitional cell carcinomas (TCCs) and Brenner Tumors .The World Health Organization (WHO) classifies Brenner tumors into three categories: benign, borderline and malignant . BorderlBorderline Brenner tumors are negative for p16, Rb and p53, and show weak immunostaining for Cyclin D1, moderate for Ras and strong for EGFR. They also express p63, as in benign subtypes but not in malignant ones, and present a diffuse staining for CK7, CA125, thrombomodulin and EMA, as in all subtypes of Brenner tumors .More recently, molecular analyses have revealed a number of molecular alterations that allowed to understand the molecular pathways involved in the pathogenesis and progression of Brenner Tumors. In particular, the loss of p16 expression in borderline and malignant Brenner tumors seems to be associated with promoter hyper methylation and a homozygous deletion . MoreoveWe report the case of a 68-year-old woman with a right ovarian mass, for which the morphology and immunohistochemistry (IHC) analyses were consistent with the diagnosis of borderline Brenner tumor. Molecular analyses were also performed to evaluate the methylation status of p16 and mutational status of KRAS and PIK3CA. Furthermore, since any activating mutation associated with EGFR over-expression has been described in the literature, we also evaluated the mutational status of the gene and verified EGFR copies number gain or amplification.A 68-year-old woman was admitted to gynecological surgery of our Institute for the presence of a suspect right ovarian mass. After a careful medical history, we discovered that the patient had been operated 12 years before for a neoplastic lesion in the left colon, with a diagnosis of “moderately differentiated adenocarcinoma infiltrating the muscularis B1 sec. Dukes, pT2 N0 G2”. No history of endometriosis was reported.The patient complained of abdominopelvic pain for two months. Vaginal examination revealed a solid mass in the right adnexal site attached to pelvis and uterus (diameter 15 cm Ø approximately) and CA125 levels were slightly increased (49.4 IU/ml). Contrast-enhanced CT scan of abdomen and pelvis revealed a “complex” pelvic mass (with both cystic and solid components) in the anatomical location of the right adnexa (maximum diameter 17 cm), not dissociable from uterus and contiguous bowel, with slight peritoneal effusion  with deep uterine and visceral adhesions; a total hysterectomy with right salpingo-oophorectomy and infracolic omentectomy was performed Figure .Figure 2Microscopically, the lesion was characterized by a solid adenofibromatous component with solid nests of transitional epithelial cells with pale cytoplasm, indented nuclei and cystic formations lined by mucinous columnar epithelium and by an exuberant papillary transitional cellular component with mild nuclear atypia Figure .The definitive pathological examination showed a Brenner borderline ovarian tumor with atypia (FIGO stage IA) and no adjuvant treatment was recommended. The patient status was NED (no evidence of disease) at 12-month follow-up visit.IHC in transitional cells was strongly positive for CK7, CA125, thrombomodulin and EMA, and negative for CDX2, CK 20 and p53  and it showed the absence of molecular alteration. Mutational analyses of KRAS and PK3CA were performed by direct sequencing, after specific PCR amplification, as previously reported [Beside, FISH analysis  did not reveal gene amplification or gene copies number gain Figure .Figure 4However, morphology and immuno-profile of the sample were consistent with the diagnosis of a borderline Brenner tumor of the ovary.Brenner tumors of the ovary are rare cancers. Up to date, about 30 cases of Brenner borderline tumors have been described, all characterized by the absence of stromal infiltration and, by definition, associated with a benign component . These lOur case of borderline Brenner tumor is morphologically characterized by papillary projections and solid nests of transitional cells surrounded by dense fibrous tissue.Transitional cells showed only mild, focal nuclear atypia and low mitotic index; extensive sampling ruled out infiltration. Therefore, our diagnosis was “Brenner Borderline tumor with atypia” .We performed an immunohistochemical study to demonstrate the origin of the tumor from the ovary and then to exclude that it was a metastasization from the bowel.The immunohistochemical panel showed strong positivity for p63 and EGFR in the epithelial component of the tumor, while p16 was positive in the basal, parabasal and intermediate layer of the papillary epithelial component.We also found, in transitional cells, a strong cell positivity for CK7, CA125, thrombomodulin and EMA, confirming the ovarian origin, and a negativity for CDX2 and CK 20. These latter findings excluded, immunophenotypically, the presence of metastasis from the primary tumor of the colon, dating back 12 years earlier.In the light of recent findings on the role of some molecular markers in the pathogenesis and progression of this tumor , we haveOur case can be placed among the low-grade lesions, based on morphology and immunophenotype. As described for papillary serous ovarian tumors of low-grade, the presence of specific markers has been of fundamental support for the differential diagnosis with benign (low expression of EGFR and p16) and malignant (negative for p16) Brenner tumors, and with TCCs (negative for EGFR).However, the molecular alterations described in literature, currently do not seem to be so specific as to suggest the possibility of developing targeted biological therapies for these lesions. Therefore, it would be necessary to better characterize these lesions, from a molecular point of view. Although they do not show a tendency to evolve into more aggressive malignancies, very little is known about cell biology that determines its pathogenesis.Written informed consent was obtained from the patient for publication of this Case Report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Iris metastasis in patients with gastric cancer is extremely rare. Herein, it is aimed to report on a patient with gastric adenocarcinoma and iris metastasis.A 65-year-old patient with the history of gastric cancer was admitted for eye pain and eye redness on his left eye. There was ciliary injection, severe +4 cells with hypopyon in the anterior chamber and a solitary, friable, yellow-white, fleshy-creamy vascularized 2 mm × 4 mm mass on the upper nasal part of the iris within the left eye. The presented patient’s mass lesion in the iris fulfilled the criteria of the metastatic iris lesion’s appearance. The ocular metastasis occurred during chemotherapy.Iris metastasis can masquerade as iridocyclitis with pseudohypopyon or glaucoma. In patients with a history of gastric cancer that present with an iris mass, uveitis, and high intraocular pressure, ocular metastasis of gastric cancer should be a consideration. Metastasis from systemic carcinoma to the eye was reported to occur in 4 %  and 8 % Gastric adenocarcinoma is among the most common causes of cancer-related mortality worldwide . GastricA 65-year-old male presented in April 2015 with a chief complaint of redness and pain in the left eye within the last 2 weeks. Past medical history included highly dysplastic undifferentiated gastric adenocarcinoma that was diagnosed by gastric biopsy via esophagogastroduodenoscopy in August 2014. The patient had undergone 18F-fluorodeoxyglucose-positron emission tomography/computed tomography (18F-FDG-PET/CT), which showed multiple uptakes in the distal stomach at the antrum and pylorus, lesser curvature, peripancreatic lymph nodes, and at multiple sites in the liver, but no uptake by the globe or periocular region. An additional biopsy specimen was obtained from a mass in the liver in September 2014, which showed metastasis of gastric adenocarcinoma. To differentiate between primary and a metastatic liver mass lesion, immunohistochemistry was performed. The liver lesion from gastrointestinal system was considered metastatic with the strong cytokeratin positivity, strong CDX2 focal positivity, focal cytokeratin 20 positivity, and strong cytokeratin 7 positivity. Both the primary lesion from the gastric biopsy and the metastatic liver lesion shared the same high-grade dysplastic gland formation under microscopy. The patient’s final diagnosis was stage 4 gastric cancer with multiple metastases.2 in total 75 mg docetaxel with 500 cc 5 % dextrose in 1 h intravenous infusion on the first day, (2) 40 mg/m2 in total 75 mg cisplatin with 500 cc isotonic saline in 1 h intravenous infusion, (3) 400 mg/m2 in total 770 mg leucovorin in 500 cc 5 % dextrose in 1 h intravenous infusion, and (4) 400 mg/m2 in total 770 mg 5-FU intravenous bolus injection. On the first day following intravenous bolus injection, 5-FU was administered 2000 mg/m2 in total 3860 mg with infusion pump continuously during the first and second day. This treatment scheme was repeated 2 weeks apart. Follow-up 18F-FDG-PET/CT in January 2015 showed stable gastric lesions and no uptake within the globe itself. The patient’s visual complaints started in April 2015. Visual acuity was 20/20 in the right eye (oculus dexter (OD)) and 20/50 in the left eye (oculus sinister (OS)). Intraocular pressure was 16 mmHg OD and 32 mmHg OS. There was ciliary injection, minimal upper nasally located corneal edema, severe +4 cells with hypopyon in the anterior chamber and a solitary, friable, yellow-white, fleshy-creamy vascularized 2 mm × 4 mm mass on the upper nasal part of the iris within OS . The modified DCF protocol was applied as follows: (1) 40 mg/mTwo days after identifying the possible iris metastasis, orbital MRI was performed to define the posterior extension of the mass before the needle aspiration biopsy; the mass was shown to be limited to the iris; no other mass lesions were noted at the ciliary body, choroid, optic nerve, extraocular muscles, or intra-orbital fat. Fine-needle aspiration biopsy was scheduled 1 day after the report of orbital MRI was obtained, but the patient passed away after a sudden respiratory arrest, nearly 10 months after the primary tumor was diagnosed.Between 90 and 95 % cases of ocular metastasis from systemic cancer occur in the choroid . A surveGastric adenocarcinoma metastasizes to numerous sites; the most common sites are the liver and non-regional or distant lymph nodes, whereas choroidal and iris metastases are very rare . To the A recent study about iris metastasis of systemic cancer among 104 patients reported that only three patients had iris metastasis of esophageal cancer, and only two patients had iris metastasis of colon cancer; no patient had iris metastasis of gastric cancer . One casIris metastasis can manifest with a stromal nodule, ill-defined iris thickening, pain, iridocyclitis, or hyphema. In 1995, Shields et al.  publishePseudohypopyon as a clinical feature of metastatic tumors in the iris was observed in only nine cases in a study ; this raThe treatment method for iris metastasis depends on a number of factors. If a patient has advanced, widespread systemic metastasis and the affected eye is relatively asymptomatic, no immediate treatment for the iris metastasis is necessary. Many patients with iris metastasis benefit from concurrent chemotherapy, in terms of local tumor control ; howeverIris metastasis of a primary tumor originating from the gastric region is rare. The presented case shows that gastric adenocarcinoma can cause isolated metastasis in the iris. The presented case was diagnosed based on clinical history and ocular examination. Iris metastasis can masquerade as iridocyclitis with pseudohypopyon or glaucoma. In patients with a history of gastric cancer that presents with an iris mass, uveitis, and high IOP, ocular metastasis of gastric cancer should be considered.Written informed consent was obtained from the parents of the deceased 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.


Tumors which originate from the Bartholin’s glands (BGs) are very rare. Many solid masses that arise from the BGs are carcinoma, though these benign solid lesions are rare.To the best of the reporter’s knowledge, about 35 cases have been reported in the English literature so far, most of which accompanied with signs or symptoms of Bartholin’s duct cyst. In this paper we report a 43-year-old woman with bilateral solid masses in the BGs, incidentally detected during anterior-posterior colpoperineoplasty. The masses were then excised.The histopathological examination showed increased number of acini with preserved duct-acinar connection, consistent with nodular hyperplasia. The patient had used oral contraceptive pill for four years. The Bartholin’s glands (BGs) are two small mucinous glands in the posterior aspect of the major labia . These g8P6A1D1L6 43 year old lady was admitted for an elective operation (anterior-posterior colpoperineoplasty). Her main chief complaint was vaginal sound, without any other symptoms like pain, dyspareunia, incontinency or history of episodes of BG's abscess. She had normal vaginal delivery for all her children and she had one early abortion (G6). She had received oral contraceptive pills (low dose) for contraception about 4 years. Bilateral swellings were discovered incidentally during operation which were firm at palpation. They were removed quite surgically with clinical impression of vulvar lump. Grossly, two ill-defined, lobulated surface, solid masses were identified, with diameters 3 × 2.5 × 1.5 and 3.2 × 2.5 × 1 cm. No capsule was identified. Microscopic examination showed increased number of acini with preserved duct-acinar relationship (A Gtionship . The acitionship . No necrBartholin’s glands are mostly affected by duct cyst inflammation. Tumors originated from BGs are rare and carcinoma is the most common one. Benign solid lesions like NH and AD are rare , 4, 6, 7The reported average age was 36 years old; the youngest and the oldest patients with NH were 19 and 56 years old, respectively , 4-8. InThere is neither a report of malignant transformation of NH, nor an evidence to indicate that NH can result in increasing the risk of malignancy , 6. Howe


Appendagitis is an uncommon clinical entity, often not recognised, and mistaken for more serious infective conditions. We describe a proven case of appendagitis which occurred after confirmed appendicitis. We postulate that this condition can coexist with appendicitis and indeed may be the result of coinflammation. This has several implications. Firstly, clinicians must retain an index of suspicion for this condition in a patient with localised abdominal pain which occurs after appendicitis. Secondly, it would be reasonable to suggest careful examination of colocated appendages in a patient with an otherwise normal-appearing appendix. Treatment might require laparoscopic resection, as performed in this case. A 21-year-old female presented to our emergency department with a history of right iliac fossa pain, loss of appetite, and nausea.She had originally presented to a different hospital three weeks previously with the same symptoms and had been admitted. A laparoscopy had been performed, and her appendix vermiformis was removed. Subsequent histology revealed the appendix had been chronically inflamed.She informed us that neither the operation nor the course of antibiotics prescribed postoperatively had made any difference. The rest of her history was unremarkable.On examination, she had tenderness in the right iliac region, with guarding and percussion peritonism. Her temperature, blood tests, and urinalysis were normal.She was admitted and initially managed conservatively. Ultrasound scans of her abdomen and pelvic organs were organised. These were normal, as were repeat blood tests after 24 hours. Gynaecological review was unremarkable. Despite analgesia, she continued to complain of pain and remained tender on examination. A CT scan of her abdomen and pelvis was organised as she was not improved and revealed inflammatory changes around the caecum, with enlarged mesenteric lymph nodes . There wEpiploic appendages are pedunculated fatty structures on the external surface of the colon, arranged longitudinally in line with the taenia coli. They can extend from the caecum to the sigmoid colon and are covered by peritoneum and number around 50–100. They tend to spare the rectum .Inflammation of these epiploicae, sometimes called “appendagitis,” is uncommon and can occur due to torsion of the epiploica or its vascular occlusion by thrombosis (primary appendagitis) or as a secondary phenomenon caused by the presence of inflammation nearby, for example, from sigmoid diverticulitis, cholecystitis, or pancreatitis , 5.Most cases are probably due to primary causes, such as torsion, or vascular occlusion, and usually occur in the sigmoid colon , 5; SingWith the advent of CT scanning in cases of acute abdominal pain, the diagnosis of appendagitis has been increasing. A recent CT review study estimated the frequency of epiploic appendagitis to be around 1.3%, with incidence of 8.8/million/year . The conOur case is unique as appendagitis occurred after chronic appendicitis. This presentation probably occurred due to thrombosis of the appendage vessels caused by the chronically inflamed appendix. We postulate that although the appendix was removed, the appendicitis had a coproximal inflammatory effect which then manifested as appendagitis. Symptoms only resolved once the inflamed and necrotic appendage was removed at the patient's second laparoscopy.It is possible that the patient may have had evolving appendagitis at the initial laparoscopy, but this may have been missed as attention would most likely have instead been paid to the appendix as being the source of the patient's problem. This emphasises the point that clinicians should have an index of suspicion for the appendage being abnormal.As far as we are aware, there have been no other cases of appendagitis after appendicectomy reported in the literature.A previous case report has described acute appendicitis occurring in a patient one week after laparoscopic treatment of acute appendagitis . The appThis case report reaffirms the need to consider the diagnosis of appendagitis in patients who present to emergency physicians with a history of right iliac region pain and a previous appendicectomy, especially in the absence of significant blood test abnormalities. Clinicians also need to have a low index of suspicion for inflammation elsewhere in the colon if appendagitis is found, particularly at laparoscopy, and the appendix vermiformis should be removed if caecal appendagitis is present, as highlighted by Chouillard et al.


Two cases of Burkitt’s lymphoma masquerading as appendicitis are reported herein. The diagnoses were made post-operatively from the appendix specimen in one case and from an ileocecal resection specimen for cecal fistula complicating an appendicectomy in the second case. These cases highlight the importance of routine histological examination of appendicectomy specimens. Burkitt’s lymphoma is an unusual variant (5%) of malignant (B-cell) non-Hodgkin’s lymphoma, which was first described between 1958 and 1962 in children in sub-Saharan Africa .P. falciparum malaria and Epstein-Barr virus (EBV) infection very early in childhood cause B cell hyperplasia which is an essential component of lymphomagenesis [The epidemiological and laboratory findings suggest that that the combination of recurrent agenesis ,5. A spoagenesis ,6. If suagenesis ,8. HistoA 13-year-old female was admitted electively for investigation of a long history (3 years) of right iliac fossa pain. This was a recurring dull and aching pain that had not responded to analgesia and antibiotics for suspected relapsing or ‘chronic’ appendicitis. She had no anorexia or weight loss. She had no constitutional symptoms, such as lethargy, fever, or night sweats, and had no altered bowel habits or urinary symptoms. On examination she appeared clinically well. The abdomen was soft but tender on deep palpation of the right iliac fossa. Thefull blood count and urinalysis were normal. Ultrasound examination suggested an appendicitis. At operation, a Lanz incision was made in the right iliac fossa. There was some free fluid in the peritoneal cavity but the appendix did not appear inflamed. It simply had a yellowish tinge. There was terminal ileal lymphadenopathy. The histology of the appendix specimen revealed a B-cell non-Hodgkin’s lymphoma consistent with Burkitt’s lymphoma  intestinal fistula in the right iliac fossa consistent with a cecal fistula. She was resuscitated with appropriate fluids and because of the persistent output from the high-pressure caecum a decision was made for surgical correction two days later. At laparotomy, there was a cecal fistula from the dehisced appendix stump. Apart for significant ileocecal lymphadenopathy the rest of the abdomen appeared normal. Following extensive lavage of the operation field with saline solution, an ileocecal resection with end-to-end anastomosis of the ileum and ascending colon was performed. The histology of the operative specimen confirmed a B-cell non-Hodgkin’s lymphoma consistent with Burkitt’s lymphoma. She had a stormy post-operative recovery and when fully recovered underwent a 6-month course of chemotherapy  having tested as HIV seronegative prior to its commencement. Nine years later she is well with no evidence of metastases but with fertility problems.chromosome 14) that activates an oncogene (c-MYC) [Burkitt’s lymphoma is a highly aggressive B-cell non-Hodgkin’s lymphoma and is the fastest growing human tumour ,10. This (c-MYC) . Clinica (c-MYC) . Patient (c-MYC) . Both ca (c-MYC) . Because (c-MYC) . A bone  (c-MYC) . The dia (c-MYC) . Due to  (c-MYC) ,20. Para (c-MYC) ,19,20. T (c-MYC) ,17. The The two patients in the case report were tested as being HIV seronegative prior to commencing the above standard chemotherapy regimen. The successful outcome in these patients may perhaps not have been achieved if they were HIV seropositive in terms of disease progression and response to chemotherapy ,21. AdjuAlthough Burkitt’s lymphoma presenting as appendicitis is rare, it should be considered an important differential diagnosis in children and adolescents from sub-Saharan Africa with atypical right iliac fossa pain. The reported cases demonstrate the importance of histologically examining appendicectomy specimens especially in patients with atypical presentations of appendicitis. The appendix, being a lymphoid organ, may be the most appropriate and accessible extranodal tissue for diagnosing intra-abdominal Burkitt’s lymphoma or other diseases. Understanding the underlying biology of Burkitt’s lymphoma would lead to strategies for prevention and approaches to reduce acute and chronic toxicities from current cytotoxic treatment.A 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 the journal.A written informed consent was obtained from the patient’s parent for the publication of this report and any accompanying images.CT: Computed tomography; EBV: Epstein-Barr virus; HIV: Human immunodeficiency virus; PET: Posittron emission tomography; MRI: Magnetic resonance imaging.The authors declare that they have no competing interest.EPW is the main author and drafted the manuscript. MNN gave advice on the tropical perspective and helped with the literature search. JSA helped with the literature search. All authors read and approved the final manuscript.


Cavernous Hemangioma of the Bladder (CHB) is benign and rare lesions. Clinical presentation has no pathognomonic signs although gross painless hematuria is the most frequent complain. CHB is suspected by cystoscopy and radiologic findings and confirmed by pathologic examinations. Management is controversial due to the bleeding risk of this highly vascularized lesion. Partial cystectomy is the treatment of choice for surgically accessible lesions. However, it appears that small lesions could be treated using transurethral resection. Since CHB is a rare case, we report another case treated successfully with a partial cystectomy associated with an augmentation cystoplasy. CHB is an uncommon benign tumor that accounts for 0.6% of bladder tumors . It mostWe report the case of a 60 years old patient without significant medical history, who presented a macroscopic haematuria. The etiology was a cavernous hemangioma of the bladder confirmed by histological examination of transurethral endoscopic resection chips. Two years later, irritative lower urinary tract symptoms led to the realization of an ultrasound that reveals an echogen mass in the dome of the bladder. The same hypervascular mass was indicated at the same side in abdominal pelvic MRI. It contains fluid central areas that can correspond either to necrosis or vascular ectasia. There is an extra-bladder invasion but no ganglions. The upper urinary tract was normal . BimanuaBladder hemangioma is a rare cause of hematuria. Although cavernous hemangioma is mostly developed in derma and epidermis, it is rarely reported in the mucosal parts. A review of the literature in 1942 revealed 40 cases of CHB had been reported up to that time . GeneralCavernous hemangioma of bladder is rare. Electrocoagulation or YAG Laser is effective the lesion is small. Partial cystectomy is the standard treatment of voluminous and isolated lesions of the movable portion of the bladder especially in cases of local invasion Although CHB have a benign course and favorable outcome, follow up is mandatory to detect recurrence or residual disease.


Trigeminal neuralgia is a disorder of the fifth cranial nerve that causes episodes of intense, stabbing, electric shock-like pain that lasts from few seconds to few minutes in the areas of the face where the branches of the nerve are distributed. More than one nerve branch can be affected by the disorder. We report an unusual case of trigeminal neuralgia affecting right side of face presenting atypical features of neuralgia and not responding to the usual course of treatment. The magnetic resonance imaging study of brain revealed a large extra-axial mass involving right cerebellopontine angle region causing moderate pressure effect on trigeminal nerve and brain stem. The aim of this case report is to show a tumor of cerebellopontine angle, presenting clinically as atypical trigeminal neuralgia. Trigeminal neuralgia (TN), also known as prosopalgia , suicideTrigeminal neuralgia has typical clinical features and various theories have been put forward as to its etiology. It is usually associated with pathosis along the course of the nerve. Most of the times the cause is not known and trigeminal neuralgia is termed as idiopathic TN.Anomaly of the superior cerebral artery causing demyelization and pressure of the aneurysms of the intrapetrous portion of the internal carotid artery causing irritation of the trigeminal ganglion leading to paroxysmal trigeminal neuralgia are recently blamed causes of idiopathic TN.Many lesions such as trigeminal neuromas in the middle cranial or the posterior fossa, epidermoid tumors, Meckle's cave, arteriovenous malformations, aneurysms, and vascular compression have been suggested as the causes. It is also recognized that TN occurs in about 2–4% of patients with multiple sclerosis .Classical trigeminal neuralgia  typicallPain is always unilateral and does not cross the midline. The pain is of short duration and lasts from a few seconds to several minutes. Refractory period is also present between the attacks. Most of these patients respond to treatment with carbamazepine and this can be used as a diagnostic indicator in TN.Many patients present with less classic symptoms and may mimic toothache, sinusitis, stomatitis, or other inflammatory conditions and therefore, for diagnosis of neuralgic pain, proper clinical examination along with thorough history is mandatory.The neuralgic symptoms in younger age group (less than 35 yrs) should alert the clinician to a possible intracranial space occupying lesion or intracranial arteriovenous anomalies.Lesions of cerebellopontine angle (CPA) are frequent and represent 6–10% of all intracranial tumors . AcoustiIn most cases, magnetic resonance imaging and computed tomography show typical features of acoustic neuromas or meningiomas and are sufficient to establish the diagnosis. Meningiomas are usually hemispheric, semilunar masses with a broad petrous base to which they are attached whereas acoustic neuromas are usually round or oval masses in the cerebellopontine cistern that emerge from the internal auditory canal and grow posteriorly because of the anterior limit represented by the cisternal segment of the facial nerve .In this paper, we discuss the importance of investigating each and every case of TN for proper diagnosis before we term it to be idiopathic TN.A 40-year-old male patient reported to Outpatient Department of Vidya Shikshan Prasarak Mandal Dental College & Research Center (VSPM DCRC), Nagpur, with the chief complaint of pain in the right ear and preauricular region for 2 yrs. Patient had visited various dentists but did not get any relief. Patient was diagnosed as a case of glossopharyngeal neuralgia three months prior to his visit and was treated with tablets carbamazepine 100 mg thrice daily which then increased to 200 mg thrice daily but without any satisfactory relief. Severity and intensity of pain increase on the same side of face in spite of taking increased dose of carbamazepine.When patient visited our department he had severe, intermittent, and lancinating pain in that region. Pain was paroxysmal and each episode lasted from few seconds to few minutes, with trigger points in the preauricular and postauricular region on right side of face. Patient also gave history of episodes of severe awakening pain during nights but no history of pain on deglutition or during movements of tongue. There was no history of seizures, nausea, vomiting, and vision changes. On examination, trigger zones were present on malar area, zygomatic buttress area, and also body and ramus of mandible but otherwise the patient's general condition was normal. The pains are bright and stimulating and are accurately located by the patient. The behavioral characteristics of the pain are neurogenous.All these findings were suggestive of trigeminal neuralgia but as the patient was not responding to the usual course of treatment and the severity of pain increases, an intracranial pathology was suspected in this case. Magnetic resonance imaging was advised which revealed an intracranial tumor.Magnetic resonance imaging (MRI) showed well-defined intensity enhancing extra-axial mass involving right cerebral pontine angle region measuring 2.5 × 2.0 × 1.8 cm in anteroposterior, transverse, and superoinferior direction, respectively, and causing moderate mass effect on trigeminal nerve and brain stem (Figures Further MRI with contrast revealed possibility of cerebellopontine angle Schwannoma or meningioma . The patIt is estimated that 15% of patients with trigeminal neuralgia have intracranial pathology that compresses the trigeminal nerve. This often occurs in the cerebellopontine angle, pons, and medulla from where the trigeminal nerve exits. Failure to properly assess for secondary TN is a major potential pitfall. So a careful examination of all the cranial nerves along with MRI of the brain is advisable in patients younger than 60 yrs., so as to identify the presence of structural lesions like tumor, cerebral aneurism, acoustic neuroma, and so forth.One should also lookout for symptoms related to compression of surrounding tissues, which can also affect other cranial nerves and blood vessels.The typical features of meningiomas are seizures, headaches, nausea, vomiting, vision, and behavioral and cognitive changes. Sometimes no symptoms occur and tumor is detected incidentally . In thisVarious treatment modalities are available for management of trigeminal neuralgia which has been tried for many years. Medicinal therapy is usually given for the treatment of TN. Surgical intervention can be attempted only if and when the pharmacologic therapy fails. Carbamazepine is the drug of choice for TN. 100 mg tablet thrice daily usually resolves pain of TN.Side effects of this drug may cause bone marrow depression, drowsiness, blurred vision, and vertigo. Therefore, monitoring the bone marrow activity by obtaining the complete blood count prior to initiating therapy and routinely thereafter is indicated.Anticonvulsant medications pose risk of sedation and ataxia particularly in elderly patients, which may make driving or operating machinery hazardous. So patients should avoid maneuvers that trigger pain. Many times the diagnosis is not established and the clinician goes for multiple dental extractions which are of no use even if pain radiates into the gums. Patients also need to understand that medications for TN are only palliative and often are of limited and temporary value. If the patient does not respond at least partially to carbamazepine, we should suspect for idiopathic TN.Tumor arising in the CPA or the tumor of the base of the skull or brainstem encroaching upon the CPA can produce TN like symptoms. The other associated symptoms of these tumors depend on the direction of growth of these tumors. In our case although the lesion arose from CPA, it did not have any feature of this tumor and rather it presented as atypical TN; therefore, diagnosis was difficult.As the tumor grows upward into the superior aspect of the cerebellopontine angle, it encroaches upon the trigeminal nerve, producing a gradual decrease of the corneal reflex and facial analgesia and anaesthesia. Downward growth of these lesions results in hoarseness, numbness of the throat, or complaints of difficulty in swallowing. As with acoustic tumors, large meningiomas can produce cerebellar symptoms and signs or hydrocephalus with increased intracranial pressure .Patient must be informed thoroughly of the risks involved with neurosurgical interventions, including anesthesia dolorosa that results from injury to the system in an attempt to treat trigeminal neuralgia. Numbness of the face is a constant part, which has also been referred to as trigeminal deafferentation pain. It results from damage to peripheral or central sensory pathways leading to partial or total loss of sensory nerve supply to a region of the body. Deafferentation pain is usually severe in intensity and highly resistant to treatment. It can spread to other orofacial structures by extension of the receptive field.Some symptoms such as hypoesthesia, paresthesia or anesthesia, or hyperalgesia may be felt in the region innervated by the injured nerve and these symptoms may persist indefinitely .The present case report focuses on the fact that the eyes cannot see what the mind does not know. It is not always possible to determine what causes trigeminal pain. However, several possibilities exist. The present case shows an unusual lesion of cerebellopontine angle tumor, rare cause of atypical TN, emphasizing the importance of investigating each and every case of trigeminal neuralgia for proper clinical diagnosis. As we cannot establish a relation between pathology in the brain or brainstem and a clinical picture in a patient, imaging technique preferably an MRI scan with a focus on the course of the trigeminal nerve can be of utmost importance for the diagnosis of such cases. Follow-up scans are needed because meningioma can recur years or even decades after treatment.


Simultaneous dilation of both the common bile duct and the pancreatic duct (double-duct sign) is usually because of ampullary or pancreatic tumours. Here we report an unusual cause of double-duct dilation; we describe the case of a 49-year-old female who developed afferent loop syndrome after pylorus-preserving pancreaticoduodenectomy: cross-sectional imaging of the abdomen revealed a double-duct sign. Whipple’s surgery is associated with a number of complications, such as pancreatic fistula, post-surgical haemorrhage, porto-mesenteric venous, pancreatitis, hepatic infarction, delayed gastric emptying, and anastomotic strictures . AfferenA 49-year-old female presented with upper abdominal pain, accompanied with abdominal distension. She had undergone pylorus-preserving pancreaticoduodenectomy for pancreatic neuroendocrine tumour three months previously. The patient had tachycardia and icterus, and abdominal examination revealed tenderness in the upper abdomen. Serum amylase, bilirubin and alkaline phosphatase levels were elevated. Contrast-enhanced computerized tomography (CECT) of the abdomen revealed a markedly dilated afferent loop of gastro-jejunal (GJ) anastomosis  with norALS is most commonly described following Billroth Type 2 surgery, which is carried out for complicated peptic ulcer disease . On the valvulae conniventes projecting into the lumen (keyboard sign)—has been shown to be the most common MDCT features of ALS [Early diagnosis is very important in preventing life-threatening complications such as afferent loop perforation . ALS cans of ALS . MRCP cas of ALS .Management of afferent loop syndrome includes endoscopic, percutaneous or surgical approaches. Conservative management has no role except on very rare occasions, as in chronic obstruction, where emptying of the afferent loop contents into the stomach may relieve obstruction, or in patients with disseminated malignancy. Surgical correction has been the treatment of choice but radiological or endoscopic treatment has also been successfully performed in selected patients , 9. EndoConflict of interest: none declared.


Dear Editor, A female, caucasian, 22-year-old patient with no comorbidity, complaining of incapacitatingcrises of subungual pain in her left hallux, strong enough to wake her up in the middle ofthe night, starting four years ago, with progressive worsening. The symptom was triggeredby a physical stimulus such as cold temperature, local pressure, and even wind causing thecrises which improved as she immersed her feet into tepid water. The patient had alreadysought medical assistance several times, receiving multiple, different treatments,including treatment for mycosis, tendinitis and neuritis. Amongst the prescribedpharmaceuticals, she reported the use of analgesic, nonsteroidal anti-inflammatory andcorticosteroid drugs, all of them with no response.Both plain radiography and ultrasonography of the first digit of her left foot did notcharacterize any pathological finding. Magnetic resonance imaging of her left halluxdemonstrated the presence of a subungual solid, well-delimited nodule with hyposignal andhomogeneous contrast uptake at T1-weighted-image . Such fi. Suchstructures are responsible for thermoregulation by means of skin blood flow control, beingconstituted by an afferent arteriole, an anastomotic vessel named Soucquet-Hoyer canalinvolved by smooth muscle fibers, an afferent vein, nervous fibers and a peripheralcapsule,2.Glomus bodies are arterio-venous shunts present in several parts of the body, with greaterconcentration in the reticular layer of the dermis, especially located in the digits, palmsof the hand and sole of the feet,3. Approximately 75%of glomus tumors occur in the hand, and 60% of them are subungual ,4. Glomus tumors were first described by William Wood in 1812, as apainful, subcutaneous, slow-growing tumor susceptible to temperature variations. Onlylater, in 1901, Grosser described the lesion as arteriovenous anastomoses, associating themwith the body temperature regulation.Glomus tumors are rare benign lesions characterized by hamartomatous proliferationoriginating from glomus bodies. Such tumors correspond to 2% of all primary soft parttumors and to 1%-5% of all soft part tumors in the hand, at a mean age of 39. Clinically, hyperalgesia is the cardinal symptom of this tumor, andin 90% of cases the triad paroxysmal pain, local hyperalgesia and hypersensitivity to coldtemperatures is present,7. The symptoms are triggered by unimportant trauma andvariations in temperature, improving with immersion into tepid water. Physical examination if usuallyineffective, but in some cases ungual alterations and bluish nodules may be found, the latter measuring about 3 to 10 mm indiameter,5,8.Glomus tumors affect young adult individuals, particularly between their fourth and fifthdecade of life, being seven times more frequent in women,6. Specific testshave been developed to AID in the diagnosis, as follows: a) Love’s test,5 –allows for identifying the exact site of the lesion, by applying localized pressure withthe end of a paper clip, with 100% sensitivity; b) Hildreth’s test,5 –reduction of the pain after inflation of a tourniquet proximally applied to the arm, with90.5% sensitivity; c) Transillumination – visualization of subungual blue nodules (on thenail bed), with 85.7% sensitivity.The diagnosis is based on the patient’s clinical history and physical examination in anattempt to trigger the pain followed by immersion into tepid water to determineimprovement. However, clinical criteria present 50%-90% sensitivity and it takes four toseven years after the symptoms onset to have a diagnosis,6, increased distance between the dorsalaspect of the distal phalanx and the underside of the nail in 25% of cases. Ultrasonography can demonstrate a welldefined, solid, hypoechogenic and hypervascularized nodule in 83% of cases. However, ultrasonography is limitedbecause of the subungual location of most glomus tumors. On the other hand, Magneticresonance imaging presents almost 1005 sensitivity, demonstrating a solid nodule withhyposignal on T1-weighted images and hypersignal on T2-weighted images, with homogeneouscontrast uptake. Surgery is thedefinite treatment,2,5, with a recurrencerate of 12% to 24%,6. As clinical criteria present low sensitivity, imaging methods play a supplementary role inthe diagnosis. Plain radiography fails to demonstrate significant findings, and may fail todepict erosion in 14% to 60% of cases


Petersen hernia is a rare internal hernia that occurs after Roux-en-Y (R-Y) reconstruction. To our knowledge, there are a few reports on internal hernia, especially Petersen hernia after open gastrectomy for gastric cancer. Two rare cases of Petersen hernia are presented in this report. A man in his 70s was referred to our hospital due to a complaint of postprandial sudden abdominal pain. He had a history of open total gastrectomy with R-Y jejunal reconstruction through the antecolic route for gastric corpus cancer. On computed tomography (CT), bowel obstruction and strangulation of the small intestine were suspected. Emergency laparotomy was done, and an internal herniation of the small intestine through Petersen space was observed. A man in his 50s was referred to our hospital due to a complaint of severe sudden abdominal pain. He had a history of open gastrectomy and abdominal/lower intrathoracic esophageal resection with R-Y jejunal reconstruction of an antecolic jejunal limb for esophagogastric junction carcinoma. On CT, internal herniation of the small intestine was suspected. During emergency laparotomy, an internal herniation of the bowel through the Petersen space was observed. Though history of R-Y reconstruction surgery may be helpful, preoperative diagnosis of Petersen hernia is difficult to establish. Here we present two rare cases of this type of internal hernia. In 1900, Dr. Walther Petersen, a German surgeon, first described Petersen space hernia which is an internal hernia caused by the Petersen defect, a space between the Roux limb and the transverse mesocolon formed after Roux-en Y (R-Y) reconstruction . He had a history of open total gastrectomy with R-Y reconstruction with an antecolic jejunal limb for gastric corpus cancer a year prior to his presentation at our hospital and coronary bypass operation. The body mass index (BMI) was 19.1 kg/mOther medical and family histories were unremarkable. Abdominal contrast enhanced computed tomography (CT) revealed a dilated small intestine with thickened bowel wall, high density of mesenteric fat presenting with edematous changes, and spiraling and converging mesenteric fat and vascular structure, showing “whirl sign”. Accordingly, bowel obstruction and strangulation of the small intestine were suspected Fig. . Differe2 at the time of surgery for Petersen hernia, and 23.2 kg/m2 at the first gastrectomy. Medical and family histories were unremarkable, except for right leg amputation. On abdominal contrast enhanced CT, well-circumscribed high density of mesenteric fat, and strangulation of superior mesenteric artery (SMA) and vein (SMV) were present with the whirl sign, indicating internal herniation . He had a history of open total gastrectomy and resection of abdominal/lower intrathoracic esophagus with R-Y jejunal reconstruction through antecolic route for gastroesophageal junction carcinoma a year and a half prior to the presentation. He had been followed up for outpatient visits and received TS-1. The BMI was 21.7 kg/mInternal hernia is a well-known complication of R-Y gastric bypass. In laparotomy, the incidence has been reported to be 1–5 % (Paroz et al. Body weight loss is considered to be another risk factor for internal hernia after gastrectomy (Kojima et al. Another factor to consider is the position of Roux limb. R-Y reconstruction has become more popular in recent years, with the intention to avoid anastomotic leakage and reflux inflammation of the remnant stomach. Recently, many reports prefer an antecolic figure for the Roux limb, which reduces the potential hernia sites from three to two, including Petersen defect after laparoscopic R-Y bypass reconstruction (Kojima et al. In a recent report, however, all patients with Petersen hernia had undergone antecolic R-Y reconstructions (Yoshikawa et al. Petersen hernia is a specific type of hernia where the small bowel moves into the potential space between the caudal surface of the transverse mesocolon and the edge of the Roux limb. Petersen hernia is rarer than other internal hernias that have mesocolon mesentery defects and jejunojejunostomy after R-Y reconstruction (Garza et al. As the symptoms of internal hernia, including Petersen hernia, are nonspecific, making its diagnosis is challenging. Most patients complain nonspecific symptoms such as postprandial abdominal pain, nausea, and emesis (Lockhart et al. Though not specific, several CT findings are suggestive of internal hernia; including the whirl sign, target sign, small bowl obstruction, clustered loop, retraction of the mesentery, congestion of mesenteric fat and vessels, mushroom sign, hurricane eye, small bowel behind SMA, and right-sided anastomosis (Lockhart et al. In conclusion, we reported two rare cases of Petersen hernia after laparotomy with R-Y jejunal bypass reconstruction in this study. Petersen hernia presenting as internal hernia after R-Y reconstruction is rare, and its preoperative diagnosis is difficult to establish. Neither symptoms nor CT finding alone cannot lead to definitive diagnosis of Petersen hernia. Therefore, we should pay attention to any history of R-Y reconstruction and consult radiologist as needed in order to make a correct diagnosis and avoid massive bowel resection.


Introduction. Tension pneumocephalus (TP) is a very rare complication related to radiotherapy for nasopharyngeal carcinoma (NPC). Case Presentation. A 46-year-old man was admitted to the hospital with an altered mental status and aqueous rhinorrhea for several hours of evolution. The computed tomography (CT) scan showed TP, a defect in the skull base and nasocranial fistula. The patient was receiving a second course of radiotherapy for local relapse. With medical treatment the patient recovered neurological status but died two days later. Discussion. In our knowledge, only 4 cases with similar characteristics have been reported in the literature. This is the first case report of TP during radiotherapy. TP was an abrupt and rapid process with neurological impairment for hours of evolution without suspicious osteoradionecrosis (OR) in previous scan images. The defect in the skull base could be due to a rapid disappearance of the tumor. The appearance of aqueous rhinorrhea and neurological symptoms must be viewed as signs of alarm. Nasopharyngeal carcinoma (NPC) is an uncommon malignant tumor. At diagnosis, the disease is often locally advanced. In these cases, the standard treatment is high doses of conformal radiotherapy with chemotherapy .Pneumocephalus is defined as the presence of gas within any of the intracranial compartments, when a valve mechanism allows air to enter but prevents it to escape a tension pneumocephalus (TP) appears. TP is very uncommon pathology and most cases occur during the postoperative posterior fossa surgery . TP relaWe report a case of a patient diagnosed with NPC who developed TP during reirradiation for tumor relapse. In our knowledge, only 4 cases with similar characteristics have been reported in the literature. This is the first case report of TP during radiotherapy.A 46-year-old man was admitted to the hospital with an altered mental status and aqueous rhinorrhea.Two years earlier, in February 2009, the patient was diagnosed of an undifferentiated NPC Stage IVA (cT4N2M0) according to AJCC's 7th edition manual, with involvement of cranial nerves and orbit. He was treated with induction cisplatin-5-fluorouracil scheme, for two cycles followed, from May 2009 to June 2009, by concurrent chemoradiotherapy with cisplatin.Gross tumor volume (GTV) was determined based on magnetic resonance imaging (MRI) and computed tomography (CT) tumor extent. Clinical target volumes (CTV) were GTV + 1 cm and nodal levels at-risk . Planning target volumes (PTV) were CTV plus a 0.5 cm margin. Radiation doses were 70 Gy to tumor and involved nodes and 50 Gy to prophylactic anatomic sites. Fractionation was 2 Gy/session. The treatment was planned using 6 MV and 15 MV photon energy. Usual head-and-neck critical structures, such as spinal cord, were contoured.3 with intracranial extension and involvement of skull base. Five cycles of cisplatin-5-fluorouracil were scheduled. At the end stable disease was evident on CT. No complaints about neurological or other type of symptoms were reported.Patient remained in complete remission, documented with endoscopy and MRI, up to May 2010 when a local recurrence was diagnosed. A CT scan showed a tumor of 3 × 2 × 1.5 cmIn March 2011, the patient complained of mild pain in left eye, moderate periorbital swelling, and ophthalmoplegia without other neurological symptoms. On MRI local progression appeared, with tumor invasion of the pterygoid and masseter muscle in the temporal fossa, sphenoid sinus, ethmoid cells, orbit apex, and meningeal involvement in ipsilateral temporal fossa. Osteoradionecrosis (OR) was not described by the radiologist.At that time, a reirradiation of the visible tumor plus 1 cm margin with an imaged-guided intensity-modulated radiotherapy (IMRT) technique, in order to decrease the toxic effects of reirradiation, was decided. CTV was GTV + 1 cm and a total dose of 66 Gy was planned. Reirradiation began in April 2011 without any neurological symptom. Treatment was stopped at 64 Gy because he exhibited abrupt deterioration.Patient was admitted to the hospital because of incoherent speech, impaired behavior, and aqueous rhinorrhea for several hours of evolution. In the emergency room, physical examination revealed disorientation as to time and place, cerebrospinal fluid rhinorrhea, left periorbital swelling, and fever (38.3°). Neurological examination confirmed a Glasgow coma scale of 10 and cranial nerves could not be properly assessed. Other findings were unremarkable. Culture of the ventricular fluid was not realized.Radiography showed the presence of cranial air in the lateral and III ventricles . The basPatient's poor clinical situation and the high local tumor invasion impeded aggressive treatments. He was treated with intravenous fluid, broad spectrum antibiotics, and anti-inflammatory drugs. With medical treatment the patient recovered neurological status, but the patient died two days later.Pneumocephalus refers to the presence of intracranial gas and is a very rare complication of radiotherapy in head and neck. It is usually caused by several etiologies, such as craniofacial surgery, trauma, tumors, or meningitis from gas forming organisms. TP associated with radiotherapy is an exceptionally rare and potentially fatal complication. The majority of cases of pneumocephalus are asymptomatic, but in TP the mass effect can produce related symptoms. Treatment depends on cause. The majority of postcraniotomy cases with no treatment is necessary with the air being reabsorbed. Surgery is necessary in the presence of TP in order to relieve pressure or in cases of cerebrospinal fluid leakage.Generally, only minor complications are observed as a result of a single course of radiotherapy for NPC: xerostomia, trimus, fibrosis, and so forth. However, severe complications can result in patients subjected to second salvage radiation, such as myelitis, paralysis of nerves, OR, temporal lobe injury, and severe trimus .Our review of the literature revealed 6 cases of pneumocephalus related to NPC and only 4 cases of TP. Ng et al. [Kiu et al.  presenteWu and Lee  reportedWang et al.  presenteIn the reported cases, TP appeared between 2 and 4 years after radiotherapy. All cases detailed the presence of OR as the cause of the skull base defect. As in previous reports, in this case TP appeared 2 years after the first radiotherapy course.Wang et al. presented a subacute case, with rhinorrhea months before TP. In the other cases, the presence of rhinorrhea was not documented. In our case, the patient who was in radiotherapy treatment did not complain about rhinorrhea, dizziness, headache, or other neurological symptoms. The clinical onset was abrupt with symptoms and neurological impairment for several hours of evolution.We believe that our patient suffered from an OR in the skull base. This event usually occurs 1 year after radiotherapy. It is thought to be secondary to osteoblastic destruction. The skull base, cervical spine, and the mandible are commonly affected. Imaging findings include areas of osteolysis and mixed sclerosis . If an OHowever, patient presented an abrupt and rapid TP so another possibility could be the creation of a defect in the skull base due to a rapid tumor disappearance, which previously invaded the skull base. In addition, signs of OR in MRI or CT were not described by the radiologist. The main drawback of our work is the lack of autopsy, precluding verify if tumor disappearance and/or OR were the responsible of the TP. The autopsy would confirm or refuse our hypothesis.Despite high-dose reirradiation being difficult due to the previous doses to the nearby critical structures, IMRT allows achieving better dose distributions than previous techniques, so doses higher than 50 Gy can be delivered for salvage irradiation. Doses higher than 60 Gy are associated with better outcomes –10.The principles of treatment of TP are to provide adequate supportive care and antimicrobial therapy and to modify the hot's inflammatory response . In addiRadiotherapy is a very useful arm in the treatment of NPC, in primary or salvage treatment. High-dose irradiation is necessary to attempt curative treatments, and serious complications have been described.TP is a very rare complication of radiotherapy related to NPC, but this possibility must be kept in mind when treating primary cancer patients with tumors invading the skull base, with neurological symptoms or when planning a salvage reirradiation. Patients complaining about neurological symptoms or persistent rhinorrhea must been viewed as signs of alarm. Rapid diagnosis of TP could be vital for the patient.


A 69-year-old female with unremarkable past history underwent endoscopy for dyspepsia. She denied weight loss or anorexia. Upper endoscopy revealed a bulge in the lesser curvature and posterior wall of the stomach with 4-5 cm. Endoscopic ultrasound was performed which showed a heterogeneous lesion, anechogenic in the major part, with a floating membrane inside, the greatest diameter of 90.8 × 17.2 mm, originated in the left liver lobe. Surgical resection was performed. Pathologic examination revealed a cystic lesion with an acellular thick fibrous wall, surrounded by a conspicuous inflammatory reaction. The cyst wall revealed a characteristic lamellar pattern of the fibers. In the internal surface of the lesion, there were remains of membranous structures, amidst which a vestigial Protoscolex was noted. In the presented case, a floating membrane was observed, which is a pathognomonic feature, establishing the diagnosis of hydatid cyst type 3. Fine needle aspiration guided by ultrasound was not performed due to the certainty in the diagnosis. To the authors' knowledge, these are the first images by endoscopic ultrasound of hydatid cyst of liver presented as a bulge in the stomach with pathognomonic features, which allowed the definitive diagnosis with no need for further diagnostic tests. A 69-year-old female with unremarkable past history underwent an upper endoscopy for dyspepsia. She denied weight loss, anorexia, asthenia, or vomits. Physical examination was normal. Upper endoscopy revealed a bulge in the lesser curvature and the posterior wall of the stomach with 4-5 cm . EndoscoPathologic examination  revealed Echinococcus type and represents a substantial disease burden [Hydatid disease is a worldwide parasitic infestation by tapeworms ofe burden . World He burden .In English literature, we found only one report in which the EUS contributed decisively to the diagnosis of a liver hydatid cyst, allowing for sample collection with fine needle aspiration in a lesion without imagiologic diagnosis .In the presented case, a floating membrane was observed, which is a pathognomonic feature, establishing the diagnosis of hydatid cyst type 3. Fine needle aspiration guided by ultrasound was not performed due to the certainty in the diagnosis.To the author's knowledge, these are the first images by EUS of a hydatid cyst of liver presented as a bulge in the stomach with pathognomonic features, which allowed for the definitive diagnosis with no need for further diagnostic tests.


Pilocytic astrocytomas (PAs) were recognized as a discrete clinical entity over 70 years ago. They are relatively benign (WHO grade I) and have, as a group, a 10-year survival of over 90 %. Many require merely surgical removal and only very infrequently do they progress to more malignant gliomas. While most show classical morphology, they may present a spectrum of morphological patterns, and there are difficult cases that show similarities to other gliomas, some of which are malignant and require aggressive treatment. Until recently, almost nothing was known about the molecular mechanisms involved in their development. The use of high-throughput sequencing techniques interrogating the whole genome has shown that single abnormalities of the mitogen-activating protein kinase (MAPK) pathway are exclusively found in almost all cases, indicating that PA represents a one-pathway disease. The most common mechanism is a tandem duplication of a ≈2 Mb-fragment of #7q, giving rise to a fusion between two genes, resulting in a transforming fusion protein, consisting of the N-terminus of KIAA1549 and the kinase domain of BRAF. Additional infrequent fusion partners have been identified, along with other abnormalities of the MAP-K pathway, affecting tyrosine kinase growth factor receptors at the cell surface  as well as BRAF V600E, KRAS, and NF1 mutations among others. However, while the KIAA1549-BRAF fusion occurs in all areas, the incidence of the various other mutations identified differs in PAs that develop in different regions of the brain. Unfortunately, from a diagnostic standpoint, almost all mutations found have been reported in other brain tumor types, although some retain considerable utility. These molecular abnormalities will be reviewed, and the difficulties in their potential use in supporting a diagnosis of PA, when the histopathological findings are equivocal or in the choice of individualized therapy, will be discussed. The term “pilocytic” to describe astrocytoma variants has been used since the 1930s , 18 to iIn this paper, we will review our current knowledge of the histopathological and molecular aspects of PA. As defined in the current WHO Classification System , PA makeAlmost all PAs are considered WHO grade I. A rare variant termed “pilomyxoid astrocytoma,” occurring predominantly in children under 1 year of age and in the hypothalamic/chiasmatic region, has been assigned WHO grade II. Uncommonly, cellular and highly atypical PAs with frequent mitoses may be seen and could be considered on the basis of the histological findings to be anaplastic, but no WHO grade III variant is included in the WHO Classification. Both these rare variants will be discussed below. For a short review of the WHO grading system, see , 54.Presenting symptoms will generally be insidious due to the slow growth of the tumor, and the identification of early symptoms will be dependent on localization and the ability of the patient to communicate neurological change and discomfort resulting from, for example, increased intracranial pressure. Common presenting symptoms for cerebellar tumors include ataxia, cranial nerve defects, and signs of increased intracranial pressure . When present in the optic pathways, the tumors may produce loss of visual acuity or field defects and, when localized to the hypothalamus, may result in endocrine syndromes, such as diabetes insipidus, precocious puberty, or electrolyte imbalance. Blocking of CSF pathways may result in hydrocephalus with rapid deterioration.Pilocytic astrocytomas usually appear on CT scans as round/oval lesions that are well-defined iso- or slightly hypo-dense and markedly enhance with contrast media. On MRI, PAs are typically hypo- or iso-intense on T1 sequences and hyperintense on T2-weighted or FLAIR images. They are typically strongly and diffusely enhancing  processes and elongated cytologically bland nuclei Fig. c, as weln = 14) (41 %) or documented by previous biopsy (n = 10); while the remaining 10 (29 %) exhibited typical pilocytic features in an otherwise anaplastic astrocytoma. Only four had received radiation. Eight patients (24 %) had a history of NF1. Four histological patterns of anaplasia were identified: (1) “pilocytic-like” (41 %) with classic bipolar cells with Rosenthal fibers and/or microcysts with eosinophilic granular bodies but with brisk mitotic activity and hypercellularity; (ii) poorly differentiated, small cell (32 %); (iii) epithelioid or rhabdoid (15 %); and (iv) cases resembling a classic diffusely infiltrative fibrillary astrocytoma (12 %). The presence of anaplastic features was associated with decreased survival when compared with typical PA. Interestingly, however, when compared to the historical cohort of diffusely infiltrative astrocytomas graded using the Mayo-St. Ann system   , 67. A sKIAA1549:BRAF fusion is extremely common in the cerebellum (found in approximately 90 % of cases) but somewhat less so supratentorially . FGFR1 alterations are largely restricted to midline structures, while BRAF V600E and NTRK family fusions are relatively more common in supratentorial tumors as a whole  can be used together with other findings to support the diagnosis of PA. It is clear, however, that the absence of such a fusion provides no diagnostic information, as there are so many other ways the MAP kinase pathway has been found to be activated in PAs. Unfortunately, many of the non-KIAA1549:BRAF alterations are also found to be similarly aberrant in the tumor types that are frequently among the differential diagnoses one has to consider when dealing with a difficult case. For example, the BRAF V600E mutation, while it occurs in a small subset of PAs, is a common mutation in gangliogliomas as well as pleomorphic xanthoastrocytomas and has also been reported in dysembryoplastic neuroepithelial tumors , 56. ThiIn general, PAs are considered to have an excellent prognosis with overall 10-year survival reported to be over 90 % . In addiPilocytic astrocytomas are primarily treated by surgery. This may be followed up by radiotherapy, particularly when there has been incomplete resection. Chemotherapy may be given in some cases where the tumor progresses, especially where further surgery is not possible.Pilocytic astrocytomas only very seldom progress to a more malignant form, with the vast majority, even after multiple recurrences, maintaining their morphology and WHO grade I designation. However, small numbers of cases of apparent malignant transformation of a classical PA have been documented , 36, 52.NF1 or BRAF. In vitro studies have shown paradoxical activation of ERK by sorafenib in the context of the absence of wild-type NF1 [The molecular findings have resulted in preliminary clinical trials of inhibitors of BRAF or targets further down the MAP kinase pathway. Initial studies with BRAF inhibitors have been published and demonstrated our incomplete understanding of the way the inhibitors affect the components of the MAP kinase pathway as well as the connections between this pathway and the PI3K/AKT/mTOR pathway. Sorafenib treatment of a small series of patients produced an unexpected acceleration of tumor growth irrespective of whether the tumor had a mutation of type NF1 , and thetype NF1 . This matype NF1 ). Howevetype NF1 .While in its classic form and typical location PA can hardly be confused with any other CNS tumor, there are a number of situations in which making the diagnosis of PA may be challenging. The differential diagnosis among these tumors still largely relies on histopathological assessment. A finding of KIAA1549:BRAF fusion mutation and the absence of other changes, in association with the appropriate morphological features, is supportive of the diagnosis of PA. Unfortunately, most molecular markers cannot be used as definitive discriminatory findings as many have been reported to occur in primary brain tumors of various histogenesis in children and adults , 37, 55.The most frequent differential diagnosis of PA includes a number of “relatively circumscribed tumors” mostly of low grade (WHO grade I), including ganglioglioma (GG), dysembryoplastic neuroepithelial tumor (DNET), rosette-forming glioneuronal tumor of the fourth ventricle (RFGNT), but also pleomorphic xanthoastrocytoma (PXA) (WHO grade II). However, even glioblastoma can occasionally be considered.Ganglioglioma is a grade I glioneuronal tumor composed of dysmorphic, frequently multinucleated ganglion cells accompanied by a glial component, resembling most often PA and less frequently either a diffuse astrocytoma or PXA. The number of neoplastic ganglion cells can be quite variable, and examples of truly “ganglion cell-poor” GG occur not infrequently. We have repeatedly encountered cases located in the temporal lobe (the most frequent single site of occurrence of GG) in which a first biopsy/resection showed a morphologic picture consistent with PA while a second resection, either to remove residual tumor or a tumor recurrence, demonstrated a definite GG. This has prompted us to often add a “disclaimer” to the diagnosis of PA in the temporal lobe “that PA is uncommon in the temporal lobe, and cases have occurred in which a second resection has disclosed a GG.” At the same time, in the cerebellum, GG should not be over-diagnosed, as large entrapped and distorted neurons from the deep nuclei can easily be mistaken for neoplastic ganglion cells. Pilocytic astrocytoma may mimic DNET with its typical oligodendroglial-like appearance; although frequently, the presence of glomeruloid-like vessels and the imaging appearance with enhancement point to the correct diagnosis.KIAA1549–BRAF fusions or BRAF mutations. Recently, FGFR1 mutations, previously described in PAs [Rosette-forming glioneuronal tumor (RFGNT) is a very rare glioneuronal tumor, which occurs within the fourth ventricle region, affects preferentially young adults, and frequently shows histologic similarities with PA. We have encountered examples in which a small focus with features of RFGNT was present in an otherwise classic PA Fig. . In contd in PAs , 67 haved in PAs , indicatPleomorphic xanthoastrocytoma, also a tumor of children and young adults, is typically a markedly cellular, epithelioid (rather than piloid) tumor. It is nearly always supratentorial, with only rare examples occurring in the cerebellum and spinal cord . UnusualIDH1 or IDH2 mutations, 1p19q co-deletion and/or presence of BRAF alterations are very helpful in sorting out the differential diagnosis, but this does not apply in children, in whom IDH1 or IDH2 mutations and/or 1p19q co-deletion are virtually absent. We have also seen supratentorial PAs, with marked cellular pleomorphism, recent hemorrhage and/or necrosis, and with prominent microvascular proliferation that have been mistaken for high-grade astrocytoma/glioblastoma. Critical examination of these cases has revealed a very low proliferative activity and lack of other features characteristic of a high-grade tumor; this has helped in reaching the correct diagnosis.The most challenging and clinically most relevant differential diagnosis of PA, given the differences in prognosis and the treatment implications, is with diffuse gliomas of low and high grade. Histologically, PA may: lack its typical biphasic appearance; show a predominant oligodendroglial-like or astrocytic appearance without distinctive features; undergo acute hemorrhage, which may result in an “alarming” imaging appearance ; show nePiloid gliosis, a type of chronic gliosis characterized by a dense “refractile” fibrillary background with varying degrees of hypercellularity and glial atypia and associated with Rosenthal fibers, may be very difficult to distinguish from a monomorphous densely fibrillated PA. Piloid gliosis can be seen in a variety of conditions, including the wall of a spinal cord syrinx, adjacent to long-standing tumors such as craniopharyngioma or spinal ependymoma or in cyst walls, including pineal cysts. In a small biopsy from the spinal cord, a diagnosis of “dense piloid gliosis with Rosenthal fibers cannot exclude PA” may be all that is possible. Rarely, dense Rosenthal fiber deposition, often perivascular and subpial, may be indicative of Alexander disease, a rare genetic disorder caused by mutations in the glial fibrillary acidic protein (GFAP) gene, which may uncommonly manifest in children and/or adults with a spinal or brain stem mass-like lesion, prompting consideration of a neoplastic process and biopsy. The potential pitfalls we described will be magnified when only limited tissue is available, as can happen with small biopsies, common at certain sites .We have come a long way in our understanding of the molecular changes that lie behind the development of PAs (and the other common pediatric brain tumors). The rate at which molecular findings have been incorporated into the diagnostic process at many centers has been impressive, but more remains to be done in this area. The molecular findings are complex, and the fact that the same mutations/rearrangements recognized to date can occur in many of the tumor types in our current classification and, in particular, are common to tumor types that can be included in the differential diagnosis of difficult cases where PA is being considered, makes their use demanding and difficult. At some point in the future, one can hope that specific patterns of multiple (both positive and negative) molecular findings will provide definitive diagnostic biomarker patterns. However, in the emerging era of personalized medicine and targeted therapies, “classification” arguably becomes less important as the molecular findings in the individual case, rather than a morphological grouping, will determine therapy. The ongoing updating of the WHO Classification of CNS tumors will ensure that the integration of molecular information into the neuropathological work-up of brain tumors becomes the norm . While a


Squamous cell carcinoma of unknown primary site (SC CUP) is a rare malignant tumor, and its histogenesis and appropriate treatment are unclear. To the best of our knowledge, this type of carcinoma with abdominal wall lesions as the primary presenting symptom 3 months after laparoscopic surgery, has not been previously described in the literature. In the present study, a postmenopausal 54-year-old female patient was diagnosed with pain from the right abdominal puncture site 3 months after laparoscopic unilateral left salpingo-oophorectomy at a local hospital, at which time the left ovary and Fallopian tube were free of malignant tumor. Computed tomography (CT) imaging showed a subcutaneous nodule with a size of 6.2×3.3 cm. A wide excision of the lesion with safety margins and repair of the abdominal wall was performed, and the histopathological results and various investigations lead to the diagnosis of metastatic well-differentiated SC CUP. The patient underwent three surgeries and eight cycles of Taxol and cisplatin/carboplatin chemotherapy, and received a total of 10.8 Gy palliative radiation. However, the patient succumbed to intestinal bleeding, thrombocytopenia and multiple organ failure with pelvic recurrence and liver metastases at 10 months post-diagnosis. The prognosis of SC CUP, particularly with multiple metastases, is extremely poor. Although chemotherapy, surgery and radiotherapy have a certain role in the treatment, no regimen has been established as a standard therapy and palliative care could be recommended. Carcinoma of unknown primary site (CUP) is defined as metastatic cancer that is present in the absence of an identifiable primary tumor site, even following thorough clinical examinations and diagnostic studies. CUP is a heterogeneous group of nosological entities based on histological, clinical, therapeutic and prognostic characteristics, and accounts for 3–5% of all malignancies. According to its histopathological characteristics, CUP can be classified into four major subtypes: well- or moderately-differentiated adenocarcinomas; undifferentiated or poorly-differentiated adenocarcinomas or carcinomas; squamous cell carcinomas (SCC); and undifferentiated neoplasms. SCC accounts for 15% of CUP, conferring a poor prognosis, with a median survival time of only 6–9 months , and theIn March 2013, a 54-year-old female  presented with a left ovarian cyst measuring 7 cm during a regular medical examination in a local hospital. There was no history of loss of appetite, weight loss or any other bladder- or bowel-associated symptoms. A clinical evaluation was made with regard to ultrasound and serological markers. Ultrasound examination showed a left ovarian cyst of 7×6×5 cm, with a normal uterus, right ovary and fallopian tubes. The serum level of carbohydrate antigen 19-9 (CA19-9) was above normal at 94.4 U/ml . Cancer antigen 125 (CA125), SCC antigen (SCC-Ag), α-fetoprotein (AFP), carcinoembryonic antigen (CEA) and neuron-specific enolase (NSE) levels were all within the normal ranges. A provisional diagnosis of an ovarian endometrioid cyst was made.The patient underwent a laparoscopic unilateral left salpingo-oophorectomy. The left ovarian mass, adhering to the broad ligament, was removed along with the fallopian tube. The cyst broke upon separating the adhesion and ‘chocolate-like’ fluid flowed into the pelvic cavity. The cyst wall was removed through the laparoscopic puncture hole on the right abdominal wall. There were no other abnormalities in the abdominal cavity, which was irrigated with normal saline several times prior to closure. Histopathological analysis identified the mass as an endometrioid cyst.The patient reported pain from the right laparoscopy puncture site at 3 months post-surgery. A 6.2×3.3-cm subcutaneous nodule was found by computed tomography (CT) imaging . FurtherDue to the malignant nature of the disease, the patient was admitted at the Zhejiang Cancer Hospital  for immediate further treatment on October 15, 2013. The first aim was to identify the primary site using a human papilloma virus test, cervical biopsy, gastroscopy examination and laryngoscopic examination, all of which were negative. Histological materials that were prepared at the time of the first surgery were examined section by section, and were all found to be without malignancy. Positron emission tomography (PET)/CT imaging was performed and revealed hypermetabolic lesions in the right pelvic cavity  and right inguinal lymph node . A biopsy of this inguinal lymph node revealed moderately-differentiated metastatic SCC . The finImmunohistochemical analysis showed poorly-differentiated invasive or metastatic SCC in the left broad ligament, the pouch of Douglas and the left lower abdominal wall , which s2; Taxol, 150 mg/m2), which was followed by 7 cycles of carboplatin/Taxol  due to severe vomiting. During chemotherapy, the serum levels of CA19-9 declined to 18.2 U/ml until the eighth cycle. Pelvic CT imaging showed recurrence in the right pelvic cavity, dilatation of the right ureter and right hydronephrosis. Following the ureteroneocystostomy, the patient was planned to receive palliative pelvic external irradiation with a total dose of 45 Gy in 25 fractions, 5 days per week, to eradicate any pelvic residual disease. However, liver metastasis and pelvic progression (Following the surgery, the patient received chemotherapy every 3 weeks for ~6 months. The first chemotherapy cycle was with cisplatin/Taxol (cisplatin, 70 mg/mgression , which cCUP are defined as a histologically proven metastatic malignant tumors with a poor prognosis, whose primary site cannot be identified following a thorough pre-treatment workup . CUP is Prior to reaching a diagnosis of CUP, various examinations are required in order to identify the primary site. These examinations should include a detailed medical history, a complete physical examination, including pelvic and rectal examinations, a full blood count and biochemistry analysis, a urinalysis and stool occult blood testing, a histopathological review of any biopsy material using immunohistochemistry, chest radiography, CT imaging of the abdomen and pelvis, and in certain cases, mammography ,5. In thRoutine evaluation of commonly used epithelial serum tumor markers, including CA19-9, CA125, SCC-Ag and CEA, usually have no diagnostic value in identifying the primary site ; howeveret al (et al (CUP remains an extremely aggressive disease with a poor prognosis. The median survival time is between 4 and 12 months –10, and et al  recordedl (et al  stated tet al (et al (Treatment regimens differ according to the location of the tumor. Although chemotherapy has a role in the treatment of CUP, no regimen has been established as a standard first-line therapy . The majet al , cisplatl (et al  found thl (et al . In the One of the most important targets when treating cancer patients should be prolonging the survival time. Although platinum-based chemotherapy or acceptance into clinical trials may be offered to patients of a relatively young age and good PS, supportive care should be recommended for other patients . Followiet al (et al (The prognostic factors reported for CUP, including age, gender, PS, weight loss, pathological subtype, tumor location, number of metastatic sites and serum markers, have been examined in several previous studies ,17–19. Ket al  conductel (et al . The patAdditional points also require consideration in the present case study. First, although all the available pathological material from the first surgery was examined and the results were shown to be malignancy-free, the possibility of primary SCC of the ovary could not be completely excluded. In the first surgery, all the resected tissues, including the cyst wall, left ovary and Fallopian tube, were transferred into an internal bag and removed through the 10-mm puncture hole on the right abdominal wall. Due to the large volume, these tissues were cut into several sections while in the bag. Cutting the cyst wall is not optimal for good subsequent pathological examinations, and it is therefore not certain that the complete cyst wall was examined without any omission. Second, the ‘chocolate-like’ fluid that flowed into the pelvic cavity upon breaking of the ovarian cyst may have caused the spread and implantation of potential malignancy, even though the peritoneal cavity was irrigated. During such clinical work, experience from the present case study would maintain the recommendation of using a bag to remove tissues from the abdominal cavity, even in cases where the lesions are considered to be benign. In addition, thorough washing of the abdominal cavity upon tumor rupture, should be performed. During laparoscopic surgery, a method to remove resected tumor tissues that are much larger than the puncture holes should be considered with regard to the requirement for a comprehensive histopathology analysis.In conclusion, the present study describes a rare case of SC CUP with abdominal wall lesions as the primary symptom. To the best of our knowledge, this is the first case study to have investigated this tumor type. The diagnosis, pathogenesis, treatment and prognosis of this type of tumor has generally been considered poor, thus, additional case reports are required to facilitate the clinical diagnosis and the appropriate course of treatment for affected patients.


Lymphoma of the uterine cervix is very rare. We report a case of diffuse large B cell lymphoma (DLBCL) involving the uterine cervix treated at a newly commissioned semiurban cancer centre in north India in 2015. Data for this study was obtained from the hospital electronic medical records and the patient's case file. We also reviewed published case reports of uterine and cervical lymphoma involving forty-one patients. We treated a case of stage IV DLBCL cervix with six cycles of R-CHOP  and intrathecal methotrexate followed by consolidation with radiotherapy. The patient showed complete response to chemotherapy. We conclude that, in advanced stage lymphoma involving uterus and cervix, combination of chemotherapy and radiotherapy is effective in short term. Non-Hodgkin's Lymphoma (NHL) affects extranodal sites in one-third of cases. The most commonly affected extranodal sites are the gastrointestinal tract and skin. Rarely may female reproductive organs be involved, most commonly ovary. NHL of the cervix is extremely rare. In one series, cervix was involved in 1 out of 730 cases of NHL and 1 out of 175 cases of extranodal lymphoma  with abnA 61-year-old postmenopausal lady presented with bleeding per vaginum and 6 kg weight loss in preceding 3 months. Her performance status was 1 . Local examination revealed a 7 × 6 cm mass in the cervix and extending to lower uterus and upper third of vagina, involving both parametria. Rectal mucosa was uninvolved. There was no hepatosplenomegaly or lymphadenopathy. Contrast Enhanced Computed Tomography (CECT) scan of chest abdomen and pelvis revealed a 8.7 × 7.9 cm mass in cervix with extension into uterus and lower cervix, abutting the urinary bladder, laterally involving parametrium, encasing right ureter causing mild-to-moderate hydronephrosis and posteriorly loss of fat planes with rectum; bilateral external iliac lymph nodes were enlarged largest 2.5 × 2 cm); 1.3 cm mildly enhancing mass in uncinate process of pancreas; multiple hypodense lesions in both lobes of liver, largest being 1.5 cm; and 2.8 × 2.4 cm soft tissue deposit in subcutaneous plane in right lower chest wall and multiple subcentimetric nodules in bilateral lungs (Figures .5 × 2 cmNHL of the uterine cervix is a rare tumour. The most common symptoms are vaginal bleeding, vaginal discharge, and pelvic discomfort and dyspareunia . DiffereNHL of uterine cervix is a rare tumour. It may be primary or secondary. It is usually high grade B cell lymphoma. Deep biopsy is required for diagnosis as surface cytology is frequently negative. Standard treatment is not established. In advanced stage disease, combination of chemotherapy and radiotherapy is the treatment of choice with encouraging early outcomes. This treatment is feasible in semiurban settings in developing countries.


Bladder cancer constitutes the ninth most common cancer worldwide and approximately only 30% of cases are muscle invasive at initial diagnosis. Regional lymph nodes, bones, lung, and liver are the most common metastases from bladder cancer and generally from genitourinary malignancies. Muscles constitute a rare site of metastases from distant primary lesions even though they represent 50% of total body mass and receive a large blood flow. Skeletal muscles from urothelial carcinoma are very rare and up to date only few cases have been reported in the literature. We present a rare case of 51-year-old patient with metastases to sartorius muscle 8 months after the radical cystectomy performed for a muscle invasive bladder cancer. Bladder cancer constitutes the ninth most common cancer worldwide and approximately only 30% of cases are muscle invasive at initial diagnosis , 2. We pA 51-year-old male presented with gross macroscopic hematuria, and the ultrasound imaging suggested the presence of a bladder wall tumor, which was confirmed with a cystoscopy. Patient underwent a transurethral resection of the bladder tumor. The pathology report confirmed a pT2 muscle invasive bladder cancer. The computed tomography of the abdomen and pelvis was negative for metastases. After 10 days the patient was subjected to a radical cystoprostatectomy and urinary diversion to an orthotopic ileal neobladder with a modified S-pouch. The final pathology report described a tumor to the posterior wall and the dome of the bladder. The prostate, the seminal vesicles, the distal ureters, and the lymph nodes were free of cancer involvement. The surgical margins were also negative. Patient did not receive chemotherapy in a neoadjuvant or adjuvant setting. The patient 8 months postoperatively complained of left thigh pain and a palpable mass. Thereafter, a full body MRI was performed and revealed a lesion of about 8 cm in diameter in the upper portion of the sartorius muscle, with no other abnormalities or metastatic sites.The patient underwent surgical resection of the lesion after induction of general anesthesia. The mass involved only the muscular plane of the sartorius, 10 cm approximately from the origin without evident contact with the tendon or adjacent muscle. A marginal excision of the lesion was performed. Macroscopically, it was an 8 × 4 × 4 cm lesion with smooth margins and the cross-section was white and shiny Figures . The posRegional lymph nodes, bones, lung, and liver are the most common metastases from bladder cancer and generally from genitourinary malignancies . Less fr


Malignant thymomas are rare epithelial neoplasms of the anterior superior mediastinum that are typically invasive in nature and have a higher risk of relapse that may ultimately lead to death. Here we report a case of an advanced malignant thymoma that was successfully treated with neoadjuvant chemotherapy followed by surgical resection and subsequently with advanced and novel radiation therapy techniques. A 65-year-old male was diagnosed with a stage IV malignant thymoma with multiple metastatic lesions involving the left peripheral lung and pericardium. Initial neoadjuvant chemotherapy with a cisplatin-based regimen resulted in a partial response allowing the inoperable tumor to become operable. Following surgical resection of the residual disease, the tumor recurred within a year. The patient then underwent a course of targeted three-dimensional intensity modulated radiation therapy (IMRT) and image-guided radiation therapy (IGRT). Five years after radiation therapy, the localized soft tissue thickening at the left upper lung anterior pleural space had resolved. Seven years after radiation therapy the tumor mass had completely resolved. No recurrences were seen and the patient is well even 8 years after IMRT/IGRT with a favorable outcome. Chemotherapy with targeted three-dimensional IMRT/IGRT should be considered the primary modality for the management of advanced malignant thymoma patients. Thymomas are rare epithelial neoplasms arising from the thymus gland and account for nearly all primary malignancies of the anterior superior mediastinum -3. The tIn general, thymomas are indolent tumors with a tendency toward local recurrence rather than metastasis. Thymic carcinomas, however, are typically invasive, with a higher risk of relapse and ultimately lead to death ,10. ThymVarious case series and small prospective trials have shown the clinical effectiveness of chemotherapy with multimodality regimens in the management of advanced malignant thymomas. Multi-agent chemotherapy is used neoadjuvantly to downstage the tumor rendering inoperable carcinoma operable or as palliative treatment to extend the patients’ life and improve their quality of life. Specifically, platinum-based chemotherapy using cisplatin in combination with vincristine, doxorubicin and etoposide have been shown to render inoperable invasive thymomas operable tumors . Many caIn this report, we describe a challenging case of a recurrent advanced malignant thymoma complicated by the presence of metastatic lesions in the lung and pericardium. Specifically, we present our experience in managing recurrent advanced malignant thymomas using multi-agent chemotherapy with cisplatin, adriamycin, vincristine, and cytoxan followed by surgical resection, intensity modulated radiation therapy (IMRT), and image-guided radiation therapy (IGRT). IMRT is an advanced mode of high-precision radiotherapy that uses computer-controlled multiple small radiation beams of varying intensities to deliver precise radiation doses to a malignant tumor or specific areas within the tumor. By incorporating three-dimensional computed tomography (CT) imaging technology, IMRT allows the radiation dose to conform more precisely to the three-dimensional shape of the tumor while modulating the intensity of the radiation beam and minimizing its dose to those organs and tissues unaffected by the cancer. IGRT uses a variety of two-dimensional, three-dimensional and four-dimensional imaging techniques that improve the precision and accuracy of the delivery of radiation dose to the targeted tumor tissue while minimizing the dose to the surrounding normal tissue during the course of radiation therapy.A 65-year old Hispanic male presented with complaints of chest pain and heaviness prior to the diagnosis of his disease. The patient had a history of tobacco use but had stopped smoking 20 years earlier. The patient had been otherwise healthy all of his life. Approximately 4 to 6 weeks prior to his diagnosis, he experienced an episode of chest pain and some mild shortness of breath. Initially, the patient underwent a routine chest X-ray and CT scan diagnostic procedures. Subsequently, a thorascopic biopsy was performed, and using H & E staining, tissue histology was carried out to detect the tumor development. In addition, the patient underwent a mediastinoscopy, thoracoscopy, and fine-needle biopsy to confirm the diagnosis of his disease.The diagnostic evaluation revealed the presence of a stage IV malignant thymoma with multiple metastatic lesions involving the left peripheral lung and pericardium. Despite the presence of metastatic disease, these lesions are still oftentimes known to be amenable to surgical resection following treatment with induction chemotherapy. Therefore, to down- stage the tumor and to improve the patient’s chance of increased surgical resectability, neoadjuvant chemotherapy was initially employed. Since platinum with an anthracycline-based triplet or quartet regimen is currently the consensus treatment for malignant thymoma -17, thisIn attempt to salvage this patient, the patient underwent a course of post-operative and post-chemotherapy IMRT/IGRT-based three-dimensional radiation therapy to a dose of 7,440 cGy to the left chest wall, mediastinum, and left pericardium Figure . The patInitial findings from the thorascopic biopsy indicated that the patient had multiple pleural-based masses with an associated small left pleural effusion. Assessment of tissue histology using H & E staining revealed a mixture of plum epithelial cells with both vesicular nuclei and distinct nucleoli and small lymphocytes indicating the presence of malignant thymoma Figure . FollowiThe initial five cycles of neoadjuvant chemotherapy with cisplatin, adriamycin, vincristine, and cytoxan was well tolerated by the patient. However, this induction therapy only resulted in reduction in the size of the tumor masses. However, a partial response was observed after the subsequent chemotherapy with four cycles of cisplatin, adriamycin, and cytoxan. The patient then underwent surgical resection of his residual disease. Despite treatment with chemotherapy and surgical resection, the patient experienced recurrent chest disease within a year. An attempt was then made to salvage the patient and he was treated with radiation therapy.A pre-radiation therapy chest CT imaging revealed several mass structures with the largest size measuring 3.7 × 3.0 cm in the left hemithorax (Figure et al. [Malignant thymomas are rare human neoplasms accounting for less than 0.5% of all malignancies. Due to their rarity, knowledge of the optimal treatment regarding these tumors is based on case reports or small retrospective series. Management of thymic carcinoma depends upon the clinical stage of the disease. Currently, surgical resection remains the mainstay of treatment for localized thymic malignancies . Howeveret al.  has showIn general, extensive mediastinal or lung invasion or metastasis is a frequent finding in most newly diagnosed patients. Despite complete surgical resection, over 50% of patients with advanced disease experience recurrence . ConsequThe present patient was diagnosed as having an advanced stage IV malignant thymoma with multiple metastatic lesions involving the left peripheral lung and pericardium. Therefore, induction chemotherapy was employed, followed by surgery, and then post-operative definitive targeted three-dimensional IMRT/IGRT in this patient. Since cisplatin-based combination chemotherapy is effective against thymic tumors ,38,39, cLittle has been reported on the role of advanced radiation therapy in the management of malignant thymoma. Until recently, no literature has demonstrated the superiority of one radiation therapy method over another. Despite the sensitivity of thymoma to radiation, the best use of radiotherapy remains controversial. Moreover, there remains no consensus on whether or not adjuvant radiation is of any benefit in completely resected thymoma. However, in this case study, we have demonstrated that by using targeted three-dimensional IMRT/IGRT, we were able to address the patient’s extensive recurrent disease and treat critical structures that were involved by the disease. In addition, our treatment approach spared the patient from any long-term detrimental side effects, especially to his myocardium and other critical organs.Multimodality therapy involving both neoadjuvant and post-operative chemotherapy in conjunction with three-dimensional well-targeted IMRT/IGRT appears to increase the success rates of complete resection and improve survival in patients with metastasized thymic carcinoma. Neoadjuvant chemotherapy with cisplatin, adriamycin, vincristine, and cytoxan resulted in a partial response allowing the inoperable tumor to become operable in this patient. Following surgical resection, targeted three-dimensional IMRT/IGRT was well tolerated without any cardiac toxicity. Targeted three-dimensional IMRT/IGRT proved to be clinically effective resulting in no serious adverse events and more importantly achieving a complete response of the tumor leading to long-term tumor control and increased survival of the patient with minimal compromise in his quality of life.A 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.