Solitary angiokeratoma of oral mucosa is rare entity. The term Angiokeratoma is used to refer to several lesions, whose common denominator is the presence of dilated blood vessels in association with epidermal hyperplasia. Mucosal involvement, including oral cavity is occasionally found either as a component of the systemic variety, cutaneous involvement or isolated oral involvement. Clinically, the lesion is irregular, whitish to dark brown in color, with female predominance. The etiological factors include injury, trauma, or chronic irritation to the wall of a papillary dermis. Histologically, it is characterized by hyperkeratosis, acanthosis, and dilated vascular spaces with or without organizing thrombi in papillary dermis. The vascular spaces are partly or completely enclosed by elongated ret-ridges. Along with this reporting a case of solitary angiokeratoma affecting tongue in a 38-year-old male patient, along with the literature review is presented. Angiokeratoma is an acquired vascular lesion which is characterized histologically as one or more dilated blood vessels lying directly subepidermally and showing an epidermal proliferative reaction especially acanthosis and hyperkeratosis with dilated capillaries in the papillary dermis .Several clinical types have been described depending on the multiplicity and location of the lesions. They can be divided into localized and systemic types . MucosalHowever, solitary angiokeratomas of the oral mucosa seem to be a rather infrequent occurrence, and very few cases have been reported in the literature. According to the best of our knowledge, since 1997 till date, only 16 cases involving oral cavity have been reported. A 38-year-old male patient reported to the Department of Oral Pathology with chief complaint of growth on tip of tongue since last 10 years. The patient was apparently all right 10 years ago when he noticed small painless growth, and then the growth steadily increased in size up to present size involving right side of the tip of the tongue. Sometimes the Patient also experienced bleeding in that area which was associated with trauma during mastication. Bleeding subside, on its own, the patient had never taken any treatment for that growth and not even for bleeding episodes. No abnormality was reveled in his medical, personal histories and general examination. The patient had a habit of tobacco chewing for about 10–15 years.On clinical examination, it was observed that well-circumscribed sessile growth is present on the dorsal surface of tip of the tongue, and the growth is of approximately 1 × 1 cm in dimension, ovoid in shape. Growth was dark brownish in color with a granular surface texture . On palpAfter routine hematological investigations, under local anesthesia, the lesion was completely excised and taken for histopathological investigation. The gross specimen is irregular in shape approximately 1 × 1 × 0.5 cm in size, brownish in color, and soft in consistency with rough surface .Histopathologically, parakeratotic stratified squamous epithelium of varying thickness with long slender rete ridges and in some areas large bulbous rete ridges is evident. Papillary connective tissue shows numerous large dilated blood-filled spaces and lined by endothelial cells. Areas of extravasations of blood are also present. Chronic inflammatory cell infiltration around blood vessels and rete ridges is also present. All these features were suggestive of a diagnosis of angiokeratoma Figures  and 4.For the confirmation of proliferation of blood vessels, CD34 marker was used. The lesion was positive for CD34 . After diagnosis, the patient underwent further examinations, and no lesions were found elsewhere in his skin or mucous membranes. The case was considered a solitary angiokeratoma affecting the tongue. In the last followup after six months, the patient was disease-free and asymptomatic. Solitary angiokeratoma was first described in 1967 by Imperial and Helwig . SolitarPathogenesis of the lesion includes relation to trauma, high venous pressure, or vascular malformation . It is tReview of all the past cases suggests that it is more common in female, but the present case patient was male. The most common site of involvement in the oral cavity is the tongue, the anterior dorsal surface. In present case the site of involvement was also the tongue.The only clinical problems these lesions can cause are bleeding, discomfort or cosmetic changes . However Type 1: primary (purely mucocutaneous and not associated with systemic disorders) Type 1A, isolated angiokeratomas of the oral cavity Type 1As solitary  Type 1Am multiple  Type 1B, mucocutaneous angiokeratomas, that is, oral angiokeratomas associated with cutaneous angiokeratomas  Type 1Bs solitary Type 1Bm multiple Type 1C, angiokeratomas occurring simultaneously in oral cavity, skin , and gastrointestinal mucosa Type 1Cs solitary Type 1Cm multiple Type 2: secondary  Type 2A, As a component of Fabry's disease Type 2As solitary Type 2Am multiple Type 2B, as a component of fucosidosis Type 2Bs solitary Type 2Bm multipleOral mucosal involvement is a component of angiokeratoma corporis diffusum . If furtType 1As, that is, isolated solitary angiokeratoma.Considering the same classification, the present case can be categorized as The main differential diagnosis on histopathological grounds was lymphangioma, to exclude the diagnosis and to confirm the proliferating blood vessels. Immunohistochemical staining is implied. In the previous literature, antigens used were CD31, CD34, and LYVE-1 , and CD31 and CD34 were found positive and LYVE-1  was negative [The differential diagnosis of angiokeratoma is important because of its similarity to some other lesions , 12. OthIn case of lymphangiomas, multiple intertwining lymph vessels lie very close to the epithelium and are seen also in papillary connective tissue. Presence of blood-filled spaces and endothelial lining also helped to differentiate angiokeratoma from lymphangiomas .Angiokeratoma can be clinically confused with the aggressive lesions like malignant melanoma, especially in case of angiokeratoma when the vessels are thrombosed . HistopaOral angiokeratomas of the oral cavity are rare tumors. Although they can appear as isolated lesions, their presence should prompt further investigations to rule out systemic disease.


Ventriculoperitoneal shunt (VPS) placement is an established procedure for the treatment of hydrocephalus of diverse etiologies in children and adults. Abdominal cerebrospinal fluid pseudocyst, which is potentially life threatening, is a rare complication and usually occurs during childhood. However, with increasing longevity following successful treatment, it can also occur in adults.Here we describe a 22-year-old man who was admitted to our hospital because of diffuse abdominal distention. A VPS was placed 21 years earlier to treat hydrocephalus secondary to spina bifida. Abdominal computed tomography (CT) revealed a homogeneous low-density fluid collection adjacent to the VPS catheter tip, causing stomach obstruction. Thus a peritoneal pseudocyst around VPS was suspected and emergency laparotomy was performed. The large mass was localized in the left upper abdomen between the stomach and mesentery of the transverse colon, exactly at the omental bursa. The cystic mass was opened and 1500 ml of clear fluid was drained; the distal end of the VPS was repositioned outside the mass. Thus, an abdominal cerebrospinal fluid pseudocyst as a complication of VPS was diagnosed.Gastroenterological surgeons should be aware of this possible complication, and this complication should be considered during differential diagnosis of an acute abdomen complaint. Ventriculoperitoneal shunt (VPS) placement is a surgical procedure performed to relieve high intracranial pressure caused by hydrocephalus of diverse etiologies in children and adults. VariousA 22-year-old man presented to our hospital with progressive abdominal distention and nausea. A VPS had been placed 21 years earlier to treat hydrocephalus secondary to spina bifida. Shunt revision was performed only once when he was 10 months old, and no other abdominal surgery was performed after this. He had no history of malignancy or pancreatic or liver disease. Physical examination revealed a large elastic palpable mass in the upper abdomen. No neurological change was observed. His white blood cell count and amylase serum levels were normal. The patient underwent supine abdominal radiography and unenhanced abdominal computed tomography (CT).Supine abdominal radiography revealed the VPS in the left upper quadrant and a soft-tissue mass in the upper abdomen Figure . AbdominThus, a peritoneal pseudocyst around the VPS causing stomach obstruction was suspected and emergency laparotomy was performed. The large mass was localized in the left upper abdomen between the stomach and transverse colon mesentery, exactly at the omental bursa Figure . After tThe findings of fluid analysis were similar to those of cerebrospinal fluid analysis, with negative cultures and absence of microbial flora.VPS is the most common treatment procedure for hydrocephalus; however, it is not free of complication. Abdominal cerebrospinal fluid pseudocyst is a rare, but potentially life-threatening complication of VPS placement. It is seen as a thin-walled cystic mass around the shunt tip, which is filled with cerebrospinal fluid. The wall is composed of fibrous tissue without epithelial lining. The underlying mechanisms involved in the formation of the cerebrospinal fluid pseudocyst remain unknown; however, inflammation, either sterile or infectious, is usually regarded as the main causative factor. In our The symptoms of abdominal cerebrospinal fluid pseudocysts in adult patients are abdominal pain, distention, and a palpable abdominal mass. Thus, adults predominantly present with abdominal signs only. On the other hand, in pediatric patients, symptoms of shunt malfunction, such as headache, nausea, and vomiting are more common.CT indicates a definitive diagnosis of abdominal cerebrospinal fluid pseudocyst, revealing a large fluid-filled collection delimited by a thin wall adjacent to the catheter tip. Differential diagnoses of an abdominal cystic mass include abdominal abscess, lymphocele, seroma, cystic lymphangioma, cystic mesothelioma, mesenteric cyst, benign cystic teratoma, cystic spindle-cell tumor, pancreatic pseudocyst, and duplication cyst. In our et al.[et al.[The time between the last VPS surgery and collection of abdominal cerebrospinal fluid has been reported to range from 3 weeks to 10 years. Recentlet al. and SenaTreatment of abdominal cerebrospinal fluid seems controversial. Therefore, many treatment algorithms have been described, such as laparotomy with wide excision of cystic walls, paracentesis and aspiration of the cystic fluid, CT-guided or ultrasound-guided aspiration of the pseudocyst. In the present case, exploratory laparotomy and excision of the cystic walls with fluid drainage was performed. Recently, laparosopic management, which is a useful tool, has been reported; howeverIn conclusion, this report described an adult case of a huge abdominal pseudocyst as a complication of VPS. Not only neurosurgeons or pediatricians but also physicians should be aware of this possible complication because early diagnosis and treatment would improve the clinical outcome and reduce the patient’s suffering and distress.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 Series Editor of this journal.VPS: Ventriculoperitoneal shunt; CT: Computed tomography.The authors declare that they have no competing interests. No financial support has been received.TA and TK collected the data, performed the treatment and wrote the paper; SD was responsible for writing the paper and its supervision. All authors read and approved the final manuscript.The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1471-2482/13/27/prepub


A 61-year-old man presented with hematuria and intermittent right pelvic pain. Intravenous urography showed a tubular filling defect and ureteroscopy a tumor in the right mid ureter. Urine cytology and tumor biopsy showed nonmalignant results. Open surgery was performed, and an intraoperative frozen section revealed a fibroepithelial polyp of the right mid ureter. A fibroepithelial polyp is a rare benign lesion that can occur in childhood but is an important differential diagnosis of an upper urinary tract urothelial cell carcinoma in adults. The upper urinary tract urothelial cell carcinomas (UUT-UCCs) account for only 5–7% of UCCs. Of these only 25% are located in the ureter. In western countries, the incidence is 1-2 cases per 100,000 per year [A 61-year-old man presented to our department with symptoms of gross hematuria and intermittent right pelvic pain. He had a past medical history of arterial hypertension, psoriasis, an annealed lung Tbc, and a smoking history of 60 pack years. Urine analysis showed microscopic hematuria. Biochemical data were within normal limits. Ultrasound and cystoscopy were normal. Intravenous urography (IVU) demonstrated a tubular filling defect with partial obstruction of the right mid ureter. Retrograde ureteropyelography confirmed a 5 cm long tubular mass in the mid ureter proximal to the iliac vessels . FurtherA fibroepithelial polyp of the ureter is a rare cause of hematuria and imaging findings that make it difficult to differentiate from UUT-UCCs, which are usually treated by RNU. A differential diagnosis between UUT-UCCs and UFP cannot be made with imaging tests only. Urine cytology and endoscopic biopsy alone might be insufficient to confirm the diagnosis as shown in our case report. A UFP is a rare benign mesodermal tumor with approximately 200 cases documented in the literature. In recent years, most cases are reported in Asian children, but older reviews show that it can occure in every age and not only in Asia . Histologically, these polyps are composed of stroma derived from the mesoderm and covered by a layer of normal transitional epithelial cells . Most UFUFP can occur in every age, but commonly they present in the third to fourth decades of adults. Males are 1.5 times more affected with it than females. Most UFP occur in the left ureter with a predilection for the proximal segment. Mean fibroepithelial polyp diameter is reported with less than 5 cm; however, larger polyps that even can protrude into the bladder have been reported , 8. UretMost common symptoms of patients with UFP are gross hematuria and intermittent right pelvic pain. Some cases are presented with hydronephrosis. IVU or retrograde pyelography typically demonstrates a tubular filling defect. This finding combined with negative cytology and biopsy should suspect a UFP. There is no evidenced guideline for the treatment of a UFP; however, priority should be the complete excision of the tumor with minimal risk of morbidity and maximal preservation of renal function. Despite no preoperative evidence of malignancy, many case reports of UFP include management by open or laparoscopic surgery. Recent publications refer endoscopic percutaneous or ureteroscopic approach excisions or laser coagulation of the polyp , 9. EndoA fibroepithelial polyp of the ureter is a rare benign tumor mimicking typical findings of UUT-UCCs. It has to be included in the differential diagnosis of UUT-UCCs, especially when urine cytology and biopsy are negative. In this condition, nephroureterectomy would be malpractice.


Situs inversus totalis represents an unusual anomaly characterized by a mirror-image transposition of the abdominal and thoracic viscera. It often occurs concomitantly with other disorders that make difficult diagnosis and management of abdominal pathology. The relationship between situs inversus totalis and cancer remains unclear.We describe a 33-year old Guinean man with situs inversus totalis who presented with obstructive jaundice. Imaging and endoscopic modalities demonstrated a mass of distal common bile duct which biopsy identified an adenocarcinoma. The patient was successfully treated by cephalic pancreaticoduodenectomy followed by adjuvant chemoradiation and he is doing well without recurrence 8 months after surgery.The occurrence of bile duct adenocarcinoma in patient with situs inversus totalis accounts as a rare coincidence. In this setting, when the tumor is resectable, surgical management should be considered without contraindication and must be preceded by a careful preoperative staging. Situs inversus totalis (SIT) is a rare congenital condition characterized by a mirror-image transposition of both the abdominal and thoracic viscera, its incidence accounts for 1/8000 to 1/25,000 of the normal population . An incrA 33-year old previously healthy man, with known SIT presented with 3 months history of obstructive jaundice and 10 Kg weight loss. Computed tomography (CT) confirmed the complete transposition of viscera as follows: dextrocardia, right subphrenic gastric bubble and spleen with a small accessory spleen, left sided liver and reversed superior vena cava, brachiocephalic artery and hepatic artery; CT also revealed a dilatation of intra and extra hepatic bile ducts secondary to stenosis of the distal common bile duct (CBD) Figure ; EndoscoSitus inversus is an uncommon congenital condition which is defined by a left-right transposition of the normally asymmetrical organs of the body as a mirror-image; transposition of thoracic and abdominal organs is termed situs inversus totalis . This foThe association of SIT and neoplasia is a rare coincidence; only sporadic cases have been reported. The first case was published by Maekawa in 1927; it was an autopsy case of gastric carcinoma in a 43-year-old man with SIT . SIT is SIT is an uncommon entity that often occurs concomitantly with other abnormalities. The relationship between SIT and cancer is not approved; further, studies are needed to identify precisely genetic and molecular patterns involved in development of malignancy in these patients. Because of the frequency of associated malformations of transposed organs and vascular and nervous anatomical variations that make difficult surgical management, special attention should be paid to the diagnosis and preoperative staging. Herein, we have described for the fourth time in the literature a case of unusual association of SIT and adenocarcinoma of common bile duct.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.SIT: Situs inversus totalis; CT: Computed tomography; ERCP: Endoscopic retrograde cholangiopancreatography; CBD: Common bile duct; CCDC11: Coiled-coil domain containing 11; DNHA1: Axonemal heavy chain dynein type 11; TGFβ: Tumors growth factor β; HNF-3β: Hepatocyte nuclear factor 3β; KIF3: Kinesin 3 family.The authors report no competing interests. The authors alone are responsible for the content and writing of the paper. All authors have contributed to this paper.HB* was involved in the management of that patient, the analysis of the data and the literature research and wrote the manuscript. SK helped with the literature research. ST performed radiological diagnosis and revising manuscript. TB was involved in the surgical management of the patient. TNT was involved in the endoscopic diagnosis. MG performed histological diagnosis. OE helped with the final editing of the manuscript. AR approved the treatment and analyzed the literature data. All authors read and approved the manuscript.


Primary non-Hodgkin’s lymphoma of the ovary is a rare occurrence. An ovarian involvement by non-Hodgkin lymphoma (NHL) may include one of the four subtypes of lymphoma: diffuse large B-cell lymphoma, Burkitt’s lymphoma (BL), lymphoblastic lymphoma or anaplastic large cell lymphoma. Burkitt’s lymphoma is a rare entity with a specific poorly differentiated pattern.Most women experience an ovarian BL with abdominal pelvic pain, abnormal vaginal bleeding, bowel obstruction, urinary frequency, incontinence and abdominal mass. Sometimes these warning signs may be absent, causing a late and more difficult diagnosis.Here we report a case of a primary ovarian Burkitt’s lymphoma with bilateral involvement in a 57 year old patient. She firstly presented neurological symptoms in the upper limbs and she was treated with surgery and combined chemotherapy. The diagnosis of malignant lymphoma was established after bilateral adnexectomy and histological study of the excised tissue. A 57-year-old pluriparous woman from Pakistan was admitted to the emergency ward due to a rapidly progressive weakness in her arms, bilateral hypoaesthesia of the 1st, 2nd and 3rd finger and wide-spread pain in her arms, shoulders and neck. Her medical history shows a TBC infection at the age of 17. MRI revealed volumetric increase of right C5-C6-C7 and left C6-C7 nerve roots with a mild contrast enhancement as idiopathic neuritis. Rachicentesis was negative. Serological HIV, HBV, HCV, neurotropic virus tests and autoantibodies were negative. The hematologic findings showed a low lymphocyte count and a slight increase in neutrophils.A Computer Tomography (CT) scan revealed multiple enlarged lymph nodes involving the bilateral supraclavicular, left laterocervical, left and right paratracheal, prevascular, aortopulmonary, left and right hilar and subcarinal groups Figure .In the pelvic cavity there was a solid inhomogeneous mass, with necrotic areas Figure .The patient was transferred to the gynaecological ward for a suspected adnexal neoplasia.Further investigation showed high tumour markers with CEA level of 3,3 ng/ml and Ca 19.9 level of 81,2 UI/ml; Ca 125, α-fetoprotein and β-HCG were negative. Serum immunofixation was negative for monoclonal components.Vaginal ultrasound confirmed the presence of a left ovarian mass; it measured 88 mm. There was no free fluid in the abdominal cavity.A bone marrow biopsy and aspiration were performed, molecular evaluation showed a polyclonal rearrangement of the IgH gene.The patient underwent a laparotomic bilateral annessiectomy; operative time length and mean haemoglobin drop value results were 60 min and 125 ml respectively. No adhesions of tumors and other organs, no intra-operative and post-operative complications were observed. Cytological examination of the peritoneal washing was negative for neoplastic cells.A histopathological examination revealed a high grade B-cell ovarian lymphoma.Immunohistochemical evaluation revealed the presence of tumour cells that were positive for B-cell markers with a high proliferation rate  Figure  and 4.The FISH test was negative for c-myc t  translocation and PCR was negative for t  translocation. These patterns indicated a not translocated Burkitt's lymphoma.2 iv and prednisone 60 mg/m2 ), cycle A1 from day 7, cycle A2 from day 49 and a cycle A3 from day 98 , cycle B1 from day 28, cycle B2 from day 77 and cycle B3 from day 119 .According to Ann Arbor Staging a diagnosis of stage IV A was confirmed. After surgical resection the patient was treated with an intensive G-mall chemotherapy protocol. In older patient (> 55 years) G-m all protocol contemplates a pre-phase of 5 days  is a highly aggressive, mature B-cell lymphoma that shows an extremely rapid growth rate ,2. In 20Primary ovarian lymphoma (POL) is an event that accounts for 0.5% of NHL and 1.5% of all ovarian neoplasia, usually bilateral and often associated with ascites -6.In 1976, POL was defined by Fox and Langley with the following criteria [(a) the disease has to be confined to the ovary,(b) absence of disease in the blood and bone marrow,(c) the extraovarian deposits, if any, should appear at least after few months.The most frequent clinical symptoms of gynaecologic BL are pelvic pain, abnormal vaginal bleeding, bowel obstruction and abdominal mass with a rapid growth; sometimes it shows unspecified symptoms with a more difficult differential diagnosis . BurkittMedical studies suggest that the origin of lymphomas in the ovaries is associated with the presence of preexisting lymphoid tissue in the ovary. Nevertheless other authors reveal that reactive lymphocytes may secondarily involve the ovary in response to inflammation  or autoimmune diseases and then they may undergo malignant change and give rise to POL ,6. The mThe prognosis of our patient, according to the International Prognostic Index of non-Hodgkin’s lymphoma, is a 5-year survival of 43%, corresponding to an high-intermediate risk . The IntWritten informed consent was obtained from the patient for publication of this Case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The patient of our case gave her consent concerning the case report.The authors disclose any commercial interest, any financial interest and any potential conflict of interest of other nature. No financial support has been required for this study.PB has conceived and designed the study, has been performed the surgical intervention and has given final approval of the version to be published; FT and GC have supervised the work and were involved in acquisition of data; MM has made CT scan images processing in final form for publication; VG and MV have followed the patient during hospitalization and have drafted the manuscript. All authors have read and approved the final manuscript.


Solitary fibrous tumors of the kidney are extremely rare tumors with unpredictable behavior. We describe a case of a patient with a solitary fibrous tumor of kidney with malignant findings with distant metastasis and nephrectomy managed with subcutaneous interferon achieving 23 months of progression-free survival. To date there is no prospective evaluation of any specific modality of treatment, but the surgical management and long-term followup are the only ones so far recommended strategies in the management of these patients. Studies are awaited with more patients to evaluate the different strategies of systemic therapy reported so far to allow adding survival benefit. Solitary fibrous tumors are rare mesenchymal neoplasms, considered a variant of hemangiopericytomas usually originates in the pleura; however, there have been reports of extrapleural origin  –4. The lA 49 year old woman with no history of importance who enters the emergency room by 2-months of dyspnea at rest, associated with pleuritic chest pain on right chest, dry cough, no fever. The chest radiograph showed a right pleural effusion and multiple nodular lesions on the pleura, so we decided to perform a CT chest and abdomen in which evidenced free right pleural effusion occupying 80% of the right chest, at least two pleural masses with solid density that capture the contrast, multiple pulmonary nodules with soft tissue density in both lungs and left kidney mass. Carried left nephrectomy with suspected metastatic renal carcinoma, shows renal mass plus liver metastases which were resected. The analysis of the tumor presented as a first option versus solitary fibrous tumor angiomyolipoma. Immunohistochemical studies that showed cell reactivity with CD34, CD99, BCL-2, and vimentin are negative for HMB-45, AMS, CD68, cytokeratin cocktail, and S100, and the Ki67 is not assessable . This prWe decided to start treatment with interferon a2b subcutaneous dose, extrapolating the cases of patients with solitary fibrous tumor of the pleura . The dosSolitary fibrous tumors are extremely rare tumors, arising mostly at the level of the pleura, and cases arising from the urogenital region are even more rare, with 49 cases reported so far in the literature. The histogenesis of this entity is still unknown, but recent studies suggest a primitive mesenchymal cells or level perivascular , 11.The differential diagnosis of these cases includes sarcomatoid variant of renal carcinoma, angiomyolipoma, fibromas, and fibrosarcomas. These results show a relatively rare entity, with peak presentation in the fifth decade of life, arising mostly in the renal parenchyma unilaterally .About 14% of patients have aggressive behavior where common management strategy is nephrectomy with complete resection of the lesions. The pathological findings that have been correlated with aggressive behavior are pleomorphism, increased cellularity and mitotic activity (>4 mitosis/10 high-power fields), necrosis, hemorrhage, and atypical sites  . HoweverIn relation to the management of this condition, there is as yet no prospective evaluation of any specific treatment modality; however, case reports and retrospective case series suggest that complete surgical resection and long-term followup are generally most recommended strategies. In cases such as the present, which present with metastatic disease, there is no clearly defined systemic therapy. Metastasectomy is thought to improve progression-free survival, but in many cases like this, this strategy is not feasible.In case reports hemangiopericytomas, entity closely related to solitary fibrous tumors, has achieved stable disease with the use of interferon with or without thalidomide. So far, this is the first case reported in the literature in which benefit is demonstrated with the use of interferon in a patient with a malignant variant renal solitary fibrous tumor with metastatic disease, achieving stable disease for about 20 months.Some authors suggest the use of antiangiogenic therapies , based on the findings of high vascularity and a possible origin of pericytes at this entity . The com


Eitel first described omental torsion in 1899, since then, fewer than 250 cases have been reported. Although omental torsion is rarely diagnosed preoperatively, knowledge of this pathology is important to the surgeon because it mimics the common causes of acute surgical abdomen. For this reason, in the absence of diagnosed preexisting abdominal pathology, including cysts, tumors, foci of intra-abdominal inflammation, postsurgical wounds or scarring, and hernial sacs, omental torsion still can represent a surprise. Explorative laparotomy represents the diagnostic and definitive therapeutic procedure. Presently laparoscopy is the first choice procedure. Omental Torsion (OT) is a condition in which a pedicle of the omental apron twists on its longer axis to such an extent that its vascularity is compromised. Eitel  in 1899 S. C., a retired man, aged 83, was admitted to our Hospital because of an intra-abdominal pain started a few days before in the absence of fever, vomiting or nausea. The patient felt a dull pain in right side of the abdomen for one day, he did not sleep during the subsequent night, then he was visited at home by his Practitioner who treated the patient pharmacologically. In the same day, when the abdominal pain became steady and dull, the patient was brought to First Aid Service of our University General Hospital. The Patient was affected by glaucoma, hypertension had a pace maker and had received right saphenectomy and right eye cataract interventions ten years before. At physical examination the abdomen appeared bloated, tenderly, with slow peristalsis, last evacuation the day before. There was moderate rigidity of the upper right side of the abdomen, with tenderness in the right and in the lower quadrants. At the admittance laboratory findings showed white blood cells count 7,640/mmc with 81.6% polymorphonuclear cells, increasing at the next days evaluations (91.6% polymorphonuclear cells), alfa-1 seroproteins 10.8 g/dl and glycaemia 173 mg/dl. As this disease may mimic other surgical emergencies, extensive imaging studies were performed.• Ultrasonography (US), which gave negative result,• Computerized Tomography (CT) Figure , 2 scan Histology Findings: omental pedicle of 18 × 13 × 6 cm in size, with lobed and cyanotic look. Evidence of hemorrhagic infarction and focal fat necrosis areas at the section. Microscopic: Omental tissue characterized by acute extensive hemorrhagic infarction and fat necrosis, with polymorph nuclear cells infiltration of vein vessels and focal necrosis areas.The operation was performed on the third day after admittance, in improved metabolic conditions. After a healthy appendix was found, we did inspect the cecum for a perforated diverticulum, the terminal ileum was then examined for Meckel diverticulum and regional enteritis, the pelvic organs were inspected and palpated, gallbladder and duodenum were observed, mesentery was evaluated for mesenteric lymphadenitis, no peritoneal pedicle torsion was found inside the umbilicus. Then we did observe the "omental ball" of 18 × 13 × 6 cm in size, which was suspended by a pedicle twisted on its axis four times (Figure POT is a rare pathological condition which presents with generic symptoms, and may mimic a variety of acute abdominal conditions such as cholecystitis, acute diverticulitis, appendicitis  and MeckPOT is a rare pathological condition with generic symptoms that may mimic many acute abdominal conditions. The pathogenesis of POT has not been established. Anatomical malformations and vascular anomalies are predisposing factors.SOT is a more common condition than POT, due to pre-existing abdominal pathology: cysts, tumours, abdominal inflammatory foci, postsurgical wounds and hernial sacs.The symptoms and the laboratory findings of POT are not specific and mimic other pathological abdominal conditions, for these reasons they make loose time to make diagnosis and provoke increasing degree and duration of OT.The differential diagnosis between POT and SOT is difficult and has seldom been made during the surgical operation. Helpful are US and/or CT scan. MRI can be effective when OT is accompanied by infarction or abscess.Explorative laparotomy represents a diagnostic and definitive therapeutic procedure.Nowadays laparoscopy is the first choice procedure for diagnosis and treatment of acute abdominal torsion.In cases of POT with extensive mass of omentum, diagnostic laparoscopy followed by laparotomy could permit the omental excision with small abdominal incison.The authors declare that they have no competing interests.All authors read and approved the final manuscript.JA drafted the manuscript and participated in the management of patient care. CC carried out a revision of the literature about the topic. OM participated in the management of patient care. MC contributed to write down the manuscript and participated in the management of patient care. NP reviewed the manuscript. DT reviewed the manuscript, carried out the surgery and participated in its design and coordination. All authors read and approved the final draft.The patient knew about this case report and he signed a consent statement. A copy of the written consent was in the patient medical record.


Internal hernia within the falciform ligament is exceedingly rare. A literature search revealed only 14 cases of internal herniation of the small bowel through a congenital defect of the falciform ligament, most of which were found intra-operatively.A 77-year-old Japanese woman presented to our emergency department with sudden hematemesis, occurring at least four to five times over a 12-hour period. No ulcer or gastrointestinal bleeding was detected on gastroendoscopy. A 40mm mass in the inferior lobe of the right lung was found on a chest X-ray, and our patient’s symptoms were therefore initially ascribed to aspirated blood from lung tumor-associated hemoptysis. However, our patient continued to show signs of severe abdominal pain and decreased urine output despite aggressive hydration, leading her examining physicians to search for a possibly severe, occult abdominal pathology. On emergent computed tomography imaging, we found an acute strangulated internal hernia within the falciform ligament. Diagnosis was made by helical computed tomography, permitting rapid surgical intervention.Our findings on computed tomography imaging assisted with the pre-operative diagnosis and enabled us to make a rapid surgical intervention. Early diagnosis may help preclude significant strangulation with unnecessary resection. While a possibility, internal hernia is not a common etiology for small bowel obstruction. Among the differential diagnoses for internal hernia, herniation through a defect in the falciform ligament is one of the most rare findings. A literature search revealed only 14 cases of internal herniation of the small bowel through a congenital defect of the falciform ligament, most of which were found intra-operatively. We report a case of pre-operative diagnosis of herniation through the falciform ligament made by characteristic computed tomography (CT) findings.A 77-year-old Japanese woman presented to our emergency department with hematemesis. As given on the report from her nursing home, she had experienced a sudden onset of vomiting dark red fluid at least four or five times over the prior 12 hours. Her medical history included hypertension for six years, and cerebral hemorrhage three years previously with left-sided paresis and decreased mentation. Her medications included a calcium-channel blocker, an atypical anti-psychotic (sulpiride), and sodium valproate.On physical examination, she was afebrile, had a pulse rate of 117 beats per minute, and a blood pressure of 111/62mmHg. The differential diagnosis included ulcer, gastrointestinal (GI) tumor, angiodysplasia and diverticulitis of the small intestine, enteroaortic fistula associated with dissection or aneurysm, and small bowel ischemia with necrosis. However, no GI bleeding or evidence of ulcer was detected on emergent gastroendoscopy. A primary pulmonary source was also considered, and a chest X-ray revealed a 40mm mass in the inferior lobe of the right lung; our patient’s symptoms were initially ascribed to swallowed and regurgitated blood from lung tumor-associated hemoptysis. However, subsequent serial physical examinations of the abdomen revealed signs of continuing, severe abdominal pain, with our patient grimacing during the examination, though without significant rigidity or guarding. Full characterization of the pain was limited by our patient’s low level of consciousness due to past cerebrovascular accident. In addition to abdominal pain, our patient continued to have decreased urine output despite aggressive hydration, leading us to search for a possibly severe, occult abdominal pathology.Laboratory test results revealed a white blood cell count of 18,000 cells/μL, C-reactive protein level of 9.03mg/dL, lactate dehydrogenase level of 342IU/L, and creatine kinase level of 1324IU/L. With an increasing likelihood of abdominal pathology, emergent CT imaging was ordered. Abdominal helical CT revealed ascites and dilated small bowel. On coronal views, a membranous structure was visualized in the center of the upper abdomen between the diaphragm and the left lobe of the liver; dilated, edematous intestine was seen on the right side of it, while air-dilated intestine was seen on the left side and in the lower abdomen (Figure In addition to ascites, CT demonstrated strangulated intestine with decreased contrast enhancement. We suspected necrosis of the strangulated intestines through the falciform ligament defect. Our patient was therefore taken to the operating room urgently. Abdominal exploration revealed bloody ascites and a gangrenous bowel. Approximately 50cm of small intestine, 2m distal to Treitz’ ligament, was looped over a defect in the falciform ligament. Prolonged strangulation had resulted in massive necrosis of tissue. The round ligament of the liver was cut, opening the falciform ligament and releasing the herniated bowel loop. The necrotic bowel was resected and primary anastomosis was performed. Our patient had a stable post-operative course with no gastrointestinal complications and was subsequently transferred five days after surgery for investigation of lung cancer.The incidence of internal hernias is 0.2% to 2%, and only a proportion of that number cause small bowel obstruction . The typThe etiology of defects in the falciform ligament is diverse, and includes congenital anomalies, trauma, inflammation, and iatrogenic causes. Some reports in the literature note these internal hernias occur after laparoscopic surgery, with defects in the falciform ligament introduced during insertion of the port cannula during laparoscopic fundoplication or cholecystectomy ,8.Congenital defects in the falciform ligament have also been reported. Development of the foregut begins after the respiratory diverticulum, and extends to and includes the hepatocystic diverticulum. It appears at the distal end of the foregut at four weeks of the intra-uterine period. The falciform ligament subsequently develops from the septum between the liver and anterior abdominal wall. Hypoplasia of the falciform ligament gives rise to the noted defect during this process .Internal hernias are more often reported in early childhood or late pregnancy. In late pregnancy, the gravid uterus expands, pressing the small bowel into to the upper abdomen, allowing relatively easy passage through congenital falciform ligament defects . In our Abdominal helical CT imaging demonstrated several specific findings. First, marked differences in both size and edema of the right and left portions of intestine, divided by membranous stricture, were identified from the abdominal wall to the point of the liver dividing laterally from internal segments. The point of stricture of the falciform ligament was visualized clearly as edematous intestines had compressed onto the anterior liver, and this represents an important visual finding in falciform ligament herniation. If a defect in the falciform ligament is caudal to the liver, it will not be readily identifiable. However, in such cases, the diagnosis may still be made by recognizing the constriction of intestine directly under the abdominal wall and directly in the medial axis of the trunk. These specific CT findings, along with a high index of suspicion for this disease entity in cases of unexplained abdominal pain, may aid in diagnosis of herniation through the falciform ligament, enabling more rapid pre-operative diagnosis. Though this diagnosis remains rare, the above clinical presentation and imaging should alert physicians to its possibility.We present a rare case of strangulated small bowel due to internal hernia through a congenital defect in the falciform ligament. Our findings on CT imaging assisted with the pre-operative diagnosis and enabled us to rapidly obtain surgical intervention. Early diagnosis may help preclude significant strangulation with unnecessary resection.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.HS interpreted the data from our patient and was directly involved in the full implementation of this report. SS was jointly involved in the surgical intervention and was a major contributor in writing the manuscript. All listed authors were involved in writing and editing the manuscript, and all authors read and approved the final manuscript.


Malignant lymphomas may originate from any area of the body and cause a variety of symptoms. However, a malignant lymphoma causing urinary symptoms is uncommon. We report a unique case of a 77-year-old woman who presented with a persistent pollakiuria. Radiographic imaging showed a large pelvic mass (13 × 13 × 11 cm) remarkably compressing and invading the bladder wall and accompanied with bilateral hydronephrosis. Urinary cytology revealed malignant lymphoma, and a final diagnosis of malignant lymphoma was made on the basis of transvaginal needle biopsy. Urinary cytology facilitated the definite diagnosis, following which we initiated a rapid and successful treatment with cyclophosphamide, doxorubicin, vincristine, and prednisone with rituximab. Malignant lymphoma (ML) may originate from any area in the body and often causes symptoms such as fever, drenching night sweats, and weight loss. However, the lower urinary tract is an unusual site for early ML manifestations, and patients with ML rarely present with urological symptoms . Herein, A 77-year-old woman was referred to our department, because of increased urinary frequency over the previous month, which had not improved despite the administration of an oral antimuscarinic agent. She lost 3 kg over two months, which was approximately a 5% loss in her body weight. However, she did not report a fever or drenching night sweats. On physical examination, a palpable lower abdominal mass (10 × 15 cm) with elastic consistency, a smooth surface, and mobility, was detected. Palpable lymphadenopathy was not significant. Laboratory data showed slightly elevated levels of serum creatinine , C-reactive protein , and lactate dehydrogenase . Urinalysis results were close to normal. Abdominal computed tomography (CT) revealed a lobulated mass (13 × 13 × 11 cm) in the pelvis, adjacent to the left pelvic wall. The mass compressed the urinary bladder anteriorly, possibly invaded the posterior bladder wall, and resulted in bilateral hydronephrosis. No apparent lymphadenopathy or metastasis was revealed. Magnetic resonance imaging (MRI) showed a tumor demonstrating hypointensity on T1-weighted and hyperintensity on T2-weighted imaging. MRI also demonstrated the tumor invasion of the bladder wall . These fClinically, urogenital secondary involvement of ML is less frequent, especially involvement of the bladder. Watson et al. described the following classification for secondary vesical lymphoma: (1) circumscribed single or multiple foci limited to the bladder wall, 2) direct invasion of the bladder by a perivesical tumor, and 3) vesical extension from prostatic foci [ direct i vesical Urinary frequency as a symptom of secondary ML of the bladder is uncommon, excluding gross bladder involvement. In 72 patients with microscopic vesical infiltration, 10 patients reported vesical symptoms during their lifetime, which occurred relatively late in the course of the disease . In the Urinary cytology is a diagnostic method to detect urinary tract malignancies and may be useful to diagnose primary or secondary ML of the urinary tract. In a report of 50 consecutive patients with ML, 14 (28%) had positive cytological findings , and it It remains controversial whether a bulky disease influences the efficacy of R-CHOP, a standard treatment for DLBCL. In some reports, bulky diseases were not prognostic factors in subgroup analyses , 9. In c


Pulmonary benign metastasizing leiomyoma characterized by the growth of uterine leiomyoma in the lung is a very rare disease. We herein report the case of a 46-year-old asymptomatic woman who underwent a total abdominal hysterectomy for her multiple uterine leiomyomas 5 years ago, with the presence of multiple shadows in her chest roentgenogram during the regular check-up. Chest computerized tomography (CT) showed multiple solitary nodules in both lungs. Video-assisted thoracoscopic surgery with a wedge resection of the lesion was performed. Histopathologically, the pulmonary nodule was composed of benign smooth muscle cells and demonstrated low mitotic activity and absence of necrosis. Immunohistochemical staining for smooth muscle actin (SMA) and Desmin were extremely positive. CD10, CD117 and S-100 were negative in the tumor cells. Positive immunoreactivity for estrogen receptor (ER) and progesterone receptor (PR) were detected. The pathological diagnosis was pulmonary benign metastasizing leiomyoma. Pulmonary benign metastasizing leiomyoma is a very rare disease characterized by the growth of uterine leiomyoma tissue in the lung . In mostA 46-year-old Chinese woman, who was a nonsmoker, was admitted for further examination of scattered shadows in both lungs that were found incidentally on the chest roentgenogram during a regular checkup in 2011. A chest computerized tomography (CT) was subsequently performed. The patient’s past medical history showed a diagnosis of multiple uterine leiomyoma in 2005 for which a total abdominal hysterectomy had been performed. The pathology revealed a uterine smooth muscle tumor with vascular invasion. During the 5-year follow-up, no recurrence of the benign metastasizing leiomyoma was detected. The clinical examination was unremarkable, and the results from routine laboratory examinations were normal. CT demonstrated multiple solitary nodules in both lungs with maximum diameters of 2 cm Figure . PositroIn August 2011, the patient underwent right video-assisted thoracoscopic surgery with wedge resection of the lesion. One of the lesions from the right lower lobe was resected for pathological diagnosis. There were multiple small nodules in the right lower lobe, which were purplish red, soft and smooth, with clear demarcations. Intraoperative pathological examination (frozen pathology) revealed a benign leiomyoma. Postoperative pathological examination showed a pulmonary benign metastasizing leiomyoma, consisting of revealed spindle-shaped cells with abundant clear cytoplasm and distinct cell borders. It demonstrated low mitotic activity (<0-1/10 high-power fields), little cytological cellular atypia and no tumor cell necrosis Figure . ImmunohThe patient was discharged from the hospital without any early postoperative complications. She started taking mifepristone (12.5 mg daily) and receiving Zoladex injections (3.6 mg every 4 weeks). Her current general condition was satisfactory without any radiological evidence of recurrent disease or distant metastases. The remaining lesions in both lungs were stable during the follow-up period.Benign metastasizing leiomyoma is a very rare disease in which tissue from a benign uterine leiomyoma is detected as a solitary nodule or as multiple nodules in the lungs of patients with a previous history of hysterectomy for uterine leiomyoma. In 1939, Steiner et al. reported the first case in which a leiomyoma transferred from the uterus to the lung, showing a benign histological appearance. It was called “metastasizing fibroleiomyoma” [Most patients with benign metastasizing leiomyoma are asymptomatic. It is usually found during physical examinations. Abramson et al. reported that the average age of patients with benign metastasizing leiomyoma is 48 years old, with the period from hysterectomy to nodule detection varying from 3 months to 26 years .The nature and etiology of benign metastasizing leiomyoma are still controversial. Uterine leiomyoma has been reported to depend on hormones ,6. EstroMaking the diagnosis of benign metastasizing leiomyoma is very difficult, and it has strict pathological diagnostic criteria. It is based on the results of the immunohistochemical staining and the history of hysterectomy for uterine leiomyoma. In our case the pathological examination showed spindle-shaped cells without mitotic activity or nuclear atypia. The tumor cells are strongly positive for SMA, confirming the smooth muscle origin of this benign tumor. In addition to these features, immunohistochemical staining is negative for CD117, ruling out an extra-gastrointestinal stromal tumor, and all of the cells are negative for S100, essentially excluding a tumor of neural origin. Positive immunoreactivity for estrogen receptor (ER) and progesterone receptor (PR) suggests the spindle-shaped cells are uterine smooth muscle cells.There are currently no standard treatment guidelines for benign metastasizing leiomyoma. Reported treatment modalities include careful observation, surgical resection, hysterectomy and bilateral oophorectomy, progestins, aromatase inhibitor and medical castration using luteinizing hormone-releasing hormone analogs  Table 11. HoweveAlthough BML is a very rare condition, it should be considered in asymptomatic women of reproductive age with a history of uterine leiomyoma who present with solitary or multiple pulmonary nodules of the lung. Long-term close surveillance is required for patients with BML for early detection of recurrence or distant metastases.Written consent was obtained from the patient for the study and publication of this case report and accompanying images. A copy of written consent is available for review from the Editor-in-Chief.The authors declare that they have no competing interests.YF collected information and prepared the original draft. HL researched the relevant literature and revised the draft. BT helped with the literature research and preparing the manuscript. BH helped prepare the manuscript. All authors read and approved the final manuscript.


Providing a more comfortable life and a longer survival for pancreatic corpus/tail tumors without metastasis depends on the complete resection. Recently, distal pancreatectomy with celiac axis resection was reported as a feasible and favorable method in selected pancreatic corpus/tail tumors which had invaded the celiac axis. Additional organ resections to the celiac axis were rarely required, and when necessary it was included only a single extra organ resection such as adrenal or intestine. Here, we described a distal pancreatic tumor invading most of the neighboring organs—stomach, celiac axis, left renal vein, left adrenal gland, and splenic flexure were treated by en bloc resection of all these organs. The patient was a 60-year-old man without any severe medical comorbidities. Postoperative course of the patient was uneventful, and he was discharged on postoperative day eight without any complication. Histopathology and stage of the tumor were adenocarcinoma and T4 N1 M0, respectively. Preoperative back pain of the patient was completely relieved in the postoperative period. As a result, celiac axis resection for pancreatic cancer is an extensive surgery, and a combined en masse resection of the invaded neighboring organs is a more extensive surgery than the celiac axis resection alone. This more extensive surgery is safe and feasible for selected patients with pancreatic cancer. Unlike pancreatic head tumors, distal pancreas tumors rarely cause jaundice. Mostly these lesions cause upper abdominal pain radiating to the low back as a result of local invasion to the celiac axis. Left-sided pancreatic tumors are usually delayed in diagnosis because of the difficulties at the differential diagnosis of low back pain which is very frequent in elderly people. Therefore, liver metastasis, peritoneal carcinomatosis, or major vascular invasion to celiac axis or superior mesenteric artery is frequent at the time of diagnosis and up to 75% of the tumors are evaluated as unresectable –6. Provitransvers mesocolon—which was treated by en bloc resection of all these organs. Recently, distal pancreatectomy with celiac axis resection was reported as a feasible and favorable method in selected pancreatic corpus/tail tumors [Our aim was to present a case of a distal pancreatic tumor invading most of the neighboring organs—stomach, celiac axis, left renal vein, left adrenal gland, and l tumors , 4, 6, 7A 60-year-old male patient was admitted with complaints of low back pain and loss of appetite. He had a history of several diagnostic and therapeutic interventions for low back pain. At last, an abdominal computed tomography revealed a solid lesion (5 × 3.5 cm) at the pancreatic corpus and tail, which had indistinguishable fatty interplains with superior mesenteric artery . Right hOn postoperative day one, the patient was extubated, and the nasogastric tube was removed. During the whole postoperative course liver enzyme levels were always within normal range (al range . PancreaHistopathological examination reported as pancreatic ductal adenocarcinoma with an extensively invaded peri-pancreatic tissue with perineural, lymphatic, and venous invasion. It had invaded adrenal gland, stomach wall, mesocolon, and renal vein. There was no histopathologically confirmed celiac axis and splenic tumor invasions, but there were fibrotic adhesions to the celiac axis. Of 29 lymph nodes resected, two had tumor metastasis; among nine lymph nodes resected around superior mesenteric artery, none of them had metastasis. Following medical oncology consultation, the patient was given chemotherapy, and after that he was planed for reversal of end colostomy. His back pain was completely relieved in the postoperative period. While this paper was prepared, the patient was at the end of the third month of his followup. One of the aims of preoperative radiological evaluation of an invasive intra-abdominal cancer is to identify patients with unresectable or incurable advanced disease in order to prevent unnecessary laparotomy. Our previous studies have demonstrated that preoperative radiological evaluation sometimes may be inadequate for determining tumour invasion of the main vasculature and for detecting peritoneal metastasis. Patients who are considered fit to undergo tumour resection should not be denied operative assessment on the basis of radiological findings of adjacent organ involvement, except in cases of large circular mass invasion. Since there is a high rate of false positives and false negatives for tumoural invasion on radiological evaluation, there may be a risk of undertreating patients who are actually operable , 9. PreoDuring surgery, we could dissect out the tumor from the superior mesenteric artery without leaving any affected tissue around the artery. However, celiac resection was performed due to the adherence of the tumor there. Celiac resection was first described by Graham et al. which was performed on a patient who underwent gastrectomy for gastric tumor . Later, In our case, the tumor had invaded the gastric wall posteriorly. Initially, gastric wedge resection was performed, however, almost complete ischemia following celiac axis resection led to high-subtotal gastrectomy. Various studies reported similar to our case that gastric ischemia may develop following celiac axis resection and some stated that left gastric artery might be reanastomosed to celiac stump for prevention of gastric ischemia . In our During the postoperative followup, the patient described that his back pain was reduced markedly. Celiac axis resection was reported to provide such a pain relief , 17. We


Ossifying fibroma is a rare benign tumor of the nasal cavity and the paranasal sinus, and is easily misdiagnosed. In the present study, we report the clinical data in the case of a 46-year-old female patient, treated due to 5-day forehead swelling accompanied by dizziness. CT examination revealed dilation of the right frontal sinus, bone wall integration, dense masses in the cavity, multiple punctate calcification foci internally and no nasal obstruction, nasal mucus or epistaxis. After hospitalization, a right frontal sinus fenestration and tumor resection plus nasofrontal duct reconstruction combined with nasal endoscopic frontal recess open surgery was conducted under general anesthesia. Following the tumor resection, the frontal sinus bone lamella was reset and fixed with a titanium bone fixation set. The postoperative pathological diagnosis was of ossifying fibroma. At the postoperative 5-year follow-up there was no tumor recurrence and nasal endoscopy revealed an unobstructed nasofrontal duct opening. Ossifying fibroma is a rare benign tumor of the nasal cavity and paranasal sinus, and is easily misdiagnosed. Since the initiation of nasal endoscopies in the Department of Otolaryngology  we have treated two cases of ossifying fibroma in the nasal cavity and paranasal sinus, confirmed by pathology, over a period of 5 years (2003–2008). Of the two, only one case has complete clinical data. In the current study we summarize the clinical and pathological data and follow-up results of this case. The study was approved by the ethics committee of Gongli Hospital. Informed consent was obtained from the patient and the patient’s family.A 46-year-old female patient was treated due to 5 days of forehead swelling accompanied by dizziness. The patient received a head CT in the Neurology Department, which revealed a right frontal sinus lesion , and was subsequently transferred to the Department of Otolaryngology, Gongli Hospital for treatment. The patient was hospitalized on April 17, 2007. From the onset, she had no vertigo, apparent nasal obstruction, nasal mucus, epistaxis, hyposmia or headache. Furthermore, she denied any history of trauma to the head or face.At the admission examination, the systemic signs were good and the external nasal shape was normal. Right middle meatus mucosal edema was visible under the anterior rhinoscope but there were no newplasm or purulent secretions in the nasal cavity. The right part of the forehead bulged slightly, the skin was normal, there was no local tenderness or fluctuation, the position of the right eyeball was slightly lower than that of the left eyeball but eye movements, visual acuity and visual field were all normal. CT scans revealed that the right frontal sinus had dilated, the bone wall was integrated, dense masses were present inside the cavity and there were multiple punctate calcification foci internally. Furthermore, the boundary was clear, there were shadows of bone-like density, the superior wall of the eye socket had moved down under stress, and sclerotin was attenuated due to mild absorption .On the 4th day of hospitalization, a right frontal sinus fenestration and tumor resection plus nasofrontal duct reconstruction combined with nasal endoscopic frontal recess open surgery was performed under general anesthesia. During the surgery, a curved incision was made 0.5 cm from the right side of the inner canthus outwards to the interior superciliary arch, and a bone window of ∼4×2 cm was prepared at the anterior wall of the frontal sinus in order to expose the frontal sinus cavity. It was observed that the sinus was filled with a solid mass, ∼4.5×4×0.6 cm in size (grey-white block mass) and sand-like tissues. The tumor had capsule and like bone, and the boundaries were clear. Following complete dissection, the surrounding bone wall presented stalactite-like changes in appearance, and the surface was rough. At the frontal sinus opening, there was a bony nodular prominence (0.5×1 cm), the opening of the frontal sinus at the sinus cavity surface was completely affected by bony atresia. An electric drill was used to grind the bony prominence in the sinus cavity, and the anterior ethmoid sinus and frontal recess were opened using a nasal endoscope. From the bottom wall of the frontal sinus, sclerotin was gradually ground downwards along the normal nasofrontal duct. Subsequently, the nasofrontal duct was reconstructed. A silicone tube was placed into the frontal sinus, led out from the anterior naris though the nasal cavity and fixed. The frontal sinus bone lamella was reset and fixed with a titanium bone fixation set, and the frontal incision was sutured layer by layer. A pressure dressing was applied and the anterior ethmoid sinus cavity was filled with calcium alginate gauze . There wAfter half a month, reexamination revealed that the frontal sinus drainage tube in the nasal cavity was unobstructed and the frontal sinus was rinsed. After one month, the frontal sinus drainage tube was removed, and nasal endoscopy was performed, revealing that the nasofrontal duct opening was unobstructed. At the 5-year postoperative follow up, there was no tumor recurrence. Nasal endoscopy 1 year postoperatively revealed an unobstructed nasofrontal duct opening . The 1 ySince the first case of ossifying fibroma was reported in 1872, there was a period of time when ossifying fibroma and fibrous dysplasia of bone were considered to be the same disease . The frontal bone, ethmoid bone and sphenoid bone may all be affected. Occasionally, the occipital and temporal bones have been affected . Ossifyilescence . The caulescence  and trauThe clinical symptoms of this disease have no marked specificity, and early detection is difficult. The patient in this study was 46 years old, and the lesion had possibly occurred earlier. Due to the lack of clear clinical symptoms, she had not visited a doctor. Her clinical manifestations were mainly dizziness, local swelling and eyeball dislocation and she had no nasal symptoms. Therefore, the diagnosis was missed by doctors in the otorhinolaryngology department. The case was initially diagnosed in the neurology department due to the dizziness symptom.The diagnosis of the case was dependent on paranasal sinus CT and pathological examination. As a routine means of examination, paranasal sinus CT assists diagnosis and defines the lesion range for preparing a surgical intervention, and it should act as a routine presurgical examination. CT scanning images of the nasal cavity and paranasal sinus present round or oval high-density shadows, where the density is uniform. If there is bleeding or a cystic lesion in the tumor, partial areas present as low density shadows, there are uneven-thickness bone shells on the surface and the boundary is clear. Additionally, pathological examination is critical for a definitive diagnosis. Pathological examination reveals a clear boundary, a fibro-osseous lesion and sand-like material distributed in the fibrous stroma. These are always the main features of ossifying fibroma. A preoperative biopsy is difficult to perform, as it is essential to cut open the mucus in order to take out the central lesion of the tumor. However, this method may easily induce the enclosed mass to bleed. It is recommended that a biopsy is not conducted prior to surgery. Furthermore, it is necessary to differentiate ossifying fibroma from fibrous dysplasia of the bone. A CT of the latter generally shows a more even mass density and the boundary is not clear, giving an appearance more similar to that of frosted glass. Pathological characteristics include irregular bone trabeculae, disorganized and crisscrossed collagen fibers and a large number of osteoclasts in the bone matrix, and no definite boundary between the lesion and the surrounding tissues. Some investigators are of the opinion that histochemistry and immunohistochemistry may aide the identification of these characteristics.Ossifying fibroma in the nasal cavity and paranasal sinus is a benign tumor histologically, but in the clinic it has invasive characteristics. It usually invades the eye socket, the base of the skulland calvarium to induce symptoms in the eye and brain, and its malignancy rate is 0.4–0.5% .The previous literature, as well as the findings from the current case, suggest that the prognosis of this case is good. There was no tumor recurrence following the complete resection of the tumor. Recurrence mostly occurs due to incomplete surgery, and following recurrence, the only solution is to perform a complete resection of the tumor again. Radiotherapy and chemotherapy are invalid for this disease, as radiotherapy may promote a malignant transformation resulting in the formation of an osteosarcoma .


Penile metastases are rare and usually secondary to genitourinary and colorectal cancer.We present a case of a 77-year-old man with penile metastasis who was operated for rectal carcinoma. He was referred to our clinic for penile ulcerous lesion, semierectile penis and voiding dysfunction. Imaging studies showed nodular lesion at glans penis and multiple bone metastases. He did not respond to chemoradiotherapy and he had bad prognosis.Imaging methods and biopsy may help to clarify the diagnosis but the treatment modalities are insufficient in these patients. The metastatic involvement of the penis is extremely rare, despite rich vascularisation between the penis and the neighbouring organs. The first report of secondary penile malignancy from an adenocarcinoma of the rectum was defined by Eberth.In this article, we report a case of a penile metastasis secondary to a rectal carcinoma three years after the surgery.A 78-year-old male presented in March 2002 with a 2 months history of change in bowel habit and rectal bleeding. The examination revealed a tumour of the lower rectum. An abdomino-perineal excision was performed and the pathological examination revealed a Dukes C undifferentiated adenocarcinoma with the lymph nodes involvement (15/22). He had received adjuvant chemo-radiotherapy postoperatively. He remained well until 2 years later, when he presented with urogenital complaints: penile ulcerous lesion, semierectile penis and voiding dysfunction.A follow-up examination was performed in April 2005 following the development of indurations, hyperaemia, and oedema of the penis and demonstrated 0.5 cm and 0.3 cm ulcerative lesions on glans penis . The exa3 / μL , anaemia (with a haemoglobin of 10.2 g/dL), normal level of PSA but increased levels of CEA  and CA19.9 . Penile US was normal for corpus spongiosum and cavernosus. CT demonstrated metastases of several thoracic vertebras and sacroiliac joint. Furthermore, MRI of the penis showed multiple pathological focuses in T2 signal and a nodular lesion that measured 15 mm in size, occupying the glans penis (2 i.v. day 1) followed by leucovorin (500 mg/m2 i.v day 1) and 5-fluorouracil (2300 mg/m2 continuous infusion day 1). Two days after chemotherapy administration of the third cycle the patient was admitted to our department with fatigue, weakness, dysuria and hypotension. Increased level of BUN, creatinin and decreased level of blood count were registered. Despite the whole blood transfusion, the values of blood count decreased (down to 5.4 g/L) during hospitalization. He was admitted for further medical therapy but during this admission his general condition deteriorated and he died with progressive disease.A complete blood cell count showed an increased white blood cell count of 16 x 10ns penis ,3. The pOverall it appeared that this patient had a disseminated metastatic rectal carcinoma, with an unusual settled for the penis.The penile metastasis in the course of rectal cancer is an unexpected complication, despite its proximity to the rectum and its rich vascularity. Recently published studies showed that the penile metastases are associated with prostate, urinary bladder cancer, and infrequently with rectal carcinoma.1The most common symptoms and signs are dysuria, voiding dysfunction, perineal pain, priapism, penile nodules and mass.We believe that it would be beneficial in the differential diagnosis to take into consideration the findings related to voiding dysfunction, priapism and ulcerous lesions of the penis. Peyronie’s disease, traumatic and infectious skin and syphilitic lesions or primary carcinoma of penis, must also be in the differential diagnosis of the penile metastasis.7corpus cavernosum on T1-weighted images, and low signal intensity against the high background intensity of the cavernous bodies on T2-weighted imaging.Tissue tumour markers: AFP, CEA, HCG can be used for differential diagnosis. Penile US and CT is the first choice for imaging but its sensitivity is limited. Cavernosogram was reported a preferable application in some patients.Treatment options are palliative with local surgery, systemic chemotherapy and local radiotherapy.10In conclusion, penile metastases are rarely seen as a form of metastasis among rectal cancer patients. Imaging methods may help to clarify the diagnosis, but penile lesions should be biopsied to confirm the diagnosis. On the other hand, the treatment modalities are insufficient for a long survey and quality of life in these patients.


Dienogest is a novel synthesized progestin used for treatment of endometriosis. This is the first case report describing a therapeutic effect of dienogest on a gynecologic malignancy. The patient was a 44-year-old woman with advanced adenosarcoma arising from the endometriosis in the rectovaginal space and infiltrating the left pelvic wall, left ureter, rectum and vagina. The residual tumor after tumor debulking surgery was resistant to both chemotherapy and radiotherapy. Dienogest was used as a substitute for medroxyprogesterone acetate because of the presence of deep vein thrombosis. Based on the RECIST criteria, partial response was obtained with oral dienogest therapy at six months and the serum CA125 level also decreased from 70 U/ml to 16 U/ml. The tumor remained stable up to 21 months. Thromboembolism or other adverse effects did not occur during the dienogest therapy. Dienogest may be useful for the treatment of adenosarcoma arising from endometriosis. Müllerian adenosarcoma is a rare tumor characterized by a benign epithelial component and a sarcomatous stromal component , a synthesized progestin, has a therapeutic effect on gynecological malignancies such as adenosarcoma  and magnetic resonance imaging (MRI) showed a polymorphic mass in the pelvis. A major portion of the mass formed a 5-cm cystic lesion in the pouch of Douglas, infiltrating to the left acetabular, left ureter and rectum Figure A. The re2 day1, CDDP:20mg/m2 day1-day4; 4 week intervals). After three cycles of chemotherapy, an MRI revealed that the residual tumor appeared to be stable disease (SD) by RECIST. The CA125 was 24.7 IU/ml. Subsequently, she received cisplatin-based chemoradiotherapy . However, a follow-up MRI after CCRT demonstrated progressive disease by RECIST and the CA125 again rose to 70 U/ml. Therefore, the patient commenced oral DNG therapy at a dose of 2 mg daily. Written informed consent for DNG therapy was obtained from the patient. Based on the RECIST criteria, a partial response was obtained at six months after initiating oral DNG treatment  than at extraovarian sites of endometriosis (1.6%)  to activate or repress gene expression in target cells. Synthetic progestines are structurally classified into two major chemical classes  is a novel ‘hybrid progestogen’ that has pharmacodynamic properties typical of the two main classes of progestogens (Ruan et al. In the presented case, the adenosarcoma underwent very rapid growth. In addition, this aggressive tumor was insensitive to either chemotherapy or radiotherapy. Several case reports have suggested that MPA may be effective in the treatment of adenosarcoma; however, MPA was contraindicated in the present case because she had deep vein thrombosis. Therefore, DNG was used as a substitute for MPA for progesterone therapy. A successful partial response was obtained at six months after initiating oral DNG treatment and the tumor was stable up to 21 months.The rapid regrowth of the tumor following a long-term stable status may have been due to the reduction of ER and PR expression via histological evolution to sarcomatous overgrowth. In previous reports of adenosarcoma, successful treatment with MPA was demonstrated only for ER- and PR-positive tumors (Hines et al. In conclusion, we experienced a case of advanced adenosarcoma that was treated with DNG. To the best of our knowledge, this is the first case report describing a therapeutic effect of DNG on a gynecologic malignancy. In this case, no adverse effects, including thromboembolism, were observed. DNG may be useful in the treatment of gynecologic malignancies, such as adenosarcoma, low-grade endometrial stromal sarcoma and grade 1 endometrial carcinoma, even for women who are at risk of thromboembolism.Oral informed consent was obtained from the patient for the publication of this report and any accompanying images before her death. Written informed consent was obtained from the family of the patient for the publication of this report and any accompanying images.


Metastasis from renal cell carcinoma (RCC) to the testis is rare. This case report presented an extremely rare case of simultaneous bilateral testicular metastases from RCC in a 65-year-old man who had experienced indolent scrotal enlargement over a period of several months. Scrotal ultrasonography showed 4.0- and 2.0-cm-sized masses in the left and right testes, respectively. Contrast-enhanced computed tomography identified multiple tumors in the kidneys, the pancreas and the left adrenal gland. Left orchiectomy and pathological examination were performed and indicated testicular metastasis from clear cell RCC. The patient underwent complete surgical resection of all residual lesions. Postoperative follow-up examination without adjuvant therapy identified no recurrence over 11 months. This study also reviewed existing literature and determined that retrograde venous spread from the primary kidney tumor to the testis may be an important pathway for testicular metastasis from RCC. In conclusion, RCC can result in testicular metastases not only unilaterally, but also bilaterally, as was observed in the present case. Physical examination revealed a stony, hard, hen’s egg-sized mass in the left scrotum. A normal-sized testis with a small nodule was observed in the contralateral scrotum. Superficial lymph nodes were not palpable. Serum levels of α-fetoprotein, β-human chorionic gonadotropin and soluble interleukin-2 receptor were all within normal limits. Scrotal ultrasonography revealed a 4.0×3.3-cm mass in the left testis and a 2.0×1.7-cm mass in the right testis. Contrast-enhanced computed tomography of the abdomen identified multiple tumors in the two kidneys, the pancreas and the left adrenal gland, in addition to the testes . ImagingWe performed left orchiectomy for pathological diagnosis. The resected testis contained a yellowish-white tumor with clear margins accompanied by parenchymal hemorrhage. Pathological examination revealed that the tumor cells had small, slightly oval nuclei with optically clear cytoplasm and were arranged in nests separated by a rich network of sinusoidal vascular channels . These rAll disseminated tumors were surgically resectable and the patient’s general condition was good. Therefore, the patient underwent partial pancreatectomy, left adrenalectomy and left partial nephrectomy, followed by right radical nephrectomy and right partial orchiectomy. Complete surgical resection was achieved. The pathological findings of the resected tumors were compatible with metastases from the right RCC . Postoperative follow-up examination without adjuvant therapy showed no recurrence for 11 months. The patient provided written informed consent.Secondary neoplasms of the testis are rare with a reported incidence of testicular metastasis of 0.02%  and 0.06Although RCC commonly results in metastases to various organs, it rarely spreads to the testes. The testes are regarded as a ‘tumor sanctuary’, as it has been hypothesized that tumor cells are not able to grow easily in that environment. The relatively low temperature of the scrotum could provide unacceptable conditions for the establishment of metastatic tumor cells . AdditioThis study also searched previous medical literature using the Medline/PubMed databases and identified 30 reported cases of unilateral testicular metastasis from RCC, excluding autopsy cases . Of the In conclusion, to the best of our knowledge, this study was the first to present an extremely rare case of simultaneous bilateral testicular metastases from RCC. Following a review of the current literature, ipsilateral testicular metastasis from RCC is more frequent and, thus, retrograde venous spread via the spermatic vein may be one of the main pathways of testicular metastasis from RCC. As demonstrated in this case, RCC can result in testicular metastasis, not only unilaterally but also bilaterally.


Spigelian hernias are rare hernias, representing only about 1-2% of all abdominal hernias. An accurate preoperative diagnosis of this condition is often difficult because the physical presence of these hernias is often not demonstrable owing to its peculiar anatomic location. Many patients with Spigelian hernias, therefore, may have no obvious findings on clinical examination. The diagnosis is even trickier in obese patients wherein the hernia sac may lie in an intraparietal plane, masked by the abdominal subcutaneous fat. Here we describe a case of incarcerated Spigelian hernia where clinical and radiological findings were inconsistent and the accurate diagnosis was made by laparoscopy and was followed by a laparoscopic-assisted repair using an open anterior approach with an onlay mesh. We therefore feel that laparoscopy can be a useful tool for confirming the diagnosis and also for performing a definitive repair in doubtful cases of Spigelian hernias. The Spigelian hernia (SH) is named after the Belgian anatomist Adriaen Van Den Spigehel who described the semilunar line (linea spigeli) in 1645 . A SpigeA 45-year-old-woman presented with a history of sudden onset of right-sided lower abdominal pain since five hours and was associated with vomiting. She gave a history of recurrent episodes of right-sided lower abdominal pain since the past 2 years and several imaging studies done over this time period, including ultrasonography and computed tomography scans, failed to reveal the etiology of her symptoms. She had no previous surgical history, but was recently detected to have type 2 Diabetes Mellitus. Clinical examination revealed an obese abdomen with maximal focal tenderness in the right lower quadrant. However, a vague fullness was palpable in the lower right quadrant which corresponded to the site of maximal tenderness. She was afebrile, the heart rate was 90/minute, blood pressure was 140/90 mm of Hg, and the bowel sounds were hyperperistaltic. Laboratory investigations were unremarkable except for neutrophilic leucocytosis. Plain abdominal radiograph and ultrasonography were inconclusive. Hence, with a provisional clinical diagnosis of an appendicular pathology, the patient was taken-up for laparoscopic surgery.Prior to the induction of anaesthesia, the patient's abdomen was palpated and the area of maximal focal tenderness and the fullness was outlined with a marking pen . PneumopThe exact etiology of Spigelian hernia is uncertain. It could be either congenital or acquired . The conThe accurate diagnosis of SH is often difficult and can be attributed to its rarity and the absence of classical symptoms coupled with a lack of personal clinical familiarity. It can simulate a variety of other commoner lower-quadrant abdominal diseases. Only about 50% of cases are diagnosed preoperatively . The anaRadiological investigations, such as ultrasonography and Computed Tomography (CT) scans may help diagnose incarcerated Spigelian hernias. Papierniak et al. have demonstrated the usefulness of computerized tomography in diagnosing Spigelian hernias . Yet in Spigelian hernias have conventionally been repaired with an open surgical technique. Zacharakis et al.  have shoIn our case, the diagnosis of SH was made laparoscopically. The clinical examination revealed only a tender area with a vague fullness and no palpable mass. This was probably because the hernial sac was small and was masked by an apron of subcutaneous fat. Moreover, as the hernia was incarcerated, there was no demonstrable expansile impulse on coughing. The defect could be clearly visualised with the help of the laparoscope and the sac could also be identified intraabdominally and isolated extraperitoneally. The hernioplasty was done by an anterior approach using a polypropylene mesh similar to an open technique and was therefore cost-effective. The immediate integrity and adequacy of the repair could be checked laparoscopically after insufflating the peritoneal cavity with air. However, we do feel that some of the reported benefits of a totally laparoscopic hernia repair could not be obtained in this case.In conclusion, the diagnosis of Spigelian hernias is not always straightforward, especially when a mass is not palpable. Laparoscopy can be a useful tool to confirm the diagnosis of Spigelian hernias especially when clinical and radiological findings may be equivocal. Laparoscopy can help delineate the exact site of the defect in the anterior abdominal wall and also help identify the sac. A laparoscopic-assisted technique, like in this case, may be cost-effective especially in rural hospitals.This paper has not been published in its current form or a substantially similar form , that it has not been accepted for publication by another journal and that it is not under consideration by another publication. One of the authors hereby transfers, assigns, or otherwise conveys all copyright ownership, including any and all rights incidental thereto, exclusively to the journal, in the event that such work is published by the journal. The authors have no conflict of interests. The patients consent has been obtained for publication. The entire paper was conceived, designed, and drafted by Dr. Sanoop K Z, and Dr. Priya J was involved in revision and drafting the final paper.


Introduction. A glomus tumor is a benign vascular tumor derived from glomus cells and occurs mainly in the subcutaneous layer of the subungual or digital pulp. Extradigital glomus tumors have been reported within the palm, wrist, forearm, foot, bone, stomach, colon, cervix, and mesentery. Glomus tumors can originate from the intraosseous, intramuscular, periosteal, intravascular, and intraneural layers. However, a glomus tumor originating from the intravascular layer of the forearm is a rare condition. Case Report. A 44-year-old woman had a 7-year history of chronic pain and focal tenderness of the forearm. No hypersensitivity or sensory alterations were observed. Contrast magnetic resonance imaging (MRI) showed a mass measuring 5 × 3 × 2 mm leading to a vein. Surgical excision was performed, and the tumor was completely resected. Finding of gross examination revealed a dark-red, well-defined soft tissue tumor, and histologic examination confirmed that the mass was a glomus tumor. The patient's symptoms were completely resolved postoperatively. Conclusion. Intravascular glomus tumors rarely occur in the forearm; therefore, a thorough physical exam, comprehensive medical history, in-depth imaging, and early surgical excision upon clinical suspicion may be helpful to prevent a delayed or incorrect diagnosis. The normal glomus body is a specialized arteriovenous anastomosis that plays a role in thermoregulation . A glomuA 44-year-old woman presented with a 7-year history of occasional pain on the volar-radial aspect of her right mid-forearm. The patient reported that the pain had gradually worsened and was significantly increased on palpation. She recalled no traumatic event and reported no relevant family history. On physical examination, “spark-like” pain was caused by touching a well-localized area, but no sensory alterations or temperature hypersensitivity was observed. No contour deformities or visible palpable skin lesions were identified.Laboratory examinations, plain radiographs, and computed tomography (CT) revealed normal findings. Magnetic resonance imaging (MRI) with contrast enhancement demonstrated an oval-shaped subcutaneous mass leading to a small vessel. The size of the mass was determined to be 5 × 3 × 2 mm on MRI. The mass had intermediate intensity on T1-weighted images and displayed an inhomogeneous high signal on T2-weighted images and homogenous strong enhancement with contrast .During the operation, a 3 cm longitudinal incision was made on the radial aspect of the forearm at the site of tenderness. A dark-red, well-defined, and oval-shaped mass  was identified in a branch of the cephalic vein . The masAfter complete surgical excision, the patient's symptoms fully resolved within days. There was no recurrence of pain up to 6 months postoperatively.The normal glomus body is involved in thermal regulation through control of the skin circulation , 8. It iThe triad symptoms of a glomus tumor are paroxysmal pain localized tenderness and cold intolerance. A glomus tumor can occasionally cause pain by directly compressing the forearm nerves , 15. HowIt is difficult to objectively detect a glomus tumor through clinical palpation because of the subcutaneous or deeper layer location and the small size (within a few millimeters). Imaging studies such as MRI or ultrasonography are useful for diagnosis , 10. MRIThe only treatment for a glomus tumor is surgical excision. Recurrence is infrequent and is usually due to incomplete excision. Cutaneous and extracutaneous glomus tumors are biologically benign, and metastasis or malignant changes are extremely rare , 2, 4, 8Compared to the well-known subungual glomus tumor, extradigital glomus tumors may have atypical features, and the absence of objective findings often leads to a delay in diagnosis. A careful and comprehensive approach, including a thorough physical examination, comprehensive medical history, surveillance of the disease progress, and imaging studies, is essential upon clinical suspicion.Although glomus tumors of the forearm are rare, an orthopedic surgeon should be aware of glomus tumors in the differential diagnosis of intravascular lesions of the upper limb. This tiny mass can cause severe pain to the patient, and a long history may unfortunately result in an improper diagnosis such as neuralgia, arthritis, and psychosomatic pain. Clinical suspicion plays an important role in management of this lesion, and only early surgical excision alleviates the patient's pain.