Solitary osteochondromas are common benign long bone tumors originating from cartilage. They may produce a wide variety of symptoms and complications depending on their spinal location. These may include compressive myelopathy, nerve root compression, pathologic fracture and malignant degeneration, or in some cases only pain. Solitary cervical spine osteochondromas have been reported mostly in the neural arch or vertebral body. This report describes a patient presenting with neck pain, with a benign osteochondroma arising in the right bifid C5 lamina. Solitary osteochondromas are common lesions and account for 30–40% of all benign bone tumors . OsteochA 28-year-old male patient presented to our clinic with neck pain. He had neck and right upper extremity pain for two years. Pain was especially on the right shoulder and was extending to the right wrist and hand. His past medical history was unremarkable with no trauma reference. He experienced no benefit from medical treatment. His complaints increased during the last 1.5 months. We admitted the patient to our clinic for further examination and treatment.At physical examination, cervical neck movements were limited and painful in all four directions and there was local tenderness on the right side of C5 and C6 dermatomes. Muscular strengths were normal. There was no sensorial deficit. Deep tendon reflexes were all normal. Hoffman reflex and clonus were bilaterally negative.The patient was referred to the radiology department for further evaluation of the spinal region. Cervical radiographs, computed tomography, and magnetic resonance imaging were performed. No significant lesion was detected in anteroposterior and lateral radiographs. In cervical CT (computerized tomography), there were coarsening of bifid lamina of C5 vertebra and a hyperintense expansile lesion on the lamina . In cervThe patient was operated with the primary diagnosis of an expansile sclerotic, osteoblastic lesion on the bifid lamina of C5 vertebra. During the operation, a vertical incision was performed between the levels of spinous processes of C3 and C7 vertebrae. Bifid lamina of C5 vertebra was seen and the expansile lesion on the lamina was removed microscopically following a right-sided hemilaminectomy. The tumoral lesion was reported as osteochondroma in the histopathological examination . SkeletaThe patient had no neurological deficits after surgical treatment. His pain on the right upper extremity disappeared just after the surgery. The patient was then discharged and recommended for clinical control.Osteochondromas or osteogenic exostoses can be both solitary and multiple as in the case of hereditary familial exostosis. Osteochondromas represent 30–40% of all benign bone tumors . OsteochSpinal osteochondromas are generally seen at the cervical and thoracic regions , 3, 8. AThe detection of spinal osteochondromas is difficult on plain radiographs because of the complex image formed by the spinal bony elements , 13. CT In case progressive neurological symptoms develop, the treatment of choice for spinal osteochondroma is surgical removal of the tumor . The manSpinal osteochondromas are rare entities and clinical manifestations due to spinal cord compression by the tumor are rarely seen. Total removal of the tumor is the choice of treatment and it is a must for avoiding the recurrence of the tumor. Partial hemilaminectomy must be chosen especially in younger patients to prevent kyphosis due to laminectomy.


The purpose of this study was to review the imaging and anatomopathologic findings and to discuss the main differential diagnosis of bizarre parosteal osteochondromatous proliferation (BPOP) or Nora's lesion, a rare benign surface lesion of the bone. Histologically confirmed plain radiographs, ultrasound, CT and MRI images of four patients with BPOP were obtained and retrospectively reviewed. Three cases involving the hand and one involving the foot are reported. On plain radiographs, BPOP is a wellmarginated, calcified or ossified mass arising directly from the cortical surface of the underlying bone. Ultrasound images show a low echoic peripheral cap around the lesion. CT images show the wide base of the lesion. On MRI, BPOP was of a low signal on T1, enhancing following gadolinium administration. Underlying bone and adjacent surrounding soft tissues were normal. Bizarre parosteal osteochondromatous proliferation (BPOP) or Nora's lesion  was nameCase 1: A 52-year-old man presented with a five-year history of a mass on the palmar aspect of the right index finger. He claimed to have no history of trauma. On examination, the mass was hard, indolent and located at the level of the middle phalanx.Antero posterior and lateral radiographs of the right index finger demonstrated a dense mass approximately 1.5 cm in size. The mass extended from the palmar aspect of the second phalanx. There was cortical erosion, but no abnormality of the underlying bony architecture .Case 2: A 24-year-old woman was seen for a nine-month history of a hard mass on the second phalanx of the right medius. She denied any history of injury. Antero posterior and lateral radiographs showed a well-circumscribed calcified juxtacortical mass.CT showed lack of continuity between this lesion and the medullary canal of the affected bone, but the cortex was normal .Case 3: A 38-year-old woman presented with swelling and discomfort affecting her right forefoot. There was no significant past medical history nor history of trauma. On examination, there was evidence of a fusiform swelling regarding the proximal phalanx of the third right toe. Radiographs showed an ossified mass with well-defined margins and a wide base on the plantar surface of the first phalanx of the third right toe .Case 4: A 45-year-old woman working as a tailor complained of swelling and pain of the distal phalanx of the right medius. Radiograph showed a welldefined bony mass originating from the distal phalanx of the right medius. In the T1 weighted MR image, a lesion of low signal intensity extending from the distal phalanx of the right medius was demonstrated. The cortex and bone marrow of the underlying bone were of normal signal intensity. The soft tissues showed no abnormality. T2 MR imaging of the lesion showed slightly increased signal intensity centrally with the periphery having much higher signal intensity. T1 weighted image following gadolinium administration showed uniform enhancement of the lesion .All patients underwent surgical excision of the lesions with pathological diagnostic confirmation. Two patients were lost to follow-up evaluation. Regarding the other two patients, the post operative follow-up period ranged from six to eighteen months without clinical or radiological signs of recurrence.BPOP is a benign and rare surface lesion of the bone that usually affects the proximal and middle phalanges, and the metacarpal or metatarsal bones.4]5] Th[5] Th4][ Th[5] ThBPOP is less commonly found in other sites. Some unuBPOP affects males and females equally.5]6]7][6][7]5][[7][6][7][6][7]5]The typical clinical presentation is a painless swelling that grows over a period of months to years.12] Pain Pain12] Radiographically, BPOP is a well-marginated, calcified or ossified mass arising directly from the cortical surface of the underlying bone.15]17][[17][15][[[17][15]Fine cut computed tomography (CT) scan shows a well-defined, intensely calcified or ossified mass, arising from the cortex of the affected bone. CT is better than plain radiography in showing the absence of continuity between the cortex, the medullary cavity of BPOP and the bone of origin and the absence of cortical flaring in this affected bone.21]22][22]21][2[22]21]17] [17] 12][ [17] 12]While most of the authors agree about these latter MRI findings, Orui  reportedBPOP has an atypical histological appearance, as reflected by the use of the word "bizarre" in its name. Microscopic typical findings include highly cellular, disorganized and irregular cartilage associated with proliferation of bizarre fibroblasts, disorganized bone and spindle shaped fibroblasts located in the intertrabecular space.12]23][[23][12][[[23][12]The presence of an unusual form of calcified cartilage that stains blue on hematoxylin and eosin (H and E) stain is characteristic. This feature, which has come to be known as "blue bone", was first noted by Meneses 2 and is found even after decalcification.13]17][17]13][1[17]13]25] T[25] T8][ T[25] T8The absence of continuity between the lesion and medullary cavity of the bone has been singled out as a key radiographic finding to differentiate BPOP from osteochondroma.6]8][10[8][106][[10[8]1014]15][[15][14][[[15][14]Recently, Rybak et al. presented four cases of pathologically proven BPOP in which corticomedullary continuity with the underlying bone was demonstrated on imaging. He indicates that what used to be a critical radiological finding distinguishing BPOP is not a reliable method for either identifying or excluding this lesion.Grossly BPOP may also be confused with osteochondromas, but some histological differences are apparent.28] In a In a28] Parosteal osteosarcoma is very rarely found in the hands and feet. It typically appears as a dense lobulated mass attached by a broad-based pedicle to the cortex and has heavy mineralization with a sclerotic appearance.Because of the increased cellularity and fibrous tissue proliferation within the bony portion, BPOP needs also to be distinguished histologically from parosteal osteosarcoma and a grade I or II chondrosarcoma. In parosPeripheral chondrosarcoma, typically found in adults, is rare in the hands and feet. Radiographically, it appears as a mass with ring-like or popcorn calcification.Peripheral chondrosarcoma is histologically different from BPOP by the well-differentiated hyaline cartilage with lobular architecture and no mitosis in grade I variant. In grade II, the cellularity increases and foci of necrosis are seen.Turret exostosis or ossified hematoma is a smooth dome shaped parosteal bone proliferation related to trauma and may also mimic BPOP. It is usually located on the dorsal aspect of the proximal or middle phalanx.14]27][[27][14][[[27][14]A stress fracture with extensive callous formation may as well look like BPOP. Typically, it presents as a thin lucent fracture line with periosteal reaction and cortical thickening. The diagnosis of stress fracture should be made from the history in conjunction with radiographic features.The other radiologic and histologic differential diagnoses of BPOP are florid periostitis and myositis ossificans. Florid reactive periostitis is a rare benign lesion usually affecting the small bones of the hands and feet in second and third decade patients. It is frequently related to trauma and is characterized by soft tissue swelling, variable lamellated or mature periosteal reaction, slight periosteal elevation and juxtacortical soft tissue calcification.14] Hist Hist14] Although agreement exists with regarding most common locations of BPOP, much debate still exists about its pathogenesis. Horiguchi  considerThese facts support the theory first published by Yuen  which evLy et al. proposesThese theories discussed earlier are based on a history of trauma initial to BPOP, whereas, many patients do not complain about previous injury or trauma. In addition, in the series by Meneses , there wOruiRecently, Nilsson  reportedBPOP is a benign lesion; therefore no treatment is required if it is asymptomatic. If the pIn fact, BPOP has a remarkable tendency to recur.16]21][[21][16][[[21][16]Ly et al. found thDespite this high recurrence rate and atypical histological appearance, no malignant transformation, metastases or deaths have been described so far in patients with BPOP.2]9][16[9][162][[16[9] Duri Duri30]


Hydatid disease is a helminthic anthropozoonosis with worldwide distribution due to the close associations among sheep, dogs, and humans. It can occur almost anywhere in the body with a variety of imaging features, which may change according to the growth stage, associated complications, and affected tissues. A definitive diagnosis requires a combination of imaging, serologic and immunologic studies. Ultrasonography, computed tomography and magnetic resonance imaging are highly accurate in detecting a hepatic hydatid cyst. However, hepatic hydatid cysts in an unusual location and/or of an unusual dimension, with atypical imaging findings, may complicate the differential diagnosis. Surgical treatment remains the best treatment.We describe an unusual case of a giant hydatid cyst, with exophytic growth from the right lobe of the liver of a 55-year-old Egyptian man. The cyst was strongly adhered to his ipsilateral kidney, which was displaced in a downwards and anterior direction, close to his abdominal wall, simulating a retroperitoneal origin. This atypical growth raised doubts about the most appropriate surgical approach. Magnetic resonance imaging easily clarified the origin of the cyst as our patient’s liver, allowing accurate surgical planning.Rarely, hydatid cysts can reach an extremely large size without any additional symptoms. Giant cysts need radical therapy because they might lead to perforation and anaphylaxis in some patients. Magnetic resonance imaging is very useful in the study of hydatid disease because of its capacity to allow a large field of view, multiplanar acquisition, and high contrast resolution. In some unusual hepatic presentations, magnetic resonance imaging can be used to determine the correct anatomical relationships. Echinococcus tapeworm. It can occur almost anywhere in the body with a variety of imaging features, which may change according to the growth stage, associated complications and affected tissues.Hydatid disease (HD) is a helminthic anthropozoonosis with worldwide distribution, caused by the larval stage of the A definitive diagnosis requires a combination of imaging, serologic and immunologic studies. Hepatic hydatid cysts can be treated by surgery, chemotherapy and/or percutaneous aspiration. Although a number of scolecoidal agents have been developed against HD, surgical resection is the only curative treatment. HD primarily affects the liver and demonstrates characteristic imaging findings on ultrasonography (US); computed tomography (CT) and magnetic resonance imaging (MRI) are highly accurate in detecting hepatic HD.However, hepatic hydatid cysts in an unusual location or of an unusual dimension, with atypical imaging findings, may complicate the differential diagnosis. Large cysts, today fairly rare even in endemic areas, are called giant hydatid cysts (GHCs) ,2. GHCs We describe an unusual case of a GHC originating from the right lobe of the liver with exophytic growth into the retroperitoneum. This atypical growth raised doubts about the most appropriate surgical approach. MRI easily clarified the origin of the cyst as the liver, allowing accurate surgical planning.A 55-year-old Egyptian man reported to our department with a large cystic mass occupying his entire right abdominal quadrants, detected by abdominal US during a routine check-up. The features of the mass on US were consistent with HD and it was diagnosed as three cysts - hepatic, renal and retroperitoneal. Our patient had no history of liver or kidney disease or pain in his right abdominal quadrants. Serologic and immunologic studies  performed during his hospitalization confirmed the HD diagnosis and a surgical approach was the first choice because of the large dimension of the mass. However, on the basis of US images alone, it was difficult to understand the origin of the cysts and to determine if there were three cysts or just a single giant cyst.Our patient underwent unenhanced MRI to determine preoperatively the extent of the disease. The MRI scans showed a huge single 30×18×16cm cystic multiloculated mass, misdiagnosed as three single cysts, hepatic and renal, during the US investigation Figures . The cysen bloc hydatid mass resection with an anterior approach, leaving his liver with the mass in place during hepatic transection  and lungs (15%); less frequently they involve anatomical locations such as the brain, bones and heart .The clinical manifestations of hepatic HD depend on the site, size and stage of development of the cyst  and HD iIt has been reported that hepatic hydatid cysts grow one centimeter in diameter during the first six months and then two to three centimeters annually; this is strictly related to the surrounding tissue resistance . GHCs arTypical radiological findings are well known and very helpful in the diagnosis of the disease. US, CT and MRI are highly accurate in detecting hepatic HD ,11, and CT and MRI may display the same findings as US; however, calcification of the cyst wall or internal septa is easily detected with CT. It is widely accepted that abdominal CT, with its higher rate of accuracy, is the diagnostic tool of choice. However, MRI, due to its multiplanar capabilities and the excellent contrast resolution for soft tissues, has a particular importance if the diagnosis of HD is questionable, because it is more accurate in demonstrating parietal features and defining anatomical relationships.Furthermore, hepatic HD may have a low-signal intensity rim on T2-weighted MR images, which probably represents the outer layer (pericyst), rich in collagen and generated by the host. It has been proposed as a characteristic but nonspecific sign  when comThe treatment options for hydatid cyst of the liver depend on stage, localization, size and complications of the cysts, and include nonoperative and operative methods . OperatiIn our case, the large dimension and proximity to vital organs induced us to operate so as to avoid cystic complications, such as a rupture in the pleural cavity through the diaphragm or inferior vena cava compression. Moreover, the challenge of this case lay in the conspicuity of the HD. Indeed, the hepatic cyst reported here was an extremely rare large asymptomatic cyst originating from the right lobe of our patient’s liver, was well-encapsulated, was not calcified, had a unique retroperitoneal growth, and was strongly adhered to his right kidney, peritoneum and diaphragm, thus suggesting a retroperitoneal origin of the mass.Hepatic HD may use two common routes of exophytic growth: the bare area of the liver and the gastrohepatic ligament. Growth through the bare area leads to involvement of the diaphragm and extension into the thorax; growth through the gastrohepatic ligament appears to be the path by which the cyst reaches the stomach. In our case, the giant size of the mass created doubt about its origin - hepatic or retroperitoneal. This was because the cyst rotated and displaced his right kidney down and anteriorly, simulating the growth of a retroperitoneal mass.In this case, multiplanar MRI was the best imaging technique to demonstrate exophytic cyst growth and its migration toward the retroperitoneum and diaphragm, and allowed accurate presurgical diagnosis and surgical planning . For these reasons, we believe that MRI is mandatory in the case of a complex hydatid cyst.We report that a hydatid cyst can, rarely, reach an extremely large size without any additional symptoms, and that these giant cysts need radical therapy because they might lead to perforation and anaphylaxis in some patients.MRI is very useful in the study of HD, because of its capacity to allow a large field of view, multiplanar acquisition and high contrast resolution. In some unusual hepatic presentations, MRI can be used to determine the correct anatomical relationships. Finally, radical surgical resection remains the best treatment option.Written informed consent was obtained from our patient for the 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.CT: computed tomography; GHC: giant hydatid cysts; HD: hydatid disease; MRI: magnetic resonance imaging; US: ultrasonography.The authors declare that they have no competing interests.GME and GV wrote the paper and performed the surgery. AL, CC, RS, PL, EBR, MA and ADC collected data, and performed the MRI imaging and surgery. All authors read and approved the final manuscript.


Transverse colon volvulus is an uncommon acute surgical presentation associated with a higher rate of mortality than volvulae at other locations along the colon. Surgical resection or correction is the only treatment, and various methods have been described in case report literature to relieve the volvulus and prevent recurrence.We present the case of a 25-year-old Caucasian woman who was admitted with a three-day history of abdominal pain, absolute constipation and abdominal distension. Subsequent radiographic and computed tomography imaging revealed right-sided colonic dilatation suggestive of a volvulus. An emergency laparotomy was performed during which the dilated proximal bowel was decompressed and colopexy executed by using the greater omentum to fix the transverse colon at the hepatic and splenic flexures.Volvulus of the transverse colon is rare but must form part of the clinician's differential diagnosis when encountering a patient with suspected bowel obstruction, especially in younger patients with no previous surgical history. Laparotomy is the treatment of choice and the technique of using the greater omentum as a fixing point for redundant bowel to the lateral abdominal wall is an option that may be considered especially when the bowel appears viable. Transverse colon volvulus, first reported by Kallio  in 1932,A 25-year-old Caucasian woman presented to our emergency department with a three-day history of abdominal pain, distension and absolute constipation with vomiting. There was no significant medical or surgical history. She took no regular medications, and was a non-smoker and non-drinker. Bedside observations were normal. A physical examination revealed a distended tympanic abdomen with tenderness across the lower abdomen but no signs of peritonism. A urine dipstick test was unremarkable and blood tests showed a raised white blood cell count (14.4 ×109 cells/L) and C-reactive protein (35mg/L). Urea and electrolytes, hemoglobin and liver function tests were all normal.Radiographs of the abdomen Figure  and chesA nasogastric tube was inserted, intravenous fluid resuscitation commenced and our patient underwent an emergency exploratory laparotomy. Intra-operatively, the bowel was found to be significantly dilated from the caecum to the distal transverse colon and the distal small bowel was mildly dilated, giving the impression of decompression through an incompetent ileocaecal valve. There was a volvulus of the transverse colon in the right upper quadrant. The transverse mesocolon appeared to take origin from the right upper quadrant as opposed to the usual location as an extension of the peritoneum from the posterior abdominal wall centrally. There was no evidence of colonic ischemia or serosal injury.The large bowel was decompressed with a 14 gauge needle allowing the tightly held bowel to be manipulated more easily. The large bowel was fully unraveled and the volvulus corrected then decompressed distally into the descending colon. The greater omentum on the transverse colon was then fixed at both the hepatic and splenic flexures by creating pouches in the lateral anterior abdominal walls, and fixing the omentum within this with polydioxanone sutures (PDS). The central portion of the omentum was purposefully caught in the mass closure of the wound to ensure additional central fixation.Post-operatively our patient made a good recovery. By day five she was tolerating a full diet and was discharged six days post-operatively. Despite a superficial wound infection at two weeks, she made a full recovery with no complications noted at her three month and six month follow-up.Volvulus of the transverse colon is a rare surgical emergency and as of 2008 there were only 100 published cases worldwide. Transverse volvulus accounts for only 2% to 4% of cases of colonic volvulus, but carries a 33% mortality rate ,6.Etiology can be acquired or congenital in nature -7.The former commonly is due to adhesions, inflammatory strictures, carcinoma or malposition of the colon following previous surgery -8.Congenital abnormalities include midgut malrotation, resulting in abnormal fixation ,4,8, conTransverse colon volvulus has been described in the literature as subacute-progressive or fulminating . FulminaRadiologically, it is classic to find a ‘bent inner tube’ sign on plain abdominal films  or a ‘biWith regards to management, in contrast to sigmoid volvulus, which can often be decompressed during sigmoidoscopy, transverse colon volvulus must be surgically corrected . When neThe argument in favour of colopexy over resection for viable bowel resides in eliminating the risks associated with the latter option. These include risks of anastomotic leak, paralytic ileus, stenosis and the need for a stoma; all of which carry considerable morbidity as well as mortality . Thus itWhile rare, transverse colon volvulus must form part of the clinician’s differential diagnosis when encountering a patient with suspected bowel obstruction, especially in younger patients with no previous surgical history. Exploratory laparotomy allows full identification of the pathology and access for corrective and restorative management. The technique of fixing the greater omentum in anterior abdominal wall pouches to correct anatomy has been demonstrated here as a successful method of treatment in the shortterm that may be considered.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.The authors declare that they have no competing interests.MS was involved in the initial diagnosis and management of our patient upon presentation to hospital, and in reviewing the literature and writing the initial manuscript. JY and KS were in theatre at the laparotomy and were major contributors to the manuscript and edited its contents. KG was the senior clinician involved in overseeing the care of our patient and approved the final version of the manuscript. All authors read and approved the final manuscript.


Intra-cortical osteosarcoma is the rarest subtype of osseous-producing tumor. Most reported cases present a low-grade histology with slow progression and good oncological control after adequate treatment. In this report, we describe a case and review the literature to propose adequate treatment.We present the case of a 21-year-old Thai woman who was thought to have an intra-cortical osteosarcoma of the right tibia. We performed a wide resection and reconstruction with bone transportation using an Ilizarov external fixator. The tumor recurred five years later at the same site with a similar histology. We performed a new resection and reconstruction by ankle arthrodesis with adjuvant chemotherapy. At the last follow-up, she had remained active and free from disease for seven years.This case report of recurrent intra-cortical osteosarcoma describes an atypical presentation. The low-grade histology, adequate surgical margin and adjuvant chemotherapy of the recurrent lesion were favorable factors, and our patient has remained free of any tumor recurrence. Only 18 cases have been reported in the literature, and the most common sites are the tibia (nine cases) and the femur (nine cases). The sex ratio of females to males is 5:13, and the median age is 19 years . Most cases present with slow progression, and treatment results in good oncological control. However, three cases have been reported in which the patient presented with distant metastasis . Two hadA 21-year-old Thai woman was evaluated for a lesion in her right tibial diaphysis. The mass exhibited slow, progressive enlargement over a one-year period, and she experienced occasional pain. Her physical examination revealed a hard-consistency mass at the anteromedial aspect of the right tibia affixed to the bone. The area was mildly tender, but there was no inflammation. A bone scan showed an increased uptake of radionuclide at the site of the lesion. Her chest roentgenogram, complete blood count, blood urea nitrogen, creatinine, liver function test and serum alkaline phosphatase were within normal limits.X-rays demonstrated an oval intra-cortical lytic bone lesion located at the diaphysis. It had an irregular endosteal border and sclerotic density that appeared to surround the osteolytic lesion. Multiple radiodense speckles were seen within the lytic area, suggesting the presence of an osteoid matrix Figure . An openAlthough free fibular graft has been reported as the suitable technique for large defect reconstruction , in someFive years later the patient felt pain in her right tibia at the same site as the primary tumor. Radiography revealed an abnormal osteolytic lesion at the lateral side of the distal tibia. Coronal T1-weighted and short-tau inversion remedy (STIR) images showed a recurrent tumor mass in the lateral portion of the right distal tibial metaphysis. The tumor showed hypointensity on T1-weighted images and mixed signal intensity on T2-weighted images with invasion into the surrounding soft tissue. Diffuse cortical thickening of the distal tibia and marrow edema was also evident Figure . A compuIntra-cortical osteosarcoma is the rarest variant of osteosarcoma described since the first one was reported by Jaffe in 1960 . Althougen bloc excisional resection and adjuvant radiation. The patient underwent above-knee amputation, since the tumor recurred as an anaplastic spindle cell sarcoma at the same site. He died at age 26 years, five months as a result of distant metastasis. The second reported case was a 25-year-old patient with an intra-cortical osteosarcoma of the right femur. The patient underwent pre-operative cobalt radiation therapy followed by hip disarticulation and died 14 months after the diagnosis with lung metastasis. These are the only two cases in which patients underwent high-dose adjuvant radiation, and both died. Picci et al. [In Jaffe's original report , a 14-yeet al. [en bloc. This patient has remained asymptomatic without evidence of metastasis for 30 years [Lichtenstein  initiallet al.  reportedet al. [et al.'s patient [Currently, several reports are in agreement that an adequate surgical margin plays an important role in controlling local recurrence ,8. Althoet al.  and us w patient  had a hiAmong 13 well-controlled cases, eight were treated without adjuvant chemotherapy and were free from recurrence and metastasis for 21 months . Five patients received adjuvant chemotherapy and remained well without recurrence and metastasis for 48 months . Our patient was treated without adjuvant chemotherapy at the first presentation because her tumor was considered to be a histologically low-grade lesion and an adequate margin had been used during surgery. We eventually had to combine chemotherapy with surgery during the second episode to minimize the risk of recurrence.The ideal treatment of intra-cortical osteosarcomas is surgical resection with an adequate margin. In cases of uncertain margin resection, adjuvant chemotherapy should be considered. For a recurrent lesion, low-grade histology is a favorable prognostic factor. However, adequate margin resection as well as neoadjuvant and adjuvant chemotherapy must be proposed in cases of lesion recurrence to offer the patient a good prognosis.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.DP wrote the draft manuscript. OA, TL, NS and DP carried out the operation and the patient follow-up. CS participated in the radiological evaluation. JS participated in the pathological studies. All authors read and approved the final manuscript.


We report a rare case of synchronous duodenal carcinoma, colonic carcinoma and renal oncocytoma successfully treated using a one-stage surgical approach. Potential risk factors for multiple primary malignancies associated with duodenal carcinoma are discussed. This case illustrates several practice points for consideration: 1. Patients presenting with small intestinal carcinomas have a higher than average chance of developing second primary tumors in other organs; this should be taken into consideration during staging and follow-up. 2. For full staging of patients presenting with small bowel tumors, upper and lower gastrointestinal endoscopy and PET scanning should be considered. 3. A one-stage surgical procedure can be used safely and successfully for multiple synchronous primary tumors. Primary carcinomas of the duodenum, excluding carcinoma of the ampulla of Vater, have been reported to occur in 0.019-0.5% of all autopsies and in 35-45% of all cases of small intestinal cancer . There hA 67 year old male presented with a history of weight loss and generalized weakness of 2 months duration. General investigations revealed anemia with hemoglobin of 9.2 g/dl. Upper gastrointestinal endoscopy was unremarkable, lower gastrointestinal endoscopy revealed a rectal polypoid mass (2.5 cm) with wide base, 12 cm from the anal verge  sutures. The biliary anastomosis was performed using 4/0 (PDS) sutures in an interrupted fashion end to side with the same jejunal loop. The gastro-jejunal anastomosis was performed in an end to side fashion using 3/0 PDS. The colonic anastomosis was performed with an EEA stapler. The operative time was 6 hours with minimal blood loss.Pathology of the periampullary tumor revealed a moderately differentiated duodenal adenocarcinoma with 10 benign peri-pancreatic lymph nodes. Pathology of the colonic tumour showed a moderately differentiated infiltrating adenocarcinoma reaching but not crossing the muscularis propria with eight benign pericolonic lymph nodes (T2N0M0). Final pathology of the kidney mass was oncocytoma with had been completely excised.The patient recovered well postoperatively and was discharged home.th and 6th decade with a median age of 55 years [Primary adenocarcinoma of the duodenum is very rare with an incidence of 0.035% of all gastrointestinal cancers . It cons55 years . PresentThe etiology and pathogenesis of small bowel and duodenal cancer is poorly understood. Several risk factors have been identified including Crohn's disease, familial adenomatous polyposis (FAP), celiac sprue, cystic fibrosis and colon cancer ,9. SeverData from 13 cancer registries from Europe and Canada was analyzed in terms of incidence of second primary cancers following a diagnosis of small intestinal malignancy. This study reported a 68% overall increase in the risk of a new primary cancer after small intestinal carcinoma [Dietary factors, alcohol consumption and high body mass index which are known risk factors for colon cancer are possibly acting as risk factors for small bowel adenocarcinoma in the same individual . Renal oIn conclusion, we are presenting an unusual case report of a patient presenting with three synchronous primary tumors who treated with a successful on-stage surgical approach.This case illustrates several practice points:1. Patients presenting with small intestinal carcinomas have a higher than average chance of developing second primary tumors in other organs; this should be taken into consideration during staging and follow-up.2. The use of upper and lower gastrointestinal endoscopy and consideration of PET scanning for full staging of patients presenting with small bowel tumors.3. A one-stage surgical procedure can be successfully used for multiple synchronous primary tumors.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.The authors declare that they have no competing interests.ES drafted the manuscript, AcS and A1S participated in the design of the study, MK assisted with the collection of data and conceived of the study, WF and DM participated in the design and coordination of the study. All authors read and approved the final manuscript


Facial cosmetic procedures are commonplace nowadays, especially techniques that aim to increase lip volume. Full lips provide a youthful, healthy, feminine and sensual appearance. There are many techniques and materials used to recover the loss of contour that occurs with age.An unusual case of fat tissue accumulation following cosmetic upper lip augmentation in a 61 year-old female was reported. Surgical treatment was performed for esthetic concerns. Microscopically, the tissue removed was composed of muscle fibers and mature adipocytes.Undesirable effects of esthetic treatment can occur and the clinician should be familiar with such complications to diagnose and manage them. Full and well-defined lips represent beauty, attractiveness and sensuality ,2. GraviA 61 year-old female was attended at our department complaining about an unesthetic “fold” on her upper lip when smiling and talking. During anamnesis, she reported having type 2 diabetes, hypertension and a history of cosmetic augmentation of the upper lip. Twelve years ago, she was submitted to an injection of autologous fat tissue in her upper lip; however, she did not know any details about the procedure.Physical examination revealed bilateral submucosal enlargement of the upper lip, which was soft on palpation, especially on the left side, and showed no color alteration of the mucosa Figure . She wasAutologous fat transplantation is indicated for the correction of wrinkles, depressed or atrophic areas in the face  and voluLongevity of the correction is unpredictable, depending on the harvesting and transfer techniques of the fat . UndesirDifferential diagnosis of this case was unsatisfactory esthetic treatment and double lip. Double lip is a rare oral abnormality, characterized by a deformity of the lip, in which a fold of labial mucosa is evident at rest or smiling . Double Surgical treatment was performed for esthetic concerns. However, the treatment of patients with similar clinical appearance could be necessary due to alterations in phonetics, mastication and difficulty in wearing a prosthesis . The patNowadays, esthetic treatments are widely performed, especially in the perioral area. In some cases, undesirable effects can occur and the clinician should be familiar with such complications in order to diagnose and manage them correctly.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 of this journal.The authors declare that there are no conflicts of interest.EK, JAR and JDA were involved in clinical attendance and drafting the manuscript. YRC performed the microscopic analyse. All authors read and approved the final manuscript.


To gain biological insights into lung metastases from hepatocellular carcinoma (HCC), we compared the whole-genome sequencing profiles of primary HCC and paired lung metastases.We used whole-genome sequencing at 33X-43X coverage to profile somatic mutations in primary HCC (HBV+) and metachronous lung metastases .In total, 5,027-13,961 and 5,275-12,624 somatic single-nucleotide variants (SNVs) were detected in primary HCC and lung metastases, respectively. Generally, 38.88-78.49% of SNVs detected in metastases were present in primary tumors. We identified 65–221 structural variations (SVs) in primary tumors and 60–232 SVs in metastases. Comparison of these SVs shows very similar and largely overlapped mutated segments between primary and metastatic tumors. Copy number alterations between primary and metastatic pairs were also found to be closely related. Together, these preservations in genomic profiles from liver primary tumors to metachronous lung metastases indicate that the genomic features during tumorigenesis may be retained during metastasis.We found very similar genomic alterations between primary and metastatic tumors, with a few mutations found specifically in lung metastases, which may explain the clinical observation that both primary and metastatic tumors are usually sensitive or resistant to the same systemic treatments. Hepatocellular carcinoma (HCC) is one of the most common cancers worldwide with an estimated 500,000-1,000,000 new cases per year -4. Althode novo nonsynonymous coding single-nucleotide variants (SNVs) were identified in metastatic lesions from acral melanomas [Recently, whole genome sequencing of metastatic acral melanomas and matched primary tumor and adjacent normal tissues showed high similarity of global gene copy number alterations, loss of heterozygosity and single nucleotide variation in primary and metastasis tissues . Meanwhielanomas . In addielanomas ,10. HereFour patients who were pathologically diagnosed of HCC and lung metastases were included in this study.A 50-year old man presented with a 9-cm sized mass at right hepatic lobe. The patient was a HBV carrier and his liver function was of Child-Pugh classification of A. The patient underwent right liver lobectomy when freshly frozen tissue of primary tumor and blood DNA was collected. The pathologic review of the surgical specimen revealed Edmonson grade II, tumor necrosis of 10%, microvascular tumor emboli and chronic active hepatitis with bridging fibrosis in background liver. Three years later, the patient developed a 2-cm sized single metastatic nodule in posterior basal segment of right lower lobe. The patient underwent right lower lobectomy and the pathologic diagnosis was metastatic hepatocellular carcinoma. Currently, the patient is in remission without recurrence since lung metastatectomy for more than 5 years.th TACE, two nodules were detected in right lung. The patient underwent video-assisted thoracoscopic surgery wedge resection of right upper apical segment, posterior segment and RLL lobectomy. The pathologic diagnosis was metastatic hepatocellular carcinoma. Fresh frozen tissues were collected during the surgery at this time. Seven months later the patient developed another 2 cm lung metastatic nodule in right middle lobe and underwent metastatectomy. The patient is currently in remission for 4 years.A 46 year-old man presented with a 5.2 × 4.4 cm massive HCC involving S6 and S7. The patient was a hepatitis B carrier. The patient underwent right hemi-hepatectomy (tissue procurement of primary tumor). The pathologic features were as follows, Edmonson grade II, 0% necrosis, intrahepatic metastasis, segmental portal vein microinvasion and negative resection margin. Three months later, the patient developed small recurrent tumor in left lateral segment of left lobe of liver and underwent radiofrequency ablation and 6 cycles of transcatheter arterial chemoembolization (TACE) thereafter. Shortly after the 6A 43-year old male presented with an 11-cm sized HCC-B (hepatitis B) and underwent right hepatectomy. The pathologic examination revealed Edmonson grade II HCC with 10% necrosis, no portal vein invasion with mild portoperiportal inflammation in background liver due to chronic hepatitis B. Four months following the surgical resection, a newly developed enhancing nodule in S3 of liver was detected which was subsequently treated with 5 cycles of TACE. Two years from the initial hepatic resection, a 1 cm nodule in left upper lobe was detected which was surgically removed with wedge resection. The tumor specimen was confirmed of metastatic HCC in lung and freshly frozen tissue was collected at this time. The patient underwent repeated multiple pulmonary metastatectomies for recurrent metastatases. The patient still has slowly progressive, multiple lung metastases which were refractory to sorafenib and sunitinib. The patient died of disease ten months later after failing to treatment due to disease progression.A 51-year old male had a 6.6 c m sized liver mass and was diagnosed of HCC-B. The patient underwent S6 segmentectomy and the pathologic examination revealed Edmondson grade II, 60% necrosis, no microvessel or portal vein invasion and no intrahepatic metastasis. Two years later, a 14 cm solitary lung nodule was detected in left lower lobe superior segment which was surgically resected. Thirty months later, the patient developed multiple HCC nodules and received ten 10 cycles of TACE. The patient is still undergoing periodic TACE for multiple HCCs at the time of this writing. The core information is marked with specific notes , and SOAPsnv [Alignments to the reference genome with opened gaps were performed using BWA ( SOAPsnv  was usedP = 0.0003531)  and C:G > G:C (P < 2.2e-16) transversion, respectively, in metastasis-specific mutations  events affecting protein coding (18/24 frameshift and 7/10 in-frame) in 42 genes  in primary tumors and metastases of 4 cases were detected by comparing with their respective matched normal samples. From primary tumors to metastases, the lengths of CNAs were changed, but the total sizes of CNAs were not significantly different  in sequencing data . We idenTo identify genes as “driver” somatic mutations in either primary tumorigenesis or metastasis process, the genes with significant higher nonsynonymous mutation rates against a background were estimated . Tumor sBy integrating altered genes in primary tumor and specifically altered genes in metastasis from four cases, we concluded several cancer and cell migration relevant pathways. Altered pathways in primary tumor were Wnt, JAK-STAT, cell cycle and focal adhesion pathways Figure , which fIn summary, genetic alteration patterns are very similar between primary tumor and metastasis of 4 cases Figure . For somHCC is the fifth most common cancer and the third most common cause of cancer-related deaths worldwide. It has a high prevalence in Asia, including Korea, due to endemic hepatitis B virus infection. numerous clinical trials using cytotoxic chemotherapy or targeted agents have failed, except sorafenib . While iIn this study, we employed whole-genome sequencing to profile somatic mutations and structural variations in primary HCC (HBV+) and their matched metachronous lung metastases. Several recent studies on HCCs of different etiologies  using whole-genome and exome sequencing have not only confirmed previously known mutations of β-catenin and TP53 but also identified novel genetic alterations in genes that are involved in epigenetic regulation, such as ARID1A, ARID1B, ARID2, MLL, and MLL3 . Wnt/β-cIn summary, comparison of structural variations between primary and metastatic tumors shows very similar and largely overlapped mutated segments Figure . In addiDespite similarities between primary and metastatic pairs, a few somatic mutations were detected only in metastatic tumors. Multiple mutations of zinc finger (ZNF) genes were detected in the metastasis samples, and mutated genes that are involved in apoptosis and transcription regulations were enriched in metastases   in the regulator of TAT binding and a missense mutation N131D) in the DNA-binding domain, both were previously reported by COSMIC , respectively.Qualified genomic DNA samples were randomly fragmented by Covaris. After ligation of sequencing adapters to both ends of resulting fragments, successfully adapter-ligated DNA fragments were purified, and those with an insert size of approximately 500 bp were selected by electrophoresis. Insert fragments with correct sizes were harvested and prepared for cluster amplification by cBot (Hiseq 2000).The qualified DNA library was then loaded on Hiseq2000 for high-throughput sequencing, to meet required sequencing depth with 90-bp paired-end reads. Raw image files were read by the Illumina’s base calling software with default parameters, and raw data were collected.http://soap.genomics.org.cn/index.html) using a sensitive score threshold. Next, p values were calculated using Fisher’s exact test for all putative mutation sites, based on the distribution of read support with high stringent specificity and sensitivity [http://refgene.com/) using an in-house annotation program.Sequencing reads from primary and metastatic tumors, as well as matched normal tissues, were gap-aligned to the hg19 reference genome by BWA program, followed by removal of PCR duplicates. Somatic variants in primary and metastatic tumors were detected by comparing tumor reads to those of matched normal samples. Somatic mutations in tumors were predicted by SOAPsnv  by re-implementing the GISTIC algorithm  using coribution .We further applied CREST  to wholeDriver genes were predicted using in-house software by re-implementing the algorithm in which driver genes are the genes with a significant higher non-silent mutation (nonsynonymous/splice/coding indel) rate against a background mutation rate estimated from collected silent mutations . SimilarPathway enrichment analyses of genes harboring somatic SNV, small coding indel, CNV, or SV were performed using the Gene Set Enrichment Analysis ,36.p < 0.05). The distribution of inter-somatic mutation distance in primary and metastatic tumors of sample D473 were compared to the distributions of inter-somatic mutation distances in HCC and other types of tumors, using published data downloaded from ICGC (ftp://data.dcc.icgc.org/version_11/). We used 2 lung adenocarcinoma samples, TCGA-05-4382-01A-01D-1265-08 and TCGA-05-4424-01A-22D-1855-08, from TCGA (http://cancergenome.nih.gov/), 2 prostate cancers, CPCG_0099_Pr_P_P1 and CPCG_0184_Pr_P_P4, from OICR (http://oicr.on.ca/), 2 liver cancers, HCC122T and RK020_C01, from NCC (http://www.ncc.go.jp/en/), and 1 liver cancer sample (sample 180) sample, sample180, from a collaboration HCC project of which some results have been published [We computed the inter-somatic mutation distance within individuals by defining the inter-somatic mutation distance as the distance from a somatic mutation to the very next one. We computed null distributions by randomly sampling positions from the genome (excluding assembly gaps), while maintaining the number of somatic mutations per chromosome . Using tSequencing data for this publication has been deposited in NCBI Short Reads Archive and is accessible through accession number SRA076160.Written informed consent was obtained from the patient (who was not deceased at the time of writing or waived by the IRB if deceased) for the publication of this report and any accompanying images.HCC: Hepatocellular carcinoma; SNVs: Single-nucleotide variants; SVs: Structural variations; CNAs: Copy number alterations; SSMs: Somatic SNV mutations; NGS: Next-generation sequencing; TACE: Transcatheter arterial chemoembolization; COSMIC: Catalogue of somatic mutations in cancer; ICGC: International cancer genome consortium; TCGA: The cancer genome atlas; OICR: Ontario institute for cancer research; NCC: National cancer center.The authors declare that they have no competing interests.JL, HL, JGK, CKP initially designed this study. LO, YZ performed research and analyzed data. LO, JL, CKP, JGK, HL, MM, YS, ZG and HZ designed the research and reviewed the manuscript. LO, JL, JGK, HL, CKP, MM, YS, ZG wrote the paper. All authors read and approved the final version of the manuscript for publication.The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1755-8794/7/2/prepubContains tables of summaries of variants, variant lists, gene lists and pathway lists.Click here for fileContains figures of clinical images and variants.Click here for file


Gastric outlet obstruction is a clinical syndrome caused by a variety of mechanical obstructions. Peptic ulcer disease used to be responsible for most gastric outlet obstruction, but in the last 40 years the prevalence of malignant tumors has risen significantly. Adhesive disease is an infrequent and insidious cause of mechanical gastric outlet obstruction.We report the case of a 78-year-old Caucasian man who had a clinical history of a right nephrectomy for malignancy three years earlier and who was admitted for a severe gastric outlet obstruction (score of 1) confirmed both by an upper endoscopy and by a fluoroscopic view after contrast injection. A computed tomography scan and a laparotomy, with omental biopsies, showed a peritoneal carcinomatosis with the development of abdominal adhesions that prompted an abnormal gastric rotation around the perpendicular axis of his antrum with a dislocation in the empty space of his right kidney. Symptoms disappeared after surgical bypass through a gastrojejunostomy.Our patient experienced a very rare complication characterized by the development of adhesions due to peritoneal carcinomatosis caused by a renal carcinoma treated with nephrectomy. These adhesions prompted an abnormal dislocation of his antrum, as an internal hernia, in the empty space of his right kidney. Gastric outlet obstruction (GOO) is a clinical syndrome caused by a variety of mechanical obstructions . GOO is typically characterized by epigastric abdominal pain, early post-prandial vomiting with or without nausea, and weight loss. Before 1970, peptic ulcer disease was responsible for most GOO, but since the introduction of proton pump inhibitors in clinical practice 40 years ago, the prevalence of malignant tumors as the cause of GOO has risen to between 50% and 80% of all cases .A 78-year-old Caucasian man, referred to our institute by another hospital, was examined in our out-patient clinic for frequent episodes of post-prandial vomiting in the previous 30 days. The hospital referred him with a clinical and endoscopical suspicion of gastric lymphoma (severe stricture of his gastric antrum), although the results of his biopsy analysis were negative. A computed tomography scan confirmed the findings seen on upper endoscopy but offered no clear explanation of its nature. His clinical history included a right nephrectomy for malignancy three years earlier, although he underwent no chemotherapy. At examination, he appeared thin and malnourished and had a Gastric Outlet Obstruction Scoring System (GOOSS) score of 1  [Symptomatic adhesions after surgery are frequent (25% of readmissions in the first post-operative year) . HoweverTo the best of our knowledge, no data on the development of post-nephrectomy adhesions in patients operated on for renal malignancy have been published. In a 10-year study of 871 living-donor nephrectomies, less than 1% of patients experienced major complications and a mere 8% developed minor complications. There were no reports of adhesive disease . A recenIn the long-term post-nephrectomy follow-up of patients with renal malignancy, the major concern is metastatic disease. The greatest risk of recurrence following resection for renal cell carcinoma is within three to five years after the operation, with predominant lung, bone, liver, brain, and local-regional involvement . HoweverOur patient experienced a very rare complication characterized by the development of adhesions due to peritoneal carcinomatosis caused by a previous renal carcinoma treated with nephrectomy but not chemotherapy. These adhesions prompted an abnormal gastric rotation around the perpendicular axis of his antrum, with a dislocation, as an internal hernia, in the empty space of his right kidney. This case is interesting for two reasons: (a) GOO can occur as a late adhesive complication after abdominal surgery or peritoneal carcinomatosis or both, and (b) despite the low frequency of incidence, a late metastasis from renal carcinoma can involve the peritoneum without ascites but with severe obstructive symptoms.This report highlights the importance of regular out-patient visits in patients with a history of neoplasms, even if they have undergone surgery and especially if they have not been treated with chemotherapy. Particular attention should be paid to new obstructive symptoms as possible consequences of late post-surgical or unusual peritoneal metastatic complications.GOO: gastric outlet obstruction; GOOSS: Gastric Outlet Obstruction Scoring System.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.FM collected the data and wrote the article. GC, IT, and LB were involved in drafting the manuscript and revising it critically for important intellectual content. GB, SG, and SC were involved in revising the manuscript critically for important intellectual content. MT was involved in revising the manuscript critically for important intellectual content and gave final approval of the version to be published. All authors read and approved the final manuscript.


Cervical thymic cysts are nearly 0.3% of all congenital cervical cysts. Thymic cysts are asymptomatic, but they rarely complain of dysphagia or tracheal obstruction symptoms. A soft, mobile, and painless mass increasing with valsalva maneuver directs the diagnosis of laryngocele. There has not been any study in the literature in which thymic cyst presenting like laryngocele. We hereby present a case of thymic cyst mimicking laryngocele that has not been reported so far. Thymic cysts are rare lesions and may occur anywhere from mandible to mediastinum, along with the migration path of the thymus. However, they are mostly seen in the mediastinum. Thymic cysts are generally misdiagnosed as branchial cyst and cystic hygroma, since they may cause almost similar symptoms to cervical cysts. However, a thymic cyst mimicking a laryngocele is extremely unusual. We herein present a boy with a cervical thymic cyst mimicking laryngocele. To the best of our knowledge, this is the first case in the literature showing that a cervical thymic cyst presenting as a laryngocele in childhood age.A 7-year-old patient admitted to our clinic with a painless mass on the right side of the neck, which was 2 cm in diameter . The detThe thymus gland is the central organ of the lymphoid system during infancy. It embryologically develops from the third and the fourth pharyngeal arches in the 6th gestational week of development. During the 7th and 10th weeks, as the primordial thymus migrates caudally and medially into the mediastinum, the proximal portion of the thymopharyngial duct usually undergoes atrophy. In the children, cervical thymic cysts constitute 0.3% of all congenital cervical cysts. Thymic cysts are rare lesions and there are less than 100 cases, together with histopathological diagnosis-reported in the literature , 3. ThymIn general, the patients with thymic cysts are asymptomatic, but they rarely complain of dysphagia or tracheal obstruction symptoms. The clinical differential diagnosis is not easy and they are often misdiagnosed as cystic hygromas or branchial cleft cysts, which are also frequently seen in this age group and which have similar symptoms –7. AdditIn the present case, the size of the mass increased with valsalva maneuver. Besides, there was a relationship between larynx and the mass, like a tail sign. The mass extended to the right hemithorax and there was a relationship between the mass and pleura. Our hypothesis is that during resting, the mass was located into the thorax while during valsalva maneuver, the pleura pushed the mass to the neck and the mass seemed to inflate by breath. Therefore, preoperative diagnosis of the present case was a giant external laryngocele extending into the mediastinum. We performed total excision of the mass with surgery. The excision of the lesion was curative and no reoccurrence was reported. Long-term prognosis was perfect. Our patient was followed for 16 months without recurrence. To the very best of our knowledge, this is the first case of a cervical thymic cyst mimicking laryngocele in childhood age.In conclusion, although extremely rare, cervical thymic cysts should be considered in the differential diagnosis of the cases with the external laryngocele, particularly in the childhood age. The definitive method of treatment of the cervical thymic cyst is surgery.


A 19-year-old male patient was admitted with flank pain, which had lasted intermittently for four years. In X-ray, there was a radiopacity with a dimension of 6 × 4 cm on the left pelvic bone. Intravenous pyelography revealed a huge left megaureter with a stone in the lower end and grade V hydronephrosis. A left ureterolithotomy, left nipple ureteroneocystostomy, and psoas hitch operation was performed. A voiding cystourethrogram taken three months after the operation showed no reflux, and in IVP there was reduced dilatation of the collecting system when compared to the ureter before the operation. Primary obstructed megaureter (POM) is a result of obstruction or an adynamic ureteral segment in the ureterovesical junction . POM occA 19-year-old male was admitted with the complaint of intermittent flank pain with a history of four years. There was no record of stones in his history. The systemic and urogenital examination was normal. In his direct urinary system radiography there was a radiopacity with a dimension of 6 × 4 cm on the left pelvic bone .In intravenous pyelography (IVP) advanced hydroureteronephrosis (grade V) was seen and there was no transit of the radiopaque dye through the bladder. In IVP it was determined that the opacity belonged to the lower end of the left ureter .In abdominal ultrasonography (USG) it was observed that the left calyceal structures and pelvis were extremely dilated and the left ureter had dilated from the proximal end through the distal end. Left kidney function was 39.6% in 99 m Tc-dimercaptosuccinic acid (DMSA). The renal function test and electrolyte values were normal in serum analysis. A direct nipple ureteroneocystostomy and ureterolithotomy operation was performed with a left Gibson incision as reported previously . During At the patient's follow-up examination in the postoperative third month there was no proliferation in his urine culture. A postoperative voiding cystourethrogram showed no reflux and the appearance of a filling defect due to the left nipple .IVP revealed residual dilatation on the left side . In USG In adults primary megaureter rarely causes symptoms and does not require surgery. Symptomatic cases generally admit urinary tract infection (UTI), hematuria, and flank pain. The most frequent reason for megaureter is the occurrence of an adynamic segment in the distal ureter . GeneralA few incidences of calculus in megaureter cases have been reported in the literature . HoweverConsequently the direct nipple ureteroneocystostomy technique may be a viable treatment method for POM because of its easy appliance; there is no need to prepare a submucosal tunnel for plication and there is a high success rate.


Glomus tumors are benign, subcutaneous neoplasms of the perivasculature. Though facial location is rare, the diagnosis of a glomus tumor should be considered in cases of undiagnosed painful facial nodules or chronic facial pain. Imaging aids in defining the tumor and planning a complete excision in order to avoid recurrence. Histological examination is mandatory after every attempted excision. A case of glomus tumor of the cheek along with the possible pitfalls of diagnosis and treatment and a brief review of the limited associated literature are presented. Glomus tumors are benign neoplasms of the perivasculature accounting for 1-2% of soft tissue tumors [A 24-year-old man presented at our clinic with a four-year history of a painful nodule in his left cheek that reoccurred after two attempted excisions in the last four months by different specialists. No biopsy result or other histological information was available. Otherwise healthy, the patient complained of pain and local tenderness. Clinical examination revealed a well-defined, round, firm nodule in the middle of the left cheek, approximately 1 × 1 cm in size that appeared to be subdermic with no apparent fixation to the underlying tissues. The overlying skin was normal and the nodule was extremely painful on palpation. Examination of the oral cavity was normal and no neck lymph nodes could be palpated. Complete head and neck examination and laboratory tests were also unremarkable. Contrast enhanced CT (puffed cheek method) revealed a well-defined, contrast-enhanced, round soft-tissue mass of the left cheek over the buccinator muscle measuring 13.8 × 9 mm, with no apparent relations to the surrounding tissues . AttacheGlomus tumors are neoplasms of the glomus body, a neuromyoarterial unit found within the reticular dermis that serves as a specialized arteriovenous anastomosis . The artAccounting for 1-2% of soft tissue tumors , 9. MaliThe symptoms and signs of a facial glomus tumor may vary from an asymptomatic (rarely) subcutaneous nodule to excruciating chronic facial pain. The most expected presentation is that of a small, painful, subcutaneous nodule. A long history of consultations by various specialists without a definite diagnosis can be expected. The classic triad of symptoms described for glomus tumors consisting of pain, localised tenderness, and cold hypersensitivity may or may not be present. Overlying skin may or may not be discoloured. Though usually not larger than 1 × 1 cm in size, large facial glomangiomas mimicking venous malformations have been reported . A 4 : 1Magnetic resonance imaging has proven to be the most sensitive imaging modality for the diagnosis of glomus tumors in the extremities –15. Cont


An intraductal papillary neoplasm of the bile duct is a biliary, epithelium-lined, cystic lesion that exhibits papillary proliferation and rarely causes large hemorrhagic cystic lesions. Here, we report a case of an intraductal papillary neoplasm of the bile duct mimicking a hemorrhagic hepatic cyst in a middle-aged man with large hemorrhagic hepatic cysts who experienced abdominal pain and repeated episodes of intracystic bleeding. Following portal vein embolization, extended right hepatic lobectomy was performed, and intraoperative cholangiography revealed communication between the intracystic space and the hepatic duct. Although histological studies revealed that the large hemorrhagic lesion was not lined with epithelium, the surrounding multilocular lesions contained biliary-derived epithelial cells that presented as papillary growths without ovarian-like stroma. A diagnosis of oncocytic-type intraductal papillary neoplasm of the bile duct was made, and we hypothesized that intracystic bleeding with denudation of the lining epithelial cells might occur as the cystically dilated bile duct increased in size. Differential diagnosis between a hemorrhagic cyst and a cyst-forming intraductal papillary neoplasm of the bile duct with bleeding is difficult. However, an intraductal papillary neoplasm of the bile duct could manifest as multilocular hemorrhagic lesions; therefore, complete resection should be performed for a better prognosis. Intraductal papillary neoplasm of the bile duct (IPNB), which is histologically characterized by the prominent papillary growth of atypical biliary epithelium with distinct fibrovascular cores, is considered to be a biliary counterpart of intraductal papillary mucinous neoplasm of the pancreas. IPNB caThe common clinical signs of IPNB are abdominal pain, jaundice and cholangitis. Radiological imaging studies commonly indicate diffuse bile duct dilatation with or without a papillary mass,3; howevA 65-year-old man presented at the hospital with epigastric pain. Laboratory tests indicated mild anemia , elevated liver enzymes , and increased concentrations of total bilirubin 1.4 mg/dl), γ-glutamyl transpeptidase (116 IU/l) and C-reactive protein (5.68 mg/dl). Carcinoembryonic antigen (CEA), carbohydrate antigen 19–9 (CA19-9) and α-fetoprotein were at normal levels. Computed tomography (CT) of the abdomen revealed a large hemorrhagic cyst in the right lobe of the liver. Transarterial embolization of the peripheral portions of the right and middle hepatic arteries was performed for hemostasis. The patient was referred to our hospital and was recommended surgical treatment because additional episodes of intracystic bleeding and abdominal pain were anticipated. Abdominal CT showed a large cystic lesion (13 cm in diameter) with a thickened wall, around which we observed several small cystic lesions  is routinely performed before hepatic resection in our institute to map the intrahepatic bile duct[The large cystic lesion exhibited a white thickened wall and was filled with rust-colored hematoma, as seen in the resected specimen Figure a. The leThe patient had an uneventful recovery and was discharged 13 days after the operation. At his one-year follow-up visit, the patient was healthy and did not show any signs of recurrence.Simple hepatic cysts are commonly observed in the general population using various imaging modalities. Although most hepatic cysts are not associated with symptoms, clinical presentations such as pain, biliary obstruction, infection, rupture and compression of other organs infrequently occur. Spontaneous intracystic bleeding is also a rare complication-14. Accoet al. observed mucinous fluid associated with hemorrhage in two out of nine cases of cyst-forming IPNBs[By contrast, according to the WHO classification system, an intrahepatic IPNB is defined as a cystic lesion that is lined with biliary, mucinous or oncocytic epithelium in papillary configurations without ovarian-like stroma. Luminalng IPNBs. Recurreet al. reported that only 11.5% and 13.5% of patients with an IPNB had high serum levels of CEA and CA19-9, respectively[et al. reported that some cyst-forming IPNBs might arise from peribiliary glands given their diverticulum-like appearance[A preoperative differential diagnosis between a hemorrhagic hepatic cyst and cyst-forming IPNB with bleeding is difficult when based solely upon clinical and radiological information. Yang ectively,19 but aectively,21; therectively,18. Howeectively, as was pearance. Althougpearance or magneAs with an intraductal papillary mucinous neoplasm of the pancreas, IPNB iset al. reported that surgically treated patients with pancreatobiliary-type IPNB demonstrated poorer survival than those with the gastric and intestinal types because pancreatobiliary-type IPNB was associated with a higher frequency of invasive carcinoma[Surgical therapy is required for IPNB treatment. Kim arcinoma. A positarcinoma, but lonarcinoma. We perfWe report a rare case of IPNB mimicking a hemorrhagic hepatic cyst. Although differential diagnosis between a hemorrhagic cyst and cyst-forming IPNB is difficult, multilocular hemorrhagic lesions could represent IPNB. Because IPNB may harbor carcinoma components, complete resection should be performed for a better prognosis.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.CA19-9: Carbohydrate antigen 19–9; CEA: Carcinoembryonic antigen; CT: Computed tomography; IOC: Intraoperative cholangiography; IPNB: Intraductal papillary neoplasm of the bile duct; MUC: Mucin core protein; WHO: World Health Organization.The authors declare that they have no competing interests.All authors contributed equally to this work. All authors read and approved the final manuscript.


First described by Hirsch and Helwig in 1961, chondroid syringomas (CSs) are rare, benign tumors of the skin arising from the eccrine sweat glands with tumor differentiation in the epithelial and mesenchymal tissues. They most commonly occur in the head and neck, although they may be also found in the axilla, trunk, limbs, and genitalia. The incidence of CS is <0.01% of all primary skin tumors. Malingnant chondroid syringomas (MCS), which are also called malignant mixed tumors of the skin, are extremely uncommon. MCSs commonly involve the limbs and rarely head and neck. In this article, we present a case of malignant chondroid syringoma localized in the face at the left nasolabial region in the light of literature review. Chondroid syringomas (CSs) are rare, benign tumors of the skin. In 1859, Billroth first described CS as a mixed tumor of the skin due to its histopathological resemblance to the benign mixed tumor originating from salivary gland . The terChondroid syringomas most commonly occur in the head and neck with a size ranging from 2 mm to ≥1 cm and usually present with solitary, solid, painless, nonulcerative, subcutaneous, or intracutaneous nodule , 6. TheyThe malignant variant of CS is rare. In most cases of MCS, anaplastic changes are present from the beginning. Malignant chondroid syringoma is a mixed cutaneous tumor, with epithelial and mesenchymal components, which compromises principally the trunk and extremities. Rarely, a chondroid syringoma of many years duration suddenly undergoes malignant changes with widespread metastasis .There have been no reports reporting effectiveness of chemotherapy and radiotherapy, and an early wide excision with a broad margin may be the most reliable treatment to date.In this paper, we present a case of MCS localized in the face at the left nasolabial region in the light of literature review.A 34-year-old female was admitted with a slowly growing nodular lesion at the left nasolabial region for three years . ExcisioChondroid syringomas are tumors arising from sebaceous glands, sweat glands, and ectopic salivary glands . They usAnalysis of the reported cases showed that the mean age of the patients at the time of diagnosis was 48.3 years (range 13–84 years). The neoplasm tends to affect limbs mostly , while its benign counterpart occurs mainly in the head and neck  , 12. TheIn our case, the patient had a history of a slowly growing lesion at the left nasolabial region and recurrent lesion within the first year of following local excision with a size of 1.5 cm. Malignant chondroid syringoma may occur de novo or rarely develop in a chondroid syringoma. In contrast to the benign counterpart, which is common in the head and neck region, the malignant variety occurs predominantly on the trunk and extremities –15. In oIn our case also, prior studies that had mitosis, nuclear atypia, pleomorphism, lymphatic invasion, and local recurrence have been recognized as helpful signs for the diagnosis of malignancy –16, 18. This highlights the need for microscopic diagnosis of all tumors that are excised irrespective of the clinical diagnosis. Treatment consists of complete excision of the tumor. Though local radiotherapy is often unsuccessful, skeletal metastasis has been shown to respond to radiotherapy . CombinaIn terms of prognosis of the reported cases, 8 of the 30 (27%) patients died from their disease. Death occurred as early as 9 weeks following surgery; one patient survived 12 years after diagnosis , 21. Malignant chondroid syringoma tends to follow an unpredictable clinical course. Of the reported cases, 50% had local recurrences –22. NodaIn conclusion, negative surgical margin should be attained using wide excision technique. These patients should be also monitored closely due to high potential of recurrence.


Benign gastro-colic fistula is a rare occurrence in modern surgery due to the progress in medical management of gastric ulcer disease. Here we report the first case of benign gastro-colic fistula occurring whilst on proton-pump inhibitor therapy. This is a case study of benign gastro-colic fistula and review of the available literature in regards to etiology, diagnosis, management and prognosis.An 84-year-old woman of Caucasian background presented with 12 months of worsening abdominal pain, nausea, vomiting, diarrhea and weight loss on a background of known gastric ulcer disease.en bloc resection of the fistula tract is the preferred treatment. Benign gastro-colic fistulae are becoming exceedingly rare in the context of modern medical management of gastric ulcer disease. Surgical management is the gold standard for both benign and malignant disease.The leading cause of gastro-colic fistulae has changed from benign to malignant due to improved medical management of gastric ulcer disease. The rarity and non-specific symptoms of gastro-colic fistula make the diagnosis difficult and it is best made by barium enema; however, computed tomography has not been formally evaluated. Surgical management with  Gastro-colic fistulae are described as presenting with the clinical triad of diarrhea, nausea/vomiting and weight loss . HoweverMalignant gastro-colic fistulae were first described in 1755 by Haller . Gastro-The overall incidence of gastro-colic fistula has decreased since the advent of effective medical management of gastric ulcer disease. Post-surgical-resection-associated fistulae and fistulae related to the use of non-steroidal anti-inflammatory medications were the most reported causes of benign gastro-colic fistulae ,4,6. In A barium enema is the radiological modality of choice for diagnosis of gastro-colic fistulae, with specificity of 90-100% compared with a barium meal that has a false negative rate of 30-70% [en bloc surgical resection of the fistula tract with a margin of adjacent tissue [The treatment of choice for a gastro-colic fistula is t tissue ,3,4,8. Tt tissue .Several cases of medical or minimally invasive management of gastro-colic fistulae have been described and may be suitable where malignant disease has been excluded and/or surgical intervention is not appropriate. Endoscopic injection of the fistula tract with fibrin has shown to be effective in several case reports .Prognosis for gastro-colic fistula has been thought to be quite poor. Between 1963 and 1994, the longest recorded survival post-resection for gastro-colic fistula due to malignant disease was nine to ten years . Post-opOne case series of six patients reported one post-operative death due to underlying co-morbid conditions. The remaining cases were followed for a mean of 66 months, with one further death due to an unrelated underlying co-morbid condition . HoweverAn 84-year-old Caucasian woman presented for repeat gastroscopy for follow-up of a benign gastric ulcer. She gave a 12-month history of worsening abdominal pain, nausea, non-feculent vomiting, diarrhea and approximately 20 kilogram weight loss. She denied any hematemesis, melena or fever. At presentation our patient was frail and emaciated. Regarding clinical examination, there were no abnormal abdominal findings.Helicobacter pylori had never been identified. Our patient was taking aspirin for cardiovascular prophylaxis and had been started on pantoprazole at 40 milligrams twice daily when the ulcer was first identified. Our patient's general practitioner confirmed prescription requests for this medication.A chronic gastric ulcer on the greater curve of her stomach had been first diagnosed at gastroscopy eighteen months earlier. Since then she had undergone four further gastroscopies without any change. Biopsies had only demonstrated features of chronic inflammatory change. On this occasion, gastroscopy revealed a deep ulcer of the greater curve of the stomach that appeared to penetrate the muscular layer and was highly suspicious of a fistula. The pathological report of the performed biopsy showed chronic inflammatory changes. An abdominal CT demonstrated a fistula between the stomach and transverse colon and excluded malignant disease. Contrast CT successfully diagnosed a fistula, excluded locally invasive disease and allowed pre-operative planning in a single step. A colonoscopy showed no evidence of primary colonic disease and failed to visualize the fistulous opening Figure .en bloc and primary anastomoses performed as malignant disease could not be definitely ruled out. A feeding jejunostomy was performed (Figures At laparotomy there were dense adhesions between the greater curve of the stomach and the distal transverse colon. The gastric ulcer together with the fistulous track and colonic opening were excised  Figures , 3, 4. HOur patient was discharged to a peripheral hospital on the twentieth post-operative day tolerating an oral diet.A gastro-colic fistula commonly presents with non-specific symptoms of diarrhea, nausea and vomiting and weight loss, thus making it a difficult diagnosis. The rarity of this condition, and alteration in the underlying etiology due to the advent of medical management of gastric ulcer disease, make benign gastro-colic fistula a very rare diagnosis. This case is important as it highlights the non-specific presentation of the disorder and is the first case documented in which benign gastric ulcer disease treated with proton-pump inhibitors progressed to gastro-colic fistula.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.MH and RC were the surgeons involved in the care of the patient. KB researched the background management of the patient and performed the literature review. All authors read and approved the final manuscript.


Cutaneous infection is an uncommon presentation of mucormycosis, usually seen after trauma, at the site of surgical drains or after occlusive dressings. The involved area is erythematous and painful, with varying degree of central necrosis. We report the case of chronic coetaneous infection of one year duration and without apparent necrosis in an immunocompetant patient.A 32-year-old immunocompetent woman presented with a large unilateral firm infiltrative plaque resembling cancer lesions, disfiguring the eyelids, nose and lips. The punch biopsy and then surgical debridement was done and the diagnosis of cutaneous mucormycosis was confirmed on histologic examinations with granulomatous reaction and characteristic broad, nonseptate, pale-staining hyphae.Mucomycosis should be in differncial diagnosis of any chronic infiltrative lesions even without visible necrosis and normal immune status of the patient. Cutaneous mucormycosis has been related to contaminated elastic bandages as a distinct entity , 2 but hA 32-year-old immunocompetent woman without any history of medical or surgical diseases, referred to our clinic, with unilateral edematous plaque on the left side of the face involving cheek, eyelids, nose and lips, with some ulcers which had purulent exudates on them .The lesion had begun from 18 months ago. The lesion persisted till 5-6 months ago with no cure. Since then, the surrounded skin gradually became red, erythematous, and other ulcers appeared on the skin. On physical examination, the left side of the face was edematous and red and some ulcers with purulent exudates were seen. The lesion involved the nose and the lips but nasal and buccal mucosa was intact. CT-scanning of paranasal sinuses showed soft tissue thickness of the left cheek without involvement of the ipsilateral sinuses. Evaluations on exudate specimens revealed branched and aseptate hyphae, indicating mucormycosis. Punch biopsy and then surgical biopsy were done and both showed granulomatos dermatitis accompanying broad branched and aseptate hyphae that invaded the vascular endothelium. These findings confirmed the diagnosis of mucormycosis and therapy with Amphotricin-B deoxycholate was begun. After the patient became stable, extensive repetitive debridement of the involved area were performed. Therapy with amphotricine-B deoxycholate continued for a total dose of 2 g with good outcome and recovery of the lesion. The patient was then referred for plastic surgery.Mucormycosis is a rare human infection and may result of decreased efficacy of human immune system. Cutaneous infections with these organisms show a great deal of variability. Necrotic and hemorrhagic lesions with black scars or gangrenous cellulitis may be seen. Erythema is often noted surrounding the lesions. Erythematous papules, nodules, plaques without superficial necrosis, shallow ulcerations and chronic infections that persisted for months or years have also been described . Also th