The ‘double bubble’ sign on antenatal ultrasound is often associated with duodenal atresia although there are numerous causes. We present a case of cystic biliary atresia presenting with a “double bubble” at 36-weeks gestation. Postnatal ultrasound and MRCP confirmed a cystic lesion at the porta hepatis, mandating early laparotomy and a successful Kasai portoenterostomy.Although diagnosis of such lesions may be imprecise antenatally, awareness and detection does allow early postnatal investigation and management, which is vital to optimise outcome.This case highlights the need to be mindful of other important anomalies that can give this appearance and that may require early intervention. Fetal anomaly scanning is key to the diagnosis of various congenital conditions. It is highly sensitive for the detection of abdominal wall defects (e.g. gastroschisis) and duodenal/ proximal intestinal atresias, but is less so for a number of other abdominal conditions. In addition, ultrasonography (US) can be used to identify extra abdominal pathology such as congenital diaphragmatic herniae and hydrocephalus.The ‘double bubble’ sign seen and originally described on plain radiography, but now also appreciable on US, is a result of fluid-filled structures seen in the either hypochondrium or epigastrium. Historically this sign has been strongly associated with duodenal atresia or other causes of duodenal obstruction. Nonetheless, to consider this sign as pathognomonic for duodenal atresia is dangerous and it is important to be mindful of other important anomalies that can give this appearance and that may also require urgent intervention.The parents of the child involved have kindly given us their written consent to share the case with our colleagues. Our aim is to remind clinicians of an important pathology.A male infant born at term was referred to our tertiary paediatric surgery centre at 2 days old with a history of bile-stained nasogastric aspirates. Although his 20-week fetal anomaly scan had been normal, a maternal US scan at 36 weeks for high fundal height and suspected polyhydramnios showed the appearance of a “double bubble” Figure , but witEnteral feeding was started and tolerated well. However, within the week, nursing staff noticed that the stools were pale.Liver biochemistry at day 9 showed total bilirubin of 113 μmol/L with a conjugated bilirubin of 83.9 μmol/L ; alkaline phosphatase (ALP) 280 iu/L  and alanine transferase 71 iu/L (reference range <41 iu/L).Magnetic Resonance Cholangiopancreatography (MRCP) was requested to evaluate the biliary system  is a disease of unknown aetiology with an incidence in the UK of about 1 in 17,000 live births (Livesey et al. Identification of a “double bubble” on maternal US mandates a search for possible causes post-natally Table . In thisIn this instance, the double bubble on antenatal ultrasonography, was in fact created by the extrahepatic cyst and the fetal stomach.Antenatal diagnosis of hepatobiliary disease is notoriously difficult and rarely correct (Davenport and Hadzic In this child’s case biliary malformations were not considered in the antenatal period. This is not uncommon. In the Kings College Hospital experience of neonates and infants with CBA and abnormal antenatal scans, none of the children had the correct diagnosis amongst the prenatal differentials (Davenport and Hadzic US does have a key role in the post-natal investigation of a persistently jaundiced infant, either to confirm the presence of choledochal malformation, inspissated bile syndrome or spontaneous biliary perforation; or in the actual diagnosis of BA. In non-cystic BA, the “triangular cord” sign has been advocated as a reliable diagnostic sign (Humphrey and Stringer The group also suggested, as much of the literature in the case of noncystic BA, that other features such as dilatation of the intrahepatic bile ducts and hepatic artery; the sizes and morphologic characteristics of the gallbladder and liver may also be helpful identifying these cases (Zhou et al. A management algorithm has been previously suggested in which confirming the cyst along with a non-dilated intrahepatic biliary tree and deranged liver biochemistry lead to urgent laparotomy and reconstructive surgery (Davenport and Hadzic Although the influence of age on outcome following surgery in BA is controversial, it is incontestable in the CBA variant. Caponcelli et al. showed a clear relationship between age at portoenterostomy and clearance of jaundice in a series of 29 infants with CBA. All those operated on at <40 days cleared their jaundice compared to none at >70 days (Caponcelli et al. With the advances in antenatal scanning, there is the opportunity to offer early intervention for potentially serious hepatobiliary disease in the neonate. Currently antenatal diagnosis of cystic biliary malformations is imprecise, which may be permissible as antenatal intervention is not warranted. However, antenatal detection does mandate early postnatal investigation. This can allow for an early diagnosis and treatment, but only if clinicians are mindful of the possible diagnoses. The first step in this is recognising that cystic biliary malformations, and crucially CBA, may be a cause of the “double bubble” sign on antenatal US.Written informed consent was obtained from the patient’s guardian/parent/next of kin for the publication of this report and any accompanying images.


Intraductal papillary neoplasm of the bile duct (IPNB) is classified as a biliary cystic tumor with a tendency of causing obstruction. Neoplastic cases involving hemobilia are rarely reported. We herein describe a case of biliary cystic tumor with repeated hemobilia. A 57-year-old woman was histologically diagnosed with cavernous hemangioma. During the follow-up period after transcatheter arterial embolization (TAE), she experienced repeated hemobilia, and multiple other TAE sessions were performed for hemostasis. She was referred to our hospital 8 years after the first surgery owing to a growing tumor. Histopathological examination after extended right hepatectomy and caudate lobectomy indicated IPNB with an associated invasive carcinoma. Six months thereafter, computed tomography revealed a recurrent liver tumor and a nodule in the abdominal cavity. She died 36 months after the second surgery, despite chemotherapy. Our experience suggests that IPNB should be considered during differential diagnosis of dilated hepatobiliary tumors with hemobilia. Hemobilia is usually caused by trauma, iatrogenic events, cholangitis, and cholelithiasis, but rarely associated with a tumor . On the A 57-year-old woman was previously admitted to a hospital because of abdominal pain. Gastrointestinal fiberscopy showed hemobilia, whereas abdominal ultrasonography and computed tomography (CT) performed at the time showed two tumors in the liver: one in the lateral segment measuring 4 × 3 cm in diameter and the other in the posterior segment measuring 10 × 5 cm in diameter. CT showed no clear differences between the two tumors; they were multiple capsulated lesions filled with cystic contents and highly enhanced mural nodules (data not shown). Additionally, the needle biopsy for both tumors did not reveal a malignant finding - a fragment of liver tissue with no malignancy. Despite the tumors being thought as benign, she underwent a diagnostic partial resection of the lateral segment. Histological examination indicated cavernous hemangioma. She then received transcatheter arterial embolization (TAE) for hemostasis of the right lobe tumor. During the follow-up period thereafter, she experienced six episodes of hemobilia, and four other TAE sessions were performed. She was referred to our hospital 8 years after the diagnostic surgery owing to the growing tumor in the right lobe.The patient's medical history included an appendectomy at 9 years of age, Cesarean section at the age of 26 and 29 years, and right lower lobectomy with partial resection of the lungs' left upper lobe for adenocarcinomas at the age of 53 years. She had no history of liver disease, including hepatolithiasis.Laboratory results on admission were as follows: hemoglobin, 11.1 g/dL; total bilirubin, 0.5 mg/dL; aspartate aminotransferase, 65 IU/L; alanine aminotransferase, 54 IU/L; alkaline phosphatase, 819 IU/L; and γ-glutamyl transpeptidase, 227 IU/L. Levels of alpha-fetoprotein and protein induced by vitamin K absence were within normal limits, while those of carcinoembryonic antigen and carbohydrate antigen 19–9 were elevated to 12.8 ng/mL and 862 U/mL, respectively. Viral markers for hepatitis B and C were negative.On physical examination of the abdomen, the liver edge was palpable four fingerbreadths beneath the xiphoid process. Abdominal ultrasonography revealed a multilocular oval mass with papillary infolding measuring 10 × 15 cm located in the right hepatic lobe. CT scans showed a low-density hepatic tumor measuring 15 cm in diameter that occupied almost the entire right and caudal lobes, compressing the portal vein. The nodules were contrast-enhanced Figure . Fluorodin situ or papillary adenoma with severe atypia.Endoscopic retrograde cholangiography demonstrated obstruction of the right hepatic bile duct, compression of the left hepatic duct, and dilatation of the peripheral bile ducts Figure . The cysThe patient underwent right hemihepatectomy (stipulate + Sg1) with cholecystectomy and concurrent splenectomy owing to the growing aneurysm of the splenic artery. Macroscopic observation indicated a direct connection between the cystic lesion and the right hepatic bile duct. During the operation, we ligated bile ducts and gave attention not to spill bile into the abdominal cavity. The resected multilocular tumor was filled with clear mucus, and the cut surface showed multiple yellowish papillary nodules lining the cystic wall. The cyst had a direct luminal communication with the right hepatic duct. Histological examination indicated the biliary cystic lesion was a well-differentiated adenocarcinoma Figure . OvarianSix months after surgery, CT scans revealed a recurrent liver tumor and a nodule in the abdominal cavity. Unfortunately, the patient died 36 months after surgery, despite chemotherapy.Hemobilia was first reported by Sandblom in 1948. It is a rare condition often caused by trauma, iatrogenic events, cholangitis, or cholelithiasis [Angiography is recommended for the examination and treatment of uncontrolled hemobilia . If hemoCommon symptoms and complications of IPNB are abdominal pain, liver function disorder, jaundice, or fever caused by the bile duct obstruction due to hypersecretion of mucin or the tumor itself ,7. We coAccording to the 2010 WHO Classification of Tumors of the Digestive System, biliary cystic tumors are no longer defined as biliary cystadenoma, papillomatosis, papillary adenocarcinoma, or biliary cystadenocarcinoma, but instead are redefined as IPNB and MCN of the liver . IPNB isOgawa et al. reported several CT findings of IPNB , includiThe CT findings in our case showed well-enhanced massive papillary contents in the dilated bile ducts with extravasation. The intraductal papillary contents evident by contrast CT further indicated that the tumor was present in the bile ducts instead of the liver parenchyma. Moreover, the repeated hemobilia episodes were also indicative of the bile ducts' involvement.IPNB are recognized as slow-growing neoplasms compared to normal intrahepatic bile duct carcinoma . SeveralIn this case, only the lateral lesion was resected for diagnostic purposes in the initial surgery, owing to the lack of any malignant impression. The residual lesion in the right lobe showed slow progression, with repeated hemobilia. IPNB also exhibits slow clinical progression, despite the possibility of direct invasion to the bile duct, leading to its dilatation, jaundice, or hemobilia.Our case presented a thought-provoking hepatobiliary course. The patient was initially followed for a slow-growing hepatic hemangioma, but eventually diagnosed with IPNB. It was challenging to distinguish the two conditions. Furthermore, although there are several reports of hemobilia originating from hepatic hemangioma, it is very rare in IPNB. Nonetheless, our experience in this case suggests that IPNB should be considered during differential diagnosis of dilated hepatobiliary tumors with hemobilia.Witten informed consent was obtained from the patient for publication of this case report and any accompanying images.


Many complications have been reported over tattoos, some of which are tumours, such as dermatofibromas. It is important to establish a differential diagnosis because they can resemble other malignant lesions as dermatofibrosarcoma protuberans. We report the development of a dermatofibroma in a 21-year-old man with a tattoo painted two years ago. Tattooing consists in the introduction in the dermis of indelible and exogenous pigments by needle and may be associated with complications such as infections, inflammatory and granulomatous reactions, as well as neoplasms .A 21-year-old man was referred to the emergency department with a 2-month history of a painful lesion arising from a black tattoo he had been done 2 years ago. He had no significant past medical history. The physical examination revealed a nodular lesion measuring 10 mm involved in a black tattoo on his right arm. The nodule was firm, tender, mobile, and light-red coloured . A skin Benign and malignant tumors, such as seborrheic keratosis, keratoacanthomas , melanomThe association between dermatofibromas and tattoos may be coincidental, given the great number of people with tattoos nowadays and the anecdotal presence of these tumors. However, these two conditions have a link because the skin was free of any type of lesion before the tattoo and there is a clear chronology between the development of the dermatofibroma and the tattoo. Furthermore, dermatofibromas can appear after trauma, and tattooing represents an action which triggers a nonspecific inflammatory reaction at punctured areas in an attempt to eliminate the toxic or carcinogenic agents contained in the ink. This process may induce a local fibrosis reaction.Dermatofibroma is a frequent tumor and the number occurring in tattoos is very rare. Evidence for a clear causal relationship is lacking due to limitations imposed by the relative rarity of these tumors arising within tattoos and by the nature of retrospective analysis. Therefore, the extent of this association and its underlying mechanisms remains uncertain. However, we suggest that dermatofibroma should be included in the differential diagnosis of tumors arising in sites of tattoos.


Retiform hemangioendothelioma (RH) is a rare low-grade variant of angiosarcoma mostly reported on dermis or subcutaneously. A 30-year-old woman suffering from dry cough, dyspnea and pleural effusion has been described. Distinctive symptoms and lesions on high resolution computed tomography (HRCT) scan and common histological, immunological feature are discussed. Diagnosis was made by thoracoscopy as RH. RH is known as a rare kind of low-grade malignant angiosarcoma with high rate of local recurrence and low potential of metastasis which is a sparsely distributed disease counting 35 cases Table  whereas A 30-year-old female patient was admitted to pulmonary department because of dyspnea and dry cough denying chest pain, hemoptysis, fever, weight loss or other systemic symptoms. Her symptoms were stable and had not been given any medication prior to admission. Her chest X-ray by local hospital showed pleural effusion on the right side. She received modified radical mastectomy in 2006 removing a breast mass on her right chest. The biopsy confirmed a diagnosis of poorly-differentiated invasive ductal carcinoma with axillary lymph nodes metastasis therefore the patient received adjuvant chemotherapy and radiotherapy. Her father died of liver cancer. The rest of her personal history, social history and the review of systems were unremarkable. Decreased vocal fremitus and dullness of percussion on her right thorax were detected in physical examination. No enlarged superficial lymph nodes were palpated and laboratory tests revealed normal level of carcino-embryonic antigen (CEA). Her ultrasonic examination of abdomen and pelvis cavity alarmed no metastatic signs. The patient’s HRCT results showed merely pleural effusion on the right side Fig. . No resiHistological and immunological examination of biopsy specimen pictured disarranged, jagged, blood vessels of thin walls and slit-like fissures were prevailing. At higher magnification the vessels’ walls were lined with highly nuclei/cytoplasm ratioed endothelial cells with minimal cytological atypia and mitosis. Atypical cells protruding into lumina mimicking “hobnail” appearance or endothelial papillae was absent as well as their invasion into adjacent tissues. Infiltration of lymphocytes was also rare Fig. . CombineAccording to our knowledge and clinical experience, endostatin was recommended for treatment. The patient revisited 6 months later with relieved symptoms when her HRCT showed diminished and encapsulated pleural effusion after receiving symptomatic treatment and passed away at home in 2013.Hemangioendothelioma (HE), an intermediate lesion between hemangioma and angiosarcoma, has been incipiently delineated as a kind of vascular tumor highlighted by its marginal biological features . It geneRetiform hemangioendothelioma is a low-grade variant angiosarcoma which has been first introduced by Calonje in 1994 . HyperchRH demonstrates a predilection for female adults . The majHistological diagnosis lays the foundation of clinical diagnosis also plays the key role in differentiating RH from angiosarcoma and other subtypes of hemangioendothelioma. RH comprises of blood vessels ”weaving a net” by infiltrating into bundles of collagen. Endothelial cells fashion intraluminal papillea with scarce cytoplasm , 12. HowHistochemical markers of RH include CD31, D2-40, CD34, factor VIII-related antigen , 16 amonThough histological and clinical features permits differential diagnosis between RH and other vascular tumors , it sharThe etiology of RH remains obscure. Latent relations between malignant medical history and RH should be elucidated , 9. In tNo tumor-related  deaths iIn summary, a case of RH situating on pleura has been depicted which exhibited peculiar pathological, immunological features and lesions. It has been reported as intrathoracic nodules predisposing a patient to unilateral pleural effusion. However discussion has been placed on the causes and treatment of RH. It implies that RH on location other than skin or subcutis may lead to corresponding symptoms or even threatened survival.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.


Alveolar rhabdomyosarcomas of the mediastinum in children are rarely reported. Multimodality therapy including chemotherapy, surgery and radiotherapy make up the backbone of the treatment of childhood rhabdomyosarcomas. Complete resection whenever achievable is an important prognostic factor. However, complete resection of tumors in the mediastinum often poses a unique challenge to thoracic surgeons due to their close proximity to important neurovascular structures. Complete resection may not always be possible and judicious peri-operative planning and preparation are required to avoid creating unnecessary surgical morbidities resulting in delay of adjuvant therapy.A 4-month-old Chinese baby boy was presented to our hospital with stridor, shortness of breath and episodes of cyanosis. Imaging studies found an anterior mediastinal mass compressing the trachea and other neurovascular structures and he was diagnosed to have alveolar rhabdomyosarcoma. Our patient received upfront chemotherapy and subsequently open resection of the mass was attempted via median sternotomy. Intraoperatively, the mass had invaded into the great vessels, precluding a complete resection. Debulking surgery was performed instead and our patient received timely postoperative chemoradiotherapy.We report a rare case of childhood alveolar rhabdomyosarcoma of the mediastinum with vascular invasion treated with chemoradiotherapy and debulking surgery. Complete resection was not possible due to the close proximity to the great vessels. Different surgical approaches to the mediastinum have been reported in adults and children alike. Regardless of the surgical access, the treatment of childhood rhabdomyosarcomas should be individualized, with careful balance between surgical clearance and surgical morbidity. Rhabdomyosarcoma (RMS) is the commonest soft tissue sarcoma in children and adolescents. Around 20% of RMS is alveolar type, and is associated with poorer prognosis. Treatment of RMS varies widely according to histology, site and age of the patient. Despite the advent of combined chemotherapy, complete surgical resection remains an important prognostic factor in the overall management of RMS. However, tumors in the mediastinum present a unique challenge to surgeons due to their intimate relationship with various important neurovascular structures. RMS in the mediastinum in pediatric patients is rarely reported and experiences with resection of cervicothoracic region tumors in childhood in general have been limited. In this report, we present a rare case of alveolar RMS of the anterior mediastinum with vascular invasion in a baby, and discuss the various considerations in the surgical approach for children with mediastinal tumors.A 4-month-old Chinese baby boy had experienced noisy breathing for 2 months. He presented to the pediatrics unit with increasing shortness of breath with episodes of cyanosis. A computed tomography (CT) scan of his chest with contrast enhancement revealed a 7 × 3 × 5cm anterior mediastinal mass extending to the cervical region, compressing the trachea and veins as well as displacing the aortic arch posteriorly Fig. . An inciUltrasonography of the abdomen, a bone scan and bone marrow study did not reveal any metastatic lesions. Vincristine, actinomycin and cyclophosphamide were commenced, with radiological evidence of tumor regression. After 11 weeks of chemotherapy, a CT scan of his chest showed good response to chemotherapy with shrinkage of the tumor. Our patient subsequently underwent an exploratory median sternotomy at the age of 8 months.A hard mass was found at the left superior part of the anterior mediastinum along with a sizeable thymus. The mass spanned from the lower border of the brachiocephalic vein to the neck and from the midline to the area between the left brachiocephalic vein and left common carotid artery. Total thymectomy with preservation of the bilateral phrenic nerves was performed. Complete excision of the tumor was attempted, but failed due to tumor invasion of the left brachiocephalic vein and the left common carotid artery. Debulking of the tumor was performed leaving behind a small volume of residual tumor in the area adjacent to the invaded great vessels Fig. . The sitA pathologic examination revealed an 8 × 5.5 × 0.8cm tumor invading the thymus, with associated thyroid tissue and skeletal muscle fibers Fig. . MicroscRadiotherapy with a dose of 50.4 Gy was given to the marked area of residual tumor. Unfortunately, a subsequent CT scan 9 months postoperatively and at week 39 of chemotherapy revealed tumor recurrence around the surgical clip site, confirmed by biopsy. The tumor progressed rapidly despite further therapy and our patient ultimately developed respiratory failure and died at the age of 3.Rhabdomyosarcoma (RMS) is the commonest soft tissue sarcoma in children . The comSpecific chromosomal translocations are detected in around 80% of cases of ARMS. Translocation t2;13) occurs in 60% of ARMS, and translocation t occurs in approximately 20%. These translocations result in the expression of transcription factors PAX3-FOXO1 or PAX7-FOXO1, which induce angiogenesis and activate cell proliferation. The prognostic value of the presence of fusion gene PAX3-FOXO1 in alveolar rhabdomyosarcoma is still under debate and a latest meta-analysis did not find significant difference in overall survival between patients with the positive and negative fusion gene. Indeed, better quality studies are needed to validate its prognostic significance [;13 have improved cure rates for RMS in children from 25% in the 1970s to 70% in the 1990s , 4. ThisNonetheless, complete resection of tumors in the cervicothoracic region remains a challenge to thoracic surgeons due to their close relationships with important neurovascular and lymphoid structures. In order to balance completeness of resection with debilitating postoperative morbidity, comprehensive preoperative planning, adequate surgical exposure and meticulous dissection are often required for favorable outcomes. To date, experiences with resection of childhood mediastinal tumors have been limited to case series and reports , 3, 5.et al., a case of tumor invasion into pulmonary artery was not identified until open surgery, ultimately resulting in aborted surgery [Magnetic resonance imaging (MRI) is recognized to be superior to CT in the assessment of tumor relations with adjacent soft tissues. Various adult case series and reports of mediastinal sarcomas had MRI performed to assess for degree of tumor encasement with respect to adjacent great vessels. Vascular involvement is considered absent if there is a less than 180 degree circumferential relationship or a clear plane can be seen between the tumor and vessel . Nonethe surgery . In our et al. performed the transmanubrial osteomuscular-sparing technique popularized by Grunenwald to achieve more than 90% gross total resections in four children with cervicothoracic neuroblastomas [et al. from the Sloan-Kettering Cancer Center in New York [Methods of surgical approach and preservation of important neurovascular structures have been discussed and published extensively in studies relating to resection of adult cervicothoracic tumors. In contrast, similar reports are very limited for childhood mediastinal tumors. Sauvat lastomas , 8. Thislastomas . To dateNew York . They peNew York . We deciIn conclusion, the management of mediastinal sarcoma remains challenging to both oncologists and surgeons. Reports of surgical resection in the literature for childhood, in particular childhood mediastinal rhabdomyosarcoma, are rare. Although complete resection is desirable, a proportion achieves only near-complete resection. Whatever the surgical access, some infant mediastinal tumors are found unresectable intraoperatively, usually because of vascular invasion. The risk and benefit between achieving complete resection and postoperative morbidity leading to delays in chemoradiotherapy must be weighed and balanced accordingly. Ultimately, the management of each case of mediastinal sarcoma should be individualized and multidisciplinary.Written informed consent was obtained from the patient’s legal guardian(s) 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.


We present a 56-year-old man with known diabetes mellitus and a 10-year history of hidradenitis suppurativa (HS) and highlight, through MR imaging findings, the relentless natural progression of the disease, characterized by recurrent exacerbations of abscesses and fistulae and complications of perianal fistulae and sacral osteomyelitis. We also demonstrate the appearance of this condition on PET-CT with F-18 FDG, which was performed for staging after postexcision tissue specimen revealed well-differentiated squamous cell carcinoma. The association of arthritis and possible dactylitis was also manifested in our patient. Discussion of HS in the radiology literature is limited and, to our knowledge, there has been no case report describing these imaging findings in the same patient.  Hidradenitis suppurativa (HS) is a chronic debilitating cutaneous disorder characterized by recurrent development of inflammatory subcutaneous lesions, abscesses, fistulating sinus tracts, and eventual cicatrization. Uncommonly encountered by the radiologists due to the diagnosis being based largely on clinical assessment, HS is more than just “recurrent abscesses” and has several important associations such as Crohn's disease and arthritis that may warrant further evaluation as well as potential complications, including complex draining sinus and fistula tract formation, fistula-in-ano, lumbosacral epidural abscess from deep extension, osteomyelitis, and squamous cell carcinoma developing in chronic inflammatory tracts. Imaging plays a supplementary but important role in both the early and chronic stages of the disease, aiding in the characterization of active lesions, assessment of the extent of involvement, and the recognition of complications.The use of advanced imaging modalities such as MRI, the modality of choice, has greatly aided in the management of these patients. To our knowledge, there has been limited discussion of HS in the radiology literature. We present a case report of a 56-year-old man with known diabetes mellitus and a 10-year history of HS and highlight through MR imaging findings, the relentless natural progression of the disease, characterized by recurrent exacerbations of abscesses and fistulae and complications of perianal fistulae and sacral osteomyelitis. We also demonstrate the appearance of this condition on PET-CT with F-18 FDG, which was performed for staging after postexcision tissue specimen revealed well-differentiated squamous cell carcinoma. The association of arthritis and possible dactylitis, which were also manifested in our patient, will also be discussed.A 56-year old man presents to our institution with complaints of increasing right buttock pain and pustular skin lesions, which have worsened in the recent month. Medical records showed recurrent admissions over the past 9 years for gluteal-femoral and perianal abscesses and multiple perianal fistulae requiring repeated surgical debridement. He has had gradual but certain weight loss in the recent years.On clinical examination, extensive erythematous and pustular lesions were noted in the gluteal-femoral region bilaterally, with extension to the upper thighs (worse on the right), on background of rope-like dermal thickening and induration. Multiple fistulous openings were identified with visible purulent and haemoserous discharge. Perianal erythema is observed.The patient was admitted for multidisciplinary management under colorectal surgery after being identified as a long-term sufferer of hidradenitis suppurativa (HS).An MRI of the pelvis was performed, which showed extensive subcutaneous abscesses, sinus, and fistula tracts in the right gluteal region where communicating and noncommunicating rim-enhancing collections were seen after IV contrast, extending deep into the right ischioanal fossa Figures  and 2 anSimilar to prior presentations, the deep right ischioanal abscess is closely related to a chronic transphincteric perianal fistula, better shown on a previous MRI  done 2 years before the current admission . There i Proteus mirabilis sensitive to Augmentin, with negative blood culture. A peripheral intravenous central catheter (PICC) was inserted and the patient was started on IV Augmentin, which was subsequently converted to IV vancomycin.Biochemical tests were consistent with sepsis and chronic inflammation, with raised total white count, polyclonal gammopathy and elevated erythrocyte sedimentation rate (ESR), and C-reactive protein (CRP) levels. Anemia, attributed to chronic disease, was also present. Initial wound culture grewHe also underwent wide debridement and drainage of the multiple subcutaneous abscesses with discharging sinuses affecting bilateral gluteofemoral and pararectal regions. Tissue specimens that were sent for histology incidentally revealed well-differentiated squamous cell carcinoma that has arisen from the chronic sinus tracts at the right thigh .A PET-CT scan following IV F-18 FDG was performed for staging, which showed no evidence of focal hypermetabolic lesion to suggest distant metastases. FDG-avid inflammation was however observed at the lesions of HS consistent with the subcutaneous inflammation .During this admission, the patient also developed painful swelling of the 2nd digit of the right hand. Radiographs demonstrated periarticular soft tissue swelling at the 2nd proximal interphalangeal joint and a small juxta-articular erosion at the 3rd distal interphalangeal joint . These aOur patient underwent sigmoid loop colostomy for faecal diversion after debridement of the chronic perianal fistulae. He was also started on palliative radiotherapy and chemotherapy for treatment of the right thigh squamous cell carcinoma.First described by Velpeau in 1839 with subsequent work done by Verneuil, Schiefferdecker, Brunsting, and Pillsbury et al. , hidradeDiagnosis is primarily clinical with recognition of typical lesions in characteristic distribution and recognizing its recurrent nature. Tender erythematous skin lesions are often identified on clinical examination during active inflammation, on background of dermal contractures and scarring from episodic recurrences. The anogenital regions and axillae show predilection for disease.Known associations have also been described, with Plewig and Kligman introducing the concept of acne tetrad, including acne conglobata, dissecting cellulitis and pilonidal abscess and suggesting acne inversa as a more accurate name, reflecting the pathogenetic link to follicular occlusion , 7. OtheAs is commonly described, our patient presents at middle age with recurrent perianal abscesses and has since progressed from his initial presentation a decade ago, where there were isolated abscess formation  in terms of MRI, PET-CT, and HS-related arthritis and highlight important complications that radiologists should be aware of such as bony involvement, that is, osteomyelitis, possible epidural abscess formation, perianal fistulae, and the rare instance of degeneration to squamous cell carcinoma.


Neuroendocrine tumors (NETs) are derived from neuroendocrine cells that are capable of producing functional peptide hormones. These tumors occur most frequently in the GI tract and lungs. GI NETs frequently metastasize into the liver, though NETs of primary hepatic origin are extremely rare. Ultrasound, CT, and MRI are typically all employed for characterization of these lesions but their appearance on diagnostic imaging can be highly variable. Reported here is an interesting case of a primary hepatic neuroendocrine tumor (PHNET), along with a discussion of the imaging characteristics of these tumors. Additionally, the current standards for definitive diagnosis and treatment of PHNETs are discussed. Neuroendocrine tumors (NETs) are tumors derived from neuroendocrine cells that are capable of producing functional peptide hormones. Typically, these tumors are found in the GI tract (55%) and lungs 30%), though they can also arise in the pancreas 2%), reproductive system 1%), biliary tract (1%), and head and neck (0.4%), among other areas [%, biliar%, reprod0%, thougDiagnosis of primary hepatic neuroendocrine tumors (PHNETs) requires histological confirmation of a NET as well as the exclusion of disease elsewhere, with final diagnosis often not achieved until after the tumor is resected. Ultrasound, CT, and MRI are typically all employed for characterization of these lesions. However, their appearance on diagnostic imaging can be highly variable, often mimicking more common hepatic malignancies such as cholangiocarcinoma, hepatocellular carcinoma, or hepatic metastases .Here, we report a hepatic lesion presenting at our institution diagnosed as a PHNET. Since these tumors are exceedingly rare, a PHNET was not considered initially but was diagnosed after treatment and histological analysis. A previous case of PHNET was confirmed at our institution and presented with similar ambiguity on initial diagnostic workup. It has been reported previously by Gurung et al.  and is uA 31-year-old female presented with a two-year history of intermittent sharp epigastric pain that began to increase in frequency in recent months. Episodes of pain lasted for up to 24 hours and were associated with severe nausea and vomiting. She had recently returned from Africa where she had been working for the past three years and was treated for malaria on three separate occasions. Her past medical history was otherwise unremarkable and her family history was significant only for celiac disease. Laboratory studies demonstrated a lipase of 57 U/L , an ALP of 196 U/L , and a GGT of 178 U/L , with otherwise normal liver enzymes. Workup for malaria, Hepatitis B and C, and celiac disease were all negative. An esophagogastroduodenoscopy was also performed and revealed no findings to explain the patient's abdominal pain.An abdominal ultrasound was performed and demonstrated a 2 cm ill-defined echogenic lesion in the medial left lobe of the liver as well as marked intrahepatic bile duct dilatation. Subsequent MRI revealed a lobulated lesion involving segments 2 and 4 with mild hypervascularity on arterial phase images and washout on delayed images . There wEndoscopic retrograde cholangiopancreatography (ERCP) was performed to assess the patency of the biliary tree and revealed a focal left hepatic duct stricture with moderate to severe peripheral left biliary duct dilatation. Stenting of the stricture was performed and required later revision because of ongoing cholangitis.CT was performed for tumor characterization, demonstrating an ill-defined heterogeneous soft tissue density mass in segment 4 of the liver measuring 4.2 × 3.8 cm as well as extensive intrahepatic biliary duct dilatation in segments 2 and 3 with pneumobilia . There wBased on the imaging, the presumed diagnosis was cholangiocarcinoma. The differential diagnoses for this lesion included hepatocellular carcinoma (HCC) and metastatic disease, though there was no obvious primary source.Left hepatectomy with bile duct resection was performed, along with hepaticojejunostomy using the remaining right hepatic duct. No peritoneal carcinomatosis was noted upon exploration.Grossly, the tumor was white-gray in appearance and more-or-less well circumscribed. It measured 4.5 × 3.5 × 4.5 cm. Immunohistochemistry was diffusely positive for synaptophysin and MIB-1 was positive in 5–10% of tumor cells. The tumor was determined to be an intermediate grade NET with a small amount of admixed adenocarcinoma in situ present within the bile duct. There was also evidence of nodal disease in two hepatic artery lymph nodes.Postoperatively, the patient developed a collection of bilious fluid in the resection bed of her left hepatic lobe requiring the placement of a drain but subsequently did well following her surgery.Neuroendocrine neoplasms are a relatively rare tumor of the gastrointestinal system, accounting for less than 2% of all GI neoplasms . PHNETs While the origin of PHNETs remains unclear, three hypotheses have been proposed: (1) they arise from neuroendocrine cells scattered in the epithelium of the intrahepatic biliary tract; (2) they originate from heterotopic pancreatic or adrenal tissue located in the liver; and (3) they arise from the neuroendocrine differentiation of a single malignant stem cell that is the precursor of other hepatic tumors .Clinically, PHNETs have a clinical presentation that is distinct from other NETs. They are more frequent in women and more likely to affect the middle-aged population . PHNETs It is very difficult to differentiate PHNETs from other solid hepatic tumors, chiefly hepatocellular carcinoma (HCC) and cholangiocarcinoma, making postoperative histological analysis the main method for final diagnosis. Radiological imaging currently lacks specificity for these tumors, which are often mistaken for more common lesions. Ultrasound, CT, and MRI are all employed for lesion characterization; however, no formal techniques exist that are specific for PHNETs. Gross radiographic features can be highly variable with some lesions appearing solid or cystic, as well as having well-circumscribed or diffuse margins. Areas of necrosis have also been described . BecauseLimited CT and MRI characterization of PHNETs has revealed enhancement patterns similar to hepatic NET metastases from the pancreas or gastrointestinal tract . On MRI,Octreotide scanning has been shown to be a useful technique for identifying NETs, with a sensitivity of up to 90% and specificity near 83% , 12. In Immunohistochemistry is performed as the definitive diagnosis for PHNETs, typically after they have already been resected. NETs have previously been shown to be associated with immunoreactivity for chromogranin A, neuron specific enolase, and synaptophysin . The tumTreatment of PHNET has been largely reliant on surgery, with hepatectomy being the most effective treatment of localized tumors . TransarAn overlap of imaging findings with other hepatic neoplasms means that diagnosis of PHNET continues to rest primarily on pathological analysis and immunohistochemistry following biopsy or resection. Given the typical lack of neuroendocrine symptoms as well as a nonspecific and variable radiographic appearance, PHNET will undoubtedly rarely be the favored diagnosis of a hepatic lesion discovered on imaging. However, it remains important to consider a NET when developing a differential diagnosis, especially if the lesion is not entirely typical of the other more common tumors.Once a hepatic NET has been confirmed, a final diagnosis of PHNET must be that of exclusion. Only once a primary extrahepatic source is excluded can the diagnosis be made confidently. Surgery continues to be the treatment of choice for these rare tumors.


Proliferating trichilemmal tumors (PTTs) are benign adnexal skin neoplasms that arise from the outer root sheath of the hair follicle. These tumors are most commonly observed on the scalp and occur, most of the time, in elderly women. Malignant transformation of these neoplasms is a rare event; less than 50 cases have been reported in the English medical literature. We present the case of a 39-year-old Hispanic woman with a tumor located on the skin of one of her breasts that in her third surgical procedure the histologic examination revealed the presence of a malignant proliferating trichilemmal tumor (MPTT). Furthermore, a review of the medical literature and a discussion of the clinical and pathologic features of this rare entity are provided. Proliferating trichilemmal tumors (PTTs) are benign adnexal skin neoplasms that arise from the outer root sheath of the hair follicle. Most of these tumors arise within the wall of a preexisting trichilemmal cyst . MoreovePTTs encompass only 0.1% of all skin tumors. Additionally, most of the patients that present these lesions are elderly women, and in 90% of the cases these tumors occur on the scalp . PTTs raIn this paper, the first case reported in Latin America regarding the malignant transformation of a PTT, we present the case of a woman with a tumor located on the skin of one of her breasts that in her third surgical procedure the histologic examination revealed the presence of a MPTT. In addition, a literature review and a discussion of the clinical and pathologic features of this rare entity are provided.A 39-year-old Hispanic woman was referred to our hospital due to the sudden appearance of a painful, mobile, fixed mass of about 5 cm in diameter in the internal upper quadrant of her right breast. The lesion was classified as a breast cyst, and a puncture of it was performed. Hyaline fluid was aspirated from the mass, with a subsequent decrease in its size. After two months, in the next follow-up, the growth reappeared. Therefore, the mass was surgically resected. The resected lesion was sent to the pathology department and a diagnosis of chronic granulomatous mastitis and fat necrosis was made.Three months after the surgery, the patient presented again for an evaluation due to the presence of a keloid scar of about 5 cm. When the breast was examined a 2 cm nodule located under the scar was palpated. The mass had well-defined borders and was not fixed to superficial or deep tissue . A breasThe patient underwent surgery in order to remove the tumorous growth. The lump was excised with amplified margins of 2 cm. The macroscopic appearance of the tumor was of a pale yellow, firm, solid, lobulated, poorly defined mass, of 2.8 cm in length and 2.5 in width, covered by mature adipose tissue . MicroscPTTs are rare, and the malignant transformation of these tumors is a rarer pathological finding. Moreover, only two cases in the literature have reported the presence of a MPTT over the skin of the breast , 9. PTTsHistologically, PTTs are characterized by an abrupt transition of the nucleated epithelium to anucleated keratinized cells without a granular layer, a phenomenon known as trichilemmal keratinization. MPTTs can occur de novo but most often occur due to the malignant transformation of a PTT. A stepwise transformation of MPTTs has been described from an adenomatous to an epitheliomatous and then to a carcinomatous stage . Additio(i) Group 1 Tumors. They are considered completely benign and present minimal nuclear atypia, trichilemmal keratinization, and stromal invasion with mononuclear cells, plasma cells, lymphocytes, and giant cells.(ii) Group 2 Tumors. They are considered locally aggressive and present irregular and local invasive contours, with moderate cytological atypia, foci of single cell necrosis, and abrupt keratinization with desmoplastic stromal response.(iii) Group 3 Tumors. They are considered malignant and present marked nuclear polymorphism, atypical mitosis, foci of single cell necrosis, abrupt keratinization, and lymphovascular invasion.Even though the diagnosis of MPTTs is mostly done in a histopathological fashion, the use of immunohistochemistry can help to distinguish MPTTs from PTTs and SCCs. CD34 is a marker that is closely associated with trichilemmal keratinization, and because it is absent in SCCs and present in MPTTs it can help in differentiating one condition from the other (an important distinction because MPTTs have a greater chance of recurring and metastasizing) , 12. As In order to evaluate possible local and distant metastases, imaging studies are warranted. It has been reported that on imaging studies MPTTs can manifest as either a cystic or a solid mass. Computed tomography scan is useful for the evaluation of focal bone involvement and erosion, and assessment of possible metastases; meanwhile, magnetic resonance imaging is reserved to evaluate soft tissue infiltration and signs of malignancy. Malignancy findings include poorly defined margins, penetration of tissue planes, and local invasion , 4.As with other skin lesions, surgical excision remains the treatment of choice. Wide excision with margins of at least 1 cm is recommended . With a Due to the rarity of MPTTs, the efficacy of alternative treatments cannot be evaluated. In cases with distant metastases the use of CAV  chemotherapy, a regimen that has been used for advanced squamous cell carcinoma, has been attempted. Nonetheless, the results have not been promising . TherefoWe report a case of a MPTT that occurred in an atypical location, which represented a clinical challenge in both the diagnosis and definitive treatment, due to the nonspecific clinical presentation and rarity of this condition. The recurrences that the patient presented may have occurred due to the fact that excision margins of less than 1 cm were used in the previous resection. Furthermore, it is important to state that the clinician should be alert to this diagnosis, especially in the presence of a cyst with recent rapid growth after remaining unchanged for a long time and tendency to recur after the tumor has been excised.


Spinal paragangliomas are very rare neuroendocrine tumors often presenting with low back pain and radicular symptoms; once resected, they often show benign clinical outcomes. Radiographically spinal paragangliomas mimic more commonly described tumors, such as ependymomas, schwannomas, meningiomas, and even hemangiomas, but a “salt and pepper” appearance related to a serpiginous vascular structure is instructive. Indeed, the rarity of this tumor makes the diagnosis rather challenging radiographically. Graded as a WHO Grade I tumor, they are slow-growing with low proliferation indices. Gross total resection is the mainstay of operative treatment but is often limited by tumor adherence to functional nerves. Here, we present a case of this rare tumor and its management, including a review of the pathology and literature related to this tumor. Paragangliomas are neoplasms originating from the autonomic nervous system, found generally in adrenal and extra-adrenal locations . Extra-aThis is a case of a 54-year-old man with a history of diabetes mellitus Type 2 and hypertension with a four-month history of back pain, radiating to his buttocks, worse on the right compared to the left. He denied any weakness, numbness, or tingling in his lower extremities. The pain was rated 10/10 upon initial presentation but decreased to 3/10 upon presentation for neurosurgical evaluation. Two months after the initial onset of pain, he was bedbound and incapacitated, unable to fulfill activities of daily living.Other than back pain, this patient complained of urinary hesitancy upon initiation and bearing down to initiate bowel movements caused a lancinating pain in his back. He denied any changes in perineal sensation but has had erectile dysfunction, which has been attributed to low testosterone for which is he undergoing replacement therapy.This patient presented to our clinic in May 2015 with a complaint of minimal back pain and a localized patch of numbness on the lateral aspect of his right leg. His neurologic review was negative. His social habits were negative for illicit drug use or alcohol use; he is a former smoker of 22.5 pack-years and denies any exposures. Family history was negative for schwannomas, neurofibromas, or other brain and spinal cord tumors; his father died of renal cell carcinoma. He reports having had multiple skin and subcutaneous lesions in his arms, legs, and trunk, one of which was biopsied and diagnosed as benign without further specification.On physical examination, he demonstrated normal vital signs. His heart, lung, abdominal, and skin examinations were normal. Cranial nerves, 2-12, were intact as was his motor exam in all four extremities. He experienced a patch of numbness on the lateral aspect of his right calf. His reflexes were 2+ throughout and without clonus. His gait was narrow-based and normal.Neuroimaging with an MRI of the lumbar spine with and without contrast demonstrated an intradural mass at the level of the conus measuring 1.4 x 1.1 x 2.2 cm with heterogeneous contrast enhancement , and the microscissors. Upon stimulating the tumor capsule, it was apparent that the S1 nerve roots were adherent to the tumor, and thus, the decision was made to leave some residual tumor in order to preserve S1 nerve function. Therefore, a near gross total resection was completed , and a dural closure was performed after meticulous intradural hemostasis was obtained. Hemostasis was then achieved in the surgical cavity and copious antibiotic irrigation was applied. The muscle, fascia, subdermal, and dermal layers were subsequently closed in a standard fashion.The patient was then turned to the supine position and extubated. His immediate postoperative examination demonstrated a stable preoperative exam, including 5/5 strength in his bilateral lower extremities. Histologic sections demonstrated a neuroendocrine neoplasm with architectural features of a paraganglia, including Zellballen configurations of neoplastic neuroendocrine cells surrounded by delicate vessels and occasional sustentacular cells Figure .The neoplastic cells showed amphophilic to mildly eosinophilic cytoplasm and regular round nuclei with finely stippled chromatin. A few scattered mitotic figures were seen. Immunostains for synaptophysin, chromogranin, and S100 were positive in the neoplastic cells. S100 highlights scattered few sustentacular cells and nerve fibers with intense staining.Patient MA was seen three weeks postoperatively in our neurosurgery outpatient clinic with minimal incisional pain and muscle spasms. He had self-titrated on narcotics and antispasmodics with a desire to return to work. Since resection, his initial preoperative complaint of right leg pain and numbness had resolved. He denied any paresis, numbness, or incontinence.On neurological assessment, his examination was intact. Postoperative imaging was deferred at the time of this postoperative evaluation with a plan to undergo magnetic resonance imaging at the three-month postoperative time point; at that time, should progression be seen, we would consider stereotactic radiosurgical boost to the small residual at the S1 nerve. He was advised that he could return to light work and activity and begin physical therapy.Paragangliomas are neoplasms originating from the autonomic nervous system, found generally in adrenal and extra-adrenal locations . Extra-aParagangliomas are classified as World Health Organization (WHO) Grade I given their characteristically slow growth and histologically benign appearance. As they are neurogenic and arising from the autonomic nervous system, they are sub-classified into sympathetic – secreting catecholamines – or parasympathetic and thus functionally non-secretory , 19. In As a subset, spinal paragangliomas are even less common and generally occur at the lumbosacral extent of the spine without reported variation in distribution , 4. TherPreoperative diagnosis is determined using magnetic resonance imaging, but due to the rarity of paragangliomas, radiographic diagnosis is challenging. Early on, the MRI characteristic of paragangliomas had been described -29. On MSurgical pathology provides the definitive diagnosis of resected lesions. A “Zellballen” pattern as a solid mass of the paraganglion-type nests or cords is often described -3, 15. ILong-term follow-up data for management of spinal paragangliomas is sparse. In the largest analysis of spinal paragangliomas, Xu and colleagues followed 19 cases and determined the mean age of presentation to be 47.7 years. Initial presenting symptoms typically included back pain with sciatica, which immediately resolved with operative resection; three of five cases with incomplete resection showed recurrence of disease at 62.1 months . Gross tData for adjunct treatment modalities also remains sparse. Conventional radiotherapy affords control rates of 90-100% for intracranial paragangliomas and is often advocated as a first-line therapy for asymptomatic patients with intracranial paragangliomas -33.Spinal paragangliomas are a very rare entity, often presenting with low back pain and radiculopathic symptoms. Their clinical course is generally benign with operative resection as the first-line treatment with nearly immediate improvement in symptoms. Radiographic characteristics include a vascular, “salt and pepper” appearance with prodigious flow voids and an occasional hemosiderin rim. Gross total resection is favored when possible with the realization that often-functional nerve roots may complicate the ability to fully resect the lesion. The role of adjunctive radiotherapy is unknown but, borrowing from the intracranial paraganglioma literature, may be useful in long-term progression management.


Heterotopic pancreas is an aberration in the developmental profile of the pancreas. This entity is rarely symptomatic and its malignant transformation is even rarer. However, when present, it poses a diagnostic dilemma to clinicians, as little help comes from gastroenteroscopy and imaging. Surgical exploration remains the only option at times, and it is the histopathological examination that finally clears the mist. This case report reveals the elusive nature of malignancy in heterotopic pancreas in the duodenum. Heterotopic pancreas (HP) is the presence of pancreatic tissue outside the normal anatomical boundaries, with no vascular connection with the main pancreas. Several theories have tried to define this aberration from normal embryological development, but none stands up to scrutiny . Though A 51-year-old male, with no known comorbidities, presented to us with history of nausea, pain abdomen and post-prandial vomiting for previous three months. Initially he used to have 2–3 episodes of post-prandial vomiting, which progressively increased to 8–10 episodes over a period of three months. He experienced a weight loss of 19 kilograms in the same time. There was no history of distension of abdomen, fever, haematemesis or melena. The physical examination was within normal limits and he was haemodynamically stable. Haematological and biochemical reports were normal.An upper gastrointestinal endoscopy revealed oesophagitis, with a dilated stomach and first and second parts of the duodenum, and a fleshy, mucosal growth in the medial wall of the distal end of the third part of duodenum . The endThrough a mid-line laparotomy, the duodenal lesion was subjected to a segmental resection and end-to-side duodenojejunostomy with a distal feeding jejunostomy. The resected lesion measured 5 × 4 × 3.5 cm and involved the fourth part of the duodenum. A 2 × 1 cm suspected malignant nodule was also resected from the jejunal mesentry, approximately 3 cm from the duodeno-jejunal flexure  and 4. HThe credit for first description of HP goes to Jean Schultz in 1727 . HoweverAlthough its cause is unclear, three theories have been put forward. The ‘theory of metaplasia’ states that it is the metaplasia in the pancreatic endodermal tissue, during embryological development, that leads to HP , while tHP is usually an asymptomatic aberration but, as and when clinical symptoms arise, the manifestations are site-specific. The usual symptoms are nausea, vomiting and abdominal epigastric pain, increasing in severity in the post-prandial period. Malignant transformation of the HP is exceedingly rare. All disease conditions that can affect the normal pancreatic tissue, such as pancreatitis and pseudocyst formation, can occur in HP. Very few cases of duodenal adenocarcinoma arising from HP have been reported in the world literature , 12. Owiet al. Type I had ducts, acini and Langerhans cells, similar to normal pancreas. Type II had ducts only and Type III showed acini and type IV islets [Confirmatory diagnosis is usually histological. Histologically, HP has been classified into three types by Heinrich in 1909 . Type I V islets .With regard to management, a surgical intervention is essential in a symptomatic patient. Although open surgery was once preferred, the recent world literature boasts several reports of successful laparoscopic and endoscopic interventions . HoweverIn conclusion, we have described a case of adenocarcinoma arising from HP in the duodenum. This phenomenon is extremely rare and difficult to diagnose pre-operatively. The clinical profile of the patient included nausea, post-prandial vomiting and significant weight loss. The endoscopic findings were inconclusive, as were the biopsies. The diagnosis was established through histopathology after surgery. The rarity of this condition makes the diagnosis and treatment a herculean challenge. We require more detailed and in-depth studies on this matter to solve the elusive riddle of HP.Conflict of interest: none declared.


A primary clear cell adenocarcinoma of the colon is a rare oncologic entity. The current study presents a case of such a tumor in the transverse colon of a 26-year-old male, and describes the computed tomography features of the neoplasm. The tumor appeared as an extensive extracolic mass, which displaced the loop of the small bowel and pancreas, and invaded the spleen. A laparotomy was performed and a huge mass measuring 12 cm maximally was revealed, arising from the transverse colon close to the left colonic flexure, with invasion of the spleen. The tumor and the spleen were resected concurrently. Histopathological examination of the excised mass revealed features of clear cell adenocarcinoma. A primary clear cell adenocarcinoma of the colon is a rare tumor, with only 13 cases reported in the English literature at present. The present case is reported here due to its rarity. A clear cell carcinoma of the colorectum is a rare oncologic entity and, thus, the clinical and imaging features of such have not been adequately investigated. Thus far, only 13 cases have been documented in the English literature, according to the results of searches using PubMed. The majority of reported primary colonic clear cell adenocarcinomas are located in the left colon and are more common in elderly men . ClinicaA 26-year-old male was admitted to Yantai Yuhuangding Hospital  with a palpable abdominal mass, which had gradually increased in size for three months. The patient had no history of abdominal pain, melena, body weight loss or change in bowel habits. Standard blood tests and chest X-rays revealed no abnormalities. Ultrasound of the abdomen showed no abnormal findings from the liver and gall bladder, pancreas, testes and genital tract or kidneys. General examination was negative, with the exception of an extensive, immobile mass (13 cm in diameter) in the left upper quadrant of the abdomen.CT examination of the abdomen was performed prior to and following the administration of an intravenous contrast agent. Sagittal and coronal reformatted images were obtained. Abdominal CT revealed an ill-defined mass (12×10 cm in size) located in the left upper quadrant. On unenhanced images the mass was hypo-attenuated in relation to the liver . The masDuring surgery, an extensive mass, measuring 12 cm maximally, was found arising from the transverse colon close to the left colonic flexure, with invasion of the spleen. The tumor was exophytic, with a lobulated and irregular surface. The cross section revealed transmural invasion. The tumor and the spleen were resected concurrently.Histological examination of the resected specimen revealed a tumor entirely composed of polygonal and oval cells arranged in lobules, which were separated by fibrous septa containing chronic inflammatory cells. The cells had abundant cytoplasm, which varied from clear to eosinophilic. The nuclei included one or more prominent vesicular and pleomorphic nucleoli . ImmunohThe patient improved and was discharged 10 days following surgery. However, one and a half years following surgery, the tumor recurred in the peritoneal cavity, at the site of the left colonic flexure. The patient succumbed to the disease three years following surgery.Clear cell adenocarcinomas are well characterized in the kidneys , lower uRegarding the clinical characteristics, more male patients exhibiting clear cell adenocarcinoma of the colorectum have been reported, and the tumor tends to be located on the left side , which iIn contrast to previous studies, the patient in the current case presented with a palpable abdominal mass without exhibiting any symptoms that indicated colonic adenocarcinoma, including melena, a change in bowel habits or symptoms of bowel obstruction. This may be associated with the exophytic growth pattern of the tumor. Due to the absence of specific clinical manifestations, the tumor was not identified until it had enlarged enough to be palpated. This condition is uncommon for a primary colonic adenocarcinoma.On CT imaging, the present tumor appeared as an extensive extracolic mass in the left upper quadrant, which encased the left part of the transverse colon, displaced the loop of small bowel and the pancreas, and invaded the spleen. It was difficult to identify the origin of the mass due to its large size and prominent extraluminal location. However, the tumor site and its association with the transverse colon may provide an indication.Noteworthy considerations in the differential diagnosis of primary clear cell adenocarcinoma of the colon are metastases from other organs, including the kidneys, lower urinary tract, ovaries and female genital tract. The majority of reported cases of clear cell adenocarcinoma in the colon have been identified later as metastases originating from renal clear cell malignancies, which may occur 16 years following resection . In the Clinical data regarding tumor-associated mortality and disease-free survival in patients with primary clear cell adenocarcinoma of the colorectum is rare, as a result of the short-term follow up and small number of cases. Previous studies have indicated that the behavior of this tumor is not significantly different from that of conventional intestinal carcinomas; however, further studies are required to investigate this.In conclusion, in this study, a rare case of clear cell adenocarcinoma of the colon in a young male was presented. The case was reported due to rarity, and the clinical and computed tomography features were analyzed. The tumor presented as an extensive extracolic mass, which is uncommon for a primary colonic tumor. The predominantly extraluminal growth pattern exhibited by the tumor, as well as the patient’s clinical characteristics, may be key features of advanced colonic clear cell adenocarcinoma.


Oncocytoma of major salivary gland is a fairly common benign tumour encountered, but its occurrence in oral minor salivary gland is a rare entity. Here we report a case of a giant minor salivary gland oncocytoma mimicking a jaw tumour which was successfully excised along with a review of literature. Oncocytoma is a benign tumor arising from the oncocytes which line the duct of salivary glands and are fairly common in major salivary glands. They occur very rarely in the minor salivary glands. Histologically, World Health Organization (WHO) 1991) classified them into three distinct types: oncocytosis, oncocytoma, and oncocytic carcinoma. Oncocytoma is also known by oxyphilic adenoma and oxyphilic granular cell adenoma. Oncocytoma is a rare benign salivary gland neoplasm composed of large epithelial cells with characteristic bright eosinophilic granular cytoplasm (oncocytic cells). It accounts for approximately 0.4–1% of all salivary gland neoplasms, occurring primarily in parotid glands, with only a small percentage occurring in minor salivary glands of palate, tonsillar fossae, larynx, nasal cavity, maxillary sinus, and the lacrimal gland. It occurs primarily in persons older than 50 years of age. Only 17 cases of histologically verified oncocytoma of an intraoral minor salivary gland are reported in literature with the 18th case being reported by Palakshappa et al. in 2014 [991 classA 53-year-old female patient presented to the outpatient department of E.N.T. at the Barasat Cancer Hospital, Kolkata, India, with complaints of a slowly growing large swelling in the left lower jaw for a duration of 14 months. There was slight difficulty in chewing and swallowing due to the size of the tumor. On inspection a firm smooth lobulated mass was seen arising from the lingual aspect of the left side of lower jaw of 5 cm by 2 cm in size extending from canine to the premolar teeth occupying the edentulous portion of left half of mandible and adjacent floor of mouth. The mass was painless and nontender on palpation. The teeth on the left half of mandible were loose and patient was unable to approximate the jaws completely; hence, he could not chew or swallow effectively.The rest of oral cavity oropharynx and laryngopharynx were normal on examination. There were no palpable cervical lymph nodes. Other than diabetes patient had no significant systemic ailments. Other than elevated blood sugar the routine haemogram, serological studies, X-ray of chest, and ECG were within normal limits. An orthopantomogram of the jaw showed a lobulated swelling in the left side of floor of mouth. The hyperglycemia was controlled with insulin therapy on admission of the patient. A punch biopsy from the mass done elsewhere was reported as oncocytoma.The patient was planned for surgery. By a left lip splitting cervical collar line incision a flap was elevated and tumor was exposed. The tumor was attached to the inner  aspect of the left half of mandible encroaching upon the edentulous part of the mandible. The tumor was excised using electrosurgical instrument along with a marginal mandibulectomy of the involved part of the mandible. The mucosal flaps were elevated from the floor of mouth and buccal mucosa was released to bury the mandibular defect. The wound was closed in layers over corrugated rubber drain. The patient was kept on broad spectrum antibiotics, anti-inflammatory agents, and insulin on a sliding scale along with antiseptic mouth washes. The postoperative period was uneventful. Patient recovered well and was discharged from hospital 10 days after the removal of sutures.Oncocytoma is a rare benign tumor of the salivary gland representing not more than 1% of salivary tumors. It is composed of large epithelial cells, the oncocytes, which are predominantly found, in senior adults, being more prevalent in the eighth decade of life. It is located mainly in larger salivary glands especially in parotid glands . Among oIn our case we encountered a slowly growing large swelling in the left lower jaw for a duration of 14 months. Following all preoperative evaluation, the patient underwent surgery. The excision biopsy was sent for histopathological examination that turned out to be “oncocytoma of minor salivary glands.” Patient is doing well postoperatively and is on follow-up for the last one year with no evidence of recurrence. This case is being reported due to its rarity and unusually large size of presentation.


Various presentations of essential palatal myoclonus, a condition characterized by clicking noises and palatal muscle spasm, have been reported in the literature. We are reporting the first case of essential palatal myoclonus following dental treatment.A 31-year-old Caucasian man presented to our Ear, Nose and Throat department complaining of objective clicking tinnitus occurring immediately after he had undergone root canal treatment on his right lower third molar 3 months ago. Magnetic resonance imaging of his head revealed no abnormalities in the cerebrum, cerebellum or brainstem making the diagnosis essential palatal myoclonus. He returned a week later, and 20 units of botulinum toxin A (Allergan) were injected into his left tensor veli palatine muscle. He reported an immediate improvement; however, symptoms recurred 6 months later.Dental treatment can be a trigger of essential palatal myoclonus. Botulinum toxin injections are an effective treatment for short-term relief of symptoms. Palatal myoclonus  is a rare condition affecting the muscles of the soft palate. It typically presents with clicking noises and muscle spasms felt at the back of the throat. PM is divided into two subtypes: essential (EPM) and symptomatic (SPM). EPM describes an objective clicking secondary to rhythmic movements of the tensor veli palatini (TVP), occurring intermittently throughout the day but not during sleep. PreviouSPM involves the levator veli palatini muscles Figure , and is We report the first case of EPM following dental treatment.A 31-year-old Caucasian man presented to our Ear, Nose and Throat department complaining of bilateral objective clicking tinnitus occurring immediately after he had undergone root canal treatment on his right lower third molar 3 months ago. The dental surgery was uneventful but the patient felt his mouth was over-extended during the procedure. The tinnitus was constant and he could not exert any control over his symptoms. He reported his symptoms were bilateral but alternating in severity between the right and left sides. As the patient lived alone, it was not known whether his symptoms occurred during sleep. He denied any symptoms of the pharynx, larynx or esophagus or any other auditory symptoms. He appeared anxious but there was no significant past medical history. He was not taking any regular medications.Examination of the patient revealed objective clicking predominantly on the right side. On inspection of his oral cavity, muscle spasms of his soft palate were observed. He was subsequently sent for magnetic resonance imaging of his head, which revealed no abnormalities in the cerebrum, cerebellum or brainstem making the diagnosis EPM. He returned a week later, and 20 units of botulinum toxin A (Allergan) were injected into his left tensor veli palatine muscle. He reported an immediate improvement with no side effects; however, symptoms had recurred at follow-up 6 months later. He was subsequently referred for consideration of radio-frequency ablation with the aim of achieving long-term symptom relief.As previously mentioned, the primary muscle affected in EPM is the TVP. The left and right TVP share a common aponeurosis which extends between the pterygoid hamuli. This anatomical connection provides an explanation for the typically bilateral symptoms of EPM.Psychogenic PM is a subtype of EPM which has a similar presentation. Physical triggers for the condition such as endoscopy have been reported in this condition. HoweverThe exact precipitant of PM in this case is unclear. Wisdom tooth surgery is occasionally complicated by damage to the branches of the mandibular nerve, most commonly the inferior alveolar and lingual nerves, but not the medial pterygoid nerve which supplies the TVP. However, this case raises the possibility of medial pterygoid nerve damage following dental surgery as a cause of EPM.Botulinum toxin is the only pharmacological therapy which has consistently been proven to be effective in the treatment of PM. Up to 3Systemic treatments that have been evaluated in the management of PM include phenytoin, barbiturates, benzodiazepines, carbamazepine, sodium valproate and anticholinergics, however, many of these drugs are associated with troublesome sedative and metabolic side effects. A previDental treatment can be a trigger of EPM. Botulinum toxin injection is an effective treatment for short-term relief of symptoms.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.EPM: Essential palatal myoclonus; PM: Palatal myoclonus; SPM: Symptomatic palatal myoclonus; TVP: Tensor veli palatini.The authors declare that they have no competing interests.AB was the consultant responsible for diagnosing and treating the patient in this case report. AB provided the information to JL and MF, who wrote the paper under AB’s supervision. All authors have read and approved the final version of this manuscript.


We report the diagnostic and successful therapeutic images of an acute occlusion of the abdominal and suprarenal aorta. This lesion is a rare but catastrophic pathology which can cause severe ischemic manifestations, depending on the site of obstruction, with high rate of mortality even after treatment. In the majority of cases it represents a surgical emergency. Although the mechanism of the thrombosis has not been delineated, the proposed etiologies include propagation of thrombus from distal artery occlusion, cardiac thromboembolism, dislodgment of a mural thrombus, or coagulation disorders. Frequent risk factors include advanced atherosclerosis combined with a low flow state because of poor cardiac performance. The management of this condition includes immediate intervention with systemic heparinization, improvement of the cardiac condition, and surgical revascularization based on the clinical and anatomical presentation. In this case the authors highlight the importance of an early detection and early intervention to enhance survival rates and reduce morbidity. A 71-year-old white male, with a history of heart failure, hypertension, hypercholesterolemia, and major abdominal surgery, was admitted to the emergency room hemodynamically stable with complaint of sudden and severe pain in the lower back and both lower limbs, which was accompanied by lower limb cyanosis, paresthesia, numbness, and progressively paralysis. No recent trauma was reported. Computed tomography (CT) scan revealed occlusion of the infrarenal abdominal aorta and bilateral iliac  arteries with retrograde partial propagation of the aortic thrombus along the aortic posterior wall up to the level of the patent celiac trunk . No aneuAcute aortic occlusion is a rare but catastrophic pathology with high hospital mortality and morbidity even after early recognition and intervention . It may


Laryngeal chondrosarcoma (CS) is a very rare entity. It is usually seen in 50–80-year olds. It is developed from cricoid cartilage largely. Patients have laryngeal CS complaint of respiratuvar distress, dysphonia, and dysphagia generally. A submucous mass is usually seen in physical examination with an intact mucosa. Distant metastasis is rare in CSs. Main treatment is surgical excision. An 82-year-old patient who has respiratuvar distress is presented in this paper and laryngeal CS is reviewed in the light of the literature. Laryngeal CS is a rare entity and it is less than 1% of all larynx cancers . The youThe aim of this paper is to present a laryngeal CS case and review its diagnosis and treatment in the light of the literature.An 82-year-old patient was admitted to our clinic with a complaint of shortness of breath. She had for 2 years this symptom and it had been worsened. She also had hoarseness and difficulty in swallowing. In her medical history, she was being treated for hypertension and asthma. She was not a smoker or an alcoholic. There was no malignant disease in her relatives. She had stridor and supraclavicular-suprasternal retractions. Her subglottic area was very narrow in physical examination. There was a mass but mucosa on mass was steady. Her laryngeal airway was narrowed by mass in form of expansion . There wLaryngeal CS is most frequently seen in the range of 50–80 years. Male to female ratio is 4/1. Its etiology is still unknown but the most acceptable theory is irregular ossification of laryngeal cartilages. Radiotherapy to head-neck area and Teflon injection can also be a reason . The pre75% of laryngeal CS develops from the cricoid cartilage like the presented patient and 17% from the thyroid cartilage . ClinicaA mass covering intact laryngeal mucosa is seen in endoscopic examination. But in advanced cases mucosal ulceration can also be seen . In CT, There is no reliable evidence to differentiate between benign chordoma and low-grade CS radiologically . Both maFor the definitive diagnosis, direct laryngoscopy and biopsy must be done, because submucosal, deep biopsy should be performed . DiffereThe main treatment of laryngeal cartilaginous tumors is surgical excision. Surgery that protects laryngeal functions is recommended if possible . But 75%Primary radiotherapy is not recommended for the treatment. A study involving 12 patients who had primary radiotherapy for laryngeal CS showed long-term remission in only two of the patients . LaryngeAbout 2–10% of the reported cases are described as metastatic disease. Lung, bone, and liver metastases have been reported . MortaliLaryngeal CS is a very rare and slow-growing entity. Its distant metastasis is also rare. Its symptoms can be confused with asthma and laryngitis. So we must be careful for the differential diagnosis especially in elderly patients.


Penile urethral stones are a rare occurrence resulting from a number of causes including migration of stones within the urinary tract, urethral strictures, meatal stenosis, and obstructing tumours such as adenomatous metaplasia of the uroepithelium, hypospadias, urethral diverticulum, and very rarely primary fossa navicularis calculi. We report the case of a 54-year-old male presenting with penile gangrene and sepsis resulting from impaction of multiple stones within the penile urethra. This paper summarises the topic and discusses the pathophysiology of this unusual condition. Penile urethral stones are a rare occurrence with an incidence of less than 0.3% , resultiA 54-year-old male presented to the Accident and Emergency Department with inability to pass urine and a painful, foul-smelling, and swollen penis for 3 days. He had no known medical problems and was not diabetic. On admission, the skin of the penis was partially necrotic with progressing gangrene, it was malodourous, and the abdominal wall and scrotum appeared normal.Ultrasound, retrograde urethrogram, intravenous urogram, and urethroscopy were done within 24 hours. These confirmed a normal upper urinary tract and bladder and showed obstruction of the penile urethra secondary to multiple calculi; however, the calculi were embedded and the tissues were grossly inflamed and ulcerated. He was started on intravenous ampicillin, gentamicin, and metronidazole which were continued for 5 days during his hospital stay followed by amoxicillin/clavulanic acid upon discharge for a further week.The patient consented for extraction of the urethral stones as well as aggressive debridement of the penile tissues with the risk of amputation. The exploration was done under general anaesthesia and an incision was done on the ventral  surface of the penis. Through a ventral urethrotomy, three urethral stones were successfully removed and the soft tissues of the penis aggressively debrided. A suprapubic cystostomy was also done and a catheter was inserted at this level (see Figures Penile urethral calculus is a rare form of urolithiasis with an incidence lower than 0.3%. Patients may present with acute urinary retention, interrupted or weak stream, gross haematuria, or pain affecting the penis, urethra, or perineum.Etiological factors include a range of pathologies including the migration of calculi originating higher up in the urinary tract, penile urethral strictures which are strongly associated with chronic infections of the urethra , 3, tumoEven rarer associations have been documented whereby urethral calculi form within the fossa navicularis which is the dilated portion of the male urethra just distal to the narrower cavernous portion . It is aIn an analysis done by Verit et al. in 2006 there were fifteen patients studied, eight of which were paediatric cases affecting the fossa navicularis. The stones were all fusiform in shape and solitary. Six of these also had urethral pathologies including hypospadias and one case with a diverticulum.The patient herein presented in this case report had clinical features of anuria, penile and perineal pain, and penile gangrene due to obstruction. There was a chronic inflammatory process at the time and quite possibly in the weeks leading up to the acute event. The stones at the time of surgery were embedded in the urethra making it difficult and near impossible to be removed via standard minimally invasive techniques such as urethroscopy.This case highlights two main points, the first emphasizing that there is still a place for performing open operations especially in this difficult situation with chronic, impacted, and multiple stones as well as being able to efficiently debride and control sepsis and the second noting that this association with gangrene and urethral stones has not been previously documented clearly in the surgical literature. A standard search of the medical literature on PubMed reveals no article associating penile gangrene with urethral stone disease to the best of our knowledge. We hope this case is useful for our colleagues practising in the field of emergency medicine, nursing and urology and brings attention to this particular condition and its management.


Mesenchymal chondrosarcoma is a rare malignant tumor arising from bone or soft tissues. Instraspinal dumbbell-shaped mesenchymal chondrosarcoma is even rarer; however, it should not be neglected by clinicians.A 26-year-old female was referred to our hospital with a 1.5-month history of sciatic pain and numbness in the left leg. Computed tomography and magnetic resonance imaging scans revealed an intraspinal dumbbell-shaped mass which had distinguishing features of neurogenic tumors, surprisingly, with massive calcifications, and no tumor metastasis was found. Then the patient underwent a total resection of the tumor, and during the operation, we found that the right nerve root of the fifth lumbar almost disappeared. The tumor was diagnosed as mesenchymal chondrosarcoma by histopathological examination after operation. Adjuvant therapies were not performed. However, recurrence of the tumor occurred 5 months later, and she underwent a total resection again combined with radiotherapy after second surgery.To the best of our knowledge, this case study presents the first report in literature about primary instraspinal dumbbell-shaped mesenchymal chondrosarcoma with massive calcifications, which may provide some evidence for clinical practice. As the clinical symptoms and radiographic findings of mesenchymal chondrosarcoma are usually not specific, clinicians should consider it as a possible case and diagnose it through careful histopathological examination. Sometimes, calcification could be seen in tumors, which may influence or reflect the growth of tumor and disease prognosis. Although prognosis in mesenchymal chondrosarcoma varies from person to person, generally, complete resection, adjuvant therapy, and regular examinations are recommended to perform for patients with mesenchymal chondrosarcoma. Mesenchymal chondrosarcoma (MCS) is a malignant tumor arising from bone or soft tissues , 2, whosA 26-year-old female patient suffering from sciatic pain and numbness in the left leg with a 1.5-month history was admitted to our department after a magnetic resonance imaging (MRI) scan which showed a tumor in her lumbar spinal canal. She had trouble in walking because of the pain, which can be relieved by maintaining on the right side with coxa and knee bending. Besides, she complained of urgency of urination and irregular bowel movement. Physical examination showed kyphosis of the lumbar spine, muscle atrophy of the left triceps surae, weakness of the anal sphincter, drop foot deformity on the right, hyporeflexia of the patellar tendon reflex on the left, absence of the Achilles tendon reflex, and the straight leg raising test, Yeoman test, and Ely test were positive. The muscle strength was 4/5 in left anterior tibial and extensor hallucis longus, while 0/5 in right. Signs and symptoms of drop foot, bladder, and bowel dysfunction were suspected to cauda equina syndrome, but she told us that she had the difficulty of dorsiflexion on the right foot since she was 14 years old, and aggravated gradually.Further checks were performed after admission. Results of laboratory tests including complete blood count, electrolytes, biochemical profile, erythrocyte sedimentation rate, and C-reactive protein were all normal. MRI revealed an intraspinal dumbbell-shaped mass measuring 1.8 × 2.3 × 6.2 cm with well-defined margin which extended from central canal to the intervertebral foramen. The tumor pushed forwards the dural sac, resulting in the cauda equine compressed and curved impression at the posterior of L4 vertebra. The mass showed isointense on T1WI images, low-signal intensity and mildly hyperintense mixed on T2WI images and fat-suppression T2WI, and heterogeneous density on the Gd-DTPA-enhanced T1WI. Computed tomography scans (CT) indicated that most parts of the tumor had calcified which extended towards both sides of intervertebral foramen at L3/4 and left side at L4/5, with intervertebral foramen enlarged, bone cortex thinned, and bone density of vertebral pedicle decreased , CD 34 (vessels), S-100, and NSE (chondroid areas), was used for further diagnosis, whose results were all positive  is a rare malignant chondrosarcoma. It was in 1959 that Lightenstein and Bernstein  reportedHere, we report a 26-year-old female patient with MCS in the lumbar spinal canal. Generally, most of the spinal tumors with dumbbell shape are from nervous system, such as schwannomas or meningiomas . To datePain or swelling of the lesion often leads to the early diagnosis of MCS. However, currently, detection of MCS is often delayed for nonspecific signs and symptoms, unless when the compression of the spinal cord or nerve roots cause initial symptoms of radicular pain, numbness, or muscle weakness. Yet, intraspinal MCS may later have onset of symptoms , probablAlthough imaging examinations like MRI or CT scan, which are already the best choices, can provide some evidence for diagnosis of this tumor , 25, theThe MCS is a hard or fish-meat like, grayish-white or grayish-red mass, mixed with calcifications sometimes , 20. DurThe therapy and prognosis of MCS still remain controversial, especially for the use of postoperative radiotherapy and chemotherapy , 20. InfPrognosis in MCS varies from person to person. Previously, the prognoses of MCS were based on the research of 111 patients of Nakashima et al. . In the Mesenchymal chondrosarcoma (MCS) is a very rare tumor, and it is difficult to distinguish it from many other kinds of tumors. When masses were discovered in spinal canal by imagings, we could not identify it merely through its shape, intensity, and other features. MCS, rare as it is, should also be considered. Actually, sometimes, solitary or punctate calcifications could be seen in tumors, which may influence or reflect the growth of tumor and disease progression. In this case, the patient had a long history of drop foot due to the injury of L5 nerve root and no tumor metastasis, which may be related to its calcification. According to imagings, decisions about making appropriate operation plans seem to be more important than to assume what the tumor is. Through CT or MRI, we can clearly see the margin of the tumor and decide how to remove the tumor. Although we performed a total resection of the tumor, she experienced tumor recurrence half a year later. Hence, complete resection, adjuvant therapy, and regular examinations are recommended to perform for MCS patients.CT, computed tomography scan; L5, the fifth lumbar; MCS, mesenchymal chondrosarcoma; MRI, magnetic resonance imaging scans


Sustained-release fluorouracil (FU) implants have been extensively used in peritoneal interstitial chemotherapy, and during surgery for gastrointestinal tumors, breast cancer and hepatic tumors. Currently, studies regarding the complications associated with sustained-release FU implants are rare. The present study describes the case of a 61-year-old male who presented with a space-occupying lesion of the left lobe of the liver six months after undergoing a radical total gastrectomy. Thus, laparoscopic exploration was performed to remove the tumor. Postoperative histological examination demonstrated that the lesion in the left lobe comprised of necrotic tissue with granulation tissue hyperplasia. Based on the surgical and postoperative histological findings, the mass was proposed to be due to a high concentration of local sustained-release FU implants. Furthermore, the drug was partially surrounded and had been insufficiently metabolized over a long time period, which was proposed to have caused necrosis, proliferation and fibrillation, and induced granuloma. In conclusion, local high concentrations of sustained-release FU implants may be associated with granuloma and this finding may enable improved management of sustained-release FU implants during surgery. In the early 1990s, the concept of interstitial chemotherapy was proposed. Interstitial chemotherapy involves loading antineoplastic agents into degradable or non-degradable vehicles to provide a sustained drug release system to maintain a prolonged drug concentration at the site of implantation, as well as to reduce toxicity and achieve drug targeting –4. The pA 61-year-old male presented with a space-occupying lesion of the left lobe of the liver, which was identified using computed tomography (CT) on July 27, 2013. The patient had undergone a total gastrectomy for an ulcerated adenocarcinoma of the greater curvature of the middle of the stomach six months previously at the Department of General Surgery at the First People’s Hospital of Yangzhou . During the surgery, a 600-mg sustained-released FU implant  was implanted. A post-surgery histological examination revealed a moderately-low differentiated adenocarcinoma. No tenderness or palpable mass was detected in the abdomen of the patient. Upon admission to hospital, laboratory examinations revealed that the serum concentrations of carcinoembryonic antigen, α-feroprotein and carbohydrate antigen 19-9 were 1.6 ng/ml, 2.1 ng/ml and 3.2 IU/ml, respectively.The patient underwent laparoscopic exploration in order to remove the liver tumor. During the surgery, a mass  was observed between the left lobe and the diaphragm, which presented with milky white necrotic matter and a cylindrical granule in the center. Ultrasonography indicated that no other tumors were present in the liver during the surgery.A post-surgery histological examination demonstrated that the mass was granuloma, presenting with tissue necrosis at the center and granulation tissue hyperplasia, which was surrounded by hyperplastic fibrous tissue.Locoregional recurrence and distant metastases are common following surgery for gastric adenocarcinoma. Thus, in the present case, the patient was initially diagnosed with liver metastases based on the history of gastric cancer and the findings of the CT scans. Radiofrequency ablation or alcohol injections are commonly used to treat single, small, metastatic liver lesions from gastric cancer. However, in the present case, the lesion was located on the surface of the left lateral lobe and there was adhesion to the intestine in the local area, thus, laparoscopic exploration rather than local treatment was used. The lesion was resected with minimal trauma.When gastrointestinal tumors metastasize to the liver, enhanced CT of the lesion edge commonly reveals intensification in the early period, however, in the later period, the density is low. In the present case, no obvious change in intensification was observed . This maA sustained-release method of 5-FU adminstration prevents the peak tissue levels that are induced by subconjunctival injections. Another potential advantage of this mode of administration is that 5-FU is delivered directly to the required site of action, further reducing the potential of toxic side-effects.In the present case, the cylindrical granule that was observed at the center of the granuloma was identified to be undegraded sustained-release FU implant polymers . Thus, iBiodegradable and non-biodegradable polymers are often used as implant base materials. The release pattern from biodegradable implants is controlled by the composition and molecular weight of the polymer, as well as the morphology, manufacturing technique and the structure of the implant. With developments in materials science, a novel biodegradable drug carrier, which is constructed using nano-controlled release technology, has been developed, which is capable of accessing the circulation of the human body and reaching the targeted area via vessels and cell absorption. This may be a promising targeting vector for use in the future –7.In conclusion, the present study has shown a rare complication associated with sustained-release FU implants and has illustrated that higher local concentration implants may lead to granuloma. Therefore, the diagnosis of a metastatic tumor should be based on the results of pathological examination.


Granulocytic sarcoma (GS) is a solid tumor that is the extramedullary presentation of acute myelogenous leukemia, other myeloproliferative disorders, or myelodysplastic syndromes. Less commonly, it also may arise as an isolated mass. In this report, we describe a 23-year-old female patient, with a GS in the uterus and we stress the value of diffusion weighted imaging for the evaluation of uterine neoplasms. To our knowledge, our case is the first in the literature to report diffusion weighted imaging (DWI) findings of GS. A 23-year-old female patient came to our clinic for the evaluation of heavy vaginal bleeding. The patient was diagnosed with AML three years ago and treated with bone marrow transplantation. Two years after the diagnosis she relapsed with GS of the breast. Following chemotherapeutic treatment, the patient was in remission. Sonographic evaluation of the pelvis revealed an enlarged uterus with areas of hypoechogenicity with no confines of a mass lesion. Magnetic resonance (MR) and diffusion weighted imaging of the lower abdomen were performed.MR imaging was performed with a 3T scanner . MR imaging showed an increase in size of the uterus and a large heterogeneous mass in the cervix. Compared to muscle, the mass was isointense on T1- and slightly hyperintense on T2-weighted imaging. The lesion had central cystic areas and showed strong heterogeneous enhancement after intravenous gadolinium administration . There wb value of 600 sec/mm2 was performed. ADC values were measured from the center of the lesion. For comparison, ADC values were also measured from muscle and bladder. ADC values were 0.43 × 10−3, 1.65 × 10−3 and 3.24 × 10−3, respectively  [−3 [DWI has been widely used and routinely implemented into imaging protocols of the different parts of the body. DWI has become integral imaging sequence for gynecologic malignancies . Bowel p sec/mm2 , 13. Cur3 mm2/s) . In a la3 mm2/s) . Tamai e3 mm2/s) . However3 mm2/s) . Recent 2/s) [−3 –16. We hIt is important to differentiate hematoma, abscess, cervical carcinoma, and lymphoma in a patient with a history of myeloproliferative disorder. MRI and DWI characteristics can be quite helpful under these circumstances. Although low ADC values are characteristic with abscesses, we expect to see high T2 signal in the center of the lesion. This contradicts the finding in highly cellular tumors such as lymphoma or granulocytic sarcoma and cannot be differentiated with imaging findings alone. Hemorrhage will reveal heterogeneous diffusion restriction and susceptibility artifacts due to blood degradation products.When a solid mass lesion arises in a patient with a history of AML, it is important to consider GS among differential diagnoses regardless of the localization. We believe combining MRI and DWI findings in the evaluation of these tumors is quite helpful, especially when there is a possibility of a coexisting benign lesion, such as hemorrhage, abscesses, or leiomyomas. Extremely low ADC values may aid the radiologist to reliably report an initial diagnosis.