IntroductionThe large nested variant of urothelial carcinoma (LNVUC) is a newly described and rare subtype of urothelial carcinoma. It is characterised by bland cytological features and a large nested architecture similar in appearance to low grade urothelial carcinoma with an inverted growth pattern. To date only 23 cases in a single series have been described.Case ReportWe describe the case of a 59 year old male with LNVUC whose tumour was initially misdiagnosed as a non-invasive low grade urothelial carcinoma. At a subsequent re-resection, his tumour was correctly re-classified as LNVUC with extensive invasion of the muscularis propria. Radical cystectomy and formation of an ileal conduit was performed. His operative specimen revealed invasion of prostatic stroma and perivesical fat, with all surgical margins clear. He is currently free from clinical recurrence 12 months after his cystectomy.ConclusionLNVUC is a newly described and rare urothelial carcinoma subtype. It characteristically possesses bland cytological features and may mimic low grade urothelial cancer. Despite its bland appearance it behaves aggressively with invasion, metastasis and death being common. This aggressive UC variant has deceptively bland cytological features, which may confound correct tumour classification. We present the case of a 59 year old male with a large bladder tumour who was initially diagnosed histologically as non-invasive low grade UC on initial resection. At re-resection the tumour was correctly identified as LNVUC.The large nested variant of urothelial carcinoma (LNVUC) is a newly described variant of urothelial cancer (UC), with a single series of 23 cases being the only examples reported thus farA 59 year old Caucasian male was transferred to our unit from a regional hospital with a two week history of macroscopic haematuria. He sought medical attention only after he developed clot retention. He denied any previous history of haematuria or urinary problems prior to the two week period immediately before his hospital admission.His medical history was unremarkable other than extensive carcinogen exposure, with both a 40 pack-year smoking history and significant occupational exposure, working as a fly-in, fly-out diesel fitter on a mine site.On admission he required placement of an indwelling urinary catheter and continuous bladder irrigations. His initial serum creatinine was elevated, but soon normalised following catherisation. He was transferred to our secondary referral centre following failure of conservative therapies to control his persistent haematuria.On his arrival to our facility we arranged Computerised Tomography (CT) to assess his bladder and upper renal tracts. CT demonstrated a grossly thick walled bladder with a large enhancing intra-vesical mass, and bilateral hydroureteronephrosis . His haeAt cystoscopy, there was a large papillary tumour involving the prostatic urethra, the trigone, and both lateral walls of the bladder.  Neither Histologically the tumour was classified as a low grade urothelial carcinoma with no evidence of superficial or muscle invasion. We found this finding inconsistent with the operative and radiologic findings and repeated a cystoscopy four weeks later.At repeat cystoscopy large volume tumour regrowth had occurred and a further 90 minute resection was performed. Tumour histology this time demonstrated invasion into the muscularis propia by a large nested variant of UC  with an A staging Positron Emission Tomography (PET) CT was negative for metastatic disease and a cysto-prostatectomy and formation of an ileal conduit was performed. The operative specimen histology again revealed the large nested variant of UC with focal invasion into peri-vesical fat  and the The patient’s post-operative period was unremarkable and he made a swift recovery. He was discharged from hospital one week post-operatively. He was referred to medical oncology for consideration of adjuvant chemotherapy, however after discussion with oncologists the patient declined any additional treatment. He is presently twelve months post cysto-prostatectomy and he remains clinically well and free from clinical disease recurrence. We will continue to closely monitor this patient.1. They describe tumours with universally bland histologic appearances but with invasion of large nests resembling von Brunns nests into the underlying stroma. In contrast to the normal nested variant of UC, a surface papillary component is present and there is abundant fibrous stroma between individual tumour nests2. LNVUC is most commonly mistaken for low grade urothelial cancer with an inverted growth pattern2.The large nested variant is a newly described subtype of UC. The first and to date only case series was published in 2011 by Cox and Epstein and describes 23 cases1.LNVUC behaves aggressively, of the 17 cases with adequate follow-up in Cox and Epstein’s series, 3 had died of their disease and another two were alive but had developed metastatic spread of their cancerThe large nested variant is an extremely rare, newly described variant of UC. Our case is only the 24th described in the literature, and the first case reported since the condition was first classified in 2011. LNVUC can confound accurate diagnosis by masquerading as Von Brunn’s nests or, in our case, low grade non-invasive UC. Despite the bland macroscopic and histologic appearance of LNVUC it behaves in an aggressive manner, and should be treated the same as any invasive urothelial malignancy.Written informed consent for publication of their clinical details and/or clinical images was obtained from the patient. This case report does an excellent job of highlighting the deceptive nature of LNVUC and the clinical importance of early detection. This aggressive and rare subtype of urothelial cancer can very easily be mis-diagnosed as a low-grade urothelial tumour. The article identifies the key demographic risk factors seen in LNVUC  and highlights the clinical factors that point towards an aggressive pathology . Early recognition is vital in allowing prompt radical treatment.In our unit we have also had a very similar case of a bladder tumour which on resection was found to contain discohesive nests of invasive urothelial carcinoma. This patient also underwent radical cystectomy and final histology confirmed T3 disease.As LNVUC can cause a diagnostic dilemma, we would be interested to learn if any immunohistochemical studies were carried out and what these showed.We have read this submission. We believe that we have an appropriate level of expertise to confirm that it is of an acceptable scientific standard. This is a timely report on large nested variant of urothelial carcinoma. It may pose a diagnostic confusion since histological appearance may resemble more indolent forms of the disease although it certainly has a high invasive potential and may be associated with metastatic disease. Therefore, the pathologists must be aware of this variant when they evaluate surgical specimens and urologists must discuss with their pathologists the possibility of the presence of such a variant when they receive a report indicating low grade disease yet the clinical presentation is not compatible.I have read this submission. I believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard. et al., 2007Hong).Nested variant of UC is indeed rarely reported. It is however, not certain whether the actual incidence is low or it is less frequently recognized. The estimated incidence is 0.3%  with the pathology indicating low grade cancer (pTa LG).Most of the recognized clinical indicators of nested variant including male gender, age above 50 years, ureteral obstruction and cystoscopy showing  tumor slightly raised and erythematous or nodular were present in this case.Tumor is known to have aggressive behavior, however, growth in 4 weeks needing 90 minute resection is rather surprising.Were any cold cup deep biopsies taken at the time of initial resection in view of the significant clinical findings of aggressive UC.Were biopsies from the bladder neck, prostatic fossa or para collicular area taken at the time of initial resection.I have few queries on which i request the authors' to commentsI have read this submission. I believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard. Thanks for your review.The described pathology in this case report is actually "large nested UC" which is a distinct clinical entity to "nested UC" and one only recently described. I agree that this entity, like "nested UC" is undoubtedly more common than is documented due to under reporting.In regards to your queries.1. I agree that the amount of tumour re-resection needed after only a 4 week interval following a macroscopically complete resection is surprising, it certainly was for us. One explanation is that a surgical trainee and  not a consultant urologist was performing the resection, which would account for some of the additional time required.2. No cold cup biopsies were taken at the time of the initial resection as the resection was felt to have adequately sampled the muscularis propria. Indeed, there was plentiful muscle present in the initial resection specimen histologically.3. Cold cup biopsies were not taken from the bladder neck, prostatic fossa and paracollicular area at the time of the initial biopsies. However both bladder neck and prostatic fossa proximal to the prostatic utricle were involved with tumour at initial resection and these areas were consequently extensively resected with loop diathermy.Thanks again for your comments and review.Andrew Keller


Synchronous malignancies of the gall bladder and common bile duct are a rare entity. Much of our knowledge on this topic comes from Japanese literature. Most of the synchronous carcinomas described in Japanese literature are associated with the presence of an anomalous pancreatic-bile duct junction (APBDJ).We report a case of synchronous malignancy of the extrahepatic biliary tree involving the fundus of the gall bladder and the intrapancreatic portion of the common bile duct (CBD). A 50-year-old female patient presented to us with clinical features of obstructive jaundice and on radiological evaluation was diagnosed to have a periampullary carcinoma; the patient underwent a pancreaticoduodenectomy, and histopathological examination revealed adenocarcinoma of the gall bladder and the intrapancreatic portion of the CBD.Synchronous malignancies have been rarely reported from the Indian subcontinent; therefore, it is essential for the clinician as well as the pathologist to maintain a high index of suspicion while evaluating such lesions and to look for the presence of an anamolous pancreatic-bile duct junction whenever indicated. Synchronous malignancies of the gall bladder and common bile duct are a rare entity. Much of our knowledge on this topic comes from Japanese literature , 2.Most of the synchronous carcinomas described in Japanese literature are associated with the presence of an anomalous pancreatic-bile duct junction (APBDJ) , 2. HoweThe data from the rest of the world is sparse. Shukla et al. from India have published an original article describing four cases of simultaneous malignancies of the gall bladder and common bile duct of which two are synchronous.Here, we report a synchronous malignancy of the gall bladder and common bile duct, its diagnosis, and successful management.A 50-year-old lady presented with complaints of pain abdomen and jaundice for one and a half months; she also complained of intermittent type of fever in the last 1 week. Physical examination revealed presence of icterus and a palpable tender globular mass in the right hypochondrium measuring 3 × 3 cm. A clinical diagnosis of obstructive jaundice secondary to periampullary carcinoma was made.Ultrasonographic examination of the abdomen and pelvis revealed dilatation of the intrahepatic biliary radicles (IHBR), with multiple freely mobile gall stones and a dilated common bile duct (CBD) measuring 1.8 cm with sudden narrowing at its distal end.CT scan showed moderate dilatation of the extra and intrahepatic biliary radicles, cholelithiasis with suspicious thickening of the gall bladder wall at the fundus, and a distal CBD stricture. There was no evidence of any distant metastasis Fig. .Fig. 1CEA preoperative diagnosis of periampullary carcinoma with chronic cholecystitis was made, and the patient planned for a pancreaticoduodenectomy (Whipple’s procedure) after optimization.The patient underwent a classical Whipple’s procedure; table examination of the resected specimen revealed multiple gall stones in the gall bladder with area of mucosal thickening at the fundus and an irregular circumferential growth involving the intrapancreatic portion of the CBD.Postoperative period was uneventful, and the patient recovered satisfactorily.Histopathological examination of the resected specimen Figs.  and 3 reMicroscopic sections obtained from the distal CBD region showed adenocarcinoma of the common bile duct with invasion into the muscular wall of the duodenum.Sections studied from the resected lymph nodes were free of tumor cells.A pathological staging of pT1aN0M0 for adenocarcinoma of the gall bladder and pT2N0M0 for adenocarcinoma of the distal CBD was made, and the patient was not subjected to any adjuvant therapy.The patient was disease-free 2 years after surgery.The data from Japanese literature suggest that synchronous malignancies of the biliary tree are not as rare as they were thought to be; its incidence is found to be as high as 5.0 to 7.4 %. The reason for their earlier reported low incidence was probably due to inadequate sampling of the gall bladder specimen after resection for extrahepatic tumors .When simultaneous malignancies of the gall bladder and common bile duct are discovered, it is important to distinguish between synchronous primary malignancies and secondary deposits. Warren et al. and Gertsch et al. have described certain criteria to differentiate synchronous primaries from malignant deposits, and they include (I) lack of anatomical continuity between the two tumors, (II) a growth pattern typical of a primary tumor, and (III) clear histological differences between the two tumors , 5. In oAPBDJ is one of the main etiological factors for synchronous malignancies of the biliary tree , 2. In aAs we were unable to diagnose the gall bladder carcinoma preoperatively, the presence of a synchronous malignancy was a histological surprise. But fortunately, as the gall bladder carcinoma was not involving the muscularis, no further treatment was needed and an R0 resection was achieved with a classical Whipple’s procedure.Shukla et al. have proposed various pathways for development of synchronous malignancies of the biliary tree and have reported four cases through which they explain the various possible pathways of development of simultaneous malignancies of the biliary tree; our case is similar to the first case described in the article in which the authors hypothesize that these are truly synchronous malignancies and sometimes may be associated with APBDJ but can occur even in the presence of a normal pancreatic-bile duct junction .Synchronous malignancies of the biliary tree are becoming more common in current hepatobiliary practice, and it is essential for the clinician as well as the pathologist to maintain a high index of suspicion while evaluating such lesions and to look for the presence of an anamolous pancreatic-bile duct junction whenever indicated.Informed consent was taken for publications of photographs in a scientific journal according to the guidelines of the Declaration of Helsinki and its later amendments.


Epithelial ovarian tumors are the most common malignant ovarian neoplasms and, inmost cases, eventual rupture of such tumors is associated with a surgical procedure.The authors report the case of a 54-year-old woman who presented with spontaneousrupture of ovarian cystadenocarcinoma documented by computed tomography, both beforeand after the event. In such cases, a post-rupture staging tends to be lessfavorable, compromising the prognosis. Mostcommonly, rupture of ovarian cystadenocarcinoma is associated with surgicalmanipulation, significantlychanging the staging and prognosis of the patients.Epithelial ovarian tumors correspond to the most lethal malignant neoplasia of thefemale genital tract. Mucinous and serous tumors are the two most common types ofepithelial neoplasia-5and patients using anticoagulant drugs. The authors report the first case of spontaneous rupture of ovarianserous cystadenocarcinoma documented by computed tomography both before and after theevent.Few reports are found in the literature about spontaneous rupture of ovariancystadenocarcinoma and these reports are mainly regarding pregnant womenA 54-year-old woman attended the emergency department presenting with sudden-onsetdyspnea associated with pain and increased abdominal volume. Chest computed tomography(CT) demonstrated the presence of pulmonary thromboembolism (PTE). Abdominal CT showed alarge pelvic solid-cystic mass with gross septations and solid component, compatiblewith epithelial ovarian tumor .The patient was admitted for treatment of the PTE, using heparin and, at the ninth dayafter the admission, she presented a sudden worsening in her condition with abdominalpain and a 3 g/dL drop in hemoglobin levels. A new abdominal CT revealed a voluminousascites and reduction in the dimensions of the adnexal solid-cystic mass with parietaldiscontinuity, compatible with spontaneous rupture .The patient was submitted to exploratory laparotomy that revealed a great amount ofhematic ascites and an adnexal tumor with solid component and ruptured cystic area. Anatomo. Epithelial ovarian tumors correspond to 60% of all ovarianneoplasms and 85% of the malignant ovarian neoplasias.The most recent Brazilian population data analysis indicates a risk for ovarian cancercorresponding to 6 cases/100,000 women. The main risk factors for development of ovariancancer include family or personal history of breast or ovarian cancer; post-menopausalhormone replacement therapy, smoking and obesity ormetastasis. Such tumorsrupture is frequently reported as occurring intraoperatively in cases where a largelesion is attached to adjacent organs. Spontaneous rupture rarely occurs, generally as a result eitherfrom internal tumor bleeding or from increased intralesional pressure in associationwith some risk factor,4,6, such as anticoagulant therapies. A previous study reports spontaneousrupture of an ovarian cystadenocarcinoma in a patient with congestive heart failure andundergoing treatment with heparin who progressed with hemoperitoneum, characterized bythe presence of hyperdense (65-70 Hounsfield units) fluid in the peritoneal cavity, butwithout clearly demonstrate the tumor lesion, probably because the scan was performedwithout using an intravenous contrast agent. In the other cases described in the literature, imaging studieswere not performed.Some of these tumors may present complications such as rupture, torsion,hemorrhage. The authors believe that the heparin anticoagulation must havebeen responsible for a bleeding within the cyst, leading to increased intralesionalpressure and subsequent rupture with hemoperitoneum.In the present case, the tumor lesion was clearly identified at the initial examination,presenting a sharp change in shape and reduction of dimensions at the subsequent scan,associated with the presence of ascites, which has allowed for establishing thediagnosis of tumor rupture, corroborated by the drop of hemoglobin levels resulting fromthe use of heparin due to the previous history of PTE. Heparin inhibits the Xa factorand thrombin, blocking the coagulation cascade at these levels with therapeuticalpurposes; hemorrhage is the main complication.Thus, the authors call the attention to the fact that the use of anticoagulant drugs inpatients with voluminous cystic lesions must be done cautiously, taking the risk/benefitbalance into consideration, with careful follow-up  along the treatment for evaluation of possible severe complications,like in the present case. Additionally, the extravasation of the cystic content mayresult in intraperitoneal malignant cells dissemination, modifying the staging, theapproach to be adopted and the prognosis of the patient


On palpation, it was nontender, firm and pedunculated. The differential diagnosis included fibroma, myxoma, mucocele, lipoma and pleomorphic adenoma. An excisional biopsy was performed and the microscopic features suggested “Fibroma”. The patient was recalled after 2 months to evaluate healing, which was uneventful (C). Fibromas are benign tumors of connective tissue origin. In the oral cavity, they occur in response to irritation from local trauma, and the most common sites are buccal mucosa and tongue. Their size usually ranges from 0.5 cm to 1.5 cm. They are asymptomatic, dome shaped, smooth surfaced, sessile or pedunculated masses that usually occur in females between 20-50 years. The recurrence rate is very low, but when it occurs, it may be due to the persistence of offending irritant.A 29 year old female presented with a growth in relation to the right cheek mucosa (A). On inquiry, she revealed that it had been gradually increasing in size, since she first noticed 4 months earlier. History revealed that she habitually bit her cheek. Constitutional symptoms and lymph node enlargement were absent. The nodule was 2 X 2 cm in size, yellowish in color, located on the right buccal mucosa, along the line of occlusion corresponding to the sharp cusps of maxillary 1


We describe here the case of a 62-year-old man with acute abdominal syndrome and severe hemorrhagic shock following successful thrombolysis for acute cardiac infarction. Emergency surgical exploration revealed extensive intraperitoneal and retroperitoneal hemorrhage resulting from the rupture of a large adrenal tumor. The diagnosis of pheochromocytoma was confirmed by histological findings. The patient died a few hours after surgery from multiorgan failure despite resuscitation attempts. This report discusses the diagnosis difficulties, treatment approach, and relevant literature. Pheochromocytoma presenting with acute hemorrhage is extremely rare. This report highlights a case of fatal retroperitoneal and intraperitoneal hemorrhage in a patient with undiagnosed pheochromocytoma who had been thrombolyzed for an acute myocardial infarction. To our knowledge, this is the first case of acute hemorrhage into pheochromocytoma associated with thrombolysis to be reported in the literature.A 62-year-old man with a medical history of diabetes mellitus and smoking was admitted to the cardiology unit with acute chest pain. The electrocardiogram showed ST-segment elevation in the inferior leads aVF, D2, and D3 Figure A. The laPheochromocytomas are catecholamine-secreting neuroendocrine tumors that arise from chromaffin cells of the adrenal medulla. The typA brief review of the literature reveals 65 cases of acute ruptured pheochromocytomas from its first description by Cahil in 1944. The cliThe reviewed patients ranged in age from 15 to 80 years, with a mean age of 50; 57% of patients were men. Pheochromocytomas were right-sided in 48% of cases, left-sided in 48%, and bilateral in 4%. The median lesion size was approximately 7 cm. In mostIf we analyze causal relationship between pheochromocytoma and cardiac infarction in our case, two hypothetic scenarios arise. First, the pheochromocytoma was spontaneously ruptured and induced cardiac disorders that simulated a cardiac infarction. The mechanism in this case, as described in a few published cases illustrating cardiac involvement in pheochromocytoma, may be Whatever the clinical picture in the foreground, computed tomography (CT) scanning of the abdomen is still the most useful examination for diagnosis. It showWhen requiring an accurate diagnosis, the optimal management is based on the appropriate medical preparation before any surgical procedure. This should include adequate alpha-adrenergic blockade, infusion therapy, blood resuscitation, and correction of coagulopathy. These mThe mortality rate as observed in the literature was 28% 18/65), with death often resulting from bleeding, heart failure from excessive catecholamine, or postoperative severe hypotension or pulmonary edema[5, with dRecently, to avoid emergency surgery in patients with ongoing bleeding, the use of transarterial embolization (TAE) was increasingly reported as being a safe alternative to achieve hemostasis and maintaining the patient in good condition until elective surgery can be performed, 21, 28.While a rare event, ruptured pheochromocytoma continues to be highly lethal in modern times. Usually spontaneous, this accident may also be initiated by trauma or medication. Thrombolytic therapy as described in the current case may play a role in accelerating bleeding. Proceeding to surgery with an occult pheochromocytoma, however, could be catastrophic. For this reason, a high index of suspicion is required for preoperative accurate diagnosis, which is a crucial step for adequate management. Computed scanning of the abdomen is the most useful examination for diagnosis, while medical preparation followed by delayed elective surgery is the optimal treatment. Alternatively, TAE may be employed to stop bleeding in an instable patient in order to avoid emergency surgery associated with a high mortality rate.Written informed consent was obtained from the patient’s relatives for the publication of this report and any accompanying images.


The small bowel rarely suffers from metastatic tumors from outside the abdomen. Small bowel obstructions caused by the metastatic spread of squamous cell carcinoma (SCC) of the hand to the intestines are even rarer. A 71-year-old man with intermittent abdominal distension and pain for 4 months was diagnosed with partial bowel obstruction. The patient underwent a video capsule endoscopic examination; however, the patient was unable to pass the capsule, which worsened the abdominal distension. He was transferred to our department for acute intestinal obstruction, and an emergency exploratory laparotomy was performed. Intraoperatively, a tumoral stricture of the intestine at a distance of 150 cm from the ileo-cecum and dilation of the proximal bowel was found. The involved segment was resected, and ileo-ileal anastomosis was performed. The pathological sections confirmed the lesion to be a moderately differentiated SCC with whole bowel layer infiltration. Immunohistochemical staining showed positive expression of cytokeratin 5/6 and p63. The patient had an uneventful recovery. However, 6 months later, he was hospitalized again with intestinal obstruction. Reoperation was performed and revealed multiple metastases in the small bowel. He died 4 months later. In this unusual case, metastasizing SCC of the hand skin led to intestinal obstruction and poor prognosis. Therefore, follow-up procedures regarding intestinal spread should be performed in patients with SCC who present with abdominal symptoms. Symptomatic intestinal or other intra-abdominal metastases from hand skin squamous cell carcinoma (SCC) are very rare. The majority of these metastatic lesions originate from the lung and esophagus -4. Katz 2N0M0 stage SCC with moderate differentiation. Unfortunately, he did not receive any other treatment except for one course of adjuvant chemotherapy with 5-fluorouracil and methotrexate. Three years ago, a wide surgical excision of recurrent skin lesions was performed . Although VCE is relatively non-invasive, it is contraindicated in patients with swallowing disorders, implanted electromedical devices or known or suspected gastrointestinal obstruction, strictures or fistulas . HoweverRadiation and chemotherapy are generally used as adjuvant or palliative therapies in patients with hand skin SCC, especially for those with involved lymph nodes or distant metastases. In our case, the patient refused additional treatment after the first laparotomy and soon developed a recurrence.Surgical resection should be considered for treating primary and metastatic SCC lesions and should include wide local excision of the tumor and lymph node-bearing mesentery. Some patients may require a second resection at a later date. The longest survival time without evidence of recurrent disease following resection of small bowel metastasis is 8 years . In our Hand skin SCC can metastasize to the thoracic wall and lung through lymph nodes in the epitrochlear and medial axillary regions. Skin SCC commonly metastasizes to the lung, liver, brain and bone, whereas intestinal obstruction caused by skin SCC metastases is an unusual presentation. SCC metastasizes to the small intestine through retrograde spread to the mesenteric lymph nodes or via the vertebral venous system. Although recurrences occur in most skin SCC cases, radical resection of the lesion is needed. Standardized chemotherapy after operation may improve the outcome for patients with distant metastasis of skin SCC.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.CK: cytokeratin; CT: computed tomography; SCC: squamous cell carcinoma; VCE: video capsule endoscopy.The authors declare that they have no competing interests.RL participated in the operation, and drafted and completed the manuscript. ZC was involved in the operation and review of the literature. QW was involved in the acquisition of clinical pictures and pathological examinations. ZC conceived the study, participated in the operation, and supervised manuscript preparation. All authors read and approved the final manuscript.


We present here a case of extrathyroid CASTLE (the third case reported in the English literature) treated with excision and neck dissection without radiotherapy. Also, we reviewed the literature and analyzed the therapeutic results of each treatment modality for CASTLE. A 27-year-old male had initially presented with a painless, right neck mass for 2 months. Computed tomography of the neck showed a 3.8 × 3.2 × 3.8 cm heterogeneously enhancing mass at right level IIa, and no definite thyroid lesion was found. An excisional biopsy was done and the pathologic diagnosis was CASTLE. Then we performed a right modified radical neck dissection and right thyroid lobectomy. After three years, no evidence of tumor recurrence was noted. Total excision followed by neck dissection could be a sufficient surgical treatment option for CASTLE. Postoperative radiotherapy might be an alternative treatment option for neck dissection in patients with positive nodal status. First d in 1985 , these l in 1985 . The maj in 1985 . GeneralAccording to reports in the English-language literature, less than 30 cases of CASTLE tumors have been reported, and only two cases were extrathyroid tumors . One casHere, we report a case of extrathyroid CASTLE, which was diagnosed by excisional biopsy, successfully treated by unilateral neck dissection with ipsilateral thyroid lobectomy, without concomitant radiotherapy.3 with 64.2% neutrophils, and the serum concentrations of thyroxine, thyroid-stimulating hormone and thyroglobulin were all normal. Neck ultrasonography revealed multiple tiny cystic-hypoechoic nodules in both thyroid glands, and a 3.8 × 3.2 × 3.8 cm mass with heterogeneous echogenicity in the right submandibular area. Computed tomography (CT) with enhancement of the neck showed the same-sized heterogeneously enhancing mass at right level IIa  showed no metabolic evidence of malignancy, and an ultrasonogram guided thyroid nodule FNA biopsy result was non diagnostic. Finally, two months after the first operation, a right modified radical neck dissection (levels I to V) and ipsilateral thyroid lobectomy were performed. A thyroid lobectomy was added to our surgery because most CASTLE tumors are intrathyroid, and also to give a definite diagnosis of the patient’s thyroid nodule. On intraoperative inspection and palpation of the thyroid, no noticeable intrathyroid mass was found. The surgery was finished without difficulty, and no other specific intraoperative findings were noted. The pathologic report of the right thyroid lobe was multinodular goiter and no definite neoplastic lesion was identified. All the lymph nodes in right levels I to V were non-neoplastic (reactive hyperplasia). The patient was discharged on the ninth hospital day without any postoperative complications, and was then regularly followed up in the outpatient department. After six months, a follow up PET-CT was taken and no metabolic evidence of regional tumor recurrence or metastasis was reported. At the most recent follow-up, i.e. three years after the second operation, no evidence of tumor recurrence was present.CASTLE is a rare malignant neoplasm that histologically resembles thymic carcinoma, and arises in the thyroid gland or adjacent soft tissue of the neck. During embryological development, primordial thymic tissue derived from the third and fourth branchial pouches migrates through the neck and into the mediastinum . Persistet al. identified 27 cases of reported CASTLE that mentioned an FNA biopsy [An FNA biopsy seems to offer limited diagnostic value for CASTLE. Youens A biopsy . ReporteA biopsy ,9, as seA biopsy .et al. found 26 reported cases of CASTLE, 25 intrathyroid cases, one extrathyroid case, and added one extrathyroid CASTLE, which they reported [.) with or without radiotherapy. A review of all reported cases showed that 16 of 27 patients had no evidence of recurrence at follow-up after treatment, and 10 of 27 had a recurrence. There was one case of recurrence with initial misdiagnosis. Four of the ten recurrent cases showed no evidence of recurrence after salvage treatment; one patient died of a cerebrovascular disorder, and five died of the disease with local recurrence or distant metastasis to the lung or liver. Most of the recurrences were noted in patients with invasion of the neck lymph nodes or surrounding tissue. Two cases of extrathyroid CASTLE were treated by resection and radiotherapy, and neither showed locoregional recurrence.In their study, Luo There is no consensus on the management of CASTLE, due to the rarity of the disease, but complete surgical excision with or without radiotherapy seems necessary to improve the long-term survival rate and reduce the locoregional recurrence rate. Table *’ in Table In summary, the most successful treatment from a review of the literature was complete surgical excision followed by neck dissection with or without postoperative adjuvant radiation therapy depending on nodal state. In our case, complete excision by neck dissection without radiotherapy achieved a positive result as there was no metastatic node in the neck specimen. Interestingly, all extrathyroid CASTLE cases (‘We present a case of extrathyroid CASTLE, which was successfully treated by excision plus unilateral neck dissection. From a review of the literature, the most successful treatment of CASTLE is complete surgical excision followed by neck dissection with or without postoperative adjuvant radiation therapy depending on nodal state.Written informed consent was obtained from the patient for publication of this case and for the accompanying images.CASTLE: carcinoma showing thymus-like differentiation; CT: computed tomography; FNA: fine-needle aspiration; NED: no evidence of disease; PET-CT: positron emission tomography-computed tomography.None of the authors involved in manuscript preparation have any competing interests regarding the manuscript itself, nor are there any financial or moral conflicts. None of the authors received support in the form of grants, equipment or pharmaceutical items.KYC collected data and drafted the manuscript. MJK and HKA supervised the pathology and managed the figures. JHK supervised the writing of the manuscript. DJL conceived of the study and corrected and revised the manuscript. All authors read and approved the final manuscript.


Background. Laparoscopic cholecystectomy represents the gold standard approach for treatment of symptomatic gallstones. Surgery-associated complications include bleeding, bile duct injury, and retained stones. Migration of surgical clips after cholecystectomy is a rare complication and may result in gallstone formation “clip cholelithiasis”. Case Report. We report a case of a 55-year-old female patient who presented with right upper quadrant pain and severe sepsis having undergone an uncomplicated laparoscopic cholecystectomy 10 years earlier. Computed tomography (CT) imaging revealed hyperdense material in the common bile duct (CBD) compatible with retained calculus. Endoscopic retrograde cholangiopancreatography (ERCP) revealed appearances in keeping with a migrated surgical clip within the CBD. Balloon trawl successfully extracted this, alleviating the patient's jaundice and sepsis. Conclusion. Intraductal clip migration is a rarely encountered complication after laparoscopic cholecystectomy which may lead to choledocholithiasis. Appropriate management requires timely identification and ERCP. Cholecystectomy represents the gold standard for definitive treatment of symptomatic gallstones. Estimated 57,000 cholecystectomies were performed in the UK in 2012 . Irrespe9/L, ALT 78 IU/L, ALP 242 IU/L, and total bilirubin 97 umol/L. After fluid resuscitation the patient was commenced on broad spectrum antibiotics and transferred to the high dependency unit (HDU) for vasopressor support. The working diagnosis at this stage was biliary sepsis, likely secondary to a retained stone in the common bile duct. Computed tomography (CT) imaging of the chest, abdomen, and pelvis revealed a moderate right sided pleural effusion and evidence of intra- and extrahepatic ductal dilatation. Hyperdense material was noted in the lower common bile duct, and a surgical clip was visible in the gallbladder fossa  and hypotension (systolic BP ~80 mmHg). The only feature of note in her past medical history was an uncomplicated laparoscopic cholecystectomy some 10 years earlier. Initial laboratory indices were as follows: WBC 14 × 10er fossa . The pater fossa . This reAlthough rare, clip migration following laparoscopic cholecystectomy has been described in a number of case reports and should be recognised as a potential late complication after surgery. Though first described as a potential postcholecystectomy complication in 1979 , the exaIn our opinion, thorough skeletonisation of the CD and distal application of the minimum number of metallic clips/ligatures are measures that are more likely to reduce the likelihood of CD stump invagination and may reduce the risk of inadvertent clip migration. Although this is a rare cause of cholangitis after cholecystectomy of unproven pathogenesis, it should be recognised early with correct imaging and treated with ERCP and stone extraction.“Clip cholelithiasis” is a rarely encountered complication following laparoscopic cholecystectomy which can lead to obstructive jaundice.The precise pathophysiological mechanism by which surgical clips migrate from the original site of application remains uncertain.The time interval between cholecystectomy and clip migration can vary, and symptoms may not manifest until years after the initial procedure.


Introduction. Metal-on-metal (MoM) hip resurfacings have been associated with a variety of complications resulting from adverse reaction to metal debris. Pseudotumors have rarely been reported to cause deep venous thrombosis (DVT). Study Design. A case report and a review of the literature. Case Presentation. A 75-year-old female who had left metal-on-metal hip resurfacing 6 years ago presented with left groin pain associated with unilateral lower limb edema and swelling. By duplex and MRI studies, our patient had an extensive soft tissue necrosis associated with a large pelvic mass causing extensive DVT of the lower limb secondary to mechanical compression of the left iliac vein. Results. Our case was initially treated for DVT followed by dual surgical approach. The pseudotumor was excised through a separate iliofemoral approach and revision of the hip implant was undertaken through a posterior approach in the same setting. An inferior vena cava (IVC) filter was inserted to minimise the perioperative risks of handling the iliac veins. Conclusion. A combined approach with vascular surgeons is required. Combined resection of the pseudotumor and revision of the metal bearing surfaces is essential, in order to achieve a good surgical outcome in this rare complication. Metal-on-metal (MoM) hip resurfacing was popularised in the late 1990s. Pseudotumors or soft tissue reactions, so-called adverse local tissue reactions (ALTRs), have been reported as complication. These effects are thought to be due to local tissue necrosis or possibly hypersensitivity reactions.We present a case of large pelvic pseudotumor associated with MoM hip resurfacing resulting in deep vein thrombosis (DVT). Such cases have been described in association with polyethylene debris but rarely reported with resurfacing hip arthroplasty.A 75-year-old female presented to our hospital in April 2014 with left groin pain associated with unilateral lower limb edema and swelling for four weeks.She had previously had left hip resurfacing arthroplasty in 2008, using Cormet prosthesis . She had no history of febrile illness. Metal ion levels were within the normal range recommended by MHRA . A metal artifact reduction sequence (MARS) MRI scan of both hips was also performed one year ago. This showed a cystic lesion measuring 30 mm × 25 mm in size. Our patient was asymptomatic with regard to both hips and mobilizing independently and was fully active prior to the onset of recent symptoms.On examination, the left leg was swollen from thigh to ankle. She had generalized tenderness starting from the groin and the lateral aspect of the thigh radiating down her leg. Her hip arthroplasty wound was well healed, and there were no inflammatory changes. Her hip was painful on all movements, but worse with rotation in flexion. Patient also showed signs of psoas irritation.On abdominal examination, there was fullness in the left iliac fossa with tenderness on deep palpation. Her right hip examination was normal.Investigations revealed an elevated erythrocyte sedimentation rate (ESR) of 48 mm/hour and C-reactive protein (CRP) of 37 mg/L . Her white blood cell count was 5.35 (3.5–11). Metal ion levels were within the normal range, cobalt 1.37 ppb and chromium 2.05 ppb.She was admitted to hospital. A plain radiograph of her pelvis showed satisfactory positioning of right uncemented total hip replacement with 36 mm metal-on-metal articulation (Corail/Pinnacle) and left hip resurfacing, without adverse features .Duplex ultrasound of the left leg venous system showed a distended occluded left common femoral vein which failed to respond to respiratory excursions. There was extensive induration and oedema of the skin overlying the thigh which hampers good visualisation of the deep veins within the thigh. Also, ultrasound showed an 80 mm × 32 mm left sided psoas collection which extends from just about the level of the femoral head to the level of the lesser trochanter distally. This collection was compressing the left iliac vein.MARS MRI scan of both hips and pelvis showed left retroperitoneal pelvic cystic lesion (74 mm × 32 mm) previously noted anteriorly and now extending into the iliopsoas bursa. It was thin walled and contained low signal linear debris. The external iliac vessels were displaced and flattened. Further, a smaller cystic lesion was also noted around the greater trochanter (20 mm in length × 5 mm in width) .Our patient was anticoagulated with low molecular weight heparin (LMWH) and warfarin. The swelling improved rapidly with treatment and rest. Revision surgery was planned but delayed for 3 months pending treatment of DVT.Prior to surgery, a temporary IVC filter was inserted to minimise the risks of intraoperative pulmonary emboli and CT angiography performed before surgery .At surgery, pseudotumor was resected after mobilisation of the iliac veins, via an extraperitoneal iliofemoral approach by a vascular surgeon. The wound was then closed and the patient placed in the right lateral decubitus position for revision of the left hip resurfacing to total hip replacement via a posterior approach.Intraoperatively, the external iliac vessels lie closely related to the external wall/capsule of the pseudotumor. The periarticular tissues at the left hip were abnormal and dull grey in colour. Samples were sent for histopathology and microbiology examination. Cultures were sterile after 14 days of incubation. Histologically, the tissue consisted of fibrous and fibrohyalinized tissue with evidence of acute inflammation, predominantly macrophages with only occasional lymphocytes. There were small areas of synovial hyperplasia and focal necrosis. This suggested an adverse reaction to metal debris.Subsequently, she also completed her course of anticoagulants, and a repeat sonogram revealed patent deep venous system. Her symptoms have significantly improved. She is able to carry out most of her daily activities and has an Oxford score of 35 out of 48 nine months after surgery.Metal-on-metal hip resurfacing was introduced in the 1990s with the hope that wear rates would be lower for the harder bearing surfaces, that larger head sizes would reduce the rate of dislocation, and that subsequent revision surgery would benefit from bone preservation of the femoral neck [Although there is less wear in MoM bearing compared with metal-on-polyethylene bearings, the particle size in MoM hips is small and may induce greater biological reactivity .Excellent results have been reported after MoM hip resurfacing, but concern regarding abnormal reaction to metal debris in some patients persists. Pandit et al. first described neither malignant nor infective cystic and solid masses associated with metal-on-metal resurfacing and introduced the term pseudotumor. They reported that pseudotumor may be the result of a toxic effect on cells due to particulate wear debris or an idiosyncratic response to the release of metal particles .By 7-year follow-up, the incidence of pseudotumors after hip resurfacing arthroplasty has been described at 0.3% to 3.4% . AccordiFactors associated with higher prevalence of pseudotumors are female sex, bilateral resurfacing arthroplasties, excessive cup anteversion, cup inclination of more than 50°, and higher metal ion levels and some prosthesis types .Pseudotumors may be asymptomatic. Various complications associated with pseudotumors include persistent pain, femoral or sciatic nerve palsy, rash, and ureteral obstruction , 8. AlthOur patient had bilateral MoM bearing hips. She remained asymptomatic for 6 years. She developed a pseudotumor that caused local compression of the iliac veins. This is a rare cause of DVT.Only four cases of such tumors causing DVT after MoM have been described before. One case had a revision surgery and excision of the pseudotumor using the same approach . AnotherOur approach to this case was initial treatment of the DVT followed by interval resection of the pseudotumor with IVC filter insertion to minimise the perioperative risks of handling the iliac veins. At the same operation, the hip was revised to a non-metal bearing (ceramic-on-polyethylene) total hip replacement, which removes the metal ion/particle generating force. Nine months after surgery, the patient reported complete relief of groin pain and good function and had an Oxford score of 35 out of 48.This case report is the first, to our knowledge, to describe a dual surgical approach for management of pseudotumor causing deep venous thrombosis due to mass effect. The external iliac vessels and femoral neurovascular bundles were at risk and lie closely related to the external wall/capsule of the pseudotumor. This study highlights the importance of two surgical specialties working together to reduce the risk of harm to these structures.


Gallbladder torsion with ischemic wall necrosis is a rare condition, as gallbladder herniation. We describe here an original case of a patient with a symptomatic incisional hernia containing a gangrenous gallbladder twisted about its pedicle. We report preoperative findings on CT-scan and emergency surgical management. Gallbladder torsion with ischemic wall necrosis is a rare condition, as gallbladder herniation. We describe here an original case of a patient with a symptomatic incisional hernia containing a gangrenous gallbladder twisted about its pedicle. This case represents diagnostic and therapeutic challenging.An 81-year-old woman was admitted in our institution for abdominal sepsis for 3 days. Her medical history included an obstructive left-sided colonic cancer 10 years ago, treated first by a discharge colostomy followed by open left-colectomy. On physical examination, there was a tender and painful mass in the right upper quadrant, corresponding to an incisional hernia at the site of the previous colostomy (inflammatory skin). The patient presented fever without occlusive symptom. Routine laboratory features showed an hyperleucocytosis (22.7 G/L) with an elevated c-reactive protein (343 mg/L) and an acute renal insufficiency. Hepatic tests were normal.An emergency contrast-enhanced multislice helical-computed tomography (CT) scan revealed a large incisional hernia containing some digestive structures (satisfactory vascular enhancement of caecum and jejunum) and a thick-walled gallbladder Figure . The diaGallbladder torsion (or volvulus) is a rare condition (400 cases reported in the literature), predominantly occurring in female (sex ratio 3:1) and the average age of incidence is 70  since percutaneous cholecystostomy is not adapted in this particular situation (no transhepatic passage). A direct surgical approach on the incisional hernia confirmed the diagnosis and the integrity of digestive structures. It allowed both the treatment of the gallbladder torsion and the repair of the fascial defect at the same time. Due to the high rate of postoperative infections in these septic conditions, fascial repair was performed without any synthetic mesh. A biologic mesh could have been discussed . Ultrasonography and CT-scan may be helpful to perform the correct diagnosis before surgical management.


Introduction. Hepatic peribiliary cysts (HPCs) usually originate due to the cystic dilatation of the intrahepatic extramural peribiliary glands. We describe our rare experience of pure laparoscopic left hemihepatectomy (PLLH) in a patient with HPCs accompanied by a component of biliary intraepithelial neoplasia (BilIN). Case Presentation. A 65-year-old man was referred for further investigation of mild hepatic dysfunction. Contrast-enhanced computed tomography showed dilatation of the left-sided intrahepatic bile duct, and biliary cytology showed class III cells. The patient was highly suspected of having left side-dominated cholangiocarcinoma and underwent PLLH. Microscopic findings revealed multiple cystic dilatations of the extramural peribiliary glands; hence, this lesion was diagnosed as HPCs. The resected intrahepatic bile duct showed that the normal ductal lumen comprised low columnar epithelia; however, front formation on the BilIN was observed in some parts of the intrahepatic bile duct, indicating that the BilIN coexisted with HPCs. Conclusion. We chose surgical therapy for this patient owing to the presence of some features of biliary malignancy. We employed noble PLLH as a minimally invasive procedure for this patient. Hepatic peribiliary cysts (HPCs) are a poorly recognized liver disease characterized by multiple tiny cysts along the portal radicle. HPCs usually originate due to the cystic dilatation of the intrahepatic extramural peribiliary glands around the intrahepatic large bile ducts . AlthougBiliary intraepithelial neoplasia (BilIN), a microscopic biliary dysplasia with various cellular atypia, is one of the major intraductal neoplasms that can progress to invasive cholangiocarcinoma. Therefore, BilIN has to be accompanied by HPCs to progress to cholangiocarcinoma; however, there are no known reports of this process.Laparoscopic liver resections have dramatically advanced in recent years but still remain a somewhat demanding modality for major liver resections. Pure laparoscopic major liver resection is therefore associated with concerns about intraoperative and postoperative morbidities , 5.In this report, we describe our rare experience with pure laparoscopic left hemihepatectomy (PLLH) in a patient with HPCs accompanied by a component of BilIN.A 65-year-old man was referred to our hospital for further investigation of mild hepatic dysfunction. Routine laboratory examination revealed liver dysfunction and increased tumor markers, with aspartate aminotransferase levels of 94 IU/L, alanine aminotransferase of 160 IU/L, carcinoembryonic antigen of 6.1 ng/mL, and carbohydrate antigen 19-9 of 129.1 U/mL. Contrast-enhanced computed tomography showed dilatation of the intrahepatic bile duct in the left hemiliver , and abdThe patient was placed in the right semilateral position under general anesthesia, and we used four port techniques in accordance with PLLH. During the hepatic parenchymal transection, the upper limit of the pneumoperitoneum pressure was set to 8–12 mmHg with monitoring of the central venous pressure. After the falciform and left triangular ligaments had been transected, cholecystectomy and regional lymphadenectomy were performed as usual. The left hepatic artery was then transected, and the left branch of the portal vein was dissected and temporarily clamped with a nonabsorbable clip  . The demMacroscopic findings of the resected specimens showed multiple cystic lesions surrounding the intrahepatic bile duct . MicroscHPCs originate from the cystic dilatation of the extramural peribiliary glands of the bile duct located in the connective tissue of the hepatic hilum and within the larger portal tract . Owing tTo the best of our knowledge, HPCs are usually harmless. In addition, no cases of malignant degeneration from HPCs have been reported. Based on these reasons, we usually consider further surgical treatment to be unnecessary when HPCs are diagnosed accurately; however, there has been one report of HPCs accompanying bile duct carcinoma . In addiDepending on the surgical procedure, HPCs should be resected in a manner similar to that employed for other types of intrahepatic cholangiocarcinomas and extrahepatic bile duct carcinomas; therefore, major hepatectomy with or without extrahepatic bile duct resection or pancreaticoduodenectomy should be chosen. On the other hand, laparoscopic procedures for HPCs have not been described in detail, and laparoscopic resection for HPCs has not been reported until now. Major hepatectomies with biliary or vessel reconstruction are not presently indicated for laparoscopic procedures owing to the high rate of conversion and severe complications . In addiIn conclusion, HPCs may have the potential to complicate biliary malignancies, including BilIN; hence, a surgical procedure should be considered when some features of biliary malignancy are revealed with HPCs. We employed PLLH as a minimally invasive procedure for our patient. Although PLLH can have the clinical benefit of early postoperative recovery, PLLH requires adequate experience of laparoscopic liver resections to ensure safety during the surgical procedure.


We herein describe a case involving spontaneous rerupture of a nonparasitic liver cyst successfully treated with cyst fenestration and an omental flap. A 59-year-old Japanese woman was transferred to our hospital for evaluation of acute abdominal pain. She had a history of conservative treatment with antibiotics for spontaneous rupture of a liver cyst 1 month previously. On arrival, she exhibited abdominal tenderness and muscular defense. Enhanced computed tomography showed ascites and a large ruptured hepatic cyst (diameter of 10 cm). We diagnosed rerupture of a liver cyst and performed laparotomy for cyst fenestration and intraperitoneal drainage. During the operation, we found the perforation site on the ventral side of the cyst and brown, muddled ascitic fluid. Cholangiography showed no bile leakage on the inner wall. Pathological investigation revealed no evidence of malignancy. The patient recovered without any adverse events and was discharged on postoperative day 8. No recurrences or complications occurred for 2 years. A nonparasitic liver cyst (NLC) is a common benign liver disease. It is potentially asymptomatic and is often incidentally diagnosed with abdominal imaging such as ultrasonography or computed tomography (CT). With the advancements and spread of these abdominal imaging techniques, NLCs are becoming more frequently detected and have been found in approximately 5 % of the population . In manyWe herein describe a rare case of spontaneous rerupture of an NLC that had become exacerbated after conservative treatment and was successfully treated with surgical fenestration.A 59-year-old Japanese woman was transferred to the emergency unit of our hospital for evaluation of acute abdominal pain. She had a history of conservative treatment for a spontaneous NLC rupture 1 month previously in another hospital Fig. .Fig. 1CTOn examination, she had a pulse rate of 115 beats/min, blood pressure of 112/68 mmHg, and no fever. Her abdomen was flat but hard and painful. She also exhibited obvious tenderness and muscular defense upon arrival. Blood tests revealed acute inflammation and anemia Table . The cysBased on the patient’s clinical course and investigation findings, we diagnosed panperitonitis associated with rerupture of the liver cyst and accompanied by hemorrhage. Laparotomy was performed for cyst fenestration and intraperitoneal drainage.During the operation, we found the perforation site on the ventral side of the cyst Fig. . The perEchinococcus species, is a well-known complication of such cysts and is often reported as hydatid cyst rupture [Rupture of parasitic liver cysts, which are mainly caused by the  rupture , 4. In c rupture  stated t rupture –19. The  rupture , 16, 20.In general, treatment options for symptomatic NLCs include surgical procedures and conservative management such as percutaneous needle aspiration and drainage . PercutaThe optimal treatment strategy and surgical indications for NLC rupture are not clearly defined. Conservative management including percutaneous drainage might be useful for cases without critical features such as signs of peritoneal irritation and shock . HoweverIn conclusion, rupture of an NLC is a highly rare complication but can be a cause of the acute abdomen. Clinical observation and conservative treatment including percutaneous needle aspiration and drainage might be beneficial; however, careful consideration of the optimal therapy and performance of close follow-up are necessary owing to the possibility of relapse.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.


Conventional chondrosarcomas rarely metastasize andit is extremely unusual to see multicentric- behaviour inmalignant cartilage tumour. We report a 40 year old ladywith presentation of two non-contiguous metachronousfoci of low to intermediate grade of chondrosarcoma overleft pelvic bone and right scalp respectively in the absenceof pulmonary or visceral metastasis.vacuum assisted closure, infection, instrumentation, spine It constitutes approximately 20 % ofall primary malignant bone neoplasms 3,4. There is higherpreponderance in axial than the appendicular skeletonwith the pelvis being the most common site, accountingfor about 40% of all reported cases 3. The spine and thecraniofacial bones are very rarely involved. Althoughmetastasis is rare with conventional chondrosarcoma,it can metastasize to lungs, liver and other bones 1. It iseven rarer for chondrosarcoma to metastasize to extraosseousregions, like the brain as accounted by Faris et al2. In addition, it is also well-known that multicentricity,which is defined as the presence of two or more separatechondrosarcomas in the absence of visceral involvementbetween the time of diagnosis of both tumours, is extremelyunusual in malignant cartilage tumours, except for thehighly aggressive mesenchymal chondrosarcomaChondrosarcoma is the third most common primarymalignant tumour of bone, following Ewing’s sarcomaand osteosarcoma To our knowledge, we have encountered a case of multicentricchondrosarcoma with metachronous involvement of pelvicbone and scalp (extra-osseous), a feature that has beenreported previously.A 40 year old female, presented with a painless left hipswelling for six years prior to presentation. She had firstnoticed the swelling in 2008 and sought treatment ata private hospital, but defaulted treatment for six yearsuntil two weeks prior to hospital admission, when shedeveloped left hip pain which radiated to the left knee, andhad walking difficulties. The patient appeared cachexic andpale. Physical examination revealed a painless huge bonymass, measuring about 20 x 15cm arising from her lefthip extending over the suprapubic region and beyond themidline of the stomach. It had ill-defined margins and therange of movement in the left hip was limited. Otherwise,distal pulses were present normally with no neurologicaldeficit over the left leg. No lymph nodes were detected.MRI findings showed a huge lobulated intermediate tohigh T2 signal intensity mass in the left iliac fossa arisingfrom the pelvis, measuring 165 x 80 x 210 mm. The massextended from the level of iliac crest down to upper third leftfemur crossing the midline to the right iliac fossa. There wasinvolvement of the left ilio-psoas muscle with destructionof the anterior column of the left acetabulum. The leftfemoral vessel was encased within the mass. Otherwise,the outline of the mass was fairly well circumscribed. Therewas no infiltration of the urinary bladder or uterus. The leftfemoral head was intact. CT scans of the thorax, abdomenand pelvis showed no evidence of distant metastasis.Trucut biopsy of the mass showed abundant hypocellular,avascular, hyaline chondroid matrix separated byfibrocollagenous septa into lobules. Embedded withinthe matrix were malignant chondrocytes located withinlacunae . These chondrocytes exhibited mild to moderatenuclear pleomorphism, with occasional binucleation andlow mitotic activity. No tumour necrosis was evident.These findings were consistent with well-differentiatedchondrosarcoma.A multi-disciplinary intervention by the orthopaedic,surgical, radiological, anaesthetic and oncology teamwas adopted. She underwent pelvic resection Type II(periacetabular), Type III  and partialType I (iliac) as classified by the Musculoskeletal TumourSociety. The resection revealed the extensive growth ofthe tumour. A large 220mm x 150mm mass, weighingat 2100g was resected from the pelvis Due to the extensiveness of the tumour, the surgical teamhad to perform peritoneum and abdominal closure afterresection of the tumour. The bladder was noted to bethinned-out and was repaired.Post-operatively, the patient made an uneventful recoveryand was discharged well with wheelchair ambulation.Post-operative histopathological findings of the resectedtumour showed narrow surgical clearance of 1mm awayfrom all soft tissue surgical margins except at the lateralmargin which was 2mm away. The pelvic bone was invadedby the tumour including the superior margin of the ilium.However, the articular surface of the acetabulum, pubicsymphysis margin, pubic rami and femoral head weretumour-free. The pelvic tumour showed well-differentiatedchondrosarcoma.On follow up review of the patient at two months, thepatient’s surgical wounds had healed well and she wasambulating with walking frame. Serial radiographsshowed bone healing with callus formation at thereconstruction site. However, a painless scalp swellingwhich was firm in consistency, measuring 3 x 2 cm overthe right temporo-occipital region was found An MRI brain showed a well-defined 36 x 31 x 16 mmmixed enhancing lesion in the soft tissue over the rightoccipital region, with no bone erosion or enhancementnoted. No focal lesions were seen in the cerebralhemispheres and cerebellum.An incisional biopsy of the scalp swelling demonstratedtumour fragments with overlying epidermis lined bybenign stratified squamous epithelial cells. The tumouritself appeared lobulated and separated with fibrous septaand composed of large amount of chondroid matrix, withmild cellularity. Malignant chondrocytes were seen within swollen lacunar spaces and the nuclei exhibited moderatenuclear pleomorphism, consistent with well-differentiatedchondrosarcoma.At six months follow-up after the major resection, shepresented with a firm mass over the right pubic region.CT scans of the pelvis revealed recurrent chondrosarcomawith extensive bilateral pelvic involvement. There weremultiple lobulated rim enhancing hypodense lesions inthe soft tissue and muscle around the bones in the pelvis.The lesions were seen on both sides of the iliac bone,proximal portion of adductor and obturator muscles,sacroiliac joints and lateral wall of pelvis, anterior wallof pelvis, perineal region. Large lesions were noted inthe right pubic region with destruction of the bone. Someof the lesions extended into the right hemipelvic cavitycompressing and displacing the pelvic structures. Therewere also similar lesions in the course of the externaliliac vessels bilaterally, which were likely metastasis toadjacent lymph nodes. Currently, the patient is undergoingpalliative management by the oncology team.Multicentric presentation for skeletal sarcomas is arare occurrence. With regard to chondrosarcomas,multicentricity is extremely unusual, especially in thenon-mesenchymal variants. In our case, this condition wasdemonstrated by the presentation of two non-contiguouschondrosarcomas, one originating in the left pelvic regionand the other originating in the right scalp, withoutevidence of visceral or pulmonary metastases.There is much ongoing debate in the literature on whethermulticentric chondrosarcomas represent multiple distinctprimary lesions or metastatic disease. From the history,we know that the patient had two separate swellings atpresentation without evidence of pulmonary disseminationon CT scans. The absence of metastases at presentationsupports the possibility that the chondrosarcoma lesionsare multicentric and metachronous.The present case also suggests that when a patient presentswith chondrosarcoma, a thorough physical examinationof the patient is imperative, during which not only mustthe primary lesion be examined, but care must also betaken to be vigilant of other secondary lesions becauseof the reasons as mentioned above. This is especiallyso during follow-up care so as to detect small insidiouslesions that may be masked. As in this case, the patienthad a small swelling over her scalp. It had been hidden underneath the thick weaves of her hair. It was onlydetected when it grew large enough to peek though thethick covering of hair.Reiner et al conclude that the prognosis and survival ratein patients with pelvic chondrosarcoma is determinedby the tumour stage and the surgical margin achieved,where the incidence of local recurrence is influencedby the surgical margin achieved and the incidence ofdistant metastases is influenced by the tumour stage.5Early diagnosis is difficult because patients with pelvicchondrosarcoma are usually relatively asymptomatic earlyin the course of the disease and tend to present at a laterstage where the tumour - has already reached a large size.This is because of the wide space within the pelvic cavitythat can accommodate a growing tumour without anycompressive symptoms until it is large enough to causemass effect. In this case, the patient had the painless hipswelling for six years before finally seeking treatmentonly after she started experiencing pain over her left lowerlimb, incapacitating her ability to ambulate. The patient’ssurvival rate in this case was further complicated because,due to the extent of the tumour growth, it was impossibleto get a wide surgical clearance. Histopathological findingsof the surgical resection demonstrated a narrow surgicalclearance margin especially over the site of the femoraliliacreconstruction. Unfortunately, six months later, thepatient presented with multiple skeletal metastases andextra-skeletal lesion, making any subsequent surgeryalmost impossible.In summary, pelvic chondrosarcoma poses a challenge fororthopaedic oncologists because of the often late diagnosis,which results in extensive growth of tumour invading andjeopardizing vital structures and compromising pelvicstructure stability. Thus, careful surgical planning andconsideration of method of reconstruction is warranted.Long-term follow-up is also necessary in view of the highpossibility of local tumour recurrence.


A unilateral tumour in the nasal cavity or paranasal sinuses is commonly caused by polyps, cysts, and mucoceles, as well as invasive tumours such as papillomas and squamous cell carcinomas. Schwannomas, in contrast, are rare lesions in this area . We present a case of a 52-year-old female who presented with a 4-year progressive history of mucous hypersecretion, nasal obstruction, pain, and fullness. Imaging of the paranasal sinuses showed complete opacification of the entire left nasal cavity and sinuses by a tumour causing subsequent obstruction of the frontal and maxillary sinuses. The tumour was completely excised endoscopically. Histopathology was consistent with that of a schwannoma. Schwannomas are benign tumours originating from peripheral nerve sheaths. Previous reports indicate that 25–50% of schwannomas occur in the head and neck region, but tumours originating from the nasal cavity or paranasal sinuses are rare, with a reported rate of approximately 4% , 2. In 2We report the case of a huge nasoethmoidal schwannoma excised endoscopically.A 52-year-old female presented with a 4-year progressive history of left nasal obstruction, pain, and fullness with intermittent epistaxis. Her symptoms began in China where she described an episode of an upper respiratory tract infection with subsequent development of ongoing mucous production. Other features of her history were otherwise unremarkable.Findings on nasendoscopy showed a huge left nasal polyp completely obstructing the anterior nasal cavity limiting further examination. On nasendoscopy through the right nostril, the tumour could be seen occupying the left nasopharynx but the right nasal cavity was clear. Examination of the eye and the oral cavity was unremarkable.High resolution CT of the paranasal sinuses showed complete opacification of the left frontal, ethmoidal, maxillary, and sphenoidal sinuses and nasal cavity. The tumour significantly displaced the left lateral nasal wall into the maxillary sinus. There was also hyperostosis of the sphenoid and maxillary sinus walls. The right nasal cavity and sinuses were clear. Axial, coronal, and sagittal views are demonstrated in Figures An MRI was performed to better visualise the soft tissue mass. The mass was identified to fill the entire left nasal cavity, extending into the choana to completely fill the nasopharynx. This mass was measured at 78 × 28 × 51 mm (AP × ML × SI). The mass also occupied the left anterior and posterior ethmoid air cells, obliterating all bony septations. The left maxillary antrum and frontal sinus were filled with secretions secondary to the obstruction caused by the mass. Axial and coronal T2 weighted images are demonstrated in Figures We did not acquire a preoperative tissue biopsy given how promptly a complete excisional procedure could be performed. Furthermore, given the patient's severity of symptoms, complete excision was necessary regardless of the diagnosis, and therefore we felt a biopsy would not have changed immediate management.Endoscopic resection of the schwannoma was undertaken. The patient was placed under general anaesthetic in a reverse Trendelenburg position. The superior extent of the tumour was dissected off the anterior skull base with no attachment found. Clearance of the frontoethmoidal recess was followed by free evacuation of mucus from the frontal sinus. Inferiorly the tumour was not attached to the nasal floor. Obstruction of the left maxillary sinus ostium was addressed with clearance of the maxillary sinus contents. The tumour was attached posteriorly to the basisphenoid, the point of presumed focal origin. The nasopharyngeal component was easily removed with no attachment found. The floor of the sphenoid sinus was drilled further as the pterygopalatine ganglion was felt to be the likely point of origin and hemostasis was achieved. The tumour was removed in two sections.The patient was extubated and had an uneventful postoperative recovery.The two separate sections of the nasal mass were sent for histopathological examination  Both sections were composed of Antoni-A and Antoni-B areas of variable cellularity consistent with those of a schwannoma. As expected, immunoperoxidase stains for S100 and SOX10 were positive. Tumour was noted to compress thin strips of bone in a submucosal distribution in all margins. There was no evidence of malignancy. Fungal microscopy and culture showed no elements or growth.The patient had a very good response to surgery at two-month follow-up, with complete resolution of symptoms. On repeat nasendoscopy, there was no evidence of residual or recurrent disease.Unilateral tumours in the nasal cavity causing nasal obstruction, pain, fullness, and epistaxis are usually caused by benign disease processes such as polyps, cysts, and mucoceles. A unilateral tumour originating from the nasal cavity should also stimulate the consideration of the rare esthesioneuroblastoma, a neoplasm originating from the olfactory neuroepithelium that has significant heterogeneity in management and variation in prognosis .Schwannomas of the nasal cavity and sinuses produce similar symptoms but are much rarer. Previous reports suggest that sinonasal schwannomas represent less than 4% of all head and neck schwannomas, with only approximately 40 cases reported as of 2001 and 100 cases as of 2014.Schwannomas are benign tumours of peripheral nerve sheaths, and it has been proposed that sinonasal schwannomas may originate from the ophthalmic or maxillary branches of the trigeminal nerve or from sympathetic or parasympathetic fibres from the carotid plexus or sphenopalatine ganglion . The majThe diagnostic workup for sinonasal schwannoma should include nasendoscopy, CT, and MR imaging of the paranasal sinuses to examine the extent of disease and to guide surgical approach for excision. As most schwannomas have focal origin and are in the most part encapsulated, these tumours are commonly amenable to endoscopic resection.This case report highlights the need for schwannoma to be included in any differential diagnosis of any soft tissue mass of the sinonasal spaces.


The ovarian cellular fibrous tumor with mitotic figure >4 per 10 high power field without moderate to severe atypia is defined as mitotically active cellular fibroma according to the 2014 World Health Organization classification. As this category is new and rare now, we described here a case of MACF and reviewed the literature.We present a case of mitotically active cellular fibroma of the ovary with 10-year history that was treated with laparoscopic surgery.We reviewed the relevant literature using PubMed search system and analyzed the previous cases.To date, only 5 cases of mitotically active cellular fibroma have been reported. Our patient is the first case of mitotically active cellular fibroma of the ovary treated with laparoscopic surgery.MACF of the ovary is a newly defined category and few cases have been reported, while prognostic factors have also not yet been fully characterized. Long-term clinical follow-up is necessary. The ovarian cellular fibrous tumors with mitotic figures >4 per 10 high-power fields (HPF) had been defined as fibrosarcoma [We treated a case of MACF, a new diagnostic category, with a long term history. And we searched previous cases using PubMed search system from 1966 to July 2015 with the terms “mitotically active cellular fibroma”, “ovary”, and “fibrosarcoma”. Then we analyzed and clarified that data of the cases have been recorded for finding important factors.A 36-year-old woman  consulted a doctor for further examination of an ovarian tumor. She had been diagnosed with a right-sided 6-cm ovarian tumor 10 years previously, since her first pregnancy at another hospital. Pelvic examination showed an adnexal hard mass without tenderness on the right side. Vaginal echography and pelvic computed tomography (CT) showed a 6-cm ovarian solid tumor. Magnetic resonance imaging (MRI) revealed a 61-mm solid tumor with heterogeneous low signal intensity in the pelvis. As such, a benign tumor such as a fibroma/fibrothecoma or malignant tumor such as a germ cell tumor or lymphoma was suspected Fig. . SubsequIn paraffin sections, the tumor cells were spindle shaped and arranged in intersecting bundles Fig. . They haIn connection with ovarian tumor, five cases were found by a PubMed search system and shown in Table In the 2003 WHO histological classification, ovarian cellular fibrous tumors with mitotic figures 3 or less per 10 HPF and no severe nuclear atypia were defined as cellular fibroma, while the fibrous tumors with mitotic figures >4 per 10 HPF and severe nuclear atypia were defined as fibrosarcoma. However, ovarian tumors with mitotic figures >4 per 10 HPF but no severe nuclear atypia were not categorized. These kinds of tumors were mostly diagnosed as ovarian fibrosarcoma. However, the clinicopathologic characteristic of this tumor is that it is significantly different from the malignant behaviors of ovarian fibrosarcoma. In 2006, Irving et al. first defined these kind of ovarian tumors as ‘MACF’ . In the To date, only five cases of MACF of the ovary have been reported in the literature. All cases were alive at time of publication. However, there were no specific diagnostic or prognostic factors.Our patient is the first case of MACF of the ovary treated with laparoscopic surgery. Ovarian benign fibrous tumors such as fibroma/fibrothecomas are often misdiagnosed as uterine myomas and malignant ovarian tumors preoperatively. However, laparoscopic surgery has been used for the management of ovarian fibroma/fibrothecomas , 9 and uWe report a case of MACF with a long term history treated by laparoscopic surgery. MACF of the ovary is a newly defined category and few cases have been reported, while prognostic factors have also not yet been fully characterized. Treatment is still not well established therefore long-term clinical follow-up including image processing, such as echography, CT, MRI etc., is necessary.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.


Richter’s hernia has an early misleading presentation with tendency to strangulation due to common lack of obstructive symptoms which may lead to delay in diagnosis and hence increased mortality. Rarely inguinal Richter’s hernia may present with an uncommon complication of spontaneous fistula. The development of spontaneous faecal fistula secondary to incarcerated inguinal hernias is much rarer among the adult population as compared to the paediatric age group. Most of these fistula have been reported from developing countries like India and Nigeria and is usually the result of poverty, lack of knowledge, neglect, late presentation and lack of proper management.A 62 years old male presented with chief complaints of multiple openings with faecal discharge in the right groin for last 20 days with no history of constipation, trauma, and urinary or other abdominal complaints. CT scan revealed a small gut loop communicating with anterior abdominal wall. Exploratory laparotomy revealed a loop of distal ileum adherent to the internal inguinal ring which was retrieved back into the abdominal cavity. There was perforation over the loop. Resection of the segment of ileum involved was done with ileo-ileal hand sewn anastomosis and the internal inguinal ring was closed from inside of the peritoneal cavity. The openings in the skin over the inguinal region were communicated with each other and laid open due to cellulitis of the area involved and pus discharge.Spontaneous faecal fistula in inguinal region following rupture of strangulated Richter’s hernia especially in adults is very rare and can occur even in absence of obstructive symptoms. In presentation of any groin swelling, there is need for an early accurate diagnosis followed by prompt treatment. The delay in its diagnosis and management may result in this rare complication of spontaneous faecal fistula. This reflects the state of health care in the developing world and needs to be addressed by the concerned authorities. Richter’s hernia is an uncommon condition in which only a circumference of the antimesenteric bowel wall is incarcerated within the hernia sac leading to ischemia, gangrene and perforation of the hollow viscus . It occuNon-availability of proper medical care and unawareness of the condition are the major factors having potential for transformation of a relatively benign condition of inguinal hernia into complicated state of incarceration and strangulation. It is extremely rare to have progression of strangulation towards the development of spontaneous faecal fistula.A 62 years old male presented to surgery emergency with chief complaints of multiple openings and faecal discharge from the openings in the right groin since 20 days. One month back he noticed a small swelling in right inguinal region after exertion. A day later, he developed fever with redness over inguinal region and few days later developed multiple openings with discharge of faecal material in the right inguinal region. No history of abdominal pain, constipation or any other bowel disturbances were present. There was no history of trauma and urinary or other abdominal complaints. There was no history suggestive of an inguinal hernia in the past, and nor was there a history of any type of surgical intervention.Patient was clinically examined and multiple openings were noticed in right inguinal region with faecal and pus discharge Fig. . Diagnos was found to be adherent to the internal inguinal ring which was retrieved back into the abdominal cavity Fig.  involvedRichter’s hernia occurs in small hernia rings large enough to entrap the partial circumference of the bowel wall, but small enough to prevent protrusion of a loop of the intestine, with firm margins commonly occuring in the femoral ring (72 %-88 %), followed by inguinal canal 12-24 %) and the abdominal wall incisional hernias (4 %-25 %). Recently several cases have been reported at laparoscopic port insertion sites also . Unusual2-24 % anAny part of intestine may get incarcerated but most commonly involves distal ileum, caecum and sigmoid colon . As onlyIn 1598, Fabricius Hildanus , reporteMajority of faecal fistula occur because of surgical intervention. The development of spontaneous scrotal faecal fistula secondary to incarcerated inguinal hernias is much rarer among the adult population as compared to the pediatric age group. Most of these spontaneous faecal fistula have been reported from developing countries like India and Nigeria  and is uEarliest in 1980 Rao et al.  reportedA case of port site Richter’s hernia presenting intestinal obstruction following laparoscopic surgery for the inguinal hernia was reported by Wegener et al.  Faecal fIn 1956 Gillespie classified patients with Richter’s hernia into three clinical groups according to the presentation of this disease. The obstructive group characterised by nausea, vomiting, peritonitis and constipation which if untreated leads to shock. The second group was post necrotic group characterised by strangulation with necrosis and perforation causing enterocutaneous fistula. The third group was dangerous group which includes patients with minimal abdominal signs . This grFriedman stated that death is the inevitable fate of the patient with a strangulated hernia, unless the resulting obstruction is relieved by operative interference, spontaneous reduction of the hernia or the formation of an external faecal fistula. He further states: “The pathologic changes taking place in the formation of a faecal fistula are in the beginning like those of any strangulated hernia: first, there is an exudation of a bloody fluid into the hernial sac, and with impairment of integrity of the bowel, infection of the fluid. As the sac wall becomes infected and edematous, the bowel perforates into, and then through the sac, thereby involving the external hernia coverings. Infection and necrosis then spread rapidly through the subcutaneous tissues, and finally rupture occurs externally through the skin, forming an external faecal fistula” .The presence of fistula allows decompression of the bowel and temporary relief of the intestinal obstruction. Unrelieved strangulation will, however, increase the likelihood of septic complications and mortality associated with intestinal obstruction. Strangulation may be associated with testicular ischemia and infarction , 23. ThiThis case report highlights one of the very few cases of spontaneous faecal fistula in inguinal region following rupture of strangulated Richter’s hernia especially in adults and stress upon suspicion of Richter’s hernia even in absence of obstructive symptoms presenting as faecal fistula and in presentation of any groin swelling, the need for an early accurate diagnosis followed by prompt treatment. The delay in its diagnosis and management may result in various complications including this rare complication of spontaneous faecal fistula. This reflects the state of health care in the developing world and needs to be addressed by the concerned authorities .Written informed consent was obtained from the patient for publication of this Case report and any accompanying images. A copy of the written consent is available for review by the Editor of this journal.


Neonatal hepatic abscess is a rare disease seen mainly in preterm following umbilical catheterisation. Liver abscess in term neonates without any predisposing factor is still rarer and only few cases have been reported in the literature. Here we report two cases of liver abscess in term neonates presenting with abdominal mass due to rupture.  Neonatal liver abscess is rare entity and till date fewer than 100 cases have been reported in literature. Generally they occur in preterm infants with certain risk factors like umbilical vein catheterization [1]. Treatment consists of aspiration, drainage of abscess with antibiotics [2]. Presentation in a full tern new born with an abdominal mass due to ruptured liver abscess into parietal wall has not been reported earlier.Case 1: A 26-day old female full term neonate was admitted to the surgical ward with history of swelling in the left upper abdomen  and fever of 5 days duration. Apart from leukocytosis , there was no sign of overt sepsis with CRP being negative. Ultrasound abdomen revealed 30×50×20 mm hypoechoic lesion in the left lobe of liver, with capsular breach and exophytic extension to the anterior abdominal wall . Liver functions tests and coagulation profile were normal. Intra-venous antibiotics with ceftriaxone and metronidazole were started and a cruciate incision was given for drainage of abscess from the parietal wall. Culture of pus from the abscess did not grow any organism. Post drainage recovery was uneventful and the patient was discharged after receiving 5 days of intra venous antibiotics which was switched to oral antibiotics for a further period of total three weeks. Follow-up at 6 weeks did not show any evidence of recurrence with ultrasound revealing a 5X2x2 mm residual cavity. Case 2:A 1-month old male child born by full term normal vaginal delivery with no history of umbilical catheterization, presented to surgical clinic with a right upper abdominal swelling of one week duration. Total leukocyte counts were 12,500/ cu.mm and CRP being negative. Ultrasound abdomen revealed a heterogeneous mass lesion in right lobe of liver segment V of size 55×34 mm with a 31×14 mm exophytic extension in the parietal wall. Computerized tomography (CT) scan showed a hypo dense multiloculated lesion of 55×34 mm in size in the right lobe of the liver with exophytic cystic component projecting into the abdominal wall measuring 32×15mm and showing thick peripheral rim enhancement . Liver function tests were normal. Alpha- fetoprotein level was 607.70 IU/ml. Aspiration of the parietal wall swelling revealed thick pus of about 15cc volume. Intravenous antibiotics with combination of ceftriaxone and metronidazole were started and subsequent pus culture showed growth of methicillin resistant Staphylococcus aureus sensitive to vancomycin and linezolid. A cruciate incision was given over the parietal swelling and drainage of the abscess was done and intravenous vancomycin was added. Patient recovered after drainage and was discharged on oral linezolid for further two weeks. A review ultrasound at 3 months showed near complete resolution of the lesion.Neonatal hepatic abscess is generally seen in premature neonates who undergo umbilical vein cannulation. Systemic sepsis, necrotizing enterocolitis, and central parenteral nutrition catheters are the other risk factors. The causative agents for neonatal liver abscess have been variable. Gram negative enteric bacteria and Staphylococcus aureus are the most common organisms isolated [5]. Rarer organisms like Candida [6], Serratia are generally seen in premature neonatal hepatic abscess. In one of our cases the culture grew methicillin resistant Staphylococcus aureus (MRSA) indicating extraneous source of infection. Community acquired MRSA (CA-MRSA) is one of the emerging pathogen which unlike nosocomially acquired MRSA (HA-MRSA), affects the children who are not exposed to health care system [7].The clinical features of these neonates are very different from infants and children. Non-specific signs like delayed capillary refill time, refusal to feed, abdominal distension are reported in neonates; thus making the diagnosis difficult. In our cases, the presentation was with abdominal wall swelling. Ultrasound abdomen is one of the main tools for the diagnosis. However sometimes it is difficult to distinguish liver abscess from other hepatic masses like hepatoblastoma, infantile hemangioendothelioma or hamartoma where computerized tomography scan may be helpful [8].Complications like rupture of liver abscess into peritoneum, pleural cavity or portal venous thrombosis are rare in neonates. Till date no cases of rupture of neonatal liver abscess into parietal wall have been reported in the literature. Treatment of hepatic abscess has evolved from open surgical drainage [9] to more conservative imaging guided percutaneous aspiration, and antibiotics therapy [2]. However, the ideal management still remains controversial. One study on neonatal hepatic abscess showed imaging guided aspiration or drainage has a good long term outcome [2]. Medical management with intravenous antibiotics is crucial and should be started at the earliest and must be guided according to the sensitivity pattern. Duration of antibiotics should be for a minimum period of three weeks [10].To conclude, rupture of liver abscess in to the anterior abdominal wall in full term neonates without any predisposing factor is a rarity. Diagnosis can be aided by clinical examination, imaging and aspiration. Outcome is favourable when managed by adequate drainage with appropriate antibiotics.Source of Support: NilConflict of Interest: None


Escherichia coli infection. Although malakoplakia usually affects the genitourinary tract, it may also be observed in the colon, stomach, lungs, liver, bones, uterus, and skin. Malakoplakia of the genitourinary system usually involves the bladder, whereas it may also affect the prostate along with the bladder. Malakoplakia of the prostate is very rare, and it may be clinically mistaken for prostatic malignancies. Definitive diagnosis is only possible through histopathological examination. This study elaborates on two patients who presented to our hospital in 2013 with high PSA levels. The primary clinical consideration was prostate carcinoma. However, these two cases were diagnosed as malakoplakia based on the results of histopathological analysis of the transrectal prostate biopsy specimen.Malakoplakia is a rarely seen inflammatory condition that is considered to develop secondary to a chronic  Malakoplakia is a rare granulomatous inflammatory condition, which usually involves the urinary bladder, and it is considered to develop as a result of a defective immune response to bacterial agents , 2. Lesi Escherichia coli sp. was grown in his urine culture. Although cystoscopy did not yield any specific results, his pelvic ultrasonography revealed a very large prostatic hypertrophy. The patient underwent transrectal ultrasound-guided 14 quadrant needle biopsy of the prostate with the primary clinical consideration of malignancy. Histopathological examination demonstrated diffuse infiltration of eosinophilic histiocytes (von Hansemann cells), plasma cells, and lymphocytes within fibromuscular stroma of the prostate. Histiocytes had distinctive intracytoplasmic inclusions with targetoid appearance (MGB)  . PAS stace (MGB) .Immunohistochemical staining demonstrated diffuse positivity and strong immune reactivity for CD68 . Levels  Escherichia coli sp. was grown in his urine culture. His serum PSA level was 100 ng/mL. Pelvic ultrasonography revealed nothing but an increased prostate volume and parenchymal calcification. The patient underwent 6-quadrant transrectal needle biopsy of the prostate with the primary consideration of malignancy. Histopathological examination of the biopsy material indicated diffuse infiltration of von Hansemann histiocytes into prostatic tissues. Like in Case  1, immunohistochemical staining revealed strong immune reactivity of these cells for CD68. Levels of pancytokeratin, alpha-methylacyl-CoA racemase (AMACR), and leucocyte common antigen (LCA) were within normal limits. Like in Case  1, PAS histochemical staining revealed intracytoplasmic bodies (Michaelis-Gutmann bodies) (MGBs). The case was diagnosed as malakoplakia. Since suspicion for malignancy could not be ruled out, thoracic and lower abdominal CT, and whole-body scans were obtained without any evidence of malignancy. Neither of the patients had bladder lesions.A 69-year-old male patient presented to our urology outpatient clinic with complaints of burning and discomfort during urination. Urinalysis revealed the presence of bacteriuria. E. coli infections of the urinary system [Michaelis and Gutmann were the first to define malakoplakia in 1902. In 1903, Hansemann coined the term malakoplakia, which was derived from a combination of malakos (soft) and plakos (plaque) in ancient Greek , 5. Malay system , 6, 7. E. coli and Klebsiella pneumoniae grow [ Escherichia coli propagation was observed in urine cultures of both cases. Malakoplakia may be mistaken for nonspecific granulomatous prostatitis in the histopathological analysis of the prostate needle biopsy specimen. Cases that morphologically resemble malakoplakia but do not contain MGBs are referred to as the nodular histiocytic prostatitis. Intravesical Bacillus Calmette-Guérin (BCG) therapy for bladder carcinoma may cause granulomatous prostatitis [Malakoplakia is a rarely seen infection, and in 80 to 90% of patients' urine cultures predominatelyiae grow –4. Some iae grow , 3. MGBsiae grow , 7, 8. Mstatitis , 6. Our Malakoplakia of the lower genitourinary system consists of sharply bordered lesions usually without pelvic organ invasion.Treatment alternatives include antibiotherapy or surgery, depending on the localisation of the involvement and degree of invasion . AntibioIn Case  1, one month after receiving antibiotherapy, the patient underwent open prostatectomy, whereas the patient in Case  2 received antibiotherapy only. Both patients had not demonstrated any pathological findings in routine controls.This study discussed the above-mentioned two cases in light of the literature in that they were rarely seen and clinically mistaken for malignancy. These cases can also be considered as pitfalls for the pathologist since they could be mistaken for malignancy.