A neonate presented three days after birth with left-sided unilateral inspiratory wheezing, intermittent respiratory distress, and desaturations. She was found to have a large ductus arteriosus aneurysm that caused compression of her left mainstem bronchus and left pulmonary artery. This lesion was not identified prior to birth on routine prenatal screening, which included fetal ultrasonography. Diagnosis was made on day of life (DOL) 5 by a computed tomography with angiography scan. On DOL 7, she underwent cardiac surgery which included resection of the ductal aneurysm, patch reconstruction of the transverse aortic arch and descending aorta, patent ductus arteriosus excision, and atrial secundum septal defect repair. There were no postoperative complications, and she has been asymptomatic since. Aneurysm of the ductus arteriosus is a rare condition that is most commonly found in early infancy, frequently may be associated with complications, and if isolated may undergo spontaneous rapid resolution.A 3.6-kg female born at 41 weeks gestation was noted on day of life (DOL) 3 to have unilateral left-sided wheezing. She had been transferred to the neonatal intensive care unit (NICU) for hyperbilirubinemia requiring phototherapy and subsequently developed respiratory difficulty. In the NICU, her initial chest radiograph (CXR) demonstrated a focal opacity near the medial aspect of her left upper lobe .Physical examination was notable for intermittent tachypnea with a maximal respiratory rate of 57 breaths per minute and mild subcostal retractions, pulse oximetry of 92% with intermittent desaturations, and inspiratory wheezing noted over her left unilateral chest. On DOL 5, a computed tomography with angiography scan (CT-A) was ordered following a pediatric pulmonology consultation. CT-A revealed a large 17-millimeter ductus arteriosus aneurysm (DAA) , which was complicated by an associated large ductal thrombus Figures . EchocarOn DOL 7, she underwent cardiac surgery due to the large size of the DAA, compression of adjacent left mainstem bronchus and pulmonary artery, and risk of rupture. The operation included resection of the ductal aneurysm and accompanying abnormal aortic ductal tissue, ligation of patent ductus arteriosus on the pulmonary arterial end, reconstruction of the distal transverse aortic arch, and descending aorta with a large bovine pericardial patch via cardiopulmonary bypass under deep hypothermic circulatory arrest, and upon rewarming, the small secundum atrial septal defect was closed. The DAA was very large in relation to the aortic arch/descending aorta and was very thin walled. Postoperatively, the patient did well and was discharged home on postoperative day 7 with no pulmonary or cardiac issues. Follow-up echocardiography and chest radiography reveal normal findings. She continues to have no respiratory complaints or abnormal clinical exam findings and is thriving.Unilateral wheezing on physical examination shortly after birth should incorporate imaging workup for anatomical causes, as opposed to assigning the origin of air flow turbulence and airway obstruction as a functional abnormality. Ductus arteriosus aneurysm (DAA) was first described at autopsy in 1827 by Martin-Saint-Ange  to as hiIn this case, the DAA was excised which revealed a significantly large 1.7 × 1.0 centimeter DAA that was nearly completely occluded by thrombus. Our patient was displaying symptoms of respiratory difficulty due to compression of her adjacent airway and vascular structures by the DAA and associated thrombus. In addition to her symptoms, the risk of rupture with the size of the DAA in our opinion warranted urgent surgical repair over conservative therapy as both strategies were discussed with the family. The excised DAA was examined by pathology and showed vascular tissue with myxoid degenerative change and haphazard arrangement of the collagen fibers. Elastic tissue stains were performed and showed incomplete elastic membrane layer, which was compared against normal control tissue .Vascular ring or pulmonary slingTumor (neoplasm vs. benign mass)Airway obstruction Congenital heart disease (noncyanotic)Congenital pulmonary airway malformation (CPAM)Bronchogenic cystAneurysm of the ductus arteriosus is a rare condition that is most commonly found in early infancy and may undergo spontaneous rapid resolution. While the pathogenesis is not entirely clear, DAA of the neonate occurs congenitally during fetal development and is typically associated with disruption of the elastic components of the ductus arteriosus. In asymptomatic cases diagnosed prenatally or incidentally and which have persisted until birth, conservative management with close monitoring and serial echocardiography until spontaneous closure may be appropriate. However, when associated with symptoms or sequelae of its presence, in addition to a large size, surgical treatment is recommended. Physical findings suggestive of air flow turbulence or airway obstruction shortly after birth should incorporate imaging workup for anatomical causes, rather than to assume the origin to be of a functional abnormality. The findings that can be included in the differential diagnosis are as follows:


Gastrointestinal stromal tumors are uncommon when compared to all gastrointestinal neoplasms but are the most common mesenchymal tumors of the gastrointestinal tract. The largest gastrointestinal stromal tumor ever recorded in literature weighed approximately 6.1 kg and measured 39 cm × 27 cm × 14 cm. About two-thirds of GISTs are malignant. The tumor size, mitotic rate, cellularity, and nuclear pleomorphism are the most important parameters when considering prognosis and recurrence. The definitive treatment for these tumors is resection. In the year 2000, the first patient was treated with the tyrosine kinase inhibitor imatinib and since then, gastrointestinal stromal tumors with high-risk features have been treated successfully with tyrosine kinase inhibitors. We present the largest gastrointestinal stromal tumor recorded in medical literature measuring 42.0 cm × 31.0 cm × 23.0 cm in maximum dimensions and weighing in at approximately 18.5 kg in a 65-year-old African-American male who presented with increased abdominal distention. The mass was successfully excised, and the patient was treated with imatinib without local or distant recurrence 1.5 years postoperatively. Although GISTs are relatively uncommon  compared with adenocarcinoma, they are the most common mesenchymal tumors of the gastrointestinal tract . AccordiThe majority of cases present as symptomatic disease. Small GISTs are usually asymptomatic and are incidentally found during radiologic evaluation, endoscopy, or at the time of surgery for another cause. Although a GIST is not a mucosa-based tumor, like adenocarcinoma, but instead grows from the muscle layer of the bowel wall, it still may be accompanied by bleeding in up to 25% of patients due to erosion of the mucosa resulting in melena, hematemesis, or symptomatic anemia. Other signs and symptoms include abdominal fullness, early satiety, a palpable mass, or abdominal pain . To our Tumor size and the mitotic count are the most commonly used prognostic factors to estimate the outcome and risk of recurrence of the tumor. Tumors arising from the stomach have a more favorable prognosis compared to other sites . TreatmeWe present a rare case of a gastric GIST larger in size than any previously documented, rapidly growing and invading into the surrounding structures that was successfully treated with surgical resection with total gastrectomy as well as en bloc resection of the pancreas, spleen, and colon with ostomy creation.GISTs are encountered frequently in the surgical clinical setting with varying sizes, invasion, and treatment modalities. We present this case for a review of GISTs and review of the surgical decision-making and to highlight the patient's success despite massive resection of major abdominal organs.A 65-year-old African-American male with no significant past medical history presented to facility with complaints of worsening abdominal distention for approximately one year, associated with dyspnea, early satiety, and weight loss of about 23 pounds. He denied chest pain, melena, or hematochezia. He had never had an endoscopy or colonoscopy performed. The patient did have a remote history of smoking of about a half pack of cigarettes per day for 10 years; however, he had quit 15 years prior. Family history was significant for a brother with lung cancer, otherwise noncontributory.On physical examination, he was noted to have bitemporal wasting and marked abdominal distension secondary to a large firm mass with ill-defined margins.A contrast-enhanced CT scan of the abdomen and pelvis showed a large, heterogeneous, partially necrotic mass measuring 38 cm × 25 cm, arising from the left upper quadrant with no evidence of metastases seen. There was displacement of the stomach and duodenum to the right and the left kidney was displaced inferiorly. The mass was suggestive of a sarcoma or possibly a GIST tumor with malignant degeneration . Chest r3 with 80% neutrophils. Hemoglobin of 10 G/dl and hematocrit 31.2% with a platelet count of 328 K/mm3. Basic metabolic panel and liver function tests and PT/INR were unremarkable.Preoperative laboratory analysis showed a white blood cell count of 5.9 K/mmThe patient was taken for an exploratory laparotomy, which revealed a large mass arising from the posterior gastric wall with a giant omental vessel as well as dilated gastroepiploic vessels. The mass was invading the spleen, distal pancreas, and the mesentery of the transverse colon. Over the next 7 hours, the mass was completely resected with a total gastrectomy, esophagojejunostomy and feeding jejunostomy, distal pancreatectomy, and splenectomy. Resection required division of the mesentery of the transverse colon, which resulted in ischemia of the transverse colon warranting resection with end colostomy creation. The tumor, spleen, distal pancreas, and transverse colon were all sent for frozen section. As expected of GISTs, pathology reports showed that the 6 harvested lymph nodes as well as the surgical margins were all negative for malignancy. During the 7-hour procedure, the patient lost nearly 3 l of blood and received 6 l of IV fluids, 12 units of packed red blood cells, 4 units of fresh-frozen plasma, 1 unit of single donor platelets, and 4 units of albumin with intermittent pushes of vasopressive medications. The patient was taken to the surgical intensive care unit postoperatively, was weaned off vasopressive medications, extubated, and started on tube feeds, and eventually fed orally. The remainder of his hospital course was uneventful, and the patient was discharged to home on postoperative day 14. The patient was started on imatinib at this time and has been maintained on that with no recurrence approximately 1.5 years postoperatively. The patient returned 8 months later for a planned colostomy take-down, which was successful and uneventful.Macroscopically, the mass was irregularly shaped, attached to the posterior-inferior aspect of the stomach, weighed 18,500 grams, and measured 42.0 × 31.0 × 23.0 cm in maximum dimensions .Histopathology showed gastric stromal tumor cells staged as high grade with >5 mitoses per high power field (hpf).Immunohistochemistry confirmed GIST with strong positive staining for CD117, DOG1, and CD34 . PostopeOur patient presented with progressively worsening abdominal distension for 9 months, weight loss, and was found to have a giant GIST causing early satiety, anemia, and locally invading into adjacent organs such as the spleen, distal pancreas, and the mesentery of the transverse colon. Despite the enormous size and weight of the tumor, our patient denied any severe abdominal pain or other associated symptoms other than abdominal distention with decreased diet tolerance.We report the largest GIST tumor reported to date. The size of our tumor measured 42.0 cm × 31.0 cm × 23.0 cm in dimensions. The largest tumor reported prior to our tumor measured 39 cm × 27 cm × 14 cm. The weight of our tumor was 18.5 kg, which is about 3 times heavier than the heaviest tumor previously reported, 6.1 kg.The treatment strategy for GIST depends on the tumor site, size, and presence or absence of metastasis. In this case, surgery was unavoidable due to the complex nature and size of the tumor as well as the associated early gastrointestinal obstruction identified by patient's decreased diet tolerance and findings on the imaging. For this reason, the decision was made to proceed with surgical intervention as opposed to neoadjuvant therapy with imatinib. Achieving an adequate en bloc resection was difficult due to the multivisceral involvement. The patient underwent a total gastrectomy, esophagojejunostomy and feeding jejunostomy, distal pancreatectomy, and splenectomy. The added difficulty during the case was after resection of the transverse mesocolon, finding evidence of a nonviable segment of bowel from the transverse colon to mid descending colon which was resected and a temporary ostomy was created. It was not advisable to create a primary anastomosis at this time given the patient's hypotension, significant blood loss, and transfusion requirement. There is no consensus on the use of neoadjuvant therapy for the treatment of GISTs. There have been approximately seven trials in which neoadjuvant chemotherapy was studied and utilized . RTOG  the tumor cells are whorls of spindle-shaped cells with eosinophilic cytoplasm and elongated nuclei; (ii) mitotic figures indicate high risk of progression; and (iii) immunoreactivity to c-KIT, CD117, or DOG1 confirms the diagnosis . HeinricIn 1998, Hirota et al. described the underlying gain function mutation leading to the development of GIST tumors, the c-kit proto-oncogene (CD117) coding the KIT receptor—a tyrosine kinase transmembrane receptor that controls crucial cell functions in tumor genesis, including proliferation, adhesion, apoptosis, and differentiation. The KIT gene mutation leads to uncontrolled cell proliferation due to stimulation of downstream signaling pathways .Different markers are used to identify KIT-negative GISTs including: calcium-dependent and receptor-activated chloride channel protein, known as DOG1 and protein kinase C theta , 20. CarAdjuvant therapy along with surgery is the preferred method of treatment when it comes to GIST. Imatinib for adjuvant treatment was approved based on the ACOSOG Z9001 study in 2008 in the USA and in 2009 in Europe. In this study, eligible patients had complete resection of a primary GIST of at least 3 cm in size. The median follow-up was 19.7 months. Adjuvant treatment for one year led to a RFS of 98% , 24.Most GIST patients will achieve the clinical benefits with imatinib, but an estimated 10% will progress within 3 to 6 months of initiating therapy. Such cases are described as showing primary resistance to treatment. Another 40% to 50% of patients will go on to develop resistance within the first two years . PrimaryWe present this case to demonstrate a successful management of the largest GIST recorded in literature with invasion into the surrounding structures requiring en bloc resection and a multidisciplinary-staged approach to treatment.


Primary angiosarcoma of the central nervous system is a rare malignant tumor with only 28 reported cases so far. Imaging findings have only been reported in a few cases. We report a case of intracranial angiosarcoma in a Caucasian male and present a review of the imaging features in the recent literature. The tumor mostly presents as a well-demarcated, heterogeneous, moderately to strongly enhancing lesion with signs of intratumoral bleeding and surrounding vasogenic edema. The differential imaging features of common hemorrhagic intracranial tumors are discussed. A 61-year-old Caucasian male with no significant medical history was admitted to the emergency department in 2006 with subacute, aggravating headaches. He had been vomiting for two days. Further clinical investigation and anamnesis revealed a mild fever of 38.0°C as well as alternating but progressive confusion and anomic aphasia for several months.An unenhanced CT yielded a 5.8 cm large and well-defined spontaneously hyperattenuating lesion in the left frontotemporal lobes, abutting the tentorium cerebelli, with a small component of the tumor expanding into the left cerebellopontine angle of the posterior fossa Figure . Within MRI showed that the lesion was mainly isointense with grey matter on both T1- and T2-weighted images. Multiple small T2-hyperintense and T1-hypointense cystic components could be seen within the lesion  and one by radiation therapy 21. Both Patients most frequently present with acute clinical deterioration, sometimes coinciding with a history of vague neurological symptoms. The acute worsening of their clinical state might be due to bleeding within the tumor, as in this case. The prognosis is comprised of either one of two contrasting outcomes: death within months after surgery [1456711131415172012391216Few reports have described the imaging characteristics in detail. It typically consists of heterogeneous lesions, with variable signal intensities on T1- and T2-weighted images. This heterogeneity has most often been reported to represent the presence of blood degradation products. Depending on the age of the bleeding, variable T1- and T2-signals can be seen. T1-hyperintense spots or components have been reported in almost every available MRI report in literature, but were not found in this case 2182021, m182031116182023211171820The differential diagnosis is that of a hemorrhagic lesion, including cavernous malformation, glioblastoma, and metastasis 23242526.232425T2-blooming sign, due to a complete hypointense rim of hemosiderin, and a typical popcorn ball appearance on T2-weighted images, found in approximately 50–67% of the lesions [The MRI features of a cavernous malformation typically consist of a lesion demonstrating a  lesions . Some leGlioblastoma multiforme (WHO grade IV astrocytoma) is the most common primary brain malignancy . The proTumors that frequently cause hemorrhagic intracranial metastases include choriocarcinoma, thyroid cancer, melanoma and renal cell carcinoma . Cancer In conclusion, primary angiosarcoma of the central nervous system most commonly presents as a well-demarcated, heterogeneous, moderately to strongly enhancing lesion with signs of bleeding within the tumor and surrounding vasogenic edema.The authors declare that they have no competing interests.


Lipoblastoma usually develops in extremities and trunk during infancy or childhood. Its occurrence in head and neck is rare.Here we present a case of lipoblastoma in 14 years old female who developed swelling in the nape of her neck. Initially the diagnosis of lymphangioma was made on the basis MRI findings but histopathology after excisional biopsy confirmed it to be lipoblastoma.Lipoblastoma can rarely present as swelling in the nape of neck in adolescents. Lipobla2A 14 years old female presented to OPD with complain of swelling at the nape of neck for the last 9 months. The swelling was small initially and then started increasing gradually in size in last 2 months. The swelling was painless and there was no discharge from swelling. The family history of patient was negative for any such swellings.On examination, a firm non-tender and normothermic swelling measuring 6 * 8 cm was found in the nape of neck with normal overlying skin. The swelling was non-mobile in both planes. There were no dilated veins or visible pulsation over swelling and the skin over swelling was pinchable. No bruit was auscultated. The examination for cervical and axillary lymph nodes was unremarkable.Ultrasound of neck showed a well capsulated and lobulated mass with cystic areas which contain internal echoes seen at the nape of neck .Fig. 1UlMRI neck gives impression of a large lobulated mass lesion approximately measuring 9.2 * 9.1 cm in size at the back of neck with main bulk on right side. Lesion is predominantly cystic with some solid component and appears hyperintense on T2W and FLAIR images, iso to hypointense on T1W images and appearance is suggestive of lymphangioma/cystic hygroma a, b.Fig.Tru-Cut biopsy was taken under aseptic measures from multiple areas and it showed only fibroadipose tissue without any granulomatous or neoplastic process. After detailed discussion, it was decided to go for excisional biopsy of mass under general anesthesia. Skin crease incision was given. Skin and subcutaneous tissue incised. The tumour was located muscle deep against vertebra. So, muscles were incised and tumour delivered to the surface. It was well circumcised mass a–c. HaemGross examination of specimen measured a 8.5 * 5 cm mass and histology revealed a circumscribed neoplasm comprised of hypocellular lobules of adipocytes with fibrous septa separating the lobules. Background stroma is myxoid with thin chicken wire vasculature. No atypia, mitoses or necrosis seen. The diagnosis of Lipoblastoma was made a–cFig. 4Post-operative course and follow up of patient was without any complication.3Lipoblastoma usually occurs during infancy and in young children and affects extremities and trunk. Presentation in adolescents and at the nape of neck is rare. There are only few other cases of occurrence of lipoblastoma in head and neck found during literature review. It is usually subcutaneous but can extend to deeper planes so it is imperative to remove the tumour completely and patient is on follow up to see any recurrence of tumor. In our case lipoblastoma present as swelling in the nape of neck in adolescent girl and also the tumour was muscle deep deep. So this case is unique in its presentation and characteristics.4Lipoblastoma can rarely present as swelling in the nape of the neck in adolescence and can involve deeper tissue than subcutaneous tissue.There are no financial or non-financial competing interest involved with this case report.No funding agency is involved and funds for publication will be contributed by all the authors from their own pockets.It is a case report and ethical approval not required.Written informed consent was obtained from the patient's parent for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Dr Shabbar Hussain Changazi and Dr Ghulam Mujtaba Ghuman made substantial contributions to conception and design of data.Dr Aamir Bashir was involved directly in acquiring data from the patient.Dr Samiullah Bhatti has been involved in revising it critically for important intellectual content.Dr Usman Ismat and Dr Mustanasr Iqbal have been involved in drafting the manuscript.This is a case report and research registration number not required.Shabbar Hussain Changazi.Not commissioned, externally peer-reviewed.


Background and Objectives: We describe a novel technique for percutaneous tumor debulking and cavity creation in patients with extensive lytic lesions of the vertebral body including posterior wall dehiscence prior to vertebral augmentation (VA) procedures. The mechanical cavity is created with a combination of curettage and vacuum suction (Q-VAC). Balloon kyphoplasty and vertebral body stenting are used to treat neoplastic vertebral lesions and might reduce the rate of cement leakage, especially in presence of posterior wall dehiscence. However, these techniques could theoretically lead to increased intravertebral pressure during balloon inflation with possible mobilization of soft tissue tumor through the posterior wall, aggravation of spinal stenosis, and resultant complications. Creation of a void or cavity prior to balloon expansion and/or cement injection would potentially reduce these risks. Materials and Methods: A curette is coaxially inserted in the vertebral body via transpedicular access trocars. The intravertebral neoplastic soft tissue is fragmented by multiple rotational and translational movements. Subsequently, vacuum aspiration is applied via one of two 10 G cannulas that had been introduced directly into the fragmented lesion, while saline is passively flushed via the contralateral cannula, with lavage of the fragmented solid and fluid-necrotic tumor parts. Results: We applied the Q-VAC technique to 35 cases of thoracic and lumbar extreme osteolysis with epidural mass before vertebral body stenting (VBS) cement augmentation. We observed extravertebral cement leakage on postoperative CT in 34% of cases, but with no clinical consequences. No patients experienced periprocedural respiratory problems or new or worsening neurological deficit. Conclusion: The Q-VAC technique, combining mechanical curettage and vacuum suction, is a safe, inexpensive, and reliable method for percutaneous intravertebral tumor debulking and cavitation prior to VA. We propose the Q-VAC technique for cases with extensive neoplastic osteolysis, especially if cortical boundaries of the posterior wall are dehiscent and an epidural soft tissue mass is present. Percutaneous vertebral augmentation (VA) with vertebroplasty (PVP), balloon kyphoplasty (BKP), or vertebral body stenting (VBS) is often performed in patients with painful, fractured, or at-risk-of-fracture neoplastic spinal lytic lesions. The main goals are reinforcement of the vertebral body, stabilization or prevention of a fracture, and pain relief ,3,4,5,6.Creation of a void or cavity prior to balloon expansion and/or cement injection seems to lower intravertebral pressure, thereby facilitating a more secure filing of the lytic defect , and hasRadiofrequency ablation (RFA) ,19, cryoThe aim of this study is to describe a new percutaneous image-guided minimally invasive technique for mechanical nonthermal intravertebral tumor debulking and cavity creation in vertebral body lytic lesion. This technique, called “Q-VAC”, combines mechanical curettage and vacuum suction with lavage. We have applied this technique to cases with extensive osteolysis of the vertebral body, widely eroding cortical boundaries and posterior wall, often in the presence of an epidural mass, prior to VBS augmentation .This is a technical note describing the procedural details and potential applications of this new technique, combining previously described and established procedures and devices ,22,23,25The Institutional Review Board approved this investigation and the patients signed a required informed consent to undergo the procedure .All interventions were performed in a mono- or biplanar angiography suite . The patients were placed under general anesthesia while in the supine position and then turned into the prone position. Intravenous antibiotic prophylaxis was administered at the beginning of the procedure. After percutaneous fluoroscopically guided insertion of two 4.5 mm (7G) caliber trocars via transpedicular access , a Kyphon Latitude II Curette T-Tip 7 or 8 mm  was coaxially inserted in the vertebral body via transpedicular access trocars. Subsequently, the curette tip was locked at 30, 60, or 90 degrees off-axis and the tissue present in the vertebral body was fragmented or “mashed-up” by multiple rotational (as a windshield wiper) and anteroposterior translational movements of the curette, while respecting the bony boundary of the vertebral body, under fluoroscopic control, until soft tissue consistency decreased due to tissue fragmentation. After retraction of the curette, a 10 G cannula was introduced into the now fragmented lesion via each access trocar. One cannula was connected to a 60-cc syringe filled with saline via a short luer-lock connection tubing and the second to a vacuum pump with a Penumbra Hi-Flow Aspiration Tubing  producing aspiration force of 242 Mbar. The aspiration was then activated and the saline solution was passively flushed from the contralateral cannula through the fragmented lesion with lavage of the fragmented solid and fluid-necrotic tumor parts. Depending on the amount of tumor extraction or suspected residual tumor, repetition of the procedure was possible and performed at operator’s discretion. After cavity creation, insertion and expansion of the VBS, followed by cement augmentation, was performed as previously described [Two illustrative cases present typical patients and treatments .We applied the Q-VAC technique to 35 cases (19/16 M/F)  of thoracic and lumbar (from T1 to L5) extreme osteolysis before VBS cement augmentation. Lytic lesions were related to solid tumor metastases in 27 cases and multiple myeloma in 8. In 21/35 cases, an extraosseous epidural mass was present on preprocedural imaging. We observed extravertebral cement leakage on postoperative CT in 34% of cases, but with no clinical consequences. No patients experienced periprocedural respiratory problems nor new or worsening neurological deficit. All treated vertebral bodies were stable at follow-up imaging, without secondary height loss.In patients with neoplastic lytic vertebral lesions, reducing pain, stabilizing fractures or lesions at risk of fracture, and ultimately improving quality of life are key elements of treatment. VA, with its technical variants, has an established role in achieving these goals ,2,5. HowCreation of a cavity prior to cement injection or intravertebral device expansion, such as balloons and VBS, might help increase safety and avoid severe adverse events. There are alternatives which have been proposed to reduce the cement migration from vertebrae like BKP , RFA andSpecific limits of BKP concern the raise of intravertebral pressure provoked during balloons inflation that might displace tumoral tissue through a dehiscent posterior wall and cause further central canal stenosis, or mobilize bone marrow fat cells and neoplastic cellular aggregates into the systemic circulation. In addition, due to balloon deflation and its removal before cement injection, the intravertebral neoplastic tissue may re-expand elastically again, obliterating the previously created cavity.RFA and cryoablation prior to cement injection result in reduction of tumor mass due to induction of necrosis ,19,20,21Another described technique for cavity creation, the percutaneous controlled ablation (coblation), utilizes a plasma field to evaporate tumor cells at low temperatures, in theory allowing subsequent low-pressure cement injection with a reduced risk of cement leaks and epidural tumor displacement ,27,28. TAlthough not truly a cavity creation technique, the bone marrow “washout” or lavage has been described firstly in a cadaveric spine model  and thenNevertheless, we found simple aspiration or washout attempts is only able to partially remove the fluid, necrotic, or bloody parts of vertebral neoplastic lesions, as in multiple myeloma, but solid vertebral lesions commonly occur in metastatic breast and lung cancer cannot be removed with simple aspiration and lavage through transpedicular cannulas. For this reason, in the Q-VAC technique, before vacuum suction and lavage, we implemented a purely mechanical cavity creation using intravertebral soft tissue mass fragmentation through a curette. The use of a coaxial curette has been described in case of sclerotic changes after vertebral body fractures to maximize height restoration during balloon kyphoplasty , but it Advantages of the Q-VAC are the creation of a true intravertebral cavity without increasing intraosseous pressure and without risk of thermal injuries to adjacent vital and nervous structures. The cavity creation adds no more than ten minutes to the procedure and does not require expensive devices. Nevertheless, the Q-VAC technique is only intended for creating a cavity in the vertebral body prior to VA, and by no means has the intent of local tumor control. Standardized oncological therapy should be considered as clinically indicated. Another limitation of this study is that it is single arm; Q-VAC has not been compared with any other debulking procedure.Application: We propose the Q-VAC technique for cases with extensive neoplastic osteolysis of the vertebral body, especially if cortical boundaries of the posterior wall are dehiscent and an epidural soft tissue mass is present. In these cases, the Q-VAC allows a minimally invasive percutaneous debulking of the soft-tissue tumor component centrally located in the vertebral body, resulting in the creation of a cavity that in turn allows safer expansion of balloons if a BKP is to be performed or if VBS or stent-screw-assisted internal fixation (SAIF procedure) ,25 is plIn our cohort, the Q-VAC technique, combining mechanical curettage and vacuum suction with lavage, is a safe, inexpensive, and reliable method for percutaneous intravertebral tumor debulking and cavitation prior to VA and its technical variants as VBS and SAIF, in extensive lytic vertebral neoplastic lesions.


Extrahepatic hydatid disease within the abdomen is uncommon, the most common site being the peritoneum. The alimentary tract is a rare site for hydatid cysts. Fistulization of abdominal hydatid cysts to the bowel lumen is a rare occurrence with few cases reported in existing literature. We report a rare case of fistulization of a peritoneal hydatid cyst to the stomach and duodenum with disseminated peritoneal and retroperitoneal hydatidosis, presenting with features of subacute intestinal obstruction. We briefly review the existing literature and discuss the confounding factors that we encountered during the diagnostic evaluation of this rare phenomenon. Hydatid disease is endemic in Asia, the Mediterranean and South America . The mosWe present an unusual case of disseminated peritoneal and retroperitoneal hydatidosis with one of the peritoneal cysts fistulizing to the stomach and duodenum. The patient presented clinically with subacute small bowel obstruction and passage of linear, membrane like structures in stools for which the initial clinico-etiological consideration was bowel ascariasis. However, typical findings on cross-sectional imaging and their correlation with past medical history led us to the diagnosis.A 54-year-old male, a shepherd by occupation, presented to us with a history of intermittent colicky pain within the central abdomen associated with episodes of bilious vomiting and progressive abdominal distension for three months. He also reported passage of linear membrane-like structures in the stools for two weeks. There were no other reported symptoms. Past medical history was significant for surgical excision of a hepatic hydatid cyst 20 years ago. An initial clinical evaluation had been carried out at a peripheral health centre, wherein the clinical suspicion was of subacute intestinal obstruction secondary to helminthic infestation of the bowel, possibly ascariasis.At presentation, the patient was afebrile with stable vital parameters. Clinical examination revealed a distended, non-tender abdomen which was dull to percussion with absent fluid thrill or shifting dullness. A right subcostal incisional scar was evident, consistent with history of prior surgical excision of a liver cyst. Auscultation revealed increased bowel sounds. The rest of the systemic examination was unremarkable.The patient’s hematological and biochemical investigations were unremarkable. Ultrasonography of the abdomen showed multiple cystic lesions within the peritoneum, the retroperitoneum as well as the pelvis, suggestive of hydatidosis. The small bowel loops appeared dilated with exaggerated peristalsis. Some of the small bowel loops within the umbilical region showed multiple small thin walled intraluminal cysts showing to and fro motion with peristalsis Figure , Video 1A diagnosis of transmural gastroduodenal fistulization of a peritoneal hydatid cyst with multiple small intraluminal cysts and membranes within the bowel, subacute small bowel obstruction and concomitant peritoneal and retroperitoneal hydatidosis was made. Echinococcal serological testing, esophagogastroduodenoscopy as well as a colonoscopy were planned but could not be performed in view of the limited financial resources at the patient's disposal. However, typical cross-sectional imaging features and the patient's past history of hydatid liver disease were adequate for making the diagnosis. The patient was started on albendazole therapy and referred to surgical gastroenterology. Following conservative management in the form of nasogastric suction and intravenous fluids for a period of 24 hours, the patient reported marked reduction in the degree of abdominal distension and pain with continuing passage of cyst components in stools. He was counselled regarding the need for surgery, especially the possibility of anaphylaxis. The technical difficulties associated with surgery for disseminated peritoneal and retroperitoneal hydatidosis, as well as the possibility of disease recurrence following surgery were also explained to the patient. The patient refused to undergo surgery, instead opting to continue prescribed medical treatment for a period of four weeks and is under follow-up at the time of writing the report.Echinococcus granulosus. Human beings are accidental intermediate hosts. The commonest sites involved by the disease in human beings are liver and lungs.Hydatid disease is an endemic zoonosis in Asia, the Mediterranean and South America . The cauExtrahepatic abdominal hydatid disease is uncommon and may involve virtually any site -4. HowevThere are exceptionally few clinical case reports of peritoneal or retroperitoneal hydatid cysts rupturing into the bowel lumen -9. PatieIn our case, there were a few important differences in clinical presentation as compared to that described in the existing literature. Firstly, the symptoms as reported by the patient were of subacute, rather than acute onset. Secondly, the patient reported no constitutional symptoms, especially fever and presented with stable vital parameters with no anaphylactoid features. Thirdly, the history of progressive abdominal distension, intermittent episodes of colicky abdominal pain and vomiting with passage of linear structures in stools as reported by the patient lead to an initial clinical suspicion of subacute small bowel obstruction due to bowel ascariasis. The presentation was confounded by two more considerations: the patient came from lower socio-economic strata and resided in a geographical region where ascariasis is endemic. It was only upon cross-sectional imaging and correlation with the patient’s remote history of surgical excision of a hepatic hydatid cyst that the diagnosis became clear. This leads us to an important clinical teaching point: the diagnosis of intestinal hydatidosis may be difficult even in areas where hydatid disease is considered endemic.Treatment options for hydatid cysts of the bowel are surgical as well as medical. Surgical treatment may entail segmental resection of the involved bowel with end to end anastomosis . In caseFistulization of peritoneal hydatid cysts to the alimentary tract is a rare occurrence which may present with features of subacute intestinal obstruction and passage of cyst components in stools. Accompanying constitutional and anaphylactoid features may be absent. The clinical features may significantly overlap with those of bowel ascariasis, which may confound clinical diagnosis. The diagnosis may be difficult, even in areas where hydatid disease is endemic. Correlation with prior history of hydatid disease and cross-sectional imaging is necessary for establishing the diagnosis. The choice of treatment, medical versus surgical, depends upon the expected degree of surgical difficulty and chances of post-surgical recurrence.


A foetal inguinoscrotal hernia is a rare abnormality. We report a case of foetal inguinoscrotal hernia diagnosed by ultrasonography at 37 weeks gestation and its spontaneous regression during the perinatal period. The imaging features, differential diagnosis, pregnancy management and outcome are discussed. A 22-year-old female in her first pregnancy was referred to our centre at 37 weeks menstrual age for obstetric ultrasonography to assess foetal wellbeing. The clinical history was unremarkable. Routine obstetric sonographic examination at 22 weeks gestation did not reveal any abnormality.Ultrasound examination showed a normal foetal biometry with adequate amniotic fluid. A mass was incidentally noted on the right side of the foetal scrotum measuring approximately 2.9 × 1.7 cm. The mass appeared heterogeneous in echotexture and showed echo movements within the lesion suggestive of peristalsis. The right testis was separately identified and displaced by the mass. The median raphe of the scrotum was displaced to the left and the left testis appeared normal . The con2 weeks later, she gave birth to a male baby by normal vaginal delivery. The birth weight of the baby was 3050 g. Surprisingly, clinical examination of the newborn revealed no evidence of scrotal swelling or hernia. Subsequent ultrasonography of the scrotum and inguinal region showed only patent processus vaginalis with a small communicating hydrocele on both sides . There w1 Herniation of the small bowel loops into the foetal scrotum occurs through the processus vaginalis. The processus vaginalis is an outpouch of the parietal peritoneum that evaginates into the scrotal fold during the second and third months of gestation. Between the seventh and ninth month of gestation, the testes descend into the scrotum through the processus vaginalis. Herniation of the bowel loops into the scrotum is expected to occur along with testicular descent.2 The processus vaginalis is obliterated nearing term, but remains patent at birth in 20% of the population and often closes during the first year of life. A patent processus vaginalis may result in failure of the testes to descend into the scrotum, communicating hydrocele or indirect inguinoscrotal hernia, but most of the cases of patent processes vaginalis remain asymptomatic throughout life.2Inguinoscrotal hernia is usually diagnosed in the neonatal period or early infancy but is very rare in the foetus, as the foetal intra-abdominal pressure is equal to the amniotic fluid. Occurrence of an inguinoscrotal hernia after birth is aided by conditions that increase the intra-abdominal pressure such as vigorous cry, prematurity, chronic lung disease, ascites and bowel pathology.1 MRI may be useful to differentiate a herniated bowel from other mass if there is no peristalsis.3 Usually the testis on the involved side is displaced peripherally by the mass and there is no flow within the mass on colour Doppler studies. Sometimes blood flow signals from the herniated mesentery may be seen within the mass and this should not be confused with tumour vascularity. The gestational age at the time of diagnosis is usually not less than 26 weeks.1In case of a foetal scrotal mass, any echo movements within the mass should be looked for first. This feature is consistent with bowel peristalsis and is pathognomonic for herniation. Here we would like to recommend observing for alteration in the contour of the scrotum along with the intrascrotal echo movements. If this feature is present, it strongly augments the diagnosis. Sometimes bowel peristalsis is not seen at the time of examination and may be absent in case of obstruction, strangulation or ischemia.4 It can be easily identified, as it shows the typical appearance of a fluid-filled anechoic space within the scrotum, outlining the testicles. Meconium hydrocele appears as an echogenic scrotal mass and is usually seen with findings of meconium peritonitis such as ascites, abdominal mass, calcifications and meconium pseudocyst.1 Testicular torsion has been described prenatally with features of an enlarged testicle and epididymis with accumulation of two layers of haemorrhagic fluid, one between the visceral and parietal layers of the tunica vaginalis and another outside the tunica vaginalis (“double ring haemorrhage”).5 Haemangioma and teratoma are unusual foetal testicular mass lesions and they can be differentiated by their vascularity on colour Doppler examination.1Other causes of a foetal scrotal mass are hydrocele, testicular torsion, haemangioma and other solid tumours. Hydrocele is the most common scrotal pathology in the prenatal period.1 In cases of uncomplicated foetal inguinoscrotal hernia, usually surgical correction will be carried out in the neonatal period, as there is a 30–40% risk of incarceration and possible strangulation in the first year of life.6After the diagnosis of foetal inguinoscrotal hernia is made, the foetus should be monitored for signs of obstruction such as dilated bowel loops and polyhydramnios. If features of obstruction are present, the delivery should be planned according to the gestational age and other obstetric parameters by a team that includes a paediatric surgeon.7 In our case, the foetal inguinoscrotal hernia had spontaneously regressed in the perinatal period and subsequent postnatal ultrasound revealed only a patent processus vaginalis and communicating hydrocele on both sides. Oudesluys-Murphy et al8 reported spontaneous regression of a clinically diagnosed inguinal hernia in pre-term female infants within the period of 2–6 months after birth. Spontaneous regression of foetal inguinoscrotal hernia in the perinatal period has not been reported so far.Very few cases of foetal inguinoscrotal hernia have been reported previously, of which all babies have undergone surgery except those who were dead.Foetal inguinoscrotal hernia is unusual. Echo movements within the scrotal mass is the diagnostic sign of a foetal inguinoscrotal hernia and the diagnosis is augmented if there is contour alteration of the scrotal wall. Prompt diagnosis of foetal inguinoscrotal hernia and effective monitoring of signs of obstruction help to plan obstetric management. Spontaneous regression of uncomplicated foetal inguinoscrotal hernia during the perinatal period is also a possible outcome, as in our case. However, more studies are needed to validate this possibility.


First described by a French physician Armand Trousseau, the Trousseau sign of malignancy is a classic example of paraneoplastic syndrome, caused by adenocarcinomas predominantly of the stomach, pancreas, and lung. The condition presents as recurring and migrating episodes of thrombophlebitis that can involve the upper and lower limbs, thoracic and abdominal wall, and the major blood vessels of the abdomen. These recurring episodes may lead to a detachment of a thrombus and the formation of pulmonary thromboembolism (PTE), often proving fatal for the patient. Herein we present a case of a 60-year-old male patient referred for autopsy. The patient was admitted with acute onset of gastrointestinal tract symptoms and after admission, his condition deteriorated rapidly, with new-onset neurological symptoms and an acute massive fatal episode of PTE. Previous medical history was uneventful, apart from several episodes of recurring lower limb thrombophlebitis for the past six months, resulting in two prior episodes of PTE. The autopsy revealed a massive PTE, with multiple thrombi in the venous vessels, including the two common iliac veins and the inferior vena cava. Histological evaluation revealed pancreatic adenocarcinoma with distant metastasis to a number of organs and abdominal thrombophlebitis with embolization of the pulmonary arteries. Trousseau syndrome, also commonly referred to as Trousseau’s sign of malignancy to avoid confusion with Trousseau’s sign of latent tetany is a type of paraneoplastic syndrome caused by adenocarcinomas, predominantly gastric, pancreatic and pulmonary, presenting as recurrent and migrating episodes of thrombophlebitis -3. ThromThe pathophysiology of the phenomena is not well established, but the mucin compounds produced by adenocarcinomas have been found to interact with the selectin family of adhesion molecules, leading to thrombocyte activation and aggregation , 4.These recurring episodes often result in detachment of thrombi and distant embolization, predominantly pulmonary thromboembolism (PTE); however, if the emboli persists in the venous system and traverse a patient foramen ovale, they can enter the systemic circulation, presenting as paradoxical cerebral embolism.A 60-year-old male patient was referred for autopsy in the General and Clinical Pathology department of the St. Marina University Hospital, Varna, Bulgaria. The patient was admitted to the Infectious Disease department with acute onset of gastrointestinal tract symptoms-lack of appetite, debilitating physical weakness, and multiple watery stools. After admission, the patient’s condition deteriorated rapidly, with new-onset neurological symptoms and an acute massive episode of PTE was registered on an electrocardiogram monitor prior to the patient’s death.Previous medical history was uneventful, apart from several episodes of recurring lower limb thrombophlebitis for the past six months, treated conservatively, resulting in two prior episodes of PTE.Upon autopsy, a massive PTE was established, with multiple thrombi in the venous vessels, including the two common iliac arteries and vena cava inferior.The gastrointestinal tract showed severe gas dilatation of the small intestine and colon with hyperemia in the mucosa across the stomach, small, and large intestine. The pancreas had severe gross changes suggestive of pancreatic adenocarcinoma-irregular borders and adhesion to surrounding tissues, solid and grayish-white in color.The central nervous system section revealed a massive hemorrhage on the left cerebral hemisphere and a lesion, measuring 2/2.4 cm in the left occipital lobe, sharply demarcated from the surrounding brain tissue.Other gross changes included several thickened zones in the peritoneum, pleura and pericardium, and more than 20 calculi in the urinary bladder.Histology from the pancreas revealed a proliferation of atypical glandular structures, with invasion into vascular structures and the surrounding adiposa and metastasis to regional lymph nodes Figures -2.Histology from the venous vessels of the abdomen showed an abundance of mixed thrombi Figure .Histology of the lungs revealed an abundance of thrombi, some with organization, and cellular emboli in the vascular tree Figure .Histology of the two lesions from brain revealed metastasis from the pancreatic adenocarcinoma, with the grossly hemorrhagic one including siderophages, showing a chronic bleeding nature to it Figure .Other sites involved from either tumor emboli or metastasis were the pericardium, liver, kidneys, adrenal glands, and spleen Figure .Trousseau’s sign of malignancy was first described by a French physician Armand Trousseau , amidst his many contributions to modern medicine including Trousseau’s sign of latent tetany, Trousseau-Lallemand bodies (now referred to as Bence-Jones proteins) [A classic example of paraneoplastic syndrome, predominantly in gastric, pancreatic and pulmonary adenocarcinomas, Trousseau’s sign of malignancy has largely lost its clinical value due to improved diagnostic processes . RelativHowever, in cases, as the one described, where there are recurring episodes of lower limb thrombophlebitis, migrating to the abdominal blood vessels, the diagnosis is difficult. Furthermore, when thrombophlebitis involves the venous system of the gastrointestinal tract, as in the case described, the clinical picture may be dominated by symptoms of the respective system and further sway the physician away from the correct diagnosis.The presented case shows a somewhat atypical presentation of Trousseau’s sign of malignancy, with recurrent lower limb thrombophlebitis migrating to the abdominal vasculature, on the background of advanced pancreatic adenocarcinoma, without any further symptoms. The value of extended clinical examinations in patients with recurrent PTE and thrombophlebitis to exclude any underlining causes, not only malignant, is a key providing adequate levels of healthcare and the prevention of further, possibly lethal, PTE episodes.


Leiomyosarcoma is a highly malignant soft tissue sarcoma. Most leiomyosarcomas of the extremities metastasize initially to the lungs, with few metastasizing to the liver. Also, it is difficult to diagnose metastases to other regions of the lung during follow-up.The first patient was a 51-year-old Japanese woman diagnosed with a leiomyosarcoma of the left distal femur. She underwent chemotherapy, followed by wide tumor excision and reconstruction using frozen autograft with total knee arthroplasty. Eleven months later, a focal lesion was observed in her right liver, despite the absence of lung metastases. Partial hepatic resection was performed, and the hepatic lesion was diagnosed a metastasis of leiomyosarcoma. Two years later, there has been no evidence of local recurrence. The second patient was a 60-year-old Japanese male diagnosed with a leiomyosarcoma of the left thigh. He underwent preoperative chemotherapy followed by wide excision. Three years later, a focal lesion was found in his medial liver, despite the absence of lung metastases. Partial hepatic resection was performed, and the hepatic lesion was diagnosed as a metastasis of leiomyosarcoma. At the latest follow-up, there has been no evidence of local recurrence.The lung is the most common site of metastases from leiomyosarcomas of the extremeties, because these metastases are hematogenous. Both our patients presented with metastases of the liver, despite the absence of lung metastases. Hepatic metastasis is commonly found in computed tomography (CT) scan. Periodic CT scans of the chest and abdomen are necessary in following-up patients who undergo resection of primary leiomyosarcomas of the extremities. There is no evidence for the efficacy of neo-adjuvant and adjuvant chemotherapy. Patients with leiomyosarcomas of the extremities and trunk have a reported metastasis rate of 34% and 5-year and 10-year overall survival rates of 64% and 46%, respectively.Leiomyosarcoma is a type of malignant soft tissue sarcoma of smooth muscle that may occur anywhere in the body. Leiomyosarcomas are relatively rare, accounting for approximately 10% of all soft tissue sarcoma, do not differ by sex and appear most commonly at around 65 years of age. The most common primary sites are the retroperitoneum, including intra-abdominal sites (35%); the uterus (30%); the extremities (19%); and the trunk, including the head and neck as well as the chest (16%).–7 Early diagnosis of lung metastases from sarcomas in other regions of the body is difficult. Although visceral and retroperitoneal soft tissue sarcomas often metastasize to the liver, hepatic metastases from soft tissue sarcomas of the extremities and trunk are rare. For example, one study found hepatic metastases in only one of 65 patients with primary soft tissue sarcomas of the extremities, and a second study reported hepatic metastases in 3 of 637 patients with primary soft tissue sarcomas of the extremities or trunk, with all 3 having primary tumors in the lower extremities. Because of their rarity, diagnosis and treatment of hepatic metastases of soft tissue sarcomas of the extremities and trunk has not been standardized. This report describes 2 patients with primary leiomyosarcomas in the lower extremities who, following treatment, developed hepatic metastases in the absence of pulmonary metastases.The lungs are the most common site of metastasis from soft tissue sarcomas of the extremities and trunk. Periodic routine examination by x-rays and computed tomography (CT) of the chest has been recommended for the diagnosis of asymptomatic pulmonary metastases.22.1D-glucose integrated with computed tomography (18F-FDG-PET) scan showed accumulation of 18F in the right liver (S6), the eleventh thoracic vertebra, and the right ilium. Histologic analysis of an ultrasound-guided needle biopsy specimen of the liver focal lesion resulted in the diagnosis of a metastatic leiomyosarcoma. A partial hepatic resection was performed to remove this lesion  and bilobar (74%). About 50% of patients develop hepatic metastases within 1 year of diagnosis of soft tissue sarcoma.Simple CT showed a clear tumorous lesion in the right liver (S6) of Patient 1 at 11 months after primary tumor resection. The metastatic lesion was solitary and resectable. Contrast CT showed a tumorous lesion in the middle liver of Patient 2 three years after primary tumor resection. This metastatic lesion was also solitary and resectable. Hepatic metastases differ in patients with primary leiomyosarcoma of the extremities and primary intra-abdominal leiomyosarcoma, in that the former are more likely to be solitary, and may take a longer time to develop following resection of the primary tumor. These tumors have response rates of 18% to doxorubicin and 44% to the combination of doxorubicin and dacarbazine. Surgical resection is more effective than chemotherapy or intravascular treatment in patients with leiomyosarcoma and hepatic metastasis. In the absence of surgical resection, these patients have a 5-year survival rate of only 4%. In contrast, the 5-year survival rate following resection has been reported to range from 20% to 30%.,14In the absence of treatment, the median overall survival of patients with leiomyosarcoma and hepatic metastasis is approximately 14 months.Both of our patients were candidates for surgical resection, as both had solitary metastatic lesions. Patients 1 and 2 have survived for 2 and 1 years, respectively, after surgical resection for hepatic metastasis, with neither showing evidence of recurrence or a new metastatic lesion. Patients who have undergone surgical resection for a solitary hepatic metastasis, especially when detected at an early stage, have a good prognosis. Patients who have undergone resection of primary leiomyosarcoma of the extremities should undergo every three months CT scans of the chest and abdomen to detect hepatic metastases.Because hepatic metastasis of primary leiomyosarcoma of the extremities is very rare, its features cannot be clarified. Assessments of an additional number of patients may reveal the characteristics of these lesions.4This report describes 2 patients with leiomyosarcomas originating in the lower extremities, accompanied by metastases to the liver without metastases to the lung. Periodic CT scans of the chest and abdomen are required to detect hepatic metastases following resection of the primary tumor. Patients with a solitary metastatic lesion to the liver, especially when detected at an early stage, have a good prognosis following metastasectomy.No other persons accept the authors contributed toward the article.Conceptualization: Toshiharu ShiraiData curation: Eiichi Konishi.Formal analysis: Eiichi Konishi.Investigation: Naoki Mizoshiri, Toshiharu Shirai, Ryu Terauchi, Shinji Tsuchida, Yuki Mori, Yusei Katsuyama, Daichi Hayashi.Methodology: Naoki Mizoshiri, Toshiharu Shirai, Ryu Terauchi, Shinji Tsuchida, Yuki Mori, Yusei Katsuyama, Daichi Hayashi.Project administration: Toshiharu Shirai.Supervision: Toshiharu Shirai, Ryu Terauchi, Eiichi Konishi, Toshikazu Kubo.Validation: Toshiharu Shirai, Eiichi Konishi.Visualization: Eiichi Konishi.Writing – original draft: Naoki Mizoshiri.Writing – review & editing: Toshiharu Shirai.


Meshes are commonly employed in abdominal hernia repair to reduce recurrence rates. Prosthetic repair, however, increases the risk of mesh related complications, including migration into adjacent viscera and erosion which can occur as uncommon and can be difficult to be diagnose.This is a case of transmural migration of composite mesh into the bowel, presenting as chronic abdominal pain and anemia 14 years after incisional hernia repair.Mesh implantation in hernia repair has increased the incidence of complications, such as seroma, hematoma and infection. Migration into adjacent viscera and erosion may present as complications related to the use of meshes. Their precise frequency after abdominal wall hernia repair is not well known and their late occurrence can make the diagnosis difficult.Transmural migration of composite mesh is an uncommon complication of incisional hernia repair. Its pathogenesis is still not completely clear but it has been reported many years after implant surgery. It should be considered in a typical presentation of patients with history of previous prosthetic ventral hernia repair. IncisioWe report an unusual case of transmural mesh migration of a composite mesh into the bowel, presenting at the OUR INSTITUTION as chronic abdominal pain and anemia, 14 years after incisional hernia repair.2A retired 76 year-old Caucasian man, BMI 27, in July 2017 was referred to OUR INSTITUTION with a 2-year history of persistent abdominal pain, resistant to analgesics (Paracetamol and Ketoprofen), irregular bowel habits and rectorrhagia. He reported a 1-year history of iron-deficiency anemia (≈7 g/dl), treated with blood transfusions and investigated with upper and lower endoscopy. The patient had a medical history significant for type 2 diabetes mellitus, treated with Metformin, and small cell carcinoma of the bladder, treated with radical cystectomy and orthotopic ileal neobladder, radiotherapy was not performed. In 2003 he was diagnosed an incisional hernia and underwent open prosthetic repair with a composite mesh  implanted in intraperitoneal position.®-Ethicon) was used to reconstruct the posterior fascia of the rectus muscles. A transversus abdominis release (TAR) could not be used due to the critical conditions of the posterior components of the abdominal wall.At admission to our Unit, a physical exam showed a well-healed midline laparotomy incision with no evidence of hernia. As completion to previous endoscopic procedures, a CT scan was performed showing entero-enteric fistulae and migration of prosthesis into adherent intestinal loops , Fig. 2.®, Integra LifeScience) was positioned in the retromuscular site. A Prevena™ Incisional System (KCI) was used to protect the skin and removed after 6 days. A small dehiscence of the lower third of the surgical wound was found and treated with V.A.C.® Therapy (KCI) for one week, and then with advanced wound care. The patient was discharged on the 23rd postoperative day. The 3-month clinical examination showed the surgical wound well healed. After 9 months the patient is still in good health, with complete resolution of previous anemia.According to VHWG grade III, an appropriately shaped, not cross-linked, 20 × 30 cm biological implant , can appear over a period of several years, making the diagnosis very challenging. Recently, an increasing number of cases of mesh adhesion, erosion and migration into hollow organs has been reported in the literature, suggesting the appearance of a new pathology, though the complete intraluminal migration remains a very uncommon event.Causes of mesh migration are still not clear. Two different mechanisms have been hypothesized, a primary mechanical migration and a secondary migration . The firNone of these theories would fully explain the outcome of the reported patient, having had a peculiar evolution as it presented as microcytic chronic anemia of unknown origin 14 years after open prosthetic incisional hernia repair, without evidence of chronic infection , blind loop with breath testing or short gut with other markers of malnutrition. A case of transmural mesh migration after laparoscopic abdominal wall repair, showing a similar unusual presentation was recently published . Both thThe earliest report of migration was published by Herrera  in 1976,The growing number of reports of MRVC has elicited greater concern in recent years about the placement of prosthetic material in direct contact with the viscera, due to the increase of the risk of adhesions, erosions, and fistula formation. The choice of the proper prosthetic material, suitable to minimize adhesion-formation would be elemental. Manufacturers continuously develop new meshes recommended for intraperitoneal use but their effectiveness is unproven at the moment and it’s still far from the ideal prosthesis .® layer was placed as temporary barrier between the biologic implant  and viscera below, to reconstruct the posterior rectus sheath and minimise adhesion formation.Composite meshes are particularly designed for intraperitoneal placement, with an absorbable or permanent barrier on the internal side to reduce adhesion formation. In our patient, a Composix™ E/X Mesh (Bard) was implanted as reinforcement in the previous abdominal wall repair. In this prosthesis, the inner e-PTFE barrier should decrease the risk of adhesions. Adhesions still developed, leading to erosion, followed by mesh migration. In our experimental study, we demonstrated that prostheses manufactured for intraperitoneal use decrease, but do not eliminate, adhesion formation and consequent complications . For thiOur patient had an unusual presentation as he complained non-specific persistent pain and indefinite gastro-intestinal symptoms with chronic microcytic anemia, similar to the case described by Voisard and Feldman .4Prosthetic abdominal wall repair may result in significant mesh related specific complications.Mesh migration and erosion, are uncommon complications of prosthetic incisional hernia repair. They may occur also several years after abdominal wall repair and should be considered in atypical patient presentations. We here report a case of migration of a composite mesh, 14 years after a prosthetic incisional hernia repair. The peculiarity of this case lies in the unusual presentation with persistent abdominal pain and microcytic anemia. This is a reminder that in patients complaining abdominal pain and chronic microcytic anemia after previous prosthetic abdominal wall repair, mesh-related complications, as mesh erosion and possible migration, should be considered as possible etiology, when all other causes of GI symptoms have been ruled out.All authors declare that there are no financial and personal relationships with other people or organisations that could inappropriately influence their work.All authors declare that any funding from any source has been received to perform the study.No ethical approval is requested because we are submitting a Case Report and not a research study.Consequently, no ethical approval from any committee has been submitted.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Ceci Francesca M.D.: Conception and design of the work, data analysis and interpretation, drafting the paper.D’Amore Linda M.D.: Drafting the paper, data interpretation Annesi Elena M.D.: data collection.Bambi Lucia M.D.: Data collection.Grimaldi Maria Romana M.D.: Data collection Gossetti Francesco M.D.: data interpretation.Negro Paolo M.D.: Data interpretation, critical revision of the paper, final approval of the version to be published.No registration is requested.Francesca Ceci, MD, PhD.Commissioned, externally peer-reviewed.


T2 weighted imaging. Positron emission tomography demonstrated marked fluorodeoxyglucose uptake in the solid component. One case with MRI 3 years before diagnosis indicated that the tumour arose in ovarian endometriotic cyst. EBT cases were difficult to distinguish from malignant ovarian tumours preoperatively. Intraoperative frozen section analysis may aid to determine treatment. Prognoses were excellent. Care should be taken for co-existing endometrial cancer.Endometrioid borderline tumours (EBTs) of the ovary are uncommon tumours of low malignant potential. They consist of atypical endometrioid cells lacking destructive stromal invasion. As the prognosis of EBT is excellent, excessive treatment should be avoided and preoperative diagnosis is important. Here we report four cases of ovarian EBTs along with imaging findings and a review of literature. The average patient age was 52 years. They presented with abdominal discomfort or abnormal vaginal bleeding. The final diagnoses for all four cases were EBT Stage IA with endometriosis. Pathologically, one case was an adenofibromatous type tumour, and three cases were intracystic type tumours. Two patients had concurrent endometrial cancer. MRI of the tumours showed enhanced solid components. The intracystic type tumours presented a dendritic structure in the cyst; fine papillary branches surrounded a low-signal trunk on  Here we report four cases of EBTs diagnosed at our institution.Endometrioid borderline tumours (EBTs) of the ovary are defined as tumours of borderline malignancy, which consist of atypical endometrioid epithelium but lack destructive stromal invasion.T1 weighted image (WI), indicating haemorrhage. A large enhanced solid component showed marked fluorodeoxyglucose (FDG) uptake on positron emission tomography (PET)  . A maligThe patient was a 65-year-old, gravida 2, para 2 post- menopausal female. She presented with vaginal bleeding. She was pointed out a pelvic mass on clinical examination and ultrasound. MRI showed a multiloculated mass measuring 15 cm of mixed high and low signal intensities on T1WI. Within a cyst, irregular shaped papillary projections were observed at the tumour wall on T2WI and were well enhanced indicating solid components . This leThe patient was a 54-year-old, gravida 2, para 2 post-menopausal female. She presented with lower abdominal discomfort and a right ovarian mass was pointed out by pelvic examination. Endometrial cytology detected adenocarcinoma cells. MRI and CT revealed a complex solid and cystic lesion of 8.4 cm diameter within the right ovary. A papillary lesion was observed on the tumour septum on T2WI and enhanced on post-contrast CT . PET-CT The patient was a 41-year-old, gravida 0 female. She was diagnosed with a left ovarian endometriotic cyst and uterine myomas when she was 34-years-old. At the age of 36, she was diagnosed with breast cancer and underwent left mastectomy and axillary lymphadenectomy, chemotherapy, and radiotherapy. She then commenced gonadotropin-releasing hormone agonist and tamoxifen. MRI at the age of 37 revealed an endometriotic cyst in her left ovary; the cyst showing high intensity on T2WI and intermediate-to-low intensity on T1WI without any solid component . At the 51 In the present case report, one adenofibromatous (Case 1) and three intracystic (Cases 2–4) types were described  , including 4 cases from our institution, were associated with endometriosis .3–6 Amonts (53%) . In the ts (53%) . Case 4,T2 WI. In Cases 2–4, which were intracystic type EBTs, showed a thickened stroma corresponding to low-signal intensity on T2 WI in the center of the solid portion, and at the periphery, fine branch-like structures of low-signal intensity were observed. MRI findings of the intracystic type EBTs were similar to serous borderline tumour (SBT) of the ovary,8 but reflecting abundant stroma characteristic in EBT histology, low-intensity portion on T2 WI was thicker than that in SBT. Multicystic portions showed stained-glass patterns with various intensities. Case 4 showed a solid portion in a single cyst. In this case, MRI taken 3 years prior to diagnosis showed endometriotic cyst of the ovary, indicating that the tumour arose in endometriosis tissue. Three patients in the current study underwent PET-CT, and all three showed marked FDG uptake in the solid portion of the tumour . However, in Case 1 the pathologist stated that endometrioid carcinoma could not be excluded. Cross et al reported that in their case series of 22 endometrioid tumours, 19 cases that were diagnosed as borderline tumours by intraoperative frozen section analysis were ultimately diagnosed as malignant tumours.10 This data suggests that endometrioid tumours tend to be under-diagnosed. Because of the high suspicion for malignant tumours based on preoperative imaging studies we performed staging laparotomy including lymphadenectomy in all four cases. As EBTs are very rare, more research with larger case series is needed to determine the reliability of frozen section analyses.Intraoperative consultation based on the pathology of frozen sections of tumour may aid in deciding adequate operative procedures for the ovarian tumour.We reported four cases of EBT along with a review of the literature.Imaging studies of our four cases of EBT presented mixed solid and cystic lesions, often including haemorrhagic cysts, with papillary solid projections demonstrating low signal intensity on T2WI. Enhancement, water restriction, and FDG uptake were observed in the solid components, making the cases almost indistinguishable from malignant ovarian lesions. Intraoperative frozen section analysis may help determine treatment. Concurrent endometrial lesions including endometrial cancer need to be considered and excluded.MRI in EBT of the ovary demonstrated heterogeneous tumours containing both solid and cystic components. The solid components demonstrated enhancement and water restriction, making them difficult to distinguish from malignant tumours.Many EBT cases are accompanied with endometriosis and some arise in endometriotic cysts.Concurrent endometrial lesions are frequently encountered, especially endometrial cancer.Written informed consent for the case to be published  was obtained from the patient(s) for publication of this case report, including accompanying images.


Malignant peritoneal mesothelioma is a rare aggressive tumor of the peritoneum. We report a rare case of resection of multiple localized malignant peritoneal mesotheliomas.18F-fluorodeoxyglucose positron emission tomography. It was very difficult to obtain a definitive preoperative diagnosis, but surgical resection was performed because we considered potential malignancy. Laparotomy revealed the principal site of the tumor in the lateral segment was on the hepatoduodenal ligament, and all other tumors were on the diaphragm. A left lobectomy and partial diaphragmatic resection were performed. The final pathological diagnosis was multiple malignant epithelioid mesotheliomas. Our patient has had no recurrence for 20 months postoperatively.A 55-year-old Japanese woman was admitted to our hospital because liver tumors were detected by abdominal ultrasonography during a screening examination. Blood examination findings, including tumor makers, were within normal ranges. She had no evidence of exposure to asbestos. Computed tomography showed four hypervascular, round liver tumors, one in the lateral liver segment adjacent to the hepatic hilus, and the other three on the liver surface. Computed tomography angiography revealed that the tumor in the lateral segment had strong enhancement and was fed from the left gastric artery. In contrast, the other tumors showed no enhancement, and were fed from the right inferior phrenic artery. Abnormal accumulation was identified in the four tumors only with In general, malignant peritoneal mesotheliomas are classified as diffuse tumors, which are often unresectable and have a poor prognosis. However, early diagnosis and treatment, particularly with the localized type, as in our patient, could lead to long-term survival of the patient. We recommend that multiple malignant epithelioid mesotheliomas be included in the differential diagnosis for patients with subcapsular hepatic tumors. Malignant peritoneal mesothelioma (MPM) is a rare but rapidly fatal malignancy. MPM is difficult to diagnose because clinical symptoms and findings are nonspecific. The median survival ranges from 5 to 12 months, mainly because of a lack of effective treatment [A 55-year-old Japanese woman was referred to our hospital because of hepatic tumors detected by abdominal ultrasonography during a screening examination. Her medical and family history was unremarkable; her occupational and residential history showed no apparent exposure to asbestos. She received no medical drugs, and neither smoked tobacco nor drank alcohol. On admission, her general status was unremarkable; her temperature was 36.2 °C and her blood pressure was 110/70 mmHg with a regular heart rate of 80/minute. A physical examination revealed no abnormal findings; neurological examinations were unremarkable. All laboratory data, including levels of the serum tumor markers carcinoembryonic antigen, cancer antigen 19-9, alpha fetoprotein, protein induced by vitamin K absence or antagonist-II, and cancer antigen 125 were within normal ranges -CT (Fig. CT findings during arterial portography showed enhancement in none of the tumors Fig. a and b, -CT Fig. . We cons-CT Fig. a–c. We p-CT Fig.  and partIn the present case report, we present a rare case of MPM which is an aggressive tumor and very difficult to diagnose. Moreover, the multiple localized MPMs seen in our patient are extremely rare and, to the best of our knowledge, have not been addressed in the literature. However, our patient was successfully diagnosed as having peritoneal malignant tumor preoperatively and treated with surgical resection.MPM is a rare neoplastic condition that arises from the serosal membranes of the abdominal cavity and is classified as either diffuse or localized . MPM preDiagnosis of multiple localized MPMs can be very difficult preoperatively and should be considered in any patient with subcapsular hepatic tumors. MPM often presents as an incidental finding or with nonspecific symptoms. A physical examination does not reveal abnormalities until the disease has advanced significantly, and standard laboratory and radiographic studies are often inconclusive , 5. Asciet al. described 23 cases of localized malignant mesothelioma, and 50% of patients were alive after several years of follow-up, in contrast to those with diffuse malignant mesothelioma [This case report highlights aspects of diagnosis and management of a very rare disease that presents with atypical characteristics in preoperative imaging. At first, the disease presentation resembles extrahepatic tumor growth, mimicking hepatic multiple hemangiomas. However, all the tumors in our patient were located on the edge of the liver, and the density of one of the tumors matched the density of slight peritoneal thickening of the diaphragm, suggesting a tumor arising from the peritoneal wall. The identification of feeding vessels with CT angiography proved key to the diagnosis of peritoneal tumor in our patient. We considered MPM as one of the preoperative differential diagnoses, and started early treatment. The prognosis for patients with MPM is poor, and the management of localized MPM is controversial. However, in the largest series to date, Allen and Cagle Because of its rarity and minimal available treatment information, it is difficult to make a primary diagnosis, and by the time of definitive diagnosis, most patients have wide tumor spread to other organs. However, surgeons should consider a peritoneal tumor in the differential diagnosis, and identification of feeding vessels with contrast angiography or CT angiography as an auxiliary diagnostic method for localized MPM, especially for tumors on the subcapsular liver.In general, MPM is a very rare disease and may mimic a benign tumor radiologically. MPM has a poor prognosis; however, early diagnosis and treatment, particularly with the localized type, as in our patient, could lead to long-term survival of the patient. MPM should be included in the differential diagnoses of patients with subcapsular hepatic tumors, and we believe that this case report will be useful for future clinical diagnosis and treatment of the disease.


Malignant granular cell tumor (MGCT) is a rare high-grade mesenchymal tumor of Schwann cell origin. MGCTs commonly affect thigh, extremity, and trunk; however, involvement of the abdominal wall is quite rare. It has poor prognosis with 39% mortality rate in 3-year interval. We report a 50-year-old female who had MGCT arising in the anterior abdominal wall and developed massive metastatic deposits in both lungs and in the right inguinal lymph nodes, with prolonged survival for 11 years. A brief review of the literature is presented. Granular cell tumors (GCTs), previously known as granular cell myoblastoma, are uncommon mesenchymal neoplasms of Schwann cell origin, composed of cells with granular eosinophilic cytoplasm, usually presenting as asymptomatic nodule, affecting adults, with women affected twice as men . They ocOn the other hand, malignant granular cell tumors (MGCTs) are exceedingly rare high-grade malignant mesenchymal neoplasms, representing only 1%-2% of all GCTs . In a stMost MGCTs commonly affect lower extremities, especially thighs and the trunk; however, involvement of the subcutaneous tissue of the abdominal wall is exceedingly rare . Herein, we report a case of MGCT arising in the anterior abdominal wall, who underwent surgical excision with wide resection margin and received chemotherapy, but developed massive metastases in the lungs and the right inguinal lymph nodes, with prolonged survival for 11 years, with a review of the relevant literature.A 50-year-old female presented in July 2007 to a peripheral general hospital with progressively enlarging mass in the anterior abdominal wall of one-year duration. On physical examination, she was well built with normal vital signs. A 7 x 6 cm firm irregular subcutaneous mass was felt in the right lower para-umbilical area of the anterior abdominal wall. On palpation, the abdomen was soft and lax with no intra-abdominal masses. There were no palpable lymph nodes. Her cardiac and respiratory examinations were unremarkable. Computerized Tomography (CT) scan of the abdomen revealed a mass within the abdominal wall. Her laboratory investigations were within normal limits. A plain chest X-ray was unremarkable.Wide local excision with safety margins of the tumor was done. The postoperative period was uneventful, and she was discharged on the second postoperative day in a satisfactory condition.Gross examination of the specimen showed an ellipse of skin that measured 13 x 10 cm with subcutaneous fatty tissue measuring 11 cm in thickness. On cut section, a well-defined nonencapsulated grayish yellow mass measuring 7 x 6 cm with foci of necrosis was seen. No skin infiltration or abdominal skeletal muscle involvement was noted.Microscopic examination revealed well-defined but nonencapsulated subcutaneous mesenchymal neoplasm composed of irregular islands of large cells with mostly round to oval pleomorphic vesicular nuclei, with markedly granular cytoplasm, and several large eosinophilic globules . The nucMany foci of tumor necrosis and scattered mitotic figures, 4 per 10 HPF at a magnification of 200, were identified Figures . MarginsImmunohistochemical stains show the tumor cells to be strongly positive for vimentin, S100 and CD68 Figures . Stains The patient was referred in September 2007 to the Oncology Clinic at our hospital for regular follow-up. A chest CT done on May 2008 showed small right lung nodules measuring 0.48 cm of uncertain significance. In April 2010, a follow-up of her chest CT showed bilateral multiple lung nodules; the largest was in the right upper lobe measuring 1.5 x 1 cm. Pelvic CT on February 2011 revealed a large right inguinal mass measuring 3.2 x 2.1 cm. Palliative chemotherapy was planned for the patient. She was started on doxorubicin and ifosfamide for 6 cycles; the last cycle was in October 2011 and then she refused to continue treatment.In September 2012, FNAC of the inguinal lymph node showed numerous clusters and single cells with abundant granular cytoplasm and large pleomorphic nuclei.In December of 2015 she was started on docetaxel single agent for 6 cycles; the last cycle was completed by April 2016. In January 2018, her last follow-up chest CT reveled an increase in the size of all metastatic nodules. The largest nodule was in the right upper lobe and measured up to 5.2 x 4.9 cm. It had increased in size since July 2017 when it measured at that time 4.4 x 4.3 cm . In addiAlexei I Abrikossoff, a Russian pathologist, described the first case of benign GCT in the skeletal muscle of the tongue in 1926. He thought that these tumors were of skeletal muscle cells origin and called them “myoblastoma” . AlthougThere are several large series of GCTs. In a study of 95 patients from Memorial Hospital in New York, with their age ranging from 11 months to 68 years, an average of 38.1 years, there were 92 benign GCTs and three MGCTs. There were 31 males and 64 females with male to female ratio of 1:2. The average size of the tumors was 1.85 cm. Multiple lesions were encountered in 8 patients. The tumor distribution was ubiquitous in many anatomic locations, including two in the abdominal wall . AnotherOn the other hand, MGCT is a rare and aggressive tumor, representing less than 2% of all GCT and 0.2% of all soft tissue sarcomas , 9. OrdoHistologically, sheets of polygonal to spindly cells were shown, with an abundant granular eosinophilic cytoplasm with scattered globules; both are positive for Periodic Acid-Schiff (PAS) stain . ImmunohThe abdominal wall is an unusual site for benign and malignant GCT; 29 cases were reported in the literature, including our case . Eleven In 1998, Fanburg-Smith et al. established the criteria for diagnosing benign, atypical, and malignant granular cell tumors based on histological features which included necrosis, spindling, vesicular nuclei with prominent nucleoli, increased mitotic activity (greater than 2 mitoses/10 HPF at a magnification of 200), high nuclear cytoplasmic ratio, and pleomorphism. MGCT was diagnosed when three or more of these features were present. Neoplasms were classified as atypical when one or two of those features were present and benign if there was only focal pleomorphism but no other features were present. Also, a tumor was considered malignant when it metastasized or caused patient death regardless of the cytological features. Metastases were the only conclusive criteria for malignancy .The present case shows at least 4 histological criteria set by Fanburg-Smith et al., namely, 4 mitosis/ 10 HPF x 200, nuclear pleomorphism, foci of necrosis, and high nuclear cytoplasmic ratio. However, the presence of subsequent metastatic deposits in both lungs and the right inguinal lymph nodes is definite feature of malignancy.We utilized fine-needle aspiration cytology (FNAC) to establish the diagnosis of metastatic deposits of MGCT in the right inguinal lymph node. Several papers documented the importance of FNAC in diagnosing GCTs , 44, 45.The differential diagnosis of MGCT includes pleomorphic rhabdomyosarcoma, granular leiomyosarcoma, and alveolar soft part sarcoma (ASPS) . MGCTs aMGCTs have a poor prognosis with 32% local recurrence and 50% metastatic rate . It can The treatment of choice is complete surgical resection with safe margins and regional lymph node dissection. Marked resistance of the tumor to radiotherapy and chemotherapy made them of low value , 9. SomeOur patient had prolonged survival in spite of the presence of metastatic tumor deposits in both lungs and right inguinal lymph node. She received chemotherapy but it is not clear whether this contributed to her long survival or not. The presence of metastases is currently considered as the only unequivocal sign of true malignancy. This opinion is supported by some degree of overlap in clinicopathologic data between benign and malignant GCTs , 2. Only


Its rarity makes preoperative suspicion and diagnosis very difficult in an emergency setting.we retrospectively analyzed the database of patients admitted in a single center emergency unit for small bowel occlusion (SBO) in virgin abdomen and treated by surgery from August 2013 to October 2018. The patients with intraoperative diagnosis of ISH were included in this study.from 29 patients with virgin abdomen submitted to surgery for SBO, two cases of ISH were recorded. In both cases preoperative diagnosis was made by CT scan and urgent treatment was successfully performed by laparoscopy, reducing the entrapped small bowel and closing the hernia's ring by continuous suture. No intestinal resection was needed.urgent laparotomic repair of obstructive ISH is the standard treatment although laparoscopic approach has also been described in a small number of cases. We reported our experience on two cases in which totally laparoscopic treatment was successfully performed in patients with stable hemodynamic parameters thanks to early diagnosis and limited intestinal distension. By an extensive analysis of the international literature, clinical, diagnostic and therapeutic aspects of this form of internal hernia were discussed.CT scan facilitates early ISH preoperative diagnosis, reducing the risk of small bowel resection and increasing the chances of minimally invasive laparoscopic treatment. •Internal supravesical hernia is a rare condition presenting with acute small bowel obstruction.•We present two cases in which laparoscopic treatment has been successfully performed.•A complete review of the literature was also reported. AccordiPatients with small bowel obstruction (SBO) and surgically virgin abdomen, have been retrospectively evaluated, detecting two cases of obstructive ISH treated by laparoscopy. The available international literature of the last years was also collected to highlight and discuss clinical, diagnostic and therapeutic aspect of this rare form of internal hernia. The work has been reported in line with PROCESS criteria .2The database of patients submitted to surgery for SBO in the Surgical Units of University of L'Aquila from August 2013 to October 2018, has been retrospectively analyzed. The patients with intraoperative diagnosis of ISH were included in this study. Using PubMed and Goggle Scholar database, we reviewed the international literature using the MeSH terms “paravesical”, “supravesical”, “internal hernia”, “small bowel occlusion” to identify the cases treated by surgery after 1995. This study has been registered on the Research Registry with the Unique Identifying Number 4265.3Twenty-nine patients with virgin abdomen underwent surgery for SBO during the analyzed period. Two cases of ISH were recorded. Both procedures were performed by two surgeons skilled in mini-invasive management of abdominal urgencies . From the available international literature, we identified 26 previous cases of internal supravesical or paravesical hernia treated by urgent laparotomic or laparoscopic surgery .Table 1L3.1A 48-year-old healthy man with BMI 30 was admitted to the emergency department with a 12-h duration history of abdominal pain and distension with bilious vomiting. The patient had no history of previous similar attacks. On examination, the patient was dehydrated, afebrile with a pulse minute rate of 98 and blood pressure of 130/80. His abdomen was distended, tympanic on percussion and tinkling bowel sounds were auscultated. The clinical suspect of bowel occlusion was confirmed by an abdominal X-ray in the upright position that revealed multiple fluid levels without free air. The abdominal and pelvic CT scan with intravenous contrast identified an SBO with a transizional zone in the right lower abdomen. Below the transitional zone there was a saclike mass of clustered dilated bowel loops descending downward into the prevesical space and compressing the anterolateral wall of the bladder . The pat3.2A 65-year-old healthy man with BMI 32 was admitted to the emergency department. He referred 6-h before symptoms, following heavy cough, mostly related to abdominal pain and nausea, with one episode of bilious vomiting but no clinical evidence of heavy abdominal distension. The patient had no history of previous similar episodes. Clinical examination revealed dehydratation, high body temperature (38.2 °C), pulse rate of 98/minute and mild hypotension (blood pressure 110/60 mmHg). His abdomen was little distended, mostly tympanic on percussion and tinkling bowel sounds were auscultated especially on the right inferior quadrants. To confirm the suspect of bowel occlusion, an abdominal X-ray in the upright position was performed revealing multiple fluid levels without free air in the peritoneal cavity. Abdominal and pelvic CT scan without intravenous contrast identified a small bowel obstruction with a transitional zone in the right lower abdomen, starting from a saclike mass of clustered dilated bowel loops descending into the prevesical space and compressing the anterolateral wall of the bladder . The pat4Internal prevesical hernia, judging from the number of cases reported, is a rare form of internal hernia. Rising from the supravesical fossa, it may develop anteriorly or laterally to the bladder, protruding into prevesical space of Retzius or into the paravesical space, respectively. This different way of bulging generated various titles with which it has been mentioned in the literature: prevesical, supravesical, anterior retroperitoneal, properitoneal, paravesical . The firThese data were confirmed in our patients complaining symptoms suggestive for SBO beginning 6 and 12 h respectively before the access to emergency department. Although characteristic CT findings of strangulated supravesical internal hernia have been described [Quite almost reported cases were treated by open laparotomy , whereasLike for others rare conditions, the surgical treatment is no standardized. Reduction of the herniated loops with closure of the hernia's sac using continuous or interrupted sutures is considered sufficient whereas complete or partial resection of the sac was considered unnecessary and performed in only few of laparotomic reported cases . In our Even if prosthetic repair is the gold standard for inguinal and incisional hernias, mesh reinforcement was not adopted in previously reported cases, without late recurrences , but datWhen prompt exploration is not carried out, the strangulated part of small bowel may become gangrenous much to make bowel resection necessary. In a series of 25 patients with internal hernia submitted to surgery, delayed laparotomy time (>3 days after the onset of symptoms) have increase significantly the number of bowel resections and their related morbidity . Small bPotential limitations of this study were the small number of patients and the relatively short follow-up period, in relation to the risk of ISH recurrences.In conclusion, we believe that strangulated ISH should be considered as possible cause of an unexplained small bowel obstruction, especially in adult male without previous surgery. CT scan in emergency setting may help the surgeon in choosing approach and timing of the procedure. The surgeon with experience of laparoscopic inguinal hernia repair, should consider in patients with stable hemodynamic parameters the operative mini-invasive technique as alternative to conventional laparotomy, in consideration to the advantages in terms of postoperative pain, hospital stay, cosmetic outcomes and laparotomy related morbidity, especially in obese  and younWritten informed consent was obtained from the patients for publication and any accompanying images.Not commissioned, externally peer reviewed.Not applicable.All the authors declare that they have no source of funding.Clementi M and Guadagni S contributed the original idea and steasure of the manuscript.Clementi M, Bonanni L and Di Furia M contributed by conceptualization and performing the surgical procedures.Sista F, Salvatorelli A, Puccica I and Vicentini V contributed by collecting all the data.Guadagni S. and Clementi M contributed by conceptualization and revision of the manuscript.The final manuscript has been read and approved by all named authors and that there are no other persons who satisfied the criteria for authorship but are not listed. We further confirm that the order of authors listed in the manuscript has been approved by all of us.All the authors declare that they have no conflict of interest.Researchregistry4265.Prof Marco Clementi, MD.There is no need for ethical approval because it is a case report. Written informed consent was obtained from the patients for publication of this cases series and any accompanying images.