Odontogenic Keratocyst (OKC) is a benign lesion recently considered an odontogenic cyst in the present World Health Organization (WHO) classification of Head and Neck tumors (2017) mainly based on its clinical and histopathological features. Several studies, systematic reviews and meta-analyses have shown a multimodal approach to reducing the high recurrence rates inherently exhibited by this pathological condition after long-term periods of follow-up.The patient, a 67-years-old woman, was submitted to conservative surgical management of an odontogenic keratocyst. Initially this consisted of decompression and subsequent enucleation, with a long-term period of follow-up to ensure that there was no recurrence.Conservative surgical management is the first choice and might be considered the gold standard, but combined therapy such as marsupialization, application of Carnoy’s solution, enucleation of the remnant lesion and an extensive follow-up period would considerably reduce recurrence rates.Therefore, an appropriate long-term follow-up must be done after the treatment performed in order to ensure clinical success, which in this case was described as an absence of signs of recurrent disease. In addition, the growth rate of this pathological condition is slow, and occurs in the anteroposterior direction, covering the medullary spaces of the bone. Consequently, in the majority of cases, in the initial stages, it is not possible to show it through the clinical finding of bone cortical swelling.In imaginological assessment, the OKC is characterized by the presence of a single or multillocular radiolucent area, with well-defined edges and regular margins. The larger lesions are often multillocular. In many cases, an unerupted tooth is associated with the cystic lesion , which mRelative to the pathological analysis, the OKC has a thin and friable capsule, with approximately eight to ten layers of epithelial cells. The intraluminal space visualized is composed of a cheesy material that shows evidence of the presence of keratin in its interior .In order to confirm the diagnosis of OKC, it is necessary to perform a complete intra and extra-oral clinical evaluation, thorough radiographic analysis and particularly the pathological examination to establish an accurate final diagnosis.Different treatment modalities have been described, ranging from conservative techniques to radical surgeries. Concerning the conservative approach, marsupialization and simple enucleation have shown high recurrence rates. For this reason, the combination of therapies has been selected in recent approaches reported in the literature, such as peripheral ostectomy performed with drill bits, cryotherapy and the use of chemical chelation by applying Carnoy’s Solution . On the The OKC has a relatively high recurrence rate, around 7–28 % during the first 5 years after treatment. This variation occurs due to some of the diagnostic criteria and as a consequence of the treatment, taking into consideration appropriate use of the different techniques described above, location and extent of the lesion . TherefoThe aim of this case report was to describe the results obtained with a conservative surgical approach in a patient diagnosed with odontogenic keratocyst and after a 5-year follow-up, in contrast with the results reported in the contemporary literature. This study was reported in line with the SCARE criteria .2The patient, a 67-years-old woman, who came to the first clinical consultation with a history of hypothyroidism and allergy to aspirin and penicillin, with painful symptoms in the submental and mental right side of mandibular bone region, with a duration of 3 months. The patient was attended by professionals of the Department of Dentistry, who proposed a procedure of surgical extraction of the mandibular teeth involved in the anterior section. Orthopantomography was performed, in which a large radiolucent area was observed in the chin region, so for this reason, she was referred to the Oral and Maxillofacial Surgery Department .Fig. 1OrAfter medical and dental details described above, an intraoral clinical assessment of the area was made, in which an increase in volume of the vestibular bone table//plate?/ was observed, tooth mobility between grade 1–2 found on inspection of 4.1–4.2. On the other hand, teeth 3.3 and 4.3 showed no pathological mobility. In addition, the surrounding mucosa was erythematous, especially in the region of teeth 3.1 and 3.3, in which the tissue was fluctuating due to possible perforation of the cortical vestibular bone as seen in Computed tomography (CT) assessment showed the presence of a delimited, unilocular, hypodense zone along its entire perimeter, limited by a thin bone cortex on the one hand, from the mesial-apical region of teeth 33–46, below the mandibular basilar edge. There was a short conservation of the basilar border, especially in the midline with greater resorption on both sides, so that greater involvement was shown in the upper part of the alveolar ridge, involving the teeth present, in the mesial and apical regions of tooth 33, and teeth 41, 42, 43.In the region itself of these teeth, an accentuated loss of bone support was found, at the level of the middle and apical third, only the cervical third of the roots was preserved. On the other hand, a smaller amount of resorption of the lingual bone and greater resorption of the vestibular cortical bone were observed throughout the extent of the lesion, measuring a thickness of only 1–1.5 mm, as shown in As the clinical and radiographic examination of the lesion was performed and having made an initial diagnosis compatible with odontogenic keratocyst, it was decided to perform a fine needle aspiration puncture of the content for subsequent pathological analysis. Thereby, a milky-yellow liquid content of approximately 10cc was obtained. In addition, the lesion showed a thick cystic capsule of intense red wine color, which showed some degree of resistance to surgical excision. Both surgical pieces were analyzed histopathologically confirming the initial diagnosis of moderate-grade unilocular Parakeratinized Odontogenic Keratocyst (former classification of odontogenic keratocyst that was reclassified as a keratocyst odontogenic tumor) as shown in After 30 days of initial decompression of the lesion, surgical resection of the lesion was performed under general anesthesia. A supracrestal incision was made in the mandibular residual edentulous alveolar ridge, covering the teeth present and extending bilaterally to the first molar regions. After this, the mucoperiosteal flap was lifted, exposing the vestibular cortical bone to obtain a complete and improved direct view of the lesion.Subsequently, a peripheral osteotomy was performed to enable direct visualization of the odontogenic keratocyst, using tungsten carbide surgical drills and a gouge clamp. In addition to the surgical procedure of excision of the lesion, enucleation and vigorous curettage were performed. The remaining teeth were removed and Carnoy’s solution was applied as an agent for chelating the remaining bone tissue, embedded in a gauze for 3 min. Finally, a reconstruction plate of the unlock 2.0 system was positioned to cover the middle portion of the mandibular body and extending from the region of the right first right molar to the left first molar. This was stabilized with 7 mm long mono-cortical screws to prevent a possible inter- or postsurgical mandibular fracture. The anatomical layers were repositioned using simple stitches with Vicryl 4.0 .Fig. 5SeFurthermore, post-surgical control was performed for the removal of the stitches, observing adequate healing of the postoperative wound, without signs of dehiscence and surgical bed infection. In addition, plans were made for follow-up consultations a month later, at three months, and at one year, with control panoramic radiography, in which satisfactory functional results were observed and without signs of recurrence, as shown in Clinical and radiographic control was performed after 6 months of follow-up, and a favorable healing process was observed without signs of recurrence of the lesion, as may be seen in After two years of follow-up; a satisfactory bone remodeling of the region that underwent surgery was found, showing bone characteristics similar to those of the surrounding normal bone tissue. Correct positioning of the osteosynthesis material was also verified, with the absence of resorption areas around it .Fig. 8FoIn an accurate CT assessment after the two year follow-up, isolated radiolucent areas, approximately 1 cm in diameter were identified in the region of teeth 4.1 and 4.2, which was compatible with recurrence of lesion, and therefore it was decided to undertake a second surgical procedure in order to obtain the diagnosis of recurrence. The osteosynthesis material was removed, as shown in Therefore, the histopathological assessment performed the second time confirmed a recurrence of the initial odontogenic keratocyst - parakeratotic subtype as shown in 3The spectrum of treatment modalities has been established in the literature since 2005 as conservative and non-conservative approaches to Odontogenic Keratocysts (OKC). Conservative methods described as simple enucleation, decompression or marsupialization and other techniques that are aggressive or non-conservative approaches such as cryosurgery or chemical destruction and radical surgery with bone resection ,11. HoweA recent systematic review and meta-analysis based on the rates of KOC recurrence in patients submitted to conservative surgical treatments showed considerable results, which may vary significantly depending on the type of approach applied. Moreover, in a high percentage of cases reviewed in this study, almost 19.8 % developed a local recurrence of the initial pathological condition. On the other hand, lower recurrence rates were found in cases in which more aggressive treatments were applied, such as bone resection (0 %), 7.8 % for cases treated by enucleation and Carnoy’s solution, and 11.5 % for cases treated by enucleation and liquid nitrogen . NeverthIn the present case report, decompression and enucleation alone were performed as the first-choice treatment, and the use of screw-retained plate to avoid pathological fracture in the residual bone. In the contemporary literature, it was recommended and established that more aggressive treatment have many disadvantages when compared with the clinical results of the conservative approach , because2. In a previous study, which evaluated the clinical results observed with the use of Carnoy’s solution, suture dehiscence and postoperative infection occurred, in addition to nerve injuries causing temporary toxicity paresthesia due to the undesirable effect caused by Carnoy’s solution applied directly on the inferior alveolar nerve, determined by exposure of the nerve in these reported cases [Carnoy’s solution, initially used as a slide fixator in laboratory pathology , is appled cases . Moreoveed cases ; howevered cases .There are others effects mentioned in the contemporary literature ,21 such Moreover, enucleation alone followed by peripheral ostectomy and application of Carnoy’s solution was chosen in order to promote the complete removal of cell elements associated with OKC, with the aim of minimizing the potential for recurrence and postoperative complications .Thus, clinical aspects, tumor location and particularly the tomographic examination of the lesion justified the approach adopted, since there was no bone fenestration and lesion communication with the surrounding soft tissues. The integrity of the surgical space was observed and the surgical planning ruled out the necessity for adjacent soft tissue excision. Moreover, the macroscopic aspects of the lesion with integrity of the fibrous capsule during curettage reaffirmed the choice of peripheral ostectomy, as was performed in the initial surgical management .Therefore, our case report supported the idea of conservative management of OKC, using combined therapy with an initial decompression and enucleation as described above. However, the recurrence rates continue to remain high, therefore extended long-term follow-up, with an established periodicity of months or years is necessary, in order to avoid further surgical procedures that diminish the quality of life, as well as the functional and esthetic changes that occur in patients have undergone this type of treatment.4Conservative surgical management of Odontogenic Keratocyst (OKC) with combined therapy using multimodal therapeutic approaches was shown as the first choice for treating this pathological condition, which showed a recurrence two years after the first surgical procedure. Therefore, an appropriate long-term follow-up must be done after the treatment performed in order to ensure clinical success described as an absence of signs of recurrent disease. We encourage further prospective studies to be performed in order to assess the multimodal approach and to elucidate the role played by each approach in reducing the disease recurrence rates.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.There are not conflicts of interest for this article to declare.National Council for Scientific and Technological Development of Brazil (CNPq) (870091/2001-8).This work was supported by the This report was exempt for ethical approval.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.All authors have contributed in the each step for writing this paper, participating in the whole process to retrieve medical information, review of the literature, and writing of each issue included.Wilber Edison Bernaola-Paredes has written and selected the topics for structuring this case report. Moreover, he did and worked in the Introduction and Discussion issues. On the other hand, Gisela Camolesi has done the literature review in order to support our discussion and introduction. Kleber Vallejo-Rosero did the case report (presentation and details about it) and took the all pictures during the surgery performed and follow-up in the clinical attendance. Kleber Vallejo was the first oral and maxillofacial surgeon who performed odontogenic keratocyst (OKC) surgery management, Wilber and Gisela participated as members of this team.It is a case report, observational, descriptive and retrospective study in which we have retrieved medical information from patient’s records.Wilber Edison Bernaola-Paredes/Kleber Vallejo-Rosero.


Paragangliomas, also known as chemodectomas, are rare tumors arise from chemoreceptor tissue, and most commonly locate at the bifurcation of the common carotid, the jugular foramen, aortic arch, and retroperitoneum. Paragangliomas generally are considered to be benign tumors, and rarely produce local or distant metastases. Metastasis to liver is extremely rare.We report the case of a 39-year-old woman, who had undergone resection of a retroperitoneal paraganglioma at her local hospital for 12 years. She was referred to our hospital for further evaluation of a hepatic mass, which was misdignosed as hepatocellular carcinoma (HCC) and was treated by transarterial chemoembolization (TACE) in the local hospital 6 years ago. At admission, CT scan revealed a huge hypervascular mass with many feeding arteries, almost the same size as 5 years ago. Ultrasound-guided biopsy of the liver tumor was performed and immunohistochemical examination confirmed the diagnosis of hepatic metastatic paraganglioma. Though liver metastasis failed to achieve complete response or partial response to TACE treatment, it remained stable without progression during the 7-year follow-up.Paragangliomas are slow growing tumors and metastasis may develop decades after resection of the primary lesion. Long-term follow-up is necessary, and curative or palliative treatment should be considered to control symptoms, improve life quality, reduce complications and prolong survival.The online version of this article (10.1186/s12876-019-1061-6) contains supplementary material, which is available to authorized users. Paragangliomas are rare lesions derived from the highly vascularized diffuse neuroendocrine system, with an incidence of 1:30000 . AdvanceBecause of difficulties in diagnosing malignancy, a considerable proportion of paragangliomas with metastatic potential is regarded as benign at initial presentation, but is identified as malignant during follow-up. It is generally recognised that intra-abdominal paragangliomas have a higher rate of metastases, and the most common distant metastases sites involve the lymph nodes, lungs and bones , 8. MetaA 39-year-old woman was referred to our hospital for further evaluation of a hepatic mass, which was discovered incidentally 6 years ago in the local hospital during physical examination for anemia and fatigue. At that time, the tumor was about 16 × 14.5 × 14 cm in size and was diagnosed as hepatocellular carcinoma (HCC). TACE was performed twice by injecting 20 mL iodized oil with 5-fluorouracil (500 mg), epirubicin (30 mg), and hydroxycamptothecin (10 mg) through the hepatic artery, followed by injection of gelatin sponge particles. Post-TACE CT scan revealed densely deposited lipiodol inside the tumor


The choledochal cyst is a rare congenital malformation of the bile ducts. It is considered as a precancerous state. The incidence of cancer in choledochal cyst increases with age and occurs around 32 years old. Therefore, young adults are often involved. In adults, clinical signs are rare and unspecific. We are reporting here the case of a cholangiocarcinoma in choledochal cyst in young adult diagnosed by ultrasonography, CT scan, and MRI and confirmed by histopathological examination. The cystic dilatation of the common bile duct is a rare congenital malformation of the bile ducts . It is cWe are reporting here a case of a suspected cholangiocarcinoma in choledochal cyst by medical imaging and confirmed by anatomopathological examination.In January 2012, a 38-year-old Caucasian woman went to the Emergency Room of the Dunkerque Hospital Center (France), for epigastric pain, no fever, along with jaundice which had been evolving for about a week. Her antecedents were appendectomy, tonsillectomy, and scoliosis with Carrington's stems. The clinical examination noted a swelling of the epigastrium and the right hypochondrium and, above all, a palpable mass was localized at the right hypochondrium. Biochemical analysis showed a liver cytolysis , a cholestasis , and an increase in gamma glutamyl transpeptidase: 467IU/L. Lipase and pancreatic amylase were normal.An abdominal ultrasound was performed and revealed a cystic mass measuring 15 cm x 10 cm, independent of the gallbladder, located between the portal vein confluence and the duodenum. Intracystic gallstone and an intraluminal tissue bud were present at the cystic wall. There was no color flow on Doppler exam in the mass. In addition, there was intrahepatic bile duct dilation, particularly noticeable on the left . An abdoHistopathological examination of the intracystic tissue bud and of the liver's nodules of segment VI showed an appearance of intraductal and infiltrating adenocarcinoma . During The choledochal cyst is a rare disease in Europe and Africa but more common in Asia.33 to 50% of the cases in the world are reported in Japanese literature . It is tCholedochal cyst is a precancerous condition with cancer occurring more often and earlier for these patients , 3. The The histological types of cancer observed in patients with choledochal cyst are as follows: adenocarcinoma (73% to 84% of cases), anaplastic carcinoma (10% of cases), undifferentiated carcinoma (5% to 7% of cases), squamous cell carcinoma (5% of cases), and other cancers (1.5% of cases) , 4. The The classical triad of vague intermittent upper abdominal pain, intermittent jaundice, and right upper abdominal mass is rarely seen . JaundicThe first scanning method of the biliary tree is an abdominal ultrasound. It is a noninvasive examination accessible quickly and inexpensively . The comThis case of cholangiocarcinoma in choledochal cyst showed the importance and necessity of combining several imaging modalities  to reach diagnosis. Early abdominal ultrasonography is recommended in case of intermittent abdominal pain and cholestasis in young adults, in order not to miss diagnosis of choledochal cyst.


Intestinal ganglioneuromatosis (IGNM) is a rare disease, defined by an abnormal proliferation of ganglion cells, nerve fibers and Schwann cells in the enteric nerve system.A 54-year-old woman presented with a one-year history of recurrent episodes of hypogastric pain, with vomiting, nausea, melena, and weight loss of 10 kg in recent 5 months.The patient was diagnosed as a diffuse IGNM by pathological examination.A complete excision of the tumor was performed.On follow-up after 26 months, the patient was asymptomatic without complications.This report showed a rare case of diffuse IGNM not associated with NF1 or MEN2b. Preoperative radiological examination suggested an intestinal GIST, yet the final diagnosis of diffuse IGNM was made according to the pathological examination of the resected specimen. Although the prevalence of ganglioneuromatosis is low, this condition should be considered in the differential diagnosis of intestinal mass in adults. Single polypoid ganglioneuromatosis are not typically associated with NF1 or MEN2b. However, ganglioneuromatous polyposis and diffuse ganglioneuromatosis often occur in association with NF1, MEN2b, juvenile polyposis, Cowden disease, and non-familial adenomatous polyposis. Its occurrence in the absence of the above-mentioned syndromes is extremely rare.Intestinal ganglioneuromatosis (IGNM) is a rare benign tumor characterized by proliferation of ganglion cells, nerve fibers and supporting cells of enteric nervous system. This paper reports a transmural diffuse ileal ganglioneuromatosis in an adult, unassociated with any manifestation of MEN2b, NF1, etc. Preoperative diagnosis suggested a gastrointestinal stromal tumor, yet the final diagnosis defined a diffuse ganglioneuromatosis depended on the pathological examination of the resected specimen.Diffuse ganglioneuromatosis has 2 variants: mucosal or transmural. In children, both mucosal and transmural variants can be seen, but in adults the most frequently variant is the mucosa.2A 54-year-old female presented in our department in July 2016 with a one-year history of recurrent episodes of hypogastric pain. She stated that the worsening abdominal pain associated with vomiting, nausea, melena, and weight loss of 10 kg in recent 5 months. She was previously healthy and had no special medical history. Regarding her family history, there were no other cases of gastrointestinal tumors, inflammatory bowel diseases. No physical and laboratory abnormalities were found. Upper gastrointestinal endoscopy and colonoscopy were all normal. An abdominal enhanced computed tomography (CT) showed a 2.4 × 2.3 cm soft tissue mass shadow with mild delayed enhancement extended into the distal ileal lumen of the right pelvic  is a rare disease, defined by an abnormal proliferation of ganglion cells, nerve fibers, and Schwann cells in the enteric nerve system. This disease can be found in any part of the gastrointestinal tract. IGNM is equally distributed between males and females and is more frequently seen in children. classified IGNM into 3 types: polypoid ganglioneuroma, ganglioneuromatous polyposis, and diffuse ganglioneuromatosis. The first type, polypoid ganglioneuroma occurs as solitary or a few small polyps presenting with sessile or pedunculated morphology. The lesions are mucosal or submucosal and are mostly seen in the large intestine. Patients with polypoid ganglioneuroma are not associated with NF1, MEN2b or other genetic syndromes. The second type, ganglioneuromatous polyposis is characterized by numerous (more than 20) mucosal and/or submucosal polyps with a sessile or pedunculated form, and is predominately located in the colon and terminal ileum. Ganglioneuromatous polyposis can be isolated or associated with one of the following syndromes: MEN2b, NF1, Cowden disease, non-familial colic polyposis or juvenile polyposis. Gastrointestinal ganglioneuromatosis can be the first presentation of these syndromes. The third type, diffuse ganglioneuromatosis is disseminated intramural or transmural lesions characterized by hyperplasia of the myenteric plexus. The most frequently involved location is the terminal ileum, colon and appendix. It can be solitary or associated with MEN2b, NF1, juvenile polyposis, tuberous sclerosis, Cowden disease, etc. However, the patient in our case had an isolated IGNM without any systemic syndromes or familial diseases. Diffuse ganglioneuromatosis has 2 froms, mucosal or transmural. In children, both variants can be found and are highly associated with RET-proto-oncogene abnormalities, while in adults the disease mostly involves the mucosa, and genetic abnormalities is not mandatory. The case reported in this paper is transmural.In 1994, Shekita and Sobin In our case, abdominal enhanced CT revealed a soft tissue mass in the enteric cavity, leading to luminal narrowing. An intestinal stromal tumor was suspected based on CT image and clinical manifestation. However, pathological examination defined a diffuse IGNM. Overall, it is difficult to differentiate GIST and IGNM before operation. First, Diffuse ileal ganglioneuromatosis (IGNM) and gastrointestinal stromal tumor (GIST) shared similar clinical presentations, such as abdominal pain, bleeding, constipation or diarrhea, intestinal obstruction, and changes in bowel habits. Second, the imaging features in IGNM and GIST are similar, which manifested as submucosal soft tissue masses in CT image. Hence, the final diagnosis depended only on the pathological examination. Generally, conservative treatment is invalid for diffused IGNM, and complete resection is needed in symptomatic cases. Because this disease is highly associated with NF1 or MEN2b, screening for associated clinical features such as external stigmata, skin nodule, multiple café-au-lait spots, and tumors at other sites such as breast, uterus, thyroid and colon, are recommended.Due to the unspecific clinical presentations and imaging manifestations, the defined diagnosis of IGNM is very difficult. Some differential diagnoses that can involve the gastrointestinal tract were proposed, including intestinal tuberculosis, Crohn disease, intestinal tumors , cytomegaly virus infection, NSAIDs enteropathy and amyloidosis.4This report showed a rare case of diffuse IGNM not associated with NF1 or MEN2b. Preoperative radiological examination suggested an intestinal GIST, yet the final diagnosis of diffuse IGNM was made according to the pathological examination of the resected specimen. Although the prevalence of ganglioneuromatosis is low, this condition should be considered in the differential diagnosis of intestinal mass in adults.The authors thank the patient who kindly agreed to provide her with the data we used in this case. The authors gratefully acknowledge the doctors working together for their support.Conceptualization: Zhou Zhao, Huijiao Chen, Bo Zhang.Data curation: Chaoyong Shen, Yuan Yin.Formal analysis: Xiaonan Yin, Xin Chen.Funding acquisition: Bo Zhang.Investigation: Renjia Shu, Jian Wang.Methodology: Chaoyong Shen, Yuan Yin.Resources: Zhaolun Cai.Software: Zhaolun Cai.Supervision: Renjia Shu.Visualization: Jian Wang, Zhou Zhao.Writing – original draft: Xiaonan Yin, Xin Chen.Writing – review & editing: Huijiao Chen, Bo Zhang.Bo Zhang orcid: 0000-0002-0254-5843.


Adult diaphragmatic hernia is a rare case and usually present with various clinical manifestations. Surgery can be done by thoracic and abdominal approaches. In emergency cases, mostly performed by abdominal approach. The use of mesh to close the defect is currently emerging as a viable option.We report a case of 70-year-old woman with complaints of shortness of breath and abdominal pain. The patient had a history of laparoscopic surgery with the installation of an anti-adhesive mesh for diaphragmatic hernia seven months before admission. Chest X-ray and Computed Tomography (CT) scan found recurrence of diaphragmatic hernia. The patient then received laparotomy showing diaphragmatic hernia with contents in the form of transverse colon, ileum, gastric and omentum. The self-attached mesh was placed on the supra-diaphragm with abdominal approach. The patient was hospitalised four days after surgery. At two weeks and six months follow-up, no complaints were found. CT scan and chest X-ray were within normal limits.We did a modified technique in managing the recurrence and adhesion of the diaphragmatic hernia using the hernia mesh attached onto the superior side of the diaphragm to minimise the complications and recurrences.Transabdominal supradiaphragmatic self-attached mesh for recurrent diaphragmatic hernia can be considered to be an alternative treatment for recurrent diaphragmatic hernia in an emergency setting because this procedure is relatively easier, safer, causes fewer postoperative complications, and can prevent recurrence. Adult diaphragmatic hernias are a rare condition that occurs mainly due to a direct penetrating injury to the diaphragm (3%–15%) or, less commonly, secondary to blunt abdominal trauma (5–7%) ,10.In adults, diaphragmatic hernia is more frequent in males with proportion of 3:1. Most diaphragmatic hernias occur on the left (85%) and without hernia sac (85%). Size of the hernia does not always represent the size of the diaphragmatic defect .There are four types of diaphragmatic hernia . Type I The thoracic and abdominal approaches are often used as surgery management of diaphragmatic hernia. Currently, there is no guideline for the absolute indication in the timing of the surgery. The use of mesh in the management of diaphragmatic hernia is emerging as a viable option; several studies have shown low recurrence in mesh use. However, there is still a lack of long-term research to prove the safety and effectiveness . This re2A 70-year-old woman presented with complaints of shortness of breath, abdominal pain and fullness. The patient had a history of laparoscopic diaphragmatic hernia surgery with the installation of an anti-adhesive mesh seven months ago. From chest X-ray and Computed Tomography (CT) scan, recurrence of diaphragmatic hernia was found. CT scan in 1We performed surgical procedure under general anaesthesia. 2We made an upper midline incision. 3Next, we did a general exploration of the abdomen whilepaying attention to the diaphragmatic hernia in the area of the esophagogastric junction.4We mobilised the left hemi liver by dividing the triangular ligament to expose the diaphragmatic hernia. 5After that, we performed adhesiolysis and then divided and exposed the medial and lateral crus of the diaphragm.6Then, we released and downward-mobilised the gastric fundus, transverse colon, and ileum which enter the diaphragmatic hernia.7Afterwards, a self-attached mesh sized 14 × 9 cm was inserted into the superior of the diaphragm via diaphragmatic hernia with self-attached surface facing the abdomen without fixation suture.8Subsequently, we repaired the hernia with simple sutures to approximate the lateral and medial crus. The evaluation was done by using the index finger to feel whether it cannot enter the diaphragmatic hernia, and make sure it was not too tight.Surgical procedures:3The incidence of non-traumatic diaphragmatic hernia in adults is rare with various clinical manifestations. Most cases can be associated with penetrating diaphragm trauma or blunt abdominal trauma ,7,10.In this case, the type of diaphragmatic hernia that occurred was type IV, with organ herniation containing transverse colon, ileum, gastric, and omentum. Management of a diaphragmatic hernia should be done by restoring the abdominal organ to its position and repairing the defect. The surgical approach for this pathology depends on the presence of visceral complications. There are two types of surgical approaches, thoracic and abdominal approaches. In an elective setting most authors recommend the thoracic approach, while, when there are septic complications, the abdominal approach is preferred. The current trend is to use minimally invasive surgical techniques such as laparoscopy, and especially thoracoscopy, which has been satisfactorily performed in adults. In the emergency setting, there is no consensus nor an absolute indication of the best timing of the surgery ,8,13. InThe use of surgical mesh in the management of a diaphragmatic hernia has developed and become a standard in recent years. Several types of mesh were developed. In general, diaphragmatic hernia repair uses anti-adhesive mesh. It is a second-generation multifilament mesh that is partially absorbable in approximately 20 days and has the benefit of anti-adhesion properties. There is one side of the mesh that prevents adhesion and the other side which is directed toward the abdominal wall or diaphragm to allow mesh ingrowth ,9,13. HoThe purpose of this report is to present our experience in managing recurrence and adhesion of the diaphragmatic hernia with some technical modifications to minimise the complications and recurrences. We attached the hernia mesh onto the superior side of the diaphragm to reduce the risk of adhesion with the abdominal visceral organs. Postoperatively, the patient was hospitalised for four days. We followed up for six months, and no complaints were found. We also performed CT scan and chest X-ray, and the results were normal. However, further studies are still needed with a larger sample, and a more extended follow-up period.4The abdominal approach using supra-diaphragmatic self-attached mesh is a viable option in recurrent diaphragmatic hernia therapy. The six-month follow-up showed a safe procedure with no symptoms of recurrence and adhesion. In the future, we need to do a larger scale of research so that this approach can become a full technical report.The authors declare that this study had no funding resource.The informed consent form was declared that patient data or samples will be used for educational or research purposes. Our institutional review board also do not provide an ethical approval in the form of case report.We have obtained all patient’s consent and had the statement included in the consent section in the manuscript. We also do not include any of the patients name or the institution.Adeodatus Yuda Handaya conceived the study. Aditya Rifqi Fauzi and Victor Agastya Pramudya Werdana drafted the manuscript and critically revised the manuscript for important intellectual content. Adeodatus Yuda Handaya, Aditya Rifqi Fauzi, and Victor Agastya Pramudya Werdana facilitated all project-related tasks.There is in literature already published data stating Lapatoromy from human. This is not the first one.Adeodatus Yuda Handaya.Not commissioned, externally peer-reviewedAdeodatus Yuda Handaya: Conceptualization, Methodology, Resources, Writing - original draft. Aditya Rifqi Fauzi: Writing - review & editing, Resources, Validation. Victor Agastya Pramudya Werdana: .No potential conflict of interest relevant to this article was reported.


Rhabdomyosarcomas are neoplasms with a high degree of malignancy and arise from the embryonic mesenchyme. They represent approximately 5% of all pediatric tumors and their main locations are the head and neck (45%), trunk (40%), and extremities (15%). Twenty-five percent to 30% of the head and neck rhabdomyosarcomas appear in the orbit; however, its origin from the conjunctiva is rare, with few case reports published in the literature.We present the case of a five-year-old girl with a diagnosis of primary embryonic rhabdomyosarcoma of the conjunctiva, treated with surgery and chemotherapy. After completing the treatment, it was followed up with controls for oncological ophthalmology, pediatric hematology-oncology, and radiotherapy oncology every six months with magnetic resonance of the orbits. Two years after the end of treatment, the patient is disease-free.Conjunctiva rhabdomyosarcoma is a rare lesion, with few previously reported cases. In the reported case, the histopathological findings and positivity of the different immunohistochemical markers allowed a definitive diagnosis of rhabdomyosarcoma. The excellent prognosis of this pathology is probably linked to the early diagnosis of the disease and the timely administration of radical treatment.It is essential to be able to identify conjunctival rhabdomyosarcoma from its clinical and histopathological characteristics in order to achieve early diagnosis and provide adequate treatment to patients. Rhabdomyosarcomas are neoplasms with a high degree of malignancy, which arise from the embryonic mesenchyme and have the potential to differentiate into striated muscle. They can appear almost anywhere on the body, including the orbit and eyeball, corresponding to 9% of all these tumors . About 7Although they are the most frequent primary tumors of the orbit in pediatric patients and the botryoid variant can compromise the conjunctiva in 5% to 8% of cases, primary conjunctival rhabdomyosarcomas are very rare, with few cases reported in the literature -4. In ShAs for their clinical manifestations, these tumors may present as a conjunctival mass or as a hyperemic area . Some ofGenerally, rhabdomyosarcomas are classified into four morphological categories: embryonic, alveolar, botryoid, and pleomorphic. The botryoid subtype is also considered as a variant of the embryonic type, which is characterized by a papillary configuration ,7.During the twentieth century, the only treatment was radical surgery; however, due to the development of new treatment strategies, such as chemotherapy and radiotherapy, supported by the creation of groups such as the International Rhabdomyosarcoma Study Group (IRSG), overall survival has increased from 15% or 25% to more than 70% in recent decades . As for Nowadays, most protocols for the management of rhabdomyosarcomas are based on risk stratification, which is determined by multiple staging systems, histology, and tumor location . Among tA five-year-old patient, with a clinical picture of 4 months of evolution, consisting of the appearance of the right conjunctival mass of progressive growth. Initially, she was evaluated extra-institutionally, where she had a partial mass resection. On that occasion, the pathology reported an embryonic rhabdomyosarcoma, the reason why she was referred to our institution. The patient was then evaluated by the oncology ophthalmology service, which, on ophthalmological examination, observed a mass that occupied the entire extent of the lower conjunctiva, causing palpebral ectropion of friable appearance, with soft and papillomatous edges Figure . She wasSubsequently, it is staged as a botryoid variant embryonal rhabdomyosarcoma of the right conjunctiva (favorable location), low-risk subgroup Ia, T1 (<5 cm), N0 (no ganglionic involvement), M0 (no metastasis), post-surgical clinical group (IRS), IIIa . The patient received chemotherapy for low-risk sub-group Ia rhabdomyosarcoma. Treatment was provided with eight cycles of VAC regimen chemotherapy . At the end of this treatment, a contrasted magnetic resonance of orbits was requested, where no residual mass was evidenced. Taking into account that the patient had not been taken to an oncological resection, but, instead, the surgical intervention performed was an excisional biopsy, the multidisciplinary group of treating physicians proposed to provide treatment with external radiotherapy after chemotherapy. The case was presented at a radiation oncology meeting and, taking into account the possible side effects of radiotherapy, it was decided to provide expectant treatment, and so the patient was left on follow-up with imaging controls every six months.Three years after the end of treatment, the patient remains clinically and imaging free of disease, with an adequate visual acuity and without symptoms suggestive of toxicity secondary to treatment.Rhabdomyosarcoma is the most common neoplasm of the orbit within the pediatric population, representing approximately 5% of all pediatric tumors . The maiIn this case, the clinical presentation of the tumor was the usual one, in accordance with what has been reported by other authors . DespiteThe tumor was staged as an Ia group according to the IRSG, and the current recommendation for this group of tumors is to perform a surgical resection and subsequently provide adjuvant treatment with VAC regimen chemotherapy. However, it was contemplated to provide treatment with radiotherapy since the patient was not taken to oncological resection but to excisional biopsy. Taking into account the limited available literature on conjunctiva rhabdomyosarcoma and its treatment and that radiotherapy could lead to late toxicity that is not negligible, it was decided to leave the patient under observation. Also, the results of the clinical trial of the International Society of Pediatric Oncology Malignant Mesenchymal Tumor (MMT-89) suggested that the omission of radiotherapy was justifiable for some patients if they were treated according to the guidelines of the IRSG . SimilarOur case emphasizes the importance of a careful histopathological review of all conjunctival masses in children in order to achieve timely diagnosis and treatment.So far, the patient is free from relapse. The favorable prognosis may be related to tumor location, early detection, and embryonic histological type. Taking into account the excellent prognosis, the goals of treatment are, in addition to curing the disease, minimizing late sequelae secondary to treatment.It is necessary to know that in the pediatric population, rhabdomyosarcoma can originate from the conjunctiva. Knowledge of its clinical, histopathological, and imaging characteristics is essential in order to achieve early diagnosis. It is essential to propose a multidisciplinary treatment where the risks and benefits of the different therapeutic tools are always evaluated.


A 22-year-old female presented with a large abdominal mass that was revealed to be a primary retroperitoneal mucinous cystadenoma.A 22-year-old female presented with a two day history of bloating, mid-epigastric pain, and nausea without vomiting. A CT scan of her abdomen/pelvis showed a large left retroperitoneal mass, possibly a mesenteric cyst. The patient underwent laparoscopic surgery for mass excision. Once the cystic mass was completely dissected laparoscopically, it was placed in a large endobag. The fluid was then aspirated while in the bag to decompress and then completely taken out through a port site. She was discharged the following day. Final pathology revealed a benign mucinous cystadenoma/cystadenofibroma of mesenteric origin.Primary retroperitoneal mucinous cysts are rare occurrences and benign mucinous cystadenomas are the rarest subtype. We use an innovative laparoscopic technique with complete excision of the cyst without spillage of content, thus preventing possible seeding in case of malignancy.There is some previous caution in using a laparoscopic approach for cystic masses due to potential seeding intra-operatively, in case of fluid spillage of a possible malignant neoplasm. We show through our case that it is possible to efficiently and safely use such an approach. Most neo2The patient was a 22-year-old female who presented to the emergency department with a two day history of bloating, mid-epigastric pain, and nausea without vomiting. The pain was mild, non-radiating, and unrelated to food intake. She had a past surgical history of ventral hernia repair 6 months prior and laparoscopic cholecystectomy 1 year prior. A CT scan of her abdomen and pelvis showed a large cyst partially abutting the inferior pole of the left kidney that was not clearly renal in origin . The cysThe mass was diagnosed as non-emergent although the patient elected to have surgical management scheduled for the next day due to her increasing level of pain. The surgical plan was for a laparoscopic excision of mass, possible open, with possible bowel resection. In the operating room, after access to the peritoneal cavity was obtained using an optical bladeless trocar with added ports, lysis of multiple adhesions from previous cholecystectomy and hernia repair was safely done. The greater omentum was transposed superiorly over the stomach and the small bowel was reflected to the right. The left retroperitoneum was widely exposed. The mass was then observed to be retroperitoneal in nature, abutting the left colon. Excision of the mass was done meticulously using Harmonic scalpel and blunt grasper dissection. Bleeding was negligible. The left colon abutting the mass was evaluated thereafter and noted without ischemic changes. The mass was caught with a 15 mm endobag, needle decompression of cyst fluid was done while in the bag, and the bag was delivered out through a 15 mm trocar site . A samplCEA, CA 125 and tissue pathology was ordered. The CEA and CA-125 values were 1.0 ng/ml and 10.2 units/mL. The final pathology report revealed benign mucinous epithelium without significant cytologic or architectural atypia in a background of fibrous tissue, consistent with a benign mucinous cystic neoplasm of mesenteric origin .Fig. 5(lThe patient was transferred to the surgical floor postoperatively, remained uneventful and was discharged the following day.3Mucinous cystadenomas are located in a variety of anatomical regions such as the ovaries, the pancreas, and in the retroperitoneum. Primary retroperitoneal mucinous cysts, as seen in this patient, are rare occurrences. Similar to previous cases, due to the cystadenoma location in the retroperitoneum and the appearance of normal ovaries, it is considered to be a retroperitoneal primary. The origin of these extraovarian mucinous cystadenomas is not clear. One theory claims that the extraovarian cystadenomas are a result of ectopic ovarian tissue or the overgrowth of mucinous epithelium from a teratoma. The theory with major acceptance is that of mucinous metaplasia to an invagination of the peritoneal mesothelial layer resulting in cyst formation. This theory has better recognition versus the ectopic ovarian tissue theory due to the occurrence in males as well. There are three subtypes to these primary retroperitoneal cysts: mucinous cystadenoma, mucinous cystic tumor of borderline malignancy, and mucinous cystadenocarcinoma. Case reports of mucinous cystadenoma with an origin from the mesentery are very rare with 19 case reports, 2 in males.  Of the 1One report of two cases uses a laparoscopic surgical approach for simple lymphatic cysts of the mesentery.  Other pr4Primary retroperitoneal mucinous cystic neoplasms rarely occur. We report an innovative laparoscopic removal of a mesenteric mucinous cystic neoplasm with steps to prevent spread if the neoplasm was malignant. A laparoscopic approach that prevents possible seeding should be considered for removal of primary retroperitoneal mucinous cystic neoplasm in the future.This case report is reported in line with the SCARE criteria .The authors declare that they have no competing interests. We have no personal or financial conflicts of interest related to the preparation and publication of this manuscript.This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.No ethical approval is required by our institution for the case study presented and 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.John Lung BS: Data collection, review of literature, co-author of entire manuscript, approval of final manuscriptAllison Gracey BS, BA: Data collection, co-author of case description, approval of final manuscriptAbigail Rosales MBA: Data collection, co-author of discussion, approval of final manuscriptEva Bashover MD: Conception and design, co-author of case description, critical review of the article, approval of final manuscriptAlan Sbar MD, FACS: Conception and design, critical review of the article, approval of final manuscriptM. Haris Nazim MD, FACS: Conception and design, critical review of the article, approval of final manuscriptFerdinand Rico MD, FACS: Conception and design, supervisor, co-author of entire manuscript, approval of final manuscriptThis is a case report and is not a first-in-man study, therefore registration is not required.Ferdinand Rico MD, FACSNot commissioned, externally peer-reviewed.


En bloc resection is required, role of lymphadenectomy is not clear.•In setting when patient is stable, diagnostic evaluation is performed first, to access size, location of tumour and plan best surgical approach.•Treatment of gastrointestinal stromal tumours should be multidisciplinary. Duodenal gastrointestinal stromal tumors (DGISTs) are mesenchymal tumors. Main presenting symptoms are gastrointestinal bleeding and non-specific abdominal pain. Surgical treatment is golden standard for treatment of DGISTs. Tumour location and size determine the surgical strategy. Recurrence rate depends on tumour location and completeness on removal.75 years old male fell ill with hematemesis, hemohesia and dizziness. Oesophagogastroduodenoscopy (EGD) did not reveal the source of bleeding. Upon admission, a second EGD was performed due to loss of consciousness and anaemia, which revealed only approximate source of bleeding in the third portion of the duodenum. The exact bleeding spot could not be reached and bleeding could not be controlled endoscopically. Patient was transferred to the operating room for an emergency explorative laparotomy. After extensive mobilisation of the duodenum a 2 × 2 cm intraluminal tumour with central bleeding ulceration was found in D3. The diseased part of the duodenum was excised and the duodenotomy was closed with interrupted sutures. Final pathohistological report confirmed gastrointestinal stromal tumour, pT1. Postoperative period was uneventful.Gastrointestinal stromal tumours are most common in males aged 60–65 years. Surgical treatment is a golden standard. Surgical approach depends upon tumor location and size. In case of major bleeding surgical procedure is lifesaving and diagnostics for staging purposes are omitted.Gastrointestinal stromal tumors are a potential source of life-threatening bleeding. In such cases an emergency operation is often indicated with aim to remove the tumour en bloc and achieve hemostasis as soon as possible. They can originate from the entire gastrointestinal tract, most commonly from stomach (60%), small intestine 20–30%) and duodenum (5%). About 10–30% of all neoplasms of duodenum are duodenal GISTs (DGISTs) [0–30% andThe following paper presents a case of DGIST from teaching university hospital.This work has been reported in line with the SCARE criteria .275 years old Caucasian male, smoker, with history of excessive drinking, was seen at Internal medicine Emergency department due to hematemesis, hemohesia and dizziness. Few days before he felt weak. He had history of gastritis, colon polyps resection, arterial hypertension, chronic obstructive pulmonary disease and depression. Family history was positive for breast and skin cancer. Upon admission on emergency department blood pressure was stable, he was tachycardic, skin was pale. There was slight pain in epigastrium, without signs of peritoneal irritation. Digital rectal examination was within normal.Due to GIB and anemia, he was admitted to the Department of gastroenterology. On the admission day hemoglobin (Hb) level was 71 g/dL and an emergency oesophagogastroduodenoscopy (EGD) was performed which showed some hematous content in the stomach without clear source of active bleeding being found. He received transfusion of 3 units of red blood cells (RBC). Patients’ condition was worsening, he lost consciousness hence the EGD was repeated. In the third portion of duodenum a submucous, actively bleeding tumor was found. Endoscopic hemostasis was unsuccessful and patient was in haemorrhagic shock. He was transfered in the operating room for an emergency explorative laparotomy. Procedure was performed by two experienced abdominal surgeons . After extensive mobilization of the duodenum a 2 × 2 cm intraluminal tumor with central bleeding was found in the third portion of the duodenum . The dis3GISTs present approximately 1% of the gastrointestinal tumors . EstimatGISTs are most common mesenchymal neoplasms and typically are subepithelial ,2. Most tumor related , host related  and environment related  (8). DGIST recurrence depends more on tumor biology than surgical approach/type of surgical procedure or microscopic margins [Leading symptoms are nonspecific abdominal pain, GIB , and symptomatic anemia ,7. Clinielated NF, age andTreatment should be multidisciplinary involving medical oncologist, gastroenterologist, surgeon, pathologist, radiologist, nuclear medicine specialist etc. Such patients should be treated in a reference centre . Our patSurgical treatment is a golden standard, although surgical approach varies depending on tumor size, location and invasion into adjacent organs (stage of disease) 1, 8). Preoperative radiological staging (CT/MRI) is of crucial importance for surgical procedure planning [. PreoperStandard treatment of localised DGIST is complete surgical excision (en bloc) with negative surgical margins, without lymphadenectomy of clinically negative lymph nodes . In caseIn cases of minimally invasive approach (laparoscopic or robotic), principles of oncologic surgery should be implemented ,8. Radio4DGIST may be source of life threatening bleeding. In such cases surgical treatment has priority over diagnostics. Aim of surgical treatment is to remove tumor en bloc, following the principles of oncologic surgery. Surgical approach depends upon tumor location and size. Recurrence rate depends upon tumor location and extend of removal. Because of the rarity, role of lymphadenectomy and most appropriate surgical approach are unclear.All authors have no conflict of interest to disclose.Authors did not receive any form of funding.Patient was treated according to current guidelines. Ethical approval is not required.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.Milena Taskovska - preparation of manuscript, literature reviewMirko Omejc - surgical procedure, review of the manuscriptJan Grosek - surgical procedure, literature revire, review of the manuscriptNot applicable.All authors accept full responsibility for case report presented.Not commissioned, externally peer-reviewed.


Primary carcinosarcoma is a rare neoplasm of the gallbladder, and is characterized by collision elements of the adenocarcinomatous and sarcomatous components. The tumours comprise both malignant epithelial and mesenchymal elements, and are usually very aggressive, conferring poor prognosis. Adenocarcinomas are the most common neoplasms of the gall bladder, while carcinosarcoma are extremely rare, with a global prevalence rate of less than 1%. The low incidence of carcinosarcoma greatly hinders the understanding of its aetiology.Here, we report the surgical outcome of 3 rare cases of gallbladder carcinosarcomas, two of which had a IVB stage disease and in one case the staging was II. Following surgery, the survival in the former was less than 1 year, while in the latter, it extended up to 7 years (86 months), and the patient is alive till date. This study aimed to elucidate the surgical outcomes and prognosis after curable resection for carcinosarcomas of the gallbladder.The findings suggest that carcinosarcomas are associated with a poor prognosis and short survival, particularly in those with tumours of >5 cm. In the absence of effective adjuvant treatment, surgery remains the mainstay of treatment.Complete surgical resection may improve prognosis and extend survival in patients with this rare and aggressive form of gallbladder cancer. Gallbla22.19/L, haematocrit: 28.2%, and platelet count: 672 × 109/L. The C-reactive protein level was 174.1 mg/L. Liver function tests demonstrated the following: albumin, 23 g/L, total protein: 65 g/L, alanine aminotransferase: 21 units/L, aspartate transaminase: 27 units/L, alkaline phosphatase: 52 units/L, total bilirubin: 11.32 umol/L, conjugated bilirubin: 6.28 umol/L, and amylase: 22 units/L. The coagulation profile was within the normal range. Cancer antigen markers including carcinoembryonic antigen (CEA), carbohydrate antigen 19.9 (CA19.9), and α-fetoprotein (AFP) were all within the normal range. Initial ultrasound of the abdomen revealed a large mass encasing the gallbladder, with multiple small gallstones. Computed tomography (CT) of the chest, abdomen, and pelvis revealed a large polypoidal enhancing mass in the gallbladder measuring 11.5 × 9.2 × 5 cm, and focal lesions in segment V of the liver with extension to the right colon; peritoneal deposits were also observed   based on the 7th edition of the American Joint Committee on cancer (AJCC) staging system. He was then referred to the medical oncology department, where he received 2 cycles of adjuvant chemotherapy. Hepatic recurrence was observed with multiple variably sized nodules in the omentum 3 months after surgery, and he died 3 months later.2.29/L, haematocrit: 32.5%, and platelet count: 423 × 109/L. Liver function tests showed the following: albumin: 34 g/L, total protein: 66 g/L, alanine aminotransferase: 55 units/L, aspartate transaminase: 52 units/L, alkaline phosphatase: 115 units/L, total bilirubin: 6.9 umol/L, conjugated bilirubin: 23.3 umol/L, and amylase: 39 units/L. The coagulation profile was within the normal range, and the cancer antigen markers revealed a CA19.9 level of 154.3 IU/mL, with normal levels of AFP and CEA. Initial ultrasound of the abdomen, performed in the referring hospital demonstrated thickening of the gallbladder wall, which measured approximately 17 mm, with large irregular soft tissue masses arising from the fundus, that measured 8 × 6 cm. These were seen infiltrating the surrounding sub-hepatic fat planes, and were inseparable from the transverse colon; no dilated intrahepatic biliary radicals, focal hepatic lesions, or enlarged lymph nodes were noted. CT of the chest, abdomen, and pelvis revealed distension of the gallbladder fundus with an intraluminal tumour and huge exophytic mass measuring approximately 13.6 × 12 × 9.5 cm; medially, the lesion displaced the distal stomach at the level of pylorus, and the mass protruding downwards displaced the transverse colon inferiorly. Few small portal lymph nodes were also seen  in the past 5 days prior to presentation. The patient denied any history of anorexia, weight loss, jaundice, or gallstones. On general physical examination, the patient was well-nourished, in moderate pain, and had no pallor or jaundice. Abdominal examination revealed minimal tenderness in the epigastric area with a palpable mass in the right upper quadrant, 7 cm below the costal margin. Laboratory examination revealed the following: haemoglobin: 10.7 g/dl, leucocyte count: 9.15 × 10Histopathological examination revealed a huge polypoidal tumour measuring 11.0 cm, filling the lumen of the gallbladder; the findings were consistent with those of carcinosarcoma, revealing an anaplastic sarcomatous component with a heterologous chondrosarcomatous components. The large omental mass measuring 20 × 10 cm showed an anaplastic poorly differentiated tumour consistent with metastatic carcinosarcoma; the resected liver showed direct invasion by carcinosarcoma (with dominance of the sarcomatous component) from the gallbladder. No direct invasion of the muscularis propria, submucosa, or mucosa was observed. However, peri-colonic fatty tissue invasion was seen. The stomach was not directly involved, there was only an invasion of the peri-gastric fatty tissue. The greater omentum and falciform ligament showed no metastases; 4 of 8 peri-pancreatic lymph nodes showed metastatic poorly differentiated carcinosarcoma. Immunohistochemical staining revealed positivity for both, CK (carcinomatous component) and vimentin (sarcomatous component) stains . The patient was referred to the medical oncology department; however, she refused adjuvant chemotherapy, and presented with recurrence involving multiple segments of the liver 2 months after surgery. She died 1 month later.2.39/L, haematocrit: 40.0%, and platelet count: 325 × 109/L. The liver function test showed: albumin: 39 g/L, total protein: 76 g/L, alanine aminotransferase: 39 units/L, aspartate transaminase: 17 units/L, alkaline phosphatase: 67 units/L, total bilirubin: 3.85 umol/L, and amylase: 49 units/L. The coagulation profile was within the normal range, and cancer antigen markers CEA, CA19.9, and AFP were all within normal ranges. Ultrasound of the abdomen, performed in the referring hospital showed a septate gallbladder with large stones measuring 1.6 × 1.2 cm, and another non-mobile echogenic structure measuring 2.7 × 0.9 cm in the dependent part, suggestive of a focal wall thickening or polyp. She underwent elective laparoscopic cholecystectomy in the referring hospital. The intraoperative findings revealed a distended gallbladder with an hour-glass appearance, suggestive of empyema; focal wall thickening was observed in the body of the gallbladder with an intraluminal mass measuring 2 × 2 cm. Histopathological examination revealed an intraluminal tumour measuring 2 × 2 cm, that appeared to be a well to moderately differentiated carcinosarcoma of the gallbladder. It had invaded the peri-muscular connective tissue through the muscularis layer; no lymph nodes were identified. Immunohistochemical stains showed positivity for both, CK and vimentin stains in the carcinomatous and sarcomatous components, respectively . The patient was referred to the medical oncology department where she received 14 cycles of adjuvant chemotherapy, and was on regular follow up. After 2 years of regular follow up, the CT revealed a localised anterior wall collection with small peri-hepatic fatty densities. The PET-CT scans of the chest, abdomen, and pelvis demonstrated a soft tissue lesion with areas of coarse calcification in the anterior abdominal wall, next to the midline measuring 8.3 × 6.9 cm; it was located near the site of the previous surgical incision A and B. 3In our case series, all 3 patients had tumour recurrence at 3 months, 2 months, and 25 months after the surgery, respectively. The shortest and longest periods of survival were 3 and 86 months. Two patients died from the tumour recurrence and/or the disease progression, while 1 is alive till date.The tumour size was greater than 5 cm in 2 of 3 patients; these patients had survived for 3 and 6 months. The patient with a tumour size of less than 5 cm is surviving for 86 months (7 years and 2 months) from the first surgery. The prognosis of gallbladder carcinosarcoma was therefore found to be very poor despite complete resection of the tumour. Carcinosarcoma of the gallbladder was first reported by Landsteiner et al. in 1907. The low incidence of this entity is a major hindrance to the understanding of its aetiology. In view of the absence of a definite staging system for this rare variant, most of the reported cases are staged based on the tumour-node-metastasis staging system for gallbladder cancer. The incidence rate of carcinosarcoma is approximately 1.2 per 100,000 population per year ,19. CarcTumour size may influence the prognosis and survival rate in these patients. In the meta-analysis conducted by Zhang L et al. in 2008 , patient4The present case series of 3 cases of carcinosarcoma of the gallbladder shows that in this extremely rare and aggressive variant of cancer, surgical resection is the only means of obtaining extended survival. The post-operative survival of 86 months noted in 1 patient from our series who had a tumour smaller than 5 cm, is probably the longest reported in the literature. Cumulative evidence from further cases is needed to improve understanding on this rare and aggressive malignancy.The authors declare no conflict of interest.This study did not receive any funding from governmental or private organizations.Ethical approval was obtained from the Institutional Review Board of the King Fahad Specialist Hospital, Dammam, Saudi Arabia. Reference number (SUR0333) Dated 26/09/2019.Written informed consent was obtained from all of the patients for publication of this case series and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Study concept or design – MYD, MSQ.Data collection – ASQ, AAA, YMG, FQ, AA, OAA.Data interpretation – MYD, ASQ, FQ, OAA, NA.Literature review – MYD, ASQ, YMG, AA, NA.Drafting of the paper – MYD.Editing of the paper – MYD, ASQ, MSQ.Researchregistry5150.Mohammed Yousef Aldossary.Not commissioned, externally peer-reviewed.


Though microaneurysms are the hallmark of diabetic retinopathy (DR), large aneurismal changes termed as ''macroaneurysms'' (MAs) may also occur in the course of chronic diabetic macular edema. MAs are usually accompanied by intraretinal hard exudates, fluid accumulation and retinal hemorrhages. Detection of MAs is clinically important as it implies that macular edema is usually chronic and therefore can be resistant to intravitreal anti-vascular endothelial growth factor injections. Multimodal imaging consisting of fluorescein angiography (FA), indocyanine green angiography (ICGA), optical coherence tomography (OCT) or OCT-angiography (OCTA) can be performed to detect and understand the nature of MA and thereby select proper treatment modality. Herein, we report multimodal imaging features of a 64-year-old woman with insulin-dependent diabetes mellitus presented with treatment naïve severe macular edema and a macroaneurysm at the right temporal macula. In conclusion, FA, ICGA and OCT seem to be far superior to OCTA to detect these lesions due to probable slow flow inside the MA. Microaneurysms, hard exudates, dot or blot hemorrhages, macular edema, capillary occlusion, cotton-wool spots and neovascularization are among the clinical features of diabetic retinopathy (DR) . Though Herein, we demonstrated multimodal imaging  features of a retinal MA in a patient with insulin-dependent diabetes mellitus (IDDM) and severe treatment naïve diabetic macular edema (DME).A 64-year-old female with IDDM presented with a gradual visual decline in both eyes (OU) and examined in October 2019 at Department of Ophthalmology . Patient consent was obtained to write this case report. This study was performed in compliance with the principles of the Declaration of Helsinki. She had IDDM for 18 years with a recent HbA1c level of 6.2%. On admission, her best-corrected visual acuity with Snellen chart was 0.4 in right eye (OD) and 0.5 in left eye (OS). Slit-lamp examination and intraocular pressure had normal findings in OU. A detailed fundus examination revealed multiple microaneurysms, blot hemorrhages and circinate exudates at the macula bilaterally. In addition, a MA was detected at the superior temporal area of the fovea in OD . FA showed multiple hyperfluorescent lesions in the early phase corresponding to microaneurysms in OU. In addition, a slow filling of large diameter hyperfluorescent lesion  with no leakage was observed at the superior temporal area of the fovea in OD . Full le. OCTA showed a round-demarcated faded flow area at the level of superficial and deep capillary plexus . Hypointense flow area corresponding to MA-related back-shadowing was noted at the choriocapillaris slab . OCT scan of the right eye revealed an oval lesion corresponding to aneurysm. This lesion had a hyperreflective wall, containing partially hyperreflective lumen and back shadowing. Also, there was a thickened retina surrounding the MA, hyperreflective dots and serous subretinal fluid suggestive of inflammation . OCTA shy plexus . Hypointris slab . Regarding these findings, severe non-proliferative DR, DME and DR-related MA were suggested. Although a combined treatment of direct focal argon laser photocoagulation onto the MA together with an intravitreal dexamethasone implant was recommended, but she denied any treatment due to reimbursement issues of dexamethasone implant. She was lost to follow-up.In this case presentation, comprehensive multimodal imaging features of a large retinal capillary MA were presented in a patient with IDDM and severe DME. DR-related MA was first described by Bourhis et al.  Because MAs have diameters larger than 150 μm and localized very close to microaneurysms and share similar OCT and ICGA features , 5. It iProgressive staining of intraluminal material (proteins and/or lipids) is suggested to be the cause of delayed or prolonged dye filling in ICGA. Histological studies have also shown that intraluminal materials such as lipids and/or stagnant blood and even thrombosis have a high affinity for staining with ICG . ICG thaSince FA is routinely performed in eyes with DR, many MAs can be easily overlooked. Therefore, the presence of MAs cannot be recognized. MAs may not respond well enough to intravitreal anti- vascular endothelial growth factor treatments. Thermal laser photocoagulation has been reported to be more effective in these patients. ICG-guided laser photocoagulation for capillary MAs has been investigated by Paques et al. . In thisPerifoveal exudative vascular anomalous complex (PEVAC) is a recently defined entity that should be considered in the differential diagnosis of retinal MAs, which occurs as a unilateral, isolated, perifoveal lesion. Large aneurysmal dilatation, hard exudates, intraretinal hemorrhages and fluid accumulation are the common features of both entities. However, the pathogenesis is different. It is considered that PEVAC is resulted from progressive retinal endothelial cell degeneration. In addition, while telangiectatic capillaries usually occur in retinal vascular diseases such as retinal vein occlusion (RVO) and DR, PEVAC is reported in otherwise healthy subjects , 15. TheClinicians should look for the presence of MAs in patients with severe and chronic macular edema. In light of the present case and the literature review, we believe that FA, ICGA and OCT may be equally helpful for detection and visualizing the extent of MAs. The diagnostic criteria, treatment strategies and prognosis of DR-related MAs are not well-defined. We believe that the definitive diagnosis criteria, treatment alternatives and its prognosis will be better determined by further studies.


Esophageal liposarcoma represent a rare cause of esophageal tumor. According to the literature, since the first case reported in 1983, only 42 cases of esophageal liposarcoma were reported.We present a case of liposarcoma in the lower oesophagus treated by surgical resection.Liposarcoma in the oesophagus gastrointestinal tract is an uncommon. An analysis of the literature reports 42 cases.In many cases the diagnosis was established in postoperative period.Surgery is the standard treatment including polypectomy, total or subtotal oesophagectomy.There is no conventional treatment of this pathology. The curative treatment requires surgical resection or endoscopic approach for selected tumor. It is a2A 44- year- old men presented with progressive dysphagia initially for solid then for liquid food since three years, associated with weight loss of ten kilogram. Physical examination was normal an oesophageal cancer was suspected in first intention, the patient underwent an upper gastro intestinal endoscopy visualising a sub mucosal mass at 26 cm from incisors. The gastro intestinal junction and gastric mucosa were normal. The biopsies of this mass did not show any malignancy sign. A computed tomographic scan of the chest with intra venous contrast and oral opacification showed a well circumscribed mass in the low oesophagus , Fig. 2.3Liposarcoma in the gastrointestinal tract is an uncommonpathology especially in the oesophagus  . Only 42The first case was described by Mansour et al in 1983 . The mosSome patients may presented an uncharacteristic symptoms like chest discomfort, cough, shortness of breath, making the diagnosis more difficult . Due to The combination between the CT scans and the MRI can help, by evaluating the fat component of the tumour, to differentiate between lipoma with 100% fat content and liposarcoma with less than 75% of fat content . In many4Oesophageal liposarcoma is a rare disease, there is no conventional treatment for this pathology. The curative treatment requires surgical resection or endoscopic approach for selected tumor.No potential conflict of interest relevant to this article wasreported.No source of funding.We have reported a single case with no requirement for ethical approval. This manuscript does not describe a clinical study.Written informed consent was obtained from the patient for publication of this case report and accompanying images.Ben safta Y: study concept, Data collection, data analysis or interpretation, writing the paper.Soui F: study concept, writing the paper.Maatouk M: study concept, writing the paper.:data collection.Zehani A: data collection.Mabrouk A: data collection, data analysis or interpretation.Daldoul S: data collection, data analysis or interpretation.Sayari S: data collection, data analysis or interpretation.Haouet K: advised and designed the report.Ben Moussa M: advised and designed the report.N/A.Yacine Ben Safta.Not commissioned, externally peer-reviewed.


Sciatic hernias are the least common type of pelvic floor hernias. The purpose of this study was to present a novel technique for manual reduction and to conduct a systematic review of previous reports of sciatic hernias to characterize them and review the outcomes.An 86-year-old female presented with left-sided lumbar pain. She had a past medical history of rheumatoid arthritis and was treated with prednisolone and methotrexate. Her left abdomen and left lumbar area were tender. An unenhanced abdominal computed tomography scan revealed invagination of the left ureter into the left sciatic foramen and a dilated left proximal ureter and renal pelvis. Ultrasonography showed an invaginated left ureter viewing from the left buttock. She was diagnosed with a sciatic hernia. Ultrasound-guided manual transvaginal reduction was performed. Post-procedure unenhanced abdominal computed tomography scan confirmed reduction of the ureter. After 10-months of follow-up, there is no evidence of recurrence.Previous reports of patients with sciatic hernia were identified. Clinical data associated with the hernia, reduction technique and clinical outcomes were collected for 72 patients. Open reduction was performed in 24 patients. A ureteral stent was placed in eight patients when the hernia contained the ureter. Four postoperative complications including one death were reported in adults. There were no reports of closed manual reduction.A sciatic hernia in women may be manually reduced without surgery. Further reviews of this rare entity are needed to determine the best management strategy. Sciatic hernias are characterized by the hernia contents entering the greater or lesser sciatic foramen. The greater sciatic foramen is subdivided by the piriformis muscle and atrophy of the piriformis muscle may be one cause of sciatic hernia. Sciatic hernia was first described by Papen in 1750 and observed and recorded by Verdier in 1753 [The purpose of this study was to present a novel technique for manual reduction and to review published reports of sciatic hernias to summarize the experience to date in the management and outcomes of this entity. This work has been reported in line with the SCARE criteria .2An 86-year-old female presented with left-sided lumbar pain. She had a past medical history of rheumatoid arthritis and was treated with prednisolone and methotrexate. On physical examination, her left abdomen and left lumbar area were tender. Laboratory examination showed no abnormalities. An unenhanced abdominal computed tomography (CT) scan revealed invagination of the left ureter into the left sciatic foramen and a dilated left proximal ureter and renal pelvis . Ultraso3A search of English-language abstracts in PubMed and Igakuchuo-Zasshi through 2017, with keywords of “sciatic hernia” or “ureterosciatic hernia” revealed a total of 71 patients with sciatic hernias . Of 72 pOf 61 adults including the present patient, 57 (93%) were female. Of the 11 children found in this review, five (45%) were female. This suggests that sciatic hernias tend to occur more frequently in adult females. However, there is no difference in incidence between genders in children. Atrophy of the piriformis muscle has been described as a predisposing factor. Therefore, elderly patients with decreased body mass index tend to have this condition. Common symptoms include unilateral lower abdominal pain, lumbar pain, and bulging of one buttock. In adults, the hernia contents have been reported to include the ureter (N = 26), small bowel (N = 14), tumors  (N = 8), colon (N = 2), ovary (N = 2), appendix (N = 1), ascites (N = 1), preperitoneal fat (N = 1), multiple organs (N = 6).Formerly, the diagnosis of sciatic hernia was made by physical examination (e.g. bulging) or at the time of operation. After the advent of the CT scan, it is the mainstay of diagnostic modalities to identify a sciatic hernia. Intravenous pyelogram or retrograde pyelogram have been performed for some patients with ureterosciatic hernias. The “curlicue” sign of the ureter was specific for this entity if the hernia contains the ureter .The treatment of a patient with a sciatic hernia depends on the hernia contents and commonly includes surgery  or placement of a ureteral stent if the ureter is involved. Open reduction with laparotomy was performed in 24 patients in the series reviewed. Recently, nine patients were reportedly treated laparoscopically and two by robotic-assisted surgery. Two patients underwent conversion from laparoscopy to laparotomy. There are no reports of successful transvaginal closed manual reduction.Transvaginal closed manual reduction was used to treat the present patient. With the patient in the prone position, the assistant places the ultrasound probe on the left buttock. The exact location of the hernia was confirmed by the CT scan. After confirming the location of the hernia, the operator inserted the right hand into the vagina, while extending the index and middle fingers . The entIn the combined series of 72 patients, postoperative complications include one death from sepsis, one anastomotic leak, one patient developed heart failure, one patient developed pneumonia, and two recurrences occurred in adults. In children, there was one death from bronchopneumonia. Two recurrences are reported after a repair without using mesh (1/20) and after removal of the ureteral stent (1/3). There are deaths reported after operative repair.Transvaginal manual reduction is less invasive and easier than other reported approaches. If there are no suspicion of strangulation of the invaginated tissue, it may be considered as the first modality to be used. However, there is a possibility of recurrence because the hernia defect has not been definitively closed. In addition, this maneuver is not applicable to men, children , and possibly, young females whose vagina may not be able to accommodate the examiner's hand.4An incarcerated sciatic hernia in women can be manually reduced. To determine the best management strategy, further studies and collection of data regarding this rare entity, treatment and follow-up are necessary.All authors have no conflict of interest.Authors had no sources of funding.IRB/Ethics Committee ruled that approval was not required for this study.Written informed consent was obtained from the patients 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.The work presented was carried out in collaboration between all authors. JK, KY, TS, AKL, and TK defined the research theme, discussed analyses and approved the final version to be published. JK analyzed the data, interpreted the results and wrote the paper.There is no need to register because it is a case report.Jiro Kimura.Not commissioned, externally peer-reviewed.


Lymphangiomas are unusual benign malformations, which can often be misdiagnosed due to their relatively mild and nonspecific symptomatology. Their appearance in the anal canal is extremely rare. Correct diagnosis is necessary for formulating appropriate management. We present the case of a 42-year-old man, with a medical history of ulcerative colitis and mild symptomatology, who was diagnosed with lymphangioma of the anal canal after undergoing a colonoscopy. Tumors of the anal and perianal region are relatively uncommon, especially mesenchymal benign ones . LymphanA 42-year-old man with a history of ulcerative colitis was referred to the surgical department by the gastroenterologist, after finding a lesion in the anal canal during follow-up colonoscopy . The patThe patient complained of periodic anal pain, discomfort, and rectal bleeding during defecation, which started approximately 6 months earlier. On physical examination, he had an unremarkable abdominal exam. On digital and anoscopic examinations, there were third degree prolapsed hemorrhoids and an ulcerated, soft lesion on top of the 3 o'clock hemorrhoid. Colonoscopy revealed no additional findings from the rest of the rectum and colon. No biopsy was taken during colonoscopy due to the location of the lesion. Consequently, a surgical biopsy of the anal lesion was performed under local anesthesia, as a day case, without any complications. Histopathological examination showed lymphangioma of the anal canal.A wide excision of the lesion under general anesthesia followed . The newLymphangiomas are uncommon malformations of the lymphatic system that can occur anywhere in the skin and the mucous membranes. They can become evident at any age, but the greatest incidence occurs at birth or early in life. The most common sites are the head and neck, although they can be sometimes found in the intestines, the pancreas, and the mesentery .Cases of lymphangiomas occurring on the lower gastrointestinal tract have been rising in the past years, probably due to the extensive use of specialized diagnostic examinations. So far, lymphangiomas of the anal canal have been extremely rare, since only 3 other cases , 3, 4 haThe clinical presentation of lymphangiomas can vary, from asymptomatic to iron deficiency anemia or rectal bleeding , 3, 4. ODue to the rarity of lymphangiomas, the detection of a tumor in the anal canal always prompts an extensive differential diagnosis. They can be clinically confused with other lesions that can appear in this area such as malignancies and less frequently other benign neoplasms, such as lipomas, papillary hidradenoma, and epidermoid cysts . Also, iThe endoscopic findings using magnifying endoscopy with narrow-band imaging techniques could be useful in making the diagnosis of a lymphangioma, as suggested by a report . MucosalAs a result, it is suggested by the authors that the diagnosis of lymphangiomas of the anal canal, even though highly unlikely, should be considered when having to differentiate a newfound tumor of the anatomic area. Also, we suggest total excision and biopsy of these tumors as the treatment of choice, as well as any excised lesion of the area, due to the possibility of missing a potential histologic diagnosis. The excision can be done either surgically as in this case or endoscopically as described in other reports, by snare polypectomy  or endosLymphangioma of the anal canal is a very rare condition but should be considered during differential diagnosis of tumors in the area. In the future, endoscopy could play an important role in the diagnosis of this rare entity. The treatment consists of excision of the lesion, either surgically or by endoscopy.


Primary cutaneous anaplastic large cell lymphoma is a CD-30 positive lymphoproliferative disorder with good prognosis, usually treated with radiation therapy and surgery. Head, neck, and extremities are the most frequently involved sites. In this paper, we describe an unusual case of oral localization, recurring after skin-involving radiotherapy, successfully treated with sixteen cycles of brentuximab vedotin. This could be a more effective approach with a less detrimental toll for treating these rare disorders. Primary cutaneous CD-30 positive lymphoproliferative disorders are a rare and heterogeneous group of primary skin tumors, accounting for about 30% of all cases of primary cutaneous T-cell lymphomas (PCTCL) . The groPCALCL is most commonly found in adults (median age at diagnosis is 60 years old), occurring more often in men than women (3 : 1). Macroscopically, PCALCL usually presents itself with red to violaceous lesions, rapidly growing and frequently ulcerative. Head, neck, and extremities are the most frequently involved sites, both for solitary lesions or for multiple grouped nodules. Systemic symptoms, such as fever, fatigue, and mucosal involvement, are rare in PCALCL. However, in at least 10% of cases, neoplastic CD30+ cells can be found in locoregional lymph nodes. Its course is generally indolent (dissemination is uncommon); spontaneous regression occurs in about 40% of cases, even though partial remission is documented more frequently than complete ones –4.The first line treatment currently accepted is radiation therapy or surgical removal, whenever possible. In case of multifocal lesions, methotrexate is accepted followed by low-dose maintenance . HoweverHistopathologically and microscopically, PCALCL exhibits a high expression rate of CD30+ antigen , positivity for CD4+ antigen and does not present ALK positivity . In thisIn November 2016, a 40-year-old Caucasian male came to our attention for a nodular, erythematous, nonulcerated, well-defined, and indurated lesion localized in his right forearm.18F-FDG in the right forearm region . As we made diagnosis of PCALCL, the patient began radiation therapy (dose of 3600 cGy), achieving complete response.A skin punch biopsy of the lesion was performed, showing an inflammatory multinodular infiltration of lymphohistiocytic cells in both deep and superficial dermis, including many large, atypical, anaplastic cells with frequent mitosis, characterized by the following immunohistochemistry: CD45+, CD2+, CD3+/–, CD7+/–, CD20–, CD30+, and ALK–. To rule out systemic anaplastic large cell lymphoma, we performed a bone marrow biopsy  and a CT/PET scan that revealed a significant and singular uptake of A new nodular erythematous lesion of 1 cm appeared in March 2017 in the right inguinal region and was unsuccessfully treated with topical steroids. One month later, when it expanded up to 3 cm, a new CT/PET scan was performed and revealed the lesion as the only pathological uptake in the right inguinal region. The patient underwent another radiation therapy of 3600 cGy, obtaining complete response again.In May 2017, two more nodular lesions appeared in the patient's oral cavity, in the left hard palate . A punchTo avoid radiation therapy and its side effects and taking into account the short time to relapse that the patient experienced with previous radiation therapy in different sites, it was decided to start systemic immunochemotherapy with brentuximab vedotin (BV) 1.8 mg/Kg every 21 days. Interesting later reviews about reduction in doses and numbers of cycles of BV have been published recently , showingSince the patient did not present any neuropathy and experienced early relapse after initial remission obtained with radiotherapy, we continued treatment with BV for sixteen cycles  with no To the best of our knowledge, this is the first documented case of PCALCL present only in the hard palate, while other few cases of similar processes involving mucosal sites of the head and neck have been previously reported with one case of systemic anaplastic large cell lymphoma involving the hard palate .We demonstrated that oral PCALCL could be successfully treated with systemic immune-chemotherapy with brentuximab vedotin, an anti-CD30 antibody conjugated to monomethyl auristatin E and an effective antimicrotubule agent that was previously approved for Hodgkin lymphoma, ALK-positive anaplastic large cell lymphoma , and othUsing immune-chemotherapy instead of radiation therapy can prevent several side effects, such as mucositis, candidosis, dysgeusia, radiation caries, osteoradionecrosis, soft tissue necrosis, progressive periodontal attachment loss, trismus, and xerostomia, which are all well documented while treating mucosal lesions with radiations .Brentuximab's most common side effect is peripheral sensitive neuropathy that can lead to dose reduction or even drug withdrawal. However, our patient did not experience such neuropathy and could complete therapy within the sixteen cycles at full dose.Due to the rarity of this group of cutaneous lymphoproliferative disorders , it is still a challenge to make a trial comparing radiation therapy with immune-chemotherapy using BV. From this case, we can infer that although BV is not side-effect free, it offers a safe choice to treat primary cutaneous CD-30 positive lymphoproliferative disorders no longer responding to radiation therapy and standard chemotherapy or cases where the anatomic site involved does not allow RT or must be avoided due to its side effects.


Migration of endoclips and stitches into the duodenum after laparoscopic hepatectomy is incredibly rare with a poorly understood mechanism.A 56-year-old woman who underwent laparoscopic left hepatectomy and cholecystectomy in August 2016 was admitted to our hospital with nausea and vomiting in December 2017.Abdominal computed tomography (CT) scan showed high density shades in duodenal ampulla. Esophagogastroduodenoscopy showed deformation of the duodenal ampulla into two lumens; hem-o-lock clips and stitches were detected in the upper lumen. Contrast enhanced CT scan revealed gastric cancer with liver metastasis (GCLM).The hem-o-lock clips and stitches were present in the wall of the duodenum; therefore, no attempt was made to remove them. High quality liquid diet, partial parenteral nutrition, and chemotherapy were administered to the patient.In September 2018, the patient died of hepatic failure caused by GCLM.This rare complication of the migration of endoclips and stitches into the duodenum after laparoscopic hepatectomy can cause epigastric pain and duodenal obstruction. The complication could be potentially avoided using absorbable endoclips and stitches or by performing of ultrasonic dissection by a skilled operator. A previous study reported that most clips migrated into common bile duct (CBD) and few migrated into the duodenum or other body parts. In this study, we present a rare case of migration of hem-o-lock clips and stitches into the duodenum after laparoscopic left hepatectomy and cholecystectomy.Laparoscopic hepatectomy and cholecystectomy are the currently preferred procedures for patients with hepatolithiasis and cholecystolithiasis, respectively, resulting in good aesthetic outcomes and rapid recovery. In these procedures, surgical clips and stitches are used laparoscopically to control bile duct, hepatic vessels, and cystic artery. However, wide use of surgical clips, such as hem-o-lock clips, in laparoscopic cholecystectomy has led to a variety of complications.The Ethics Committee of Qilu Hospital of Shandong University (Qingdao)  approved this study, and the written informed consent was obtained from the patient.2A 56-year-old woman had epigastric pain in June 2016; her abdominal computed tomography (CT) scan showed gallbladder stones and hepatoliths. Laparoscopic left hepatectomy and cholecystectomy were performed in August 2016. Preoperative imaging showed normal extra-hepatic biliary anatomy (no short or long cystic ducts). One year later, the patient experienced epigastric pain which radiated to the back and nausea which worsened after eating. She lost 7-kg weight in 3 months. There was no history of gastrointestinal bleeding or jaundice. In December 2017, abdominal CT scan revealed gastric cancer with liver metastasis (GCLM)  are rare. However, no evidence of ulcer or active bleeding was present in this case.In previous cases, only metallic or hem-o-lock clips were found in the CBD or duodenum. However, in this case, apart from hem-o-lock clips, stitches were also found by EGD. In most reported cases, clips were usually found in duodenal ulcers with active bleeding.A variety of mechanisms of clip migration have been proposed.(1)The first aspect to consider here is the procedure. Because of the incomplete closure of the cystic duct caused by ineffective clips, the duct remains partially patent resulting in bile leakage.(2) Moreover, the difficult dissection at the Calot's triangle and clip-induced inflammatory process may cause adhesion between the duodenum and ligated cystic duct and arterial stump with subsequent formation of choledochoduodenal fistula.,9Anatomically, the duodenal wall lies adjacent to the ligated cystic duct and cystic artery stump; therefore, the clips may directly erode the duodenal wall.(3),10 However, none of these mechanisms was found to cause clip migration in our case.Despite the low rate of occurrence, an undiagnosed pre-existing duodenal ulcer may cause adherence of the duodenum to the gallbladder fossa, capturing the clip in the inflammatory process.(4)In the present study, the patient had GCLM. Normal anatomic relationship was destroyed by the growing tumor, which compressed the hem-o-lock clips and stitches into the duodenal wall.,11 Ultrasonic dissection without the use of clips is also a viable option and could be broadly used in laparoscopic hepatectomy. Both the meticulous dissection of the Calot's triangle and the accurate placement of clips depend on the surgical skill of operators. Misplaced and wandering clips should be quickly detected and removed during the procedure. Comprehensive preoperative assessments such as CT, magnetic resonance imaging (MRI), magnetic resonance cholangiopancreatography (MRCP), are also important to identify anatomic variations.Absorbable clips and stitches have been used more commonly since several years. Although the cost is marginally higher, absorbable clips and stitches should be the first choice to eliminate the possibility of clip migration.Data curation: Xiao-fei Gao, Cheng-yu Shi.Formal analysis: Cheng-yu Shi, Yuan-hui Jiang.Resources: Yuan-hui Jiang.Supervision: Xin Yi.Writing – original draft: Yan Xia.Writing – review & editing: Yan Xia.