The authors present an extremely rare case of an aggressive and progressive vertebral capillary hemangioma of the lumbar spine secondary to a trauma. A 40-year-old man who complained of back and leg pain due to a hemangioma of L1 that had begun a year after the fracture of the same vertebra was subsequently operated on. Due to the profuse bleeding, only a subtotal removal was possible. Histopathological diagnosis of the lesion revealed a capillary hemangioma. Postoperative control MRI taken at eight months showed that the lesion and destruction of the L1 vertebra were progressive. A second embolization procedure was performed and this time the hemangioma was totally removed via an anterior approach and corpectomy. Fusion was achieved by Th12-L2 graft and plaque. In the fourteenth year of follow-up, he was symptom-free and radiologically clear of this lesion. We propose that progressive hemangioma is extremely rare and that its cure is possible by total surgical removal of the lesion. This case is the second extradural capillary hemangioma secondary to spinal trauma ever to have been documented in English literature. The emergence of a hemangioma in a fractured vertebra suggests that its pathogenesis can be related to the deviation of the angiogenetic pathways from the normal healing process. A 40-year-old man had a car accident in 1996 which caused the burst fracture of L1 as diagnosed on a spinal CT scan and fracture of left femur diaphysis. This examination revealed absolutely no hemangioma or any other spinal soft tissue lesions . The L1 At one-year follow-up, neurological status examination remained unchanged, but the CT scan showed that the tumor was more destructive , while tVertebral hemangiomas are frequently encountered on plain radiographs, CT scans, and MRIs. These lesions are found in between 10 to 12% of routine autopsies but those which cause clinical signs are very rare; of spinal tumors treated, only between 2 and 3% are hemangiomas [On plain radiographs and CT scans, vertically oriented vertebral lucencies separated by thickened trabecular bone are typical and give the appearance of honeycomb.MRI shows the degree of soft tissue extension and the severity of the compression of the spinal cord. Vertebral hemangiomas can contain large feeding or draining vessels; hence, preoperative spinal angiography and preoperative embolization are highly recommended.The formation of new blood vessels (neovascularization) is essential for normal tissue growth during embryonic development. Physiological neovascularization in adults is observed only in a few conditions such as wound and fracture healing, placenta formation, and the menstrual cycle. Abnormally activated neovascularization may occur in pathological states such as in cancer growth, diabetic retinopathy, and age-related macular degeneration.A history of trauma has been elicited in up to 35% of the synovial hemangiomas of the knee. Also, in many patients with hemangioma of the skin, trauma has been regarded as an etiological factor , 3. RecuIn the therapeutic armory, there are radiotherapy, intra-arterial embolization, direct ethanol injection, vertebroplasty, and surgery. They all have varying degrees of success. Surgical decompression is undertaken when significant or progressive neurological deficit is present.One of the main causes of perioperative morbidity for patients undergoing surgical treatment for vertebral hemangioma is “excessive intraoperative blood loss” and postoperative hematoma . PreoperIntra-arterial embolization followed by laminectomy is a safe and effective procedure for the treatment of cord compression by intraosseous vertebral body and posterior element hemangiomas. Gross total resection obviates the need for postoperative irradiation and minimizes the likelihood of recurrence .Vertebral hemangiomas are radiosensitive lesions and doses as low as 30 to 40 Gy are effective , 12.To our knowledge, this is the second report of posttraumatic extradural spinal hemangioma. The first case of extradural capillary hemangioma secondary to spinal trauma was published in 2011 by Shilton and involved an extradural homogeneously enhancing mass on a basis of T7 and T8 vertebral compression fractures following a motor vehicle accident ten years previously . AnotherThe present case demonstrates particularly the “growing” ability of the hemangiomas, as shown by the MRI from the first postoperative period. This growth potential obliged us to wait sixteen years before publishing our case; we believe that sixteen years of follow-up is a more than acceptable period of time in order to pronounce the absence of recurrence.Although absolute proof of cause and effect as in our case is very rare, the emergence of a hemangioma in a fractured vertebra suggests that it is related to the deviation of the angiogenetic pathways from the normal healing process. An aggressive vertebral hemangioma that has been complicated with vertebra destruction and neural compression indicates the need for preoperative embolization and total surgical removal. Such a total resection can be safely achieved and seems to prevent recurrence without adjuvant therapy.


Tension-free repair using mesh is a common inguinal hernia surgical procedure. However, various complications such as mesh-related infection and recurrence may develop as a result. Moreover, although rare, there are also reports of intestinal obstruction caused by adhesion of the mesh to the intestinal wall and cases of mesh migration into various organs. Here, we report our experience with a patient in whom mesh extraction was performed due to migration of mesh into the intestinal tract following inguinal hernia surgery and formation of a fistula with the bladder.Our patient was a 63-year-old Japanese man who had a history of operative treatment for right inguinal hernia during early childhood. Because a relapse subsequently occurred, he was diagnosed as having recurrent right inguinal hernia at the age of 56 years for which operative treatment (the Kugel method) was performed. He presented to our hospital 6 years later with the chief complaint of lower abdominal pain. Computed tomography findings revealed a mass shadow in contact with his bladder and cecal walls, and enteric bacteria were detected in his urine. Furthermore, because lower gastrointestinal endoscopic findings confirmed mesh in the cecum, we performed operative treatment. The mesh had migrated into the cecum and a fistula with his bladder had formed. We removed the mesh through ileocecal resection and partial cystectomy.It appeared that a peritoneal defect occurred when the mesh was placed, allowing the mesh to migrate into our patient’s intestinal tract. Because contact between the mesh and the cecum resulted in inflammation, a fistula formed in his bladder. It is important to completely close the peritoneum when placing the mesh. Tension-free repair using mesh has become a widely utilized technique in inguinal hernia surgery because it has a lower relapse rate and fewer postoperative complications than other procedures . Howeveret al. [et al. reported a case of a sigmoid colonic fistula secondary to a mesh plug [Chuback et al.  reportedesh plug . Also, tesh plug .Normally, the peritoneum is between the mesh and the abdominal cavity, so the mesh cannot come into direct contact with the intestines or other organs. However, mesh migration is believed to occur because of a defect in the peritoneum due to incomplete peritoneal repair  or becauThe present report describes a case of mesh migration into the intestinal tract complicated with bladder fistula that occurred 6 years after surgical repair using the Kugel method.Our patient was a 63-year-old Japanese man who had a history of operative treatment for right inguinal hernia during early childhood, although the type of procedure was unknown. Relapse subsequently occurred, and at the age of 56 years, he received a diagnosis of a recurrent right inguinal hernia, for which the Kugel method was performed. Six years later, he visited a local physician with the chief complaint of lower abdominal pain. Computed tomography (CT) showed thickening of his bladder and cecal wall; mesh infection was suspected and he was referred to our department.Mild tenderness was apparent in his lower abdomen at presentation. Hematologic findings revealed that although his white blood cell count was normal, his C-reactive protein was slightly increased to 1.44 mg/dl. His urine analysis was positive for occult blood and bacterial contamination, and enteric bacteria were detected in urine cultures. An abdominal CT showed a poorly defined mass in contact with the cecal wall in the right inguinal region; the mass was also in contact with his bladder wall Fig. .Fig. 1AbBecause we suspected that the fistula formation in his intestinal tract and bladder was due to the mesh, we scheduled a mesh removal procedure, but our patient felt left testicular pain 2 days before surgery, and an ultrasound examination revealed swelling of the left epididymis. Therefore, we diagnosed epididymitis. We thought that a mesh-related urinary tract infection was the cause of the epididymitis and performed surgical removal of the mesh.Following laparotomy with a midline abdominal incision in which intraperitoneal observation was performed, the cecum was found to be firmly adhered near the right internal inguinal ring. There was no mesh exposure in his abdominal cavity, but when the adhesion was separated, we confirmed the presence of mesh migrating into his intestinal tract and piercing the cecal wall from the preperitoneal space. The inner side of the mesh was in contact with his bladder wall. We performed ileocecal resection to extract the mesh, and the mesh, including bladder wall, was finally excised by partial resection Fig. . PartialIn the excised specimen, the mesh had penetrated the cecal wall and was exposed in the intestinal tract Fig. . On histWhen inguinal hernia surgery is performed, the tension-free method is standard, but operative techniques such as the plug and Kugel method utilizing various meshes are also performed. However, as the use of mesh has become more common, mesh-specific complications such as intestinal obstruction and migration of the mesh into the bladder have been reported –11. A seDirect contact between the mesh and the organs  and stroIn the case of the present patient, there were adhesions around the hernia sac caused by inguinal hernia surgery during early childhood, and it is possible that peritoneal closure was incomplete. Moreover, because the mesh had migrated so as to protrude into the intestinal lumen, it is possible that the border of the mesh may have caused excessive strain on the intestinal wall. Thus, it can be assumed that the mesh and the cecum came into contact with each other, causing a foreign body reaction, and postoperatively, the mesh gradually migrated into his intestinal tract over time due to movement of the mesh and intestinal tract , 15.As to the fistula formation with his bladder, there is a possibility that the mesh and his bladder wall came into contact, similar to what happened in the intestinal tract. However, the Kugel patch is normally placed so as to be in contact with the bladder wall, and the tissue intervening between them is the preperitoneal fat; the peritoneum is not present. Nevertheless, there are almost no reports in which the Kugel patch formed a fistula with the bladder, and it is unlikely that only contact between the mesh and the bladder caused the fistula in the present case. Furthermore, because in the present patient a trace of mesh was found in the fibrosis site around his bladder, and a fistula covered with inflammatory granulation tissue was present in the bladder lumen at this site, it can be postulated that the fistula was formed because of the infection caused by the mesh after it migrated.In the present patient, approximately 6 years had passed since the mesh was placed, and it is unknown when the migration occurred. A review of the literature on mesh migration revealed that symptoms develop from 1 to 20 years after surgery , 15. If The frequency of migration of mesh to the internal organs and fistula formation is low, but it is difficult to treat when the condition occurs. Although there are various causes, in terms of the surgical technique, in order to prevent complications it is important to completely close the peritoneum when placing the mesh and sufficiently separate the preperitoneal space so as to not allow the mesh to place tension on the peritoneum and surrounding organs.


Blunt abdominal injury in children can be a significant diagnostic and therapeutic challenge. The extent and localization of organ damage cannot be always thoroughly investigated noninvasively and in spite of modern imaging techniques and a laparotomy may be necessary for diagnosis, even though it carries a significant morbidity. We present a rare case of isolated gastric rupture after blunt abdominal injury in a 12 year old boy that sustained a bicycle accident. He was hemodynamically stable, had signs of acute abdomen and axial tomography was inconclusive as of the site of visceral perforation. Definitive diagnosis and treatment were carried out laparoscopically with excellent results. Laparoscopic surgery in cases of blunt abdominal injury with gastric rupture can serve both as a diagnostic and therapeutic modality with the additional advantage of being less traumatic. The accumulation of relevant experience is mandatory in order to establish this modality in the diagnostic and therapeutic protocols. Laparoscopic surgery (LS) is gradually substituting open surgery not just as an alternative operative technique but also in the planning of patient management in pediatric blunt abdominal injuries (BAI) , 2. Our A 12 years old boy was referred to our facility one hour after sustaining a BAI caused falling on the steering bar of his bicycle. He was hemodynamically stable and laboratory report was normal. He reported epigastric pain and his upper abdomen was tender, with guarding. The child was cooperative and did not carry any other major wounds. No pain could be elicited in the thorax, the pelvis, or other areas of the body. An IV line was established. A supine chest X-ray and a CT scan of the abdomen and pelvis with IV contrast were performed. Free intraabdominal air, especially in the upper abdomen and significant amount of free fluid in the right and left paracolic gutter and around the stomach with considerable soiling of the area were evident. A large amount of free fluid was present in the pelvis. Radiologists indicated the anterior gastric wall, the duodenum and the sigmoid colon as the most likely points of injury. No other pathology was apparent. A nasogastric tube was placed which revealed bloody gastric content.2 pneumoperitoneum at 10mmHg and 4L/min flow was established. Upon introducing the scope, a frothy liquid material was observed between the liver and the stomach  as the body accommodates the high energy of sudden velocity change into increased luminal pressure, causing gastric blowout . BecauseLaparoscopy can enhance the strategic algorithms for BAI. It allows both the diagnosis and treatment of these occult injuries, while mitigating the concern about the morbidity of a laparotomy. More specifically, in gastric rupture, this approach is effective, allowing a thorough inspection of the abdomen, evacuation of the irritable and contaminated material and reconstruction of the gastric wall.The authors declare no competing interest.


To determine the incidence of regional lymphadenopathy in gastrointestinal (GI) schwannoma and to evaluate the relationship between peritumoral lymphoid cuff and lymphadenopathy.We queried 118 GI tract schwannomas and reviewed radiologic findings, intraoperative findings, and electronic medical records of all cases for enlarged regional lymph nodes.p = 0.001). Of the 106 cases for which clinical or radiologic data was available for, 76 cases (71.7%) showed regional lymphadenopathy. The presence of peritumoral lymphoid cuff showed significant correlation with regional lymphadenopathy (p < 0.001) and the size of enlarged lymph nodes (p = 0.002).Location of tumors included 85 gastric (72%), 11 colonic (9.3%), 7 esophageal (5.9%), 3 pancreatic (2.5%), 1 hepatic (0.8%), and 11 mesenteric (9.3%). The size of the tumors ranged from 0.2 to 11 cm (mean 3.8 cm). Histologically, 70.3% showed a peritumoral lymphoid cuff ranging in thickness from 0.3 to 6 mm (mean 1.6 mm). The peritumoral lymphoid cuff was significantly more frequent in gastric schwannomas (78.8%) followed by colonic (72.7%), esophageal (57.1%) and rare in other locations , a lymphoid cuff was not present warranting continued need for caution in the preoperative radiologic and postoperative pathologic diagnoses. The differential diagnosis for subepithelial tumors of the gastrointestinal (GI) tract is broad and includes a wide range of benign and malignant entities including gastrointestinal stromal tumor (GIST), schwannoma, lymphoma, neuroendocrine tumor, leiomyoma, and metastatic carcinoma , 2. Of tIn addition to their subepithelial location and similar histologic findings, schwannomas and GISTs also show similar macroscopic features . AdditioIn this large series of GI tract schwannomas, we reviewed the clinical, pathologic, and radiologic findings of schwannomas and positively correlated the presence of a peritumoral lymphoid cuff with regional LAD.Patients’ ages at diagnosis ranged from 25 to 80 years . The female to male ratio was 1.7:1. The anatomic location of GI tract schwannoma varied with gastric being the most common  followed by colonic , esophageal , pancreatic , hepatic , and intra-abdominal soft tissue around the GI tract   (Table p < 0.001) (Table p = 0.002)  showed a peripheral peritumoral lymphoid cuff ranging in thickness from 0.2 to 6 mm (mean 1.63 mm) Figure . Interes) Figure .In this first and largest series of GI tract schwannomas, we found that a peritumoral lymphoid cuff is frequently seen in GI tract schwannomas, and it correlates highly with the presence and size of regional LAD. However, it should be noted that in a significant subset (29.7%) a lymphoid cuff was not present in schwannomas of the GI tract.The differential diagnosis of subepithelial tumors of the GI tract includes GIST, leiomyoma, lymphoma, and schwannoma. Among these lesions, GIST is the most common tumor with the majority of them occurring in the stomach. Primary schwannomas of the GI tract are a rare subepithelial tumor also occurring most often in the stomach thus making it an important differential diagnosis for GISTs. They account for 0.2% of all gastric tumors and 4% of all benign gastric neoplasms . As bothet al., reported 30 cases of primary gastric schwannoma seen at a single institute over 8 years and found a peritumoral lymphoid cuff in 26 patients (86.7%) [Due to the rarity, there are relatively few case studies and literature reviews of primary GI tract schwannomas –21. Most (86.7%) . This inIn 2002, our institute reported 12 cases of GI schwannomas collected from 1995 to 2001 with regards to clinicopathological features and to the differential diagnosis with GIST . In the In conclusion, a peritumoral lymphoid cuff is frequently seen in GI tract schwannomas, however, in a significant subset, a lymphoid cuff was not present warranting continued need for caution in the preoperative radiologic and postoperative pathologic diagnoses.The Samsung Medical Center pathology database was queried for all resected gastrointestinal schwannomas from January 1998 to May 2016. Two pathologists (H.B and K.-M.K.) reviewed all cases. The pathologic diagnosis was defined as a well circumscribed lesion with an admixture of compact cellular spindled (Antoni A) and hypocellular (Antoni B) areas with collagen fiber deposition and positive expression for S-100 protein  and negative for c-kit  by immunohistochemistry. In cases suspicious for schwannoma but lacking a peritumoral lymphoid component, we also applied DOG1  by immunohistochemistry to completely exclude GIST. Finally, a total of 118 GI schwannoma cases were identified. Among them, 5 cases were initially misdiagnosed as c-kit negative GIST because of their histologic similarity and absence of lymphoid cuff within the tumor. Written informed consent was obtained from all patients.All resected tissues were fixed overnight in 10% buffered formalin and were embedded in paraffin. Hematoxylin and eosin-stained tissue sections (4 μm) of all cases were examined, and the characteristics of anatomic location, tumor border, nodularity, peritumoral lymphoid cuff, and regional LAD status were collected. Tumor size, the thickest area of peritumoral lymphoid cuff, and the length of the short axis of enlarged lymph nodes were also measured. For correlation, experienced radiologists (T.W.K. and D.C.) reviewed all the computed tomographic (CT) images and reports. Enlarged lymph nodes were defined as >4 mm of its length of short axis when measured in CT images in the picture archiving and communication system (PACS) workstation . When two or more preoperative CT scans were performed, the most recent scan was used for analysis. Also we reviewed intraoperative findings, and electronic medical records of all cases describing the presence of enlarged regional lymph nodes.p < 0.05 was considered to indicate a statistically significant difference.Statistical analysis was performed using SPSS version 23.0 software . Significant differences were evaluated using Fisher's exact test for categorical data. A


Mucocele of the appendix is a rare clinicopathological entity simulating acute appendicitis. The most common form of the mucocele is cystadenoma, which is characterized by luminal dilatation producing large amounts of mucin. We present a new case of a giant mucocele of appendix with mucinous cystadenoma. A 61-year-old female was admitted with complaints of severe lower right quadrant pain. Ultrasonography and computed tomography (CT) suggested that it was a mucocele, but due to severity of pain, she underwent an emergency operation. Fortunately, without a perforation, it was a giant mucocele and the operation was terminated with an uneventful appendectomy with segmental cecal resection. The histopathological evaluation of the specimen reported to be a mucocele with mucinous cystadenoma with negative surgical margins. The patient was discharged postoperative 6th day, and a control colonoscopy and abdominal CT was planned for 6 months following surgery. Appendicular mucocele is rare and difficult to diagnose preoperatively, and sometimes it may be of large size which increases the risk of perforation. Pseudomyxoma peritonei (PP) is the most feared complication of mucocele perforation. Appendectomy with negative margins is a requirement for adequate treatment for most cases. Utmost care should be taken during surgery to avoid perforation of mucocele.Sertkaya M, Emre A, Pircanoglu EM, Peker O, Cengiz E, Karaagaç M. Giant Appendicular Mucocele Due to Mucinous Cystadenoma. Euroasian J Hepato-Gastroenterol 2016;6(2):186-189. Appendicular mucocele is a rare pathological entity characterized by cystic dilatation of the appendix due to abnormal appendiceal mucinous secretion by benign or malign lesions. Retention cyst or simple mucocele, mucosal hyperplasia, cystadenoma, and cystadenocarcinoma are four main groups of mucoceles. Cystadenoma is the most common form and is characterized by tubular adenomatous epithelium with varying degree of epithelial atypia. It manifests itself in different clinical features, such as producing large amounts of mucin which could sometimes result in giant luminal dilatation that might perforate spontaneously or during surgery. Perforation of a mucocele can cause pseudomyxoma peritonei (PP) which is the most feared complication leading to a worse clinical condition. The most important point is preoperative accurate diagnosis and maximum care about perforation risk during surgery to avoid having to deal with PP. The most important diagnostic tools are ultrasonography and computed tomography (CT). Periodic follow-up after surgery should be emphasized due to possible recurrences. Here, we present a new case of a giant appendicular mucocele with mucinous cystadenoma.A 61-year-old female patient with past history of hypertension, diabetes mellitus, hyperlipidemia, and a coronary artery bypass surgery 8 years ago was admitted to the emergency department with complaints of sudden onset of a severe blunt pain in the right lower quadrant. She had no episodes of vomiting or nausea, but had a chronic constipation complaint. On her general and physical examination, she was moderately obese, had right lower quadrant tenderness, and guarding and rebound was positive. The blood tests revealed leukocytosis around 13.5 K/µL, an elevated C-reactive protein value around 124 mg/L, and an elevated glucose value around 340 mg/dL. Other biochemical values were at within normal limits. The abdominal ultrasonography detected a 113 × 44 × 55 mm-sized, hypoanechoic encapsulated lesion with distinct contours located in the right lower quadrant adjacent to anterior wall of the abdomen. A planned abdominal CT to evaluate the differential diagnosis and the presence of additional pathologies revealed a tubular-shaped cystic mass without any solid components, superiorly extending to anterior abdominal wall from the level of right ileocecal valve, about 14 × 5 cm in size, with a thin wall, having a density of about 20 HU . Due to 1 the term “mucosal” was first coined by Karl Freiherr von Rokitansky in 1842.3 It is an obstructive and cystic dilatation of the appendiceal lumen caused by intraluminal accumulation of mucoid material.23 Although recognized as a pathological condition by Rokitansky, it is claimed that the term “appendicular mucocele” was first defined and used by Feren in 1876.34 As the name suggests, it is a simple and understandable term which provides ease for surgeons and radiologists to report it before any exact histopathological diagnosis.1 The importance of easy recognition of mucocele is to plan an optimum surgical procedure and to avoid rupture as it could result in a worse entity called PP.3 Being with high mortality rate,4 this is the most feared complication of appendiceal mucocele which occurs due to perforation of mucocele by increased intraluminal pressure, spontaneously or during surgical manupulations.135 The term “PP” was first introduced by Werth in 18841 and signifies the presence of pools of mucin- and mucin-secreting cells within the peritoneal cavity. Other complications of mucocele are reported to be intestinal obstruction by intussusception or volvulus, gastointestinal bleeding, ureteral obstruction, and hematuria.35Being a morphological term that describes the transformation of the appendix into a bag full of mucus depending on several etiologies,7 It is reported that the rate of a correct preoperative diagnosis had been achieved in up to 30% of cases according to the published series.2 The incidence of mucocele is ranging from 0.07 to 0.7% of all appendectomy specimens,8 and it affects both sexes between 4th and 7th decades of life,126 with a female predominancy.7 However, there is still disagreement about gender distribution, and different studies have reported different results.23Despite its high risk of perforation and serious complications, due to its low incidence rate, mucocele can be difficult to diagnose preoperatively, and thus it is more likely to be diagnosed during surgery or after pathological evaluation.3 and often mimics acute appendicitis since the most common presenting symptom is right lower quadrant pain.1 Other symptoms of mucoceles encountered in symptomatic patients are a history of painless or painful mass sensation in the lower right quadrant, nausea and vomiting due to intestinal obstruction or intussusception, genitourinary complaints, anemia, weight loss, chronic intermittent colic-type pain, abdominal discomfort, and nonspecific complaints like change in bowel habits.5 Interestingly, almost 50% of cases are asymptomatic and usually diagnosed incidentally during surgery performed for other reasons, or during radiographic imaging.4 As far as we can understand, becoming symptomatic for mucoceles depends on the size and location of the lesion. In the present case, the diameter of mucocele and appendix together was 14 cm and led to severe lower abdominal pain that justified an emergency operation.Mucocele symptoms are nonspecific5 Ultrasound may show an encapsulated cystic lesion adjacent to cecum, but it alone may not be sufficient to make the diagnosis.3 We can say that CT is a more accurate diagnostic tool and can reveal more clearly a pericecal well-encapsulated cystic formation with thick or thin wall, sometimes accompanied with parietal calcification.3 In the present case, ultrasonography and CT suggested that it was a mucocele , appendiceal mucocele with hyperplastic epithelium and moderate luminal dilatation (5–25%), mucocele through mucinous cystadenoma (63–84%), and malignant mucocele through cystadenocarcinoma (11–20%).46 Being a noninvasive tumor, it produces large amounts of mucin with prominent luminal dilatation of up to 6 cm and has an association with perforation risk about 20%.46 The luminal diameter of the lesion we measured was about 5 cm, and it was about to perforate (Cystadenoma is characterized by tubular adenomatous epithelium, with varying degree of epithelial atypia, and morphologically resembles adenomas in the colon.erforate  and 3.4 The mentioned two lesions may occur de novo or from a simple mucocele preexisting in the appendix.4 In these two cases, complete and accurate excision of the appendix is considered curative surgery if histopathologically it can be proven that negative margins are assured, and lymph nodes are not invaded when evaluated perioperatively.34 If lymph nodes are invaded, a cecum resection or right colectomy is required for curative surgery.3 Perioperative assessment of the present case suggested that it was a benign lesion, and there was no sign of lymph node invasion (Cystadenoma and cystadenocarcinoma are the two neoplastic groups of appendiceal mucoceles which encompass about 35% of primary neoplasms of the appendix.invasion . So we dinvasion . For theinvasion . So it w7 For the first two groups which are also classified as nonneoplastic lesions, a simple appendectomy is an adequate treatment option. Due to the fact that the final diagnosis will not be possible before pathological evaluation of the removed lesion, all kinds of mucoceles perforation should be avoided during open or laparoscopic surgery due to the possible most feared complication, PP.8Accordingly, it is known in the literature that for all four groups of mucoceles surgery is the required treatment.45While postoperative prognosis of patients with benign mucoceles is reported to be excellent with 5-year survival rates of 91 to 100%, with malignant mucoceles, the 5-year survival rate is markedly reduced due to complications of PP.Because the mucocele of the appendix may be simultaneously accompanied by solid organ tumors of other locations and the later risk of PP, it is reasonable to explore thoroughly during operation and to follow-up patients periodically after surgery. In the present case, the patient was discharged planning a control colonoscopy and a whole abdominal CT to be performed 6 months after surgery.Mucocele of the appendix is a very rare entity, but often confused with acute appendicitis. The most important diagnostic tools are ultrasonography and CT. Preoperative diagnosis is very important to decide an appropriate surgical procedure, to make sure there is no any other pathologies, and to avoid perforation of the mucocele which can cause PP. Simple appendectomy with negative margins is generally the sufficient and required treatment, but depending on the size and location of the mucocele, sometimes cecum resection or right hemicolectomy may be required. It is important to follow-up periodically after surgery due to the risk of possible recurrences.


Among classes of ovarian tumor, granulosa cell tumors are the least common. In approximately 10% of cases of granulosa cell tumor, androgen will be secreted which will present with hirsutism and hyperandrogenemia. We describe a woman with ovarian granulosa cell tumor who presented with hirsutism.2. She had hair on her upper lip, chin, and lower abdomen; she had a Ferriman–Gallwey score of 10. A pelvic examination revealed that her uterus was of normal size and there was no adnexal mass. Ultrasound finding: her right ovary measured 5 × 4 cm. Her serum testosterone was 254 ng/dl; she was counseled to undergo an exploratory laparotomy but she declined. She presented to our out-patient department 10 months later with a complaint of excessive vaginal bleeding of 18 days’ duration. A sonographic evaluation showed a 12 by 15 cm right adnexal cystic mass. With preoperative diagnosis of testosterone-producing sex cord–stromal tumor of the ovary, an exploratory laparotomy was performed. The laparotomy revealed a 20 by 30 cm right ovarian mass with pathology result of adult granulosa cell tumor.A 50-year-old woman of Amhara ethnicity, para III, abortion I (induced), presented with excessive hair on her face and lower abdomen of 4 years’ duration which affected her quality of life. Her menopause started 7 years ago. Her body mass index was 29.8 kg/mIn postmenopausal women with new hirsutism that is severe or rapidly progressive, the possibility of an androgen-secreting tumor must be suspected and a thorough evaluation is needed before initiating treatment for idiopathic hirsutism. Among the major classes of ovarian tumors, sex cord–stromal tumors are the least common , 2. GranA 50-year-old woman of Amhara ethnicity, para III, abortion I (induced), presented to our out-patient department with the complaint of excessive hair on her face of 4 years’ duration. The hair growth was on her face and lower abdomen; it had gradually increased in amount despite frequent shaving. Her menopause started 7 years ago. She had no vaginal bleeding or discharge; she had no abdominal or pelvic pain. She did her daily chores normally as before but the hair growth over her face affected the quality of her personal life and work life.2. Her vital signs were in the normal range and there were no abnormal findings from tests of her chest and cardiovascular system. She had hair on her upper lip, chin, and lower abdomen with a modified Ferriman–Gallwey score of 10, which is mild hirsutism  of 29.8 kg/mHirsutism is defined as the presence of terminal coarse hairs in females in a male-like distribution and it affects approximately 5 to 10% of women . MoreoveSimilarly, sex cord–stromal ovarian neoplasms are rare; the overall incidence of these neoplasms is 0.2 per 100,000 women. Malignant sex cord–stromal neoplasms are often diagnosed at an early stage, at diagnosis 90% of them are stage I, like in our patient; our patient was staged surgically as International Federation of Gynecology and Obstetrics (FIGO) IC which indicates the tumor was confined to her right ovary with ruptured capsule, but no seeding into her peritoneum, omentum, or other intra-abdominal organs and ascetic fluid was free of malignant cells. However, since outcome is less favorable with stage I disease in which the tumor has ruptured, our patient took five cycles of BEP , 7, 8. AFor a young patient desiring fertility, unilateral conservative surgery is appropriate. However, for a 50-year-old woman, like our patient, a total abdominal hysterectomy with bilateral salpingo-oophorectomy may be preferred even if the mass seems benign and unilateral.One notable difference between sex cord–stromal neoplasm and other ovarian neoplasms is that lymph node metastases are rare so performing pelvic and para-aortic lymphadenectomy may not be necessary. Nodes should be palpated; lymphadenectomy is required for women with palpable nodal enlargement. Pelvic and para-aortic lymphadenectomy should also be performed in women in whom there is a suspicion of a different histologic type of ovarian malignancy . Our patIn postmenopausal women with new hirsutism that is severe or rapidly progressive, the possibility of an androgen-secreting tumor must be suspected and a thorough evaluation is needed before initiating treatment for idiopathic hirsutism.


Rectal gastrointestinal stromal tumours (GISTs) are uncommon tumours and usually present with large sizes. We present two cases of rectal GIST. Imatinib was used in the setting of neoadjuvant and adjuvant therapy. Both tumours were resected transanally by the transanal endoscopic operation (TEO) platform. Oncosurgeons are recommended to implement sphincter-sparing surgeries for these cases. Gastrointestinal stromal tumours (GISTs) are the commonest mesenchymal tumours (sarcoma) affecting the gastrointestinal tract. However, GISTs represent only less than one percent of the gut tumours .Rectal GISTs are even rarer, representing 3% of all GIST cases, while most cases are detected in the stomach and small intestine (90%) .The discovery of the c-Kit tyrosine kinase receptors (75–80% of cases) and the platelet-derived growth factor receptor α (PDGFRA) mutation (10–15% of cases) led to the use of Imatinib mesylate therapy. Imatinib is a tyrosine kinase inhibitor that inhibits proliferation and induces apoptosis in GIST cells [However, surgery remains a cornerstone in the treatment and the only curative option for those with rectal GISTs. Several surgical options are available. In spite of this, large tumours with aggressive features have been commonly treated with abdominoperineal resection .The use of transanal minimally invasive surgery (TAMIS) technique for the resection of GISTs in the rectum is documented in several studies ,4, but tA male patient aged 36 years old sought medical advice for constipation and a sense of incomplete defecation. MRI pelvis was done, which revealed a mass 8 × 3 cm at the ano-rectum. Fine-needle aspiration biopsy (FNAB) revealed a cellular spindle cell tumour, which was assigned as GIST by immunostain. Clinical oncologists started Imatinib (Gleevec®) at 400 mg daily dose for three months with good radiological response, the lesion downsized to 1.5 cm in maximum diameter . AnotherA 55-year-old female patient presented to the internal medicine department with constipation and heaviness in the pelvis. Abdominal examination was free, per-rectal examination revealed a mass, 4 cm from the anal verge occupying anterior and left wall with intact overlying mucosa. Per-vaginal examination showed a palpable mass pushing the posterior vaginal wall with intact vaginal mucosa. Pelvic ultrasound showed a 5.9 × 5.6 cm mass at recto-vaginal pouch with mass effect upon the rectal lumen. Pelvic MRI reported intraluminal mass in the rectum 4.5 cm, infiltrating the left lateral rectal wall . EndorecFour different approaches of local resection of GISTs have been described in the literature. The transanal (Parks) and the transsacral (Kraske) approaches can achieve a complete resection in low- and mid-rectal lesions with low morbidity and mortality but usually require fragmentation of the specimen which increases the chance of local recurrence .Resection of the anterior wall rectal lesions is technically difficult. Transvaginal excision was described for lesions higher on the anterior wall of the rectum, with the advantage of avoiding anal dysfunction . AnotherMore recently, transanal minimally invasive endoscopic excision of rectal GISTs has been applied as a fifth approach. This includes TEM  and TAMIS commonly using either GelPOINT path transanal access platform  or SILS  . In our 2 insufflation to 14 mmHg. Subsequently, we mark the excision margins by a monopolar diathermy 1 cm around the visible border of the tumour. Excision of the lesion is done by an advanced bipolar or an ultrasonic shear. Finally, we close the defect with sutures. We think the TEO platform is both feasible (comparable to TAMIS) and relatively cheap, because it is reusable (comparable to TEM). The main advantage of this technique over Kraske or Park’s technique is the delivery of an intact specimen.In our technique, the operative field must be in a position corresponding to six o’clock of the rectoscope. The operation starts by washing the anal canal by diluted povidone-iodine and saline in order to remove residual faeces and mucous secretions. Then, gentle dilatation of the anal sphincters for a few minutes to allow insertion of the TEO platform without damage to the internal anal sphincter, followed by CORectal and anal GISTs are rare and thought to be of worse prognosis. Limited evidence from case reports and small case series suggests that neoadjuvant imatinib therapy can successfully downsize tumours and aids organ-preserving surgery in patients with high-risk rectal GISTs . One of Sphincter-sparing procedures for rectal GISTs using minimally invasive techniques should be the treatment of choice whenever possible. The TEO platform is an adequate method for achieving this goal. Imatinib as a neoadjuvant therapy for rectal GIST is a promising option, which increases the chance of tumour downstaging and thus anal sphincter preservation.The authors declare no conflict of interest.


Dear Editor,We report the case of a 63-year-old black female who presented with complaints ofdifficulty in urinating and pollakiuria. She also reported an eight-month history ofepisodes of dysuria and hematuria. She had smoked for 10 years and had quit 30 yearsprior. She had three pregnancies, all with vaginal delivery, and had undergone totalhysterectomy 17 years prior.Magnetic resonance imaging (MRI) of the pelvis revealed, below the urinary bladder, acystic formation involving the urethra, consistent with urethral diverticulum (UD),within which there was a solid component showing paramagnetic contrast enhancement,suggesting an expansive process. Communication with the urethra was well defined after aurethral catheter had been inserted. The diverticulum was surgically resected. On thebasis of histological and immunohistochemical studies of the surgical sample, thepatient was diagnosed with papillary urothelial diverticular carcinoma.-8. Some studies have reported that theincidence of UD is higher in black individuals,4,7. Typically, UD is underdiagnosed because, in most cases, theclinical profile is nonspecific,4-6and up to 20% of patients are asymptomatic,8. The site most oftenaffected is the middle third of the urethra, where the paraurethral glands (Skene'sglands) are typically located, and 96% of diverticular orifices areposterolateral-5,8. Most patients with UD have the acquired form, which probably arisesfrom dilation/abscess in paraurethral glands. Other causes include trauma andsurgery-5,8. Typicallymeasuring 0.2-1.6 cm, UDs can besingle or multiple, simple or multiloculated, and locally restricted or surrounding theurethra (in a "horseshoe" shape), with one or more (narrow or broad) orifices,2. Differential diagnoses include cervical cysts, vaginal cysts,abscesses, tumors, urethral endometriosis, and ectopic ureterocele,4,5.The reported prevalence of UD is 0.6-6.0%, and the condition is most common in womenbetween 30 and 60 years of age-8. The chronic inflammation and urinary stasis seen in UDs result incomplications,4, including calculi (in 1.5-10%) and malignanttumors-8. As for the tumors, UDs are responsible for less than5% of all urethral neoplasms,8, fewer than 200 cases having beenreported,2,7,8. Malignancies in UD include adenocarcinoma, in 49-61%of cases, transitional cell carcinoma, in 27-30%, and squamous cell carcinoma, in10-12%-5.Clinical findings include the classic triad of dysuria (in 30-70% of cases), dyspareunia(in 10-25%) and postmicturition dribble (in 10-30%), as well as pollakiuria or urinaryurgency (in 40-100%), urinary incontinence (in 32-60%), recurrent urinary tractinfections (in 30-50%), hematuria (10-25%), and bulging in the anterior vaginal wall (in35%), accompanied by purulent urethral discharge on palpation (in 12%),4,5; double-balloon urethrography-a methodwith an accuracy of 90%, showing findings similar to those cystourethrography, with thedisadvantage of being invasive and complex,4,5; ultrasound-a method with excellent accuracy (near 100%) whenintraurethral (highly invasive) or translabial/transperineal (less invasive) thatcharacterizes the cystic formations and any vascularized solid content, althoughexaminerdependent and limited in the evaluation of collapsed UDs,4,5; computed tomography-a method that isuseful in identifying calculi and tumors (evident solid components), albeit with lowsensitivity for small UDs,4,5; and MRI-the method of choice, with near 100% sensitivity, which isnoninvasive, with excellent contrast between tissues and discrimination of thecomplexity of the structures, capable of detecting small UDs and identifyingneoplasms,2,4-6,8. In T-2weighted MRI sequences, UDs show hyperintense signals, although they can be hypointenseif they have thick content,2,4,6. Solid tumor components present asvegetative lesions with intermediate signals on T1- and T2-weighted sequences,potentially restricting the diffusion, and show significant enhancement afterintravenous administration of contrast,2.Diagnostic imaging methods include the following: voiding cystourethrography-atechnically simple method, with an accuracy of 85%, that demonstrates the diverticulumthrough contrast and identification of filling gaps suggestive of calculi or tumors,although it uses an iodized agent might not detect UDs with small orifices


Dear Editor,A 43-year-old female patient with no known diseases sought medical attention complainingof increased abdominal volume. The patient underwent ultrasound and subsequent magneticresonance imaging (MRI) of the pelvis ,which s-3. MCTs are typically asymptomatic and slow-growing,3. They are usually seen in women of reproductive age and are rarelydiagnosed before puberty. Its growth ceases at menopause-7. An MCTtypically contains well-differentiated tissues of the three germ layers,5: the ectoderm, ; the mesoderm(osteomuscular and adipose tissues); and the endoderm (ciliated and mucinousepithelium). The diversity of tissues in teratomas results in a wide variety ofcharacteristics in imaging studies.Also known as a dermoid cyst, MCT is the most common benign ovarian tumor, accounting for10-25% of cases in adult patients and 50% of those in pediatric patients-12. However, MCTs often present typical imagingfeatures, which facilitate the diagnosis. Among such features, one of the most common isthat of a fatty tumor. In suchcases, the most common ultrasound finding is that of a cystic mass with an echogenictubercle (a Rokitansky nodule), presenting posterior acoustic shadowing secondary tocalcifications, strands of hair, or foci of fat,5,7.In most cases, pelvic tumors do not present imaging features that are considereddiagnostic,5,7. In rare cases,the presentation of MCT is atypical, which can be a diagnostic challenge forradiologists,6. Multiple small floating spheres within a large cyst,as observed in the case presented here, is one of those rare presentations, known as the“floating ball” presentation,6. Histologically, the spheres arecomposed of keratin, fibrin, hemosiderin, sebaceous debris, hair, and fat, in variableproportions,6,13. Although themechanism of formation of these spheres has yet to be clarified, it is speculated thatit involves aggregation of sebaceous material around a nidus,4,14. The mobility of the spheres is due to their lowdensity relative to the other content of the cyst,4,6. A finding of multiple floating spheres within a single largecyst has not been reported for other types of tumors and is therefore consideredpathognomonic of MCT,6,14-16.Characteristic findings on computed tomography include areas of fat attenuation, with orwithout foci of calcification. On MRI, the fat seen within the lesion produces ahyperintense signal on T1-weighted images and signal loss in fat-saturatedsequences


The aim of this study is to report a case of cystoid macular oedema (CME) associated with Rosai–Dorfman Disease (RDD).A 52-year-old male initially presented with a two-month history of a congested left eye. At presentation, visual acuity was 20/20 in the right eye and 20/80 in the left eye. Biomicroscopy showed conjunctival hyperaemia in the left eye with a slight elevation, suggesting a subconjunctival mass. Retinal fluorescein angiography and optical coherence tomography (OCT) revealed the presence of CME in the left eye. A clinical examination revealed nodular lesions in the ears and a lump in the subcutaneous tissue of the left arm. A biopsy of the subcutaneous lesion showed histological and immunohistochemical characteristics of RDD. The patient was treated with intravitreal triamcinolone (0.1 mL/4 mg). One month after treatment, there was complete regression of the oedema with a significant improvement in visual acuity to 20/20.This is the first reported case of RDD associated with cystoid macular oedema. Macular oedema responded to intravitreal treatment with triamcinolone. Rosai–Dorfman disease (RDD), or sinus histiocytosis with massive lymphadenopathy, was first described by Destombes  in 1965.In the following report, we describe the first case of cystoid macular oedema (CME) associated with RDD.A 52-year-old male initially presented with a two-month history of congestion of the left eye.At presentation, he underwent a complete ophthalmic examination. Visual acuity was 20/20 in the right eye and 20/80 in the left eye. Biomicroscopy showed conjunctival hyperaemia in the left eye with a slight elevation, suggesting a subconjunctival mass Fig. , but no A biopsy of the subcutaneous lesion showed a cellular infiltrate in the superficial and deep dermis with histiocytes with phagocytized lymphocytes and plasma cells Fig. . A charaDue to the low visual acuity secondary to CME, the patient was treated intravitreally with triamcinolone (4 mg/0.1 mL). One month later, the patient showed significant improvement in visual acuity (20/20) and OCT showed complete regression of the oedema Fig. . The folRDD (sinus histiocytosis with massive lymphadenopathy) often presents as a painless bilateral cervical lymphadenopathy with systemic symptoms. Consists of a rare disease, non-hereditary, benign histiocytic proliferative disorder. The Extranodal impairment have been reported in 28–43% of cases with rare ocular involvement [The characteristic findings of RDD include emperipolesis, positive histiocyte staining for S100 and CD68, and negative staining for CD1a. There is no standardized protocol available for treatment of ocular RDD because of its rarity and the possibility of its spontaneous regression , 5. ExciExtradonal impairment has been reported in 28–43% of cases, with locations including the skin, upper respiratory tract, salivary glands, epidural space, bone, and ocular adnexa. Ocular manifestation is rare. In the largest series of 243 cases as described by Foucar in 1990, the eye was involved in 36 cases 8.5%) and was highly associated with nasal sinus involvement. Infiltration of the soft tissues of the orbit and eyelids was the most common manifestation of ocular involvement % and was. In addiUveitis was present in three isolated cases of anterior uveitis  involving one patient with coexistent corneal infiltration , one patTo the best of our knowledge, this is the first report of macular oedema associated with RDD. The patient also presented with a subconjunctival mass and dermatologic abnormalities in the arm and ear. There was a prompt clinical response to intravitreal treatment with triamcinolone, suggesting a probable inflammatory cause of the macular oedema as previously reported in other forms of ocular involvement, such as chronic uveitis and uveitic glaucoma , 17.The short follow-up time of 6 months was a limitation of this study, particularly with regard to drawing conclusions regarding the efficacy of long-term therapy.In summary, RDD can be associated with CME; therefore, RDD patients should undergo routine eye examinations.


Biphasic malignant pleural mesothelioma is a rare malignant tumor, usually presenting as a pleural-based mass in a patient with history of chronic asbestos exposure. We herein report a case of a 41-year-old man who presented with chest pain and had a chest computed tomography (CT) scan suggestive of a primary skeletal tumor originating from the ribs (chondrosarcoma or osteosarcoma), with no history of asbestos exposure. CT-guided core needle biopsies were diagnosed as malignant sarcomatoid mesothelioma. Surgical resection and chest wall reconstruction were performed, confirming the diagnosis and revealing a secondary histologic component (epithelioid), supporting the diagnosis of biphasic malignant mesothelioma. Mesothelioma is an uncommon and highly aggressive malignant tumor  usually A 41-year-old Caucasian man presented to our institution with left lateral chest pain for approximately one year. He was previously evaluated at an outside institution where he underwent an unrevealing cardiac workup for his chest pain, including cardiac enzyme testing, a treadmill stress test, and echocardiogram. This was followed by a chest CT scan that showed a destructive lesion involving the posterior-lateral aspect of the left seventh and eighth ribs. He had no history of chest wall trauma or injuries. He had no cough, dyspnea, fever, chills, or night sweats. However, he reported a five-pound unintentional weight loss over the last three months. His past medical history was unremarkable, with no history of surgeries, cigarette smoking, or tobacco use and no occupational or environmental exposures. Family history was negative for chronic diseases or malignancies. Physical examination revealed a tender mass along the left lateral chest wall, corresponding to the area where the destructive rib lesion was seen on the previous CT scan.A repeated chest CT, with additional imaging of the abdomen and pelvis, demonstrated an 8.5 × 8.2 × 3.0 cm destructive tumor involving the posterior-lateral aspect of the left seventh, eighth, and ninth ribs , which aA CT-guided core needle biopsy of the rib lesion and adjacent soft tissue was performed . It reveA positron emission tomography- (PET-) CT scan was performed for staging and showed FDG uptake within the destructive chest wall lesion with standardized uptake value (SUV) of 17, an additional area of FDG uptake was also seen on the left side of the twelfth thoracic vertebra (T12) with SUV of 4 .Treatment with combination pemetrexed-cisplatin chemotherapy was initiated, with a total of 4 cycles. A follow-up chest CT showed a stable 8 cm rib-pleural mass. After presenting and discussing the case at a multidisciplinary thoracic oncology conference, consensus was in favor of surgical resection with chest wall reconstruction.One month later, the patient underwent a left thoracotomy procedure with tumor resection, left upper and lower lobe pulmonary wedge resections, and chest wall reconstruction with titanium mesh plate and latissimus dorsi flap. Surgical gross pathology showed a large, rib-based 8 cm tumor and two lung parenchymal wedge biopsies with white-tan pleural-based nodules. Interestingly, while the tumor revealed histological features similar to the previous biopsy, the lung specimens showed involvement by a mesothelioma with epithelioid features Figures  and 6. IAfter allowing time for the surgical recovery and wound healing, the patient was referred to radiation oncology, where he received left chest wall radiation for positive resection margins. Follow-up CT imaging has not shown recurrence after ten months of follow-up .MPM is a highly aggressive cancer typically associated with asbestos exposure –8. PatieBased on the postmortem analysis of 172 patients with MPM, most commonly involved distant organs include the liver (55.9%), adrenal glands (31.3%), kidneys (30.1%), and contralateral lung (26.8%) . ExceediThis case of biphasic MPM is unique and challenging, as it affected a young patient, with no history asbestos exposure, and exhibited an unusual presentation, suggesting a primary skeletal malignancy. Furthermore, it succinctly highlights limitations and possible clinical pitfalls in the diagnosis of MPM. As with other tumors, the larger the lesion is, the less representative a core biopsy or fine needle aspiration (FNA) may be. In addition, if a tumor is poorly differentiated , loss of phenotypic marker expression is not uncommon , 17. KaoMPM is highly resistant to therapy and the preferred treatments are surgery (at early stages) and/or combination treatments with radiotherapy and chemotherapy . ConventWhen evaluating suspected skeletal tumors based on thoracic imaging, variants of malignant pleural mesotheliomas must always be considered in the differential diagnosis of a chest wall lesion, even in relatively young individuals with no history of asbestos exposure. The identification of nonepithelioid subtypes of malignant pleural mesotheliomas may require extensive workup, exhausting the limited material in core/FNA biopsies. In addition, they may not be entirely representative of all the histologic components present in the tumor. Nonetheless, these procedures remain invaluable as the first step in guiding further diagnostic and therapeutic interventions.


Anal adenocarcinoma is very rare and usually occurs in the elderly. We present a case of a 12-year-old girl with an anal margin painful tumor infiltrating the lower rectum, with perineal and vulvar permeation nodules and bilateral fixed inguinal and iliac lymph nodes. Histology showed anal adenocarcinoma with mucosecreting component and independent cells. She had no extra pelvic metastasis on CT scan. She underwent a colostomy and palliative care. This exceptional case challenges us on the diversity of forms of anal cancers that require a multidisciplinary approach. The precarious social context and the age of onset make it difficult to manage this rare cancer. Anal cancers are rare. They represent 1.5 to 2.5% of digestive tract cancers . Most arA 12-year-old girl, whose parents are separated, was sent to us in May 2017 for painful swelling and pruritus of the anal margin. Eight months before the first signs, a notion of multiple anal intercourses is reported. Because of the notion of anal intercourses, she was examined in several hospital and social departments without successful outcomes.She presented a large circumferential tumor, budding, ulcerated, and painful tumor of the anal margin, with perineal and vulvar permeation nodules and bilateral fixed inguinal and iliac lymph nodes .Histology showed anal adenocarcinoma with mucosecreting component and independent cells . ImmunohFor our 12-year-old girl who presents an ADK of the anus classified cT4N3M0, we realized a colostomy in gun barrel. Anemia was corrected after transfusion of globular concentrate. Chemotherapy could not be administered because of lack of financial resources. Evolution without anticancer treatment was a locoregional tumor progression. The patient is under tramadol to calm the sometimes intense perineal pain.Anal adenocarcinoma occurrence in a 12-year-old child is extremely rare. The average age is 65 years or older . The oriThis exceptional case challenges us on the diversity of forms of anal cancers that require a multidisciplinary approach. The precarious social context and the age of onset make it difficult to manage.


Dear Editor,A newborn male patient presented with suspected obstetric trauma due to increased forearmdiameter. An X-ray of the forearm showed fAn incisional biopsy was performed. Histological and immunohistochemical analysis of thebiopsy specimen demonstrated positivity for vimentin alpha-actin and for S-100 protein,together with negativity for desmin. On the basis of those findings, the definitivediagnosis of fibrous hamartoma of infancy (FHI) was made., FHI was firstdescribed in 1956 by Reye, whodubbed it subdermal fibromatous tumor of infancy. There have been fewer than 200 casesreported to date, only 8 having been reported in the literature of Latin America; 91% ofall cases occurred in the first year of life, 25% having been diagnosed atbirth.A benign soft-tissue tumor that typically occurs in the first two years oflife,5. Theidentification of fat within the lesion helps narrow down the differential diagnosis, asdo patient age and form of presentation. In the appropriate clinical context, a findingof fibrous tissue trabeculae interspersed with fat in an organized pattern is stronglysuggestive of FHI.The differential diagnosis of FHI includes all other soft-tissue tumors. When the tumoris hard and fixed to the deep planes, it is important not to confuse FHI with malignantneoplasms such as juvenile fibromatosis and sarcoma . Neural tumors (mainlyneurofibromas) and vascular tumors should also be excluded. Larger lesions typically involveneurovascular structures. Although the tumors are infiltrative, with ill-defined bordersand no capsule, the typically do not invade the surrounding bone structures.The occurrence of FHI is not related to syndromes or a positive family history.Reportedly, FHI is painless and its growth is unpredictable. It can grow rapidly in earlychildhood, its rate of growth slowing after the child has reached five years of age.There have been no reports of spontaneous involution or malignancy. Together with thebands of connective tissue, an FHI presents nests of primitive mesenchyma represented bysmall, rounded, immature cells, without areas of atypia but occasionally with mitoses,immersed in a myxoid matrix, possibly constituting an anomalous process of tissuematuration.Histopathological examination of an FHI shows compounds of mature adipose tissueinterspersed with bands of dense fibrous tissue rich in myofibroblasts andcollagen, no adjunctive therapies being required. Recurrenceafter complete resection is uncommon, having been reported in only approximately 10% ofcases.The preferred treatment for FHI is complete local resection


Mucinous cystadenomas (MCAs) of the liver  are rare tumors that comprise about 5% of cystic masses of the liver in adults. These slow-growing lesions most commonly occur in middle-aged individuals, with a female sex predominance. Herein, we present a MCA in a 6-year-old male, one of only very few such cases described in the pediatric literature to date. Although MCAs are generally considered benign lesions, malignant transformation rarely occurs. The recurrence rate is high when partial cyst excision is performed. Therefore, complete surgical cyst resection with clinical follow-up, including imaging, is warranted. Mucinous cystadenomas (MCAs) of the liver are rare intrahepatic lesions seldom discovered in childhood. These lesions are typically found incidentally, often during evaluation of non-specific abdominal complaints. Like other more widely described hepatic masses, MCAs can cause symptoms such as abdominal pain, nausea, vomiting, and jaundice from mass effect. These “great pretenders” are easily mistaken for abscesses, cysts, or even malignancy, which prompts diagnostic resections. Characteristically, MCAs are considered benign, although secondary malignant transformation has occurred in as many as 20% of affected adults . DefinitHerein, we report a case of a 6-year-old healthy male diagnosed with MCA after a liver mass was incidentally discovered during an echocardiogram. Through this report, we hope to educate gastroenterologists and surgeons about a rare mass that they may encounter more frequently in practice as abdominal imaging modalities improve. We will discuss the role of imaging and histology in the diagnosis of MCAs, definitive management, and propose a standard for routine surveillance in affected patients.A 6-year-old previously healthy male presented to the Pediatric Gastroenterology Clinic for evaluation of a liver mass that was incidentally seen on an echocardiogram while assessing a heart murmur. At the time of the discovery of the liver lesion, the patient was asymptomatic, without abdominal pain, anorexia, weight loss, vomiting, jaundice, or other liver-related complaints. Vaccinations, including hepatitis B, were up to date.Findings on physical examination were normal. Results of serum blood chemistry including markers of liver synthetic function , transaminases, bilirubin, alpha-fetoprotein (AFP), cancer antigen (CA19-9), and carcinogen embryonic antigen (CEA) were normal. HAV, HBV, and HCV serologies were also negative. An abdominal ultrasound showed a cystic lesion with irregular borders in the left hepatic lobe. The hepatic mass was characterized by computerized tomography (CT) as a circumscribed, complex, multi-lobulated, and septated cystic lesion measuring 5.5 cm × 4.2 cm × 5.2 cm.Abdominal magnetic resonance imaging (MRI) done 7 months later showed the same complex cystic lesion in the left hepatic lobe without interval change in size. Given the absence of symptoms and benign laboratory and imaging results, we pursued a conservative approach with clinical, biochemical, and radiological follow-up every 6 months. Results of the patient’s laboratory tests remained normal, but the lesion showed slight growth on ultrasound over the following 2.5 years. A repeat abdominal MRI (with Eovist contrast) showed the complex multi-cystic lesion had increased slightly in size to 6.1 cm × 4.1 cm × 6.4 cm , palpable abdominal mass up to 53%), nausea, vomiting and rarely, jaundice 0–90%, pa, 11. In %, nauseaOn imaging, lesions may be unilocular or multilocular and resemble abscesses, echinococcal cysts, teratomas, hematomas, necrotic tumors, and polycystic disease , 11, 13.Histologically, MCAs are usually lined with cuboidal or non-ciliated columnar cells, with basal nuclei and mucin-containing cytoplasm , 4, 13. Because it is difficult to diagnose MCAs by imaging, the gold standard for treating these lesions is complete surgical resection with confirmed clear margins , 5, 9. OPercutaneous biopsy and analysis of cystic contents have been proposed as a less invasive diagnostic option , 7, 8. FIn summary, given the lack of consistently reliable non-invasive diagnostic methods and high tumor recurrence rate with partial resection, complete surgical excision is considered the gold standard. Even with complete removal, the recurrence rate is 5–10% in adults (Written informed consent for the presentation and publication of this case was obtained from the patient’s legal guardian.DF and MM drafted the manuscript. IZ edited the manuscript, and obtained and interpreted the gross pathology images. DH-G edited the manuscript and obtained and interpreted the microscopic pathology images. RG-P critically reviewed the manuscript and is the author guarantor.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. The reviewer NK and handling editor declared their shared affiliation.


Soft tissue metastases are an uncommon presenting feature for primary solid tumours. This case highlights a rare presentation in which a soft tissue mass is the first clinical manifestation of a widespread disseminated malignancy of the esophagus. A 73-year-old woman presented with a soft swelling in the left upper quadrant of the abdomen arising from the anterior abdominal wall, suspicious of liposarcoma. Core biopsies revealed squamous carcinoma. Immunohistochemistry suggested the most likely diagnosis was that of metastatic carcinoma with a number of potential primary sites. Computed tomography scanning showed widespread metastatic disease, including lung, liver, kidney, omentum, subcutaneous and intramuscular lesions. The distal esophagus was noted to be circumferentially thickened. Finally, upper gastrointestinal endoscopy revealed carcinoma of the esophagus. The patient remains well awaiting esophageal stenting and palliative chemotherapy. In conclusion, it is important to be able to distinguish the origin of a soft-tissue swelling as the management will depend significantly on the histological type. Soft-tissue metastases are rarely encountered as a presenting sign of an occult cancer. Primary cancers that most commonly metastasise to soft tissues include those arising within the lung, colon and kidney. The most frequent histological diagnosis is adenocarcinoma. This case demonstrates the utility of biopsy in the investigation of soft tissue masses when the clinical presentation is unusual. Soft tissue metastases are an uncommon presenting feature for primary solid tumours -3 and reWe present a unique case of an elderly woman who presented with a short history of a left-sided abdominal wall swelling. Further investigation with a series of imaging studies, tissue biopsies and endoscopy revealed widespread visceral and soft tissue metastases arising from a squamous cell carcinoma of the esophagus. This case highlights a rare presentation in which a soft tissue mass is the first clinical manifestation of a widespread disseminated malignancy of the esophagus: an aggressive cancer which has a poor prognosis owing to its late presentation.A 73-year-old woman was referred to a ‘Sarcoma’ clinic suspected of having a soft-tissue malignancy. The woman had noticed a slow-growing lump on the left side of her abdomen, which had been present for 2 months. The abdominal lump was her primary complaint and she had no significant past medical history. On examination she was noted to have a soft swelling in the left upper quadrant of the abdomen, which felt as if it was arising from the anterior abdominal wall.An ultrasound scan of the abdomen wall demonstrated a complex mass lying within the fatty layer. The lesion extended to 3.5 cm in diameter with a central lobular, heterogenous portion surrounded by fat, which demonstrated marked internal vascularity on Doppler study. This was suspicious of liposarcoma, and 3 core biopsies of the lump were taken which revealed extensively necrotic moderately to poorly differentiated carcinoma. There were both glandular and focal squamous differentiation histologically with keratinisation. The CT scan showed widespread metastatic disease. Beginning cranially, within the lungs there were multiple rounded nodules of varying size, the largest measuring 23 mm, consistent with metastases. In the right superior mediastinum there was an enlarged node measuring over 2 cm. This node was separated from the right lobe of the thyroid by the jugular vein but posteriorly shown to be inseparable from the upper thoracic esophagus. The distal esophagus and gastro-esophageal junction was noted to be abnormally circumferentially thickened and irregular; however, there was no proximal dilatation of the esophagus. There were further metastatic deposits in the liver, kidneys and omentum. A number of subcutaneous and intramuscular lesions were identified throughout the thorax and abdomen, but no lytic bone lesions. The original left abdominal mass was identified as originating from the anterior abdominal wall and extending into the subcutaneous tissue: An upper GI endoscopy revealed an extensive, friable and bleeding carcinoma of the esophagus at between 25 cm and 31 cm. Biopsies of the lesion revealed squamous cell carcinoma. The biopsies of the abdominal wall and the esophageal lesion were compared and confirmed as showing identical pattern.The patient was subsequently referred to the upper GI cancer team and at the time of writing, despite the substantial tumour burden, remains well awaiting esophageal stenting and palliative chemotherapy.It is important to be able to distinguish the origin of a soft-tissue swelling as the management will depend significantly on the histological type. Although not in themselves infrequent, soft-tissue metastases are rarely encountered as a presenting sign of an occult cancer as by the time these appear, the patient has commonly developed symptoms pertaining to the primary tumour. Primary cancers that most commonly metastasise to soft tissues include those arising within the lung, colon and kidney -7. FurthThe prognosis and treatment of esophageal cancer is dictated by the extent of local invasion, but more importantly by both nodal and metastatic spread. Early detection with surgical resection and appropriate adjuvant therapy confers the best chance of survival; however, by the time most patients who present this may not be feasible. Palliative techniques such as esophageal stenting with self-expanding metallic stents (SEMS) may provide symptomatic relief from dysphagia. Initial staging of esophageal cancer involves CT scanning and endoscopic ultrasound, although PET scans have been suggested to be more useful in detecting soft-tissue metastases .There are few reported cases of metastatic spread of esophageal cancer to soft-tissues and all were known cases of primary cancer. As far as the authors are aware, there are no reported cases of soft-tissue metastases presenting as the first clinical sign of an underlying squamous cell carcinoma of the esophagus.In conclusion, it is all too easy to attribute superficial soft tissue swellings to common conditions and plan excision based on the likeliest diagnosis. As shown in the case described, there is no substitute for thorough clinical questioning and examination, and any disparity between the clinical presentation and examination findings should prompt additional investigation before further management can be appropriately planned. This case also demonstrates the utility of biopsy in the investigation of soft tissue masses when the clinical presentation is unusual.


Primary appendiceal adenocarcinoma is a rare tumor, mucinous variety being common. This case is reported to highlight the unusual presentation and diagnostic difficulty of appendiceal adenocarcinoma.Patient presented with acute appendicitis with ill-defined tender lump which responded to conservative management.High index of suspicion should be kept in mind for elderly patients presenting with appendicular lump. Every effort should be made during elective appendectomy to remove stump in case of sloughed out appendix. Primary appendiceal adenocarcinoma is a rare entity frequently discovered by pathologist after appendectomy. Patients commonly present as acute appendicitis or may present as a palpable mass in the right iliac fossa . Primary3. The rest of the investigations were normal.A 55-year-old female presented with recurrent abdominal pain in the peri-umbilical region and right iliac fossa of 6-month duration which had increased since 5 days, associated with one episode of non-bilious vomiting. There was no history of fever, altered bowel or bladder habit or loss of appetite. On examination, patient was febrile with pulse of 104/min. Systemic examination was normal. Abdominal examination revealed an ill-defined tender lump in the right iliac fossa with no guarding and rigidity. Rectal examination was unremarkable. Investigations revealed white blood cell (WBC) count of 13,800/mmUltrasonography of the abdomen showed thickened loops of ileum and caecal wall suggestive of inflammatory etiology. In view of ill-defined lump, possibility of appendicular abscess or ileo-caecal Koch’s was considered. Erythrocyte sedimentation rate (ESR) was 22 mm (0–20 at the end of 1 h), and serum adenosine deaminase (ADA) level was 48.8 IU/L (0–40 IU/L) which were within normal limits.Computerized tomography (CT) scan of abdomen Fig.  suggeste4N0M0 (Modified Astler Coller B3). Multidrug adjuvant chemotherapy of 5-fluorouracil, leucovorin, and oxaliplatin (FOLFOX) was given. Radiotherapy has been given for 15 cycles. Patient was asymptomatic, and CEA levels at the end of 12 months were normal. CT scan at the end of 12 months revealed no evidence of recurrence.Post appendectomy, CT abdomen did not show any mass or evidence of metastasis. Serum carcino embryonic antigen (CEA) levels were 2.02 ng/ml (0–5 ng/ml). Right radical hemicolectomy with regional lymphadenectomy was performed after 2 weeks of appendectomy after histopathology report was available. Postoperative course was uneventful. Histopathology revealed moderately differentiated mucin secreting adenocarcinoma. Resection margins were negative, and all 12 lymph nodes resected from the specimen were negative for tumor. The tumor was staged as TThe emphasis of the case report is on highlighting the difficulty in preoperative diagnosis of appendicular carcinoma in patients presenting with appendicular lump and sepsis. In elderly patients presenting with appendicular lump, the differential diagnosis of malignancy should be kept in mind. During interval appendectomy, even if sloughed off appendix, specimen should be sent for histopathology. Conformation of carcinoma even if rare would save patient from dissemination of malignancy due to violation of planes at the time of surgery and better adherence to oncologic principles during the single-stage procedure as opposed to a two staged one.The tumors of the appendix are of three histological types: carcinoid tumors (88%), cystic adenocarcinoma (8%), and primary adenocarcinoma 4%) . Primary% . PrimaMalignancies of appendix are rarely diagnosed preoperatively or intraoperatively. Most tumors are identified only after histopathological examination of resected specimen following appendectomy or as an incidental finding during exploratory laparotomy for other surgeries . While sIn cases of adenocarcinoma of appendix with intra-abdominal metastasis, the treatment consists of aggressive debulking followed by adjuvant chemo-radiotherapy –11. The


Sarcomatoid hepatocellular carcinoma (SHC) is a rare malignant hepatic tumor. Recurrent interventional therapies such as transcatheter arterial chemo-embolization (TACE), radiofrequency ablation (RFA), and percutaneous ethanol injection have been reported previously utilized in a majority of SHC cases. To date, the exact pathogenic mechanisms underlying sarcomatoid transformation of hepatocellular carcinoma (HCC) remain unknown.In this study, we report a 68-year-old female SHC patient admitted to our hospital due to discrete abdominal distention for more than 20 days. Abdominal computed tomography (CT) with tri-phase enhancement revealed portal vein tumor thrombi (PVTT) and a left hepatic lobe lesion measuring 110.0 mm × 160.0 mm. The patient subsequently underwent liver resection, after which pathological examination revealed proliferation of spindle-shaped SHC cells. A sarcomatoid, T4 stage carcinoma was eventually diagnosed. Forty-seven days after the operation, tri-phase enhanced CT detected extensive lesions in the liver, spleen, peritoneum, omentum majus, and mesentery, indicating SHC recurrence and metastases. Combination chemotherapy with pirarubicin and cisplatin was initiated for 1 cycle, but terminated due to resultant severe myelosuppression and medication intolerance. The patient was lost to therapy after 3 months of follow-up.This case is unique because of hepatitis C virus infection. We should consider the possibility of this disease in patients with atypical clinical presentation. Sarcomatoid carcinoma, a relatively rare form of malignant tumor, is characterized by features of epithelial and mesenchymal tumors. It is reported in a wide range of organs, with the lungs being most susceptible –3. SarcoClinical studies have suggested recurrent interventional therapies such as transcatheter arterial chemoembolization (TACE), radiofrequency ablation (RFA), and percutaneous ethanol injection to potentially be major causes of sarcomatoid transformation of hepatocellular carcinoma (HCC) –8; only In this case report, we describe a patient previously diagnosed with hepatitis C on discontinuous antiviral therapy manifesting with SHC. Additionally, we review the histopathologic, immunohistochemical, and diagnostic characteristics of SHC.A 68-year-old female patient was admitted to our hospital due to perceived abdominal distention for over 20 days. Past medical history included an 8-year infection with HCV and discontinuous anti-viral treatment. Laboratory tests revealed levels of HCV antibodies of 32.900 S/CO < 1.00), carbohydrate antigen 125 of 361.69 U/ml (< 35.00), neuron-specific enolase of 62.05 ng/ml (< 25.00), and alpha- fetoprotein (AFP) of 113.77 ng/ml (< 20.00). Abdominal computed tomography (CT) scan with tri-phase enhancement revealed an irregular, dense shadow (dimensions 110.0 mm × 160.0 mm) in the left hepatic lobe with cystic fibrosis and scattered uneven calcification within. An abnormally low-density filling defect was observed in the left portal vein .Immunohistochemistry revealed carcinoma cells positive for CD34 and CK8 . Cells were also partially positive for smooth muscle actin (SMA), vimentin, and cytokeratin-pan (CK-pan), but negative for desmin and AFP Fig. a–c. HistSHC is a rare form of a malignant hepatic tumor with combined characteristics of both HCC and hepatic sarcoma. In this case report, we described a unique case of an SHC patient previously diagnosed with HCV infection not having received anticancer therapies. We reviewed associated pathological and imaging features as well as relevant literature.As in primary hepatic carcinoma, symptoms of SHC are non-specific, which may simply result in a diagnosis of HCC at an advanced stage. However, in contrast to patients with HCC, those suffering SHC are more likely to develop local recurrence and distant metastases after surgical excision. In the present case, laboratory tests indicated abnormally high levels of AFP, which declined and fell within the normal range after surgery, suggesting that AFP may be associated with sarcomatoid transformation. Imaging may provide information assisting localization of the lesion. Currently, surgical excision is the preferred effective treatment for SHC, as the efficacy of alternative treatments such as radiotherapy, chemotherapy, and targeted therapy remain unclear.Final diagnosis of SHC depends upon precise histopathological, immunohistochemical, and pathological techniques. The primary hepatic sarcoma cell is histologically characterized by a spindle shape, possessing a clear nucleolus and acidophilic cytoplasm, atypia, and significant mitotic attributes . SarcomaThe pathogenesis underlying SHC remains unclear. Most reported SHC cases manifested in the elderly patients who previously received recurrent interventional therapies such as TACE, RFA, or percutaneous ethanol injection –8. A numImaging remains the primary modality used in localization of lesions. It also provides essential information in establishing an accurate diagnosis, which is obviously vital for choosing and optimizing the type of surgery for patients with SHC. The rapid progression of SHC usually results in a massive, cystic-solid mass of mixed density noted on imaging exhibiting characteristic CT findings. A lower-density shadow in the mass is frequently detected, likely caused by ischemia and necrosis of the tumor due to rapid cellular proliferation. On enhanced CT scan, central mass signal intensity decreases due to necrosis, while parenchymal signal intensity remains greater. Similar enhancements can be found in the margins of the arterial and delayed phases. SHC exhibits weak signal intensity on T1W1 MRI, but high signal intensity on T2W1. On enhanced MRI, a substantial margin of the irregular septum is enhanced during the arterial phase, while the central cystic necrosis does not, and the portal venous phase reveals decreased enhancement . In our Literature regarding SHC mostly consists of case reports, due to the low incidence of the condition and unconfirmed diagnostic criteria. It is important that SHC is differentiated from other liver tumors, such as hepatapostema and hepatosarcoma. Common clinical symptoms of hepatapostema include pain in the right upper abdominal quadrant, fever, and chills. Laboratory tests usually indicate the occurrence of an inflammatory reaction manifesting with anemia, increased white blood cell count, elevated erythrocyte sedimentation rate, and higher expression of C-reactive protein. Imaging of hepatapostema typically reveals an isolated mass without liver lobe infiltration. Pathological examination exhibits phlogotic infiltration into the lesion instead of sarcomatous and carcinomatous constitution . HepatosSurgical excision is accepted as the most effective treatment of SHC, but patients are more likely than those with typical HCC to develop local recurrence and distant metastases after operation, mainly due to the high grade of malignancy and rapid progression of the disease. Indeed, the prognosis of SHC is quite poor, with 3-year survival rates reported as low as 18.2% after hepatectomy . To dateIn summary, we report a rare case of SHC, in which HCV infection might have contributed to its sarcomatoid transformation. When treating atypical HCC patients with viral hepatitis, even those not undergoing anticancer therapies, SHC should be considered as a possible diagnosis. A poor prognosis, with extremely low 5-year survival rates, should be expected. Future studies are urgently needed for exploring more effective therapies in order to extend the survival period and improve the quality of life of patients with SHC.


The omphalomesenteric duct (OMD) or the vitelline duct (VD) is the embryonic structure connecting the vitelline sac to the primitive gut. It undergoes obliteration at 5–9 weeks of gestation. Failure of this duct to close, which occurs in approximately 2% of the population, can lead to various types of VD residual diseases. A persistent OMD remnant is pathological, and it typically presents in the pediatric population. Meckel diverticulum is the most common anomaly that results from failure of resorption of the OMD. In extremely rare instances, OMD remnant adenocarcinomas have been reported in the adult population.In this study, we present a case of OMD remnant adenocarcinoma with axillary lymph node metastases in an adult male.Because OMD remnant adenocarcinoma is rare, few relevant studies have been reported. The final diagnosis of navel VD residual adenocarcinoma depends on postoperative pathology and immunohistochemical analysis. The follow-up treatment in OMD is similar to the chemotherapy regimens of postoperative gastrointestinal malignant tumors.In this report, the patient experienced no complications after surgery and was discharged on the seventh postoperative day, followed by 12 courses of postoperative FOLFOX6 scheme chemotherapy. By the end of chemotherapy, the patient had no evidence of recurrent disease and metastasis across the reexamination of PET–CT. The omphalomesenteric duct (OMD) or the vitelline duct (VD) is the embryonic structure connecting the vitelline sac to the primitive gut, which undergoes obliteration at five to nine weeks of gestation (Bagade and Khanna –computed tomography (PET–CT) scan of the umbilicus mass revealed a focally increased metabolic rate, and there was no clear border line with adjacent bowel loops  Salemis . A previIn the present case, OMD remnant adenocarcinoma was suspected from the physical and imaging examination findings. Surgery provided a definitive diagnosis of an OMD remnant and curative treatment by resection with minimal invasion (Morita et al. Omphalomesenteric duct remnant adenocarcinoma is very rare, and few relevant studies have been reported. The combined pathological type, operation findings, imaging findings and even a colonoscopy are needed for diagnosis. Final diagnosis of the navel VD residual adenocarcinoma is made on the basis of postoperative pathological and immunohistochemical results; follow-up treatment should choose one type of chemotherapy regimen of postoperative gastrointestinal malignant tumors.Patients with axillary lymph node metastases require adjuvant chemotherapy 6 months after resection of the primary tumors (Des Guetz et al. We have presented an extremely rare case of OMD remnant adenocarcinoma safely resected by surgery in an adult patient. Imaging plays a critical role in the diagnosis of OMD remnants. An awareness of the variety of these anomalies is essential to their proper management.


The dysplasia epiphysealis hemimelica (DEH) is a rare disease of unknown etiology consisting in an abnormal osteocartilaginous growth at the epiphysis, usually hemimelic with histological findings similar to benign osteochondroma. In this case series, we described the results of the arthroscopic treatment of 2 consecutive cases of intra-articular ankle localization of DEH in 2 patients aged 9 and 10 years. The good result obtained, persistent at the 5-year follow-up, leads us to consider the arthroscopic approach as a reliable treatment in patient affected by intra-articular ankle DEH. Dysplasia epiphysealis hemimelica (DEH), firstly reported by Muchet and Berlot in 1926 and called tarsomegaly . It is aGenerally diagnosed between 2 and 14 years of age, it consists of an abnormal osteocartilaginous growth localized at the epiphysis, usually hemimelic  . The DEHAzouz classified the DEH in 3 types: localized, when a single epiphysis  is involved; classic, when more than one epiphysis of the same limb (2/3 of cases) is involved; generalized, when a whole limb is involved .Histologically, however, the lesions are similar to osteochondroma; DEH lacks the EXT1 and EXT2 gene mutations typical of the osteochondroma , 13. TheDEH treatment is controversial because the rarity of the disease and absence of high level study. Asymptomatic lesions can need only clinical observation , while tIn July 2010, a 9-year-old Caucasian male presented to our attention for persistent left ankle pain that began 40 days before following an ankle sprain playing soccer. The pain was not present during night but occurred with sport activities and walking for long time. The clinical examination showed pain in the anteromedial and posteromedial region of the ankle without remarkable soft tissue swelling and a slight reduction (5°–10°) of the dorsiflexion in comparison to the contralateral ankle; there was no limping and the stability tests of the ankle were negative. Plain radiographs showed bony-like irregular protuberances localized at the anteromedial and posteromedial aspect of the talus . ComputeThe patient was recalled in October 2016 (5 years after surgery) for a clinical and radiographic examination. The X-rays showed that the left ankle was free of recurrence and there were not ankle arthritis signs . The cliIn January 2011, a 10-year-old Caucasian male referred to our hospital for a 3-month ongoing story of pain and swelling of the medial aspect of the left ankle. There was no history of trauma, fever, other joints pain, and abnormalities. He experienced pain walking and playing sports, with limping at the end of the day. Clinically, the ankle joint was painful at the medial edge from the anterior to the posterior aspect. Both dorsiflexion and plantar flexion were reduced of 10°, with local swelling and mild warmth.X-rays examination showed a protuberance of bony-like nature starting from the medial articular portion of the talus and with lobulated appearance and a large intra-articular cauliflower fashioned loose body . The 3D Because of the symptoms described and the imaging evidences, the first choice of treatment was the surgical arthroscopic removal by a posterior two-portal approach. The arthroscopy showed a flush reactive synovitis that was removed using the motorized tool. The intra-articular loose body was completely removed. The large pedicled prominence was arthroscopically detected but because of the need to perform the histological examination, the removal was performed by an open anteromedial approach. The rehabilitation program was the same as the previous case described. The histological examination showed a cartilaginous and bony-like mass without any evidence of neoplastic cells; then the clinical and pathological diagnosis was DEH of the ankle. The outpatient examinations were performed 2 weeks, 4 weeks, and 3 months after surgery, when the return to his days and sport activity was allowed. At the 6-month follow-up, there was no recurrence of pain and swelling or any other issues and there were no evidences of any perioperative and postoperative complication.The patient was recalled in October 2016 (5 years after surgery) for a clinical and radiological examination. The X-rays  did not The aim of this paper was to describe the good results obtained, in a 5-year follow-up examination, in the treatment of two cases of symptomatic DEH of the ankle by the arthroscopic approach.The first patient referred to pain after a first-grade ankle sprain with symptoms that were refractive to a prolonged physical therapy and anti-inflammatory drugs. The second case was a patient with an atraumatic ankle pain, with swelling and reactive synovitis.Despite the different presentation, in both cases the arthroscopic removal of the lesions restored a good function with results that lasted after 5 years.The arthroscopic approach to these kind of lesions seems to be growing in the literature, although the rarity of the condition does not allow us to draw definitive conclusions. In a recent accurate review of the literature, Gökkuş et al. suggested that carrying out excisions is a better choice because of a large number of good results, although the number of cases in the literature is limited. They highlighted the chance to treat these lesions by an arthroscopic approach . PreviouAn important issue that has to be highlighted is the role of the MRI in the diagnosis of this condition. Although the traditional radiological images provide some important information for the diagnosis, the MRI examination is extremely useful for the differential diagnosis, in identifying the extension of the epiphyseal lesion, and in identifying the eventual joint deformity and the soft tissue changes associated with DEH. Also, the role of MRI is important both in the follow-up and in the recurrence .After an accurate review of the literature and after our good experience, because the natural history of the lesion is to grow until skeletal maturity, when localized intra-articular, the removal of the osteochondromatous lesions should be performed arthroscopically, although asymptomatic, to avoid the risk of articular cartilage degeneration and early osteoarthritis .In conclusion, the arthroscopic removal of the ankle lesions can be considered a viable option for the DEH treatment.