Teratomas are neoplastic tumors derived from totipotent germ cells containing a wide assortment of tissues originating from all three germ cell layers. Teratomas can be mature or immature depending on the presence of immature tissues; typically neuroepithelial tissue. Immature teratomas can be oncologically benign or malignant, and can be divided into three grades with increasingly aggressive biological behavior. The most common site for this tumor is the sacrococcygeal region. The nasal septum is an exceptionally rare site for immature teratomas, with very few cases reported.We discuss a 14-year-old male patient with a left nasal mass which, on histopathological examination, turned out to be a Grade-3 immature teratoma. Imaging revealed the mass to be confined in the left nasal cavity with erosion of the anterior skull base. During endoscopic excision, the tumor was seen extending intracranially but remaining extradurally. Complete resection was achieved, albeit with mild cerebrospinal fluid (CSF) leakage, which was closed successfully. The patient was subjected to adjuvant chemotherapy. A regular follow-up of 2 years showed no recurrence.The purpose of this report is to document the first case of a high-grade immature teratoma arising from the nasal septum with intracranial extension, as well as the efficacy of combined endoscopic resection and adjuvant chemotherapy for this pathology. Teratomas are congenital or developmental tumors which are derived from multi-potent cells and differentiate into diverse types of tissue, representative of all three germ layers . TeratomHere, we discuss a case of immature teratoma (Grade 3) arising from the nasal septum in a 14-year-old boy, with emphasis on management and a brief review of literature.A 14-year-old boy visited the ear, nose, and throat (ENT) outpatient department of our center, with a history of left-sided nasal obstruction gradually progressive over a period of 1 year, with recurrent history of associated rhinorrhea. There was no history of epistaxis or any other ENT complaints, and the patient’s past medical history and family history were insignificant.On anterior rhinoscopy, a single polypoidal mass was seen filling the left nasal cavity extending up to the vestibule. The mass was firm in consistency, insensitive to touch and pain, and did not bleed upon touch. General and systemic examinations and an examination of the rest of the ENT were all within normal limits. A contrast-enhanced computed tomography (CT) scan showed a homogenous soft tissue lesion measuring 44×68×12 mm in the left nasal cavity, extending posteriorly into the nasopharynx . The mass extended superiorly into the left ethmoid air cells up to the cribriform plate with its thinning and breach. The rest of the sinuses was clear. On contrast study, there was heterogeneous enhancement. Based on the CT findings of the anterior skull base breach, gadolinium-enhanced magnetic resonance imaging (MRI) was performed to rule out intracranial extension, and revealed small sub-centimeter focus posteriorly without any significant intracranial extension . Rigid endoscopy was carried out and the mass was seen attached to the anterior part of the nasal septum. A punch biopsy was taken, which presented as an inflammatory polyp.The patient was referred for standard endoscopic sinus surgery under general anesthetic after providing informed consent. The mass was removed endoscopically, and was seen to be extending intracranially but extradurally. Complete resection of the tumor was achieved and while managing the tumor in the region of the cribriform plate, there was an obvious CSF leak. The site of leakage was identified and closed using an underlay technique with septal cartilage, fascia lata, and tissue glue. The intra-operative blood loss was minimal.The post-operative recovery period was uneventful. Histopathology of the excised specimen showed the tumor mass lined by pseudostratified columnar epithelium with squamous metaplasia. The subepithelial tissue was composed of loose myxoid areas with mature adipose tissues and foci of cartilage. Foci of neuroepithelial cells occupying more than four low-power fields were seen. Grade 3 immature teratoma was given as final diagnosis -6. Adjuvant chemotherapy was started based on the histological grading, with four cycles of bleomycin, etoposide, and cisplatin. A regular follow-up for a period of 2 years showed no signs of recurrence.Teratomas are tumors of totipotent cells, consisting of mature and immature tissues originating from all three germ cell layers. The predominant component is ectodermal, which usually consists of neural tissue, teeth, skin, and hair. The mesodermal tissue is represented by fat, bone, and cartilage, whereas the endodermal component includes respiratory or intestinal epithelium. Primitive neuroepithelium with rosettes, pseudorosettes, or neurofibrillary matrix are observed in some tumors . ImmaturThe histogenesis of the head and neck teratoma is controversial, and the most accepted theory is displaced totipotent germ cells during embryogenesis and subsequent differentiation into teratomas . Head anUnlike teratomas of other regions, which are more common in females, head and neck teratomas do not have a gender predilection . We encoAnother case study reported erosion of lamina papyracea with intraorbital extension .Immature teratomas are histologically graded into three subtypes, with worsening prognosis, based on the amount of immature neuroectodermal tissues. If the foci of the neuroepithelial cells occupy no more than one low-power field per slide then it is characterized as Grade 1, while Grade 2 is established if the foci occupy no more than three low-power fields per slide. When the foci of the neuroepithelial cells occupy at least four low-power fields per slide then this is described as Grade 3 immature teratoma . It is necessary to at least identify tissues originating from two out of three germ layers; the ectoderm, mesoderm, and endoderm .Complete surgical excision is the treatment of choice for sino-nasal teratomas irrespective of their histological nature, and the approach depends on the size of the tumor and its extent. Sreetharan et al. employed a lateral rhinotomy approach in their first documented case of teratoma of the nasal septum . In our This is a far superior modality compared with an open approach, as the associated angled telescopes and magnification help ensure complete removal of the tumor from every corner. Furthermore, this approach is associated with less intra-operative bleeding, shorter hospital stays, fewer surgical morbidities, and excellent cosmetic acceptability. Use of powered instruments such as a micro-debrider and electric scalpel has also been reported ,10. TumoWe conclude that sino-nasal teratoma, although an exceptionally rare pathology, can originate from the nasal septum, and this tumor should be included in the differential diagnosis of unilateral benign nasal tumors. The first documented case of nasal septal teratoma was reported in 2004 , and verAfter a MEDLINE® search of the available literature, we failed to find any case report describing such advanced-stage immature teratoma of the nasal septum causing bony erosion of the skull base with intracranial extension. We further conclude that endoscopic resection with adjuvant chemotherapy is a safe option for these tumors, despite having a high recurrence rate and significant mortality.


Background: Schwannomas are tumors originating in Schwann cells of the peripheral nerve system and uncommonly develop in the gastrointestinal tract. Sigmoid colon schwannomas are very rare and only 28 cases have been reported. This study aims to report a case of a sigmoid colon schwannoma and present a literature review.Case report: We report a case of a 66-year-old female with asymptomatic sigmoid colon schwannoma. The patient underwent a screening colonoscopy and about 4cm sized submucosal tumor was identified at the sigmoid colon. A colonoscopic biopsy was performed and the microscopic exam revealed an ulcerated lesion with a proliferation of fibroblast-like spindle cells beneath ulcer, which was insufficient for diagnosis. Abdominopelvic computerized tomography (CT) scan showed a well-defined, well-enhancing, round shaped and slightly heterogenous mass at the sigmoid colon. No distant metastasis was identified in abdominopelvic CT and chest CT scans. Carcinoembryonic antigen level was within a normal range (1.33ng/mL). The patient underwent laparoscopic anterior resection. Immunohistochemical staining of the resected specimen showed positivity for S-100 protein in tumor cells and schwannoma was diagnosed post-surgically. Surgical resection margins were free from tumor and no regional lymph node metastasis was reported.Conclusion: Colon schwannomas are rare diseases. Most cases of colon schwannomas are accidentally identified during screening colonoscopy. The tumors usually present as submucosal masses and colonoscopic biopsies are mostly non-diagnostic. Surgical resection is required, and definitive diagnosis is made by confirming S-100 positive tumor cells in immunohistochemical analysis. Most cases are benign; a few cases have been reported to be malignant. Surgical resection with free negative margins is the treatment of choice GI tract schwannomas develop most frequently in the stomach (83%), and less frequently in the small intestine (12%), colon and rectum3. Sigmoid colon schwannomas are very rare and only 28 cases have been reported so far4. Most colon schwannomas are incidentally identified as submucosal tumors on screening colonoscopy5. Colonoscopic biopsies alone usually provides limited information and definite diagnosis is made after surgical resection1. Most of the colon schwannomas are benign and surgical resection with adequate free resection margins is the treatment of choice6. Here we present a case of sigmoid colon schwannoma and discuss the clinical features of the disease with a literature review.Schwannomas are a common type of tumor of peripheral nerve in adults which originate in Schwann cells. These tumors mainly present along the peripheral nerves and are rarely identified in the gastrointestinal (GI) tractA 66-year-old Asian female patient, who was a housewife, visited a local clinic for a routine screening colonoscopy in mid-January 2018. During the colonoscopy, a submucosal tumor sized about 4cm was identified at the sigmoid colon  and biopThe patient was referred to our hospital at the end of January 2018. She presented no specific symptom and physical examination showed no specific finding. She had a history of hypertension and a benign breast mass. She had a positive family history of cancer: her father had gastric cancer, and her uncle had lung cancer.An abdominopelvic computerized tomography (CT) scan revealed 4.4cm sized well-circumscribed, well-enhanced, round-shaped mass in the sigmoid colon, which was slightly heterogenous inside. No intraabdominal metastasis was identified . Chest CThe patient underwent a laparoscopic anterior resection. On laparoscopic exploration, an extruding mass was identified at the anterior wall of the sigmoid colon and no metastasis was observed. The sigmoid colon was mobilized and the inferior mesenteric artery was low ligated. Sigmoid colon resection with end-to-end anastomosis was performed.On examining the resected specimen, about 4.5 × 4.0cm sized round mass was observed on the surface of the serosa and there was no tumor infiltration to the serosa . The tumThe patient recovered from surgery uneventfully and was discharged on postoperative day 5. When she visited the out-patient clinic two weeks after discharge, she did not present any complication. No postoperative adjuvant therapy was performed.2. GI tract schwannomas represent about 2–6% of all mesenchymal tumors3.Schwannomas are peripheral nerve sheath tumors which rarely develop in GI tractet al. clarified the entity of the nerve sheath tumors developing in the GI tract and proposed these tumors to be designated as “benign schwannoma of the GI tract” in 19887. Lymphoid cuffing, benign nuclear atypia and positive immunostaining for S-100 protein were the distinct features of the schwannoma of the GI tract, which distinguish the schwannoma from other spindle-cell stromal tumors of smooth muscle origin7. Until the early 1990s, most GISTs traditionally had been classified as smooth muscle tumors8. Ueyamaet al. suggested that most of the GIST had smooth muscle differentiation and excluded schwannomas from the GIST8.For the first time, Daimaru9. And GI schwannomas are considered distinguished from conventional soft-tissue schwannomas and CNS schwannomas2.Currently, GI tract schwannomas are classified as non-epithelial tumors of which disease entity is clearly distinct from leiomyomas, leiomyosarcomas, gastrointestinal autonomic nerve tumors (GANTs) and GISTs3. They are most frequently diagnosed among people in their sixties and the incidence rates are identical for males and females10. Most of them are incidentally identified during screening endoscopy or imaging studies because they are usually asymptomatic. However, just like any other GI tumors, they can present some clinical symptoms such as abdominal pain, tenesmus, rectal bleeding or melena1. Sometimes these tumors manifest as colonic obstruction or intussusception10.GI schwannomas are mostly identified in the stomach and less frequently seen in colon, rectum, small intestine or esophagus3. On CT scans, the tumors usually present as exophytic masses with homogeneous enhancement and cystic change, necrosis, or calcification within tumors are uncommon2.Endoscopically, these tumors usually present as a submucosal tumor with smooth mucosa or with mucosal ulceration3. In Inagawa’s study, only 15% of the colon schwannomas were diagnosed on preoperative endoscopic biopsy11; in Bohlok’s study, 24% of the colorectal schwannomas were diagnosed preoperatively3.A preoperative diagnosis is challenging because endoscopic mucosal biopsy usually provides limited information to differentiate them from other mesenchymal tumors of GI tract such as GISTs, NETs, leiomyomas, or leiomyosarcomas11. Schwannomas can be distinguished from other smooth-muscle tumors by strong s-100 positivity in immunohistochemical analysis13. Additionally, CD34 or c-kit protein is useful to distinguish the schwannomas from GISTs13. Schwannomas are S-100 positive, but CD-34 and c-kit negative; most GISTs are s-100 negative, but CD-34 and c-kit positive.Diagnosis is confirmed pathologically with immunohistochemical analysis. Histopathological features of schwannomas are mainly elongated bipolar spindle cells with variable cellularity and sometimes peripheral cuff-like lymphocyte infiltration is exhibited around the tumor, which helps to differentiate schwannomas from other spindle-cell tumors like fibromas or leiomyomas3. However, even though many researchers reported the benign features of GI schwannoma, some of these tumors present local recurrence or distant metastasis. In Bohlok’s study, 3 (3.1%) out of 93 cases of colorectal schwannomas were malignant3. High mitosis rate, high Ki-67 index, and large tumor size are considered to be associated with malignancy3.Prognosis is generally promising because most of the GI schwannomas are benign and malignant potential is low3, because tumor recurrence is generally owing to incomplete surgical resection with inadequate margins2. In some limited cases, patients can be treated by endoscopic resection or transanal resection without undergoing radical surgery3.Complete surgical resection obtaining free resection margins is the best therapeutic option14. Currently, limitation of our knowledge is that there is no consensus for subsequent treatment after surgical resection in case of malignant transformation5.Adjuvant therapies are not commonly recommended if surgical resection achieving free margins is completedSigmoid colon schwannomas are very rare colonic neoplasms. To our knowledge, only 28 cases of sigmoid colon schwannomas have been published. Because of its rarity and challenge to diagnosis, review of the clinical features of the disease with a presenting case would be of help for physicians and surgeons. We believe that our study presents the clinical manifestations including endoscopic, imaging, histopathologic and immunohistologic findings of this rare disease with a thorough literature review and it provides guidance in diagnosis and treatment of the disease. Limitation of this study is that the treatment strategy for metastatic diseases was not suggested because only very limited cases were reported and no consensus exists for now.Sigmoid colon schwannomas are usually found incidentally during screening colonoscopy and present as submucosal tumors. Preoperative diagnosis is challenging because clinical manifestations, as well as colonoscopic and CT findings, are nonspecific. No specific tumor marker exists either. Histopathologically, the tumors consist of spindle cells. However, colonoscopic biopsies have limitations in terms of a definite diagnosis and differential diagnosis includes schwannoma, GIST, NET, leiomyoma, leiomyosarcoma, etc. Conclusive diagnosis can be made by confirming s-100 proteins in immunohistochemical analysis and mostly confirmed post-surgically. Complete surgical resections with adequate free margins are required because although the majority of the diseases are benign, some are reported to be malignant. There is no consensus for adjuvant chemotherapy.All data underlying the results are available as part of the article and no additional source data are required.Written informed consent for publication of their clinical details and clinical images was obtained from the patient. The authors presented a case report and a review of related literature about sigmoid colon schwannomas. This is a rare disease and an interesting subject.The authors claimed segmenter resection is enough for sigmoid colon schwannoma but in their case report they prefer to anterior resection. it is necessary to explain this in the discussion section.The authors shared a photo of the specimen they resected in figure 3. This image resembles segmental resection rather than anterior resection. Although it is a beautifully written review in general, some corrections are required:I confirm that I have read this submission and believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard. Overall this is a good case report and review of a rare tumor. There are a few small grammatical errors, but the paper is otherwise well organized and easy to follow.Minor suggestions:When referring to the size of the tumor, especially when giving dimensions that seem fairly exact ("4.5 x 4.0 cm") it is unnecessary to say "about", you can just state the size.Replace "Most of the" with "Most", as in "Most GI tract schwannomas" or "Most colon schwannomas"Figure 6: "proper muscle layer" should be "muscularis propria layer"I confirm that I have read this submission and believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard.


Rectus sheath hematoma is an unusual but well-known clinical problem. Our hospital admitted a 54-year-old woman complaining of harsh right-sided hypogastric pain that started while muscle training. Computed tomography of the abdomen and pelvis demonstrated a right rectus sheath hematoma. As the hematoma did not increase, the patient was conservatively treated. Despite increased awareness of rectus sheath hematoma, its early diagnosis and treatment still present a challenge to emergency physicians. Swift acknowledgement of this rare cause of abdominal pain may avoid more intrusive examination, unnecessary hospitalization, and laparotomy. Careful consideration of the patient's medical history and a high index of suspicion are needed to diagnose this complication. Rectus sheath hematoma is a relatively uncommon but important cause of acute abdominal pain encountered in emergency room patients , 2. Due Herein, we report a case of spontaneous rectus sheath hematoma following abdominal exercise. Rectus sheath hematoma is usually associated with abdominal trauma  and/or a3 (3500-9500 /mm3), hemoglobin 13.4 g/dL (12.1-15.1 g/dL), hematocrit 38% (37-46 %), and platelet count 365 x 103 cells /mm3 (150-450 x 103 cells /mm3). Her serum electrolyte, renal function, and urinalysis test results were not notable.Our hospital admitted a 54-year-old woman complaining of strong, right-sided, hypogastric pain two hours after muscle training. The pain was exacerbated by breathing and moving. She was not taking anticoagulants and did not have any known blood dyscrasia. Her vital signs were pulse rate 80 beats/minute and rhythmic, blood pressure 115/85 mmHg, respiratory rate 18 breaths/minute, body temperature 37.8°C, and arterial oxygen saturation 97%. The patient had no symptoms of fever, nausea, chills, vomiting, or diarrhea. Physical examination revealed muscle defense and a tender, palpable 10 cm mass in the abdomen. Bruising around the umbilicus and flank was noted. Bowel sounds were normoactive. Testing revealed a white blood cell count of 11200 /mmAbdominal computed tomography (CT) was performed to determine the reasons for acute abdomen, along with acute appendicitis. Enhanced abdominal CT revealed a right rectus sheath hematoma with extravasated contrasting agent . The hemOne of the emergency physician's most important tasks is determining lethal causes of abdominal pain among cases mimicking a myriad of acute abdominal conditions such as rectus sheath hematoma. Rectus sheath hematoma may occur from not only a direct blow, but also strenuous non-contact exercise and trivial movements like twisting or sudden muscle strain and change in position without direct trauma. It occurs due to rupture of the upper and lower epigastric arteries and their branches or rupture of the rectus muscles themselves during contraction . In our Risk factors include anticoagulation therapy, old age, asthma exacerbation, recent surgery, medication injection in the abdomen, paroxysmal coughing, obesity, pregnancy, and chronic liver disease , 14, 15.With careful recording of the patient's medical history and paying close attention to a few critical points on examination, one could reliably differentiate a rectus hematoma from intra-abdominal pathology, thereby avoiding unnecessary surgery. Abdominal ultrasonography was also a very helpful available bedside tool and valuable screening technique in this case. Abdominal CT provides information helpful for differential diagnosis and can depict precise hematoma localization, with near 100% sensitivity during the first five days of rectus sheath hematoma formation . In partAlthough uncommon, rectus sheath hematoma is a significant cause of abdominal pain that can imitate surgical acute abdomen. Physicians should consider this diagnosis in patients with the above described predisposing factors. It is important for emergency physicians to keep this differential diagnosis in mind along with other pathologies of acute abdomen.


Thyroglossal duct carcinoma is a rare pathologic entity. The surgeon's main concern is whether to perform thyroidectomy or not. In this paper, we report another case of thyroglossal papillary duct carcinoma in a 14-old girl suspected preoperatively and confirmed postoperatively on the histological analysis of resected specimen by a Sistrunk procedure. Therapeutic strategy was completed by a total thyroidectomy with radioactive iodine therapy and suppressive levothyroxine therapy. In the absence of clear guidelines, the management of thyroglossal duct carcinoma is depending on the clinical situation and the experience of the team of surgeons. It occurs in 0.7-1% of cases and most are undiagnosed preoperatively. TDCa in children is even rarer than in adults, with few cases reported in the English literature [Thyroglossal duct cysts (TDCs) are the commonest congenital anomaly in the development of the thyroid gland. Classically, it manifests as a swelling in the midline of the neck that moves with deglutition and protrusion of the tongue. The development of a carcinoma in a cyst is rare but well recognized. Brentano A 14-year-old female patient presented with an 11-month history of neck swelling. She had no dyspnea, dysphagia or hoarseness. There was no history of prior radiation. Familiar medical history was negative for thyroid gland or neoplastic diseases. She did not have any symptoms of hyper-or hypothyroidism. Physical examination revealed a 3cm mass of located in the midline region of the neck covered by a normal skin . It was Treatment: A Sistrunk surgical procedure was then performed. Peroperatively, a cystic mass was noted close to the hyoid bone; it was adherent to the digastric and to the mylohyoid muscles. The mass was removed along with entire duct from the gland to the level of the foramen caecum and the middle portion of the hyoid bone. Extemporaneous examination of the surgical specimen showed a cystic mass with a smooth external surface with no signs of malignancy  are the most common anomaly of the cervical region in childhood accounting for 70% of all congenital neck lesions . They oret al. reviewed a treatment of twenty-six of childhood TDCa. Thirteen of the patients underwent a total thyroidectomy but none of them had showed a malignant involvement of the thyroid gland. They concluded that Sistrunk procedure and local lymph node resection is sufficient for adolescent patients with TDCa that show no signs of thyroid involvement on ultrasound or uptake scan. Close follow up with these patients is warranted to monitor for future signs of thyroid involvement [et al. concluded that the addition of total thyroidectomy to the Sistrunk operation did not have a significant impact on recurrence and survival [et al. recommend in addition to Sistrunk procedure, a total thyroidectomy and excision of large regional lymph nodes in treating patients with TDCa. Post-operative radioiodine ablation and suppressive levothyroxine are further steps in treatment [In all instances, a Sistrunk procedure should be performed, consisting of excision of the cyst, the middle portion of the body of the hyoid bone, and a core of tissue around the thyroglossal tract at the foramen caecum , 14. Thesurvival . Howeverreatment . The incThe lack of consensus in the management of childhood is due to the rarity of the disease and the small number of cases managed, even in tertiary referral centers. Considering literature and our observations through this case report, we thought that total thyroidectomy must not systematically performed in children and discussed when there is clinical and radiological criteria of gland invasion.The authors declare no competing interests.


Cholecystoduodenal fistula (CDF) is a rare complication of gallbladder disease. Clinical presentation is variable, and preoperative diagnosis is challenging due to the non-specific symptoms of CDF. We discuss a 61-year-old male with a history of atrial fibrillation who presented with severe abdominal pain out of proportion to exam. The patient was diagnosed promptly and successfully managed non-operatively. This case presentation emphasizes the need to maintain a broad differential diagnosis for abdominal pain out of proportion to exam, with the possibility of a biliary-enteric fistula as a possible cause. It also stresses the importance of a multimodality imaging approach to arrive at a final diagnosis. A 61-year-old male with a history of atrial fibrillation presented to our emergency department with intermittent, post-prandial abdominal pain over the prior week. The patient localized the pain to the epigastric region and reported associated diarrhea. On examination, he was afebrile but appeared markedly distressed with an irregularly irregular rhythm at 108 beats per minute. Abdominal exam revealed mild generalized tenderness without guarding or rebound. Laboratory study results were unremarkable. Right upper quadrant ultrasound revealed gallstones with gallbladder wall thickening measuring up to 9.0 millimeters. In conjunction with the ultrasound findings, computed tomography (CT) angiography of the abdomen and pelvis revealed acute cholecystitis with gallbladder adherence to the right hepatic lobe and a fistula between the inflamed gallbladder and proximal duodenum  and 2. MCholecystoenteric fistula (CF) is defined as a spontaneous tract between an inflamed gallbladder and one or more parts of the adjacent gastrointestinal tract. Cholecystoduodenal fistula accounts for approximately 75–80% of all such fistulas.4What do we already know about this clinical entity?Cholecystoduodenal fistula (CDF) is a rare complication of gallbladder disease with chronic cholecystitis as the primary etiology.What is the major impact of the image(s)?These images clearly characterize CDF and will help raise awareness for this particular disease.How might this improve emergency medicine practice?This presentation emphasizes the need to maintain a broad differential diagnosis for abdominal pain out of proportion to exam, with CDF as a possible cause.


The objective of this article was to report the clinicopathological characteristics, treatment modalities, and outcomes of patients with clear cell carcinoma (CCC) of the abdominal wall. Medical records of six patients diagnosed with CCC of the abdominal wall between May 2003 and May 2018 at the National Taiwan University Hospital were reviewed. All patients had prior obstetric or gynecologic surgeries. The primary clinical presentation was enlarging abdominal masses at previous surgical scars. Four patients underwent initial/primary surgeries with/without adjuvant chemotherapy. One patient received neoadjuvant chemotherapy followed by surgical intervention and adjuvant chemotherapy, the other received chemotherapy and sequential radiotherapy without any surgical intervention. Two of four patients undergoing initial/primary surgeries had disease recurrence and the remaining two cases without initial surgery experienced disease progression during primary treatment. Inguinal lymph nodes were the most frequent sites of recurrence. In conclusion, previous obstetric or gynecologic surgery can be a risk factor for CCC of the abdominal wall. Complete resection of abdominal wall tumor and suspected intra-abdominal lesions with hysterectomy and bilateral inguinal lymph nodes dissection may be the primary treatment. Adjuvant chemotherapy would be considered for potential benefits. For patients without bilateral inguinal lymph nodes dissection, careful inguinal lymph node palpation during postoperative surveillance is necessary. More cases are still needed to elucidate the clinical management of this disease. It is estimated that cancers arise in approximately 1% of woman with ovarian endometriosis . MalignaIn 1925, Sampson described the first case of malignant transformation of ovarian endometriosis, and defined three criteria for the diagnosis of malignant transformation of endometriosis: (1) Demonstration of both neoplastic tissue and endometriosis within the tumor, (2) histological appearance resembling endometrial stroma surrounding characteristic glands, and (3) no other primary tumor site . In addiThe management of CCC of the abdominal wall is not well established because of its extremely low incidence rate. Therefore, we comprehensively reviewed the characteristics of six cases with this disease in our institution over a 15-year time period. To our knowledge, the current case series is the largest one to date. It is noteworthy that this is the first study reporting the recurrence pattern of CCC of the abdominal wall, which may have potential to modulate the primary treatment strategy for the improvement of clinical outcomes for these patients.Following approval by the National Taiwan University Hospital Institutional Review Board, a retrospective review was conducted (201805083RIND). All medical records of 6 patients treated for CCC of the abdominal wall in the National Taiwan University Hospital between May 2003 and May 2018 were reviewed. These cases were confirmed via pathological diagnosis. The medical parameters, including basic characteristics, primary treatment modalities, pathologic characteristics, and clinical outcomes, were comprehensively reviewed.The basic characteristics included age at diagnosis, body mass index (BMI), parity, previous obstetric and gynecologic surgery histories, the interval between the most recent obstetric or gynecologic surgeries and disease diagnosis, tumor size, presenting symptoms, pretreatment serum cancer antigen 125 (CA-125) level, and findings of pretreatment computed tomography (CT). The treatment strategies included the methods of surgery, timing of administration of chemotherapy, and use of radiotherapy. The pathologic characteristics included histology, status of surgical margin, co-existing endometriosis, and existence of granuloma caused by suture materials of the abdominal wall tumor. When the removal of gynecologic organs was performed, pathologic examination for CCC and endometriosis in these specimens were recorded.The clinical outcomes included recurrent sites, types of salvage treatment, disease-free survival (DFS), overall survival (OS), and current status of these patients. Periodic examinations during follow-up included history-taking, pelvic and rectal examinations, and regional lymph node palpation every 3 months for 3 years, and every 6 months thereafter after completion of the primary treatment. Levels of serum CA-125 was determined on each visit. Smears of the vaginal cuff were done annually. CT or magnetic resonance imaging (MRI) was done for suspected recurrence. CA-125 levels ≥2-fold the upper limit of normal in two consecutive tests with 2-week intervals, abnormal results of imaging studies or tissue proven from a biopsy was considered as recurrence. DFS was defined as the length of time after completion of the primary treatment until the date of disease relapse or last follow-up. OS was calculated as the time from initial diagnosis of CCC of the abdominal wall until the date of death or last follow-up.2 . All patients received obstetric or gynecological surgeries. Five patients had cesarean section histories and one had laparoscopic surgery for ovarian endometriosis. The mean time from the most recent obstetric or gynecologic surgery to diagnosis of CCC of the abdominal wall was 20.2 years . The shortest interval (4.0 years) was noted in the patient with laparoscopic surgery (case 3). The mean tumor size was 10.1 cm . Five patients with cesarean section histories presented with an enlarging lower abdominal wall mass containing scars of a cesarean section  and carboplatin (AUC = 5 or 6).The primary treatment strategies of six patients for CCC of the abdominal wall are demonstrated in 2) and carboplatin (AUC = 5) combined with bevacizumab (7.5 mg/kg), and (2) gemcitabine (800 mg/m2) and carboplatin (AUC = 5). Surgical intervention with tumor excision, TAH, BSO, and infracolic omentectomy was performed for tumor progression. Postoperative adjuvant chemotherapy was prescribed using gemcitabine (800 mg/m2) and carboplatin (AUC = 5) combined with bevacizumab (7.5 mg/kg). However, the disease progression was still noted. Case 6 received tumor biopsy other than resection for disease diagnosis. This patient received two regimens of chemotherapy: (1) paclitaxel (175 mg/m2) and carboplatin (AUC = 5), and (2) liposomal doxorubicin (40 mg/m2) and carboplatin (AUC = 5). In addition, sequential radiotherapy with a total dose of 4400 cGy was administered after completing primary chemotherapy. However, the tumor progression still could not be controlled.Case 5 initially underwent two regimens of neoadjuvant chemotherapy: (1) Paclitaxel , which suggested a slow evolution of the tumorigenesis . The typPrimary wide tumor resection combined with total abdominal hysterectomy and bilateral salpingo-oophorectomy or cancer staging surgery was the preferred modality of primary treatment at our institution. For complete removal of all visible tumor tissue, a synthetic mesh or flap may be required for abdominal wall reconstruction. All the specimens without malignant suspicion on preoperative CT scanning histologically showed no evidence of clear cell carcinoma. Only right pelvic lymphadenopathy on preoperative images was finally confirmed as metastasis by pathologic examination F. Thus, Disease control of primary surgery with or without adjuvant chemotherapy was good. In our study, 50% of patients  had disease remission after primary operation. One patient  without evidence of disease was under postoperative chemotherapy. Two patients  who did not receive initial tumor resection with a free margin experienced uncontrolled disease progression. Therefore, initial complete tumor resection with/without adjuvant chemotherapy (case 1–4) seemed to have better disease control in comparison with other primary treatment modalities, such as neoadjuvant chemotherapy followed by operation and adjuvant chemotherapy (case 5), or chemotherapy and sequential radiotherapy (case 6). The role of adjuvant chemotherapy was not established. In our cohort, one patient not receiving adjuvant treatment eventually encountered disease recurrence (case 1). Four patients received adjuvant chemotherapy (case 2–5), two of them remained without evidence of disease (case 3 and 4), and the other two experienced disease recurrence (case 2 and 5). Overall, it would be difficult to draw a conclusion based on such limited cases. However, the results seemed to support the potential benefits of adjuvant chemotherapy. Therefore, administration of adjuvant chemotherapy after primary operation could be considered. At our institution, platinum-based chemotherapy was the most used.The survival intervals and prognostic factors are difficult to investigate due to the limited number of cases with standard management to treat CCC of the abdominal wall . TaburiaPatterns of disease recurrence/metastasis have not been reported in the literature before. The most common site of recurrence and metastasis was the inguinal lymph node. Inguinal lymphadenopathy can be detected in all cases with disease recurrence or progression . The reaCCC of the abdominal wall is a rare and distinct entity. Our study addressed comprehensive clinical courses and outcomes of these cases in a single institution. Complete resection of the abdominal wall tumor and intra-abdominal suspected lesions on preoperative image studies with hysterectomy and bilateral inguinal lymph node dissection may be suggested as the first step to treat CCC of the abdominal wall. Adjuvant chemotherapy would be recommended for potential benefits. For those patients without bilateral inguinal lymph node dissection, careful inguinal lymph node palpation during postoperative surveillance is needed. More such cases are still needed to elucidate the survival benefits of this surgical procedure, the role of adjuvant chemotherapy or radiotherapy, and the protocol of disease follow-up.


Biliary mucinous cystic neoplasms are rare cystic lesions of the liver which carry pre-malignant potential. Given the scarcity of reports in the literature, they pose a considerable challenge to clinical management, particularly with regards to accurate pre-operative diagnosis.We present the case of a 37-year-old Tunisian woman who presented with subacute right upper quadrant pain and a large multi-loculated cystic lesion, most consistent with a hydatid cyst. She underwent an open right hepatectomy, and pathology surprisingly revealed a biliary mucinous cystadenoma. Herein, we review the current literature on biliary mucinous cystic neoplasms, with a particular emphasis on diagnostic investigations, key radiological features and optimal treatment modalities.Biliary mucinous cystic neoplasms require a high index of suspicion and should be managed with complete surgical resection, as conservative techniques are associated with high recurrence rates. Considering the potential for malignant transformation, periodical surveillance imaging is recommended in the post-operative period. Biliary mucinous cystic neoplasms (BMCNs) are rare liver tumors that pose a considerable diagnostic challenge, as they can mimic various other liver lesions. Accurate pre-operative diagnosis may be difficult. A thorough understanding of the characteristic features of BMCNs and a high index of suspicion are essential to minimize the risk of malignant transformation or recurrence.Herein, we present an interesting case of a 37-year-old female with a biliary mucinous cystic neoplasm mimicking a hydatid cyst. We review the current literature on BMCNs, including clinical presentation, diagnostic investigations  and surgical management.An otherwise healthy 37-year-old Tunisian woman presented to the emergency department with a 3–4 month history of right upper quadrant pain. She also reported general malaise and a 2–3 kg weight loss. She denied any jaundice, fever or infectious symptoms. She had been living in Tunisia up until the previous year, and had been exposed to dogs and sheep. On physical examination, she had mild right upper quadrant tenderness, without peritoneal signs. Computed tomography (CT) scan revealed a 7.1 × 6.5 × 10.7 cm complex, multi-loculated cystic mass with partially calcified septations, partially embedded within the liver (segments V/VIII) and causing deviation of the right portal vein  are rare cystic lesions which predominantly occur in the liver, but can occasionally arise in the extra-hepatic biliary system. They account for less than 5% of non-parasitic liver cysts . There aBiliary mucinous cystic neoplasms are histologically divided into two types, depending on the presence of mesenchymal stroma, which is a subepithelial stroma resembling ovarian stroma . BMCNs wUltrasonography (US), CT and magnetic resonance imaging (MRI) are the most commonly used imaging modalities in the workup of BMCN . They tyThe presence of an irregular and thickened cystic wall, hypervascular mural solid nodules, thick calcifications and papillary projections may suggest an increased risk of malignancy . HoweverThe radiological appearance of hepatic hydatid disease is also quite variable. On US, the cyst wall is usually seen as double echogenic lines separated by a hypo-echogenic layer. Calcifications can be present in the cyst wall, although internal calcifications are also seen. Hydatid sand may be visualized as small echogenic foci falling to the most dependent portion of the cyst (i.e. snowstorm sign) , 35. DauCore needle biopsy is not routinely recommended, because of its low diagnostic accuracy and the risk of seeding and dissemination, in case of malignancy . In addiManagement of biliary mucinous cystic neoplasms is dictated by two key concerns. First, if misdiagnosed as a simple cyst or a hydatid cyst, incomplete excision  may be undertaken. Incomplete excision of BMCN is associated with a high recurrence rate, with some authors reporting recurrence rates over 90% , 40. ThuSecond, because differentiating BMCN from cystadenocarcinoma pre-operatively is exceedingly difficult, complete surgical resection remains the gold standard , 43. TheEnucleation is a viable alternative in cases in which formal resection would be technically difficult or associated with excessive morbidity , 46. AltFinally, given the high rate of recurrence and the potential risk for malignant transformation , surveilBiliary mucinous cystic neoplasms are rare liver neoplasms, which may strongly mimic a hydatid cyst in patients with a contributory history. Pre-operative differentiation on the basis of radiological, biochemical and serological features is often unreliable. Suspicious multilocular cystic lesions should be managed with complete surgical resection, as conservative techniques are associated with high recurrence rates in cases of BMCN. Considering the potential for malignant transformation, periodical surveillance imaging is recommended in the post-operative period.


Schwannomas of the colon and rectum are rare among gastrointestinal schwannomas. They are usually discovered incidentally as a submucosal mass on routine colonoscopy and diagnosed on pathologic examination of the operative specimen. Little information exists on the diagnosis and management of this rare entity.The aim of this study is to report a case of cecal schwannoma and the results of a systematic review of colorectal schwannoma in the literature.PubMed, Scopus, and Cochrane database searches were performed for case reports and case series of colonic and rectal schwannoma.Ninety-five patients with colonic or rectal schwannoma from 70 articles were included. Median age was 61.5 years . Presentation was asymptomatic (28%), rectorrhagia (23.2%), or abdominal pain (15.8%). Schwannoma occurred in the left and sigmoid colon in 36.8%, in the cecum and right colon in 30.5%, and in the rectum in 21.1%. Median tumor size was 3 cm and 56.2% of patients who underwent preoperative colonoscopy had a typical smooth submucosal mass. At pathology, 97.9, 13.7, and 5.3% of schwannomas stained positive for S100, vimentin, and GFAP, respectively. The median mitotic index was 1/50.Colorectal schwannoma is a very rare subtype of gastrointestinal schwannoma which occurs in the elderly, almost equally in men and women. Schwannoma should be included in the differential diagnosis of a submucosal lesion along with gastrointestinal stromal tumor, neuro-endocrine tumors, and leiomyoma-leiomyosarcoma. Definitive diagnosis is based on immunohistochemistry of the operative specimen. Rarely malignant, surgery is the mainstay of treatment. Schwannomas of the gastrointestinal tract are spindle cell tumors originating from peripheral nerve lining Schwann cells and represent a very rare entity, accounting for approximately 2–6% of all mesenchymal tumors , 2. In fThe aim of this study is to report an incidental finding cecal schwannoma with a systematic literature review of colorectal schwannoma, to describe the clinical, diagnostic , pathologic, and prognostic features of colorectal schwannomas.We performed a systematic review of the literature using three databases . In the PubMed search, we used the following search terms:  AND “Neurilemmoma”[Mesh] with filter for case reports and  AND “Neurilemmoma”[Mesh] AND case series. Free terms included  AND Schwannoma and  AND Schwannoma AND case series. In the Scopus database search, we used the terms (TITLE-ABS-KEY ({case report})) AND (TITLE-ABS-KEY(schwannoma) AND TITLE-ABS-KEY) OR ((TITLE-ABS-KEY(schwannoma) AND TITLE-ABS-KEY) AND ({case series})). In the Cochrane search, we used the following: Title-abstract-keywords: Schwannoma and Title-abstract-keywords: Neurilemmoma. The search strategy had no publication date, or publication type restriction. In addition, the reference lists of relevant reviews or included articles were also searched to find other eligible studies. We included only studies published in English.Study characteristics such as patient age, sex, and presenting symptoms, diagnostic exams, tumor characteristics , timing of diagnosis (endoscopic biopsy or post-operatively), type of resection , tumor immunostaining , and mitotic rate reported in the pathology report, lymph node status, and the degree of aggressiveness of the tumor were evaluated in the literature review.An asymptomatic 70-year-old female known to have surgically treated squamous cell carcinoma of the nose underwent a routine screening colonoscopy that revealed an uncomplicated diverticulosis and a cecal submucosal mass Fig. . The appAfter multidisciplinary team discussion, a differential diagnosis of mesenchymal tumor of the colon  was suggested and we decided to perform an exploratory surgery. The patient was consented for open exploration by mini-laparotomy and possible right hemi-colectomy. The right colon was mobilized at the white line of Toldt, the 3 cm white cecal mass was well circumscribed, and a wedge resection, including the appendix, using GIA 75  was performed. The operative specimen was sent for frozen section at pathology. The temporary diagnosis was a benign spindle cell tumor. The intra-operative decision was to wait for the definitive histopathologic examination report in order to try to avoid a right hemicolectomy. The final pathology report revealed a benign spindle cell tumor that stained negative for CD117 and DOG-1 and was diagnosed as cecal schwannoma with a reactive lymph node Fig. .Fig. 4a The post-operative course was uneventful and the patient started oral feeding the same night and was discharged on day 5 with pain killers. The final multidisciplinary committee decision was follow-up without further treatment needed. At 1-year follow-up, the patient is disease free.A total of 521 articles were identified from the PubMed, Scopus, and Cochrane databases. After removing duplicated articles, 230 articles remained for further assessment. A total of 171 articles were excluded on the basis of the titles and the abstracts. Of the remaining 120 articles, only 78 articles had full text published in English. After full text review of these remaining articles, 70 were eligible and included in the systematic review  and 56 female (58.3%) patients with a mean age of 61.2 years (range 14 to 95 years). Thirty-five patients (36.4%) were asymptomatic at presentation, including eight with positive fecal occult blood test (8.3%). The most common presenting symptoms were rectorrhagia (22.9%) followed by nonspecific abdominal pain (15.6%), constipation (7.3%), tenesmus (7.3%), and increased abdominal girth (2.1%).Schwannoma occurred most frequently in the cecum and right colon (30.5%) followed by the sigmoid (28.1%), the rectum (21.1%), the left colon (8.3%), the transverse colon (5.3%), and the appendix (1.1%). The tumor size ranged from 0.3 to 28 cm with a mean of 3.78 cm (median 3 cm).On colonoscopy, results were available in 73 out of 96 patients (76%). Schwannoma had the typical submucosal mass appearance with smooth mucosal surface in 41 (56.2%) and with ulcerated mucosa in 5 (6.8%). Fifteen patients (20.5%) had a tumor described as submucosal polyp, and nine patient tumors (12.3%) were described as a mass, either fungating (8.2%) or polypoid (4.1%).Pre-operative tumor imaging results were available for 46 patients (48%). The most common imaging done was abdominal CT scan . In the majority, the CT scan report lacked a description of the schwannoma and identified a colorectal mass. In 12 cases (34.3%), the schwannoma was described as a well-circumscribed homogenous lesion with low enhancement on arterial phase and in one case as a well-circumscribed homogenous lesion without arterial phase enhancement. Endorectal ultrasound (EUS) results were available for only seven patients (7.3%) and all of these showed a transmural hypo-echogenic mass. FDG-PET/CT scan was done in only four patients (including our patient) and showed a hypermetabolic lesion in three of them.The diagnosis of schwannoma was made on the operative specimen in the majority of patients (74%), on endoscopic or transanal biopsy in 23 patients (24%), and diagnostic method was not reported in 2% of cases.All patients with no biopsy or inconclusive biopsies underwent radical colonic resection either open (60.4%) or laparoscopic (11.5%). Three patients were diagnosed with schwannoma pre-operatively and underwent wedge colonic resection. Fifteen patients had colonoscopic resection at their initial examination and resection was judged sufficient and they did not undergo further treatment. Transanal surgery was performed in seven patients with rectal schwannomas including two treated by transanal microsurgery.high-power fields). Lymph node status was available in only 11 pathology reports (11.5%) and was negative in all of these. Schwannoma was judged to be benign in 93 out 95 patients (96.9%), and local and hepatic pattern recurrences were observed in 3 patients (3.1%) and were reported as “malignant” schwannomas [On pathologic examination, the Antonini subtype was available or deduced in 26 out of 96 patients, 57.7% were type A (14 patients), half of the remaining were type B, and the other half were both types A and B. In all available immuno-histologic examinations, schwannomas stained positive for S100 in 97.9%, for vimentin in 13.5%, for glial fibrillary acidic protein (GFAP) in 5.2%, for CD34 in 2.1%, and for CD68 in 1%. Tumor mitotic activity results were reported in only 41 patients (Ki-67 in 5 patients (5.3%) the highest below 5%, MIB-1 in 6 patients (6.3%) the highest < 5%, and low mitotic count in 30 (31.6%) with a mean of 2.1/50 and a median of 1/50 wannomas , 71.Schwannomas are extremely rare tumors of the nerve sheath, developing from Schwann cells. In the gastrointestinal tract, they present as spindle cell tumors, originating from Auerbach’s myenteric plexus rather than Meissner’s submucosal plexus, and account for approximately 2–6% of all mesenchymal tumors .Colorectal schwannoma is a very rare neoplasm and is the least frequent location for a GI schwannoma , 2. BaseFor differential diagnosis of schwannoma, abdominal CT scan can help differentiate between schwannoma and other mesenchymal tumors. Schwannomas appear as well-defined homogenous mural masses with low enhancement , 51, 70 In this review, four patients (4.16%) underwent metabolic imaging (FDG-PET/CT scan). Schwannoma exhibited hypermetabolic activity in three of these (75%) and no metabolic activity in one of them (25%). In the majority of cases, FDG-PET/CT scan was performed in the preoperative work-up of an atypical colorectal mass to differentiate between a malignant and a benign lesion, as in our case report. In patients with and without metabolic activity, all lesions were reported as benign. Although reported data are limited, current data do not suggest a role for FDG-PET/CT scan to differentiate between benign and malignant gastrointestinal schwannoma. In addition, octreotide receptor PET/CT scan can help to exclude the diagnosis of NET, as in the present report.The definitive diagnosis is made on immunohistopathologic examination of the operative specimen. Macroscopically, schwannomas tend to be lobulated well-defined tumors ulcerating into the mucosa . FurtherTwo histological growth patterns have been described: Antoni A, characterized by the dense growth of fusiform cells compactly arranged in palisades to form Verocay bodies and Antoni B in which the fusiform cells are more loosely distributed with rounded or elongated nuclei, with a great quantity of myxoid stroma and xanthomatous histiocytes. Recognition of these patterns has proved useful in the histologic identification of schwannomas .Colorectal schwannomas are reported as benign in more than 98% of cases. They are characterized by a low rate of mitosis, the absence of atypical mitotic figures, and nuclear hyperpigmentation. The degree of aggressiveness depends on the Ki-67 index and the mitotic index. Ki-67 index is recommended as an indicator of malignancy. A value of more than 5% correlates with greater tumor aggressiveness and a value of more than 10% is considered malignant . A higheThe best therapeutic option is complete surgical resection with free negative margins. Radical surgery is not necessary. In reported cases, the observed high frequency of radical resection is due to the absence of accurate preoperative diagnosis. When diagnosed preoperatively, schwannomas were resected either endoscopically or by a wedge resection , 56. AccOur review was limited by the small number of published case reports and the low index of pre-operative suspicion which resulted in somewhat hazardous diagnostic examinations. Most patients either did not undergo an abdominal imaging modality or lacked a detailed description of imaging results. Moreover, mitotic index was not calculated in all patients.In conclusion, schwannoma of the gastrointestinal tract is a rare, usually benign, tumor, and colonic schwannoma is an even more rare occurrence. Differential diagnosis of a submucosal lesion should include schwannoma as well as GIST, NET, and leiomyoma-leiomyosarcoma. Submucosal or deep biopsy might help to make a pre-operative diagnosis. Contrast-enhanced CT scan with low enhancement could help differentiate the well-defined schwannoma from other mesenchymal tumors or adenocarcinoma and exclude distant metastasis in cases of other diagnoses. The definitive diagnosis is based on immunohistochemistry of the operative specimen. Schwannoma stains strongly for S100, and the mitotic index should be calculated to help differentiate benign from malignant lesions. Surgery is the mainstay of treatment, and as for other mesenchymal tumors, wedge resection rather than classic regional resection is advised.


Osteoid osteoma is a benign bone-forming tumour, which very unfrequently has multifocal or multicentric presentation. We report the first known case of a multicentric, multifocal and recurrent osteoid osteoma treated using radiofrequency ablation.A 39-year-old man with two-year history of left hip pain was admitted at our Institution. The pain was more intense during the night and partially relieved by salicylates. Pelvis CT demonstrated two lytic lesions  with surrounding sclerotic reactive bone, both with a central focal area of high attenuation, located in the femoral neck and along the anterior portion of the acetabulum, respectively. Both lesions had clinical and imaging findings consistent with multicentric osteoid osteoma. Thus, the two lesions were biopsied – with pathologic confirmation of osteoid osteoma – and treated using radiofrequency ablation. Hip pain decreased but did not disappear, actually increasing a few months after treatment. CT and MRI were performed showing a smaller lesion (5 mm) with the same imaging features, surrounded by marrow oedema, along the posterior column of the acetabulum. The lesion was considered suspicious for osteoid osteoma, overlooked on previous examinations. Therefore, a diagnosis of multicentric and multifocal osteoid osteoma was established. The new lesion was again treated with radiofrequency ablation with symptom disappearance. However, hip pain relapsed after 18 months, and CT and MRI showed an osteoid osteoma recurrence on the posterior column of the acetabulum, which was biopsied and successfully treated using radiofrequency ablation.To our knowledge, this is the first reported case of multicentric, multifocal, recurrent osteoid osteoma. Our case report highlights the importance of considering a diagnosis of multifocal osteoid osteoma when dealing with multifocal lytic lesions of the bone and with pain persistence after treatment. It also emphasises the combined role of CT and MRI in this setting. It accounts for 10–13% of all benign bone tumours and 2.5–3% of all primary bone neoplasms [nidus –6.en-bloc resection, which are currently considered as invasive procedures. Currently, percutaneous radiofrequency ablation (RFA) is considered the standard of care [Although benign, OO is generally treated with the twofold purpose of removing the pain and to avoid growth disturbance in immature skeletons. Traditionally, treatment relied on surgical curettage or  of care , 7.OO can be missed on a plain radiograph, especially if small, obscured by reactive bone sclerosis, or located in difficult anatomic locations such as the spine or the pelvis. In this setting, computed tomography (CT) may be helpful in early identification of OO, being considered the imaging modality of choice to detect the nidus and later to guide the RFA procedure .Very unfrequently, OO can be multiple, presenting as multifocal or multicentric lesion . ‘MultifIn this paper, we report a case of a multicentric, multifocal and recurrent OO in a 39-year-old man presenting with 2-year history of left hip pain.A 39-year old male presented with a two-year history of spontaneous, intermittent, non-traumatic left hip pain. Symptoms were worsening at night, with no irradiation to the leg, fever, or other associated signs/symptoms. Pain was initially reported as mild, then it worsened and became unresponsive to non-steroidal anti-inflammatory drugs, especially salicylates. At first outpatient examination, diffuse tenderness of the hip was noted, with pain not changing with movement. No palpable swelling or decrease of muscular tone was observed. Hip range of motion was preserved, with negative flexion, abduction and external rotation (FABER) test. No neurological signs were found and Laségue maneuver was negative on both sides.The patient underwent pelvis and left hip plain radiography, which was unremarkable. Clinical suspicion of OO was raised and the patient underwent CT examination. It revealed the synchronous presence of an intracortical radiolucent nidus (7 mm) with central hyperdensity and mild sclerosis of the adjacent bone, located in the anterosuperior portion of the left femoral neck. A second lytic lesion (8 mm) with similar features was also seen, surrounded by denser sclerosis, located along the anterior column of the acetabulum. A diagnosis of multicentric OO was made Fig. . Thus, tTypically, OO is a solitary, intracortical lesion usually developing in long tubular bones , althougIn our case, the patient was affected by OO which was both multicentric, since it involved the femur and the acetabulum, and multifocal, due to its double localization in the anterior and posterior acetabulum. In addition, recurrence after treatment is another possible and well-known event after OO treatment, easily identified on the basis of the clinical scenarion (pain recurrence) and CT/MRI findings . Frequen“wake-up call” on MRI scans of patients with OO.In our case, the third OO located in the posterior acetabulum, which required re-treatment with RFA, was initially overlooked. This is related to the small size and non-specific features of the lesion. Also, this may also be due to a satisfaction of search error due to the identification of the other two lesions, which justified patient’s clinical conditions . NotablyPlain radiography is usually the first line examination, but may result inconclusive especially in cases of intramedullary lesions or tricky locations . The typical finding is a solid and sclerotic bone reaction, containing a lucent oval-shaped central nidus . CT alloSurgery of OO has been traditionally used as standard of care. Currently, RFA represent the best treatment option, allowing to treat the nidus by thermal coagulation through a percutaneous applicator, with well documented safety, and success rates higher than 90% , 18, 19.In conclusion, this is the first known case of both multicentric and multifocal OO, also associated with lesion recurrence. Our case report highlights the importance of considering a diagnosis of multifocal OO when dealing with multifocal lytic lesions of the bone and with pain persistence after RFA. It also emphasises the role of combined CT and MRI evaluation.


A tumourectomy was successfully performed, during which manipulation of the retroperitoneal tumour triggered a sharp rise in blood pressure. Histological analysis confirmed the diagnosis of a paraganglioma. The clinical complaints of headache, paroxysmal palpitations and night sweats disappeared postoperatively. This case is a classic presentation of a paraganglioma occurring in the organs of Zuckerkandl, a collection of paraganglia. The diagnosis should be suspected in the presence of a heterogeneous, hypervascular mass in the retroperitoneum and typical clinical symptoms of hypertension, headache and palpitations. Treatment involves surgical resection, after accurate preoperative management. Genetic counselling is required, allowing a personal and genotype-based follow-up.A 31-year-old West-African female attended our emergency department presenting with palpitations, headache, fatigue and night sweats during the last 2 weeks. Clinical examination revealed tachycardia and a painful, palpable infraumbilical mass. Ultrasound examination of the abdomen showed a smoothly rounded soft-tissue mass with a diameter of 5 cm. On contrast-enhanced CT, a prevertebral mass with intense contrast enhancement was seen, located caudal to the aortic bifurcation. On PET-CT, there were no distant  In the last 2 weeks, she had a sense of general malaise, fatigue and night sweats. Moreover, she also complained of palpitations and persistent headaches. In the emergency department, clinical examination revealed tachycardia and a painful, palpable infraumbilical mass.A 31-year-old female from West Africa was referred to the hospital by her general practitioner following a blood test showing inflammation and anaemia , inhomogeneous intense 18F-FDG uptake was seen in the lesion, with a high maximum standardized uptake value (SUV) of 12.2. There were no distant 18F-FDG-avid locoregional nodes or masses.To further differentiate and rule out metastatic disease, a positron emission tomography (PET)-CT was performed . One houBased on the retroperitoneal location, the differential diagnosis is limited to neuroendocrine tumours , soft-tissue tumours and lymphadenopathy.Because of the paravertebral location, neurogenic tumours must be considered. Among neurogenic tumours, schwannomas are the most common tumour of peripheral nerves. Although a schwannoma is also a well-defined mass, diagnosis is less likely because there is no continuity with the adjacent neural foramen. In case of a neurofibroma, we would expect a hypodense mass with only minimal or no contrast enhancement. This does not fit the presented case.A liposarcoma, one of the most common primary retroperitoneal neoplasms, typically presents itself in a different way. A varying amount of fat and soft tissue, and multiple septae are expected. Leiomyosarcoma is another frequently encountered primary retroperitoneal sarcoma and large tumours have extensive areas of necrosis and occasional haemorrhage. Malignant fibrous histiocytomas are also large, moderately hypervascular, and heterogeneously enhancing soft-tissue masses with areas of necrosis and haemorrhage. However, we exclude both leiomyosarcoma and malignant fibrous histiocytoma due to the lack of invasion of adjacent organs. Lymphomas are presented as a lymph node mass. Due to their hypovascular nature, minimal contrast enhancement is seen. Often there is encasement of the large retroperitoneal vessels. Moreover, blood analysis can present associated abnormalities.Given the age of the patient, the clinical presentation, and the morphology and location of the mass, the preferred diagnosis was a retroperitoneal paraganglioma.A laparotomy was performed to remove the lesion. The tumour was situated closely to the left ureter and left iliac artery, but there was no invasion. After ligation of the immediate vasculature, the well-defined tumour could be removed completely. During the operation, manipulation of the tumour triggered a sharp rise in blood pressure. After removal of the tumour, the blood pressure remained within normal limits. The clinical complaints of headache, paroxysmal palpitations and night sweats disappeared postoperatively. Although not measured before surgery, the plasma-free metanephrines were negative several days after the tumourectomy.Pathology revealed a firm, tan mass, measuring 6 by 6 by 4 cm. Macroscopy showed a solid white surface with haemorrhagic zones and microcysts . The his1 Their prevalence has been estimated between 1:6500 and 1:2500.Paragangliomas are rare neuroendocrine tumours, arising from the paraganglionic system composed of neural crest cells. Tumours that arise from the neuroendocrine cells of the adrenal medulla (80%) are referred to as pheochromocytomas, those that are found in an extra-adrenal location (20%) are called paragangliomas.4Up to now, 14 different genes have been reported, with different genotype- phenotype presentation and risk of malignancy and recurrence. These familial tumours can also be associated with multiple endocrine neoplasia syndrome and neuroectodermal syndromes; for example., tuberous sclerosis, Type 1 neurofibromatosis and von Hippel-Lindau disease.5Paragangliomas mainly affect adults in their fourth or fifth decade of life. There is no sex predilection. They may occur isolated or as part of a hereditary syndrome. Up to 30% of patients with a paraganglioma have disease-causing genetic mutations, inherited in an autosomal dominant manner.5 A typical location is in the organs of Zuckerkandl, a collection of paraganglia located at the bifurcation of the aorta or at the origin of the inferior mesenteric artery. Forty percent produce high levels of catecholamines, which results in symptoms such as headache, palpitations, excessive sweating and hypertension.1 Approximately 10% of the paraganglioma are clinically silent, presenting as an incidental finding.5The majority of extra-adrenal tumours are seen in specific locations in the neck, chest and abdomen.When there is clinical suspicion for a catecholamine-secreting tumour by physical examination and clinical history, gathering biochemical evidence is recommended. Plasma free metanephrines or 24 hr urinary fractionated metanephrines and catecholamines are measured for the evaluation of catecholamine hypersecretion.Either CT or MRI is recommended for initial tumour localization. Contrast CT is the primary imaging modality, given its excellent spatial resolution and good sensitivity (88–100%), but as with MRI, it lacks specificity (70–80%).5On contrast-enhanced CT scans, a paraganglioma appears as a large well-defined soft-tissue tumour with intense contrast enhancement due to their hypervascular nature. Areas of necrosis may be seen in the tumour, as well as punctate calcifications or focal areas of high attenuation caused by acute haemorrhage. The key to diagnosis is the location of the mass; when a hypervascular mass at the bifurcation of the aorta is found, the possibility of a paraganglioma should always be considered.T1 weighted MR images, these tumours are usually hypointense or isointense compared to liver parenchyma. On spin-echo sequences, signal voids can be seen, resulting in the characteristic “salt and pepper” pattern. On T2 weighted images, a paraganglioma presents as a markedly hyperintense mass, but the tumour often displays complex and heterogeneous signal intensity due to haemorrhage.On Only the presence of distant metastases allows differentiation of malignant from benign paragangliomas on imaging. Patients with large size of tumours, extra-adrenal or recurrent disease have an increased risk for distant metastasis. For detection and follow-up, nuclear medicine is indicated.4  Most paragangliomas show uptake of 18F-FDG in PET imaging and even benign tumours can be highly 18F-FDG-avid.6 Aggressiveness and dedifferentiation alone do not explain the high 18F-FDG uptake, this in contrast to other neuroendocrine tumours, for example. thyroid cancers and endocrine pancreatic tumours. Specific genetic defects, tumour localization and malignant potential may all play a role in the degree of 18F-FDG uptake. However, the exact mechanism for the molecular changes is yet to be determined.Progress in PET imaging allows for reliable early detection of metastatic disease, with a sensitivity of 74–100%.68Ga-DOTA-coupled peptides DOTATATE, DOTANOC and DOTATOC. Especially 68Ga DOTATATE PET-CT has been shown to have a higher sensitivity and uptake intensity compared to all other imaging modalities, and is a suitable first line investigation when a paraganglioma is suspected.7Paragangliomas express somatostatin receptors, enabling imaging with somatostatin receptor PET-tracers such as the Microscopically, all paragangliomas have a similar morphological appearance. They are highly vascularised and the cells that produce catecholamines are intimately related to the capillaries. In the presented case, the cells were highly immunoreactive for the neuroendocrine markers CD56, synaptophysis and neuro specific enolase (NSE).1 In up to 10% of patients, metastases are already present at the time of diagnosis.4Paragangliomas are more aggressive tumours compared to their adrenal counterparts, with 22–50% versus 2–10% developing metastases.3 This emphasizes the role of the radiologist in the preoperative diagnosis, in order to notify the treating physician.Treatment involves surgical excision to reduce the symptoms of excess catecholamine. The decision for laparoscopic or open resection depends on the tumour size and location, as well as on the surgeon’s preference and expertise. Before surgery, patients with a hormonally functional tumour should undergo preoperative blockade to prevent perioperative cardiovascular complications, with α-adrenergic receptor blockers as the first choice.3 Patients require long-term periodic clinical and imaging follow-up, because metastatic disease or recurrence can appear even after decades free of disease.Molecular genetic testing should be considered in all patients with a paraganglioma. Based on the results, decisional algorithms are available for imaging follow-up.The possibility of a paraganglioma should be considered in the presence of a retroperitoneal, enhancing mass at the bifurcation of the aorta and typical clinical symptoms of hypertension, headache and palpitations.The diagnosis is suggested based on a careful history, physical examination, biochemical and imaging studies.68Ga DOTATATE PET-CT is a newer and more specific imaging modality, suitable for first line investigation when a paraganglioma is suspected.The There is an important role for the radiologist in the preoperative diagnosis, in notifying the treating physician to prevent perioperative catecholamine excess and complications.Treatment involves surgical resection and the final diagnosis is made by pathological examination of the resection specimen.Genetic counselling is not only important to predict the malignancy and recurrence risk, but also for personalized, genotype-based management and follow-up.Written informed consent for the case to be published  was obtained from the patient(s) for publication of this case report, including accompanying images.


Tumefactive multiple sclerosis (TMS) is a rare entity which can be difficult to diagnose unless definitive diagnostic measures are taken. TMS is characterized by solitary or multiple lesions that are sized > 2 cm, with/without mass effect, edema, and ring enhancement on magnetic resonance imaging (MRI). The demyelinating lesion can mimic infections, vascular lesions, and inflammatory lesions. The clinical presentation is highly dependent on the area of the brain which is affected, and this can lead to a variety of signs and symptoms. Herein, we present the case of a 40-year-old immunocompetent female with a history of right-sided numbness of her face, arm, and leg associated with muscle weakness for about a week. Workup included an MRI showing ring-enhancing lesions in the white matter of the brain, zero oligoclonal bands in the CSF, a normal immunoglobulin G (IgG) index, and an elevated myelin basic protein (MBP) in the CSF. A biopsy was obtained that showed predominant macrophage infiltrate with loss of myelin but the preservation of axons. Suspecting a demyelinating pathology, the patient was informed that she would be started on intravenous dexamethasone for an eight-day course. With subsequent completion of this course in the hospital, the patient was discharged on oral prednisone daily for a month and a referral leading to a definitive diagnosis of TMS. The patient was started on interferon beta-1a and subsequently relapsed due to noncompliance. However, further workup showed a reduction in the mass-like lesions and a response to therapy. If suspicion for TMS is high despite workup, steroids can be used with immunomodulators in the interim to combat symptoms and potentially reduce lesions and potentially subvert the need for biopsy. Neurological symptoms require a physician to take into consideration many differentials ranging from vascular, metabolic, to infectious causes. Multiple sclerosis (MS) is a chronic inflammatory demyelinating disease known for relapsing and remitting episodes . TumefacTMS as an entity is a demyelinating disease, and it tends to present as a space-occupying lesion that has tumor-like effects . DiagnosWhile the prognosis is mostly benign in nature, the symptoms typically remit with doses of high-intensity steroids . In addiA 40-year-old Caucasian female presented to our facility with a five-day history of right-sided numbness. She was referred to us from a small area hospital with a concern for a mass following computed tomography (CT) of the head. The CT report showed a 2 x 1.5 cm hypodense lesion adjacent to the posterior left basal ganglia/lateral thalamus and deep white matter adjacent to the posterior left lateral ventricle , T1-weighted (T1W), and T2-weighted (T2W) images  analysis for aerobes and anaerobes, toxoplasma, Cryptococcus, fungal, and human immunodeficiency virus (HIV) infections. All studies proved to be negative. CSF workup for multiple sclerosis displayed zero oligoclonal bands. CSF concentration of myelin basic protein was elevated with a value of 7.1 (0.0 - 0.12 ng/ml), in addition to cytology results revealing mature lymphocytes. CSF protein and antibody analysis yielded insignificant results and included a normal IgG index. A serum IgG of 564  and a CSF IgG being 1.4 (0.0 - 8.6 mg/dL) were obtained and were unremarkable. This further directed the need for a biopsy of the left thalamic lesion.Histologic sections demonstrated a predominant macrophage infiltrate that was relatively demarcated from the surrounding white matter. The immunohistochemical stains performed at an advanced medical center demonstrated olig2 to label glia, which can be interpreted as promoted recovery. A myelin stain  showed loss of myelin with relative preservation of the axons on neurofilament protein (sm31). Oncologic markers were negative. Findings were significant for a macrophage-rich lesion and suggestive of demyelinating disease.After ruling out infectious and malignant conditions, we ultimately determined this to be an unknown inflammatory condition with a strong suspicion of multiple sclerosis (MS) at the forefront of possible etiologies. The patient was started on an eight-day course of intravenous dexamethasone, 4 mg every six to eight hours. The course of intravenous dexamethasone was completed in the hospital, and the patient was discharged to rehab with a month of per os (p.o.) prednisone, 20 mg daily, followed by a taper. She was also referred to a specialized medical center for MS confirmation, where they were better able to confirm the diagnosis as complex tumefactive multiple sclerosis . As a result, the patient was started on interferon beta-1a which was taken three times per week via subcutaneous injection.The patient subsequently returned to our facility for a similar episode four months following the initial presentation but with the now confirmed diagnosis of TMS. This was assessed as a relapse due to noncompliance of weekly injections. On imaging, MRI showed a reduction in the mass-like lesions Figure , leadingDemyelinating pathologies of the central nervous system are regularly encountered, with MS being the most common . ConventBefore considering biopsy, imaging plays a crucial role in distinguishing cerebral lesions. MRI is considered the gold standard neuroimaging modality for a TMS diagnosis. Characteristically, these lesions are hyperintense, over 2 cm in size, and show ring enhancement with gadolinium -13. The Another diagnostic modality that can help differentiate neoplasms from TMS lesions is perfusion imaging. This modality measures the mean relative blood volume within the lesion. TMS lesions will show significantly less relative blood volume compared to neoplasms, but this may be inconclusive because of the variability in different types of tumors, especially in glioblastoma multiforme and primary CNS lymphoma. TMS lesions have contrast enhancement with gadolinium on MRI in 95% - 100% of patients with varying patterns, such as open or closed rings, diffuse, concentric, homogenous, or punctuate pattern. The ring enhancement in TMS lesions represents an advancing area of active inflammation away from the non-enhancing core, which is indicative of chronic inflammation .Diagnostically, in addition to imaging, CSF analysis plays an important role to help distinguish TMS lesions from other pathologies. The main features typically include an elevation in the IgG index and oligoclonal bands (OCBs). Literature has illustrated that OCBs are present in 90% of patients with pre-established MS compared to 52% of patients who present with tumefactive demyelination as the first event . With thAccording to a retrospective study by Altintas et al., treatment with high-dose IV steroids resulted in 82.7% of cases showing a significant response with regression of lesions in 76% of cases . In our TMS is a rare variant, and in our case, it showed that with unclear diagnostic imaging, a normal IgG index, and negative OCBs, the differential of TMS couldn’t be excluded. As a result, a biopsy to attain a histopathological diagnosis was required in this case. However, multimodal imaging approaches may help distinguish tumor presentations from their mimics and avoid biopsy in some cases. If clinical suspicion is high despite workup, steroids can be used with immunomodulators in the interim to combat symptoms, reduce lesions, and potentially subvert the need for biopsy.


Gallbladder cancer commonly spreads by direct extension to the liver and adjacent organs of the gastrointestinal tract. Ovarian metastases by biliary origin, though known, are a very uncommon finding.We report a rare metastatic localization by gallbladder cancer, Krukenberg tumor mimicking a primitive ovarian cancer. A comprehensive critical review was performed and suggested strategies were analyzed.The prognosis of ovarian metastastes by biliary origin is very poor with an overall survival estimated at around 6 months. The variable clinical presentation, radiology and serum markers make the appropriate histological diagnosis mandatory.The presence of Krukenberg tumor should be considered in the work-up of gallbladder cancer. •Gallbladder carcinoma is a rare malignancy with a significant source of mortality in selected areas of high incidence.•Krukenberg tumor by gallbladder cancer is a very uncommon finding with diagnostic difficulties for clinicians.•The variable clinical presentation, radiology and serum markers make the appropriate histological diagnosis mandatory.•The presence of Krukenberg tumor should be considered in the work-up of gallbladder cancer. In 2018, about 219 000 people were estimated to have been diagnosed with gallbladder cancer .However, 1–2% of surgical specimens demonstrated a gallbladder cancer as an incidental finding . Most gaHere, the authors describe an uncommon clinical presentation of gallbladder cancer with the presence of Krukenberg tumor by biliary origin, mimicking a primitive ovarian cancer in an academic medical center.2first case in man”, neither a new device or surgical technique was performed; for this reason, it was exempt from registering into the Research Registry.The work has been reported in line with the SCARE 2018 criteria . However3A 82 years-old multiparous Caucasian woman was referred by family physician as she presented abdominal distension and pain and a palpable mass in the right iliac fossa. The past medical history included essential hypertension under pharmacological treatment and gastro-esophageal reflux. She denied any alcohol abuse or smoking. Blood tests showed CA 125 277 U/mL (reference range 0–36 U/mL) and CA 19.9 64.5 U/mL (reference range 0–37 U/ml). An abdominal ultrasonography and computed tomographic (CT) scan were obtained. Abdominal ultrasonography identified a heterogeneous, multi-septated mass with solid components occupying whole pelvis and a sclerotrophic gallbladder with wall thickening and biliary sludge. Abdominal CT-scan confirmed the thick-walled gallbladder close to the duodenum and the ovarian mass presenting hypodense areas with necrosis and calcifications with 17 × 11.5 cm in size, as shown in 4Gallbladder carcinoma is a rare malignancy, but in selected areas of high incidence, such as India, Chile and Japan, it is a significant source of mortality . The diaFriedrich Ernst Krukenberg, a German physician who reported what he thought was a new type of primary ovarian malignancy in 1896, a fibrosarcoma of the ovary [The ovarian metastatic localization by biliary origin is a very uncommon finding in everyday clinical practice. Krukenberg tumor refers to a malignancy in the ovary that metastasized from a primary site, classically the gastrointestinal tract, although it can arise in other tissues. Gastric adenocarcinoma represents the most common source. Krukenberg tumours are often found in both ovaries, consistent with its metastatic nature. Krukenberg tumours are named after he ovary ; six yeahe ovary  reportedhe ovary . The highe ovary . Kim SH he ovary  also sugHarcourt and Dennis  emphasiz5The most important learning points are represented by the difficulty of clinical and radiological diagnosis of ovarian metastases, which may mimic a primary ovarian cancer, the enormous importance of histopathological evaluation and the need of multidisciplinary approach in order to avoid certain pitfalls. Indeed, the occurrence of Krukenberg tumor should be considered in the work-up of gallbladder cancer, by keeping in mind this rare occurrence in clinical practice.This study is exempt from ethical approval in our institution.This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.Pesce A and La Greca G designed the paper, Pesce A performed review, Amore FF and Magro G performed histological analysis and provided histological pictures; Pesce A and Li Destri G wrote the paper, La Greca G and Puleo S supervised the paper; all the authors read and approved the final manuscript.first case in man”.Not applicable. This case report is not the “On behalf of all authors, I am the guarantor who accept full responsibility for the work.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Not commissioned, externally peer reviewed.All the authors declare that they have no competing interests.


A 74-year-old woman presented with a vague suprapubic pain and progressive deterioration of her general condition. CT examination showed a partially air filled bladder with disruption of the bladder wall on the right posterolateral side. There was no intra-abdominal free air or free fluid. The bladder was slightly thickened Figure . In LungMost cases of bladder rupture are traumatic. Spontaneous rupture is much more rare. Risk factors of spontaneous bladder wall rupture are urine retention due to diabetes, neurologic bladder, prostatic hyperplasia, history of pelvic radiotherapy or recurrent infections. There are also cases reported of bladder rupture due to binge drinking and due to obstructed labour. Bladder diverticula are an extra risk factor because they are responsible for an additional weakness of the bladder wall. In our case, an emphysematous cystitis caused bladder wall weakening and secondary rupture.Patients with bladder rupture can present with haematuria, anuria, suprapubic pain and fever. Rupture can be divided into intraperitoneal (10–20% of cases) or extraperitoneal (80–90% of cases) depending on the site of disruption. Intraperitoneal rupture is associated with an acute abdomen. Our patient had an extraperitoneal rupture that was limited to perivesical space.Intraperitoneal rupture needs open surgery while most extraperitoneal rupture can be treated conservatively with placement of Foley catheter. Cystography can identify the type and extend of rupture. Nowadays, CT cystography is the procedure of choice, especially in an emergency setting.Emphysematous cystitis (EC) is a relatively rare variant of a urinary tract infection caused by gas-forming microbes. E. Coli is by far the most common isolated urinary pathogen, as in our case. It is typically seen in elderly women (60–70 years of age). Risk factors are the same as in uncomplicated cystitis like diabetes mellitus, neurogenic bladder and urinary stasis due to bladder outlet obstruction. The clinical course can range from asymptomatic to severe sepsis. Abdominal pain, dysuria and haematuria are most common. EC can be diagnosed radiologically by conventional radiography as a curvilinear increased radiolucency in the bladder wall. Intraluminal gas in the bladder will be seen as a dynamic air-fluid level. The mucosal surface may have a cobblestone or beaded ‘necklace’ appearance reflecting the irregular thickening produced by submucosal blebs as seen at direct cystoscopy. In our case we could also see these blebs on CT using a lung window. Furthermore, CT is also useful to rule out associated findings such as pyelonephritis, intra-abdominal abscess, malignancy or other causes of intraluminal gas such as an enterovesical fistula. EC may be suggested on ultrasound when dirty shadowing of the bladder wall is seen. Therapy of EC is similar as treatment of extraperitoneal bladder rupture: hospitalization with intravenous antibiotics, bladder drainage with Foley catheter and handling of predisposing conditions.Spontaneous rupture of the bladder wall is rare, especially due to emphysematous cystitis. Clinical presentation can range from asymptomatic to severe sepsis, so a high degree of suspicion during diagnosis is essential, especially in patients with some cause of urine retention. Findings at initial CT can be negative, so CT cystography is the modality of choice to rule out a tear and allow accurate diagnosis of the type of bladder wall rupture.


Parotid tuberculosis remains a very rare localization in the Department of Otolaryngology and Cervico-Facial Surgery (ENT) sphere. It is presented in the form of a deceptive clinical picture causing confusion with other pathologies of the parotid gland, including tumor pathology. In addition, its lack of knowledge by practitioners increases the risk of missing the diagnosis. Often, the diagnosis is a histological surprise on a piece of excision after an exploratory parotidectomy. However, its treatment is primarily medical if the positive diagnosis is well established. We report medical observation of two new cases aged 44 and 45 respectively, who consult our center for parotid swelling. Radiological examinations were in favor of intraparotid cystic lesions. Both patients benefited from an excision whose histopathological study was in favor of primary parotid tuberculosis. The subsequent evolution was favorable under antituberculous treatment. Tuberculosis is a chronic granulomatous infection caused by Mycobacterium tuberculosis or bovis that can affect all organs. Still a public health problem in Morocco, lung localization remains the main problem. At the level of the ENT sphere, lymph node involvement is the most frequent. The isolated location of tuberculosis in the salivary glands, especially the parotid gland, is extremely rare. There is no clinical, radiological or biological evidence to guide the diagnosis .Observation 1: this was a 44-year-old woman, followed by iron deficiency anemia under iron supplementation, who developed an isolated swelling of the right parotid region, evolving for 5 months. Clinical examination: The patient was in good general condition, apyretic. The tumefaction was pretragal and measured 3cm of long axis, firm, well limited, mobile and painless, associated with multiple, hard and fixed homolateral jugulodigastric adenopathies. The controlateral parotid was with no particularities. There was no chewing discomfort, no trismus or endobuccal pus exit, no peripheral facial paralysis, and the upper aerodigestive tract was free. At Parotid MRI (otid MRI , the lesotid MRI . Anatomootid MRI  concludeObservation 2: a 45-year-old woman, with no notable pathological history, consulted in our facility for a left sub-angulo-mandibular mass evolving for approximately 4 months. There were slight inflammatory signs nearby, particularly a slight redness with pain and fever at 38°C without weight loss, chewing discomfort, trismus, or pus exit at salivary orifices. The young woman was in good general condition and had a swelling of the left parotid area, firm, painless, measuring about 4 cm in long axis, and fixed. There was no limitation of the mouth opening or peripheral facial paralysis. Examination of the oral cavity and the opening of the stenon canal did not reveal any inflammation or pus output. Examination of the cervical lymph node areas was normal. The rest of the ENT and general examination was without any particularity. A cervical ultrasound was done to show a tumor lesion, measuring 29 x 28 x 14 mm, of mixed echostructure with central liquid predominance at the lower pole of the left parotid. Parotid MRI showed a lesional process in the deep compartment of the left parotid gland, with low intensity T1 signal, and in the other hand, high intensity signal in diffusion sequence with peripheral enhancement, measuring 30 x 29 x 15 mm. The right parotid and submandibular glands, in addition to the thyroid are without signal or morphological abnormalities. This was in favor of a left parotid abscess ( abscess . The resParotid tuberculosis is a rare entity of extrapulmonary tuberculosis and particularly of extranodal ENT localization. After Robert Koch had discovered the bacillus responsible in 1882, the first case of secondary parotid tuberculosis was reported in 1894 by De Paoli, and Von Stubenrauch described a second case in 1893, which is of primary disease. Since then to the present day the literature has reported 220 cases. It is a pathology that can reach all ages. But often, the young adult between 20 and 40 years, remains more incriminated. The child is rarely affected. Both sexes, male and female, are equally affected. Ninety percent of published cases come from the African and Asian continents, countries where tuberculosis is a public health problem. Parotid localization poses a real differential diagnostic challenge for practitioners because of the non-specific clinical picture, and because many paraclinical examinations are non-conclusive .The cervico-facial MRI allows a good analysis of the parotid lodge and asserts the parotid nature of the lesion. It is considered superior to CT in the detection and analysis of a parotid tumor process. In the normal state, the parotid parenchyma shows a T1 signal more intense than the muscle but less than the peripheral fat. The glandular capsule appears fine and regular. In parotid tuberculosis, the lesion shows a low T1 signal, and high T2 signal, which can be delimited by a shell enhanced after injection of Gadolinium. However, this aspect is not specific. Other very frequent benign parotid tumors pose a problem of differential diagnosis, such as pleomorphic adenoma, which manifests itself by a low T1 signal and high T2 signal, always enhanced but sometimes heterogeneously ,6. A cheTuberculosis of the parotid gland is rare and can occur in different clinical forms. Often confused with neoplastic pathology. Since Morocco is a country where tuberculosis is a major public health problem, the ENT surgeon must evoke this diagnosis before any parotid swelling, and this requires knowledge of the epidemiological, clinical and paraclinical aspects allowing an early positive diagnosis before arriving at surgery. Since the antibacterial treatment alone is effective allowing the regression of the parotid swelling and the elimination of the tubercular focus; the use of parotidectomy for diagnostic and/or therapeutic purposes is no longer a hot topic.The authors declare no competing interests.