Giant cell reparative granuloma is a rare, locally benign tumor with an aggressive behavior resembling malignant neoplasm, originating mainly from the mandible and maxilla; however, it can originate from other sites, such as zygomatic and temporal bones, with a young adult female predilection.A 28-year-old female presented to the Department of Otolaryngology and Head and Neck Surgery with a history of a slowly enlarging swelling over the left parotid region for four months. Physical examination revealed a non-erythematous, non-tender, firm mass with no overlying skin changes. Fine needle aspiration cytology of the lesion revealed a multinucleated giant cell-rich tumor and the histopathological examination of an incisional biopsy from the mass confirmed giant cell reparative granuloma. Therefore, a total surgical excision of the mass with curating of the zygomatic and temporal bones was performed with uneventful postoperative course and regular follow ups for one year.A growing mass in the parotid region in a young adult female with no history of trauma should raise the suspicion of giant cell reparative granuloma. Histopathological examination is the definitive tool for diagnosis, and surgical excision is the treatment modality of choice in such cases. •Similar GCRG case has been only reported once in the literature.•This is the second case reporting a GCRG.•Young females with no history of trauma should raise suspicion of GCRG.•We must give more thoughts into etiologies causing such reported cases. On further questioning, there was no history of facial trauma or injury, family history nor smoking. The patient has not been on any drugs with no past psychological history and no genetic investigation was carried as it was irrelevant. Furthermore, the swelling was not related to any meal intake nor feeding problems and there were no constitutional symptoms reported. Upon physical examination, a 4 × 3 cm well-defined, non-erythematous, non-tender, non-fluctuant, firm swelling over the left parotid region with no overlying skin changes was seen. Moreover, no findings were noted on bimanual and ENT examinations and there were no palpable cervical lymph nodes. Therefore, a decision to take a Fine Needle Aspiration Cytology (FNAC) of the mass was made revealing the presence of multinucleated giant cells-rich tumor of uncertain malignant potential .Regarding radiological investigations, a Contrast-Enhanced Computed Tomography (CECT) scan displayed an ill-defined mass that measured 3.2 × 2.0 cm and infiltrated the superficial lobe of the left parotid gland. In addition, erosion of the left glenoid fossa, left temporal and zygomatic bones was noted with an exertion of a mass effect over the external auditory canal. Even though the mass was extended intracranially with bulging impression on the temporal fossa  resultinThe procedure was decided and was conducted by one otolaryngology, head and neck oncology and reconstruction surgeon of 10 years' experience and a second otolaryngology and head and neck surgeon with 7 years’ experience. Intraoperatively, a pre-auricular incision was made through modified Blair incision with extension superiorly to the hairline margin in which a posterior flap was then raised at the level of the sternocleidomastoid fascia. Afterwards, the greater auricular nerve branches and the external jugular vein were identified and preserved, and the plane was developed where a hard mass was found being attached closely to branches of the facial nerve. Furthermore, retrograde dissection was made medial to lateral while the facial nerve was monitored and observed carefully using a facial nerve monitor (NIM 4 ports). The mass was then found deeply attached to the brain dura. At this point, the temporal bone was visualized, and a neurosurgeon joined to facilitate the need for further intervention. However, after careful evaluation only small parts of the temporal bone was eroded along with part of the zygomatic bone while the dura was intact. Tachosil was then used to seal the exposed dura and testing for cerebrospinal fluid showed no leakage present. Regarding the eroded bones, they were curetted and smoothened using a Volkmann Bone Curette with a periosteal elevator. Complete harvesting of mass was made and all facial Mercer tested positive with no reconstruction needed.Regarding her postoperative course, the patient was taken to the surgical ward in a stable condition after placement of a drain with painkiller management regime of paracetamol, PRN oral narcotics as well as prophylactic antibiotic (co-amoxicillin) and demonstrated an uneventful and stable course; therefore, the drain was removed on the second day with minimum output. Additionally, the facial nerve was reassessed and was intact with no evidence of CSF leak on further examination. On the third postoperative day, the patient was discharged home in a good stable condition. Regarding the histopathological examination of the mass excised, it was 3.5x2.5 × 1 cm in size, infiltrating parotid tissue, zygomatic and temporal bones, muscle and fat without lymph nodes involvement, atypia, necrosis or increased mitotic activity of the cells. The final histopathology report demonstrated the presence of a mixture of multinucleated giant cells, osteoclast-like with more than 20 nuclei per cell. In addition, the stromal cells were found to be oval, spindle and round while satellite lesions were seen away from the main mass as a sign of infiltration of bony and fat tissues. Furthermore, thick walled vessels with endarteritis obliterans were seen at the periphery of the lesion . MeanwhiAccordingly, the oncology tumor board planned for follow-up only without any further interventions. Follow up was initially weekly post operatively for the first month, then monthly for the next 3 months. Afterwards, it became every 3 months for the following 6 months then every 6 months for 2 years, first surveillance scan was done 3 months postoperatively in form of an MRI and it was negative for recurrences followed by another MRI on a yearly basis for 2 years with future surveillance plan of having one MRI per year for the next 5 years.3In 1953, Jaffe first described GCRG as fibro-osseous dysplasia, located mainly in the mandible and maxilla and to a lesser extent in the temporal, zygomatic, ethmoid, sphenoid and hand bones ,7. Despi4GCRG occurs more frequently in children and young adult female. However, this is the second reported case in the literature to be reported arising from the parotid region. As more cases likely to be present, it is important to have such a rare pathology in mind in young adult females with no history of trauma presented with a slowly progressive swelling over the parotid region. Surgery is the mainstay of treatment for such cases.Written informed consent was obtained from the patient for publishing this report with the included radiological and histopathological images.No funding was received.All included authors contributed in writing, proofreading and referencing of this case report.Not commissioned, externally peer reviewed.The authors declare no conflict of interest.


Annular pancreas is a rare entity. It clinically presents with intestinal obstruction. We present a case of a young female with incomplete annular pancreas presented with recurrent acute pancreatitis. A 28‐year‐old woman presented to the Gastroenterology out ‐ patient department (OPD) with a history of recurrent episodes of mild pancreatitis for the last 2 years. Her physical examination was unremarkable. All the laboratory investigations were normal. The Magnetic Resonance Cholangio‐pancreatography (MRCP) report was suggestive of pancreatic divisum. The Endoscopic Retrograde Cholangio‐Pancreatography (ERCP) was performed, and pancreatogram showed the main pancreatic duct wrapping around the duodenum and going toward the spine Fig. ; suggestAnnular pancreas is a rare congenital malformation with an estimated incidence of 5–15 cases per 100 000 adults.


If successful surgery is the primary quest of any surgeon, unintentionally leaving behind surgical items in the operative field remains his most feared obsession. This rare but dramatic accident can lead to potentially fatal complications and turn both lives of the surgeon and the patient upside down. We present the case of a 29-year-old female patient who presented to the ER with three days history of severe diffuse abdominal pain associated with fever, biological inflammatory syndrome and well-tolerated iron deficiency anaemia. She had no past medical history except for a lower segment cesarean section 5 months ago. Abdominal MRI allowed the diagnosis of two gossypibomas responsible for two intra-abdominal collections. An emergency laparotomy allowed the removal of these foreign bodies and the management of their serious complications of intestinal perforation by the construction of a double intestinal stoma. The patient made a post-operative uneventful recovery. This observation emphasizes the need to raise the practitioner´s awareness about this differential diagnosis in every case of any poorly localized abdominal pain occurring after surgery. The term gossypiboma refers to any type of non-absorbable surgical swab mistakenly left inside the human body after surgery. It concerns primarily the abdominal cavity (56%) then the pelvis (18%) [1]. This iatrogenic condition may have unfortunate health consequences with severe morbidity for the patient. Its real incidence is difficult to estimate given its medico-legal [2] implications and it is reported to occur in 1/1000 to 1/1500 intra-abdominal procedures. The diagnostic delay added to the severity of the possible complications may jeopardize the vital prognosis.A 29-year-old female patient presented to the emergency room with a several month history of diffuse abdominal pain associated with episodes of diarrhea nausea and chills. Further investigations revealed a history of cesarean section via pfununstiel incision for fetal macrosomia 5 months ago. There was no history of any other previous disease or surgery. The patient claimed that few weeks following her delivery; she started to suffer from dull abdominal pain, vomiting and diarrhea. Her general physician attributed her symptoms to a puerperal infection and she was sent back home with an array of antibiotics and painkillers. Starting one month before admission, she had multiple episodes of melena totally neglected by the patient. On physical examination, her body temperature was 38.9°C. She had a blood pressure of 110/80 mm/Hg, a heart rate of 88 beats/min and respiratory rate of 16 breaths/ min. The abdomen was slightly distend and tender especially in the right and left iliac fossa without guarding.3 and a CRP rate of 180mg/l. It also showed an iron deficiency anaemia with a hemoglobin value of 9g/dl and a mean corpuscular volume of 69fL. Given the unavailability of the CT scan that day for technical reasons, an emergency abdominal MRI showed two well-circumscribed collections located in the right and left iliac fossa with spongiform pattern and entrapped air bubbles (Bi-digital examination did not find any abnormalities. Initial screening tests showed a biological inflammatory syndrome with a WBC count of 21,000 cells/mm bubbles . The pat bubbles  which be bubbles  and anot bubbles  having c bubbles . After tForgetting surgical material of cotton matrix in the operating field is most commonly described in emergency surgical procedures, those with prolonged operative duration and hemorrhagic interventions [3]. High body mass index has also been cited as another contributing factor [1]. Literature data corroborates our observation indicating that cesarean section alone exposes to this risk with an incidence of 17.9% [4]. Physiopathologically, the retained sponges or gauze left by mistake in the peritoneal cavity can induce an exudative inflammatory reaction. In case of secondary infection, these surgical materials and the surrounded foreign body reaction may turn into a real intra-abdominal abscess. It can also encyst in adherent tissue and become calcified. Aseptic fibrosis may in some cases give rise to an encapsulated mass that remains silent for years [3]. Sankpal et al. have described a case of a completely accidental discovery of a textiloma during a laparoscopic cholecystectomy following a cesarean section occurring 5 years earlier [3]. Longer delays in finding a textiloma after a cesarean section have been reported in literature. Abdulrehamn et al. reported another case of a textiloma discovered up to 15 years after a classical caesarean delivery [5].The clinical presentation lacks specificity and outside the framework of accidental discovery, the signs generally reflect a complication such as abscess, intra-abdominal collections, acute intestinal obstruction, acute peritonitis due to intestinal perforation or internal or external fistulas [6]. Chief complain of the patient is persistent abdominal pain. Clinical symptoms may include abdominal tenderness or guarding in case of peritonitis, fever, gastro intestinal disorders, alteration of the general condition, bleeding and a palpable abdominal mass [1]. The case presented in our observation highlights the ability of these foreign bodies to cause progressive erosion through the intestinal wall leading to perforation. In such a case, routine blood investigations may show a biological inflammatory syndrome. Trans-mural migration can mimic a pseudo-tumour responsible for gastro intestinal disorders, melena and anaemia, as it is the case in our patient. Hence, the danger of this incident, which could completely change the outcome of the simplest commonly performed surgical intervention and worsen its prognosis. It should be noted however, a case of complete trans-mural migration of a spontaneously expelled textiloma without damage by natural rectal route described by Butt et al. [7].The clinical sings are strongly helped by the radiological findings in order to establish a timely diagnosis [4]. The plain abdomen X-ray may be sufficient to detect the radiopaque marker if it is present on the textile used but with a false negative rate of 25% [4]. Abdominal CT is the best imaging modality that allows making the diagnosis, indicating the exact location of gossypiboma and precising its possible complications. It usually shows a highly suggestive typical image of a heterogeneous spongiform cystic lesion with a hyperdense line indicating the radio opaque ribbon [6]. A calcified “reticulate rind sign” may also be noticed in cases of long-lasting gossypiboma and represents a very useful characteristic radiological feature [8]. MRI is another diagnostic alternative and it shows a well-defined hypointense cystic lesion on T1 weighed images and hyperintense on T2 weighed images. However, it does not visualise the radiopaque marker usually lacking radio-magnetic characteristics and giving poor MRI signal [2].Consequently, the diagnosis seems difficult and it requires the contribution of multiple associated arguments. The history of a previous surgery, the type of the intervention performed, the biological inflammatory syndrome if present and above all the often very suggestive radiological data are the pillars of the diagnostic process. Once the diagnosis is made, the immediate removal of any gossypiboma associated with high specific level of intensive critical care is necessary to prevent more serious complications that could jeopardize the patient's life. Laparoscopy can be useful in some uncomplicated cases [3]. However, in the event of multiple digestive perforations, as in our case scenario, that can go unnoticed in laparoscopy, laparotomy seems a more reasonable choice. Admittedly, the best treatment remains prevention. Strict compliance with the rules of standard material counting protocols, coordination between members of successive surgical teams, the use of radio opaque marked surgical sponges, the use of modern means of retained sponge detection such as radio frequency or barcode technology help to rectify this human error and avoid its disastrous consequences [6].Despite the evolution of the health care system and the continuous modernization of the surgical techniques, gossypiboma is an iatrogenic complication that is still seen in every day´s surgical practice. The diagnosis is difficult because we refuse to admit it and even think about it given its implications for the surgeon´s reputation that mark his professional career for life. Nonetheless, human error still exists and it must be kept in mind justifying a timely radiological examination. Prompt surgical management is essential because any delay worsens the consequences for the patient and the surgeon. Increased vigilance must be shown over preventive measures and strategies aiming to improve safety that must be the subject of constant research.


Scleral buckling (SB) is usually considered an extraocular operation premeditated to have a low risk of sympathetic ophthalmia (SO). Here we report a rare case of presumed SO in a young female patient following SB.A nineteen-year-old female patient was referred for visual loss in her left eye due to macula off inferior long-standing rhegmatogenous retinal detachment (RRD). The best corrected visual acuity (BCVA) was 20/400 in the left eye. SB with 360 degrees encircling band, an inferior segmental tire with one spot cryoretinopexy at the break site, and subretinal fluid drainage was performed. BCVA was improved to 20/80 and the retina was totally attached 1 week after the operation. The patient referred to the hospital 6 weeks later with severe visual loss in both eyes as counting finger 1 m. Patient examination indicated bilateral multifocal serous retinal detachment (SRD) and vitreous cells. The patient, diagnosed with SO, received intravenous corticosteroid pulse therapy and mycophenolate mofetil for treatment. The inflammation was controlled and SRD resolved after a 5-day intravenous treatment without being relapsed after 6 months. Consequently, BCVA became 20/20 and 20/50 in the right and left eye, respectively, after 6 months. The findings of systemic workup were negative for any extraocular disease or systemic involvement.Since SB is a procedure without manipulating intraocular tissues, it is considered to impose a low risk for SO. This report presented SO occurrence after successful SB. Some factors may induce SO, including inciting the choroid and retinal pigment epithelium with cryoretinopexy or perforating for drainage. Sympathetic ophthalmia (SO) is a rare bilateral diffuse granulomatous panuveitis that may occur after surgery or trauma to one eye, as penetrating injury and exposure of the uvea . The incScleral buckling (SB) is usually considered an extraocular operation, a privilege over vitrectomy, and SO is presumed to have a very low risk after SB. Here we report an unusual rare case of SO following successful SB surgery which was combined with subretinal fluid drainage (SRFD) and cryoretinopexy.A nineteen-year-old female was referred for visual loss and superior visual field defect in her left eye (LE). Best corrected visual acuity (BCVA) was 20/20 and 20/400 in the right eye (RE) in the LE, respectively. The anterior segment examination was within normal limits except for the detection of Shaffer sign in the LE. The results of the fundus examination revealed macula off inferior long-standing rhegmatogenous retinal detachment (RRD), 3 h of proliferative vitreoretinopathy (PVR), retinal break, and lattice degeneration. The patient had no history of head or ocular trauma or any intraocular surgery. SB with 360 degrees encircling silicone band  and inferior segmental silicone tire  with one spot cryoretinopexy at the break site, and SRFD due to chronicity of the RRD was performed. During this process, SRFD was not inadvertent. Scleral thinning was performed after passing the band and tire. Moreover, the needle of polyester spatula suture  was used to drain the fluid after the cauterization of the choroid. BCVA was improved to 20/80 and the retina was totally attached 1 week after the operation. The patient was followed up receiving a topical antibiotic  and topical corticosteroid  for 3 weeks.The patient referred to the hospital with severe visual loss in both eyes 6 weeks later. On examination, BCVA of both eyes was counting finger at one meter and the anterior segment was within normal limits, except 1+ anterior chamber and anterior vitreous cells in both eyes. Bilateral multifocal serous retinal detachment (SRD) was obvious in funduscopy Fig. .Fig. 1FHere, we report a rare case of SO following uncomplicated SB surgery accompanied by cryoretinopexy and SRFD. In this regard, it is recommended that SO be taken into consideration in any case of bilateral panuveitis associated with multiple SRD and a history of penetrating ocular surgery.In several studies, the occurrence of SO has been reported after vitreoretinal surgery , 4–6. ItThe onset of SO symptoms after the operation usually occurs between 3 weeks and 6 months after surgery due to delayed hypersensitivity . RegardiIn a recent study performed by Tyagi et al., the incidence of SO following vitreoretinal surgery was estimated at 0.038% of all vitrectomy cases, and vice versa, 9% of all cases of SO had vitreoretinal surgery . In thisMost of the cases presented in studies –9 had siOne of the most important differential diagnoses of our case is related to VKH which is a common uveitis etiology with similar clinical and imaging findings with SO , 10. AltThe results of SO imaging, based on FAG, revealed multiple hyperfluorescent pinpoints leakages associated with late pooling resembling VKH and hypofluorescent foci during the early phase of angiography. Moreover, the late phase of FAG showed hyperfluorescence similar to acute posterior multifocal placoid pigment epitheliopathy . In our In conclusion, the diagnosis of sympathetic ophthalmia should be taken into account in any case of bilateral uveitis or bilateral SRD following scleral buckling with uveal tract violation, such as cryopexy or SRFD.


The incidence of small bowel tumors is comparatively rare when compared to colonic tumors. They comprise less than 10% of all gastrointestinal neoplasms. Gastrointestinal stromal tumor (GIST) is the most common mesenchymal neoplasm of the gastrointestinal tract; however it accounts for less than 1% of all gastrointestinal tumors.To report a rare case of jejunal gastrointestinal stromal tumor presenting as a life-threatening emergency in a young adult male who was treated in the General Surgery department in the Jordanian Royal Medical Services (JRMS), Amman, Jordan. PRESENTED CASE: We report a 59-year-old male who presented to our emergency department with severe generalized abdominal pain. The patient was in a hypovolemic shock. Abdominal examination revealed a massively distended, tender abdomen. Abdomenal CT scan with IV contrast showed significant fat stranding around the jejunum associated with pneumoperitoneum and free fluid. Exploratory laparotomy was emergently performed, revealing diffuse peritonitis secondary to perforated small bowel tumor. En bloc resection was performed. Histopathologic examination confirmed the presence of malignant gastrointestinal stromal tumor of the jejunum with R0 clearance.Gastrointestinal stromal tumors are relatively rare and the jejunum is the least affected among other parts of the GI tract. The majority of ruptured GISTs occur spontaneously, and are located in the stomach and small bowel. Most ruptured GISTs are associated with dismal prognosis.A jejunal gastrointestinal stromal tumor can rarely present with spontaneous perforation. However, it is important to highlight this condition, as early emergency intervention constitutes the key to a good outcome. It originates from the interstitial cells of Cajal, which are part of the autonomic nervous system of the intestine. The majority of the lesions are benign with a possibility of 20–30% for malignancy. It occurs mainly in the submucosal connective tissue of the stomach, small intestine, esophagus, colon, rectum, omentum and mesentery. It occurs more commonly in men with a median age of 50–70. GISTs are thought to be the result of mutations of proto-oncogene which encodes the cell surface tyrosine kinase receptor. Presenting symptoms in GISTs are non-specific and they are most often diagnosed incidentally .29)/L (Segmented neutrophils 70%), sodium (135 mEq/L) and potassium (3.5 mEq/L) with normal creatinine and blood urea nitrogen level . An abdominal CT scan with IV contrast enhancement was performed after initial resuscitating, it showed pneumoperitoneum with significant fat stranding in the small bowel mesentery . Adequate safety margins were confirmed. The number of lymph nodes retrieved in the specimen was 9. None of which was involved by the tumor. The pathologic stage was pT4 pN0.One-month and two-month post-surgery follow up visits to the clinic were unremarkable. The patient maintained the same body weight as when discharged. No features of short bowel syndrome were detected. The patient started oral imatinib treatment 3 weeks post-surgery.3GISTs are considered the most common mesenchymal tumors in the gastrointestinal tract  that occOne third of the patients with GIST are asymptomatic  but it'sMost of GIST are diagnosed incidentally during surgery, CT or endoscopy but several examinations of the gastrointestinal track failed to reach the correct diagnosis with 100% certainty ,7. To diImatinib gives a 14% absolute reduction in recurrence rate, achieving 97% recurrence-free survival . Our dis4In conclusion, GIST is rare and its presentation as spontaneous rupture is extremely rare. Despite all the advancement in diagnostic procedures the preoperative diagnosis remains difficult. However, local excision of the tumor associated with the use of imatinib was effective. This case is remarkable for several reasons: (1) the combination of difficult decision to undertake in emergency settings. (2) The scarcity of this pathological condition. (3) The successful treatment without complications of this patient.None of the authors have conflicts of interest.This study has no sponsors and is self-funded.Ethical approval has been taken from the ethical committe at King Hussein Medical Center, Amman, Jordan. The reference number is 19/12-2019.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.Category 1: Pre-Operative care: Emergency department care: Ghassan T. Alswaiti.Interpretation of the CT images: Walid Al-Nizami.Operation: Main Surgeon: Ghassan T. Alswaiti.Assisstant Surgeon: Mohammad A. Al-Doud.Post-Operative Care: Ghassan T. Al-Swaiti, Mohammad H. Al-Qudah, Mohammad A. Al-Doud and Alaa R. Al-Bdour.Category 2: Drafting the manuscript: Ghassan T. Al-Swaiti, Mohammad H. Al-Qudah revising the manuscript critically for important intellectual content: Mohammad A. Al-Doud, Alaa R. Al-Bdour and Walid Al-Nizami.Category 3: Approval of the version of the manuscript to be published : Ghassan T. Al-Swaiti, Mohammad H. Al-Qudah, Mohammad A. Al-Doud, Alaa R. Al-Bdour and Walid Al-Nizami.Does not need registration.Dr. Ghassan T. Al-Swaiti.Not commissioned, externally peer reviewed.


Insulinoma is a rare neuroendocrine tumor. It may occur sporadically or as part of the genetic tumor syndrome multiple endocrine neoplasia type 1 (MEN1). Diagnosis is challenging because of the small size of insulin producing tumors that lead to hyperinsulinemia. Advances in imaging modalities may provide more accurate diagnosis of primary tumors, metastasis, and tumor functional status. Advances allow for improved medical and surgical management with new tools for research of neuroendocrine tumors. Surgical excision of the primary tumor is often curative; however, insulinomas in MEN1 syndrome are often multifocal with a high rate of recurrence presenting unique challenges in management. Here, we present the case of a 34-year-old male with recurrent hypoglycemic episodes and hyperparathyroidism diagnosed with multiple pancreatic insulinomas secondary to MEN1. Furthermore, we provide a brief review of the literature and discuss the approach to diagnosis and management in patients with MEN1 syndrome and future areas of investigation. Insulinomas are neuroendocrine tumors (NET) that arise from pluripotent stem cells in the pancreatic ductal or acinar epithelium with characteristics of insulin-secreting beta cells . In funcA 34-year-old Hispanic male presented to the emergency room with an intermittent epigastric abdominal pain of one year. He endorsed a 10-pound weight loss due to decreased appetite and post-prandial nausea and vomiting. He did not take any medications regularly, and his family history was remarkable for a brother with Lynch syndrome. Five years prior, he had been diagnosed with primary hyperparathyroidism presenting with urolithiasis secondary to hypercalcemia. He also had a non-specific history of recurrent hypoglycemic episodes. More recently, he had episodic confusion for one to two weeks during which he could not recognize other people, familiar places, or even recall who he was. Multiple episodes occurred while working, which prompted his employer to send him to the emergency department.On exam, the patient was tachypneic and hypertensive with mild epigastric abdominal tenderness to palpation. There were no focal neurological deficits. Initial laboratory studies were remarkable for mild normocytic anemia, blood glucose 39 mg/dL, calcium 11.5 mg/dL, and phosphorus of 1.8 mg/dL. An intravenous (IV) infusion of 5% dextrose in half-normal saline was started for hypoglycemia. Subsequently, pamidronate along with zoledronic acid was administered for the hypercalcemia. With therapy, blood glucose improved to 107 mg/dL. After stabilization, the intact parathyroid hormone was measured at 180 pg/mg, which confirmed the presence of primary hyperparathyroidism. An ultrasound of the head and neck did not show evidence of parathyroid hyperplasia. Due to suspicion for insulinoma, fasting levels of insulin, c-peptide, and proinsulin were obtained. While random serum insulin and c-peptide were within normal limits , proinsulin was elevated , which was suggestive of a diagnosis of insulinoma. The patient was started on diazoxide therapy, while a magnetic resonance imaging (MRI) of the abdomen was performed to identify potential NETs and possible metastases. The MRI of his abdomen revealed multiple pancreatic masses, the largest measuring 2.8 x 1.3 cm, with diffusion restriction and variable enhancement within the pancreas, consistent with insulinomas , insulin-like growth factor-1 (IGF-1), adrenocorticotropic hormone (ACTH), follicle-stimulating hormone (FSH), and luteinizing hormone (LH) levels, were within normal limits. Therefore, an MRI of his brain was deferred. Surgical oncology was consulted for therapeutic options and recommended management with diazoxide and an octreotide scan for tumor staging.An indium-111 pentetreotide (octreotide) scan was performed with imaging at 48 hours post-injection. It revealed no abnormal areas of uptake, including lesions previously identified on MRI Figure . Given cAt follow-up three weeks post-surgery, the patient was enjoying an improved quality of life without any episodes of hypoglycemia or hypoglycemic symptoms. He reported checking his fasting blood glucose two times daily since his surgery with fasting blood glucose ranging from 90 to 120 mg/dL without insulin therapy. The first case of insulinoma was described in 1927 by Wilder and colleagues . It was The success of the surgical intervention relies heavily on accurate preoperative imaging and the ability to clearly localize the tumor. However, imaging insulinomas, specifically in MEN1 syndrome, is notoriously difficult due to their small size and often extrapancreatic locations. Somatostatin receptor (SSTR) scintigraphy with 111In-pentetreotide computed tomography (OctreoScan) is one method of detecting foregut NETs, but it fails to image 35%-50% of pancreatic NETs <1 cm in diameter . EndoscoAs patients with MEN1 syndrome are more prone to multifocal disease and recurrence, the development and validation of novel therapeutics holds the potential to greatly benefit these patients. Peptide receptor radionucleotide therapy using lutetium-177 or yttrium-90 conjugated to an SSA has shown promise for NETs in patients without MEN1. One controlled trial of 177Lu dotatate in 229 patients with advanced midgut NET showed progression-free survival of 11% vs 65% at 20 weeks in favor of the radioisotope group . These pWe present a patient with recurrent symptomatic hypoglycemic episodes, who was found to have multifocal insulinomas and MEN1. Clinicians should be aware of the diverse presentations of insulinoma in patients with MEN1 syndrome. Rigorous preoperative staging with imaging is critical to successful surgical management. Currently both OctreoScan and 68Ga dotatate PET scans are available imaging modalities; however, both have significant limitations when attempting to identify insulinomas. Lutetium-177 dotatate offers a potential area of receptor-based therapy that could be both diagnostic and therapeutic. However, more robust clinical trials will need to be conducted in order to establish the role of 177Lu dotatate in insulinomas related to MEN1 syndrome.


The incidence of a mature ovarian teratoma ranged from 20% to 30% of pediatric ovarian tumors , which is composed of well-differentiated tissues that derive from all three germ cell layers ; it is one of the most common benign ovarian neoplasms. In this case report, we discuss a 9-year-old female patient who presented with abdominal pain and distended abdomen, for which she had an abdominal ultrasound and magnetic resonance imaging. The histopathological exam, after a laparotomy, showed a mature ovarian teratoma. Germ cell tumors are the most common ovarian neoplasms in childhood and adolescence, and of these, the teratomas, whether mature or immature, are the most frequently found . TeratomA mature teratoma (MT), which is composed of well-differentiated tissues that derive from all three germ cell layers , is one of the most common benign ovarian neoplasms , 4.In most cases, they are easily diagnosed on imaging studies because of their characteristic intratumoral fat component. Although typical imaging findings of mature cystic teratomas are well known to radiologists, various atypical imaging features can be particularly misleading. This article illustrates atypical imaging manifestations of mature ovarian cystic teratomas through a clinical case and literature review.We present the case of a 9-year-old female patient who presented with diffuse abdominal pain, distended abdomen, and rapid increase in abdominal volume.An abdominal ultrasound diagnosed an abdominopelvic mass with different compositions .Magnetic resonance imaging was performed using 1.5 T MRI with sagittal, transverse T2-weighted, and transverse T1-weighted turbo spin-echo and transverse fat-suppressed T1-weighted turbo spin-echo images. After rapid bolus intravenous injection of gadolinium, transverse contrast-enhanced fat-suppressed T1-weighted turbo spin-echo images and sagittal contrast-enhanced T1-weighted turbo spin-echo images were obtained .The images demonstrate a solid cystic mass of approximately 126 mm in diameter in the abdominopelvic cavity.The cystic structures are of different signal, and the solid mass is raised discretely after injection of contrast medium, probably originating from the left ovary. As a result, the diagnosis proposed is an immature ovarian teratoma.A laparotomy was performed, and the patient underwent left oophorectomy. The histopathological exam pointed out a mature ovarian teratoma .The incidence of a mature ovarian teratoma ranged from 20% to 30% of ovarian tumors in children . BilaterMacroscopically, the mature teratoma is a cystic tumor in 88% of cases, rarely solid. Its wall is covered with a squamous epithelium derived from the ectoderm and limited outside by the ovarian stroma packed around the cyst . Its conA minor percentage of mature cystic teratomas have only a small amount of fat or no visible fat on imaging studies.A mature teratoma is a benign tumor, whereas the immature type, although also benign, has a more aggressive course, with a propensity to recurrence .It is important to remember that the diagnosis of a mature teratoma (MT) may be overlooked in the small number of tumors that do not contain fat and that the differentiation from other epithelial neoplasms is difficult . SometimIn some cases, MTs show atypical imaging manifestation. It is possible to distinguish the atypical manifestation caused by tumor components: MTs without fat in the cystic cavity, MTs with a pure fatty component in the cyst, and combination and collision tumors , 13. A mImaging findings of mature cystic teratomas can be atypical depending on the tumor components. Understanding the atypical imaging manifestations of mature cystic teratomas permits a more specific and accurate diagnosis.


Vascular leiomyosarcoma is an extremely rare tumor and is associated with poor prognosis among leiomyosarcoma. Surgical resection remains the main treatment option. But outcome of definitive treatment with chemoradiotherapy in inoperable patients is not clear. Here, we report treatment and outcome of definitive chemoradiotherapy in a case of vascular leiomyosarcoma. A 64-year-old man with the initial presentation of pulsatile right neck mass was diagnosed with right carotid body leiomyosarcoma. He refused surgical intervention due to risk of carotid body injury and ischemic stroke. Successful tumor control was achieved with carboplatin-based concurrent chemoradiotherapy. Investigational liquid biopsy for circulating sarcoma cells was also performed to analyze drug sensitivity profile of this rare tumor. One year after treatment, the disease remained well controlled and there was no evidence of baroreflex failure or treatment-related late toxicities. To our best knowledge, this is the first case report of right carotid body leiomyosarcoma controlled with definitive concurrent chemoradiotherapy. The approach of personalized multi-modality treatment will be a focus of our future investigation. Sarcomas are a rare type of malignancy that account for less than 1% of all adult cancers. More than 80% of sarcomas originate from soft tissue, with the remaining, less than 20%, of them arising from bone. Soft tissue sarcomas are a heterogenous group of cancers that originate from the embryonic mesoderm and can originate in any part of the body. It usually originates in extremities but can sometimes occur in the trunk, retroperitoneum, or the head and neck region. The most common subtype of soft tissue sarcomas is liposarcoma, undifferentiated/unclassified soft tissue sarcoma, followed by leiomyosarcoma (LMS). LMS comprise 12% of all soft tissue sarcomas, which usually arise from the smooth muscle layers of uterus, gastrointestinal tract, intra-abdomen origin, retroperitoneum, and rarely the blood vessels .Despite a lack of high-level evidence, surgical resection is the mainstay of treatment for these patients. The role of chemotherapy and/or radiotherapy (RT) for vascular LMS remains inconclusive. Clinical descriptions are limited to single case report or case series for vascular LMS of the arterial origin. Due to its rare incidence and poor prognosis, personalized treatment is challenging to multidisciplinary tumor board (MDT). Here, we report our experience of treating one case with primary arterial LMS arising from the right carotid body. Disease control was achieved with carboplatin-based concurrent chemoradiotherapy (CCRT). Relevant literature review on peripheral arterial LMS is also provided.A 64-year-old male presented to the oncology department of our hospital with pulsatile right neck mass for one year. It was associated with dizziness and near-syncope. He was quite healthy without past medical history. On physical examination, a pulsatile mass about 2 cm was palpable over right submandibular region. Carotid bruit was not audible while auscultation and the mass were invisible upon oral cavity inspection. The absence of symptoms of hoarseness or dysphagia indicated no pressure on the larynx or pharynx. Vocal cord paralysis was also excluded after laryngoscopy examination. Magnetic resonance image (MRI) of the head and neck revealed a 2.6-centimeter soft tissue mass occupying the right carotid space and the right parapharyngeal space at the level of carotid bifurcation Figure 1He received biopsy of the carotid body tumor. Pathology reported a high grade leiomyosarcoma with non-limited positive margins. Immunohistochemically, tumor cells are positive for desmin, actin, and h-caldesmon and negative for ER, S-100, CD34, and beta-catenin. Therefore, a diagnosis of right carotid body leiomyosarcoma, high grade, stage IIB was made. After thorough discussion in MDT, platinum-based CCRT was recommended.via 6MV photons  followed by right carotid tumor boost (20 Gy in 10 Fx) was delivered  photons Figure 2In addition to routine clinical workup, an investigational liquid biopsy to analyze circulating sarcoma cells (CSC) was performed if salvage treatment indicated after CCRT. Peripheral venous blood sample was processed for mononuclear cell fraction and was cultured in a binary colloid crystal-coated microfluidic chip to allow clonal expansion. The outgrowth cellular organoids were observed after 4 weeks and cells were stained positive for vimentin and negative for CD45 or EpCAM Figure 5Management of soft tissue sarcoma is challenging to clinical physicians due to its rare incidence, heterogeneity of natural history, and unpredictable response to treatment owing to diverse histology. In addition, special considerations were needed for site-specific tumors. For adult localized disease, surgical resection with appropriate margins is the standard treatment of all adult sarcoma patients. Regional lymph node dissection is not routinely required because the risk of metastatic lymphadenopathies is quite low. However, surgical measure is not always practicable due to the limitations of critical anatomical structures such as major nerve and blood vessels. In addition, positive margins through microscopic examination or macroscopic residual disease imply high risk of local recurrence. Adjuvant treatment, including adjuvant RT, is mandated for microscopic or macroscopic diseases and is associated with improving local control rate from 56% to 74%  as compared with LMS of other origin (9.6 years and 7 years) . AnthracTo our best knowledge, this is the first case report of vascular LMS arising from the right carotid body. Owing to its complexity, the patient received multimodality treatment with carboplatin-based CCRT instead of radical surgery after comprehensive discussion in head and neck MDT. The disease remained controlled after one year of surveillance. The patient lives with preserved quality of life and is satisfied with excellent tumor control during this period. Moreover, investigational liquid biopsy with CSC and molecular and cellular analysis demonstrated potential for personalized drug sensitivity profiling. Although CCRT is associated with promising tumor control, it is not an evidence-based approach owing to the rare disease entity. Furthermore, there are no detailed neurophysiological examinations to clarify neurological function despite the absence of symptoms of baroreflex failure. In conclusion, definitive CCRT is well tolerated and achieved promising tumor control for this patient with right carotid body LMS. Further investigation will be necessary for treating inoperable peripheral arterial LMSs with definitive CCRT.The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.The studies involving human participants were reviewed and approved by Office of Human Research, Taipei Medical University. Written informed consent to participate in this study was provided by the participants’ legal guardian/next of kin.Conceived and designed the experiments: L-SL, J-FC, and L-LT. Analyzed the data: C-SL, J-RT, Y-TK, L-SL, Y-JC, TB, and P-YW. Contributed reagents/materials/analysis tools: L-SL, Y-JC, TB, and P-YW. Critical discussion and reading: J-FC and L-LT. Wrote the paper: C-SL, J-RT, L-SL, and L-LT. All authors contributed to the article and approved the submitted version.This project is supported by the National Health Research Institutes, Ministry of Science and Technology, and Taipei Medical University .The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


Brunner’s gland adenoma is a rare benign tumor arising fromBrunner’s glands. It is mostly small in size, and patients with this tumorare asymptomatic.We report the case of a 63-year-old woman with upper gastrointestinal obstructionfor almost 10 years, who was pathologically diagnosed with large Brunner’sgland adenoma of the duodenum. Postoperatively, no sign of recurrence has beennoted until now.This study may help clinicians to understand and provide a more accurate diagnosisof Brunner’s gland adenoma. However, Brunner’s gland adenoma, also called polypoid hamartoma orBrunneroma, was first described by Curveilheir in 1835. It is a rare benign tumorarising from Brunner’s glands that may be transformed into a malignant tumor,3 and is2T1-weighted images and slightly high in T2-weighted images. Endoscopic ultrasonography , heartburn, acid regurgitation, and eructationfor 6 months. No history of hypertension, diabetes mellitus, or coronary heart diseasewas noted. Upon physical examination, a 5 × 4 cm mass with a hard textureand poor mobility was observed in the upper abdomen. Routine blood and tumor marker testresults were within normal range. Abdomen computed tomography CT,  showed anography  demonstrNeoplasm resection of duodenum was performed, and we found a mass measuring about 25× 30 × 10 mm located in the descending duodenum. It was soft,brittle, and mobile with a clear boundary. The pathological result  of the mInformed consent: Informed consent has been obtained from the patientincluded in this study.3Helicobacter pylori infection [Brunner’s gland adenoma is primarily located in the duodenum, especially theproximal duodenum, and is possibly caused by hyperplasia of the secretory tubes andstroma in the proximal duodenum ,5.Brunnnfection . It is rnfection . In our nfection  and may nfection , anemia nfection , acute pnfection ,14. In tThe accurate diagnosis of Brunner’s gland adenoma can be made throughhistopathological examination; however, such examination is difficult to performpreoperatively. Gastrointestinal endoscopy, CT, and other radiologic imaging methods areuseful for identifying the cause of the clinical manifestations. Nonetheless,Brunner’s gland adenoma is easily confused with pancreaticoduodenal tumorsbecause it is nonspecific, which may lead to difficulties in diagnosing and changes inthe treatment strategy .At pres4Brunner’s gland adenoma is a rare benign tumor of the duodenum. It is also aninsidious cause of upper gastrointestinal obstruction because some patients may presentwith upper gastrointestinal bleeding, and a few of these tumors could be malignant, andtherefore, warrant attention. Endoscopic resection is the first treatment choice whenthe tumor is small or has a stem. Surgery is reserved for cases where endoscopicresection has failed or when the tumor is large.


Ultrasonographic documentation of perforated abomasal ulcer has not been published till now. This report describes the clinical, ultrasonographic and postmortem findings in a Jersey cow with type-3 abomasal ulcer and left displacement of the abomasum (LDA).The main clinical findings were abnormal demeanour, rumen atony and tympany, positive foreign body tests, positive auscultation and simultaneous ballottement on the left side, abdominal guarding and loss of negative pressure in the abdominal cavity. The tentative diagnosis was peritonitis and LDA. Abdominal ultrasonography produced images typical of LDA, and in one location between the abdominal wall and abomasum there was a layer of fibrin, a fibrin clot, a break in the abomasal contour, suggestive of a perforated ulcer, and partial obstruction of this gap with fibrin. The diagnosis of perforated abomasal ulcer with subsequent peritonitis was confirmed during postmortem examination.The examination of this case shows that under certain circumstances, ultrasonographic imaging of a perforated abomasal ulcer in a cow is possible. Antemortem diagnosis of type-3 abomasal ulcer is preferable to relying on exploratory laparotomy and/or post-mortem examination. Abomasal ulcers in cattle are classified as types 1–5 (U1–U5) . Type-3 A 2.3-year-old Jersey cow that had calved 4 weeks previously without complications developed acute mastitis 2 weeks after parturition followed by unilateral peritarsitis 2 weeks later. Treatment with oxytetracycline ) led to transient improvement but the cow relapsed and stopped eating. Treatment with a rumen magnet, glucose infusion (500 mL of a solution containing 109 g glucose and 70 g sorbitol), brotizolam  and dexamethasone  was unsuccessful. The cow developed rumen tympany and spontaneous grunting and was therefore referred to our clinic.The general health and appetite of the cow were poor, the skin surface temperature and muzzle were cold to the touch and the scleral vessels were congested. The heart rate was 84 beats/min , the re®, Roche, Basel) produced no abnormal findings , and the rumen chloride concentration was 25 mmol/L . Ballottement and simultaneous auscultation were positive on the left side. Intestinal motility was poor and there was abdominal guarding. Transrectal examination revealed a loss of negative pressure in the abdominal cavity but the faeces were normal. The California mastitis test was positive in all quarters. The cow had peritarsitis in the left hind leg but was not lame. Urinalysis using a test strip . The maBased on the positive foreign body tests, abdominal guarding, rumen atony and tympany and reduction in negative abdominal pressure, a tentative diagnosis of peritonitis was made, and the auscultatory findings on the left were suggestive of left displacement of the abomasum (LDA). Ultrasonography of the reticulum and the abdominal cavity  and radiBased on the ultrasonographic findings, a diagnosis of localised fibrinous peritonitis attributable to a perforated (type-3) abomasal ulcer was made, in addition to LDA. Based on a grave prognosis, the cow was euthanased and underwent postmortem examination. Incision of the abdominal cavity resulted in release of malodorous gas, and small amounts of free greenish plant material were seen within the abdominal cavity. The abomasum was in an anatomically normal position at the time of examination and had two perforated ulcers measuring 4.0 and 1.5 cm in diameter, respectively, and multiple small type-1 ulcers Fig. . There wThe different types of abomasal ulcers have been described in many studies –5, 19–24A gap partially occupied by a plug-like clot of fibrin in what otherwise should have been a smooth abomasal contour was clearly imaged by ultrasonography. It was likely that bacterial gas production led to a marked increase in the echogenicity of the fibrin clot and the comet artefact medially. This notion was supported by malodorous gas that escaped after the abdominal cavity was opened. The fibrinous adhesions between the abdominal wall and the abomasum were the result of massive inflammation caused by leakage of abomasal contents. Subjective signs of abomasal ulcers such as lesions associated with peritonitis have been seen via ultrasonography in cattle with type-3  and typeWe were surprised to find the previously displaced abomasum in its correct position during postmortem examination considering the degree of peritonitis and the amount of fibrin between the left abdominal wall and the perforated ulcer. We suspected that the adhesions were at an early stage of development and that tearing occurred during movement of the dead cow from the barn to the postmortem room.This case report should encourage clinicians to continue to place emphasis on ultrasonographic examination of the abdomen when perforated abomasal ulcers are suspected, particularly in cows with LDA. Advances in ultrasound technology and the availability of high-resolution transducers have increased the probability of detecting a variety of disorders including ulcers.


Abscess of the liver ligaments is extremely rare, and abscess of the falciform ligament has been sporadically reported. We report the case of a 3 years old male who presented with a three days history of right upper quadrant abdominal pain, fever and nausea. The ultrasound and computed tomography (CT) scan showed an abdominal wall abscess located anterior to the liver. The patient underwent surgery. Abscess of the falciform ligament secondary to a ruptured liver abscess was found. Excision of the falciform ligament including the abscess was performed. Although pathology of the falciform ligament is rare, it should be included in the differential diagnosis of acute abdomen. Few cases of falciform ligament abscess have been reported. This implies that the pathology of falciform ligament abscess is poorly understood, and many surgeons may be unable to recognize it when encountered. We report a case of a pyogenic falciform ligament abscess secondary to a ruptured liver abscess.S. aureus (methicillin sensitive). Antibiotics were appropriately manipulated, and he was kept on oral antibiotic therapy for 4 weeks. Echocardiography revealed no evidence of valvular heart disease. The patient was discharged on postoperative day 7. Pathological examination revealed a fibrosis of the falciform ligament with abscess formation, and presence of non-specific granulomatous tissue. The post-operative course was uneventful.A 3-year-old male was admitted to the hospital with a three-day history of right upper quadrant pain, fever, diarrhea and nausea. There was no history of jaundice. There was no history of abdominal surgery or abdominal trauma. On physical examination, her body temperature was 38.2°C, pulse was 84 beats/min and blood pressure was 110/70 mmHg. Abdominal examination revealed a palpable epigastric mass that was slightly tender. There were no signs of peritonitis. Results of laboratory studies showed: white blood cell count 19,500/ml with 75.0% polymorphonuclear leukocytes, c-reactive protein test (CRP) was 130 mg/dl. Liver function and electrolytes were within normal ranges. Abdominal radiography was unremarkable. An ultrasound examination revealed an anechoic lesion measuring 5×4 cm in the left lobe of the liver. The biliary ducts, gallbladder, pancreas and spleen were normal . A proviStaphylococcus aureus, and most often, a solitary abscess is found such as our patient, if bacteria spread from an adjacent infected organ, the abscess is usually multiple and polymicrobial, with gram-negative enterics and anaerobes.The falciform ligament is the embryologic remnant of the ventral mesentery, and marks the separation of the most caudal part of the left lobe of the liver into medial and lateral segments. The ligament is composed of two mesothelial layers, within which lies the ligamentum teres hepatis , paraumbilical veins, muscular fibers, and a variable amount of adipose tissue . FalcifoInfection of the falciform ligament is extremely rare, but it should be suspected in patient with abdominal complaints and imaging study may be useful to demonstrate abscess formation of the falciform ligament.


Staphylococcus aureus (MSSA) infection. In our case, morphea was thought to have been potentially triggered by a post-surgical infection. We discuss the potential infectious triggers and common infections that may confound the diagnosis.Morphea or localized scleroderma is reported to be triggered through diverse stimuli. We present a case of morphea that presented as a non-healing wound with superimposed methicillin-sensitive  Morphea  is a rare chronic connective tissue disease of unknown etiology. It belongs to a broader group of disorders that constitutes scleroderma, other diseases include diffuse cutaneous systemic sclerosis (DcSSc) and limited cutaneous systemic sclerosis (LcSSc). The common pathognomonic feature of these disorders is the presence of skin fibrosis and thickening .The exact cause of scleroderma is not known; it is postulated to occur after exposure to an exogenous stimulus in genetically predisposed individuals . DcSSc aWe present a case of morphea that initially manifested as recurrent skin and soft-tissue infections (SSTIs).A 53-year-old Caucasian lady presented to our emergency department (ED) in October 2019 with complaints of diffuse abdominal pain worsened with movement, nausea, chills, non-healing abdominal wound and brown-colored drainage from an abdominal drain site.Staphylococcus aureus (MSSA) and she was discharged on oral antibiotics, with the JP drains in situ.Her past medical history was significant for bilateral breast reduction mammoplasty and abdominoplasty in 2017. Her postoperative course was complicated by recurrent abdominal wall fluid collections, managed with repeat surgical drainage. After the last surgery in March 2019, she developed hyperpigmentation and ulcer formation along the anterior abdominal wall, managed with local wound care. She was re-admitted to another hospital in September 2019 with worsening abdominal tenderness. Imaging revealed a 15 cm × 2.3 cm mixed attenuation fluid collection in the right anterolateral abdominal wall, connecting to a 3.2 cm × 1.4 cm midline fluid collection . She was directed to the ED of our hospital for further management. Review of systems was negative except for complaints of fatigue and mild exertional dyspnea. She lived with her son and denied any toxic habits.3, hemoglobin 11.9 g/dl, blood urea nitrogen 13 mg/dl and creatinine 0.73 mg/dl.On initial evaluation, she was afebrile and hemodynamically stable. Systemic examination revealed no remarkable findings. Skin examination revealed a 5 cm × 5 cm tender ulceration over anterior abdomen, with surrounding mild erythema and collarettes of scale, with no active discharge or bleeding. She was also noted to have two firm hyperpigmented, indurated and excoriated plaques over the upper left and right abdomen. As per the plastic surgeon, there were no retained foreign bodies. Initial laboratory testing was unremarkable, including white blood cell count 4,740/mmInfectious diseases service was consulted and she was initially started on empiric cefazolin. Dermatology was consulted and punch biopsies were performed from central abdomen and edge of right abdominal ulcer. Interventional radiology was consulted and she underwent placement of an 8.5-F Dawson-Mueller pigtail catheter  into the right lower abdominal quadrant, with drainage of 30cc of clear fluid.Skin biopsy Figures -6 revealBlood cultures and fluid cultures showed no growth. A repeat CT scan showed complete resolution of the fluid collection Figure . She wasMorphea is a rare disease. It is estimated to involve 0.4-2.7 per 100,000 people . It is mMorphea may be mistaken for infections, particularly in the active stage of disease. It has been mistaken for cellulitis and tinea cruris ,7. ZosteBorrelia burgdorferi infection with morphea, with no definite consensus. The researchers used different testing techniques in patients from different geographic areas and evaluated different strains [Additionally, morphea has been reported in association with multiple infections, such as tularemia, human T-cell lymphotropic virus type 1 infection, cytomegalovirus, human immunodeficiency virus and hepatitis C virus coinfection -13. The  strains -17.The diagnosis of morphea is usually clinical, while tissue biopsy helps exclude other differential diagnoses . High tiReview of the available literature revealed one recent report of morphea being mistaken for cellulitis after breast reconstruction surgery . The patMorphea may be mistaken for an infection in the acute stage. Clinicians should remember to include it in the differential diagnoses of chronic wounds with atypical, prolonged or recurrent presentations. In such diagnostically challenging cases, a multidisciplinary approach may be warranted.


A 27-year-old male patient presented with cough and right-sided, light thoracic pain. His physical appearance showed typical features of gigantism. Subsequently, further diagnostic work-up showed elevated level of growth hormone and a huge tumor of the right lung, identifying a typical pulmonary carcinoid tumor (TPCT). Curative surgery was performed leading to normalization of the elevated growth hormone levels few days after surgery. Two- and five-year follow-up showed no signs of recurrence. Respected to tumor size, we determined the largest TPCT to be reported in medical literature history. Pulmonary carcinoids were initially labeled as adenomas of the bronchial system . AlthougIn our case, we report an exceptional huge TPCT in a young male patient with gigantism syndrome.A 27-year-old, non-smoking, 197 cm tall, 118 kg weight male patient presented himself at an external medical practice with symptoms including cough and right-sided, thoracic pain. His past medical history was uneventful. Next to signs of gigantism, the physical examination determined weak breath sounds of the right lung. Furthermore, the patient suffered from augmentation of the facial configurations, large extremities with oversized hands and feet, with no correlation to the height of his parents. Even though laboratory investigations showed normal results, growth hormone levels were found to be massively elevated: 27.6 µg/l .A subsequent CT scan revealed a huge tumor in the right lower lobe accompanied with calcifications as well as moderate pleural effusion Fig. . The tum2  within few days after surgery. The patient was discharged on the 11th postoperative day in an excellent physical condition. To date, the patient is recurrence-free and under follow-up (2- and 5- and 10-year follow-up was done performing CT scans).Typical pulmonary carcinoid syndrome is a rare occurrence. Patients with TPCT almost never exhibit symptoms. TPCT are known to be active endocrinal tumors, although it is a very rare phenomenon. Hormone production is seldom but gigantism, acromegaly or Cushing’s syndrome for example is the commonest in the literature. Other rare endocrinopathies have been reported in such patients . In our Carcinoid tumors do not present specific radiological signs. Huge-size carcinoid tumors could be mistaken as benign lesions, for example fibroma or lipoma. Nevertheless, patients with typical carcinoid tumors have an excellent prognosis, with up to 87% survival rate at 10 years and 74% at 15 years –5. AccorIn summary, huge typical carcinoid tumors are extremely rare, proclaiming a surgical challenge. With respect to the literature, this is the first report about carcinoid tumors associated with gigantism syndrome. Furthermore, our report seems to be the first case with huge carcinoid tumors in a young male, who was successfully treated surgically in curative intend. In the literature one patient with carcinoid tumor associated with acromegaly has been reported . SynchroThere exists no consensus on how to follow up patients after surgery for a typical carcinoid. We recommend a CT scan once every 5 years at the latest. We suggest that, in case of gigantism, chest X-ray should be added to the investigation methods to identify such lesions and to plan a useful therapy. Typical carcinoid tumors are potentially curable, even when they reach a large size. The management of such cases requires careful investigation, planning, and an aggressive treatment strategy.Typical carcinoid is a slowly growing tumor, which could reach huge sizes. It is usually physiologically inactive but hormonal activities are rare known. In this case, the tumor produced a significant amount of growth hormone, which affected the patient’s configuration information of a gigantism. The tumor was surgically removed successfully in a curative intention. Long-term follow-up was uneventful.


Soft tissue sarcomas (STSs) are rare malignant tumors originating from mesenchymal cells. Extremities are the most commonly affected anatomical sites, and majority of them present as a painless mass. We present a very interesting case of high-grade spindle cell sarcoma of the pelvis manifested as urinary retention and bloody diarrhea. A 68-year-old male presented to the emergency department with abdominal pain, inability to void urine, and bloody diarrhea. Straight urinary catheterization retrieved 900 mL of urine, and a Foley catheter was placed. All laboratory workup including complete blood count, complete metabolic panel, and urinalysis were within normal limits, but computed tomography (CT) of the abdomen and pelvis with contrast was remarkable for bilateral moderate hydronephrosis and a large 14 x 9.1 cm pelvic mass fistulizing into the rectum. To better identify the extent of disease, magnetic resonance imaging (MRI) with contrast was performed, which also revealed a similar large pelvic mass fistulizing into the rectum. Core needle biopsy of the mass was performed, which showed malignant spindle and epithelioid neoplasm with necrosis consistent with high-grade sarcoma. This is a very rare presentation of STS, and, to the best of our knowledge, only few similar cases have been reported thus far. Soft tissue sarcomas (STSs) are a rare and heterogenous group of mesenchymal cell tumors. The annual incidence is approximately 4 cases per 100,000 population cases per year in the United States and is more common in the pediatric population. More than half of STS cases are high-grade undifferentiated pleomorphic sarcomas, leiomyosarcomas, liposarcomas, synovial sarcomas, myxofibrosarcomas, or malignant peripheral nerve sheath tumors. The most common anatomical sites are the extremities. The rarity and heterogeneity of STS make them one of the most challenging tumors to manage.A 68-year-old male presented to the primary care physician with difficulty in passing urine for two days. His past medical history was significant for hypertension only. His symptoms were attributed to enlarged prostate gland, and he was sent home on tamsulosin. In the subsequent days, his symptoms worsened and he presented to the emergency department with severe lower abdominal pain, inability to void, and bloody diarrhea. Initial vitals were stable. Emergent bladder scan showed that approximately 800 mL of urine was retained in the bladder. Subsequently, straight urinary catheterization was performed, which retrieved 900 mL of urine, with remarkable improvement in his symptoms. Initial laboratory workup including blood urea nitrogen, serum creatinine, electrolytes, hemoglobin, white blood cell count, platelet count, and lactic acid were all within normal limits. Stool studies were unremarkable, and urine culture did not grow any microorganism. Stool cytology revealed numerous malignant spindle cells. Computed tomography (CT) of the abdomen and pelvis with contrast performed in the emergency department was remarkable for a 14.0 x 9.1 cm heterogenous solid mass fistulizing into the rectum Figure , 3. The 1) Vimentin: positive.2) Actin and caldesmon: weakly positive.3) Paired box gene-8: rarely positive.4) Mixed cytokeratin, AE1, AE3, desmin, S100, PSA, PSAP, CD34, CD117, myogenin, and tyrosinase: negative.A multidisciplinary team including a musculoskeletal oncologist, urologist, radiation oncologist, and colorectal surgeon was formulated, and radiation therapy (RT) in an attempt to shrink the tumor was initiated. Finally, the patient underwent pelvic exenteration surgery considering extensive local tumor burden, and systemic chemotherapy was planned afterward. This is a very rare case of STS of the pelvis presenting with acute urinary retention and bloody diarrhea.Sarcomas are a very rare and heterogenous group of human malignant tumors of mesenchymal cells. They comprise less than 1% of all adult malignancies but are relatively common in the pediatric population, with an estimated incidence of 4 per 100,000 cases per year [According to the World Health Organization, most of the soft tissue neoplasms are classified according to the presumptive tissue of origin such as liposarcoma, synovial sarcoma, leiomyosarcoma, rhabdomyosarcoma, fibrosarcoma, and angiosarcoma. In some cases where histogenesis is uncertain, sarcomas are designated based on the architectural pattern, e.g., alveolar sarcoma of soft parts, epithelioid sarcoma, clear cell sarcoma, and spindle cell sarcoma . IHCs arSTSs most commonly present with a slowly enlarging painless mass. It could also rarely present with symptoms of compressions, including extremity edema and sensory deficits. Our patient initially presented with urinary retention and later with bloody bowel movements, making presentation very rare in itself. Several anatomical sites are identified where STS could potentially occur, with a majority of cases found in the extremities . SarcomaMRI is usually the initial imaging modality for most of the sarcomas, but some authors prefer CT for sarcomas confined to the retroperitoneum. In one study, Panicek et al. found no statistically significant difference between MRI and CT for the diagnosis of STS . PositroThe treatment of STSs is highly individualized and controversial. Several factors including tumor sizes, anatomic site, tumor grade, high-risk features, and range of histology must be considered before initiation of therapy. A multidisciplinary team including surgery, orthopedic surgery, medical oncology, and radiation oncology should be formulated . In geneThe heterogeneity and the rarity of STS have been obstacles to generating reliable evidence in favor of or against adjuvant and neoadjuvant chemotherapy. The 2014 European Society for Medical Oncology (ESMO) clinical practice guidelines state that adjuvant chemotherapy, though not the standard of care, could be considered as an option for STS greater than 5 cm and intermediate- to high-grade sarcomas . ChemothHistological grade of the tumor at presentation, outcomes of local resection, and extent of distant metastasis are considered as major prognostic factors. Several nomograms including the Memorial Sloan Kettering Cancer Center sarcoma nomogram and Helsinki University sarcoma nomogram have been developed to predict post-operative survival more accurately ,16. In mSTSs are a rare but very diverse group of tumors of mesenchymal cell origin. The complexity and heterogeneity of these tumors have been the major obstacles in their effective treatment. Several treatment options including local resection, RT, and adjuvant chemotherapy have been studied, but the standard care is still not defined. STS of the pelvis is a rare entity, which can present with significant anatomic complications such as urinary retention and fistula, as evident in our patient. Early diagnosis and prompt treatment with debulking surgery with and without radiation is crucial in the management of these tumors.


A 57-year-old male with a history of gastroesophageal reflux disease and esophageal strictures presented with melena and abdominal pain. He underwent an esophagogastroduodenoscopy, which revealed a 5-cm duodenal bulb mass causing partial obstruction of the gastric outlet. Endoscopic ultrasound showed a 5-cm, hypoechoic lesion, arising from the mucosal layer, with a large blood vessel feeding the lesion. Biopsy revealed benign Brunner’s gland hyperplasia. The large mass was causing symptomatic obstruction of the pylorus and iron deficiency anemia, and had risk for malignant transformation. Due to its size it was not amenable to endoscopic removal. Subsequently, he underwent exploratory laparotomy with pyloroplasty, duodenotomy and partial duodenal resection. Surgical pathology showed Brunner’s gland hyperplasia and was negative for malignancy.  The small intestine constitutes 75% of the gastrointestinal tract; however, small intestine tumors are extremely rare (5%), with duodenal tumors being more common than jejunal and ileal tumors . BrunnerWe present a case of Brunner's gland hyperplasia leading to a 5-cm symptomatic mass that required surgical resection due to its size. This case was presented as a poster and published as an abstract . A 57-year-old male with myasthenia gravis, gastroesophageal reflux disease (GERD) and esophageal strictures requiring dilations presented with a few weeks of melena and abdominal pain. His labs were notable for iron deficiency anemia: hemoglobin 9.4, iron 27, ferritin 7. He had been on pantoprazole 40 mg daily and was started on iron supplementation.Helicobacter pylori. Colonoscopy showed benign colon mucosa with three hyperplastic polyps and diverticulosis. He underwent an esophagogastroduodenoscopy (EGD) and colonoscopy. EGD showed chronic duodenitis and a large, benign appearing, polypoidal mass with a large stalk and normal overlying mucosa, measuring about 4 cm, in the proximal duodenal bulb. This mass was causing partial obstruction of the gastric outlet. Biopsies of this mass were benign in pathology and negative for He was referred for an EGD with endoscopic ultrasound (EUS) for further evaluation of the mass, which was delayed by seven months as the patient had relocated. After establishing care with a gastroenterologist, a CT abdomen with contrast was performed which showed an elongated, well-circumscribed, intraluminal mass, arising from the proximal duodenal bulb, measuring approximately 5.3 x 1.7 x 1.5 cm, without evidence of lymphadenopathy or metastasis (Figure Repeat EGD showed a 5-cm polypoidal mass and a thick stalk in the proximal duodenal bulb Figure , causingDifferential diagnosis at the time included tubular adenoma and carcinoid. Endoscopic biopsy revealed benign histology with foveolar cell metaplasia and reactive cellular changes. No adenomatous changes or malignancy were identified Figure .EUS showed a hypoechoic lesion arising from the mucosal layer, with a large blood vessel feeding the lesion Figure .Due to its large size, the lesion was not amenable to endoscopic removal. He was referred for surgical removal of the mass, as it was causing iron deficiency anemia and symptomatic obstruction of the pylorus, and had risk for malignant transformation. He underwent exploratory laparotomy with pyloroplasty, duodenotomy and partial duodenal resection due to the lesion being symptomatic and concern that this may have malignant potential. Histological examination of the resection on routine hematoxylin and eosin slides showed Brunner’s gland hyperplasia and was negative for malignancy Figure . He has been doing well postoperatively and reports resolution of his symptoms. Helicobacter pylori infection or inflammation stimulating the Brunner's gland cells to produce alkaline secretions and subsequently undergoing hyperplasia [The American Institute of Radiologic Pathology uses the terms “Brunner's gland hyperplasia” for lesions <5 mm in size and “Brunner’s gland hamartoma” for lesions >5 mm in size . The underplasia . Brunner’s gland hyperplasia is usually an incidental finding during endoscopy or imaging studies . SymptomDiagnosis requires histological examination of the mass, usually by endoscopy with excision or biopsy. CT scan and EUS can demonstrate the submucosal origin of the mass . Mode ofThe potential cause of our patient’s large Brunner’s gland mass could be gastric acid hypersecretion given his severe GERD with peptic esophageal strictures. Brunner’s gland hyperplasia, though often an incidental finding, can present symptomatically with larger sized masses. Although largely benign, there is rare potential for malignant transformation. The potential cause of our patient’s large Brunner’s gland mass could be gastric acid hypersecretion given his severe GERD with peptic esophageal strictures.


With the development of endoscopic techniques, endoscopic therapy began to play an important role in the management of esophageal hemangiomas.A large esophageal submucosal tumor (2.5 cm), which was suspected to be an esophageal hemangioma, was diagnosed in a 50-year-old woman.Esophageal hemangiomaEndoscopic submucosal dissection was performed for tumor removal.Histopathological results revealed hemangioma. No complication or recurrence was observed in the 17-month follow-up period.Our successful experience showed that endoscopic submucosal dissection is an effective and a safe approach to treat large esophageal hemangiomas (2.5 cm). Furthermore, endoscopic sclerotherapy alleviates discomfort of larger lesions (>2 cm) with high recurrence rates. One Previous study showed that endoscopic submucosal dissection (ESD)may be applied for esophageal hemangiomas (>2 cm). We herein, report a case of a large submucosal esophageal hemangioma (2.5 cm) in a 50-year old patient that was successfully removed en bloc by ESD.Esophageal hemangiomas are uncommon benign vascular tumors.2A 50-year-old woman presented with a 4-month history of dysphagia. The patient's physical and overall nutritive conditions were good. No abnormal physical findings in the thoracic or abdominal region were observed. No abnormalities were detected in the peripheral blood, blood biochemistry, results of blood coagulation tests, tumor markers, and viral markers. The patient underwent upper gastroesophageal endoscopy, which revealed a 10 mm diameter pale-bluish and engorged polypoid mass obstructing the esophagus 18-21 cm from the central incisors Fig. . FurtherESD was performed under general anesthesia. The first submocosal injection was administered 0.5 cm away from the lesion to avoid bleeding. In the process of incision, the transparent cap was fixed on the head of the gastroscope . The front end of transparent cap was approximately 0.3 cm away from the front end of the gastroscope, which helped to partially peel the lesion and widened the operative field. This cap prevented the tip of the dual knife  to be too close to the mass. The distance between the first incision and hemangioma was approximately 0.5 cm. Following the incision of the mucosal layer, the submucosal layer was dissected from the oral side using a dual knife Fig. . The outNo complication occurred after ESD. A broad-spectrum antibiotic and proton pump inhibitor were administered intravenously 1 hour after the surgery and for the next 3 days. The patient was fasting and receiving fluid therapy for 3 days. She was discharged 5 days after the surgery and an oral proton pump inhibitor was prescribed for the next 3 weeks. After 3 months, a follow-up endoscopy was performed that showed no recurrence or complication at the ESD site Fig. . The pat3 which represent approximately 3% of all benign esophageal tumors. Typically, the overlying bluish mucosa can be easily detected in the esophagus. On computed tomography, esophageal hemangiomas appear as homogenous and isodense mass with calcifiation. On EUS, the hemangioma appears hypoechoic and involves the mucosal and submucosal layers with intact muscularis propria. In our case, EUS was performed to detect the depth of the lesion and to evaluate the risk of perforation during ESD. In most cases, asymptomatic hemangiomas require only clinical observation. However, patients with symptoms, such as bleeding from the hemangioma and digestive tract stenosis, may require treatment. Esophagectomy is the conventional surgical approach for treatment of esophageal hemangiomas; however, recently less invasive approaches, such as endoscopic therapy, are widely used.,3Esophageal hemangiomas are uncommon benign vascular tumors, Sclerotherapy can also be utilized for the small lesion (<2 cm) with lower risk of recurrence. The common agents include 5% ethanolamine oleate, 1% polydocanol followed by 99% ethanol, and ethanoloamine. Potassium titanyl/yttrium aluminum garnet laser therapy and endoscopic sclerotherapy are less invasive strategies than surgery for large esophageal hemangiomas (>10 cm), which cannot be managed by endoscopic resection and often need repeated treatment to reduce the recurrence of tumor. The mass decreases in size and the obstructive symptoms are alleviated after sclerotherapy. Although endoscopic sclerotherapy may cause hemorrhage and necrosis on the puncture site, no perforation has been reported.,8,10 Since sclerotherapy cannot procure specimens for pathological examination and is associated with a risk of residual or recurrent hemangioma, en bloc removal is a better treatment option.A previous study reported a case of a small esophageal hemangioma (<0.5 cm) that was successfully treated by repeated argon plasma coagulation. EMR is less invasive than ESD and may reduce the risk of perforation, and thus, has been used in pedunculated esophageal hemangiomas (<2 cm).,12 However, EMR can cause bleeding. Combined EMR with endoscopic band ligation or hemostatic clipping can be performed as alternative treatment options for esophageal hemangiomas with the advantage of less bleeding and perforation risks. However, sometimes repetitive EMR is necessary to prevent recurrence.Several techniques have been used to treat esophageal hemangiomas in different sizes for endoscopic en bloc removal. EMR has been widely used in the en bloc resection of small tumors (<2 cm). ESD may be used in cases of small and large esophageal hemangiomas (1–2.5 cm) without obvious complications.,15 ESD in cases of vascular lesions in the esophagus is risky because of the hypervascularity of the tumor. The risk of severe complications (bleeding and perforation) is equal to the size of the tumor. Herein, we have presented a case of a large submucosal esophageal hemangioma successfully removed en bloc by ESD. Based on this case report, the key point is the avoidance and pre-management of the blood vessels in the wound to maintain a clear operation field. Moreover, our case highlights the benefits of ESD in shortening the recovery period. The benefits of ESD in treating esophageal hemangioma include minimal invasiveness compared to the surgery, low recurrence rate, integrity of esophagus, and short recovery time. The risk of this endoscopic surgery is local complications; hemorrhage and perforation are prone to occur in the large lesions. The limitation of this study is that we did not investigate the small and large intestine to detect hemangiomas in this patient.The number of patients undergoing ESD is increasing, especially in cases of large size tumors (>2 cm).In conclusion, the best endoscopic treatment for esophageal hemangioma should be effective, safe, and have a low recurrence rate and few complications. We suggest that appropriate submucosal injection, electrocoagulation hemostasis, and maintaining a clear operative field could contribute to successful ESD procedures. Thus, ESD may be utilized for esophageal hemangiomas. Further, the benefits and risks of ESD in the treatment of esophageal hemangiomas (>2.5 cm) need to be investigated in the future.Conceptualization: Yan Yu, Mei Liu.Data curation: Yan Yu, Mei Liu.Formal analysis: Yan Yu, Bingzheng Shen, Mei Liu.Funding acquisition: Yan Yu.Investigation: Yan Yu.Methodology: Jiqiao Zhang, Mei Liu.Project administration: Yan Yu, Mei Liu.Resources: Chao Zhang, Mei Liu.Software: Bingzheng Shen, Li Cao.Supervision: Mei Liu.Validation: Bingzheng Shen.Visualization: Chao Zhang, Panpan Lu.Writing – original draft: Yan Yu.Writing – review & editing: Yan Yu, Bingzheng Shen, Mei Liu.


The aim of this report was to present a selection of focal hyperechoic liver lesions of different etiologies, illustrating the wide spectrum of diagnostic possibilities for such lesions in the pediatric population. Focal hyperechoic liver lesions in children encompass a wide range of diagnostic possibilities including congenital and acquired lesions, neoplastic and non-neoplastic lesions, benign and malignant lesions. Knowledge of the spectrum of such lesions, their imaging aspects, the age group in which they are most prevalent, their clinical characteristics, the associated laboratory test results, and the patient history (of trauma or intervention) is essential for the diagnosis.Abdominal ultrasound images were obtained at our institution. We selected cases of the following: childhood hepatic hemangioma, congenital hepatic arteriovenous malformation, umbilical vein catheterization hematoma, hepatoblastoma, focal steatosis, hepatic lipoma, biliary rhabdomyosarcoma, regeneration nodule and biliary hamartomas.The most common benign childhood liver tumor of vascular origin is infantile hemangioma, which is typically characterized by a growth phase followed by a regression phase. Approximately 90% of cases are diagnosed in the first six months of life, and it is slightly more common in girls.Although most childhood hemangiomas manifest as an asymptomatic abdominal mass, life-threatening complications can occur: high-output congestive heart failure, due to intratumoral arteriovenous shunts; the coagulopathy known as Kasabach-Merritt syndrome, as a consequence of platelet sequestration; and severe hypothyroidism, caused by the activity of type 3 iodothyronine deiodinase, which is produced at high levels by the tumor. Laboratory tests reveal anemia and hyperbilirubinemia. Serum α-fetoprotein levels are rarely elevated.Presentation on ultrasound - Solitary, multifocal, or diffuse lesions , Ehlers-Danlos syndrome, and biliary atresiaPresentation on spectral and color Doppler ultrasound - Faint hyperechoic areas containing tortuous, dilated vessels with a pattern of low-resistance arterial flow and pulsatile venous flow, together with signs of portal hypertension.,4.Hepatic complications of poor positioning of an umbilical venous catheter are uncommon and occur due to extravasation of blood, drugs, or parenteral nutrition into the liver tissue. Hepatic complications include hematoma , fluid cA hepatoblastoma is a malignant neoplasm whose cells resemble those of the embryonic liver and is the most common primary liver tumor in children. In 68% of cases manifest in the first two years of life and 90% are observed in patients under five years of age, with predominance in boys. A typical feature of this tumor is a highly elevated serum α-fetoprotein level.Presentation on ultrasound - Circumscribed liver mass with lobulated contours , diffuse heterogeneous, and multifocal/nodular . The mulPresentation on ultrasound - Hyperechoic lesions, which do not promote a mass effect or displace branches of the hepatic and portal veins, which cross the lesions without altering their course. Lesions may mimic true nodules, causing diagnostic difficulty, especially in the event of pre-existing chronic liver disease or malignancy,7.A hepatic lipoma is a quite uncommon, benign lesion that is usually asymptomatic. Although lipomas may consist only of fat cells, they may also contain tissue that is adenomatous (adenolipoma), angiomatous (angiomyolipoma) or myomatous (myelolipoma). They are occasionally accompanied by tuberous sclerosis (in 6% of cases) or renal angiomyolipoma (in 20%).Presentation on ultrasound - Well circumscribed, uniformly hyperechoic lesion with posterior acoustic shadowing of varying intensity, as shown in . Biliary rhabdomyosarcoma occurs almost exclusively in children and accounts for 1% of liver tumors in the pediatric population. In 75% of cases, they are diagnosed before the age of five. The clinical manifestations are jaundice, bloating, fever, hepatomegaly, nausea, and vomiting. Laboratory tests reveal elevated levels of conjugated bilirubin and alkaline phosphatase, together with normal serum α-fetoprotein levels.A rhabdomyosarcoma is a highly aggressive tumor that can occur anywhere in the body and rarely appears in the biliary treeRhabdomyosarcomas are usually large. Polypoid, or “bunch-of-grapes”, projections are often observed in the lumen of the bile duct. Although the extrahepatic bile ducts are the most commonly involved, the lesion can originate or grow in the intrahepatic bile ducts and invade the liver.Presentation on ultrasound - Dilatation of bile ducts with intraluminal mass, indicating the biliary origin of the tumor , macronodules (> 0.3 cm), or giant nodules (> 5.0 cm).A regenerative nodule is a non-neoplastic lesion that develops in a cirrhotic liver and is therefore also known as a cirrhotic nodule. It is characteristically surrounded by fibrous septa and may progress to a dysplastic nodule or hepatocellular carcinoma. It is distinguished from nodular regenerative hyperplasia, which develops in a liver without fibrosis and is surrounded by normal liver parenchymaPresentation on ultrasound - The nodules are usually hypoechoic, but may be isoechoic, mixed, or, less frequently, hyperechoic (.erechoic (10).Biliary hamartomas, also known as von Meyenburg complexes or microhamartomas, are rare benign malformations of the intrahepatic bile ducts and are considered part of the spectrum of fibropolycystic liver disease due to abnormal embryonic development of the ductal plaque. Biliary hamartomas are usually asymptomatic and are detected as incidental findings on imaging, in laparotomies, or during autopsies (in 6% of adults and in 1% of children).Presentation on ultrasound - Multiple small hyperechoic or hypoechoic foci with “comet tail” reverberation artifacts produced by cholesterol crystals within cystic ductal dilations or calcifications, which can also produce posterior acoustic shadowing artifacts, as depicted in Focal hyperechoic liver lesions are commonly seen in adults, in whom hemangiomas account for most of the cases, whereas in children there is a much wider range of diagnostic possibilities for such lesions.In pediatric patients, hyperechoic liver lesions are often identified as incidental findings in abdominal ultrasound examinations. Knowledge of the spectrum of these lesions and the imaging features that characterize them, as well as of the age group in which they are most prevalent, of their clinical manifestations, and of the accompanying biochemical alterations, as well as of any history of liver disease, trauma, or intervention, make it possible to restrict the diagnosis to the most probable hypotheses. That can inform pediatrician decision-making regarding the management of such lesions-watchful waiting or further investigation-which should always have the objective of avoiding unnecessary invasive examinations.


Background: Glioblastoma (GBM) is the most malignant intracranial tumor in adults. However, the overall management of GBM in pregnancy is rarely reported. How to balance the therapeutic benefits to the mother and risks to the fetus remains hugely challenging for clinicians. The application of specific targeting therapy combined with conventional treatment sheds light on a longer lifetime for the patients suffering from GBM.Case Presentation: We present a pregnant female at 20 weeks gestation diagnosed with GBM. Surgical resection was initially performed without adjuvant therapy, and the tumor recurred de novo 2 months later. A secondary craniotomy and cesarean section were performed simultaneously at 32 weeks gestation, both the patient and infant were survived. She was subsequently treated with traditional chemo-radiotherapy. No other identified genetic alterations indicating an optimistic prognosis were detected except for BRAF V600E mutation. Thus, the BRAF inhibitor was placed on her with achieving a good clinical outcome of more than 2-year survival without recurrence.Conclusion: Personalized multidisciplinary therapy should be considered when GBMs occur in pregnancy. Response to the therapy in this presenting case suggests that BRAF V600E mutation is a favorable biomarker for GBM. The mortality of GBM might be reduced through genetic testing and targeted treatment. However, more studies must be conducted to confirm our observation. Glioblastoma (GBM) is the most aggressive brain tumor which is considered a grade IV glioma based on the WHO classification , with a With the innovation of genomic characterization, more profound insights into the molecular identity of tumors have been achieved. Increasing molecular targets aiming at the stages of initiation, development, and metastasis of tumors have been identified, through which various novel targeted treatment regimens are created. Multidisciplinary therapies that are composed of targeted therapies, surgery, radiotherapy, and adjuvant chemotherapy have shown efficacy in multiple malignant entities including tumors of the central nervous system , 10. TheHere, we present the first case of a young pregnant female with glioblastoma exhibiting BRAF V600E mutation, in whom the isocitrate dehydrogenase (IDH1), O-6-methylguanine-DNA methyltransferase (MGMT) promoter methylation and telomerase reverse transcriptase (TERT) are all negative, indicating chemo-resistance to the classical chemotherapy strategy. The tumor was short-termed recurrent after the primary surgical resection without concomitant treatment. Multidisciplinary therapy composing of mainstream modality for glioma revolved around the second surgery, radiation, chemotherapy and molecular therapy using BRAF V600E inhibitor, was placed on her, resulted in stable regression of the tumor for more than 2 years so far. Besides, the management of GBMs in pregnancy that were reported in the literature were also reviewed and summarized.A 28-year old female at 20 weeks' gestation was admitted to our hospital with progressive headache, dizziness, emesis and vision diminution for 2 months. Magnetic resonance imaging (MRI) showed a heterogeneous cystic and solid mass in the right parietal lobe with marked surrounding edema and a shift of the midline structures to the left side; the cyst wall is distinctly enhanced with contrast medium. GBM was highly suspected . Total rUnfortunately, over 2 months after the operation (32 weeks gestation), with complaints of dizziness and headache, the patient was transferred to our hospital again. An emergency CT scan supported tumor recurrence . B-mode The management of GBMs in pregnancy were diverse and customized, which was highlighted by a literature review of published cases . We condSome of the pregnant patients chose to get RT and/or chemotherapy after surgical resection by taking a risk for the fetus even at the metaphase of gestation. In the case reported in 2012 , the 14-Considering the risk for the fetus, some of the gravidas refused taking RT or chemotherapy after the surgical resection. The patient of 2014  was onlyIn some cases, the patient was diagnosed with GBMs at the relatively late stage of gestation, they chose to undergo delivery and craniotomy in the meantime and accepting adjuvant treatment afterward. The gravida reported in 2005  was almoNotably, some patients got pregnant during the period of adjuvant treatment for GBM. The patient reported in 2012  got pregGBM is the most aggressive brain tumor with infiltrating growth, which leads to difficulty of total resection , 31–33 aThe reported outcomes of variant management for GBM during pregnancy are dramatically different. Survivals of the mother range from 2 to 24 months, none of the cases accepted gene sequencing for molecular features of the tumors and genetic therapy. Regarding this presenting case, not willing taking a risk for the fetus, the pregnant patient accepted surgical resection for the primary tumor without undergoing any concomitant radiation therapy or adjuvant chemotherapy, consequently, the tumor recurred in a short time. With undergoing multidisciplinary therapy including gene sequencing and BRAF-targeted treatment , she remNowadays, more in-depth insights into genetic advances have contributed to a better understanding of the pathophysiology and molecular stratification of GBM , 39. TheWith increasing prominence, BRAF-targeted therapies have shown efficacy in specific tumor entities , 12, 44.Meanwhile, the side-effect of vemurafenib is also reported in the clinical cases, for example, granulomatous hepatitis  and cutaWe present here a significant case that suggests the personalized MDT comprising genetic therapy may provide the pregnant GBM patients with improved outcomes, it complements the evidence of the management for GBMs in pregnancy. The response to this presenting therapy indicates that BRAF V600E mutation is a favorable biomarker for GBM. The mortality of GBM might be reduced through genetic testing and vemurafenib targeted treatment. However, more studies must be conducted to confirm our observation.The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation, to any qualified researcher.The studies involving human participants were reviewed and approved by Ethics Committee of Xiangya Hospital Central South University. The patients/participants provided their written informed consent to participate in this study. Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.CQ, WL, YX, CZ, CW, YL, QX, and QL performed the surgical procedures. CQ, WL, CW, YL, and QX performed data collection and analysis. NJ and YL performed gene sequencing and target therapy. CQ, WL, and QL wrote the manuscript. QL supervised the entire work. CQ, WL, YX, CZ, CW, YL, QX, NJ, YL, and QL provided final approval for the version to be published. All authors contributed to the article and approved the submitted version.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.