Multiple myeloma is a hematological malignancy that classically results in an abnormal clonal proliferation of plasma cells in the bone marrow. Extramedullary disease in the setting of multiple myeloma, referred to as secondary extramedullary plasmacytoma, is found in 7–17% of cases of multiple myeloma at the time of diagnosis and can involve any organ system. Small bowel obstruction is a rare but important gastrointestinal manifestation of multiple myeloma that should be considered in patients with multiple myeloma who present with concerning abdominal symptoms.We present the case of a 52-year-old African-American man with a history of deep venous thrombosis (he is on anticoagulation) and pathologic fracture secondary to multiple myeloma diagnosed 4 months prior to our encounter. He presented with abdominal pain, constipation, nausea, and vomiting. An abdominal X-ray showed distended bowel loops concerning for bowel obstruction and a contrast-enhanced computed tomography scan of his abdomen and pelvis showed a 5.4 cm soft tissue mass involving a loop of distal ileum. He underwent laparoscopic exploration of his abdomen with small bowel resection and primary anastomosis for a small intussusception. He had an uneventful postoperative course and was discharged on postoperative day 6.Multiple myeloma has myriad presentations. Gastrointestinal involvement, although rare, can manifest as small bowel obstruction for which early recognition and appropriate surgical management are key to improving outcome. Intussusception is the most common mechanism of obstruction from extramedullary plasmacytoma causing small bowel obstruction and this has been seen in five of six case reports, including this case. It is important to recognize and consider the risks of immunosuppression, venous thromboembolism, and malnutrition in the surgical management of gastrointestinal complications of multiple myeloma. Multiple myeloma (MM) represents 1–2% of all cancers in the USA with an annual incidence of 4.3 per 100,000 .et al., only 24 of 2584 patients with MM (0.9%) were found to have extramedullary disease affecting the GI tract [Gastrointestinal (GI) involvement by EMP in MM is rare. In a study by Giampaolo GI tract . Given tGI tract .Data on the relevance and role of surgery in GI involvement by MM remain mostly limited to case reports. However, with improvements in therapy and overall survival in MM, coupled with more sensitive imaging, GI involvement and the need for surgical input or intervention will become relatively more common and relevant to the general surgeon , 8. It iOur patient is a 52-year-old African-American man with no prior abdominal surgeries and a past medical history of MM and venous thromboembolism who presented with a 6-day history of nausea, vomiting, and abdominal pain. He had not passed flatus for 24 hours prior to presentation. He did not have fever, chills, or malaise. He was seen by his oncologist and an abdominal X-ray was done; the abdominal X-ray was concerning for SBO for which reason he was subsequently admitted and general surgery consulted.He had been diagnosed as having MM 4 months prior, after sustaining a pathologic left humerus fracture and was on chemotherapy as well as radiation therapy to the affected humerus. He had completed his second cycle of chemotherapy 10 days prior to presenting with signs of SBO. His chemotherapy regimen included bortezomib, lenalidomide, and dexamethasone.in situ hybridization (FISH) myeloma cytogenetic analysis detected a 17p13 deletion in 30% of cells and a t(14:16) re-arrangement was detected in 5.7% of cells. These cytogenetic changes are both identified as high-risk features in the Mayo Stratification of Myeloma and Risk-Adapted Therapy (mSMART) molecular risk classification system. He had stage II disease by the International Staging System (ISS) of MM, with serum beta-2 microglobulin of 4.7 mg/L and serum albumin of 4.5 g/dl.His MM was diagnosed with a bone marrow biopsy that showed 25% clonal plasma cells and Kappa light chain restricted. Serum K/L was 222 and fluorescence His other medical conditions include hypertension, obesity, sleep apnea, vitamin D deficiency, and pulmonary embolism diagnosed 1 month after his diagnosis of MM, for which he was on therapeutic enoxaparin. He had no prior abdominal operation. His family history is significant for breast cancer in his sister.His vital signs were normal and an abdominal examination revealed tenderness in the right lower quadrant with mild guarding. A contrast-enhanced computed tomography (CT) scan of his abdomen showed a 5.4 cm soft tissue mass involving a loop of distal ileum causing dilation of proximal ileum Fig. . There wHe was taken to the operating room for exploration and underwent a laparoscopic small bowel resection with primary anastomosis. A small intussusception was noted intraoperatively. He had an uneventful postoperative course and was discharged on postoperative day 6. A small-bowel mass at the intussusception was confirmed as plasmacytomas on pathology.He was seen in the surgical clinic 2 weeks postoperatively, doing well. His midline surgical scar was healing well, without any signs of infection. He continued to follow up with his oncologist and his chemotherapy regimen was switched to daratumumab, pomalidomide, and dexamethasone because of his progressive disease  despite his initial chemotherapy regimen. He was referred to a tertiary facility because of the aggressive nature of his disease. He developed multiple new skin masses and, subsequently, weakness and sensory changes in bilateral lower extremity with magnetic resonance imaging (MRI) of his spine demonstrating spinal cord compression from T4–T6 level secondary to epidural extension of MM. Despite further aggressive chemotherapy regimen at tertiary facility, including bortezomib, dexamethasone, thalidomide, cisplatin, adriamycin, and cyclophosphamide, he developed worsening and widespread disease with metabolically active masses on positron emission tomography/CT involving right orbit, lungs, liver, spleen, bilateral adrenal glands, and multiple lymph nodes including mediastinal nodes. His clinical and functional status progressively declined, and he died 11 months following his initial diagnosis of MM.MM is an abnormal clonal proliferation of plasma cells producing a monoclonal immunoglobulin and accounts for 17% of hematological malignancies . ExtrameSmall bowel involvement in plasmacytoma secondary to a known plasma cell myeloma (PCM) is quite rare: in a review of the only 61 cases of EMP of the small intestine reported in the literature as of 2012, Lopes da Silva found that only five of these already rare 61 cases were secondary to another, known PCM . HoweverGI manifestations depend on the organ involved but generally result from direct organ invasion leading to perforation in cases of hollow viscus involvement, mechanical mass effects leading to obstruction, and, rarely, development of ascites .Although more recent data have suggested that adhesions remain the primary cause of SBO even in the virgin abdomen , 16, thiTo the best of our knowledge, from a search of the English literature using PubMed, Embase, and Google Scholar, only five cases of SBO from plasmacytoma secondary to MM have been reported –21. AmonGiven an increasing overall survival from MM, the relevance of uncommon presentations of MM, such as SBO, to the general surgeon has become greater than before. This case highlights plasmacytoma as a differential diagnosis for SBO especially in the population of patients with MM with a virgin abdomen who present with concerning abdominal symptoms.MM is a hematologic malignancy with myriad presentations that can involve multiple systems including the small bowel where EMP can lead to bowel obstruction and perforation. Small-bowel plasmacytomas appear to have a high likelihood of causing intussusception as noticed intraoperatively in five out of six case reports of secondary plasmacytoma causing SBO, including this case.Although adhesions remain the most common cause of SBO in the virgin abdomen, the differential of plasmacytoma should be an important consideration in the population of patients with MM especially those without prior abdominal surgery.Various factors such as the risk of immunosuppression due to MM itself and chemotherapy/steroid use, venous thromboembolism, and malnutrition need to be considered in the planning of surgical intervention (preoperatively and postoperatively) in patients with MM with GI complications.Surgical experience and the role of surgery with EMP involving the GI system remains limited with most reports in the literature being case reports; more research and case reports are needed to help in guiding the optimal assessment, prognostic evaluation, and decision making for surgical intervention.


BRAF mutation p.V600E, targetable by the specific inhibitor Vemurafenib. After confirming the presence of the mutation in the tumor, the patient was treated with Vemurafenib. The treatment proved to be effective, leading to a partial response and a stabilization of the disease. Usually, in patients with DS a reduction of the dose of chemotherapeutic drugs is necessary. Vemurafenib was instead well-tolerated as the only observed adverse effect was grade I skin toxicity. This is, to our knowledge, the first case of a PXA reported in a child with DS and the first DS patient treated with Vemurafenib.Brain tumors are the most common solid neoplasms of childhood, but they are very rarely reported in children with Down Syndrome (DS), who develop more commonly different types of malignancies. In particular, we hereby report the case of an 8-years-old child with DS that presented to our attention for neurological and endocrinological issues. Brain imaging revealed the presence of a mass that was partially resected revealing a histological diagnosis of Pleomorphic Xanthoastrocytoma (PXA), a rare WHO grade II tumor extending from the diencephalic region into the surrounding brain tissue. These tumors can harbor the  Down syndrome (DS) is the most common chromosome abnormality among live births, with an incidence of ~1 every 800 live births .Children with DS exhibit a wide variety of medical conditions, such as cognitive impairment, dysmorphic features, congenital heart diseases, gastrointestinal abnormalities, reduction in visual acuity and accomodation, hearing loss, endocrine, and immune deficiencies .Even though life expectancy in these patients has significantly increased over the last decade, children with DS still have a higher risk of neonatal and infant mortality when compared to children without DS, because of the above mentioned comorbidities .One of the most severe conditions associated with DS is the development of malignant tumors, mainly acute leukemia. On the other hand, solid tumors, and, in particular, brain neoplasms are rarely reported in patients with DS, and therefore the biological behavior and natural history of these tumors are not well-described and understood. Whereas, several oncogenes have been identified as responsible for the development of acute leukemias in DS, little is known about the molecular basis of solid tumors in this population.BRAF p.V600E mutated  is a rare brain tumor that most commonly affects children and young adults. The prognosis is favorable when total resection is possible, but in patients not amenable to the eradication, chemotherapy, and/or radiotherapy are the only available therapeutic options. However, these regimes rarely lead to a control of the disease and the prognosis is generally poor . Between mutated  and treaing PXAs .BRAF p.V600E mutation in a patient with DS.We hereby report, to the best of our knowledge, the first case of a PXA harboring a An 8-year old boy with DS was referred to the DS outpatient care unit of the Bambino Gesù Children's Hospital for progressively impaired gait and signs of early puberty.During neurological examination, a slight asymmetrical gait pattern was noted. This anomaly was firstly attributed to the general motor clumsiness typical of DS patients. When evaluating sexual development, a Tanner Stage of P2G2 was observed, with a bilateral testicular volume of 8 ml. To confirm the clinical suspect of early puberty, Gonadotropin-releasing hormone (GnRH) stimulation test was performed. The results showed: basal FSH of 0.7 mIU/mL and after LHRH administration: 3.78 mIU/mL; basal LH was 1.3 mU/mL, and after stimulation: 20.11 mU/mL; Testosterone basal level was 54.5 ng/dL, PRL, beta-HCG, DHEAS and thyroid function were all normal. These results confirmed the suspect of an early puberty of central origin and a brain Magnetic Resonance Imaging (MRI) was then performed. The brain imaging showed diffused pathological tissue, extending from the left diencephalic region and involving the cerebral peduncle caudally, the basal ganglia region cranially , the outer capsule laterally, the temporo-mesial cortex and subcortical white matter, which extended deeply to the anterior portion of the temporal lobe, to the optic chiasm and bilateral retrochiasmatic tract .BRAF p.V600E mutation was then assessed by immunohistochemistry  and was later increased to 480 mg twice a day. After 32 months the therapy was discontinued, and the disease remained stable 3 months after the stop therapy.The patient underwent partial resection of the lesion and the histopathological examination was compatible with the diagnosis of WHO grade II Pleomorphic Xanthoastrocytoma. hemistry  and throThe only side effect reported was a transient follicular truncal rash in the first month of administration with fickle subcutaneous nodules, treated with local topical corticosteroids. No ECG changes or/and suspected skin lesions developed.A new brain MRI after 6 months of therapy demonstrated an important reduction of the lesion and a substantial reduction of enhancement, the last MRI (30 months after diagnosis) demonstrated a stable disease .Clinical response, with gait and movements improvement, was also noted shortly after the beginning of therapy.DS is associated with several hematological disorders occurring at different ages. Neonates with DS may present with transient asymptomatic blood count abnormalities such as neutrophilia, thrombocytopenia and polycythemia. Within 1–2 months of life, 3–10% of infants with DS develop transient myeloproliferative disease (TMD) . DespiteIn a study performed on 2,814 individuals with DS, the authors have shown that the occurrence of cancer in DS is unique with a high risk of leukemia in children and a decreased risk of solid tumors, and amongst these brain tumors, in all age-groups , 13.The molecular basis of this paradox of tumorigenesis in DS is not well-understood yet. Several experimental observations have shown that DS should be a cancer-prone condition: higher rate of whole chromosome and segmental chromosome instability, increased DNA damage and defective DNA repair, immunodeficiency and susceptibility to infections, oncogenes on chromosome 21 . Also, aAPP gene, transcriptional factors such as ETS proto-oncogene 2 (ETS2), the angiogenesis suppressors Dscr1 and Dyrk1A and the Collagen-18 gene whose fragment, endostatin, is an anti-angiogenic compound  that results in substitution of valine by glutamic acid.The mutation of the is tumor , 35, invis tumor . The mosApproximately 70% of pilocytic astrocytoma contain duplication of BRAF gene, which is rare in PXAs . The BRATMEM106B-BRAF fusion in a case of PXA with anaplastic features in a 10-yr-old-female. This alteration results in replacement of the amino-terminal regulatory domain of BRAF with the amino-terminal region of TMEMB106B. They demonstrated that the fusion results in aberrant activation of BRAF signaling, with activation of MAPK/ERK pathway  and later controls proved a stabilization of the disease.In the case presented the mutation Children with DS often respond to chemotherapy treatment as well as children without DS. However, they are more likely to experience severe toxicity with standard chemotherapy regimens, particularly those requiring methotrexate, and this often leads to a reduction of the chemotherapy doses, leading to a less effective treatment , 40. TheThe use of Vemurafenib has not yet been approved for pediatric patients affected by brain tumors and DS. Although central nervous system (CNS) tumors are the most common pediatric solid organ tumor, they are very rare in patients with DS. However, the possibility of development of brain tumors in DS should be kept in mind, especially in case of unusual neurological and endocrinological symptoms. In conclusion, we have shown the safety and efficacy of Vemurafenib in a pediatric patient with DS affected by PXA.This study was carried out in accordance with the recommendations of the Internal Review Board of the Bambino Gesù Children's Hospital with written informed consent from all subjects. All subjects gave written informed consent in accordance with the Declaration of Helsinki. The protocol was approved by the Internal Review Board of the Bambino Gesù Children's Hospital.The authors declare that written informed consent was obtained from the patient's parents for publication of this case report.GP, FdB, AM, and FL designed the study. GP, FdB, GC, DV, AntC, and IA cured the collection of the data. FdB, GC, IA, AndC, AM, GSC, TC, and FD-C interpreted and analyzed the data. GP, DV, FdB, and GC drafted the manuscript. FD-C performed immunohistochemistry analysis. EA performed molecular analysis. AM and FL critically revised the manuscript for intellectual content.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.


Pain in the inguinoscrotal region had severaldifferential diagnoses and can be acute or chronic in nature. Various conditions provokeintense pain and are potentially serious. The imaging examinations used in diagnosingthe causes of such pain include X-ray, computed tomography, and ultrasound.The causes of abdominal pain are extremely variedBecause it is widely available and safe, with an excellent cost-benefit ratio, ultrasoundis usually the first imaging study used after the clinical examination, in order toelucidate or confirm the diagnosis of a palpated lesion-often identified by the patient,who is in pain and requested the consultation-as well as to inform decisions regardingthe treatment strategy . In cases of suspected hernia in theregion, the main objective of an ultrasound examination is to confirm its presence, todistinguish among the types of hernias  and to makethe differential diagnosis with torsions in the scrotum, inflammation, infection,thrombosis, and trauma.(5).When the presence of a hernia is confirmed, it is important to identify the contents ofthe hernia sac, as well as to determine whether the hernia is acute or chronic, as wellas whether it is complicated(6).In clinical practice, inguinal hernias are the most common, accounting for approximately75% of all abdominal wall hernias, are not always easily diagnosed, and can manifestwith or without bulging on clinical inspection. In addition, the clinical diagnosis canbe impeded by patient-related factors, such as being in immediate postoperative period,obesity, etc.(5).One complication of a hernia is incarceration, which occurs when the opening throughwhich the hernia sac passes is wide but the sac can not be reduced. In such cases,Doppler mapping is fundamental, allowing vascularization to be identified in theherniated fat layer and in the wall of the herniated intestinal loop. When the herniapasses through a narrow opening in the abdominal wall and is voluminous, it can becomplicated by strangulation. A strangulated hernia constitutes an emergency because thehernia is irreducible and the ischemia can progress to necrosis. Contrary to what isseen in an incarcerated hernia, Doppler mapping of a strangulated hernia shows no bloodflow in the hernia sac, underscoring the emergency nature of thediagnosisTo make the correct diagnosis, it is necessary to know the anatomy of the region, as itappears on ultrasound, and its reference points; the proper examination technique (andequipment settings); and the appropriate dynamic maneuvers and patient positioning to beused during the examination. It is important to have a systematized protocol forultrasound examination so that no steps are forgotten or neglected. In general, theultrasound approach is sufficient and there is no need for additional imagingexaminations. It is fundamental that the ultrasound technician be experienced. Thecharacteristics observed in B-mode and  Doppler ultrasoundexaminations should be reported in an assertive manner, thus aiding the prescribingphysician in the decision-making process and increasing the chances of therapeuticsuccess.(7), focuses on the daily practice of physicians who performultrasound examinations of the inguinoscrotal region. The authors explore theexamination technique, the anatomy of the region, and the major differential diagnosesof inguinoscrotal pain, as well as describing a number of cases, some common and othersmore rare. Certainly, physicians who work in the field of ultrasound, especially inemergency care, will benefit from reading the article cited.A pictorial essay and review, published in this issue of RadiologiaBrasileira


Periampullary malignancies arise in the vicinity of the ampulla of Vater, a common passage for biliary and pancreatic secretions. Determining the anatomical origin of these tumors represents a diagnostic challenge. This is especially true for large tumors due to the transitional nature of this region, proximity to different structures, anatomical variations, and overlapping features among constituting structures. This determination has significant prognostic and therapeutic implications. Among them, primary ampullary adenocarcinoma is a rare malignancy that has the best overall prognosis with high rates of potentially curative resection and possible survival even in advanced disease. Due to its rarity, it is also a vague territory with no definitive guidelines regarding management and surveillance currently available. Acute gastrointestinal hemorrhage is a rare presentation of ampullary carcinoma that occurs secondary to tumor ulceration.We report an elderly male with a previously known large, initially asymptomatic periampullary mass who came for evaluation of melena and was noted to be hypotensive secondary to acute blood loss from the large tumor, later determined to be adenocarcinoma of the ampulla of Vater. The ampulla of Vater is an anatomically complex structure within the major duodenal papilla. It is a dilated passage at the confluence of the common bile duct and the main pancreatic duct, which drains their respective secretions into the second part of the duodenum through the papillary opening . There aDespite being the most common site of neoplastic transformation in the small intestine, primary ampullary malignancies are rare with an incidence of four to six per million representing 0.5% of all gastrointestinal and 7% of all periampullary malignancies -3. Some Both benign and malignant ampullary tumors can occur sporadically or in association with a genetic syndrome. The incidence of these tumors has shown dramatic increment in association with hereditary polyposis syndromes like familial adenosis polyposis (FAP) and hereditary non-polyposis colorectal cancer (HNPCC) . These mWe present an elderly male with multiple comorbidities brought for evaluation of melena, a year after a prior incidence of hematemesis. Evaluation of hematemesis on prior admission had revealed a large periampullary mass that remained untreated. It was determined to be the cause of acute gastrointestinal hemorrhage leading to hemodynamic instability, a rare association with ampullary adenocarcinoma.A 79-year-old African American male was admitted for evaluation of two episodes of melena within one day. No associated abdominal pain, nausea, weight loss, appetite changes, diarrhea, hematemesis, or hematochezia was reported. His past medical history was significant for chronic obstructive pulmonary disease (COPD), heart failure with reduced ejection fraction of 25%, coronary artery disease, dementia, and a recent large left middle cerebral artery (MCA) stroke that had led to aphasia and residual right hemiparesis.The patient was admitted a year ago for evaluation of hematemesis with a hemoglobin level of 6.9 g/dL. At that time, esophagogastroduodenoscopy (EGD) had shown a large submucosal, ulcerated mass in the area of major duodenal papilla with histology suggestive of benign small intestinal mucosa without any atypical changes Figure . A subseExamination on this admission revealed an ill-appearing, aphasic, thin male with hypotension and tachycardia. Initial testing showed a hemoglobin level of 9.9 g/dL, a blood urea nitrogen (BUN) level of 30, an international normalized ratio (INR) of 1.1, and a total bilirubin level of 0.3. After initial resuscitation with intravenous fluids and red blood cell transfusions, an emergent EGD was performed using front- and side-viewing endoscope. A fungating, polypoid mass was seen within the ampulla of Vater with blood oozing out of the duodenal papilla that failed to be controlled with epinephrine injection Figure . A hyperA biopsy specimen taken during endoscopy showed ‘invasive adenocarcinoma’ on histopathology Figure . CA 19-9Due to his poor performance status and multiple comorbidities, he was not considered as a candidate for surgery or aggressive chemotherapy. He refused any aggressive measures and was discharged to a nursing facility with an outpatient oncology follow up for consideration of palliative radiotherapy.Malignancies in the periampullary region are diagnosed based on the endoscopic, radiologic, and histopathologic data.Ultrasound (US), CT, magnetic resonance imaging (MRI), magnetic resonance cholangiopancreatography (MRCP), or endoscopic retrograde cholangiopancreatography (ERCP) is used for evaluation of this region. The EUS is used only in a few cases requiring further delineation where there is suspicion for malignancy despite negative biopsy similar to the case described. It gives the most accurate assessment of the T stage along with tumor extension and penetration within the duct and surrounding tissues, respectively. The recent use of intraductal ultrasound (IDUS) to visualize and explore bile and pancreatic ducts has increased the diagnostic accuracy .Large periampullary malignancies like the one presented, represent a diagnostic challenge in terms of determining the anatomical origin . The impIn this case, the tumor was considered to be ampullary and not pancreatic in origin due to a more indolent course of progression. Pancreatic adenocarcinomas have a highly aggressive course with a poor prognosis and a five-year survival rate of only three percent in contrast to 30%-50% noted in adenocarcinomas of ampullary origin with limited nodal involvement . This maFurther histologic subdivision of ampullary adenocarcinoma into intestinal and pancreato-biliary subtype was first demonstrated by Kimura et al. in 1994 . HistomoThe 2017 American Joint Committee on Cancer (AJCC)/Union for International Cancer Control (UICC) TNM system is used for staging of ampullary adenocarcinoma. In nonmetastatic disease, prognosis mainly depends upon the extent of tumor invasion and lymphatic spread represented by the T and N stage, respectively.Despite pancreatic duct obstruction secondary to a T3 stage tumor, our patient did not report any typical symptoms. These tumors are a rare cause of obscure gastrointestinal bleeding and more rarely, can also lead to overt hemorrhage secondary to tumor ulceration. This is seen as blood oozing out of the major duodenal papilla on a side-viewing endoscope which also facilitates visualization and biopsy of the ampullary mass. This may initially mimic other rare causes of obscure gastrointestinal bleeding (OGIB) including hemobilia and hemosuccus pancreaticus, which refer to bleeding originating in the CBD and pancreatic duct, respectively. The differentiation may become apparent during angiography when the source and tract of bleeding are delineated. The intermittent nature of bleeding in these conditions contributes to their obscurity. The CT angiogram (CTA), a noninvasive procedure has a sensitivity and specificity of 79%-90% and 95%-99% -11, respOn the other hand, invasive angiography has both diagnostic and therapeutic applications. It can detect bleeding >0.5 mL/min with a sensitivity and specificity of 30%-47% and near 100%, respectively -14. It mIt is important to recognize the limitation of using a front-viewing endoscope while looking for the source of an OGIB as it can easily miss lesions of the ampullary complex due to its location. Endoscopic sampling is associated with a high false-negative histopathology nearing 50% due to sampling difficulties that could yield inadequate specimen. Side-viewing ERCP endoscope is able to identify causes in the gut wall and must be considered while evaluating the duodenal papilla and the ampullary complex.  Pancreatoduodenectomy (PD) or Whipple’s procedure is the standard surgical procedure for ampullary cancer with potentially curative resection achieved in as high as 90% cases . Five-yeAmpullectomy is an option that can be considered in early, limited lesions <1 cm when the patient is unsuitable to undergo PD, although it is not an alternative to it. An aggressive preoperative and intraoperative assessment is needed to rule out necessary criteria like tumor invasion, high-grade morphology, lymphovascular invasion, and lymph node involvement for such a consideration. The risk of tumor recurrence and shorter disease-free survival in comparison to PD is the most important limiting factor .Ampullectomy can be performed as a minimally invasive transduodenal surgery or as an endoscopic procedure. Endoscopic ampullectomy is the surgery of choice for benign ampullary lesions <20 mm including lesions with low-grade dysplasia . It may Transduodenal surgical ampullectomy is an infrequently performed procedure which is less invasive than PD and has a lower surgical morbidity. It can be considered in Tis and T1 disease with lesions < 1 cm . There aThere is a lack of specific guidelines regarding the management and surveillance of this malignancy. Despite good resectability, recurrence remains a major issue. There is no consensus regarding the use of adjuvant chemotherapy due to the rarity of the condition, lack of definitive data, and uncertainty of benefits. However, most physicians offer adjuvant chemotherapy with (USA) or without (Europe) chemoradiation due to high recurrence rates. The drug regimen is on the lines of adjuvant chemotherapy for resected pancreatic adenocarcinoma based on fluoropyrimidine or gemcitabine -20. ChemPoor prognostic factors include the presence of nodal metastasis, poor differentiation on histology, tumor invasion, and positive postoperative surgical margins. Other factors associated with an adverse outcome are macroscopic ulceration and perineural, lymphatic, or vascular invasion .Despite a large number of studies regarding the management and prognosis of ampullary adenocarcinoma, there remains significant ambiguity evident by lack of treatment and surveillance guidelines. Our understanding remains limited due to the small sample size of these studies, rarity of the disease, and the dilemma posed by periampullary tumors. Hopefully, the increasing incidence as reported in a few studies will be met with defined guidelines in the near future .Tumors of the periampullary region represent a diagnostic challenge. Determining the tissue of origin in such cases is necessary for appropriate management and prognosis. Among these, ampullary cancer represents a rare category that has a more favorable prognosis than others. Acute gastrointestinal hemorrhage secondary to such a lesion is a rare presentation which may mimic conditions like hemobilia or hemosuccus pancreaticus requiring emergent management. Due to its rarity and lack of large multi-center trials, currently, there are no definitive guidelines for the management and surveillance of ampullary cancer. This has presented as an area of ambiguity for a disease considered to have a much better prognosis than all other periampullary tumors.


There are an estimated 1–2 cases per million per year of adrenocortical carcinoma in the USA. It represents a rare and aggressive malignancy; it is the second most aggressive endocrine malignant disease after anaplastic thyroid carcinoma. Non-secretory adrenal masses are diagnosed late due to a mass effect or metastatic disease or found incidentally .The first case report describes a 39-year-old Greek woman who presented to our department with complaints of repeated symptoms of flatulence and epigastric discomfort over a few months. The second case report is about a 67-year-old Greek woman who presented to our department after being evaluated for fatigue, mass effect, and epigastric discomfort. Both of them were diagnosed as having a nonfunctioning adrenocortical carcinoma and underwent open adrenalectomy.Approximately 60% of patients with adrenocortical carcinoma present with symptoms and signs of hormonal secretion. Our cases’ adrenocortical carcinomas were not functional. Hormone secretion is not a discriminating feature between benign and malignant adrenocortical masses. The silent clinical nature of nonfunctioning adrenocortical carcinoma results in late diagnosis, while the majority of patients present with locally advanced and/or metastatic disease.Adrenocortical carcinoma is a rare endocrine tumor with a poor prognosis that can be diagnostically challenging and demands high clinical suspicion. The work-up for adrenal masses must include determination of whether the mass is functioning or nonfunctioning and whether it is benign or malignant. There are an estimated 1–2 cases per million per year of adrenocortical carcinoma (ACC) in the USA [1]. ACC represents a rare and aggressive malignancy .Functional ACC can clinically manifest early with virilization, feminization, or Cushing’s syndrome, while non-secretory adrenal masses are diagnosed late and incidentally  due to a mass effect or metastatic disease . NonfuncOn the other hand, adrenal cysts are usually benign lesions with a discovery at autopsy range of 0.064–0.18%, in which adrenal pseudocysts account for an extremely small amount . In cases of recurrence, surgery should be considered a first-line option. However, mitotane has a valuable role in stage IV cases or in the presence of recurrent disseminated disease [Allolio survival . In case disease .Studies demonstrated mitotane’s associated toxicity and its ability in: (1) inhibition of adrenocortical steroid biosynthesis by inhibiting cholesterol side chain cleavage and 11 β-hydroxylation, and (2) induction of hepatic clearance affecting extra-adrenal disposition of cortisol . ConcernAlternatively, in patients with proven lung metastasis, the published evidence suggests that en bloc excision of involved organs and pulmonary metastasectomy could improve overall survival.Postoperative surveillance for recurrence should be performed every 3 months for the first 2 years and then every 6 months for 5 years.Adrenal pseudocysts most commonly arise from hemorrhage within the adrenal gland, secondary to extreme stress, birth, trauma, surgery, or malignancy. The differential diagnosis of adrenal pseudocysts is from endothelial epithelial and parasitic cysts and rarely from mesothelial cysts, lymphangiomas, dermoid cysts, or cystic adrenal carcinomas.Most pseudocysts on CT reveal well-demarcated, round or oval masses with fluid density with content of septa, blood, and soft tissue components. The wall of a pseudocyst seems to have occasional calcification on CT. MRI is pathognomonic for visualizing the complicated intracystic components .Regarding surgical treatment, the open approach is preferable if the patient is symptomatic, the mass is > 6 cm, or there is a possibility of malignancy. Laparoscopic surgery is indicated for small tumors or pseudocysts provided there is no peri-adrenal infiltration and subsequent capsular disruption , 16.ACC is a rare and aggressive malignancy. Non-secretory adrenal masses are diagnosed late by a mass effect, metastatic disease, or found incidentally. The work-up for adrenal masses must include determination of whether the mass is functioning or nonfunctioning and whether it is benign or malignant.We present the cases of two women who presented to our department after being evaluated for fatigue, mass effect, and epigastric discomfort. Both of them were diagnosed as having a nonfunctioning ACC and underwent open adrenalectomy. To the best of our knowledge, the first case’s nonfunctioning ACC is considered to be one of the biggest that has ever been described in the literature.


Hydatid disease is a zoonotic disease which is caused by a parasite belonging to the Echinococcus species. It may affect all part of the body mostly the liver and the lungs, involvement of the fallopian tube is a very rare.A 30-year-female presented with acute lower abdominal pain, fever and vomiting for 2 days. The past medical history was negative and she had history of surgery for hydatid cyst of the right lobe of liver 3 years before presentation. During examination there was tenderness at the right iliac fossa with guarding. Ultrasound of the pelvic organs showed a left adnexial cyst. During surgery there was an evidence of hydatid cyst involving the left fallopian tube. Left salpingectomy was performed with excision of the cyst. The patient remained three days in the hospital with no reported complication.Hydatid cyst is one of the differential diagnoses of any cystic lesion in patients from an endemic areas of hydatid disease. Hydatid cysts of the female pelvic organs may be diagnosed initially as ovarian cyst, ovarian tumor, or pelvic infection. Most of the cases are diagnosed during surgery which is the main treatment modality; anthelminthic medications may help to decrease the recurrence rate. This disease is caused by a parasite belonging to the Echinococcus species. The route of the transmission of this disease is through the fecal-oral route when the eggs are ingested with the contaminated food or water and reach the bowel of the accidental intermediate host; i.e. the human. The eggs are hatched and transmitted by the blood circulation to all parts of the body, hepatic and pulmonary involvements are the commonest, other organs may be affected with variable frequencies, hydatid cyst of the fallopian tube is a rare finding and in most case it is associated with involvement of other body organs ,2.Most cases are diagnosed incidentally or the disease may cause nonspecific symptoms. Pain is the most frequent symptom; large cysts may cause pressure symptoms, or the cyst may present with complications such as infection and rupture .Imaging may be useful in making a correct preoperative diagnosis especially when the cyst contains the floating membrane or when contains many daughter cysts, it may also be helpful in delineating the anatomy and showing the involvement of other organs .The work of this report case has been reported in line with the SCARE 2018 criteria .2A 30-year-female presented with acute lower abdominal pain, fever and vomiting for 2 days. The past medical history was negative and the patient had history of surgery for hydatid cyst of the right lobe of liver 3 years before presentation, no spillage was reported during the first surgery, the patient received postoperative anthelminthic drugs for 3 months after the first surgery.2.1There pulse rate was 110 beats /minutes, the blood pressure was 105/65 mmHg, and the temperature was 38.6 degrees of Celsius.During abdominal examination, there was tenderness at the right iliac fossa with guarding.2.2Ultrasound of the pelvic organs showed a left adnexial cystic lesion measuring 60*50 mm with clear contents.2.3Diagnostic laparoscopy was performed which showed an evidence of inflammatory mass in the left adnexial region, the surgery was then converted to the open one. There was an evidence of hydatid cyst involving the left fallopian tube Left salpingectomy was performed with excision of the cyst, the surgical field was isolated with surgical sponges soaked with chlorhexidine solution to control any intraoperative spillage 2.4The patient remained three days in the hospital with no reported complication, she received anthelminthic drugs for 3 months after the surgery.3Hydatid cyst is one of the differential diagnoses of any cystic lesion in any part of the body in patients from an endemic areas of hydatid disease such as the Mediterranean region. Once hydatid cyst is diagnosed, other organ involvement should be excluded particularly pulmonary involvement as it has a priority for management before any surgical intervention for other anatomical sites ,5.Hydatid cysts involving the female pelvic organs may be diagnosed initially as simple or complicated ovarian cysts, dermoid cyst, ovarian tumor, or pelvic infections and abscesses. Most of the cases are diagnosed during surgery ,6.The blood tests are normal in the majority of cases, however, in cases of infected cysts the white blood cells may be elevated, the eosinophil count may be elevated in some cases as an allergic manifestation to the parasite .In the review of literature only few cases of primary involvement of the female pelvic organs are reported. Involvement of the fallopian tubes is very rarely reported, and less than 30 cases are reported. Such cases are usually diagnosed as para-ovarian cysts and most cases are diagnosed during surgery ,8.Surgery is the main treatment modality, but medical treatment with anthelminthic medications such as albendazole may be effective in some occasions, the main benefit of the medical therapy is in the form of adjuvant therapy after surgery to decrease the recurrence rate. During surgery the most important step is preventing spillage of the content of the cyst to avoid recurrence, and some patients may develop severe allergic reaction to the cyst fluid, cases of intraoperative deaths have been reported. Follow up is recommended to detect early recurrence and to detect other organs involvement ,9.None.Ethical approval has been exempted by my institution for reporting this case.Written informed consent was obtained from the patient for publication of this case report and accompanying images.The concept of reporting the case, data recording, and drafting the work done by Dr Dildar Haji Musa and Dr Ayad Ahmad MohammedDr Dildar Haji Musa took the consent from the patient for publishing the case.Final approval of the work to be published was done by Dr Ayad Ahmad Mohammed.This work is case report and there is no need of registrationDr Ayad Ahmad Mohammed is guarantor for the work.Not commissioned, externally peer-reviewed.After surgery I am afraid of recurrence of the cyst, I should keep regular checks with my doctor.The author has no conflicts of interest to declare


In this report, we want to emphasize how a laparoscopic bariatric surgical procedure, in experienced hands, has shown to be a valid alternative for the hemorrhage control and the removal of a gastrointestinal tumor in a life‐threatening situation. They arise from the Interstitial cells of Cajal (ICCs) that are implicated in the regulation of gut peristalsis, with a pacemaker role. GISTs are more often located in the stomach (56%) although they can arise in any portion of the digestive tract.Although the treatment of gastrointestinal stromal tumors with tyrosine kinase inhibitors is one of the best examples of the advances of molecular targeted therapy over the last two decades, surgical resection with clear margins remains the mainstay of cure.Herein, we report the cases of two patients from two different Italian hospitals who have undergone a successful laparoscopic sleeve gastrectomy in emergency following the acute bleeding of a gastric GIST.Laparoscopic sleeve gastrectomy (LSG) is the most commonly carried out bariatric surgical procedure worldwide; by removing approximately 75% of the stomach from the greater curvature side, it acts as a restrictive procedure also capable of changes in the secretion of gastrointestinal hormones involved in the weight loss.In this report, we present two cases in which a gastric GIST is not an incidental finding during a laparoscopic bariatric operation but a laparoscopic bariatric surgical procedure, in experienced hands, has shown to be a valid alternative for the hemorrhage control and the removal of a gastrointestinal tumor in a life‐threatening situation.22). Electrocardiogram (ECG) and serum troponin levels excluded an ischemic cardiac event, but the results for complete blood cell counts and hemocoagulation revealed a hemoglobin value of 9.6 g/dL. Two days before, the patient had undergone an esophagogastroduodenoscopy  was used to aspirate and lavage the stomach as well as confirming the localization of the bleeding lesion in the greater curvature at the gastric fundus. Subsequently, a laparoscopic sleeve gastrectomy (LSG) was carried out. Five trocars have been placed in the upper abdominal quadrants: one 12‐mm trocar above the umbilicus in the midline, three 12‐mm trocars in right subcostal, subxiphoid, and left subcostal, and one 5‐mm trocar in the left anterior axillary line. The gastric resection was performed using a linear stapler (Echelon Flex™ 60 Endopath®) applied alongside a 38‐Fr bougie. The next step has been a methylene blue dye test in order to check the sealing of the staple line that was additionally reinforced by nebulization with cyanoacrylate sealant (Glubran 2®) used also to create an adhesion with the greater omentum as a chemical omentoplasty.E coli urinary tract infection which was successfully treated with antibiotics. The diagnosis of gastric GIST was confirmed on the surgical specimen that showed a polypoid mass with hemorrhagic foci 40 × 35 mm in size; surgical resection margins were negative (R0). The mitotic rate was <1/50 HP; CD34, CD 117/C‐kit, and smooth muscle actin were positive; the Ki67‐MIB1 immunostaining indicated a low proliferative rate (count rate 4%). The tumor was staged as GIST of gastric origin in the very low‐risk category so that a systemic adjuvant chemotherapy was not needed2).The patient's postoperative course was uneventful, except for a left subcostal wound infection which was opened and drained at the bedside and for a 32 and a past medical history of hypertension, hyperthyroidism, and atrial fibrillation (AF) under treatment with direct oral anticoagulants (DOACs). The patient also had a history of previous surgery represented by a laparotomic left hemicolectomy for a benignant colonic stenosis. She was initially managed with intubation and received mechanical ventilation, and once hemodynamic stability was achieved, her conditions worsened due to unexpected upper gastrointestinal bleeding (UGIB). She was given a blood transfusion of 4 units of red blood cells, but her hemoglobin value continued to stay under 9.0 g/dL. The following OGD showed an ulcerated submucosal lesion of 3 cm at the greater curvature of the gastric fundus  in emergency state for the acute onset of a respiratory failure accompanied by high‐frequency atrial fibrillation (AF). She had a body mass index (BMI) of 29.52 kg/mIn the management of this acute situation in order to avoid any further delay of surgical intervention, the patient was prepared for an emergency laparoscopy. Four trocars have been placed in the upper abdominal quadrants: one 12‐mm trocar above the umbilicus in the midline, two 12‐mm trocars in right and left subcostal, and one 5‐mm trocar in the right anterior axillary line. The exploration of the abdomen cavity showed the adhesions attributable to the previous lower abdominal surgery as well as the cirrhotic changes of the parenchymal status of the liver. Once the stomach has been exposed, a nodule appeared on its surface near the greater curvature at the fundus. Subsequently, a laparoscopic sleeve gastrectomy (LSG) was carried out. The gastric resection was performed using a linear stapler (Echelon Flex™ 60 Endopath®) applied alongside a 38‐Fr bougie. The gastric specimen  is the only potentially curative option for GISTs. In the case of gastric location, which is the most common, based on the assumption that GISTs do not have the typical lymphotropism, infiltrating behavior and propensity to metastatize of adenocarcinomas, a less extensive surgery like in the case of wedge resections, is suitable to treat the disease and spare the stomach.In the Case 1, the patient was admitted to the emergency department with hematemesis and melena 48 hours following the endoscopic biopsy of a suspicious mass of the greater curvature at the gastric fundus, 30 mm in size. Conventional endoscopic forceps biopsy of GISTs is difficult and does not obtain enough tissue for a definitive diagnosis; EUS‐FNA (Endoscopic ultrasound‐guided‐fine needle aspiration biopsy) is the most established tissue sampling method.In the Case 1, the exploration of the abdomen cavity showed no evidence of gastric exophytic lesions; an intraoperative endoscopy was useful in order to locate the mass and guide the chose of the proper resection. In the Case 2, the location of the tumor was well defined by the presence of a nodular lesion located on the greater curvature at the anterior wall of the gastric fundus.In both cases, in light of the favorable location of the mass at the greater curvature, in the absence of distant disease, laparoscopic sleeve gastrectomy (LSG) has been chosen in order to treat the bleeding, remove the potential GIST, and spare the stomach calibrating the resection by the use of a 38‐Fr bougie placed against the lesser curvature. In addition, the fact that the procedure was carried out by experienced bariatric surgeons allowed to be more confident in facing emergency surgery with the use of a standardized laparoscopic technique, which reduced also operative time. Although LSG is considered an easier technique if compared with other bariatric procedure since no anastomosis is required, it includes some key technical points like the dissection of the stomach from the spleen, necessitating adequate laparoscopic expertise. Moreover in our cases, the presence of a blood‐filled stomach due to the hemorrhage may have had implications on the sealing of the staple line. In the Case 1, aerosolized cyanoacrylate sealant Glubran 2® has been used as a reinforcement of the staple line to prevent leak and bleeding.2). Wang Y et al reported their experience of laparoscopic sleeve gastrectomy in the treatment of GISTs in morbid obese patients. They found the LSG to be the best choice for obese patients combined with GISTs. It can remove tumors and excess stomach away at the same time.The patients’ postoperative recovery was uneventful for major complications. Pathology reports showed in both cases complete surgical resection (R0); the tumors were diagnosed as gastric GISTs in the very low‐risk/low‐risk category, and adjuvant chemotherapy was not necessary.5Gastrointestinal stromal tumors (GISTs) are the most common mesenchymal tumors of the gastrointestinal tract. Surgical resection with free margins of tumor disease (R0) is their only curative option. In the case of potentially resectable gastric tumors, laparoscopic wedge resections are considered the standard therapy. This report describes the application in two cases of the laparoscopic sleeve gastrectomy for the emergency treatment of a bleeding gastric GIST. It can be concluded that a laparoscopic bariatric surgical procedure, in experienced hands, has shown to be a valid alternative for the hemorrhage control and the removal of a gastrointestinal tumor in a life‐threatening situation.The importance of this surgical speciality has too often been underestimated, while the clinical, social, and economic benefits cannot be ignored.The authors declare that they have no competing interests.Consent for publication: Written informed consent was obtained from the patients for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor‐in‐Chief of this journal.NC: researched the data, wrote the manuscript, and contributed to the discussion. AP: wrote the manuscript, contributed to the discussion, and reviewed the manuscript. CN: performed one of the bariatric interventions. AB: contributed to the discussion and reviewed the manuscript. GM: wrote the manuscript, contributed to the discussion, and reviewed and edited the manuscript. All authors read and approved the final manuscript.


H3F3A point mutations at G34 (NET-H3-G34) occur most often in cerebral hemispheres of teenagers and young adults, and have a generally adverse prognosis. These tumors have been histologically classified as glioblastoma or primitive neuroectodermal tumor (PNET) in the past, and have not been defined as a separate entity in the revised WHO classification of tumors of the CNS 2016. Here, we report two cases of NET-H3-G34 with glial and dysplastic ganglion cell components affecting teenagers. Patients were treated with surgery and radiochemotherapy with temozolomide. One patient underwent partial resection and deceased 21 months after diagnosis, while the other patient is alive without evidence of disease 15 months after total resection. So far, a dysplastic ganglion cell component has not been described in NET-H-G34, and its presence raises a possible relation to (anaplastic) gangliogliomas. Genome-wide copy number analysis did not provide unequivocal evidence that these tumors represent anaplastic variants of gangliogliomas, as opposed to NET-H3-G34. Our observations expand the morphologic spectrum of NET-H3-G34. Further cases of NET-H3-G34 with dysplastic ganglion cells should be clinically followed to find differences or similarities in their biological behavior, as compared to NET-H3-G34 and anaplastic gangliogliomas.The recently described malignant neuro-epithelial tumors with histone  H3F3A point mutations at G34 (NET-H3-G34) occur most often in cerebral hemispheres of teenagers and young adults, and have a generally adverse prognosis, with reported median PFS and OS of 8–9 months and 12–22 months [H3F3A G34 mutations has not yet been reported.The recently described malignant neuro-epithelial tumors with histone Case 1 affected a 16-year-old male patient with significant weight loss in 1 year, headaches and visual impairment developing over 2 months. Magnetic resonance imaging (MRI) demonstrated a left-sided fronto-temporo-insular mass, hypointense on T1 (Fig. 3, extending to the falx, hyperintense on FLAIR-weighted images, slightly hypointense in T1 with small hyperintense spots, compatible with calcifications and blood. The tumor was sharply demarcated from the surrounding brain parenchyma, which presented no significant signs of edema. The patient underwent total resection and radiochemotherapy with temozolomide, and 15 months after surgery was asymptomatic, without radiological evidence of residual or progressive disease.Case 2 involved a 14-year-old male patient presenting with headaches for 8 weeks. Computed tomography scans showed a partially hyperdense tumor with calcifications and slight contrast enhancement. MRI revealed a right occipital cortical/−subcortical tumor with a cystic component, measuring 5.3 × 4.6 × 6.8 cmH3F3A G34R mutation in both cases (Fig. CDKN2A locus, as well as indication of chromothripsis of chromosome 10. Case 2 displayed amplification of CDK6 and loss of chromosome 3q and 4q (Fig. Histologically, both tumors showed mixed neuronal and glial components Fig. f, j posises Fig. e. Classises Fig. . Case 1 MYCN and DLX6, among others [A ganglion cell differentiation in NET-H3-G34 is not surprising, as the specific pattern of H3K36me3 binding in these tumors upregulates genes particularly involved in neuronal morphogenesis and differentiation, such as g others . Howeverg others . ChildreCDNK2A, which have been described in a case of AGG [CDK6 amplification seen in 10% of NET-H3-G34 [The radiological findings described for gangliogliomas and “NET-H3-G34” do not enable a clear distinction between them , 13. Cyte of AGG , and aree of AGG , as welle of AGG . Case 2 T-H3-G34 . These aT-H3-G34 , and alsT-H3-G34 . HoweverT-H3-G34 .H3F3A-K27 M mutations have been detected in AGG [H3F3A-G34 mutations have not been reported in AGG. One should keep in mind that the histological spectrum of mutations associated with tumor entities is often wider than initially described – a good example is the growing list of tumors with BRAF-V600E mutations or histone H3-K27 mutations. Occurrence of a certain mutation, therefore, may not per se define a tumoral entity. On the other hand, ganglion cell differentiation is not sufficient for the diagnosis of a ganglioglioma, as this feature has been documented in different glial tumors, including oligodendroglioma IDH-mutant and 1p/19q codeleted [Histone d in AGG , but so odeleted . Howeverodeleted  seems ad


Herein, we present a case of a 64-year-old male presenting with diplopia that was found to have a left orbital Masson’s tumor. It is necessary to differentiate it from malignant angiosarcoma as complete excision is often curative. The following case emphasizes the modalities that can be used for diagnosis as well as the appropriate treatments. Masson’s tumor, also known as intravascular papillary endothelial hyperplasia (IPEH), is a rare benign neoplasm that affects the head, neck, and upper extremities. There are few (less than 20) periorbital cases that have been reported in ophthalmologic literature -4. MassoA 63-year-old Caucasian male with past medical history of diabetes mellitus and past ocular history of bilateral cataract extraction with intraocular lens (CEIOL) presented to an outside vitreoretinal specialist one-month post-CEIOL due to diplopia and possible retinal detachment. Best corrected visual acuity (BCVA) was measured as 20/25 OU, pupils were reactive without afferent pupillary defect (APD) OU, and intraocular pressure (IOP) was measured as 21 and 18 mmHg, respectively. Optical coherence tomography of both eyes revealed a flat and attached macula OU. Fundus photos revealed irregular appearance of nasal retina OS. The patient was subsequently diagnosed with nasal choroidal effusion and was given cyclopentolate, prednisolone, and oral dexamethasone.The patient returned for follow-up one week later with no resolution of symptoms and new complaint of an inferior “knot” OS with pain and pressure. BCVA and pupillary examination were unchanged. IOP was measured as 16 and 18 mmHg, respectively. Dilated fundoscopic examination was performed, and treatment was discontinued as the choroidal effusion had resolved.Further follow-up three weeks later revealed no change in subjective pain and pressure symptoms OS. The patient described his diplopia as horizontal and binocular. Further questioning revealed that his symptoms began two months ago after CEIOL. BCVA, pupillary examination, and IOP were unchanged. Dilated fundoscopic examination was notable for a return of his choroidal effusion. MRI of the orbits with and without contrast ordered which revealed an enhancing 2.5 x 1.6 x 3.2 cm left orbital lesion involving both the intraconal and extraconal compartments and contributing to the left-sided proptosis (Figures At this time, the patient was referred to oculoplastics for further evaluation. Nineteen days after the MRI, the patient reported continued diplopia, pain, and proptosis OS. His ocular examination was as follows: BCVA of 20/25 OU, pupils reactive with no APD OU, IOP of 21 and 27 mmHg, respectively. His extraocular movements were full OD and -1 in all gazes OS. His MRI was reviewed, and the patient was scheduled for a biopsy of the orbital mass. An approximately 2.1 x 0.5 cm mass submitted for routine sectioning revealed papillary formations and arborizing channels contained within a fibromuscular wall compatible with a dilated vascular space. Variably sized vascular channels intersect the interior of the mass, and these channels contain erythrocytes and serum. Papillae are characterized by a hyalinized fibrovascular core lined by plump mononuclear cells that occasionally form syncytia. CD31 immunohistochemical marker for endothelial cells richly outlines the furcating vascular network Figures -7.The final diagnosis was IPEH, also known as Masson’s tumor.The patient subsequently underwent orbitotomy with excision with removal of approximately 80% of the tumor. The other 20% was inaccessible as it was circumferential to the optic nerve. The patient had improvement of pain, diplopia, and proptosis post-surgery, and a follow-up CT was scheduled three months later.IPEH was first described by Masson in 1923 as “vegetate intravascular hemangioendothelioma”. It is cIPEH has a predisposition for the head, neck, and upper extremities, and it may present in both the orbit and eyelid. It often presents as a firm, cystic, freely mobile, reddish/purple swelling that may increase in size ,4. The dThere are three forms of IPEH. Primary IPEH forms within a dilated vascular space with a preference for extremities. Secondary or reactive IPEH is the most common and is predominately found in the musculature. It originates within pre-existing vascular malformations such as varix, lymphangioma, or cavernous hemangioma. Tertiary IPEH is the rarest, forms within hematomas, and is extravascular.Evidence of unilateral proptosis in an elderly patient should prompt imaging to rule out mass effect. MRI is the preferred imaging technique for identifying IPEH. Lesions appear isointense on T1-weighted imaging and hyperintense on T2-weighted imaging with homogenous enhancement following contrast administration .Recognizing IPEH and differentiating from malignant lesions such as angiosarcoma is important to prevent morbidity related to unnecessary chemotherapy, radiation, and surgery. This case emphasizes the importance of early diagnosis and a strong referral base for appropriate diagnosis and treatment. Even in cases where surgical correction is not entirely curative, a significant improvement of symptoms can be obtained post-surgery. This patient received appropriate care and surgical correction to remove 80% of the tumor, and he subsequently had significant quality-of-life improvements.


Florid cemento-osseous dysplasia (FCOD) is a rare, benign, slow growing, multifocal fibro-osseous tumour originating from the periodontal ligament. The lesion is characterised by regular bone-structure replacement with fibrous tissue and dysplastic bone. The initial characteristics of FCOD, depending on localisation, resemble those of periapical lesions of inflammatory origin, potentially leading to misdiagnosis. In the absence of infection signs, the treatment of choice is conservative therapy with antibiotics and regular follow-up examinations.We report the case of a 39-year-old Caucasian woman with sensory disturbance along the inferior alveolar nerve for >2 weeks. The involved teeth were vital and the digital orthopantomogram indicated multiple bilateral periapical brightening. Biopsy was indicated to exclude malignancy and to confirm the FCOD diagnosis with predominantly reactive calcification and cell-rich connective-tissue reaction. The sensory disturbances disappeared after the biopsy. Further conservative therapy included regular radiological examinations every 2 years to prevent surgical intervention due to infection risk.Sensory disturbances around the nervus alveolaris inferior are well-known complications of dental and maxillofacial operations and of malignant tumours. Sensitivity disorder as a first symptom of fibro-osseous dysplasia has not been previously described. Although uncommon in its clinical behaviour, FCOD was finally diagnosed after thorough clinical, radiological, and histological examinations.Paraesthesia combined with FCOD has not been previously described. Correct diagnosis is essential for proper treatment and to avoid further complications even when the clinical symptoms appear unrelated. The current classification of bone-related lesions, launched in 2005 by the World Health Organization, does not account for the various phenotypes of fibro-osseous lesions .The term florid cemento-osseous dysplasia (FCOD) was introduced in 1976 to describe an extensive variant of a reactive fibro-osseous process, known as cementoma . FCOD isIn radiological examinations, the lesion manifests as multiple, lobular, well-marginated, non-expansile intraosseous masses of varying internal lucency and sclerosis, surrounding the root apices of vital teeth .While all CODs share similar microscopic features, FCOD is distinguished by a multifocal distribution, involving two or more quadrants of the maxilla and mandible, often in a bilateral symmetric fashion. Female patients, specifically of African-American ethnicity, are more often affected than their male counterparts. The age at the time of diagnosis ranges from 19 to 76 years [A systematic review found that in 59% of cases, the lesion occurred in Afro-Americans, in 37% of cases in Asians, and in 3% in Caucasians. Of all patients, 97% were female .The origin is unknown, but currently, a reactive process is assumed. In most cases, the patients have no related heredity and no family history of similar lesions .Because of the asymptomatic presentation of FCOD, the lesion can remain undetected for several years and is therefore often diagnosed accidentally during regular radiological examinations ,11. In sDue to their rarity, histological diagnosis of fibro-osseous lesions is challenging, as they are easily confounded by similar tumors ,7.Unnecessary biopsy and surgery should be avoided, as the hypo-vascular lesions are susceptible to infection, osteomyelitis, and bone necrosis . AccordiSensory disturbances in the region of the nervus alveolaris inferior (NAI), including anaesthesia, paraesthesia, hypoaesthesia, and hyperaesthesia, are well known complications of malignant tumours and of dental and maxillofacial operations . InfectiThe aim of this case report is to highlight the clinical and radiographic features of FCOD to raise the general awareness for this rare disease. The patient was managed in a private dental office. We report the present case in accordance with the SCARE criteria .2We report the case of a 39-year-old Caucasian woman who had persistent paraesthesia of the right NAI for longer than 2 weeks.She had unremarkable drug, family, and psychosocial histories and was a non-smoker. Clinical examination revealed throbbing pain at the lingual and buccal sites of the mandible.The orthopantomogram revealed multiple radiolucent, periapical lesions in different sizes and shapes. The findings were non-symmetrical, arranged bilaterally, and covered the entire lower jaw. Teeth 36 and 37 showed a large radiolucent periapical area, only separated via a small sclerotic septum. The origin of the lesion at tooth 36 appeared to be the distal radix and clearly expanded over to the apical of tooth 35. The canalis alveolaris inferior sinister appeared to be infiltrated at region 37. Further radiolucent lesions could be found periapically at region 33, almost reaching tooth 32. In the right mandible, similar lesions could be seen at regions 46, 42, and 41. Yet, the nerve channel appeared to be intact. The tumorous lesions to the front almost appeared as a single, large body. Although, every pathology arose around the radices, no root resorption was visible, and the performed vitality test was positive for all involved teeth. In contrast, no radiological pathologies could be found in the upper jaw.On an orthopantomogram obtained 13 years earlier, some of the radiolucent lesions of the lower jaw could already be assumed at regions 37, 36, 32, and 46 (Computed tomography (CT) of the mandible 13 years after the first CT, a high-resolution 1-mm stratification of the horizontal mandibular branch, showed multiple resorption zones periapical directly in the area of incisors 31, 41, and 42. In addition, periapically, osteolysis was observed in the area of premolar 35 and molars 36, 37, 46, and 47. The corticalis was thinned and displaced ventrally, whereas the alveolar canal had remained intact. The lesion was more distinct on the left side than on the right. There was no periosteal reaction and no spicules Based on the medical history, clinical symptoms, and radiological findings, a differential diagnosis of fibro-osseous disease was decided.However, due to the existing paraesthesia in the area of the left NAI, a histopathological examination was necessary to exclude malignancy. The specimens were acquired from the area around region 46.A combination of surgical bur and the chisel technique was used for bone removal. First, the surgical bur was cut under water cooling conditions and then the bone was removed with the chisel technique. The semi-lunar flap was selected as the ideal access path for the sampling The histological examination revealed calcifications or map-type neoplasms in front of a relatively cell-rich, fibrous stroma, with a small number of histocytic cell elements. The histological appearance was compatible with the diagnosis of FCOD. Typical of FCOD, as in our case, the teeth were vital despite the apical localisation of the lesions After the biopsy, the paraesthesia of the bottom lip disappeared. Six months later, the patient described a feeling of intense painful pressure in the area of the former FCOD. Although uncommon and aware of the possible risks, we decided to perform a surgical procedure. The patient underwent surgery under general anaesthesia performed by the head of the dental department. Using an intraoral approach, a mucoperiosteal flap was raised. The aim was to partially remove the tumour, especially at the area of the nervus mandibularis, which we suspected was the origin of the symptoms. At that time, the patient also received perioperative antibiotics. However, no evidence of FCOD could be detected upon specimen examination. The specimen appeared as regular modelled bone with physiological circulation. The patient received postoperative antibiotics, a nonsterial anti-inflammatory agent, and a proton pump inhibitor, and was discharged in stable cardiorespiratory condition. For the following 6 months, the patient remained under regular follow up. Neither further increase in size of the lesion nor newly occurred spots were detected.Biopsy and histopathological examination confirmed the suspected diagnosis. Due to the successful operation, with no postoperative complaints and uneventful wound healing, the patient was very satisfied. The therapy was very well tolerated, and the patient reported no pain.3We presented the case of a young Caucasian woman with large FCOD affecting the mandible. Our patient and her family did not have hereditary diseases. Further, there has been no documented case of familial FCOD .FCOD is normally described as a symmetrically-arranged lesion affecting the left and right part of the lower jaw ,7. HowevFurther we can confirm that FCOD radiologically appears as a well-defined lesion that includes the apices of vital teeth and can spread across multiple quadrants , as for To our knowledge, sensory disturbances, as a first sign of FCOD, have not been described in the literature . The medWe were very well aware of the fact that an invasive treatment could trigger an infection in the hypo-vascular lesion, but due to the paraesthesia, a biopsy was essential to exclude malignancy.Further therapy includes radiograph examinations every 2 years and surgical procedures should be avoided due to the risk of infection . We beliIn principle, surgery is strictly contraindicated, but in our case of a sensory disorder, it was useful to eliminate malignancy and relieve pain and remove pressure. If the size of the lesion increases, a modelling osteotomy will have to be considered.4This report describes a case of FCOD, a rare and benign pathology. Unlike its normal clinical behaviour, the patient in our case had symptomatic paraesthesia in the mandible as a first sign of the lesion. To the best of our knowledge, this is the only documented case of such symptomatic FCOD in the literature. The growth of the lesion could be documented for more than 13 years. We assume that the expanding lesion triggered the sensory disorder in the mandible when it reached the nerve. Sensory disturbances in cases of FCOD are not common and by the time they appear, they are usually due to infection of the lesion or iatrogenic injury during an operation or an endodontic treatment; none of which applied in our case. Another particularity is the slightly asymmetric growth of the lesion and the ethnicity of our patient. It is our concern that every radiolucent apical lesion on an orthopantomogram should be carefully considered for a more infrequent differential diagnosis. Therefore, a vitality test should be performed before any further treatment is undertaken to avoid misdiagnosis and unnecessary interventions.The authors declare that there is no conflict of interest.This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.The ethical approval has been exempted by our institution.The patient received a thorough explanation of this report gave her oral and written informed consent to be included in this report as well as for publication of these case, anonymous data, and pictures. A copy of the written consent is available for review on request.Pascal Grün and Patrick Bandura: study concept and design, writing the paper.Andrew Grün, Walter Sutter and Oliver Meller: data collection, analysis and discussion of data.Dritan Turhani: final approval of the version to be published.Not applicable.The corresponding author is the guarantor of submission.Not commissioned, externally peer reviewed.


Ultrasonography and MR cholangiopancreatography revealed cholecystolithiasis, obstructive choledocholithiasis, abscess of the ligamentum teres hepatis and left portal thrombosis. Under ultrasound guidance, pus was aspirated from the abscess and the patient was started on broad-spectrum intravenous antibiotics, insulin and low-molecular-weight heparin. He subsequently underwent endoscopic retrograde cholangiopancreatography with sphincterotomy and stone extraction. On the tenth day post admission, he underwent laparoscopic cholecystectomy and excision of the ligament. The patient made an uneventful recovery and was discharged on the seventh post-operative day. On follow-up, the patient remained asymptomatic with normal biochemical parameters. This article highlights the importance of suspecting and identifying an abscess of the ligamentum teres hepatis when a patient with acute abdomen presents with a sausage-shaped right parasagittal mass, especially in the setting of cholangitis, cholecystitis or omphalitis.An abscess of the ligamentum teres hepatis is a very rare cause of acute abdomen and can present a diagnostic dilemma. A 40-year-old diabetic male presented with obstructive jaundice and cholangitis. An ill-defined, sausage-shaped, tender parasagittal supraumbilical mass was palpable on the right side. Murphy’s sign was negative. Laboratory investigations revealed polymorphonuclear leukocytosis (total leukocyte count 19,000 mm Localized accumulations of pus or abscesses are ubiquitous. However, an abscess of the ligamentum teres hepatis or round ligament  is a very rare occurrence. Only about 16 cases in adults and 6 cases in neonates and infants have been reported until 2016. An abscess at this location presents as an acute abdomen and can be perplexing. It may be erroneously diagnosed as an abdominal wall abscess and managed non-surgically, which is usually unsuccessful. Hence, familiarity with this entity and its early recognition is essential for the radiologist, surgeon and paediatrician. This article presents a case of an abscess of the liagamentum teres hepatis associated with left portal thrombosis consequent to cholecystolithiasis, obstructive choledocholithiasis and cholangitis. The purported aetiopathogenesis, imaging findings and management of this condition are also discussed.−1, regular but low in volume, with a blood pressure of 110/70 mmHg and respiratory rate of 18 min−1. He had marked icterus. Murphy’s sign was negative. An ill-defined, sausage-shaped, tender parasagittal supraumbilical mass was palpable on the right side. Erythema was noted on the skin around the umbilicus; however, there was no umbilical discharge. Laboratory investigations revealed polymorphonuclear leukocytosis , with elevated liver enzymes  and conjugated hyperbilirubinaemia (16 mg dl−1). His blood glucose was 250 mg dl−1. The rest of the biochemical tests were normal.A 40-year-old male, suffering from diabetes mellitus for the past 2 years and on irregular treatment, was brought in with a history of progressive jaundice of 10 days’ duration, and fever and right upper abdominal pain for the past 3 days. There was also a history of passage of clay-coloured stools. The patient mentioned occasional post-prandial self-limiting right upper quadrant pain in the past, for which he did not get himself investigated. On examination, he was found to be febrile with a temperature of 102 °F and appeared dehydrated. His pulse was 100 minTransabdominal ultrasonography of the abdomen revealed cholecystolithiasis. However, there were no signs of cholecystitis. Bilobar dilatation of the intrahepatic biliary radicles was noted and the common bile duct measured 20.0 mm in diameter. A calculus measuring 8.2 mm was seen in the middle segment of the common bile duct. A tubular cystic structure with echogenic debris was visualized, extending from the umbilicus off midline on the right side to the inferior surface of the medial segment of the left lobe of the liver. A subsequent MR cholangiopancreatography  also showed cholecystolithiasis and choledocholithiasis with an 8.0 mm-sized calculus in the distal part of the supraduodenal common bile duct. The entire biliary tree proximal to the calculus was dilated. A sausage-shaped cystic lesion with thick shaggy walls was seen on the right side, extending from the umbilicus cranially up to the umbilical fissure. This lesion measured 16.0 × 3.5 × 3.5 cm . The normal flow void in the left branch of the portal vein was replaced by hyperintense signal intensity –3. TheseEscherichia coli on culture) was aspirated from the abscess on the day of admission and the patient was started on broad-spectrum intravenous antibiotics and insulin. He was also started on injectable low-molecular-weight heparin. The next day, he underwent endoscopic retrograde cholangiopancreatography with sphincterotomy and stone extraction. The patient gradually recovered with subsidence of fever and improvement of the clinical parameters. On the tenth day post admission, he underwent laparoscopic cholecystectomy and excision of the residual abscess cavity. The patient made steady recovery after the surgery and was discharged on the seventh post-operative day. Ultrasonography performed on the day of discharge was normal except for evidence of cholecystectomy. Colour Doppler flow imaging revealed normal colour flow in the left portal vein. On follow-up, the patient remained asymptomatic with well-controlled blood sugar and normal liver function.Under ultrasound guidance, 80 ml of pus , which is the obliterated left umbilical vein, measures approximately 17 cm in length and runs along the inferior margin of the falciform ligament from the umbilicus to the umbilical fissure (between the left and quadrate lobe) on the inferior surface of the liver.An abscess of the ligamentum teres hepatis or round ligament is a very rare occurrence. Only about 16 cases in adults and 6 cases in neonates and infants have been reported until 2016. Most cases in adults have been reported in the elderly population.10 Lipinski et al11 proposed contiguous spread of infection from omphalitis via the round ligament as the cause of this abscess in infants. The paraumbilical veins connect the superficial periumbilical venous network to the left portal vein to form a portal–systemic venous anastomosis. This venous network might explain the mechanism of spread of infection from omphalitis into the falciform ligament in the absence of a round ligament (post-operative cases).7 The cholecystic veins drain directly into the portal vein and the pericholedochal venous system. It is likely that the infection spreads from the biliary tree via the paraumbilical veins to the falciform ligament, leading to abscess formation.5 Lymphatic spread of infection has also been postulated as a cause of abscess formation.3The reported causes of abscess of the ligamentum teres hepatis are acute calculous cholecystitis, cholangitis and pancreatitis in adults, and omphalitis and infected ventriculoperitoneal shunt in children.4 In our patient, cholangitis was likely to have caused portal pyaemia, portal vein thrombosis and spread of infection to the ligamentum teres hepatis.The major causes of portal vein thrombosis are liver cirrhosis, neoplasms and haematological malignancies, infection and pancreatitis. In the absence of cirrhosis or neoplastic disease, approximately 10–25% cases of portal vein thrombosis are associated with sepsis. Portal pyaemia and thrombosis may occur secondary to appendicitis, biliary tract infection, post-abdominal surgery sepsis, amoebic colitis, acute necrotizing pancreatitis and diverticulitis.Potential complications of missed or delayed diagnosis of an abscess of the ligamentum teres hepatis are peritonitis, sepsis, mass effect causing extrinsic intestinal obstruction and portal thrombosis.4 In other cases, a more delayed approach may be followed, as in the case of our patient. Antibiotic therapy and surgical excision are the mainstay of treatment; however, conservative management with endoscopic biliary drainage and antibiotics has also been successful.9 Any predisposing condition such as acute calculous cholecystitis, cholangitis, choledocholithiasis and omphalitis will also need to be simultaneously addressed.Management of ligamentum teres abscess involves use of broad-spectrum intravenous antibiotics to cover the gastrointestinal flora and elimination of the abdominal focus of infection. Aspiration of pus followed by surgical excision of the abscess should be performed immediately on diagnosis if features of peritonitis are present, owing to the risk of extensive spread and severe peritonitis.via the paraumbilical veins to the falciform ligament can lead to abscess formation. This entity should be suspected when a patient with acute abdomen presents with an elongated supraumbilical right parasagittal mass, especially in the setting of cholangitis, cholecystitis or omphalitis.An abscess of the ligamentum teres hepatis is a very rare cause of acute abdomen that can present a diagnostic dilemma. Intra-abdominal infections leading to portal pyaemia and consequent spread of infection Abscess of the ligamentum teres hepatis is a rare cause of acute abdomen.It should be suspected when a sausage-shaped, right parasagittal supraumbilical mass is palpable in the setting of cholangitis, cholecystitis or omphalitis.via the paraumbilical veins to the falciform ligament.It occurs owing to portal pyaemia and consequent spread of infection It may erroneously be diagnosed as an abdominal wall abscess. The diagnosis is confirmed by ultrasonography and/or CT or MRI.Complications include peritonitis, portal thrombosis, sepsis and mass effect, causing extrinsic intestinal obstruction.Scrupulous umbilical cord hygiene is essential in neonates to prevent omphalitis and secondary abscess formation.Management of the abscess is three-pronged: broad-spectrum intravenous antibiotics to cover the gastrointestinal flora, elimination of the inciting focus of infection  and drainage of pus.Written informed consent was obtained from the patient to publish this case. All patient images have been anonymized and the patient is not identifiable from the images/medical information included in the article.


Uterine leiomyoma, which is very common gynecological tumor in the reproductive years, is extremely rare in adolescence. We herein report a case of a uterine leiomyoma treated with laparoscopic surgery in an adolescent.A 13-year-old girl with no gravida and her first menses at 11 years of age reported feeling bloated. She had a regular menstrual cycle but felt increased abdominal distension.Transabdominal ultrasound and magnetic resonance imaging revealed uterine leiomyoma with a diameter of 10 cm.Laparoscopic myomectomy was performed.The total weight of the leiomyoma removed was 660 g with pathological diagnosis of uterine leiomyoma. The postoperative course was uneventful. The patient was free of disease at the follow-up consultation 18 months after the treatment.Laparoscopic approach is a very useful and minimally invasive surgery for symptomatic leiomyoma in adolescents. We herein report a uterine leiomyoma in a 13-year-old adolescent treated by laparoscopic myomectomy.Uterine leiomyomas are benign gynecological tumors originating from smooth muscle cells of the uterine wall. They are very common among woman of reproductive age, being found in an estimated 20% to 30% of women under 50 year of age.2This study was approved by the Osaka Medical College Clinical Research Review Board, and the patient with her parents gave written informed consent for publication.2.13-year-old girl with no gravida and her first menses at 11 years of age had a regular menstrual cycle with 4 to 5 days of bleeding each month. She had no remarkable medical history or family history. She had felt gradual abdominal distention over the past several months. Her body mass index was 18 kg/mTransabdominal ultrasonography revealed a solid mass with a diameter of 10 cm located behind the uterus Fig. . LaparosSurgery was performed with peumoperitoneum under general anesthesia. She underwent multiport (4 ports) laparoscopic myomectomy with parallel procedures. The uterus itself was the size of a newborn's head with normal tubes and ovaries Fig. A. There The postoperative course was uneventful. Pathologically, the tumor was diagnosed as a uterine leiomyoma. Eighteen months have passed since the treatment, and no recurrence has been noted.3In the current case, we performed laparoscopic myomectomy for a 13-year-old girl with uterine leiomyoma. To our knowledge, this is the youngest case in which laparoscopic myomectomy was performed. Clinically, a number of symptoms of leiomyomas in adolescents have been consistently reported in the literature. Moroni et al described the outcomes of 19 case reports of fibroids in adolescents from 1969 to 2014, with 87.5% (17/19) of patients presenting with symptoms, including abnormal uterine bleeding (10/18), abdominal pain (6/18), and perception of an abdominal mass or increased abdominal volume (8/18). Among the identified cases, the average age was 15.35 years, and the mean diameter was 12.28 cm. The most common treatment was abdominal myomectomy (13/19), with hysteroscopic myomectomy (2/19) and abdominal hysterectomy (1/18) also reported. There has only been one case report of leiomyoma in an adolescent successfully treated by laparoscopic myomectomy.Uterine leiomyomas are a very common gynecological tumor in woman of reproductive age; however, they are extremely rare in adolescents (1.0%).,8 However, laparoscopic myomectomy may be difficult in adolescents for several reasons. In symptomatic adolescent cases, most leiomyomas are already large at the time of the diagnosis. In addition, the intraoperative space is restricted in adolescents due to their relatively small body size. The risk of complications, including blood loss and organs injury, may therefore be increased.Laparoscopic salpingo-oophorectomy for benign adnexal tumor, such as serous cystadenoma and dermoid cyst, in adolescents is not rare. Recent studies have shown no significant difference in surgery time, duration of hospitalization, blood loss, or postoperative complications between adolescents and adults. However, the incidence of postoperative pelvic adhesions is higher in abdominal myomectomy than in laparoscopic myomectomy. Therefore, laparoscopic myomectomy should be selected as the primary surgery of leiomyoma in order to create a safe environment for secondary surgery and reduce the risk of postoperative complications.Parker et al found that up to 11% of women have a history of single fibroid removal and 26% have a history of multiple fibroid removal require secondary surgery for recurrence of leiomyoma. Laparoscopic and abdominal methods of myomectomy have similar recurrence rates. However, they have not been prescribed to this end in adolescents. The side effects of GnRH analogues in adolescents (as well as adults) must be considered, such as hot flashes, osteoporosis, dyslipidemia, hypertension, and the suppression of secondary sexual development. Bone loss leading to osteoporosis after long-term use is the most serious complication and may limit the therapy. A rule of thumb for women with endometriosis is that approximately 6% of bone is lost over 12 months of therapy, and 3% is regained following the cessation of therapy. Therefore, adolescents with early puberty or gender dysphoria are generally treated with GnRH analogues; however, whether this treatment should be started at an early Tanner stage or when puberty is almost complete and the appropriate duration of treatment remain unclear at present; such decisions may depend on the individual child and how well he or she is growing.,14 GnRH analogues may be useful for reducing the size of the mass and making laparoscopic surgery easier and safer in adolescents as well as in adults. However, we advise against the use of these agents for leiomyoma in adolescents due to a lack of evidence.Gonadotropin-releasing hormone (GnRH) analogues cause the profound inhabitation of sex hormones, including estrogen and androgen, and are used for a variety of indications. GnRH analogues are particularly commonly prescribed for leiomyoma in adult woman to reduce the size of the mass and make surgery easier and safer.It is difficult to define the impact of myomas in adolescent on future fertility. Five to 10% of infertile women have uterine myomas, however, it is unknown whether the myoma cause infertility directly. Many studies showed that myomectomy increased pregnancy rate and improve assisted reproductive technology success if the myomas are in or abutting the endometrial cavity or if they are large.In conclusion, laparoscopic myomectomy can be performed depending on the size, number, and location of the mass and the skill of the surgeons involved. This approach is a very useful and minimally invasive surgery for symptomatic leiomyoma in adolescents that reduces the postoperative pain, risk of pelvic adhesions, and hospitalization duration and affords a good cosmetic appearance.The authors thank Junko Hayashi and Kumiko Satoh for their valuable secretarial assistance.Writing – original draft: Natsuko Morita, Tomohito Tanaka.Writing – review & editing: Tomohito Tanaka, Sosuke Hashida, Satoshi Tsunetoh, Kohei Taniguchi, Kazumasa Komura, Masahide Ohmichi.


The transformation of nonosseous soft tissue into bone is known as osseous metaplasia (OM). This condition most commonly affects the musculoskeletal and central nervous systems and it is a well-known phenomenon in different soft tissue organs. Rarely, OM can affect the uterus, which can extend into the cervix. OM affecting the cervix alone is a more rare condition that has multiple different clinical presentations. The presentation can be similar to that of a tumor in extremely rare cases. A 23-year-old nulligravida was complaining of irregular vaginal bleeding for one-month duration. Speculum examination revealed a foul-smelling bloody purulent discharge, tender cervix, and a brownish growth located at the posterior cervical lip. A punch biopsy of the growth was performed. Histological examination of the tissue revealed multiple bone fragments with necrosis and an inflammatory exudate. Because of the unusual findings, a repeat biopsy was performed. The biopsy yielded the same findings, which confirmed the diagnosis of osseous metaplasia of the cervix. Although osseous metaplasia is a known phenomenon in different soft tissues, it is extremely rare in the uterine cervix and can mimic malignancy. Therefore, clinicians should be aware of it. Osseous metaplasia (OM) is the benign transformation of nonosseous soft tissue into bone. The musculoskeletal and central nervous systems are the most commonly affected tissues by this condition . It is aRarely, OM can mimic a tumor formation. In this paper, our aim is to report a case of cervical OM mimicking a cervical tumor. In addition, we include a literature review of the previously reported cases.A 23-year-old presented with irregular vaginal bleeding for one-month duration. The patient was also complaining of a foul-smelling discharge, abdominal pain and unusual sensation of a mass in her vagina. The patient was never pregnant before. She is medically free. The patient reported using some herbal treatment topically on her genitalia. On examination of the external genitalia, the vulva and perineum were normal. Bimanual exam revealed that the uterus was anteverted and normal in size. The adnexa and pouch of Douglas were normal as well. Speculum examination, however, revealed a foul-smelling bloody purulent discharge, and a brownish growth located at the posterior cervical lip. In addition, the cervix was tender on palpation. Given that a vaginal bleeding can be the first symptom of a cervical malignancy together with the finding of a mass on examination the case was suspicious for a cervical malignancy.A transvaginal ultrasound was done for the patient. The ultrasound showed a normal sized anteverted uterus with normal echogenicity. The endometrium was well defined but appeared thin. The ovaries were normal in size and showed multiple small follicles. The adnexa were normal bilaterally. The pouch of Douglas was free, all consistent with the examination findings. The cervix, however, was hyperechoic Additionally, benign squamous epithelium was seen Metaplasia is the reversible substitution of one type of fully differentiated cell for another within a given tissue . The mosThere are two forms of OM, a congenital and an acquired form. In both situations, the ossification process is initiated by a local osteogenic factor, which stimulates osteoblasts to differentiate and synthesize ground substance and collagen . The acqOM of the cervix can be asymptomatic or clinically present with leukorrhea, pelvic pain, vaginal bleeding, menstrual irregularities, infertility, or dyspareunia, The exact mechanism of OM of the cervix is not known although several have been proposed. It has been suggested that inflammation and trauma leading to chronic tissue injury stimulate undifferentiated stromal mesenchymal cells to undergo OM . In caseFinally, some women develop uterine OM after abortions occurring before 13 weeks of gestation, when fetal bone starts developing , 21. NonThis condition is distinct from metastatic and dystrophic calcification. Metastatic calcification occurs when the calcium-phosphorous levels are elevated, and the calcifications involve normal tissues. Dystrophic calcification, however, occurs in the presence of normal metabolism in damaged or devitalized tissues . CalcifiOM of the cervix is not a well-recognized condition due to its rare occurrence. This condition is benign, and treatment is available with resection. Therefore, it is important to recognize its occurrence to avoid misdiagnosis as a malignant tumor, such as a malignant mesodermal tumor.