Venous thrombosis is a medical condition that occurs when a blood clot forms in a vein. These clots usually develop in the lower leg, thigh, or pelvis but can also occur in the arm. It is essential to know about Venous thrombosis because it can happen to anybody and cause severe illness and disability. Fortunately, if the diagnosis is early, the outcomes will be excellent. However, idiopathic or spontaneous internal jugular vein thrombosis is a rare but potentially fatal condition. Method: here, we presented a rare case of Internal jugular vein thrombosis (IJVT) and reviewed the literature on cases of IJVT to describe clinical features, associated risk factors, possible complications, ways of investigations, and outcomes.Among 57 cases (56 in the literature plus our case), 25 patients out of 57 had a chief complaint of neck swelling, and only five complained of neck pain; on the other hand, four patients were asymptomatic. Thirty-five patients had a risk factor of developing thrombosis, 19 patients had a malignancy, and 22 did not have an obvious risk factor. To diagnose IJVT, ultrasound alone was used in 11 patients, Ct alone was used in 13 patients, and a combination of CT and ultrasound was used in 21 patients. Conclusion: IJVT thrombosis is a rare condition, but its diagnosis requires reasonable radiological and laboratory investigations; early treatment is warranted to avoid fetal complications. •Thrombosis is the term for the growth of a blood clot within a blood vessel, which may happen in either veins or arteries.•Following a proper diagnosis, this condition is regarded as an urgent disease that has to be treated right once.•The disorder known as idiopathic internal jugular vein thrombosis is very uncommon.•Radiological and laboratory tests should rule out any other causes of type II jugular vein thrombosis. Venous thrombosis is an intravascular condition resulting from the alteration of blood constituents (hypercoagulable states), alterations in blood flow, and vascular endothelial injuries. Internal jugular vein thrombosis (IJVT) is an unusual case of vascular disease of the upper limb veins that could result in multiple complications if left untreated. IJVT can be subdivided into primary and secondary. Primary IJVT is when thrombosis happens to someone without known risk factors. Most patients present with painful erythematous neck swelling and headache. The duplex/Doppler ultrasound is the best initial test to define the extent of the thrombosis ,2. Ultra22.1We reviewed the English-language literature and checked the relevant references from 1970 to 2020 to identify all known cases of Thrombosis of the Right Internal Jugular Vein using multiple sources; MEDLINE, PubMed, search engine Google and EMBASE using the terms jugular vein thrombosis, Internal jugular vein thrombosis, idiopathic Internal jugular thrombosis. Manuscripts published in English and Spanish were reviewed and relevant references were checked, and authors were contacted where possible. In addition, we also present a new case report.2.2All cases with a diagnosis of internal jugular vein thrombosis , were included in this report . We excl2.3For all reports of Internal jugular vein thrombosis, the information extracted included, if available: patient's age and sex, clinical and investigation tools used to confirm the diagnosis, another site of thrombosis, associated medical conditions, complications as well as the outcome.2.4Fifty-seven cases that meet the criteria were included in the review. Diagnostic modalities were different among the cases: Ultrasound was used in 11 cases, computed tomography (CT) scan was used in 13 cases, but the combination of CT and ultrasound was used in 21 cases, and Magnetic resonance imaging (MRI) was used in 2 cases, the combination of MRI and US was used in two cases, the combination of CT and MRI was used in one case, the combination of US and MRI was used in two cases. Venography and US were used in one case, and MRI with angiography was used in one case.Side of IJV thrombosis was distributed equally, with 23 cases involving left IJV and 23 cases right IJV, but 7 cases involved both sides. The involvement of other veins was reported in 13 cases. In 6 cases subclavian vein was involved; in two cases, subclavian and brachiocephalic were involved. One case involved subclavian and axillary veins, and one case involved subclavian, axillary, and humeral veins. One case involved the external jugular vein, one case external and brachiocephalic, one case involved sigmoid sinus, and one involved transverse and sigmoid sinus.The clinical manifestation of IJV thrombosis showed a range of variety in most cases, 25 presented with neck swelling, five patients with neck pain, and 4 cases were asymptomatic. The other cases present with supraclavicular fossa swelling, facial and upper limb swelling, fever and dyspnea, dizziness, confusion and fall, chest pain, and parotid gland swelling.In most of the cases, 48 were treated without complications. However, two ended up with death; two patients had a pulmonary embolism, one had pulmonary hypertension, and one had peripheral facial nerve palsy.3A 27-year-old Kenyan female with no history of previous illness presented to the emergency department with a 7-day history of neck pain and swelling. Her medical and family histories are unremarkable. She denied taking oral contraceptives or IV drugs. Also, she banned fever, loss of weight, previous abortions, and a history of deep vein thrombosis. On examination, she looked well with stable vital signs. A neck examination showed right-sided neck swelling, which was tender upon palpation. Examination of the ipsilateral and contralateral arms, as well as the other systems, was routine. Urgent neck ultrasound doppler showed right internal jugular vein thrombosis. Her CBC, blood chemistry, liver function test, vitamin B12 level, and coagulation profile were within normal limits. Chest radiography did not reveal any cervical rib, and a contrast-enhanced computed tomography (CT) pulmonary angiography was unremarkable. Thrombophilia workups including ANA, antiphospholipid Ig G/Ig M, protein C, protein S, antithrombin III activity, homocysteine, and fibrinogen level were requested, and the patient was admitted to the medical ward, and subcutaneous enoxaparin was initiated.In the ward, enoxaparin was stopped, and rivaroxaban was started. Thrombophilia workups return negative, ruling out secondary causes of thrombosis and supporting the diagnosis of idiopathic thrombosis of the right internal jugular vein. CT abdomen and head were also unremarkable. The patient was discharged on the 5th day of admission in good condition to continue rivaroxaban for three months.3.1Internal jugular vein thrombosis refers to an intraluminal thrombus occurring anywhere from the intracranial internal jugular vein to the junction of the internal jugular and the subclavian vein to form the brachiocephalic vein. The internal jugular vein is an uncommon site of spontaneous venous thrombosis. It is an underdiagnosed condition that may occur as a complication of head and neck infections, surgery, central venous access, local malignancy, polycythemia, hyperhomocysteinemia, neck massage, and intravenous (IV) drug abuse. It is also reported to occur spontaneously as a rare case . Interna3.2Idiopathic internal jugular vein thrombosis is a rare condition. All other causes should be excluded radiologically and by laboratory tests related to type II jugular vein thrombosis. Idiopathic internal jugular vein thrombosis has potentially fatal complications. Therefore, a physician should provide primary care when they suspect the presence of jugular vein thrombosis or when there is neck swelling with tenderness on palpation.N/A.N/a.Written informed consent was obtained from the patient for publication of these two case reports and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.MA: Manuscript writing, Data analysis, and approval of the final manuscriptBS: manuscript writing and editing, literature review, review and approval of the final manuscriptEB: manuscript writing, approval of the final manuscriptMN: manuscript writingFK: Case selection, case identification and conceptualization, literature review, manuscript writing, obtaining informed written consent, prescribing medicine, clinical follow up, and mentorNot applicable.Fahmi Khan.All authors declare no conflict of interest.


To the best of our knowledge, this case represents the first report of an extranodal double-hit follicular lymphoma (DH-FL) as an intestinal polypoid lesion.A 72-year-old woman presents with constipation. Colonoscopy reveals a sessile polypoid lesion of the colon bearing morphological, immunohistochemical and molecular hallmarks of DH-FL. Complete clinical staging and bone marrow biopsy showed no signs of disseminated disease. The patient, after two years of follow-up is still free of disease confirming the indolent behaviour of this limited lesion.A synoptic view at all the features of the patient and not merely at the molecular hallmarks of a disease are essential to establish the correct clinical approach. Primary double-hit follicular lymphoma (DH-FL) is rare. We describe the first case of follicular lymphoma with double-hit cytogenetics as a primary colonic localization. Moreover, our study discusses the clinical impact of MYC translocation in FL given the fact that data reported in literature are scarce. So far, DH-FL does not belong to high-grade B-cell lymphoma with MYC and BCL2 and/or BCL6 rearrangement category according to the revised 2017 WHO classification and no consensus exists on how to manage patients with this disease. Our case prompts the necessity of a re-classification of such entities.MYC Dual Color Break Apart Rearrangement Probe; Vysis LSI BCL2 Dual Color Break Apart FISH Probe; Abbott, Chicago, Illinois) detected both IGH/BCL2 . The diagnosis of an extra-nodal follicular lymphoma grade 3A with BCL2 and MYC rearrangements was made (DH-FL). The patient underwent a complete clinical staging with FDG-PET/CT and bone marrow biopsy. No signs of disseminated disease were found and primary colonic localization was confirmed. Although bearing molecular hallmarks of malignancy, a watch-and-wait approach was adopted according to the clinically favourable setting. The patient, after two years of follow-up shows no signs of relapse.A 72-year-old woman presented with complaints of constipation for three months. Physical examination, haematological and biochemical investigations were normal. Medical history was unremarkable. A colonoscopy was performed and revealed a sessile polypoid lesion of 5 mm of the sigmoid colon. The polyp had a smooth surface, without nodularity or ulceration Fig. . HistopaPrimary FLs are mostly detected in the lymph nodes; although relatively rare, they can appear in the gastrointestinal tract, being the most common site for extranodal lymphoma. Intestinal FLs account for only 1.0 to 3.6% of cases, and the duodenum is the most common site of occurrence, occurring rarely in the colorectal area [Rarely, follicular lymphoma may arise with MYC and BCL2 and/or BCL6 rearrangements, termed double-hit follicular lymphoma or triple-hit follicular lymphoma and should not be mistaken with the previously described double-hit lymphoma (DHL). While DHL classically presents with high-grade morphology resembling diffuse large B-cell lymphoma and/or Burkitt lymphoma with clinically aggressive course, DH-FL demonstrates the same translocation patterns but exhibits a follicular lymphoma histology, with no areas of high-grade cytologic transformation [Primary DH-FL is rare. So far, very few cases have been described, however presenting with nodal localization , 4. To tFor the diagnosis of such entities FISH is mandatory. However, such technique is not routinely used for localized lesions with a small cell lymphoma morphology. On the other hand, high grade morphology, high proliferation index and high MYC protein expression can prompt the pathologist for in-deep molecular analysis as in our case.This diagnosis offers a clinical challenge, given the difficulty of predicting whether the tumor will behave aggressively, as suggested by its cytogenetic profile or indolently, as indicated by its limited stage and preserved follicular growth pattern.MYC and BCL2 double-hit cytogenetic profile in FL favours the transformation to high-grade lymphoma and a poor outcome [MYC translocation in FL [Some authors have shown that  outcome . Convers outcome , 7. Howeon in FL . The patThis case illustrates that a synoptic view at all the features of the disorder and not at isolated markers are essential to establish a tailored clinical approach and to avoid overtreating.


Pancreatic rest, otherwise known as aberrant, ectopic or heterotopic pancreas, occurs when the pancreatic tissue does not have an anatomical or vascular connection to the normal body of the pancreas. This rare congenital anomaly was first described in 1727 by Hunt and Bonesteel, and it is now known to be found predominantly within the stomach or proximal small bowel. Most of the time, pancreatic rest is asymptomatic and is found incidentally. When symptomatic, the most common presentations tend to be: abdominal pain, nausea, gastrointestinal bleeding, obstruction and symptoms of pancreatitis. We report a case of a 21-year-old female with symptomatic pancreatic rest noted in two locations: antrum of the stomach and the proximal jejunum just distal to the ligament of Treitz. Pancreatic rest, or aberrant, ectopic or heterotopic pancreas, is a rare congenital anomaly . The incWe report a 21-year-old with a 10-year history of pain, nausea and diarrhea, initially treated with regimens aimed toward Crohn’s disease. Despite maximal medical management, symptoms persisted and further evaluation revealed a 2.6-cm jejunal mass. Due to the size, suspected wall involvement and location, biopsy was not feasible and the patient was referred for surgery. Resection was performed utilizing a robotic approach. Upon pathological examination of the specimen, typical pancreatic architecture was present confirming pancreatic rest. The patient is now symptom-free and off medications.A 21-year-old presented with ongoing pain, nausea and diarrhea. She had a 10-year history of abdominal pain for which she was initially diagnosed with Crohn’s disease at 9 years of age, and later, she felt to have ulcerative colitis as a teenager. Her routine management consisted of mesalamine with burst dose steroids for acute flares. She underwent repeat comprehensive evaluation to assess her persistent symptoms. The patient endured multiple imaging studies including: abdominal ultrasounds, hepatobiliary iminodiacetic acid scan, computed tomography (CT), CT enterography, magnetic resonance imaging (MRI) and MRI enterography. On MRI enterography, a 1.9-cm non-obstructing jejunal lesion was identified. Push enteroscopy was performed revealing submucosal masses in the antrum and jejunum with central umbilication. Biopsies were taken and the jejunal mass tattooed, but pathology was inconclusive.Endoscopic ultrasound (EUS) revealed a 1-cm submucosal antral mass. Due to size and wall involvement, pancreatic rest was suspected, though the jejunal lesion multifocal carcinoid tumor remained in the differential. An endoscopic mucosal resection of gastric lesion was performed, but pathology again was inconclusive. As the jejunal mass was noted to be 20–30 cm distal to the ligament of Treitz, and a miniprobe EUS was utilized to access this location. The small bowel lesion was hypoechoic, well demarcated, 2 cm and arose from the muscularis propria. Due to the size, location and depth of wall involvement, an endoscopic biopsy or resection was not feasible and formal surgical resection was recommended. Therefore, a robotic small bowel resection was performed. The previously marked jejunum was identified adjacent to a large mass with associated enlarged mesenteric lymph nodes . ApproxiOn post-operative outpatient evaluation, she reported complete resolution of all suspected Crohn’s disease symptoms. Specifically, in addition to pain resolution, she reported having normal bowel movements for the first time in 10 years.Pancreatic rest, aberrant, ectopic or heterotopic pancreas, is a rare congenital anomaly of pancreatic tissue, which occurs in the absence of normal anatomical or vascular connection to the pancreas . It was Heterotopic pancreas was categorized by Heinrich in 1909 and was modified by Gaspar-Fuentes in 1973 [Pancreatic rest generally is asymptomatic and most commonly found incidentally on endoscopy, laparotomy or autopsy , 2. ThouWhile CT still poses diagnostic challenges, there are findings that are helpful in making the diagnosis. These include flat ovoid shape, antrum, pylorus, or duodenum location, long:short diameter ratio > 1.4, endoluminal growth with ill-defined borders and enhancement of overlying mucosa . It has Overall, pancreatic rest is rare and difficult to diagnose. These challenges were highlighted herein. Diagnostic difficulties are related to nonspecific symptoms, inconclusive testing due to mass location, vague CT findings and difficult biopsy. While pancreatic rest is rare and can be elusive, it should be considered in the differential diagnoses of gastric and small bowel masses.K.C., M.L. and A.P. were responsible for the conception and design, acquisition of data, interpretation of data and manuscript writing and drafting. K.C., M.L., A.P., L.O’B. and L.R. revised the manuscript critically for important intellectual content and were in charge of the final approval of the version to be published.The authors of this report verify that the manuscript has been read and approved by all authors, requirements for authorship have been met and believe that the manuscript represents honest work. An abstract of this report has been previously presented at the Northwest Society of Colon and Rectal Surgeons (NWSCRS) on 24 July 2021. It otherwise has not been published or presented elsewhere.An abstract of this report has been previously presented at the Northwest Society of Colon and Rectal Surgeons on July 24, 2021. It otherwise has not been published or presented elsewhere.


MALAT1-GLI1 fusion has been considered to be the most relevant mutation. Herein, we present a case of an asymptomatic 58-year-old man who happened to find a submucosal gastric mass during a gastroscope and received endoscopic submucosal excavation (ESE). He turned out to have a gastroblastoma with a novel PTCH1::GLI2 fusion confirmed by Sanger sequencing. The patient was discharged two days after ESE without any complication and was recurrence-free during his one-year follow-up. According to the previous literature and our own experience, in cases with characteristic histopathology and immunohistochemistry patterns, a diagnosis of gastroblastoma should be considered even without a MALAT1-GLI1 fusion. Gastroblastoma pursues a favorable clinical outcome and endoscopic therapy could be an effective alternative treatment choice.Gastroblastoma is an extremely rare stomach tumor that primarily presents in adolescent and early adulthood, with a biphasic cell morphology of epithelioid and spindle cells. In light of its similarity to other childhood blastomas, it has been named gastroblastoma. Few patients showed a potential of metastasis and recurrence, however, most of the reported cases were alive, with no evidence of the disease after surgical treatment. Commonly,  MALAT1-GLI1 fusion was considered to be the most relevant mutation [PTCH1::GLI2 fusion in a 58-year-old male who successfully underwent endoscopic submucosal excavation (ESE) with no metastasis or recurrence post-procedure during his one-year follow-up, and summarize the relevant literature. We hope this will enlighten the diagnosis and treatment choices for gastroblastoma patients in the future.Gastroblastoma, an extremely rare stomach tumor, is characterized by biphasic morphology with nests, sheets, tubules and cords of bland epithelioid cells, as well as clusters of spindle cells . It usuamutation . HoweverThe patient was a 58-year-old male who happened to find a submucosal mass during an esophagogastroduodenoscopy (EGD) in an annual routine examination. He had no discomfort or history other than hypertension and cholecystlithiasis, and came to our hospital for further consultation and examination. We found the mass at the lesser curvature of the gastric body , with a soft tissue density and a clear boundary A. It shoPhysical examinations and laboratory tests of the patient showed no abnormalities. As his lesion had a clear boundary without infiltrative growth, the patient was admitted to our hospital for ESE using a GIF-Q260J with a LUCERA CV-290 electronic endoscope system . ESE was successfully carried out and four clips were used to close the wound after complete excavation on 26 July 2021 A. It hadPTCH1 and GLI2 (ex1:ex8), was revealed. The fusion was subsequently confirmed by a reverse transcriptase polymerase chain reaction (RT-PCR), followed by Sanger sequencing , it was named after gastroblastoma by Miettinen et al. . When coMALAT1-GLI1 fusion has been detected in five cases of gastroblastoma (5/15 cases). In addition, it can also be involved in plexiform fibromyxomas (3/16 cases) [GLI1-MALAT1 fusion was found in two gastroblastoma cases and for the other two cases with a MALAT1-GLI1 fusion, MALAT1 exon1 was fused to GLI1 intron 5 to exon 12 and GLI1 exons 5–12, respectively [MALAT1 (metastasis-associated lung adenocarcinoma transcript 1) is a long noncoding RNA, which is highly expressed in the nucleus of most cells and provides a strong promoter that drives GLI1 overexpression [GLI1 (glioma-associated oncogene homologue 1) encodes a zinc-finger transcription factor in the Kruppel family proteins, and plays a vital role in the Sonic Hedgehog (Shh) pathway, as well as in proliferation, growth, angioinvasion, invasion, migration and cancer stem cell self-renewal in a variety of malignant tumors [MALAT1-GLI1 fusion results in the overexpression of GLI1 and, in turn, the overexpression of GLI1 leads to the overexpression of several downstream targets, including PTCH1, VEGFA, SOX2 and CCND1. PTCH1 (transmembrane receptor Patched 1) is involved in cell proliferation and organization, and it is correlated with a copy number variation burden, tumor mutational burden (TMB) and loss of heterozygosity score [PTCH1 and GLI family proteins are both involved in the Hedgehog-GLI (hh-GLI) pathway. Canonical activation of hh-GLI signaling is initiated when the secreted ligand binds to PTCH1, which derepresses SMO (G protein-couple receptor Smoothened) and leads to the activation of the GLI transcription factors  [FHL2-GLI2 fusion in a sclerosing stromal tumor, which resulted in the transcriptomic activation of the Shh pathway [PAMR1::GLI1 fusion in which the Shh/GLI signaling pathway was involved [ACTB-GLI1 fusion was reported by Ambrosio et al. in a patient with a malignant epithelioid neoplasm of the ileum [ACTB activated GLI1 expression, where the Shh signaling pathway was involved in the tumorigenesis. As for our case, the gastroblastoma patient with a PTCH1::GLI2 fusion, we predicted that the Shh pathway was activated and an overexpression of GLI2 would be involved, similar to that of other GLI fusion tumors. The underlying mechanism and downstream targets require further study.A recurrent astoma 5/ cases. Iectively . MALAT1 yxomas 3/ cases [Gt tumors . MALAT1-ty score . PTCH1 a1, 2, 3) . Abnorma1, 2, 3) . Kim eta pathway . Punjabiinvolved . A transhe ileum . The ubiPTCH1::GLI2 fusion. In cases with a characteristic histopathology and IHC patterns, a diagnosis of gastroblastoma should be considered, even without a MALAT1-GLI1 fusion. For this particular case, whose lesion was relatively small with a restricted growth, a therapeutic endoscopy was carried out and no evidence of metastasis or recurrence was indicated during the one-year follow-up. Combining the previous studies, it seems that this particular tumor possesses a favorable clinical outcome and prognosis, despite its adopted suffix “blastoma”. Additionally, endoscopic therapy could be an effective alternative choice for some gastroblastoma patients in the future.We report a gastroblastoma occurring in an adult with a novel


SDHx variants are estimated to be associated with 0.3–1.8% of PitNETs. Only a few case reports have documented the association with MAX variants. Prolactinomas are the most common PitNETs occurring in patients with SDHx variants, followed by somatotrophinomas, clinically non-functioning tumours and corticotrophinomas. One pituitary carcinoma has been described. SDHC, SDHB and SDHA mutations are inherited in an autosomal dominant fashion and tumorigenesis seems to adhere to Knudson’s two-hit hypothesis. SDHD and SDHAF2 mutations most commonly have paternal inheritance. Immunohistochemistry for SDHB or MAX and loss of heterozygosity analysis can support the assessment of pathogenicity of the variants. Metabolomics is promising in the diagnosis of SDHx-related disease. Future research should aim to further clarify the role of SDHx and MAX variants or other genes in the molecular pathogenesis of PitNETs, including pseudohypoxic and kinase signalling pathways along with elucidating epigenetic mechanisms to predict tumour behaviour.Pituitary neuroendocrine tumours (PitNETs) associated with paragangliomas or phaeochromocytomas are rare.  SDHx) can associate with PPGL  can rarely occur in association with paraganglioma (PGL) or phaeochromocytoma. These tumours may develop in patients with or without identifiable germline variants. The combination of PitNET and phaeochromocytoma/PGL (PPGL) is also uncommon but well-described in the setting of multiple endocrine neoplasia (MEN) type 1 whilst the association in MEN2 is probably coincidental. Succinate dehydrogenase (SDH) gene variants  variants , miR-147b repressed SDHD activity, which is known to result in HIF accumulation , and one comprising RET and NF1-mutated  tumours ; the patient later developed a phaeochromocytoma  downstream of SDHD on chromosome 11  is located on chromosome 1p36.13 and codes for the catalytic SDHB subunit of the SDH complex . Evidence is inconclusive in the remainder of SDHB-related PitNETs analysed ). In 13 patients, no tissue analysis has been undertaken ( complex . SDHB mudertaken . Cases wSDHB c.298T>C (p.Ser100Pro) variant was reported to have a macroprolactinoma managed with dopamine agonist and surgery. LOH at the SDHB locus was confirmed in the tumour tissue, suggesting a pathogenic role of the SDHB variant  variant (SDHB-mutated PitNETs were identified and again LOH was confirmed in the pituitary carcinoma tissue (SDHB c.298T>C (p.Ser100Pro) variant was diagnosed with macroprolactinoma (SDHB variant might not have been causative and that a phenocopy was plausible. The most recent PitNET reported in association with an SDHB variant (c.587-591DelC frameshift) occurred in a 74-year-old female diagnosed with a macro somatotrophinoma on a background of metastatic PGL  is located on chromosome 11q23 and encodes the anchoring SDHD subunit  did not have any analysis undertaken in tumour tissue  variant  variant. She was diagnosed with macro somatotrophinoma. SDHA and SDHB expression was retained in tumour cells. No LOH was identified in the PitNET (The first SDHC-associated PitNETs are less frequently reported (n = 5) than those associated with SDHB and SDHD variants. To date, there has been no comprehensive report of PitNET secondary to a pathogenic SDHC variant . However, no GH expression was identified in the gangliocytoma by De Sousa and colleagues and no somatotrophinoma was present in the tissue submitted for pathological assessment. In addition, the expression of growth hormone-releasing hormone was not evaluated in tumour tissue. The microprolactinoma expressed SDHB by IHC, reinforcing the prediction that this is a benign variant  is mapped on chromosome 1q23.3 . SDHC-as variant . The firr tissue   is located on chromosome 5p15.33. To our knowledge, seven cases of PitNET in setting of SDHA variants have been reported  variant  was reported in a 53-year-old patient with an NF-PitNET and family history of NF-PitNET (father). The same patient had a history of nephroblastoma at the age of 1 year, 2 liposarcomas at 32 and 40 years, retroperitoneal PGL and renal oncocytoma both at the age of 50 years (SDHA variant was not causative. The variant c.969C>T was absent in his father’s NF-PitNET (Another SDHA c.1873C>T (p.His625Tyr) variant  was diagnosed with NF-PitNET at the age 30 years (SDHA c.91C>T (p.Arg31Ter) variant and macroprolactinoma was reported by Niemeijer and colleagues. Tumour tissue showed no SDHB and SDHA expression alongside LOH, suggesting the SDHA variant was contributory  variant and macroprolactinoma. Surgery was undertaken due to poor compliance with medical therapy. Analysis of the tissue revealed SDHB staining, no vacuoles and no LOH, suggesting a phenocopy (The most recent case of SDHAF2 variant in PitNETs has been reported (The gene encoding the SDH assembly factor 2 (OMIM*613019) is mapped on chromosome 11q12.2. To our knowledge, no evidence supportive of a causative reported .SDHx variant without tissue-based analysis to support a causative role for an SDHx variant. It is therefore possible that a considerable proportion of these cases could be phenocopies. A summary of SDHx-associated PitNET and PPGL including such cases is outlined in Many other reports have described PitNETs with PPGL associated with MAX codes for the MAX protein, a component of the MYC signalling pathway. The protein forms heterodimers with C-MYC via basic-helix-loop-helix zipper (bHLHZ) domain interactions. These heterodimers can then bind to target DNA sequences or E-BOX sequences to regulate transcription of genes involved in cell proliferation and cell growth  implicated in SDHx-related disease has roles in MNT/MAX/MYC-mediated cellular proliferation , VarSome pathogenic) has been documented   and of GATA3 by immunohistochemistry will improve the identification of cell lineages of SDHx-mutated cells  variant  missense variant resulting in maternally transmitted disease was also previously reported   at diagnosis .Written informed consent has been obtained from the patient for publication of this report. We thank our patient for his consent and for his support.P B L, F R and M K wrote the manuscript. P B L, F R, E H, P W, R T C and S J H provided diagnostic support and clinical care of the patient. R T C and M B performed the metabolomics study. All authors reviewed and edited the manuscript.


Retroperitoneal tumors are rare neoplasms that can reach great dimensions due to a slow growth pattern. Although these tumors rarely metastasize, they have a great risk of recurrence, and majority of times these lesions are a challenge for the surgeon. We report the case of a 63-year-old woman who presented with symptoms of large bowel obstruction and was diagnosed with a large heterogenous mass located in the retroperitoneal space. The fine needle biopsy revealed the histology of liposarcoma. The purpose of this article is to report our approach in management of this kind of tumor. Tumor size (27.1 × 29.1 × 36.1 cm) and involvement to the adjacent organs was a challenge for us in order to reach safe oncological margins. In these cases, the risk of recurrence is high; therefore, the patient should be screened at 6, 12 and 24 months post procedure. Chemotherapy or radiotherapy for this tumor is not yet defined, and if operable, surgery is the treatment of choice. Retroperitoneal sarcomas (RPSs) are rare tumors, accounting for 0.1–0.2% of all malignant tumors . These t12/l, hematocrit = 31%).A 63-year-old woman presented with a 6-month history of progressive weight loss, nausea, vomiting, diffuse abdominal pain and constipation. An ultrasonography exam revealed a huge retroperitoneal mass. The computed tomography with intravenous contrast revealed a large heterogenous mass having multiple linear septation and HU value of −90 of 27 × 29 × 36 cm  originatThe mass was approached by a midline incision. An extension of the mass was seen on the right side of the abdomen cavity. The diaphragm, pancreas and hepatoduodenal ligament were invaded by the tumor. We aimed to resect it with preservation of the abdominal organs . UnfortuHistologic sections revealed a well-differentiated liposarcoma , a sclerThe postoperative course was uneventful, but due to intraoperative complications—pneumothorax and major blood loss of 3600 ml, the patient’s discharge was delayed, namely, on the 13th postoperative day.Overall, local/abdominal recurrence is common  following resection of primary RPS. A high proportion of recurrences occur late (after 5 years), mandating ongoing follow-up . Tumor cSeveral studies suggest that tumor size does not have an impact over overall survival , 12, whiWell-differentiated liposarcoma is a locally aggressive adipocytic neoplasm with no potential for metastasis. We all know that lipomatous tumors are immunoreactive to S100; however, the adipocytic nature of the tumor is usually obvious and does not warrant an S100 stain. The morphologic distinction between benign lipoma and atypical lipomatous tumor relies on the identification of the hallmark diagnostic cells, which are the atypical hyperchromatic stromal cells and lipoblasts. The histologic types include adipocytic, sclerosing, spindle cell and inflammatory variants. When in doubt of this diagnosis, nuclear immunoreactivity of MDM2 and cyclin-dependent kinase 4 (CDK4) is confirmatory, which corresponds to amplification of these genes .Well-differentiated liposarcoma is a genetically distinct group of lesions. With the exception of the spindle cell variant, all well-differentiated liposarcoma subtypes share the same genetic aberration, represented by the presence of distinctive ring and/or giant marker chromosomes. Ring chromosomes contain amplified sequences derived from the 12q13–15 chromosome region, where several proto-oncogenes including MDM2, CDK4 and HMGA2 are located. Therefore, amplification of HMGA2, MDM2 and CDK4 (as well as overexpression of the corresponding proteins) can be detected by molecular or immunohistochemical techniques in well-differentiated liposarcoma, which is a helpful confirmatory diagnostic tool. S-100 immunopositivity is helpful to highlight the presence of multivacuolated lipoblasts. Sclerosing well-differentiated liposarcoma is best recognized by the presence of scattered bizarre, hyperchromatic stromal cells set in a fibrillary collagenous background .While tumor-related factors are paramount, the only intervenable predictive factor is the extent and quality of surgery . The resThe aim of the extended surgery approach is to maximize the possibility of achieving a complete resection through a standardized approach based on histologic behavior and site of origin. In liposarcoma, the extended approach often includes the resection of adjacent visceral organs  due to the difficulties in understanding tumor boundaries, multifocality and the importance of local control. Of note, the extended approach may preserve some of the adjacent organs thanks to its different patterns of growth, with a clear separation of tumor surface from the rest of the adjacent organs. Preserving noninvaded organs does not compromise the quality of the surgical resection .


Rosai-Dorfman disease (RDD) was described in 1969 by Rosai and Dorfman.JFG, a female patient aged 58 years, presented with dysphonia during the past four months; there were no other otorhinolaryngological or systemic symptoms. The patient smoked 90 packs or cigarettes per year, consumed alcoholic beverages in social settings, and had no history of tuberculosis or other comorbidities. The physical examination was within normal limits. Videolaryngostroboscopy revealed an ulcer on the right vocal fold and a decreased mucosal wave); laryngeal mobility was normal . NasofibRDD is a rare cause of enlarged lymph nodes; it is found mostly in black males.RDD is a rare atypical proliferation of histiocytes. Extranodal involvement alone, as in the present case, is even less common. The etiology is unclear. The diagnosis of RDD is made using histopathological and immunohistochemical methods. The disease progression varies, but is generally self-limited. New therapies are being investigated.


Inguinal hernias are the most commonly experienced disease in pediatric surgery. However, it is rare for the organs of the urinary system to prolapse as the contents of the hernia.We report a case of a 14-year-old boy with congenital paraperitoneal inguinal herniation of the ureter. Intraoperatively, we found an unfamiliar tubular loop structure arising from the deep inguinal ring in the left inguinal canal. The tubular structure, which may have been part of the ureter, was left in the inguinal canal to avoid damage. Postoperative drip infusion pyelography-computed tomography showed anatomical irregularity of the ureter in the inguinal canal. Follow-up in the 5th postoperative year showed no recurrence of hydrocele and complications associated with ureteral obstruction.Inguinal ureteral hernias are rarely reported in children. Paraperitoneal inguinal hernias are reported to be associated with vesicoureteral reflux and posterior urethral valve. Patients rarely present with symptoms like those observed in our case report. Whilst general surgical treatment is to return the ureter to the retroperitoneal space, we opted to leave the ureter in the inguinal canal to avoid unnecessary damage. However, this intraoperative management resulted in slight hematuria. The ureter should be placed back where it belongs, and postoperative monitoring using computed tomography may be important.children, highlighting the importance of accurate diagnosis and appropriate surgical intervention in the treatment of this disease.This case provides valuable insight into preoperative diagnostic difficulties and intra- and postoperative management of an inguinal ureteral hernia in  •Inguinal ureteral hernias are rarely reported in children.•Preoperative diagnosis of inguinal ureteral hernias is difficult.•An inguinal ureteral hernia may result in intraoperative technical difficulties.•The possibility of an inguinal ureteral hernia should be considered under surgery. However, ureter prolapse as the contents of the hernia is significantly rare, and few pediatric cases of congenital inguinal herniation of the ureter have been reported 2A 14-year-old boy, who presented with left scrotal bulging, was referred to our hospital. He had a past medical history of left inguinal hernia surgery when he was one. According to the initial operation record, two vasa deferentia were confirmed, and one of them was significantly thick at 4 mm. The general operation for inguinal hernia could not be performed, and high ligation of the hernia sac could be performed at long last after detaching a severe adhesion of “some” spermatic cords. There were thus far no complications after the inguinal hernia surgery. During the initial physical examination, the patient had an egg-sized, smooth, and painless translucent solid mass in the left scrotum. No tumor was palpable in the groin, and both the silk glove sign and pump tests were negative. Ultrasonography revealed a large hydrocele (10 cm) around the left testis.The procedure was performed under general anesthesia. A skin incision was performed along the previous surgical wound, and the inguinal canal was opened. The intestinal tract and a hernia sac were not shown in the inguinal canal. After detaching a severe adhesion of the spermatic cord, we detected an unfamiliar elastic soft tubular structure  which emanated from the internal inguinal ring separately from the spermatic cord and returned to the internal inguinal ring . We optePostoperative drip infusion pyelography-computed tomography (DIP-CT) was performed to confirm the urinary system abnormalities. DIP-CT showed no abnormalities in the shape and position of both kidneys and no hydronephrosis. Nevertheless, the left ureter reached the inguinal canal via the internal inguinal ring, looped around, changed direction, and then connected to the bladder . Based o3Since the first case of an inguinal ureteral hernia was reported by Leroux et al. in 1880, more than 140 cases of inguinal ureteral hernia have been thus far reported Whilst most cases of inguinal ureteral herniation have been reported in adults, a few cases have been reported in children, as shown in General surgical treatment for inguinal ureteral hernia involves returning the prolapsed ureter into the inguinal region of the retroperitoneal space 4This case provides valuable insight into preoperative diagnostic difficulties, as well as the intra- and postoperative management of an inguinal ureteral hernia in children, highlighting the importance of accurate diagnosis and appropriate surgical intervention associated with this disease.Written informed consent was obtained from the patient's parents for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Ethical approval was not required for this case report in our institution.No funding.Dr. Toshifumi Hosoda — corresponding author; collecting the data, writing the article, reviewing patient notes, writing articles, analyzing images, and approving the final submission.Dr. Kohei Hijikata — collecting the data, reviewing patient notes, writing articles, and approving the final submission.Dr. Shigeki Ishioka — collecting the data, writing the article, writing articles, and approving the final submission.N/A .Dr. Toshifumi Hosoda.The authors declare no financial, personal, or other conflicts of interest that could induce bias.


Scrotal pain and swelling are common presentation, the prompt evaluation and diagnosis is needed due to wide range of causes, uncommon causes of orchialgia should be kept in mind whenever there is no clear diagnosis. Spermatic vein thrombosis usually presents with orchialgia along with episodes of acute exacerbation of pain. The diagnosis is challenging and need high index of suspension and detailed radiological evaluation. We present a case of metachronous bilateral unprovoked spermatic vein thrombosis treated conservatively with anticoagulation with good response and resolution of symptoms. US:UltrasoundTVTTesticular venous thrombosisNOANew Oral Anticoagulants1Scrotal pain and swelling is common presentation, the prompt evaluation and diagnosis is needed due to wide range of causes that might need urgent intervention as torsion to more chronic cases like varicocele.1.1A40-year-old healthy gentleman presented to emergency department initially with an exacerbation of left scrotal pain has been there for 3 months, on and off with no associated fever or other features of orchitis, no trauma or strenuous activity. On physical examination left testis was firm, tender without swelling. Inflammatory markers within normal range. A Scrotal Doppler ultrasound (US) showed a non-compressible thrombus extending from the left spermatic to the inguinal veins  A and B anti-cardiolipin, and Factor V leiden mutation. The only risk factor for deep vein thrombosis was smoking which is a modifiable risk factor and patient quit smoking.Patient had work up to rule out coagulopathy patient had normal Coagulation profile, Negative lupus anticoagulant, 2Differential diagnosis of chronic testicular pain includes Varicocele, Testicular mass, Spermatocele, Hydrocele, post-procedural pain, and hernia.The origin of testicular venous thrombosis remains unclear. Although a lot of physicians and researchers relate it to pathological changes like varicocele, obstruction of the venous drainage , coagulopathies, or other systemic diseases,,The nature history of TVT varies as there it might resole spontaneously or to be complicated with pulmonary embolism.We present a case of metachronous bilateral unprovoked spermatic vein thrombosis treated conservatively with NOAs with good response and resolution of symptoms.3Spermatic vein thrombosis is a rare diagnosis needs his clinical suspicion, doppler ultrasound is the preferred diagnostic modality to rule out other causes of testicular pain and confirm the diagnosis. The main stay of treatment is conservative with anticoagulation and analgesia.The authors declare that they have no financial or non-financial conflicts of interest related to the subject matter or materials discussed in the manuscript.


IntroductionGlomus tumors are benign lesions with hamartomatous proliferation in the neuromyoarterial glomus bodies. Glomus tumors are small, reddish, painful blue nodules usually underneath the fingernail.ObjectivesThis study is mainly focusing on magnetic resonance imaging (MRI) findings of glomus tumors on T1, T2, short inversion time inversion recovery (STIR), and post-gadolinium images. Further study of clinical and histopathological findings to support the radiological diagnosis.Material & methodsThe retrospective study included an elaborate study of MRI imaging findings of 24 cases of glomus tumors of the hand and leg at a tertiary care center in Udaipur. Patients with imaging findings confirmed on histopathology were included in the study. MRI study was conducted using a 3-T MR unit and a high-spatial-resolution module.ResultsA total of 24 cases of glomus tumors were diagnosed during the six years between January 2015 and November 2020. Out of 24 patients, 14 were female and 10 were male. The most common site of involvement was a hand, followed by a foot. All 24 cases showed isointense to hyperintense lesions on T2-weighted images with a conspicuous hyperintense lesion on STIR images. Further histopathological examination confirmed the diagnosis showing nests of monomorphic tumor nuclei arranged in a perivascular pattern.ConclusionGlomus tumors can present with variable pain. A high index of suspicion is needed for diagnosis. Contrast MRI has a significant role in the diagnosis of glomus tumors. The clinical and histopathological picture further confirms the diagnosis. MRI imaging is further supportive to differentiate postoperative fibrosis from residual or recurrent tumors. Glomus bodies are present in the reticular layer of the dermis of the whole body but are numerously seen in digits, feet soles, and hand palms. These bodies have arteriovenous anastomoses, which are very helpful in thermoregulation by the regulation of skin blood flow . Glomus These tumors are also known as glomangiomas and represent approximately 2% of all primary soft tissue tumors and 1% to 4.5% of hand neoplasms ,8. GlomuGlomus tumors are small and reddish-blue nodules measuring 3 to 10 mm in diameter . The subA retrospective study of 24 cases of glomus tumors showing typical findings on MRI imaging, was carried out for the study period between January 2015 to November 2020 at a tertiary care center in Udaipur, Rajasthan. Medical records were retrieved and reviewed for demographics, symptoms, duration, physical examination, and recurrence.Inclusion criteriaThe following patients were included: 1. Patients with typical MRI features of glomus tumors of the hand and foot; 2. Patients admitted to our hospital and operated on for surgical excision; 3. Diagnosis of glomus tumor confirmed on histopathological examination.Exclusion criteriaPatients without histopathological confirmation of glomus tumors or glomus tumors other than the hand and foot were excluded from the study.MRI protocolAll MR imaging examinations were performed with a 3-T MR unit . A high-spatial-resolution module for skin imaging was used in all patients. A surface gradient coil was used in place of the anteroposterior gradient coil of the imaging system to increase the gradient strengths. A surface radio-frequency coil with a 1.5-cm radius was placed at the center of the gradient coil. All MR images were retrospectively reviewed in consensus by two radiologists with more than four years of experience who were blinded to the surgical findings.The patterns of glomus tumors were also noted. The location of each tumor in the sagittal plane and transverse plane was noted. The margins of the tumor were classified and the largest diameter of the tumor was measured in millimeters in the transverse and sagittal planes. The signal intensity of each tumor on T1- and T2-weighted spin-echo MR images compared with the signal intensity of the normal nail bed was characterized as low, equivalent (isointense), or high and as either homogeneous or heterogeneous. The appearance of the tumor after intravenous injection of gadolinium was characterized as no enhancement, slight enhancement, and intense enhancement compared with the appearance of the nail bed. On MR angiograms, an early nodular enhancement in the arterial phase was considered to be diagnostic of a glomus tumor .For excising subungual lesions trans-ungual approach, lateral or dorsal approaches were used and those with pulp lesions were excised with a direct incision over the affected site. All specimens were subjected to a histopathological study. Histopathological examination was carried out on formalin-fixed paraffin-embedded sections. Sutures were removed after 14 days.A total of 379 MRIs for hand and foot were done in the last five years. Sixty-seven MRIs were done for pain in the fingers and toes, out of which 24 patients were found to have features of glomus tumors. These 24 patients were referred from the orthopedics and dermatology department. Out of 24, 14 were female and 10 were male.Age ranges from 19 to 68 years with a mean age was 35.8 ± 8.5 years. The most common anatomical site of the tumor was the hand (19 cases) followed by the foot (five cases). The range of duration between clinical symptoms and diagnosis was four to 62 months and the mean duration was 16.9 ± 13.2 months.The left hand was affected in 11 cases while the right hand was affected in eight cases. The thumb, index, and middle finger were involved in four cases, eight cases, and six cases respectively, while one case involved the little finger. The right great toe was affected in three cases while two cases occurred in the left great toe.Fourteen patients complained of digit pain, and it was localized to the terminal phalanx of the finger. Three patients had pain localized to the great toe. Fourteen patients clinically presented with swelling at the site of pain. Cold hypersensitivity and night aggravation of pain were positive for eight patients. Love’s pin test (pinpoint tenderness) localized the lesion in all 24 cases (100%). Hildreth’s sign was positive in six out of the eight cases where it was done (75%). The pain was reproducible on palpation in all 24 hand and foot cases.The X-ray imaging and routine laboratory test were within normal limits. All 24 patients underwent contrast-enhanced magnetic resonance imaging (CEMRI). All of them revealed isointense to hyperintense lesions on T2. Twenty-three patients revealed intense contrast enhancement on T1 post-gadolinium fat saturation images, and one patient showed minimal contrast enhancement. One male and one female patient revealed well-defined, spherical, T2-weighted, iso-intense lesions in the subungual aspect of the index finger and little finger, respectively. Both lesions were more conspicuous on short inversion time inversion recovery (STIR) images and hypo-intense on T1-weighted fat-saturated image with avid homogenous post-contrast enhancement was seen on T1 post-gadolinium fat saturation images Figures -1h.One female patient present with a complaint of toe pain revealed a well-defined, spherical, T2-weighted iso-intense lesion in the subungual aspect of the great toe. The lesion was more conspicuous on STIR images and hypo-intense on a T1-weighted, fat-saturated image with minimal post-contrast enhancement seen on T1 post-gadolinium fat saturation images  sequence shows intense homogenous contrast enhancement can help differentiate glomus tumors from mucoid cysts and epithelial inclusion cysts because the latter two do not enhance. Most glomus tumors contain mainly three components and the components are vessels, glomus cells, and mucoid tissue, so the vascular form shows strong enhancement while solid and mucoid form shows less enhancement .MRI provides sufficient information about the extent and location of the tumor and is very helpful in the planning of surgery. Complete surgical removal of tumors is the treatment of choice for glomus tumors. The incidence of postoperative recurrence is 12-24% of cases. If recurrence of the symptom is within less than one year of surgery then incomplete resection is the likely cause and if recurrence is after more than one year then the development of the new lesion is the likely cause .Histopathology shows subcutaneous proliferation of well-circumscribed tumor nodules showing cuffs of glomus cells surrounding thin-walled blood vessels. Glomus cells show small, uniform round nuclei with amphophilic to eosinophilic cytoplasm and well-defined cell borders. Histopathological variants of Glomus tumor are oncocytic, epithelioid, myxoid, and hyalinized. A glomus tumor containing spindle cells and branching vasculature is known as ‘glomangiomyoma’ and ‘glomangiopericytoma’, respectively. A ‘symplastic glomus tumor’ is reserved for marked nuclear atypia without increased mitosis. A malignant glomus tumor is diagnosed in the presence of significant nuclear atypia and increased mitosis, including atypical ones .MRI is very helpful in the differentiation of recurrent/ residual tumors with postoperative fibrotic changes. Postoperative changes are usually ill-defined and appear hypo-intense on all sequences, whereas recurrence and residual lesions show similar findings as that of a non-treated lesion . Mucoid A glomus tumor is a rare tumor and its differential diagnosis should be kept in mind while assessing a patient with pain and swelling of a fingertip. Complete surgical excision is enough for the treatment of glomus tumors, with a very low incidence of recurrence.MRI imaging along with clinic-pathological findings have a very significant role in the precise and early diagnosis of glomus tumors. Apart from diagnosis, MRI is also very helpful in identifying the exact location and extent of the lesion and ruling out a postoperative residual tumor. Some rare solid and mucoid variants are difficult to confirm on MRI. A combined patho-radiological approach is demanded in such cases. Histopathological confirmation is essential to describe any variant of glomus tumors and to rule out features indicating their malignant nature.


Anaplastic astrocytoma (AA) is rarely observed in the brainstem and the clinical symptoms and imaging manifestations vary, which present a great challenge to accurate clinical diagnosis.18F-fluorodeoxyglucose positron emission tomography-computed tomography, and the clinical symptoms were not typical, leading to early misdiagnosis.A 56-year-old woman, with a month-long history of nausea and vomiting, was first diagnosed with acute cerebral infarction and demyelinating disease. The patient showed negative results on enhanced magnetic resonance and Finally, the patient was diagnosed with AA by pathological biopsy. The patient visited a different local hospital a month ago where they could not find a cause for her symptoms. The patient’s vomiting and headache became worse during the treatment and the patient was transferred to our hospital for further treatment. The patient did not report anorexia and her body weight did not change significantly.The patient had a history of uterine leiomyoma and did not have a recent history of cold, fever, diarrhea, or clinically significant family history. The patient did not have any history of brainstem tumor. Her family history was unremarkable.Physical examination on admission revealed that the patient had central facial paralysis, obvious impairment of bilateral eye movement function, and gross horizontal nystagmus.2O. The white blood cell count in the CSF was 3 × 106/L, with 1.8 × 106/L of lymphocytes, 1.1 × 106/L of monocytes, and 0.1 × 106/L of other white blood cells. The protein concentration was 0.35 g/L, and Pandy’s reaction was negative. The glucose and chloride ion levels were 4.66 and 120 mmol/L, respectively. The CSF was negative for aquaporin-4, myelin oligodendrocyte glycoprotein, and myelin basic protein antibodies.Cerebrospinal fluid (CSF) collected via lumbar puncture was colorless and transparent with a pressure of 140 mm H18F-FDG PET-CT revealed no obvious signs of tumor (Magnetic resonance imaging (MRI) of the head revealed acute pontine infarction (lack of imaging data). Urgent head computed tomography (CT) imaging after admission revealed a thickened brainstem, although the nature of the lesion was unknown. The results of the cranial MRI  indicateof tumor . Four weof tumor  revealedof tumor .Follow-up: At 6 months follow-up, the patient was still alive, bedridden, and unable to take care of herself.Ethical approval: The research related to human use has been complied with all the relevant national regulations, institutional policies and in accordance with the tenets of the Helsinki Declaration, and has been approved by the authors’ institutional review board or equivalent committee.Informed consent: Informed consent has been obtained from all individuals included in this study.3According to 2016 WHO classification guidelines for tumors of the central nervous system , AA is aThe clinical manifestations of patient with brainstem tumors vary with the lesion site and growth pattern. The focality and location of the lesion is distinguished from the clinical history, presentation, and associated imaging. In this case, the patient’s early symptoms were nausea and vomiting, rather than the usual triad of ataxia, long bundle syndrome, and multiple brain nerve damage . The iniIn this patient, T1WI showed low signal, T2WI FLAIR and diffusion-weighted imaging (DWI) revealed an enhanced signal, although enhanced scanning revealed no enhanced signal. The patient, a 56-year-old woman, who was also prone to cerebral infarction, was initially diagnosed with cerebral infarction based on the results of an MRI at a local hospital, which demonstrated the limitation of both plain and enhanced MRI in diagnosing brainstem AA. DWI can reflect the infiltration of tumor tissue into healthy brain tissue via the apparent diffusion coefficient (ADC) value but the value of ADC coexisting in brain tissue is superimposed . DWI is 18F-FDG PET-CT was the first tracer used for of brain tumors, which help in diagnosing tumors and has more advantages than MRI in judging the degree of malignancy of brainstem gliomas [18F-FDG in gliomas is higher than that in normal brain tissue and the degree of increase is related to the degree of tumor malignancy, which can reach a sensitivity of 80% with almost 100% specificity [18F-FDG to white matter is more than 1.5, or the ratio to gray matter is more than 0.6, the possibility of high-grade gliomas must be considered[18F-FDG uptake rate of the focus did not increase, which did not differ significantly from that of the surrounding normal brain tissue, nor did the blood flow increase abnormally in the cerebral blood perfusion map obtained using single-photon emission CT. This is because the metabolism of the brain cadre position is active, and the high variability and heterogeneity of a single tumor to 18F-FDG uptake further reduces the ability of PET-CT to detect these lesions, as 18F-FDG uptake and blood flow do not differ significantly between the tumor and surrounding brain tissue. gliomas . The uptcificity . When thl-phenylalanine (18F-FDOPA) display superior contrast to 18F-FDG because of low uptake of amino acids in normal brain tissue [Amino acid PET radiotracers including 3,4-dihydroxy-6-18F-fluoro-n tissue . A study418F-FDG PET-CT can be falsely negative even in high-grade glioma.In the present report, an unusual presentation of AA in the brainstem triggered the misdiagnosis of acute cerebral infarction and demyelinating disease. Although 18F-FDG PET-CT was negative, brain tumor was still considered with atypical imaging characteristics, and finally AA was diagnosed by pathological biopsy. The diagnosis and treatment of this patient reminds us that imaging diagnosis can only be used as an auxiliary diagnosis of the disease, and timely pathological biopsy is very necessary when the clinical manifestations are inconsistent with the image, because


Low-grade fibromyxoid sarcomas (LGFMSs) are typically adult-onset tumors that arise from the extremities. Here, we report an exceptional case of primary thoracic LGFMS in an 8-year-old girl that resulted in mediastinal syndrome. In reporting this case, we discuss the clinical challenges, role of molecular profiling and review reported cases of pediatric thoracic LGFMSs. Primary thoracic tumors are infrequent in children . Not onlA previously healthy nine-year-old girl presented with six months of cough, weight loss, and recent onset of dyspnea. Examination showed reduced air entry over the right lung, tracheal deviation to the left and facial puffiness, suggestive of superior vena cava syndrome. Subsequent X-ray and computed tomography (CT) confirmed a solid mass lesion occupying and expanding the entire right hemithorax, measuring 17 × 15 × 22 cm . There wGiven these imaging features, Type III PPB was initially considered, with differentials including inflammatory myofibroblastic tumors and other soft-tissue sarcomas. With such extent of disease, the patient was considered to be at an extremely high risk for anesthetic procedures, core biopsy was thus done under local anesthesia, mitigating the concerns associated with use of systemic agents. Frozen section indicated the presence of spindle cells. In view of the life-threatening picture, empirical treatment for PPB according to the IVADo regimen  was started and the patient was monitored closely in the intensive care unit .in situ hybridization study indicated the presence of FUS translocation, suggesting LGFMS  every 4 monthly, remaining in remission thus far 12 months from diagnosis . She wasFUS and CREB3L2 (95%) or CREB3L1 (5%), while MUC4 expression represents a sensitive and specific surrogate marker on IHC studies  as a result of neoplastic growth represents an oncologic emergency, which can manifest as life-threatening cardiovascular decompensation as a result of mass effects on the surrounding anatomical structures , 23. AccWe present the clinical course and molecular finding of a rare case of pediatric thoracic LGFMS. Molecular profiling facilitates the classification and management of uncommon soft-tissue tumors while pre-emptive multi-modal measures are integral to the successful management of patients with mediastinal syndrome.The datasets for this article are not publicly available due to concerns regarding participant/patient anonymity. Requests to access the datasets should be directed to the corresponding author.Ethical review and approval was not required for the study on human participants in accordance with the local legislation and institutional requirements. Written informed consent from the participants’ legal guardian/next of kin was not required to participate in this study in accordance with the national legislation and the institutional requirements. Written informed consent was obtained from the minor(s)’ legal guardian/next of kin for the publication of any potentially identifiable images or data included in this article.YC, DK, and AL: conceptualizing and initial drafting. YC, AK, LY, and AL: data collection. DK and AL: supervision. All authors contributed to the patient care, critical review, and final approval.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.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


Teaching Point: Infected urachal cysts are a rare cause of abdominal complaints and can be accurately diagnosed by abdominal ultrasound and computed tomography (CT). An abdominal ultrasound revealed a thick-walled heterogeneous cystic mass surrounded by fat stranding anteriorly in the midline on top of the bladder. The bladder dome was thickened, and the lesion was focally invading the left rectus abdominis muscle . The primary imaging modality for urachal cysts is ultrasound revealing a cystic mass in the midline anteriorly between the bladder dome and umbilicus. CT or MRI may be subsequently employed to help reach a definite diagnosis. Infection represents the most common complication of urachal anomalies and may present with nonspecific symptoms. Imaging findings that should prompt consideration of infection in the setting of an acute clinical presentation include the presence of a urachal remnant with complex echogenicity at ultrasound and heterogeneous attenuation of the content and contrast enhancement of the thick wall on CT. As in this case, it can be challenging to distinguish an infected urachal remnant from a malignant urachal neoplasm. Treatment consists of antibiotic therapy followed by complete excision to mitigate the 30% risk of infection recurrence and potential malignant degeneration [


After six courses of combined chemotherapy, pemetrexed was administered as a single agent. At the time of writing this report, she was undergoing over the 30th course of chemotherapy without any significant side effects. Diffuse malignant peritoneal mesothelioma is a rare, fatal, and progressive disease. Our patient achieved long-term survival of more than 5 years with maintenance therapy using single-agent pemetrexed.A 76-year-old woman presented with lower abdominal pain and nausea and was referred to the gastroenterology department in our institution. Previous contrast-enhanced computed tomography (CE-CT) for follow-up after breast cancer surgery had indicated a soft tissue mass below the right diaphragm, which was considered a benign change. CE-CT performed at the first visit to our department revealed further thickening of the soft tissue mass with extension to the liver surface. In addition, ascites and nodules were observed in the abdominal cavity. Histopathological examination of a biopsy specimen revealed peritoneal invasion of atypical epithelioid cells with trabecular and glandular patterns. The tumor cells were positive for AE1/AE2, calretinin, WT-1, D2-40, HEG1, EMA, BAP1, and MTAP and negative for carcinoembryonic antigen, MOC-31, Ber-Ep4, ER, PgR, TTF-1, claudin 4, and desmin. A diagnosis of epithelioid mesothelioma was made. The patient received chemotherapy with cisplatin (75 mg/m In the United States, malignant peritoneal mesothelioma (MPM) accounts for up to 15% of all cases of mesothelioma, and it is rapidly fatal. MPM is difficult to diagnose because of its nonspecific clinical symptoms and findings. The common symptoms are abdominal pain or a palpable abdominal mass. MPM often develops near the pelvis, rectum, urinary bladder, or uterus. Generally, it is treated with surgical resection or chemotherapy [A 76-year-old woman was undergoing regular follow-ups at the Department of Breast Surgery in our hospital for postoperative breast cancer. There was no recurrence of breast cancer; however, she presented with a complaint of lower abdominal pain and nausea and was referred to the gastroenterology department. Her medical history did not include any apparent episodes of asbestos exposure. Physical examination at the first visit showed normal vital signs, a flat and soft abdomen, and tenderness in the lower abdomen.2) and pemetrexed (500 mg/m2) was initiated. CT evaluation 12 months after the start of treatment showed no changes in the tumor, and the treatment was continued. After six courses of combined chemotherapy, pemetrexed was administered as a single agent because the total cisplatin dose exceeded 500 mg. At the time of writing this report, she was undergoing over the 30th course of chemotherapy without any significant side effects. CT performed 48 months after the start of chemotherapy showed stable disease with amelioration of the abdominal symptoms observed at the initial examination , cancer antigen (CA) 19-9, and CA 125 were within normal limits. Previous contrast-enhanced CT (CE-CT) for follow-up after breast cancer surgery had indicated a soft tissue mass below the right diaphragm, which was considered a benign change. CE-CT performed at the first visit to our department revealed further thickening of the soft tissue mass with extension to the liver surface. In addition, ascites and nodules were observed in the abdominal cavity . Fluorodmination .Peritoneal mesothelioma was first reported by Miller and Wynn in 1908. Among all mesotheliomas, peritoneal lesions are rare; most mesotheliomas occur in the pleura and peritoneum, with 80% involving the pleura and 20% involving the peritoneum .Measurement of biomarkers is recommended for the diagnosis of mesothelioma. In addition, the absence of BAP1 and MTAP loss is effective in distinguishing the epithelioid and sarcomatoid types; however, it is not 100% accurate, and peritoneal mesothelioma can also show the absence of MTAP loss. The treatment of mesothelioma includes chemotherapy and surgical resection. In addition, various treatment modalities are being explored, including radiation therapy and HIPEC . One stuγ-glutamate synthetase and low thymidylate synthase levels have been associated with the sensitivity of pemetrexed; however, we did not examine these biomarkers in the present case. Given that comprehensive gene panel tests for advanced solid tumors are covered by insurance in Japan, it is possible to test these biomarkers if there is sufficient tumor volume.Targeted therapy, such as gene therapy, is based on the method of administering a thymidine kinase gene conjugate to the Herpes simplex virus and then administering an antiviral agent to kill it. This option could not be explored in the present case because of insufficient tissue mass in the peritoneal biopsy. Moreover, previous studies have evaluated the predicted biomarkers for pemetrexed-treated mesothelioma. High folylpoly-2) and pemetrexed (500 mg/m2), and cisplatin administration was interrupted because irreversible side effects such as peripheral neuropathy and renal failure occurred when the total dose exceeded 500 mg. Single-agent pemetrexed was administered after six courses of combined chemotherapy. For elderly and unfit patients in particular, single-agent pemetrexed (500 mg/m2 intravenously every 3 weeks) has been recommended [In the present case, the patient was treated with chemotherapy comprising cisplatin 75 mg/m5 mg/m2 aommended . Of lateIn conclusion, diffuse MPM is a rare, fatal, and progressive disease. However, our patient achieved long-term survival of more than 5 years with maintenance therapy using single-agent pemetrexed. This may be because pemetrexed has no tolerable dose and is administered only once every three weeks with an infusion time of 30 minutes, ensuring good compliance and reducing patient burden. All these indicate that pemetrexed therapy may help in maintaining long-term tumor shrinkage without severe side effects.


Metastatic colorectal cancer has poor prognosis for many patients at time of diagnosis with <20% 5-year survival rate. Recent advancements in palliative chemotherapy have improved patient outcomes as median survival has increased almost 2-fold. We report a 44-year-old gentleman who initially underwent palliative chemoradiotherapy and subsequently a Hartmann’s procedure for ypT3N1M1 upper rectal adenocarcinoma with multiple liver metastases. Fortuitously, he made a remarkable recovery with complete radiological resolution of liver metastasis post-operatively. The patient has remained in remission for the past 10 years. Patients with metastatic colorectal cancer (CRC) often present with poor prognosis at time of diagnosis. Palliative chemotherapy, 5-fluorouracil (5-FU) with leucovorin and oxaliplatin (FOLFOX) in particular, remains the gold standard treatment of choice for unresectable metastatic CRC. Surgical interventions to remove the primary tumour in advanced disease are limited and currently only warranted in the event of obstruction or bleeding. This case would hopefully add value to the literature for clinicians to consider the role of surgical resection after palliative chemoradiotherapy in patients with advanced CRC and liver metastasis (CRCLM) in the future.A 44-year-old Mr TM presented in October 2011 with a 2-week history of rectal (PR) bleeding, pencil-like bowel motions and right upper quadrant pain. Colonoscopy revealed a non-obstructing poorly differentiated upper rectum adenocarcinoma, 10 cm from the anal verge. Completion staging computerised tomography (CT) scan demonstrated multiple liver metastases in Segments 2, 4A, 5–8, maximally 94 mm in diameter as shown in He was deemed suitable for palliative chemoradiotherapy and underwent 13 cycles of FOLFOX and 1 cycle of SIRTEX, eventually ceased because of severe peripheral neuropathy. He responded positively to treatment, with more reduction in the size of both his primary rectal cancer and liver metastasis on serial CT scans.Unfortunately, in August 2012, 6 months after the completion of palliative chemoradiotherapy, the patient began to develop worsening PR bleeding. An urgent colonoscopy demonstrated a slight increase in size of the rectal tumour from 20 to 30 mm, with evidence of ulceration, which was likely the cause of his symptoms.Following discussion within a multidisciplinary team, the patient underwent an uncomplicated palliative open Hartmann’s procedure in December 2012 for ypT3N1M1 upper rectal adenocarcinoma . Post-operatively, the patient opted for no further chemoradiotherapy and was also not deemed for surgical intervention regarding the liver metastases.Subsequent surveillance imaging including magnetic resonance imaging and CT scans demonstrated gradual reduction and eventual complete resolution of the liver metastases refer to –4. FurthCRC is the third most diagnosed cancer in Australia , with paMoreover, in incurable disease, the incorporation of palliative surgery with chemotherapy significantly improves quality of life and symptom management . SurgicaUnderstandably, there are limited guidelines for surveillance of resected Stage IV CRC as poor prognoses are expected in most patients. For patients with CRCLM, liver resection is the definitive treatment offering the best chance at long-term survival . HoweverAlthough cases of the radiological ‘disappearing metastasis’ phenomenon post-chemotherapy for CRCLM have previously been reported , 12, we None declared.None.


Wide local excision of the tumor was performed. Monomorphic spindle cells in a storiform pattern were observed by histological evaluation. The neighboring adipose tissue was invaded by the tumor cells in a honeycomb-shaped pattern. The histological features were suggestive of Dermatofibrosarcoma Protuberans. Due to the high likelihood of recurrence, it is crucial to monitor these patients for an extended period of time.Dermatofibrosarcoma protuberans is a rare, low-grade dermal soft-tissue tumor with a high propensity for recurrence but a low propensity for metastatic spread. It mostly affects the head, neck, proximal extremities, and trunk. We report a case of dermatofibrosarcoma protuberans over the right lumbar region. The patient presented with swelling in the right lumbar region measuring 3 cm × 3 cm. The local region ultrasonography (USG) revealed a well-defined hypoechoic lesion in the subcutaneous area. The patient was provisionally diagnosed with a peripheral nerve sheath tumor under evaluation. USG-guided fine needle aspiration cytology Darier and Ferrand first described dermatofibrosarcoma protuberans (DFSP) in 1924, while Hoffman first defined the term in 1925. It is an uncommon, soft tissue sarcoma (low to intermediate grade) that develops in the skin's dermal layer. Lesions may begin as a painless, skin-colored plaque with a potential dark red or blue staining and have a tendency to expand slowly. Later stages of DFSPs might enlarge and develop into protuberant or ulcerative lesions. They typically exhibit localized aggression and have the propensity to invade nearby structures, including the subcutaneous tissue, muscles, tendons, and even bone structures. However, metastatic disease is rarely documented .Patient information and clinical findingsA 32-year-old female came with a chief complaint of swelling over the right side of the back since last year. It was gradual in onset, and there was a progressive increase in the size of the swelling. The patient complained of pain over the swelling. She had a history of cyst excision done four years back from the same site.On local examination, a palpable lump of size 3 cm × 3 cm was present over the right side of the back Figure . The sweTimeline of the current episodeThe patient had swelling on the right side of the back for one year. Swelling gradually increased in size, involved the skin over the back, and caused pain and discomfort to the patient during the previous week. The patient had a similar complaint of cystic swelling over the same site four years back, which she had got excised. The patient exhibited generalized weakness. There was no history of discharge from the swelling. The patient does not have diabetes mellitus, asthma, tuberculosis (TB), hypertension, or any other known diseases.Diagnostic assessmentThe USG local region revealed a well-defined hypo-echoic lesion of size 3.8 cm x 2.1 cm x 3.5 cm in the subcutaneous area in the right lumbar region. It showed posterior acoustic enhancement and multiple septa within. Color Doppler imaging revealed a significant vascularity. The patient was provisionally diagnosed with peripheral nerve sheath tumor. USG-guided fine needle aspiration cytology (FNAC) was performed. Operative procedureWide local excision of the tumor was performed under general anesthesia, and the specimen Figure  was sentDiagnosisUSG-guided fine-needle aspiration (FNA) smears revealed moderate cellularity Figure , showingThe excised tumor was subjected to a histological examination. It was a single globular specimen with attached fibroadipose tissues and skin Figures , 7. The On microscopy, sections from different areas revealed a nodular mass composed of a proliferation of mostly uniform, medium-sized spindle cells with a storiform or cartwheel pattern of growth Figures , 9 placeFollow-up and outcomesPost-operation, the patient was relieved of acute symptoms of pain and swelling over the right side of her back. Her discomfort had subsided. Overall, the patient was doing quite well after the surgical intervention. DFSPs, a rare, slowly-growing malignant fibroblastic mesenchymal skin tumor, make up less than 0.1 percent of all malignant tumors and one percent of all soft tissue sarcomas . The claDFSPs have been discovered to be predominantly hypo-echoic or mixed hyper-echoic on ultrasound, with mainly well-defined boundaries or irregular, with projections resembling pseudopodia . DFSP's Diffuse invasion of the dermis and subcutis under a microscope identifies DFSP while typically sparing the epidermis and appendages of the skin. While entering fat lobules, it develops along preexisting fibrous septa, forming the characteristic honeycomb-shaped pattern. Dermatofibrosarcoma protuberans might sporadically manifest as an infiltrative subcutaneous tumor. Mitoses are uncommon, and atypia is modest. Tumor cells have immunohistochemical staining for CD34, vimentin, nestin, apolipoprotein D, and epithelial membrane antigen (EMA) .Broad local excision is the preferred course of treatment with negative margins of 3-5 cm from the tumor edge. To obtain negative resection margins in cases when there is a chance that the bone will be involved, the periosteum or even a small amount of the bone may need to be removed. Depending on the resection margins, the rate of recurrence will change. Recurrence rates were under 5% in series that employed five-cm resection margins .DFSP is a rare tumor that grows slowly, with a female preponderance. It usually occurs around the age of 40 years. The storiform pattern of spindle cells is observed histologically. Treatment consists of complete surgical removal. There are many recurrences. The majority of local recurrences manifest during the first three years following surgery. Recurrences later than five years have also been documented, though. Therefore, it's crucial to keep an eye on these individuals over the long term.


Penile fracture is a rare urological emergency that effect due to blunt trauma on erect penis. Mostly often occurs during sexual intercourse and is rare in blunt of trauma from impact at penile. That trauma it can caused by rupture and hematoma of tunica albuginea. In physical examination, we found swelling and hematoma of penile shaft, with abnormal angulation to the right and tenderness. Two hours after sexual intercourse, patient urinated and he felt fresh blood comes out with urine and it is painful. Penile fracture can be diagnostic with clinically and imaging radiology. Important information due clinically with précising radiologist finding can making the correct diagnosis and surgical procedure can be done immediately. Penile fracture is a rare urological emergency due to trauma to the erect penis, frequently occurring during sexual intercourse, caused by rupture and hematoma of tunica albuginea. The diagnosis is based on physical examination in which patients usually present erected penile with edema. Urethrography fluoroscopy is an excellent diagnostic tool before surgery as it shows the rupture in the cavernous body. We report a case of a 46-year-old man diagnosed with a penile fracture, focusing on the ruptured anterior urethra and right cavernous body.A 46-year-old male presented to our emergency unit with pain and edema at the penile. According to his wife, it happened after the sexual intercourse, in which the patient noticed the bloody urine, progressing to swell and darken with pain. The patient's general condition was within normal limits, with a blood pressure of 138/74 mmHg and a pulse of 85 bpm. Localized examination showed the darkened and swollen penile with tenderness . LaboratThe patient was admitted to the urology surgery department with a diagnosis of a ruptured anterior urethra and right cavernous body and planned for emergency surgical repair as soon as his general condition improved. Exploratory surgery was performed during the repair, with a circumferential subcoronal incision and degloving to the proximal penis, revealing a hematoma on the dorsolateral of the right cavernous body, which was subsequently evacuated. We discovered the ruptured site measuring roughly 1 cm and proceeded to repair it with a primary suture using 3.0 vicryl thread on the torn cavernous body . The bloAt the 2-month follow-up visit, there was no gross hematuria, swelling, or edema at the penile soft tissue. We requested a contrast urethrography, yielding urethral stricture at the cavernous part with no contrast leakage visualized .Fig. 6UrErection converts the safe, flaccid penis into a vulnerable organ. During an erection, the thick tunica albuginea becomes thin and fragile. Penile fracture is a relatively rare condition caused by blunt trauma to the erected penis, with the most frequently reported trauma mechanism being non-physiological bending of the erect penis during sexual intercourse or masturbation eggplant sign of the penis and tenderness at the ruptured site provides the clinical diagnosis of the penile fracture without requisite diagnostic tests in most situations; nonetheless, penile ultrasound, cavernosography, and MRI may be helpful in some cases Most patients with fractured penis present the symptoms of a cracking sound followed by sudden detumescence with painful penile swelling. The defect at the fractured site is often palpable and has been described as the "rolling sign," referring to a firm, mobile, tender mass where the penile skin can be rolled over the blood clot ,2. This We reported the case of a penile fracture with ruptured urethra at the cavernous part with retrograde urethrogram fluoroscopy contrast injected into the penile orifice, demonstrated. Penile fracture can be diagnosed with clinical examination and radiology imaging and confirmed upon surgery. Meticulous history taking and precise imaging findings offer a timely diagnosis and prompt surgical repair.Written informed consent was obtained from the patient for the publication of this case report. No patient identifiers are disclosed in current report.


A 46-year-old man has a diagnosis of schizoaffective disorder complained of intermittent abdominal pain for many years. Due to this, he had been reviewed by the GP and he was prescribed medication to help with his intermittent abdominal pain.Over the years the abdominal pain gradually worsened. He also has communication issues due to language barriers and was unsettled for most of his assessment.His past medical history includes a duodenal ulcer, infected swollen legs and recurrent urinary tract infections.He continued to have pyrexia despite being on regular paracetamol. Following his second episode of pyrexia, he was referred to the hospital for further investigation.This was found to be acute acalculus cholecystitis, with possible cholecystocolic fistula and pneumonia. He was managed conservatively with intravenous antibiotics and is awaiting cholecystectomy.Behavioural change in people with mental illness need not necessarily be linked to their mental state as it can very well be the atypical manifestation of physical illnesses- some of which could be fatal. Prompt recognition and referral to acute medical or surgical services is essential. Staff need training in bias, diagnostic overshadowing and atypical presentations in those with mental illness which will help reduce rates of avoidable morbidity and premature mortality.In any case physical illnesses may not present typically. Acalculous cholecystitis is a rare type of gall bladder inflammation and the cause in Mr X's case is not clear. At times of COVID-19, with the anxieties around exposure to hospitals and infections, it is important to be aware of this and ensure that people with worrying physical symptoms are promptly referred whether or not it is considered to be related to COVID-19.Due to the pandemic, we were cautious on the ward and community about COVID-19 and preventing catching and spreading the infection. During all this, patients change of behaviour shouldn't be alluded to deterioration of mental health and mental health professionals should also consider ruling out physical causes for the change of presentation.


A 61‐year‐old woman developed a pulsatile mass on the left upper limb and was diagnosed with arteriovenous malformation with pseudoaneurysm. A two‐stage operation including ligation and resection of the aberrant branches and subsequent resection of the mass with revascularization was performed. Histological analysis suggested arteriovenous malformation and pseudoaneurysm. Ligation and resection of the aberrant branches and resection of the mass with revascularization relieved clinical symptoms. Long‐term follow‐up is required for monitoring a progression of a new arteriovenous network and aneurysm.. The majority of AVMs are found in people between the ages of 20 and 40 years.2A 61‐year‐old woman with no history of trauma or hemodialysis was referred to our hospital for treatment after developing a pulsing mass on her left upper limb. The swelling was first noticed in early childhood, with a significant size disparity between her two upper limbs; however, it was left untreated. Physical examination revealed skin pigmentation, varicosities, a palpable thrill, and continuous bruit on the left upper limb, as well as a significant size discrepancy between the two upper limbs. A blood test and echocardiogram revealed no signs of congestive heart failure. Computed tomography revealed an 8‐mm dilated brachial artery with several abnormal branches, a 3 × 3‐cm pseudoaneurysm, and AVMs on the forearm Figure . AngiogrThere was a need to resect surgically the mass due to the risk of a rupture. However, a single operation of resecting the mass was considered to be associated with a risk of operative bleeding because a blood pressure of all veins was equivalent to that of an artery and a swelling of the left upper limb could lead to a surgical wound site‐related complication such as dehiscence and infection. Therefore, AVMs should have been treated to reduce the vein pressure and alleviate the swelling. As all AVMs cannot be completely removed and a large AVM cannot be treated with catheter intervention, a surgical treatment seemed to be an optimal option. Then, we decided to perform a two‐stage surgery including initial resection of the aberrant branches of the brachial artery and subsequent resection of the pseudoaneurysm.In the first surgery, three aberrant vessels branching from the middle of the brachial artery were ligated and resected under general anesthesia Figure . The sweThe second surgery was performed under general anesthesia 1 month after the initial surgery. The median nerve was encircled by a 3 × 3 cm mass that was a portion of the thin, dilated brachial artery Figure . The abeOne year after the operation, the skin pigmentation, palpable thrill, and significant size discrepancy between the two upper limbs disappeared, and the varicosities were alleviated.3We believe the AVM, in this case, was congenital because the patient exhibited symptoms in the left upper limb since childhood. The vascular system develops in four stages: blood islands, vasculogenesis, angiogenesis, and maturation.Therapeutic options for AVMs include endovascular, surgical, radiation therapy, or a combination of all of these. However, treatment for AVM is challenging because clinical conditions vary from one case to another, and, additionally, a large widespread AVM cannot be completely repaired.4The patient's symptoms were relieved after the aberrant arteries that formed the AVM and pseudoaneurysm were successfully resected and revascularized. Long‐term monitoring will be necessary since other aberrant arteries linking the vein still exist, and there are concerns about the progression of a new arteriovenous network and aneurysm.Akihisa Furuta conceptualized and designed the study, and drafted, critically revised and approved the article. Daisuke Futagami conceptualized and designed the study, interpreted the data, and approved the article. Hironobu Morimoto conceptualized and designed the study and critically revised the article. Junya Kitaura and Shogo Mukai critically revised and approved the article.None.Approval of the International Review Board was not required at our institution because this study was a case report.Written informed consent was obtained from the patient for the publication of this report under the journal's patient consent policy.Figure S1Click here for additional data file.


Fifty-four-year old male was admitted to the intensive care unit (ICU) due to impaired consciousness. Past medical history included alcohol dependence, liver cirrhosis, esophageal varices, 2 esophageal varices banding procedures in the past, pathological obesity. Computed tomography (CT) examination of the head performed in the referring hospital was normal. At admission the CT examination of the head was repeated and showed no abnormalities. Urgent esophagogastroduodenoscopy revealed presence of esophageal varices and scarification following previous banding procedures located in the middle and lower part of the esophagus. Gastrointestinal bleeding being the most likely cause of chronic liver decompensation was therefore excluded. Multimodal neurologic diagnostic assessment was negative. Finally magnetic resonance imaging (MRI) of the head was performed. Taking into account clinical picture and the MRI result, the differential diagnosis included chronic liver encephalopathy, exacerbated acquired hepatocerebral degeneration, and acute liver encephalopathy. Due to history of umbilical hernia CT of the abdomen and pelvis was performed and showed intussusception of the ileum, confirming hepatic encephalopathy. In this case report the MRI suggested the diagnosis of hepatic encephalopathy and prompted search for alternative causes of decompensation of chronic liver disease. Fifty-four-year old male was admitted to the intensive care unit (ICU) due to impaired consciousness  and acute respiratory failure. Recently the patient complained of malaise, drowsiness, problems with memory (was not able remember names of known objects), visual hallucinations (saw ‘flowers’). The patient in the last 3 days experienced several episodes of non-bloody vomiting and constipation. Past medical history included: alcohol dependence—the patient ceased to consume alcohol after the last esophageal varices banding procedure approximately 5 months earlier, liver cirrhosis (Child-Pugh class B), esophageal varices (grade II/III), 2 esophageal varices banding procedures, secondary thrombocytopenia, cholelithiasis, pathological obesity, epilepsy. Computed tomography (CT) examination of the head performed in the referring hospital was normal. At admission to the ICU the head CT examination was repeated and showed no abnormalities. The physical examination of the abdomen did not reveal any pathologic finding, however its diagnostic value was limited because of obesity. Common causes of decompensation of chronic liver disease include gastrointestinal (GI) bleeding, infection, high alcohol intake and hepatotoxic medications . As the Taking into account clinical picture the differential diagnosis involved: chronic liver encephalopathy, acquired hepatocerebral degeneration (AHD), acute liver encephalopathy. Various nonspecific, not limited to vascular supply region, lesions of the brain, including rare appearance like T1 gray matter hyper-intensity, were consistent with hepatic encephalopathy. Because liver encephalopathy was a possible diagnosis, ammonia concentration was determined. The ammonia concentration determined with spectrophotometry reached 417 µg/dL, highly above the reference range 31–123 µg/dL). Ammonia was found to be an independent risk factor for hepatic encephalopathy and intracranial hypertension, the latter occurring in 55% of patients with ammonia concentrations >200 μmol/L (340.6 µg/dL) [3 µg/dL. Due to history of umbilical hernia, computed tomography (CT) of the abdomen and pelvis was performed and showed radiographic signs of mechanical bowel obstruction with dilation of jejunum and ileum proximal to the hernia A–C.The patient underwent emergency resection of the intussuscepted ileum. The intussusception of the small intestine was described previously a cause of acute-on-chronic liver failure ,4. Due tIn this case report the MRI suggested the diagnosis of hepatic encephalopathy and prompted search for alternative causes of decompensation of chronic liver disease, whereas CT confirmed the cause of decompensation of the disease.


Extensive vaccination programs are being implemented worldwide for coronavirus disease 2019 (COVID-19). With the spread of vaccination, swelling of the lymph nodes after vaccination is frequently seen. We encountered a patient who developed left axillary lymphadenoma following vaccine administration.The patient was a Japanese woman in her 80 s who had previously undergone surgery for right breast cancer. She received two injections of the Pfizer-BioNTech COVID-19 vaccine in her left arm. Approximately 3 months later, she complained of left axillary swelling, and imaging resulted in a diagnosis of left axillary lymphangioma. In accordance with the patient’s wishes, we performed axillary mass resection. The pathological diagnosis was lymphangioma.Our examination findings indicated that congestion of the axillary lymph vessels might have been caused by upper-arm injections of the COVID-19 vaccine. Extensive vaccination programs are being implemented worldwide for coronavirus disease 2019 (COVID-19). According to the package insert for the Pfizer-BioNTech COVID-19 vaccine, the incidence of lymphadenitis as an adverse event is low , 4. HereA Japanese woman in her 80 s received a second injection of the Pfizer-BioNTech COVID-19 vaccine in her left deltoid muscle in 2021. She had a history of right breast cancer (T1N0M0) and had undergone breast-conserving surgery and sentinel node biopsy in her 70 s. Postoperative follow-up examinations were continued, and no sign of recurrence, including in the left axial region, was observed until 2021. There was no evidence of trauma to the left axial region. Her early adverse reaction following vaccination was mild pain at the inoculation site on the day of vaccination and the following day. However, 3 months after the second vaccination, she noticed left axillary swelling and visited the outpatient department at the Tokushima Breast Care Clinic. No lymphedema was found on her upper arm, but an elastic-soft left axillary mass was detected, and breast ultrasonography (US) revealed a 6-cm cystic mass. Computed tomography (CT) revealed a cystic mass without any solid pattern near the normal lymph nodes, and a lymphangioma was suspected  revealed a 6.2 × 3.4 × 7.4-cm mass having a clear boundary, smooth surface and nearly homogeneous internal pattern. Compared with the surrounding muscle, the mass had a low signal on T1-weighted imaging . The congenital cause is thought to be obstruction of the lymph vessels during the fetal period . On the We let the patient choose between conservative treatment and tumor removal. At the patient’s request, surgery was performed, which confirmed the diagnosis of lymphangioma. Although a causal relationship between vaccination and lymphangioma cannot be proven, we cannot rule out the possibility that the lymph vessels were congested because of an inflammatory response in the axillary lymph nodes and vessels. The following observations support this possibility: (1) the symptoms first manifested after the second vaccination; (2) there were no abnormal findings in the left axillary region in the 2008 CT examination or the 2021 US examination; (3) the left axillary region had incurred no trauma; and (4) no radiotherapy had been performed following the earlier surgery for the right breast cancer.Our findings indicated the possibility that occlusion of the axillary lymph vessels was caused by upper-arm injections of the COVID-19 vaccine. Further accumulation of cases is expected to elucidate the cause and pathogenesis of lymphangioma.