Escherichia coli or mixed growth of bowel organisms. Urinary tract infections caused by Lactococcus lactis are very rare, as it is rarely considered pathogenic in humans. We report the case of a 70-year-old woman who presented with symptoms of a recurrent urinary tract infection. Urine cultures were positive for L. lactis. Abdominopelvic computed tomography (CT) revealed focal thickening of the bladder wall and gas in the bladder. Cystoscopic examination and colonoscopy revealed sigmoid colon cancer and a sigmoido-vesical fistula. Laparoscopic surgical treatment was done. The patient recovered and was discharged 3 weeks later without chemoradiotherapy. On follow-up after 6 months, the patient was asymptomatic and stable. To our knowledge, this is the second reported case of L. lactis infection of the urinary tract and the first reported case in adults. L. lactis infection usually indicates the presence of serious underlying diseases such as malignancies, uncontrolled diabetes, and organ failure.A colovesical fistula is a pathological communication between the colon and bladder. The symptoms include pneumaturia, fecaluria, and a lower urinary tract infection. The diagnosis is based on clinical symptoms, but the symptoms are not specific. Therefore, confirming the diagnosis is challenging. Urine cultures performed in patients with colovesical fistulas usually show growth of  Lactococcus lactis infection is very rare since it is considered nonpathogenic in humans. Here, we present the case of a 70-year-old woman with a recurrent urinary tract infection. Urine cultures revealed L. lactis. She was diagnosed with a sigmoido-vesical fistula secondary to sigmoid colon cancer, underwent surgical treatment, and recovered. To our knowledge, this is the first reported case of L. lactis infection of the urinary tract in adults.Colovesical fistula is a rare complication of various diseases, particularly diverticulitis and neoplasms of the colon and bladder. It is difficult to diagnose because of the atypical symptoms. A 70-year-old woman was admitted to our hospital due to urinary frequency, urgency, and hematuria of 1 month duration. She also complained of lower abdominal pain and 3 kg weight loss. She presented with a fever for 3 days during this period, with a peak body temperature of 38.3°C. Approximately 10 days prior to admission, she consulted the urology outpatient department and received anti-microbial treatment. Thereafter, only a slight resolution of symptoms was observed.The patient had a history of type 2 diabetes mellitus for 10 years and was maintained with oral hypoglycemic drugs.Upon physical evaluation, her vital signs were stable. Enlarged superficial lymph nodes were not palpable. Mild tenderness was observed in the lower abdominal region, without rebound tenderness. No costovertebral angle tenderness was noted.L. lactis in two readings. Ultrasound examination showed that the right bladder wall was thickened and revealed a 3.6 cm × 1.5 cm lesion, which was considered likely inflammatory tissue . Routine urinalysis showed positive results for red cells (39/high power field), white cells (47/high power field), urine protein, and urine sugar. The fecal occult blood test results were positive. Serum tumor marker analysis revealed that carcinoembryonic antigen (CEA) was mildly elevated to 6.0 ng/ml . Fasting plasma glucose was 13.6 mmol/L, and glycated hemoglobin A1c (HBA1C) was 8.9%. Urine culture was done thrice, which detected On day 10 of admission, the temperature suddenly increased to 39.3°C with worsening lower urinary tract symptoms and lower abdominal pain. Also, foreign bodies were observed in the urine, described as similar to watermelon seeds and tea stems. Abdominopelvic computed tomography (CT) revealed focal thickening of the bladder wall and gas in the bladder , fecaluria (51%–68% of cases), and symptoms of lower urinary tract infection (57%–71%), which include frequency, urgency, suprapubic pain, and hematuria –5. Our pThe diagnosis is based on clinical symptoms; however, the symptoms and signs are not specific. It is challenging to confirm the diagnosis of a colovesical fistula, and it may take months before the condition is recognized. The patient in this case was monitored for almost a month before the diagnosis was confirmed. After admission, poor glycemic control was considered the probable cause of the urinary tract infection; however, the symptoms resolved partly after administration of insulin glargine and intravenous ceftazidime. Further work-up was performed when a sudden worsening of symptoms was observed despite ongoing treatment. A classical presentation of colovesical fistulas is Gouverneur syndrome, characterized by suprapubic pain, frequency, dysuria, and tenesmus , which wEscherichia coli, Klebsiella, and Enterobacter, which travel from the gastrointestinal tract and perineal area into the urinary tract. Previous studies reported that urine cultures performed in patients with colovesical fistulas showed growth of E. coli in approximately 33% of cases and mixed growth of bowel organisms or enterococci in approximately 65% of cases  for the publication of this case report.YA, QC, and YL were the physicians-in-charge of the patient, reviewed the literature, and contributed to manuscript drafting. LL, DW, and YY reviewed the literature and contributed to manuscript drafting. WK and DA were the patient’s surgeons, analyzed and interpreted the imaging findings. DL performed the cystoscopy and was responsible for the interpretation of the findings. All authors contributed to the article and approved the submitted version.


Intra‐dural perimedullary lipomas involving craniovertebral region are rare. There is no clear tumor‐cord border and patients are at high risk of neurological deficits after surgery. Partial resection of the mass and neural decompression are the main surgical strategies in symptomatic patients. Spinal intramedullary lipoma within the craniovertebral region is extremely rare. These lesions are often associated with spinal dysraphism. Here, we present a nondysraphic one. Our patient was a 56‐year‐old woman who presented with progressive right shoulder pain and gait disturbance. Imaging showed an intradural lipoma, adherent to medulla, and upper cervical spinal cord. The patient underwent limited suboccipital craniotomy and C1‐C5 laminectomy. Lipoma was resected partially due to the adherence of the tumor to neural tissue. Her weakness was exacerbated immediately after surgery but improved within 4 weeks. They account for <1% of spinal cord tumors.2The patient was a 56‐year‐old woman. She had suffered chronic but progressive pain in her right shoulder since 20 years ago. She also complained of clumsiness in her right upper and lower limbs, and gait disturbance started 2 months ago. She had no sensory loss or bowel/bladder involvement. In the physical examination, four limbs tone and strength were normal except mild weakness in right shoulder abduction. Light touch, pinprick, and vibration perception were normal on both sides. Deep tendon reflexes were hyperactive on the right side (3+), and Hoffman sign was present on the right side. Plantar responses were upward bilaterally. Routine laboratory data were normal.MRI showed an intradural well‐defined mass lesion extending from the medulla to C4 level. it was hyperintense in T1 and T2 sequences. Fat‐suppression sequence showed intensity reduction in the lesion in favor of fat tissue , preventive surgery is not recommended.Although the amount of resection has increased using CUSA and microsurgical techniques under microscope,4Nondysraphic perimedullary spinal lipomas are extremely rare. There is no consensus regarding the management of these lesions. Generally, trying for gross total resection can lead to severe neurological deficits and is not recommended. Subtotal tumor resection using CUSA under intraoperative neuromonitoring is a rational approach for most symptomatic cases.Faramarz Roohollahi: Conceptualization; methodology; writing – original draft. Vahid Sharifi: Data curation; methodology; validation; visualization. Setare Rostami: Investigation; visualization; writing – review and editing. Morteza Faghih Jouibari: Methodology; project administration; supervision; writing – review and editing.All the authors involved in this article are researchers with educational targets in medical universities. The authors are not funding by any corporation or government. Also, we have to clarify that all the writers involved in this article are researchers with educational targets in medical universities, and they have no government involvement or any other official representative.The authors report that there is no competing interest to declare.Written informed consent was obtained from the patient to publish this report in accordance with the journal's patient consent policy.


Malignant melanoma (MM) frequently occurs in the skin or mucosa, whereas malignant melanoma of unknown primary (MUP) is diagnosed in patients with lymph nodes or visceral organs as the site of origin, where it is challenging to detect the primary lesion by comprehensive examination. MUP is possibly related to the spontaneous regression of the primary lesion. In addition, primary hepatic melanoma (PHM) usually refers to the primary MM occurring in the liver, with no typical primary lesions and no manifestations of tumor metastasis. A 61-year-old male patient with liver as the site of origin was diagnosed with MM by Melan-A, HMB-45, and S-100 immunohistochemistry staining of liver biopsy tissue. Based on a comprehensive examination, no basis was found for melanoma in sites such as the skin, mucosa, five sense organs, brain, digestive tract, respiratory tract, or genitalia, and the patient was subsequently diagnosed with MUP. MMs require a comprehensive inspection, beginning with the liver, to search for the primary lesion; if the primary lesion is not found, the possibility of PHM or MUP should be considered. With mo2The patient was a 61-year-old male retiree who was in excellent health and had no history of skin and eye diseases or related treatment, no toxic exposure, no addiction to tobacco or alcohol, and no family history of hepatocellular carcinoma.2.1The chief complaint was abdominal distension accompanied by loss of appetite and weight loss for more than a month. Symptoms included abdominal distention, poor appetite, nausea, weakness, and progressive weight loss. On physical examination, no obvious melanocytic nevi were discovered on the skin and mucosa of the entire body. Palpation revealed that the site, a width of four transverse fingers below the xiphoid process of the liver, was harder, hypersensitive, and nodular.Informed consent: Informed consent has been obtained from all individuals included in this study.Ethical approval: The research related to human use complied with all the relevant national regulations, institutional policies and is in accordance with the tenets of the Helsinki Declaration, and has been approved by the Ethics Committee of Affiliated Hospital of Nanjing University of Chinese Medicine.2.2Blood routine examination was basically normal; liver function: GOT 50 U/L (10–50 U/L), GPT 69 U/L (10–40 U/L), Glb 35.0 g/L(35–52 g/L), Alb/Glb 1.03, AKP 859 U/L (40–130 U/L), GGT 623 U/L (8–61 U/L), TBil 42.60 µmol/L (<24 µmol/L), DBil 27.60 µmol/L (<5 µmol/L), ChE 2126 U/L ; PCT: 2.10 ng/mL (<0.046 ng/mL); HBsAg, HBcAb, and HCV antibodies were negative; tumor markers: CA19-9 49.2 IU/mL (<37 IU/mL), FER 724 ng/mL (23.9–336 ng/mL), AFP 1.34 IU/mL (<10.9 ng/mL).2.3Abdominal computed tomography (CT) enhancement suggests hepatomegaly, diffuse space-occupying lesions of the liver, uneven enhancement in the arterial phase, and splenomegaly; abdominal MRI reveals diffuse space-occupying lesions of the liver ; positro2.4Pathology of liver biopsy tissue: appearance: black, under the microscope: a malignancy with pigmentation ; immunoh2.5The patient was treated once with 200 mg of pembrolizumab monoclonal antibody, followed by target therapy. At a 5 month follow-up, the patient complained of a modest improvement in abdominal distension and weakness but continued to have a poor appetite, and imaging re-examination suggested no obvious changes in intrahepatic lesions.3MUP, with an incidence of approximately 3% in MM, is defined as metastatic melanoma detected under the skin and in lymph nodes or other visceral organs, with no obvious indications of the primary lesion ,5. DasGuRegression of the primary focus is the most likely reason for the pathogenesis of MUP to remain unclear . The freInitial consideration was given to the rare diagnosis of primary hepatic melanoma (PHM), for which only a few reports exist ,13. MelaMRI is the preferred imaging technique for detecting the liver and spleen metastases of MM . In addiIn terms of prognosis, the mean survival of MUP patients with lymphatic metastasis is 24–165 months, and that of MUP patients with visceral metastasis is 3–13.2 months . In term4MM originating from the liver necessitates the accumulation of a comprehensive medical history, a thorough physical examination, and a corresponding auxiliary examination in order to search for the primary lesion. If the primary lesion cannot be identified, PHM or MUP should be considered.


The role of immune checkpoint inhibitors (ICIs) continues to receive more attention as evidence emerges of their efficacy and improved survival in patients with advanced biliary tract malignancies including cholangiocarcinoma (CCA). In line with this evidence, we describe the case of a 52-year-old male patient who presented initially in October 2019 with abdominal pain. Investigations revealed multifocal hepatic masses that proved to be CCA that was considered unresectable. Chemotherapy with cisplatin (C) and gemcitabine (G) was initiated. In January 2020, progressive disease was noted, prompting the initiation of 5-fluorouracil (5-FU) and oxaliplatin along with pembrolizumab. Since March 2020, pembrolizumab monotherapy was pursued with radiological evidence of excellent response. Pembrolizumab monotherapy was continued through August 2022, where positron emission tomography (PET)/CT scan showed no evidence of active disease. This case serves to supplement the ongoing evidence of ICI efficacy especially as a durable, sustained radiological response was evident for more than three years from the time of diagnosis. Cholangiocarcinoma (CCA) originates from bile duct cells and accounts for 3% of all gastrointestinal tract tumors . It is kIn October 2019, a 52-year-old male patient presented to the emergency department (ED) with worsening abdominal pain for one month. The pain was described as right upper quadrant (RUQ) abdominal pain, intermittent, aching, aggravated by movement and oral intake, and occasionally radiating to the back with poor appetite and 35 pounds weight loss over the same period but no nausea, vomiting, and change in bowel habits. Past medical history was unremarkable except for diabetes type 2 on metformin. In the ED, vital signs included blood pressure of 140/80 mmHg, heart rate of 80 beats per minute, respiratory rate of 17 cycles per minute, temperature of 36.7 degrees Celsius, and peripheral oxygen saturation (SpO2) of 100 % in room air. On the physical exam, he had RUQ tenderness and moderate abdominal distention. Otherwise, the physical exam was unremarkable. Basic labs showed elevated alkaline phosphatase 313 U/L (reference 38-126), with normal aspartate aminotransferase (AST), alanine transaminase (ALT), and bilirubin. Otherwise, his labs were unremarkable. A CT scan of the abdomen and chest with IV contrast showed left paracardiac lymphadenopathy adjacent to a large 6 cm x 4 cm mass in the left hepatic lobe that was exophytic into the pericardial fat pad with another smaller 5 cm x 4 cm left hepatic lobe mass, one enlarged lymph node in the lower mediastinum measuring 3 cm, and multiple other enlarged lymph nodes affecting the porta hepatis region and left perigastric region  with worsening RUQ abdominal pain for two weeks which was uncontrollable with opioids. A CT scan of the abdomen and chest showed evidence of progression despite being on chemotherapy (Figure 020 with A repeat CT scan in February 2021 showed new development of cirrhosis but with further interval decrease in the size of left hepatic masses 3.3 cm x 2.3 cm and 2.6 cm x 1.4 cm Figure . The patClassification of CCA is based on location and includes intrahepatic CCA and extrahepatic CCA. The latter can be further classified as perihilar and distant CCA . MortaliPembrolizumab has shown good response in certain malignancies including gastric, urothelial, non-small cell lung carcinoma, and melanoma -11. EvidOnly a handful of cases of successful management of advanced CCA with pembrolizumab were reported in the literature Table  14-17].-17.14-17The role of ICIs in the management of patients with advanced unresectable biliary tract cancers including CCA is under continuous investigation. There is emerging evidence of efficacy and improved overall survival, prompting the recommendation of ICIs as first-line therapy in the appropriate setting. This case serves to supplement the ongoing evidence of this efficacy especially as durable sustained radiological response was evident for more than three years from the time of diagnosis.


Cystic mucinous neoplasms of urachal origin cover a wide spectrum of benign and malignant lesions arising from the remnants of the urachus. They display various degrees of tumor cell atypia and local invasion, with no reported cases of metastasis or recurrence after complete surgical resection. We present a 47-year-old man who referred to our Surgical Department due to an abdominal cystic mass incidentally found upon abdominal ultrasound. He underwent en block resection of the cystic mass along with partial bladder dome cystectomy. The histopathology of the resected specimen revealed a cystic mucinous epithelial tumor of low malignant potential with areas of intraepithelial carcinoma. The patient showed no evidence of disease recurrence or distant metastasis 6 months after resection and is scheduled for follow-up with serial MRI or CT scans and blood tumor markers over the next 5 years. The urachus is a tubular structure connecting the allantoid and the dome of the urinary bladder during fetal development, which is usually obliterated after birth and converted into the medial umbilical ligament. Occasionally in the event of incomplete atresia, urachal remnants consisting of tubular structures lined with transitional epithelium can be complicated with infection, cystic formation, or neoplasm development .Urachal carcinoma is a rare clinical entity usually identified as a midline, supravesical mass, with mixed solid and cystic components that may be difficult to differential diagnose from other benign urachal lesions or urothelial bladder cancer . It is aCystic mucinous neoplasms of urachal origin cover a wide spectrum of benign and malignant lesions from the common mucinous cystadenoma to the rare and aggressive frankly invasive mucinous cystadenocarcinoma. This category also includes mucinous cystic tumors of low malignant potential with/without intraepithelial carcinoma as well as mucinous cystadenocarcinoma with microinvasion .In the present work, we report the case of a 47-year-old man who referred to our Surgical Department due to a cystic mucinous epithelial carcinoma of urachal origin and we mention our considerations on this rare tumor. A 47-year-old man was referred to our Surgical Department for an abdominal mass. It was found incidentally upon abdominal ultrasound after being admitted to another hospital with symptoms of fatigue and palpitations. The patient's medical history revealed a Hodgkin Lymphoma in remission after treatment 30 years ago, hypertension, dyslipidemia, complete atrioventricular block with a dual-chamber rate-modulated (DDDR) pacemaker implantation. He had a history of tobacco use, having smoked for a period equivalent to 25 pack years. The physical examination of chest and abdomen was unremarkable. A chest X-ray was normal. Complete blood count and biochemical tests were within normal range. On admission, the patient exhibited slightly elevated serum level of carcinoembryonic antigen (CEA) of 6.12 ng/mL  and elevated serum level of carbohydrate antigen 19-9 (CA 19-9) of 72.50 U/mL . Colonoscopy was unremarkable other than a microscopic polyp which was endoscopically removed. The patient underwent a hematological assessment due to the past medical history of Hodgkin Lymphoma, which was negative for disease recurrence.Ultrasound examination of the abdomen revealed a lower abdomen mixed echo mass with cystic and solid segments separated by septa. The lesion measuring 224 mm x 141 mm displayed a blood flow towards the more solid areas of the mass.A contrast-enhanced CT scan of the abdomen Figure  demonstrAfter multidisciplinary team (MDT) council, surgical excision of the tumor was scheduled. After bilateral ureteral pigtail placement for ureter protection, we performed an en block resection of the cystic mass along with partial bladder dome cystectomy Figure , while cThe resected cystic mass measured 230 mm x 220 mm x 190 mm with a wall thickness of 1-19 mm and was filled with a cream-colored, gelatinous fluid. Histologic examination of the specimen revealed a cystic lesion with well-developed, thick smooth muscle wall lined by columnar, mucin-producing epithelium with papillary architecture  with areas of intraepithelial carcinoma -14.The patient did not receive any adjuvant treatment and six months after resection was followed-up with CT series scan that showed no evidence of disease recurrence or distant metastases. He is scheduled for a close surveillance with MRI or CT series scan and CEA, CA19-9 serum level tests over the next 5 years.The urachus is a tubular structure of allantoic origin which obliterates after birth forming the medial umbilical ligament. Microscopic remains of the urachus are identifiable in most adult, and in one third of these, the remnants remain in contiguity with the urinary bladder lumen . NeoplasMucinous neoplasms of the urachus represent the majority of the urachal neoplasms, accounting for 31 of the 55 glandular neoplasm of the urachus based on the largest cohort presented in the literature . Due to Urachal tumors usually remain latent for long period because of their obscured location between the dome of the bladder and the umbilicus. They mostly present as an abdominal mass (32% of cases) or incidental finding (32% of cases) as well as with hematuria, mucusuria, or pain . SimilarThe differential diagnosis of urachal neoplasms includes a wide range of benign and malignant conditions. The most common urachal malignancy is urachal adenocarcinoma, but other primary malignancies of the urachus are also possible, including sarcoma, lymphoma, and small cell carcinoma. Metastatic tumors from other sites can also involve the urachus. Benign urachal masses such as cysts, diverticula, and adenomas can present similarly to malignant tumors. Inflammatory and infectious conditions, such as urachitis, tuberculosis, and fungal infections, should also be considered in the differential diagnosis.Radiologic findings reveal a midline mass anterosuperior to the dome of the bladder that can be solid, cystic, or mixed. CT and MRI series provide further information on disease extension, regional lymph node involvement and the presence of distant metastases . EspeciaConsidering the spectrum of atypia within the same cystic mass, sampling can be a hazard of pathologic evaluation. Proper and extensive biopsy should be performed if atypia is found on initial assessment, or in cases of a multilocular lesion, so as to identify potentially small areas of carcinoma that have potential prognostic value , 12.As far as morphological features are concerned, mucinous urachal neoplasms are mainly unilocular, filled with mucin exhibiting often areas of calcification and rarely ossification . Tumor cThe treatment of choice for patients with mucinous cystic neoplasms of the urachus is mass surgical excision, with possible urachectomy or partial cystectomy. The largest series of this type of tumor in the current literature of 39 cases, including three patients with intraepithelial and invasive carcinoma, exhibited no recurrence or metastasis on follow-up after surgical resection. Thus, although the data remain limited, these patients appear to have a favorable prognosis after complete excision .In our study, we present the case of a 47-year-old man who was referred to our department due to an abdominal mass found incidentally upon abdominal ultrasound. Diagnostic workup included blood and liver function tests, cancer markers, and CT of the abdomen that raised the suspicion of a cystic mucinous neoplasm arising from the urachus. We performed an en block resection of the cystic mass along with partial bladder dome cystectomy. The resected mass measured 24 cm at its greatest diameter; the second largest reported in the literature, following the one described by Neo and Lee . Taking into consideration the location, the morphological characteristics and the immunohistochemical profile of the resected tumor, the initial differential diagnosis included a villous urachal adenoma of lower urinary tract, a urothelial carcinoma with glandular differentiation, a primary adenocarcinoma of the bladder, a secondary colorectal adenocarcinoma of colloid type as well as mucinous urachal tumors and mucinous tumors of the appendix.Villous urachal adenomas arise in the dome of the bladder or in the urachus and often comprise a noninvasive, superficial component of an adenocarcinoma. Histologically, they show elongated, finger-like projections with central fibrovascular cores lined by pseudostratified columnar epithelium with variable amounts of intracytoplasmic mucin. Extracellular mucin may also be present. In contrast with the neoplasm of our case, these lesions are grossly polypoid and non-cystic. Moreover, these adenomas are described as forming an excrescent focus of dysplastic mucinous epithelium arising in the setting of otherwise nondysplastic metaplastic urachal remnant epithelium tissues and do not present as mucinous cystic urachal neoplasms with multiple foci of atypia along their lining, as the one in our case .Invasive urothelial carcinoma of the bladder with glandular differentiation and primary adenocarcinoma of the bladder-especially the mucinous type-needed to be excluded as well. The distant location of the neoplasm from the bladder’s epithelium as well as the sharp delineation between the tumor’s and bladder’s epithelium by a thick muscular layer along with the normal appearance of the urinary bladder, as observed through both imaging studies and intraoperatively, ruled out a primary bladder neoplasm.Colorectal adenocarcinoma of colloid variant has almost similar morphological and immunophenotypic characteristics with urachal mucinous tumors. However, the immunohistochemical findings described above , the absence of atypical free cells floating in extracellular pools of mucin in correlation with normal colonoscopic and imaging (CT scan) findings excluded the colonic origin of the neoplasm.The mucinous epithelium of the tumor presented has also many similarities with mucinous neoplasms of appendiceal origin, especially with a low grade mucinous appendix neoplasm (LAMN). These neoplasms consist of mucinous epithelium with low grade atypia and have the potential to spread through the visceral peritoneal surface of the appendix into the peritoneal cavity -and thus in the bladder- forming mucinous, acellular deposits. This condition is called “pseudomyxoma peritonei” and is clinically recognizable. However, the patient’s appendix and peritoneal cavity appeared normal both intraoperatively and radiologically.Urachal adenocarcinomas are glandular neoplasms with specific diagnostic criteria as modified by Johnson et al. . These iConsidering the rarity of these tumors as well as the potential involvement of adjacent structures, we suggest that all case of suspected or diagnosed urachal neoplasms be referred to tertiary hospitals with specialized surgical and urological units. We note the importance of en bloc resection and proper surgical planning in order to ensure negative resection margins and to avoid tumor tear and spillage. We highlight the importance of an MDT approach composing of an oncologist, surgeon, urologist, diagnostic and interventional radiologist, and pathologist to ensure optimal patient outcomes.At present, more data and case series are required to establish therapeutic guidelines for mucinous cystic neoplasms of urachus. Although the prognosis of these patients appears to be favorable based on current evidence, we suggest close and long-term follow-up after complete surgical resection.Mucinous cystic neoplasms of urachal origin cover a wide spectrum of benign and malignant lesions that based on current data published, appears to have a favorable prognosis after complete surgical resection. More case series with close patient follow-up are required to establish therapeutic and follow-up guidelines.


Intracranial meningiomas are benign extra-axial brain tumors. Their etiology is undetermined and several hypotheses have been proposed to explain their genesis. The clinical symptomatology of intracranial meningiomas is atypical and varies according to the location of the lesion, its size, and its relationship with neighboring organs. Imaging is essential for establishing a positive diagnosis, but the way to a diagnosis of certainty remains histological. In this article, we describe the CT and magnetic resonance imaging aspects of an intraosseous meningioma discovered in a patient in her forties that presented with right proptosis, and whose brain MRI revealed a cranial lesion with adjacent meningeal involvement; the CT scan subsequently performed allowed a better analysis of the bone lesion, the appearance of which suggested an intraosseous meningioma. This diagnosis was confirmed by a histological exam. The purpose of this article is to illustrate the CT and MRI aspects of this entity by reporting a case of intraosseous meningioma of spheno-orbital location. Intracranial meningiomas are benign extra-axial brain tumors that represent up to 53% of benign intracranial lesions and 37.6% of all brain pathologies , making A 42-year-old woman, with no particular pathological or traumatic history, presented with progressive headaches evolving for 28 months, dizziness, photophobia, and right proptosis. The clinical examination of the patient revealed a non-axial right unilateral exophthalmos, and on palpation, a hard and immobile right temporal swelling was noted. There was also a decrease in visual acuity, and the fundoscopy showed papilledema. The contralateral eye showed no abnormality. MRI revealed a right intraosseous spheno-fronto-temporo-orbital lesional process in hyposignal on the T1, T2, and diffusion sequences, and enhancing after contrast administration. It was associated with the thickening and enhancement of the adjacent meninges after gadolinium injection Figure . The patMeningiomas are the most common primary intracranial tumors. They arise from the meningothelial cells of the arachnoid membrane and develop in the subdural space . They reClinically, the symptomatology is not typical, as it varies according to the location of the lesion, its size, and its relationship with neighboring organs , and manInvolvement of the oculomotor nerve or the trigeminal nerve is not exceptional , 4. NeurCondensing lesions do not often enhance in post-contrast. We must systematically look for the thickening and enhancement of the dura mater. Magnetic resonance imaging allows a better analysis of the anatomical structures by showing their different components. The tumor appears in marked hyposignal on T1- and T2-weighted sequences. The intravenous injection of gadolinium will increase contrast uptake within the intraosseous component and at the level of the adjacent meningeal envelopes. It allows for the assessment of meningeal extension which is often underestimated by the scanner because of its low differential contrast between the bone and the enhanced meninges, thus making it possible to avoid a recurrence by partial excision. MRI can also detect intracranial extension . ScintigThe differential diagnosis depends on the radiological presentation. With hyperostotic lesions, the differential diagnosis is as follows: sclerotic hemangiomas which are well-defined lesions, with honeycomb trabeculations associated with a periosteal reaction which on MRI appears in heterogeneous T1 hypersignal; fibrous dysplasias appear as ground glass with sharp outlines; the osteoma shows regular and clear contours; and finally osteoblastic metastases and Paget's disease. As for lytic lesions, the differential diagnosis is as follows: osteosarcoma which presents fine and anarchic spicules; osteolytic metastases, and myeloma lacuna. In young subjects, eosinophilic granulomas and epidermoid cysts should also be discussed , 4.Therapeutic management requires surgery by performing a fronto-temporo-sphenoidal craniectomy with resection of all the infiltrated tissues. In the event of incomplete resection, additional radiotherapy may be recommended . Tumor rIntraosseous meningiomas are infrequent benign tumors of ectopic location and increasing evolution. The clinical symptomatology is dominated by ophthalmological manifestations, especially in the temporo-spheno-orbital forms. Imaging is nowadays, the first means of diagnosis because it makes it possible to confirm or disprove the existence of a bone lesion and to analyze the radiological characteristics of the lesion. However, the gold-standard for a diagnosis of certainty remains histological.


Ectopic pituitary adenoma is a rare neoplasm located in the clivus and could mimic other clival tumors. Diagnosis and treatment could be challenging. It should be considered in the differential diagnosis of clival tumors.Ectopic pituitary adenomas (EPAs) are isolated adenomas that can be located in variable locations outside the sella turcica and have a normal‐appearing pituitary gland. These tumors are rare and are thought to often arise from embryological remnants along the route of Rathke's pouch migration. EPAs are associated with a wide range of clinical manifestations depending on hormonal activity and involvement of adjacent structures, which can represent a challenge in making the diagnosis and deciding on the most appropriate management. In this case study, we report a 47‐year‐old male who presented with visual disturbances, a headache, and generalized weakness. Magnetic resonance imaging showed a 2 cm mass located in the clivus invading the sphenoid sinus with an intact pituitary gland. The patient underwent endoscopic transsphenoidal surgery to eradicate the mass while maintaining the integrity of the pituitary gland, which was successful and uneventful. Pathological studies were consistent with prolactinoma, with no cytological malignant features. Post‐surgery, symptoms notably improved, and serum prolactin levels significantly dropped, The patient's condition was satisfactory on follow‐up with no long‐term complications reported. This paper contributes to the existing literature by sharing the clinical management of a challenging and uncommon case. : Endoscopic images of transsphenoidal surgery showing the neoplastic mass adjacent to the intact pituitary gland (A), the neoplastic mass adjacent to both the right and left ICAs , and the pituitary gland after complete resection of the mass (D). EPAs are thought to be extremely rare tumors.2A 47‐year‐old male presented to the ENT Department  with a 1‐year history of both parietal and occipital headaches, retro‐orbital pain, transient blurry vision, fatigue, and difficulty completing routine tasks. However, there were no reported symptoms of hyperprolactinemia. Family, medical, surgical, and drug histories were unremarkable. The physical examination was insignificant. However, an optic nerve examination was not performed before the surgery. Laboratory findings were normal, except for elevated serum prolactin of 269 ng/mL. MRI, with contrast of the sphenoid sinus and pituitary gland, revealed a 2 cm mass within the clivus, invading the sphenoid sinus. The mass showed moderate enhancement with peripheral calcifications. The pituitary gland and its stalk appeared to be normal, in Figure 3EPAs are rare extracellular pituitary adenomas that have no correlation with the components located inside the sella turcica. They are often found along the migration pathway of Rathke's pouch; however, the exact pathogenesis remains unclear.Herein, trans‐sphenoidal endoscopic surgery was performed as a diagnostic and therapeutic procedure in our case. Compared with other locations , the clival EPAs are relatively more difficult to reach, detect, and confirm with an endoscopic biopsy. In addition, experience in EPA treatment is often lacking, considering how rare these tumors are.Marah Mansour: Conceptualization; data curation; formal analysis; funding acquisition; investigation; methodology; project administration; resources; software; supervision; validation; visualization; writing – original draft; writing – review and editing. Zeinah Khozamah: Conceptualization; data curation; formal analysis; investigation; methodology; resources; software; writing – original draft; writing – review and editing. Abdulmonem Naksho: Conceptualization; data curation; formal analysis; investigation; methodology; resources; software; writing – original draft; writing – review and editing. Roaa Zayat: Data curation; investigation; resources; software; writing – original draft; writing – review and editing. Aliaa Al Sabbagh: Conceptualization; data curation; investigation; resources; software. Ahmad Deeb: Conceptualization; data curation; funding acquisition; investigation; methodology; project administration; resources; software; supervision; validation; visualization.No funding was required.The authors declare that they have no conflicts of interest.Not required for this case report.Written informed consent was obtained from the patient for publishing this case report and any accompanying and identifying images or other personal or clinical details of this patient that compromise anonymity. A copy of the written consent is available for review by the Editor‐in‐Chief of this journal on request.Ahmad Deeb is the guarantor of this work.


Isolated fallopian tube torsion is a rare condition presenting with abdomen pain. The diagnosis is challenging because the clinical findings mimic several other conditions. In this article, we present a case of an adolescent girl who was diagnosed with the condition during laparoscopy.A 15-year-old girl presented with complaints of sudden onset left lower quadrant pain, nausea, and vomiting. There was tenderness in the left iliac fossa. Ultrasonography revealed inconclusive findings and contrast-enhanced computed tomography of the abdomen and pelvis suggested possible left adnexal torsion. Hence, she underwent a diagnostic laparoscopy which revealed a twisted, edematous, and congested left fallopian tube. The diagnosis of isolated left fallopian tube torsion was made and she was managed with unilateral salpingectomy.Women of the reproductive age group are usually affected by this condition. The common presentations are abdominal pain, nausea, and vomiting. The physical examination may reveal abdominal and cervical motion tenderness. Per abdominal ultrasound is the first go-to modality in children. Magnetic resonance imaging, if available, is advised in children if the ultrasound is inconclusive because of the absence of radiation. However, it may require sedation. Therefore, contrast-enhanced computed tomography abdomen gives an added advantage in such scenarios as in our case. This condition is managed by surgery with salpingectomy or tube detorsion with preservation of the tube, depending on the intraoperative findings.Clinicians should be aware of the condition given the rarity and challenges in the diagnosis of isolated fallopian tube torsion. HighlightsAn isolated fallopian tube is a rare diagnosis.It usually presents in women of reproductive age group.Presentations are nonspecific.Surgery is the preferred choice for management.1. Early identification of the case is important for prompt management and prevention of future complications such as infertility. Here, we present a case of an adolescent girl who was diagnosed with isolated torsion of the fallopian tube and successfully managed with laparoscopic salpingectomy. Our article has been reported in line with the Surgical CAse REport (SCARE) criteria2.Isolated fallopian tube torsion is a rare condition, and diagnosis is frequently missed because of nonspecific presentations mimicking other conditions, such as ovarian torsion and appendicitisA 15-year-old girl presented to our emergency department with a complaint of lower abdominal pain for 12 h. The pain was sudden at the onset and continuous. It was located in the left iliac fossa and was severe, non-radiating, and progressive. She also complained of nausea and vomiting since the onset of the pain. Her last menstrual period was 15 days before the presentation. There was no history of recent vigorous activity.On physical examination, her vital signs were stable except that she was tachycardic with a pulse rate of 126 beats per minute. There was tenderness on the left iliac fossa; however, no adnexal mass could be palpated. There was no guarding or rigidity per abdomen. Her complete blood count, erythrocyte sedimentation rate (ESR), and biochemistry were within normal range.Per abdominal ultrasound Fig.  shows a On laparoscopy, the left fallopian tube was twisted multiple times and was edematous and congested as a result of torsion Figs A, B. The3. Very few cases have been reported among premenarchal girls4. The incidence of the condition is estimated to be 1 in 1.5 million women3.Isolated fallopian tube torsion is a rare cause of lower abdominal quadrant pain. Women of reproductive age group are mostly affected4. In our case, no predisposing factors could be identified.Factors predisposing to fallopian tube torsion can be divided into intrinsic and extrinsic. Intrinsic factors include congenital anomalies of the fallopian tube and acquired conditions such as hydrosalpinx, cysts, neoplasms, pelvic inflammatory disease, and abnormal peristalsis, whereas extrinsic factors include uterine enlargement, adnexal mass, adhesions, and trauma6. The pain can be associated with nausea, vomiting, and bowel and bladder symptoms6. Physical findings usually include tender adnexal mass and cervical motion tenderness7. This is consistent with our case, as she also presented with abdominal pain, nausea, vomiting, and tenderness in the left iliac fossa. Although her laboratory findings were normal, literature has described that there may be a mild elevation in complete blood count and ESR5.The most common presenting complaint is sharp and colicky abdominal pain that can radiate to the flank or thigh8. Doppler ultrasonography can also aid in diagnosis as impedance or absent flow in the tubular structure can be demonstrated7. CT findings include dilated and twisted tube with thickened wall and an adnexal cyst isolated from the ovary4. In this case, clinical and imaging findings led to a misdiagnosis of ovarian torsion, which was later found to be tubular torsion during laparoscopy.Imaging findings are nonspecific and hence, diagnosis is usually established only during surgery. Typical ultrasonography finding of isolated fallopian tube torsion consists of the dilated fallopian tube with thickened echogenic wall tapering toward either end, giving rise to a ‘beak sign’7, dextrorotation of the uterus, and relatively increased venous flow on the left9. However, our case is unique since she presented with left-sided torsion.The isolated torsion of the fallopian tube is reported to be more common on the right side because of the location of the sigmoid colon on the left side, which prevents adnexal movement10.The isolated fallopian tube torsion should be managed with prompt surgery. If the tube is not completely twisted and the presentation is of short duration, it can be salvaged. However, if the tube is necrotic, salpingectomy is the only option for the managementIsolated fallopian tube torsion is a rare clinical entity. Because of the nonspecific clinical–radiologic presentation, the diagnosis is often challenging and only possible during operation. It is recommended that clinicians remain vigilant of the diagnosis for prompt management and prevention of complications.This case report did not require review by the ethics committee.Written informed consent was obtained from the patient for the publication of this case report and accompanying images. A copy of the written informed consent is available for review by the editor-in-chief of this journal on request.None.S.A.: contributed to the preparation of the first draft of the manuscript, review of the literature, and editing of the manuscript; P.R.R.: collected information on the case and checked and confirmed the final version of the manuscript; N.P.: contributed to the preparation of the first draft of the manuscript.There are no conflicts of interest.Not applicable.Pradeep Raj Regmi.Datasets generated during the current study are publicly available.Not commissioned, externally peer-reviewed.We would like to acknowledge Professor Dr Padam Raj Panta, Dr Sandesh Poudel, Dr Atit Poudel, Dr Shree Prasad Adhikari, and Dr Ichha Upreti for providing the operative images.


Lymphatic neoplasia may occur in various types, such as lymphoma, lymphosarcoma, lympholeukemia, and plasmacytoid leukemia. Lymphoma, defined as a malignant tumour of lymphoid tissue, has been found in a number of fish families including Esocidae and Salmonidae. However, the occurrence of lymphoma is rare in those belonging to the Cyprinidae. A final diagnosis of ocular and testicular T-cell lymphoma in the present study was based on the clinical signs, morphology, and texture of the tumour masses in the macroscopic and microscopic examinations. In addition, histopathological and immunohistochemical findings corresponded to T-cell lymphoma characteristics.Cyprinus carpio Linnaeus 1758) with a large ocular mass and severe exophthalmia in the right eye was referred to the Ornamental Fish Clinic in October 2020. Under anesthesia, enucleation was performed. 57 days after enucleation of the right eye, exophthalmia in the left eye was discovered. 221 days after surgery, the fish was discovered to be dead. At necropsy, a large soft tissue mass attached to the left testis was discovered. There were also small whitish nodules on the surface of the liver. Histopathology revealed a hypercellular ocular mass with scant connective tissue. The sections also revealed multifocal hemorrhages, round to ovoid neoplastic cells, mild-to-moderate anisokaryosis and anisocytosis, and mitotic figures. Basophilic neoplastic cells were found in blood vessels within the testicular mass, raising the possibility of systemic spread. The liver showed microscopic metastasis with morphologic similarities to the ocular and testicular tumors. The neoplastic cells infiltrating the left and right eyes as well as the testicular mass were immunohistochemically positive for CD3 but negative for CD20. The masses were diagnosed as T-cell lymphoma based on histopathological and immunohistochemical findings.A 2-year-old hermaphrodite koi carp (Cyprinus carpio) in Iran.This case report provides the first evidence of clinical, histopathological, morphological, and immunohistochemical findings of an ocular and testicular T-cell lymphoma in a hermaphrodite koi carp ( Cyprinus carpio Linnaeus 1758) as a freshwater ornamental fish is a coloured variety of common carp (C. carpio) that originated in Japan and has been bred in Iran since 2002 [The aquaculture industry’s most important sectors are food and ornamental fish cultivation . Althougnce 2002 –11.Esox lucius) [Oryzias latipes) [Oncorhynchus kisutch) [Ameiurus melas) [Oncorhynchus mykiss) [Scleropages formosus) [Hypanus sabinus) [Ameiurus catus Linnaeus) [Neoplasia can occur in both lower and higher vertebrates . Fish ne lucius) , 21, Japlatipes) , 23, cohkisutch) , black bs melas) , rainbow mykiss) , flower  mykiss) , gold crormosus) , Atlantisabinus)  and captinnaeus) .C. carpio) in Iran.In contrast to the epizootics of lymphoma in Esocidae  and SalmCyprinus carpio) was referred to the Ornamental Fish Clinic, Faculty of Veterinary Medicine, University of Tehran. The koi carp (C. carpio) was referred due to a large ocular mass with an ulcerated surface, extreme exophthalmia, and right eye hemorrhage  [On histopathological investigation, the right ocular mass was hypercellular in scant connective tissue. This mass was composed of sheets and clusters of densely and uniformly basophilic lymphoid cells involving the three tunics , anterior and posterior chambers, extraocular muscles, and adipose tissue. These cells were extended from behind the bony orbit, through the extraocular muscle layer, adipose tissue, the eye globe, and onto the cornea Fig. a-d. At h2)  were oneCancer is a multistep process and a disease of the genome, arising from DNA alterations that disrupt gene structure or function , 38. TheCyprinus carpio) was based on the light microscopic features and immunohistochemical characteristics of the neoplasm as described in both mammals and poikilotherms. Histopathologically, hypercellularity of mass in scant connective tissue was observed, which was consistent with the findings of Kieser et al. [Oryzias latipes) has been described [The complementary diagnosis of lymphoma in the koi carp  were most likely of B lymphocyte origin. Manire et al. [S. tiburo). Immunohistochemically, positive staining of the neoplastic cells infiltrating the left and right eye and testicular mass for CD3 and the negative staining for CD20 were consistent with the findings of Lakooraj et al. [The presence of specific cytoplasmic immunolabelling for CD20 and CD79a suggested that neoplastic cells of lymphosarcoma in a captive bonnethead shark . Also, Schultz and Schultz [Poeciliopsis. Furthermore, Brown et al. [Esox lucius). Following observations on the ornamental fish farm, no more affected fish were discovered. In addition, no infectious agent was detected in the current study. Therefore, further research is needed to determine the cause of the lymphoid proliferation and the precise promoting factor for the development of this tumor in fish.Retroviruses are well established as a cause of lymphoma in many domesticated mammal species  and are  Schultz  showed tn et al. , 57 estaA final diagnosis of ocular and testicular T-cell lymphoma in the present study was based on the clinical signs, morphology, and texture of the tumour masses in the macroscopic and microscopic examinations. In addition, histopathological and immunohistochemical findings corresponded to T-cell lymphoma characteristics.


There are few reports describing the unusual origin of the inferior mesenteric artery (IMA). We report a rare case of advanced sigmoid colon cancer with the IMA arising from the superior mesenteric artery.A 59-year-old man with diarrhea and abdominal distention was diagnosed with advanced sigmoid colon cancer. Colonoscopy revealed a semi-circumferential cancer lesion in the sigmoid colon. Enhanced CT scan and CT angiography showed that the IMA directly originated from the superior mesenteric artery at the level of the second lumbar vertebra. PET-CT suggested metastases in the para-intestinal lymph nodes and the liver, but not in the central lymph nodes along the IMA. Preoperative diagnosis was sigmoid colon cancer cT4aN2aM1a cStage IVA. We performed laparoscopic complete resection as the radical treatment of the primary region prior to resection of the liver metastases. Intraoperative findings showed that the IMA was running parallel to the abdominal aorta; meanwhile, the colonic autonomic nerve was supplied from the lumbar splanchnic nerve at the caudal side of the duodenum. Central lymph nodes around the colonic autonomic nerve were dissected en bloc with the regional lymph nodes. Pathological radical resection including the regional lymph nodes metastasis was achieved. Two months later, complete resection of the liver metastasis was performed. After the adjuvant chemotherapy, no recurrence was observed 1.5 years after the liver resection was performed.Preoperative confirmation of the anatomy helped us to safely complete radical surgery in a patient with unusual bifurcation of the IMA. Three major arteries arise from the abdominal aorta: the celiac artery, the superior mesenteric artery (SMA), and the inferior mesenteric artery (IMA). While there are many various subtypes of the branches from the celiac artery or the superior mesenteric artery reported, there are few reports of those from the IMA , 2. HereA 59-year-old-male presented with a complaint of bloody stools. On physical examination, no mass was felt during palpation. Laboratory data on admission showed that the levels of tumor markers including CEA and CA19-9 were within normal ranges. Colonoscopy revealed a semi-circumscribed irregular elevated lesion in the sigmoid colon that was diagnosed as adenocarcinoma by biopsy  had a defective IMA, two cases 0.06%) had abnormal running of the root associated with visceral retroversion, and two cases (0.06%) branched from the SMA. All other cases bifurcated to the left side at the level of the third lumbar vertebra . The IMA% had abnFor left-sided colorectal cancer with unusual IMA bifurcation, the optimal definition of the extent of lymph node dissection has not been established , 13–16. Although anatomical variation of the IMA is infrequent, preoperative assessment using CT and CT angiography is very important because they can provide clear case-specific anatomical information. Even in patients with rare anatomical anomalies, prior confirmation of the anatomy allows us to safely complete radical surgery.We reported a rare case of sigmoid colon cancer with the IMA directly branching from the SMA. Preoperative confirmation of the anatomy enabled us to safely complete radical surgery even in a patient who had unusual bifurcation of the IMA.


Azygos vein aneurysms are rare and asymptomatic in many cases. The management for these aneurysms is controversial, and there is no clear guideline or evidence-based threshold for surgical or interventional therapy.Herein, we report the case of a giant azygos vein aneurysm in a 78-year-old man that was treated with a reversed L-shaped incision. A 56 × 77 mm saccular azygos vein aneurysm was incidentally detected on computed tomography. Subsequently, surgical resection with interventional radiology and reversed L-shaped thoracotomy was performed. First, we performed coil embolization of the azygos vein aneurysm inflow. Next, a cardiopulmonary bypass was established through a reversed L-shaped sternotomy, and the aneurysm was excised.In this case, surgical resection via reversed L incision was effective. The majority of documented patients with an azygos vein aneurysm (AVA) was diagnosed incidentally by computed tomography (CT) or magnetic resonance imaging (MRI) as a mediastinal mass –5. LargeA 78-year-old asymptomatic male patient underwent evaluation for prostate cancer. CT revealed a 56 × 77 mm mass at the posterior mediastinum Fig. ; subsequThe main causes of AVAs are idiopathic, acquired, and traumatic. Based on the form and medical history of our case, we believe that it was an idiopathic saccular aneurysm that was congenital. In patients with AVA, there are risks of rupture, thromboembolism, and symptoms caused by compression of adjacent structures , 4. It wReversed L-shaped thoracotomy is a good approach for surgically treating AVA owing to excellent visualisation.


Tumors of the salivary gland are a group of complex, heterogeneous lesions that are located either in the parotid gland, submandibular gland, sublingual gland, or minor salivary glands. These tumors have a wide range of etiology, pathophysiology, treatment, and prognosis. Multiple salivary gland tumors are extremely rare and usually occur more commonly in major salivary glands than in minor glands. A 61-year-old man with a chief complaint of swelling in the upper jaw for the past eight years reported to the department of oral and maxillofacial surgery. Incisional biopsy revealed a canalicular adenoma (CA) of the minor salivary gland of the palate. Wide local excision was done with closure using a buccal pad of fat and a collagen sheet. Surprisingly, the excisional biopsy was suggestive of synchronous low-grade polymorphous adenocarcinoma (PAC) with CA of the minor salivary gland of the palate. This appears to be the first reported case of PAC with CA found in the palate. Polymorphous adenocarcinoma (PAC) is the second most common intraoral malignant salivary gland tumor . It occuA canalicular adenoma (CA) is a benign salivary gland tumor composed of ductal epithelial cells arranged in cords. The age of occurrence ranges from 40 to 70 years . It occuA 61-year-old male patient came to the department of oral and maxillofacial surgery with a chief complaint of swelling in the upper jaw for the past eight years. The patient had a history of diabetes for the past three years. The patient was a known smoker for 20 years but had quit the habit 10 years back. The patient did not have any other comorbidities. According to the patient, the swelling was small when it started and has attained its present size gradually over the course of eight years.On examination, there was a lesion measuring 2.1 x 0.9 x 0.6 cm in the mid-palatal region with the presence of ulceration in the center (Figure On radiographic examination, contrast CT revealed a radio-opaque lesion in the mid-palatal region, which did not involve the maxillary bone Figures -4. The lAn incisional biopsy was performed for the lesion, which was suggestive of CA. Sections showed neoplasm of glandular origin showing epithelial tumor cells arranged in anastomosing branching cords and tubules, which appear to float in the stroma. The tumor cells were cuboidal to columnar showing moderate amounts of amphophilic cytoplasm and monomorphous basophilic nuclei. The intervening stroma was loose and edematous with rich vascularity, moderate inflammatory infiltrate, and extensive hemorrhagic areas. Parakeratinized stratified squamous epithelium of variable thickness suggestive of the surface epithelium was also seen.Immunohistochemistry was performed for the incisional biopsy specimen Figure . It was The patient was then planned for complete excision of the lesion under general anesthesia. After performing routine investigations and obtaining anesthetic fitness, the patient was planned for surgery. The patient was cooperative and understood the whole procedure. Informed consent was obtained for surgery as well as general anesthesia. Under general anesthesia, nasotracheal intubation was done. Standard scrubbing and draping were done according to protocols. Lignocaine 2% with adrenaline 1:200,000 was given as infiltration around the lesion. An incision was done using a number 15 size surgical blade. The incision was placed to remove the lesion in total. The lesion was removed and measured 2.1 x 0.9 x 0.6 cm Figure . The bucThe excised specimen was sent for histopathological examination and immunohistochemical analysis to the department of oral pathology Figure . SurprisMultiple sections showed a tumor of glandular epithelial origin, which appeared circumscribed in some areas and infiltrative in other areas. The tumor mass was composed of cells arranged in a variegated pattern like tubular, duct-like, strands and single file arrangement with cellular uniformity Figure . The celImmunohistochemistry revealed S100 was positive in tumor cells and strongly positive in CA areas. P63 showed diffuse nuclear positivity in PAC areas. P40 was negative.Postoperative healing of the surgical site was uneventful. Complete palatal mucosa was restored and follow-up after six months revealed no signs of recurrence.CA is a benign tumor of the salivary gland, which is mostly found in the minor salivary gland. It occurs on the upper lip 80% of the time, followed by the buccal mucosa and palate ,5. It ocPolymorphous low-grade adenocarcinoma is a rare, malignant salivary gland tumor, which is found almost exclusively in minor salivary glands. It is more frequent in the third to seventh decade of life, with a clear female predilection in a 2:1 ratio  Most of Multiple salivary gland tumors can occur as synchronous or metachronous and they can also occur unilaterally or bilaterally. The most common multiple tumors are Warthin’s tumor and pleomorphic adenoma since they have identical histological features. Multiple tumors, which are synchronous or metachronous, are uncommon. It usually occurs in major salivary glands. According to Ortega et al., their case is the first reported case of synchronous low-grade PAC and CA on the upper lip .The advantages of using the buccal pad of fat for closure of the surgical wound are that the buccal pad of fat is in an anatomically favorable position, it can be harvested quite easily, has a low failure rate, and a good rate of epithelialization. The advantages of the collagen sheet are that it has chemotactic properties on wound fibroblasts, and collagen dressings encourage the deposition and organization of newly formed collagen, creating an environment that fosters healing. The disadvantage of collagen sheet dressing is that it requires a secondary dressing.Here, we report an extremely rare case of synchronous low-grade PAC with CA of the minor salivary gland of the palate for the first time. These kinds of tumors occur very rarely. Proper examination, prompt diagnosis, and early and accurate treatment result in success and a good prognosis. The knowledge regarding the biopsy techniques and appropriate immunohistochemistry for such cases helps in proper diagnosis and treatment plans. With such a rare possibility of occurrence of these kinds of lesions, it is difficult to study the course of the disease and the effectiveness of the treatment. So, further case reports and case series need to be published to share the wisdom obtained with fellow surgeons and pathologists.To conclude, we report an extremely rare case of synchronous low-grade PAC with CA of the minor salivary gland of the palate for the first time. Even though such cases occur in rare instances, we, as oral and maxillofacial surgeons, need awareness regarding such lesions for proper diagnosis and to provide the appropriate treatment to the patients.


Adrenal schwannomas are rare benign tumors with no specific imaging and laboratory findings to diagnose preoperatively. Due to the limited number of cases in the literature, clinical, imaging, and pathological findings are presented in this study. Case 1 is a 61-year-old woman patient who has a 31-mm mass in the right adrenal gland. This mass was nonfunctional; in imaging studies, this mass had a cystic necrotic component, and high 18-fluorodeoxyglucose (FDG) uptake was seen. There was no metaiodobenzylguanidine (MIBG) uptake. Laparoscopic transabdominal right adrenalectomy was performed, and the pathology result was consistent with adrenal schwannomas. Case 2 is a 63-year-old man patient who presented with a 38-mm mass in the left adrenal gland. This mass was nonfunctional and similar to that in Case 1; this mass had a cystic component. Laparoscopic transabdominal left adrenalectomy was performed. The diagnosis of adrenal schwannoma with degeneration was revealed. Case 3 was a 72-year-old woman patient admitted to the hospital for a 125-mm left adrenal mass. Similar to Case 1, this mass also had a cystic necrotic component in imaging studies. High FDG uptake was seen, and the patient underwent conventional adrenalectomy due to the suspicion of malignancy. After pathological evaluation, a diagnosis of adrenal schwannoma was made. A main diagnostic challenge in adrenal schwannomas is the preoperative diagnosis. These masses have no pathognomonic finding or specific hormonal function. Imaging findings of these masses may increase the suspicion of malignancy, which may affect decisions for surgery and the surgical technique. These rare tumors generally appear at flexor surfaces of extremities, mediastinum, head, and neck regions. Infrequently, these tumors are seen in the retroperitoneal area , 2. RetrCommon benign tumors originating from adrenal tissue are benign adenomas and pheochromocytomas; however, schwannomas rarely originate from adrenal tissue and are considered as only 0.7% of benign adrenal tumors . Due to Adrenal schwannomas may be confused with adrenocortical cancer, adrenal metastasis, or pheochromocytoma due to their imaging findings . In this2.2.1.A 61-year-old woman patient was admitted to the outpatient clinic because of a 31-mm mass in the right adrenal gland after undergoing a computed tomography (CT) scan for COVID-19. The patient has no medical history other than COVID-19. The CT scan showed a cystic necrotic component that caused suspicion of adrenocortical carcinoma or pheochromocytoma. The patient was evaluated for excess hormone production with the 1-mg dexamethasone suppression test, serum and urine metanephrines, plasma renin activity, and serum aldosterone. There was no excess hormone production by the adrenal mass. Several imaging studies, in addition to computed tomography scans, were performed. Magnetic resonance imaging (MRI) showed a semisolid mass with cystic and necrotic components and peripheral diffusion restriction, which increased suspicion of adrenocortical carcinoma . TherefoOwing to the suspicious findings in the imaging studies, surgical excision was planned. Because of the small mass size, laparoscopic transabdominal adrenalectomy was applied. During gross examination of the excised material, two components were easily seen . One cysAt the end of the pathological examination, the final diagnosis was consistent with adrenal schwannoma. Needle-shaped cells without atypia were seen under a microscope, and diffuse positive reactivity with S100 protein was seen in immunohistochemistry. There was no staining with CD34, Desmin, and SMA. The Ki-67 index was less than 1%.2.2.A 63-year-old man patient was admitted to the general surgery clinic because of a left upper quadrant pain. The patient had no history of medical illnesses and no history of abdominal surgery. After an unremarkable routine physical examination, abdominal ultrasound imaging was ordered. Ultrasound imaging revealed a 38-mm × 36-mm mass in the left adrenal gland with a cystic component surrounded by a thick wall. A biochemical investigation was planned, and the patient was evaluated for excess hormone production with the 1-mg dexamethasone suppression test, serum and urine metanephrines, plasma renin activity, and serum aldosterone. There was no hormonal excess, and no hormone production by the adrenal mass was shown. The patient underwent an abdominal MRI scan, and a thick-walled adrenal mass with a cystic component was revealed. The size of the mass was 33 mm × 37 mm, and the mass was described as isohyperintense in T1-weighted images, hypo-hyperintense in T2-weighted images, and contrast enhancement at the postcontrast series. Due to its heterogeneous imaging characteristics, laparoscopic left transabdominal adrenalectomy was performed. The patient had an unremarkable hospital stay and was discharged on the third day postoperatively. The final diagnosis of the adrenal mass was concluded as schwannomas with degeneration in the pathology report. The patient continued to follow-up examinations for 12 years and had nothing remarkable in imaging studies and biochemical evaluations.2.3.A 72-year-old woman patient was presented to the outpatient clinic for left adrenal incidentaloma. This mass appeared in a CT scan during the investigation for hydrocephalus. The patient has a past medical history of high blood pressure, and vertigo, and laparoscopic cholecystectomy. In routine laboratory tests, no excess hormone production was shown. In the CT scan, the size of the left adrenal mass was 125 mm × 102 mm × 123 mm, and heterogeneous contrast enhancement with cystic-necrotic components was observed . This ma3.Schwannomas are frequently seen in the head, neck, and extremities; however, adrenal schwannomas are a rare clinical entity and constitute only 0.7% of benign adrenal tumors . SchwannAdrenal schwannoma cases are seen more frequently at ages 39 and 59 years and more frequently in women (1.17 times more than men) . These mThe largest diameters of the previously reported adrenal schwannomas varied between 1 and 15 cm, and 64% of these masses were between 4 and 10 cm . HoweverPatients with adrenal schwannomas mostly have complaints of vague pain on the left upper quadrant or have no complaint and incidentally detected a mass on adrenal glands. Due to the COVID-19 pandemic, many patients underwent thorax CT scans, which may increase the possibility of finding more incidentalomas. However, adrenal schwannomas cannot be diagnosed from imaging studies and biochemical tests. Hormonal inactivity of these masses may not be helpful to clinicians for their differential diagnosis; nevertheless, imaging findings may confuse clinicians for preoperative diagnosis. Heterogeneous imaging findings such as thick walls, mixed cystic solid components, calcification, and necrosis may cause suspicion of pheochromocytoma and adrenocortical carcinoma . In MRI In addition to the other imaging techniques, FDG-PET/CT scans may give information about the metabolic activity of adrenal lesions . AdrenalThe main treatment modality of schwannomas is surgical excision; however, there is no certain adrenalectomy technique for schwannomas. An impression of malignancy or invasion may direct surgeons to an open adrenalectomy; therefore, excellent preoperative imaging is crucial for choosing the right surgical technique. Masses with smooth margins are suitable for laparoscopic adrenalectomy. An appropriate size of the masses for laparoscopic surgery is debatable; however, in case reports from several centers, patients with masses larger than 10 cm were operated without complications , 12. PatSchwannomas originate from nerve sheaths and have hypocellular and hypercellular areas of spindle cells. Immunohistochemistry results show strongly positive staining of spindle cells with S100, and this result verifies their neural origin . These mRecurrence or metastasis is not expected after complete resection in adrenal schwannomas. There is no consensus on the follow-up period in these patients; however, long-term follow-up data are limited , 7, 19. 4.Adrenal schwannomas are rare tumors that are mostly diagnosed after surgical resection. Imaging findings are confounding, and these masses are nonfunctional tumors. Radiologic findings cause suspicion of malignancy, and there are no specific preoperative diagnostic criteria. The final diagnosis can be made in pathological evaluation after resection of the mass. The main treatment is surgery, and after complete resection, recurrence and metastasis are infrequent. There is limited data in the literature, and more case reports and case series are needed for better patient care.


Rhabdomyosarcoma (RMS) is a highly aggressive pediatric neoplasm that originates from striated muscle or undifferentiated mesenchymal cells. Based on its histopathological characteristics, the World Health Organization categorizes RMS into four distinct subtypes: embryonal RMS, alveolar RMS, pleomorphic RMS, and sclerosing/spindle cell RMS. Embryonal RMS represents the predominant subtype and primarily manifests in the head and neck region, with the genitourinary system being the subsequent most frequent site of occurrence. Embryonal rhabdomyosarcoma of the cervix (cERMS) is more insidious in the reproductive tract, and there is still a lack of consensus on its treatment. Patient-derived organoids (PDOs) are being prioritized for use in guiding personalized medicine. The application of PDOs to test the sensitivity of chemotherapy drugs in patients with cERMS has rarely been reported. In this case report, we delineate the presentation and diagnosis of a 16-year-old adolescent with cERMS, emphasizing the utilization of PDOs in the management of this infrequent neoplasm. We intend to elucidate the diagnostic and therapeutic processes associated with cERMS by referencing previously reported literature on this infrequent tumor, aiming to offer a foundation for clinical practice. Based oThe predominant clinical manifestation is vaginal bleeding, accompanied by soft, polypoid, grape-like masses. Pathologically, cERMS is characterized by the cambium layer, which is denoted by a subepithelial accumulation of tumor cells . A signiA unified treatment strategy for this uncommon tumor type remains elusive. With a multimodal treatment approach, including surgical intervention, and systemic chemotherapy with considerations for radiotherapy, the survival for RMS patients has been greatly improved . The ovein vitro  organoid culture of human tumor tissue has emerged as a representative platform to model tumor genetic and phenotypic features in vitro . As such2A 16-year-old adolescent presented with a history of “irregular vaginal bleeding lasting over a year, which had worsened over the past six months.” She was subsequently referred to the gynecology department of our institution on November 17, 2022. In October 2021, she experienced an episode of unprovoked, painless vaginal bleeding, which was overlooked and thus remained untreated. On November 14, 2022, due to symptoms of dizziness, fatigue, and palpitations, she sought care at a local hospital. Laboratory evaluation revealed severe anemia with a hemoglobin level of 34g/L. Magnetic resonance imaging (MRI) of the pelvis disclosed a substantial vaginal mass, which extended anteriorly into the anterior fornix and posteriorly into the uterorectal pouch. The local hospital administered a blood transfusion, comprising 7.0 U of leuko-depleted suspended red blood cells and 300ml of frozen plasma, along with hemostatic and other symptomatic interventions. The preliminary diagnosis leaned heavily towards a vaginal or cervical malignancy. Once her condition stabilized, she was referred to our center for further evaluation and management.Upon physical examination, the patient exhibited signs of anemia and had a Grade II bilateral thyroid enlargement. A rectal examination revealed a cystic-solid mass, approximately 10 cm in size, located anterior to the rectum. The mass had well-defined margins, and the deeper pelvic structures could not be palpated through the examination.Laboratory findings indicated anemia, with a hemoglobin level of 92g/L. Tests for tumor markers, thyroid function, coagulation profile, and other relevant indicators yielded results within the normal range, with no significant abnormalities detected.To gain a deeper understanding of the patient’s condition, imaging studies were conducted. Ultrasound imaging revealed a hypoechoic mass, measuring approximately 8.6×6.1×8.0 cm, situated in the vaginal region just below the uterus. A pelvic MRI further identified a mass with mixed signal intensity in the region of the external cervical and vaginal fornix, measuring about 7.4×7.9×8.5 cm. This mass extended upward, infiltrating the mid and lower cervical muscle layers, occupied the vaginal fornix, and extended downward into the mid and upper segments of the vagina. The MRI findings strongly indicated a malignant process Figure 1Vaginal endoscopic and histologic evaluation of the tumor was used to confirm tumor malignancy, tissue origin, and classification , 9. The Based on the staging criteria set forth by the Intergroup Rhabdomyosarcoma Study Group (IRSG), the patient was categorized as being in stage Ia. For subsequent postoperative management, the VAC combination chemotherapy regimen was contemplated for the patient, given its status as the contemporary first-line therapeutic approach for cERMS .2 for a duration of 30 minutes. Upon the Matrigel matrix completed solidification, an expansion medium was added. The PDOs were then placed in a 37°C carbon dioxide cell incubator for cultivation. Subsequently, the PDOs were passaged according to their growth patterns. Before drug sensitivity testing, the PDOs exhibited solid spheroid morphology with irregular edges  of each drug were determined using “GraphPad Prism 8.0”  for four cycles. During periodic follow-up evaluations, recurrence was not observed, the tumor markers level remained normal, and MRI did not show any new lesion. The diagnosis and treatment strategy timeline schematic are presented in 3We report a detailed clinical case of a 16-year-old adolescent, with cERMS and multinodular goiter. A comprehensive histopathological characterization and a Sanger sequencing analysis of this rare tumor are provided. In addition, PDOs were used to reflect drug sensitivities already known for cERMS and to be of use for such screening approaches.RMS represents the predominant soft tissue sarcoma observed in children and adolescents, comprising 2-3% of all malignancies diagnosed within this demographic . AccordicERMS typically presents with vaginal bleeding, protruding vaginal mass, cervical polyp, or expelled tumor fragments through the vaginal . Our casThe majority of cERMS typically present as a polypoid or grape-cluster-like appearance, being soft in texture, and they might be associated with hemorrhage and necrosis . HoweverPrevious studies have shown that nearly all cERMS harbor a DICER1 mutation , 16, 17.Since our patient had both cERMS and multinodular goiter, DICER1 tumor predisposition syndrome was highly suspected. Hotspot mutations of DICER1 were examined in the patient’s tumor sample through PCR amplification of exons 24 and 25 (the DNA sequences encoding the RNase III b domain) and subsequently analyzed via Sanger sequencing . HoweverGiven the rarity of cERMS, standardized guidelines are absent, compelling physicians to rely on experiential insights from published case reports and series. In clinical practice, the primary treatment approach for cERMS continues to be a combination of surgery, chemotherapy, and/or radiotherapy. At present, the methods of surgical treatment mainly include radical hysterectomy with or without lymphadenectomy, vaginectomy, cervicectomy, and local lesion excisions. Since cERMS tend to occur in young women, fertility-sparing is a major concern. The IRSG states fertility-sparing surgery and chemotherapy appropriate treatments for patients with localized disease but not applicable for advanced-stage and metastatic disease . Some caAccording to the recommendations of our country and the NCCN clinical practice guidelines in oncology, abdominal radical hysterectomy, bilateral salpingectomy, bilateral pelvic lymphadenectomy, and paraaortic lymph node sampling were performed for this patient , 9. The It is generally recognized that adjuvant chemotherapy is essential. Montag et al. in 1986 reported a patient who underwent adequate surgical excision and chemotherapy survived for 7 years without recurrence . Subsequin vitro 3D culture systems of human tumor tissue can model histopathological, genetic, and phenotypic heterogeneity across different patients  was better than that of monotherapy. With an IC50 of 1.5820μM, cyclophosphamide demonstrated a meager antitumoral impact. This is in line with previous studies showing that cyclophosphamide did not contribute to the success of the treatment, but significantly reduced treatment toxicity , and minor(s)’ legal guardian/next of kin, for the publication of any potentially identifiable images or data included in this article.XQ and ZZ composed the manuscript and literature review. MX and PC guided the analysis of radiological images and histological images analysis and enriched the discussion. MH reviewed the revised paper and provided the final approval of the version to be published. All authors contributed to the article and approved the submitted version.


Sphenoid sinus lesions grasp the attention of Otorhinolaryngologists due to their prime location and vital surrounding structures. Once detected, these lesions require prompt investigation to identify the underlying cause, usually attributed to a tumor, fungal infection, sinusitis, or polyps, thus allowing tailored treatment.We report a case of an elderly lady whose neurological presentation lead to the diagnosis of sphenoid sinus lymphoma. We discuss the diagnostic challenge in view of its interesting presenting symptoms as well as the surgical approach risk and limitations. Primary extranodal lymphoma in the paranasal sinus is generally uncommon and accounts for only 8% of all sinonasal malignancies . LymphomA 66-year-old female presented with a progressive right-sided frontal and temporal headache, blurred vision in her right eye, and numbness in the right side of the face, all of which had been developing over a period of three weeks. These symptoms were not accompanied by vomiting, dizziness, or any nasal symptoms such as anosmia, nasal blockage, nasal discharge, or epistaxis.On assessment, pain and touch sensation were absent on the right cheek. Nasal endoscopy showed normal findings. There was an absent sensation at the V2 distribution of the trigeminal nerve. CT scan of the paranasal sinuses showed soft tissue mass in the sphenoid sinus with intracranial extension. MRI of the brain and orbits showed a large enhancing tumor mass measuring about 5.0 x 3.2 x 2.5 cm in the sphenoidal sinus, more prominent on the right, with anterior extension into the ethmoidal sinuses and clival bone Figure . There was tumor invasion into the cavernous sinus and encasement of the internal carotid arteries. Extension into the pituitary fossa was noted, displacing the pituitary gland and stalk to the left. An impingement on the right optic nerve proximal to the optic chiasm, close to the orbital fissure, was also apparent. Additionally, there was meningeal thickening and enhancement in the right anterior temporal region, suggestive of tumor infiltration. A right functional endoscopic sinus surgery was performed, and a biopsy of the sphenoid mass was taken. Intraoperative findings revealed a polypoidal mass occupying the whole sphenoid sinus with extension into the right posterior ethmoid Figure .Histopathology of the mass was suggestive of a high-grade B cell lymphoma  . The tumThe tumor cells were medium-sized, exhibiting hyperchromatic nuclei, round to irregular nuclear membrane, clumped chromatin, some with prominent nucleoli, and active mitosis with the ki67 proliferative index of 100%. The tumor cells expressed CD10, CD20, CD79a, MUM 1, BCL2, BCL 6, C MYC, and nuclear stain PAX 5 Figure .Prior to the commencement of treatment, her condition continued to worsen with the onset of cervical lymphadenopathy with orbital apex syndrome secondary to the sphenoid mass. The patient received a chemotherapy regime of R-R1 protocol and R-R2 protocol alternating for three cycles. The regime included a combination of 6-mercaptopurine, vincristine, methotrexate, cytarabine, and daunorubicin. The patient's response to treatment was excellent, with tumor regression and partial restoration of right eye vision.The paranasal sinus is a rare site for primary malignancy, accounting for less than 1% of all cancers, with the sphenoid sinus being the least common among the four sinuses to contribute to these cases . The malDiagnostic challengeDue to their neurological and orbital presentations, sphenoid malignancies remain a diagnostic challenge for Otorhinolaryngologists. A high degree of suspicion and robust collaboration between teams are essential for the early detection of these lesions. Access to imaging technology also significantly influences the early detection of these lesions, many of which are found incidentally. Endoscopic sinus surgery provides a pathway toward faster diagnosis for various types of sphenoid lesions. However, it presents a challenge for rhinologists, as they must walk a fine line between obtaining adequate tissue samples to confirm a diagnosis and avoiding damage to the critical surrounding areas.Orbital apex syndrome is characterized by ophthalmoplegia, ptosis, proptosis, positive relative afferent pupillary defect, and visual impairment due to involvement of cranial III, IV, and VI secondary to sinusitis, tumor, or inflammation . The degA high clinical degree of suspicion and good collaboration between primary care, ophthalmology, and otorhinolaryngology teams are pivotal in the early diagnosis of sphenoid lymphoma. Early symptoms such as facial numbness or visual disturbances should not be dismissed, and thorough clinical examination is essential. The aim is to detect sphenoid malignancies before they reach a considerable size and before they cause irreversible damage.


Cholangiocarcinoma (CCA) is an uncommon biliary neoplasm that is more frequent in male patients. CCA is categorized into intrahepatic cholangiocarcinoma (iCCA) and extrahepatic cholangiocarcinoma (eCCA) associated with the anatomical origin location. The clinical presentation is non-specific and varies depending on the origin, iCCA is generally asymptomatic until advanced disease is present therefore this neoplasm presents a poor prognosis with a survival rate of two years. We present a case of iCCA with lung metastasis in a 29-year-old male patient with no risk factors for this malignancy. Cholangiocarcinoma (CCA) is a rare malignancy of the biliary tree and represents 3% of gastrointestinal neoplasms ,2. CCAs The clinical manifestations of CCA are often subtle and depend if it is iCCA or eCCA. iCCA diagnosis is often incidental and occurs proximal to the second-order bile ducts which are the next largest branches or ducts of the biliary system which join to form or empty into the main hepatic bile duct, so symptoms of biliary obstruction are much less common compared with eCCA -9. JaundCCA has a poor prognosis with a median survival of two years ,10. FiveA 29-year-old Ecuadorian man who had no previous medical history of diseases and denied family history of malignancies. The patient was admitted to the hospital for chief complaints of fatigue, malaise, and vomiting. Physical examination was unremarkable. Laboratory data are summarized in Table Abdominal computed tomography (CT) scan with and without intravenous contrast was obtained and showed irregular liver contours with a space-occupying lesion with indistinct borders, which has a low density in the portal phase with heterogeneous enhancements postcontrast, with a greater axial diameter of 19 cm, involves the entire left hepatic lobe and partially the medial segments of the right lobe associated with low-density nodular images in the portal phase with ring enhancement of 20 mm, 11 mm, 10 mm as well as adenopathies in the course of the celiac trunk  scan with and without contrast revealed a solid, expansive mass of approximately 20 x 12 cm that involves almost the entire left hepatic lobe and partially the medial segments of the right lobe, hyperintense on T2 with hypointense areas on T1 and a pseudocapsule on its periphery with peripheral enhancement in the early post-contrast phase, the mass causes irregularity of the hepatic contour associated with the presence of multiple nodular images inside, it is also associated with multiple multisegmented nodular lesions in the right lobe measuring 21 mm, 12 mm, 10 mm with ring enhancement in the early post-contrast phase, compatible with intrahepatic bile duct space-occupying mass. The lesion involves and infiltrates the left suprahepatic vein as well as the left branch of the portal vein and the left hepatic duct and its branches. Adenopathies in the course of the celiac trunk, measuring 17 mm, 16 mm  95% positive at the edges of the neoplastic glandular epithelium, CEA and CEA monoclonal (CEAm) were 90% positive at the apical luminal edges of the neoplastic glands, negative for CD10 and CDX2. Based on these histopathological and immunohistochemical the diagnosis was consistent with well-differentiated CCA Figure .A high-resolution chest CT scan with and without intravenous contrast was obtained and revealed multiple, bilateral pulmonary nodules of different sizes, randomly distributed, measuring between 13 mm, 12 mm, 11 mm, some of them with post-contrast enhancement, compatible with metastatic lesions. Medial supradiaphragmatic and right paracardiac lymph nodes of 17 mm and 13 mm, no pleural effusions were observed Figure .After determining that the patient was not a surgical candidate (due to metastasis), he was started on systemic therapy with gemcitabine and cisplatin for 21 cycles. Over the course of this regimen, slight dose adjustments were necessary due to weight variations and increase in metastatic lesions.Chemotherapy was changed to oxaliplatin, 5-fluorouracil (5-FU), and folinic acid due to persistent mass increase with a duration of four cycles after the patient presented a decompensation due to hematemesis, he underwent upper gastrointestinal endoscopy, which showed esophageal varices controlled by six-rubber-band ligation and red blood cell transfusion. Follow-up laboratories were performed while receiving gemcitabine + cisplatin and oxaliplatin, 5-fluorouracil (5-FU), and folinic acid. Results are summarized in Table The patient was stabilized and some days after he refused to continue with chemotherapy. He required frequent paracentesis for two months for nonmalignant ascites. The overall follow-up duration was 19 months, and the patient died 21 months after he was diagnosed.CCA is a rare neoplasm with an incidence rate of 0.3/100,000 habitants per year with a predominance in males between the fifth and seventh decade of life from Western countries with an annual incidence increase of 4% ,11. TherUnfortunately, CCAs are generally diagnosed at advanced stages as a result of their non-specific clinical presentation. Signs and symptoms rely upon growth pattern, tumor location, and stage, due to this a large part of the cases are found incidentally. Although the clinical presentation, laboratory, and imaging studies serve as a guide, biopsy and immunohistochemistry are required to confirm the diagnosis ,14. The Serological tumor biomarkers such as CA 19-9 and CEA are usually elevated in CCA, however, the diagnostic accuracy is limited, since they can be elevated in benign conditions such as cholestasis, liver injury, and other cancers. Nevertheless, CA 19-9 values> 1000 U/mL have been associated with advanced-stage or metastatic iCCA ,16. EvenSystemic chemotherapy is often the option for iCCA subjects with metastasis or unresectable tumor . The ABCiCCA is a very aggressive type of cancer with a high mortality rate that in most cases presents symptoms when it is in an advanced stage and surgery is often no longer an alternative treatment. Despite correct diagnosis and treatment, the disease is very aggressive and the patient was already in an advanced stage with metastases in both lungs, therefore, chemotherapy was administered to prolong the time and quality of life for several months until the patient stopped responding to treatment and ultimately succumbed to the disease.


Plexiform neurofibromas are benign tumors that arise from neuronal cells and are commonly associated with neurofibromatosis type 1 (NF1) patients. However, the occurrence of plexiform neurofibromas in the pharyngeal region is extremely rare. In this particular case, we report the successful diagnosis of a retropharyngeal plexiform neurofibroma in an adult male patient without a history of neurofibromatosis. The diagnosis was made using magnetic resonance imaging (MRI) and confirmed by a biopsy. Following the diagnosis, the tumor was surgically excised, resulting in a successful removal of the neurofibroma. Plexiform neurofibromas (PN) are benign neoplasms that originate from neuronal cells. They consist of both neuronal and associated connective tissue, with their composition varying based on their specific location . These tIn this context, we present a case report that highlights the presence of a plexiform neurofibroma in the retropharynx of a patient with no previous pathological history. This report provides insights into the clinical and radiological findings, successful surgical removal of the tumor, and histological aspects of the case.We present the case of a 51-year-old male with no significant medical history who was admitted to the hospital due to a six-month history of dysphagia, a sensation of a foreign body in the pharynx, orthopnea, rhinolalia, and significant weight loss.During the physical examination, the vocal fold mobility was preserved and was assessed by rigid endoscopy, no swollen lymph nodes were found in the neck, and the oropharynx appeared normal. Cervical magnetic resonance imaging (MRI) was performed, revealing a tumor-like mass measuring 40 x 24 x 55 mm located in the retropharyngeal space Figure .Laryngoscopy confirmed that the mass was abutting the epiglottis Figure . AlthougThe surgical procedure was performed successfully without any technical complications or postoperative issues. During the postoperative course, the endotracheal tube (ETT) was successfully decannulated. The excised mass was polyp-like and measured 5 x 3 cm Figure . The patLaryngeal neurofibroma is a rare tumor, with squamous cell carcinomas originating in the mucosa being the most common laryngeal masses that can invade surrounding tissues. Diagnosis of these tumors is typically achieved through direct examination using a rigid endoscope . HoweverIn individuals with a history of neurofibromatosis, tumor onset, particularly in the head and neck region, is more frequent . MoreoveSimilarly, plexiform neurofibromas affect both sexes equally and typically manifest in patients without a history of neurofibromatosis type 1 between the ages of 30 and 60 years. Hoarseness is often the initial symptom observed, followed by dysphagia and, subsequently, dyspnea , mirroriPlexiform neurofibromas are mostly benign neoplasms that should be considered in the differential diagnosis of head and neck tumor masses. Despite being benign, they exhibit slow growth and can develop complications, such as hoarseness, dysphagia, and dyspnea. If not diagnosed in a timely manner, these complications can lead to high mortality rates. Additionally, the assistance of auxiliary exams is important for surgical decision-making, and proper characterization of the pathological sample will help determine the origin and degree of recurrence in the patient.