Urinary tract infection (UTI) is the most commonly occurring serious bacterial infection in young infants. Uncircumcised male infants have a higher rate of UTI when compared with circumcised male infants and girls. A prostatic abscess is a very rare clinical variety of UTI, especially in neonates.We present the case of a 15-day-old male neonate who developed a rare variety of urosepsis. The baby was evaluated and found to have a prostatic abscess. Ultrasound of the abdomen showed an enlarged prostate gland with diffuse heterogeneous hypoechogenicity. Magnetic resonance imaging (MRI) of the pelvis showed an enlarged, lobulated prostate with T2 hyperintense signal and T1 hypointense signal and diffusion restriction. The post-contrast images in the pelvis-MRI also showed peripheral rim enhancement suggestive of a prostatic abscess. Urine culture showed growth of methicillin-resistant Staphylococcus aureus (MRSA). The baby was treated with intravenous vancomycin, and pus was drained through a transurethral approach.Phimosis can cause purulence in the prostate. Prostatic abscess usually has a good prognosis in neonates when diagnosed early and appropriate treatment was instituted. Urinary tract infection (UTI) is the most commonly occurring serious bacterial infection in young infants. UTI in neonates (<30 days of age) is confederated with bacteremia and congenital anomalies of the kidney and urinary tract. The true incidence of UTI in the neonatal age group is not known because the prevalence of bacteriuria in healthy term neonates is very uncertain and difficult to assess. UTI affects approximately one in six febrile neonates ≤30 days of age and the prevalence of UTI in febrile infants <8 weeks of age is 13.6%. UTI in neonates have a male predominance; males are affected 2.5-times greater than females [Uncircumcised male infants have a higher rate of UTI when compared with circumcised male infants and girls. The presence of underlying renal abnormalities increases the risk of neonatal UTI. Vesicoureteric reflux is associated with approximately 20% of neonatal cases of UTI. Neonates with UTI often present with fever, poor feeding, lethargy, vomiting, and diarrhea. Neonatal UTIs are sometimes associated with jaundice, also .The occurrence of pyogenic infection of the prostate in a neonate is very rare. We report the case of a prostatic abscess in a 15-day-old neonate, who was treated successfully.A 15-day-old neonate was brought to the out-patient department (OPD) with complaints of fever, hematuria, and pyuria. The baby had a temperature of 37.8 degrees Celsius. Urine analysis revealed slightly turbid urine with more than 50 leucocytes per high power field. Abdominal ultrasound was initially performed and revealed an enlarged prostate gland with heterogeneous hypoechoic appearance, prominent peripheral vascularity, and no definite central vascularity on color Doppler imaging  x 2.3 (transverse) x 1.8 (anteroposterior) cm in size. The volume of this region measured about 5.2 cc. This region showed significantly restricted diffusion with a low apparent diffusion coefficient (ADC) signal. A thin rim of hypointense prostatic parenchyma was noted surrounding this region. On post-contrast images, no enhancement was noted within the above-described region, with significant peripheral contrast enhancement confirming an abscess collection , which was sensitive to vancomycin, linezolid, and nitrofurantoin. Then the antibiotic was changed to vancomycin (injection). The prostatic abscess was drained through a transurethral approach. The fever spikes then decreased, and CRP gradually normalized. The ultrasound was repeated, which showed normal-sized prostate with a tiny central hypoechoic focus suggestive of the residual collection Figure . InitialUrinary tract infections in infants are common, but prostatic abscess is not a common clinical entity in infants. A prostatic abscess is a very rare clinical variety of UTI, especially in neonates. Only 13 cases have been reported in the literature before our case report. Some factors are responsible for the occurrence of prostatic abscess in neonates. The first factor is that neonates, themselves, are at risk of any infection. The second factor is an obstruction to the urinary flow because of physiologic phimosis. The third factor is the presence of squamous metaplasia of the epithelium of the utricle, prostatic urethra, and prostatic glands at birth, which undergoes regression during the first week of life .Two mechanisms can explain the occurrence of prostatic abscess. The first mechanism is the hematogenous spread of organisms to the prostate, most commonly after Staphylococcal bacteremia . The othNeonates with prostatic abscess usually present with fever, hematuria, or pyuria. The clinical diagnosis of prostatic abscess in neonates is difficult because rectal examinations are not routinely performed in neonates; also, only one-third of neonates present with flocculence. Even though ultrasound imaging helps diagnose a prostatic abscess, MRI is the investigation of choice, especially MRI with contrast, which will differentiate between acute prostatitis and prostatic abscess. Staphylococcus aureus is the most commonly encountered pathogen in a neonatal prostatic abscess.Treatment of prostatic abscess mainly focuses on the drainage of pus by transurethral, transperineal, or transrectal approach plus appropriate antibiotic therapy based on the culture and sensitivity reports .Williams and Martins reported a series of cases of neonates with prostatic abscesses or periprostatic hematoma. Three cases were of prostatic abscesses, and two had a periprostatic hematoma. Retention of urine was the predominant clinical feature in all those neonates. The diagnosis was easily made by rectal palpation after the bladder had been emptied by a catheter. Escherichia coli and Staphylococcus species were the predominant organisms grown in culture. Urinary retention was relieved by drainage of abscess or hematoma cavity through the transperineal route; the prognosis is good if the abscess cavity is adequately drained .Stewart Mann reported three cases of acute staphylococcal prostatic abscess presented with urinary obstruction in neonates. Urethrogram done in these cases demonstrated enlarged, distended prostate causing obstructive uropathy .Heyman and Lombardo reported a case of metastatic prostatic abscess in a 34-day-old infant who developed staphylococcal pneumonitis and enteritis during the first admission and later developed funiculitis with epididymo-orchitis and urinary retention. Rectal examination revealed enlarged fluctuant prostate. The authors attempted a cystoscopic examination but were unable to pass the cystoscope beyond the prostatic urethra. The cystoscope punctured the prostate, which was followed by a large volume of purulent drainage from the urethra. Culture of the purulent drainage showed growth of Staphylococcus aureus. The infant improved with antibiotic therapy .Collins also reported a case of prostatic abscess in a 10-day-old neonate who died within 36 hours of presentation because of this fulminant urosepsis. A prostatic abscess was found in the autopsy, and culture showed growth of Serratia marcescens .A prostatic abscess is a very rare entity of urosepsis in neonates. A prostatic abscess usually presents with urinary retention, fever, or pyuria. Imaging modalities like ultrasound or magnetic resonance imaging help in the early diagnosis of a prostatic abscess. Treatment of prostatic abscess involves drainage of pus with appropriate antibiotic therapy. A prostatic abscess usually has a good prognosis in neonates when diagnosed early and if appropriate treatment was instituted.


Spinal cord compression (SCC) is a rare initial presentation and complication of acute lymphoblastic leukemia (ALL) with nearly all reported cases occurring in the pediatric population. We report a 38-year-old previously healthy man who presented with acute on chronic lower back pain, gait instability, urinary retention, and severe thrombocytopenia. Radiologic examination revealed two soft tissue masses of the thoracic spine associated with compression fractures causing spinal canal narrowing and cord compression. Bone marrow biopsy confirmed the diagnosis of ALL. Immediate initiation of high-dose corticosteroids and systemic chemotherapy resolved the patient's symptoms without radiation therapy or surgical intervention. After two courses of chemotherapy, the patient achieved complete remission in the bone marrow. Rapid administration of chemotherapy alone in this case resulted in a complete resolution of SCC. Given the rarity of this complication in adults, no standardized treatment has been established. The success of this case recommends chemotherapy as the initial management of SCC in chemotherapy-naïve ALL. Acute lymphoblastic leukemia (ALL) is a malignant neoplasm of early lymphoid precursors. Its incidence is bimodal, with the first peak occurring in childhood (80% of ALL cases) and a second peak around age 50 . ALL has3/μL, and platelet count of 13 × 103/μL. A peripheral smear showed innumerable blasts. Lactate dehydrogenase was 4179 IU/L, and uric acid was 9.9 mg/dL. Magnetic resonance imaging (MRI) of the thoracic spine and computed tomography (CT) of the chest, abdomen, and pelvis both showed compression fractures of the T2 and T9 vertebral bodies. The MRI revealed a 3.0 cm enhancing soft tissue mass that encased and infiltrated the T2 vertebral body and bilateral pedicles and extended through the bilateral T2-T3 neural foramina into the ventral epidural space which resulted in mild canal narrowing. In addition, a 5.0 cm mass encased and infiltrated the T9 vertebral body and extended through the T9-T10 neural foramina and into the epidural space from the level of T7-T8 to the top of T10. Epidural tumor encased the thecal sac and resulted in severe spinal canal narrowing , supporting the diagnosis of ALL. Bone marrow biopsy showed >95% blasts. Cytogenetics and fluorescence in situ hybridization (FISH) panel for ALL were normal; Philadelphia chromosome was not present. There was no B-lymphoblastic population detected in the CSF fluid analysis, albeit CSF sampling occurred after chemotherapy was initiated.Corticosteroids were initiated with immediate improvement of neurologic symptoms within 24 hours. Neurosurgery and radiation oncology were initially consulted. Surgical decompression and radiotherapy were deferred given suspected diagnosis of leukemia. The patient was immediately started on high-dose methotrexate, cytarabine, and rituximab as part of the R-Hyper-CVAD  regimen, as well as intrathecal methotrexate. After one course of chemotherapy, his neurological symptoms improved, with resolution of saddle anesthesia, normal patellar reflexes, regular bladder/bowel control, and increased strength with ambulation. After two courses of R-Hyper-CVAD, the patient achieved complete remission in the bone marrow, and subsequent MRI of the spine showed near resolution of the mass at T9 and resolution of previous spinal canal stenosis.While CNS involvement in patients with ALL is common, there have been only a few documented reports of previously healthy adults presenting with SCC due to extramedullary leukemia in the spinal column at initial diagnosis of ALL. Astute recognition of acute leukemia as a cause of SCC is critical as the management may differ from the typical approach to SCC in patients with solid tumor malignancies.There are only three cases reported in the literature of a previously healthy adult presenting with SCC in association with ALL –8. The cThere is no standardized management of patients presenting with SCC due to ALL. For SCC associated with solid tumors, rapid initiation of steroids to decrease inflammation and to prevent worsening neurological symptoms provides better outcomes . After aThe exception to this general approach occurs when SCC develops in patients with highly chemosensitive tumors. ALL is a very chemosensitive malignancy. Other chemoresponsive malignancies include lymphomas and germ cell tumors, as well as high grade neuroendocrine tumors such as small cell lung cancer . When chOur patient received neither surgery nor radiotherapy, and high-dose dexamethasone followed by expeditious initiation of R-Hyper-CVAD quickly relieved neurological symptoms and increased motor strength of the lower extremities. High-dose methotrexate and cytarabine, routinely administered during the even courses of the R-Hyper-CVAD protocol, were initiated first in this patient with CNS symptoms because of effective penetration of the blood brain barrier with these agents. Complete remission was achieved in the bone marrow after two courses of R-Hyper-CVAD, and repeat spine imaging obtained after four courses demonstrated nearly complete resolution of the T9 mass without cord compression . The patAlthough rare, back pain and SCC could be the initial manifestation of ALL in an adult, particularly in the setting of blood dyscrasia. Malignant SCC is an emergent presentation that often requires rapid management to prevent further neurologic deficits and improve the chance for neurologic recovery. Steroids, followed by surgical decompression and/or radiation therapy, are most often utilized in this setting, particularly when SCC is associated with solid malignancies. However, in the setting of highly chemosensitive cancers , rapid initiation of steroids and systemic chemotherapy offers the advantage of treating both systemic and local disease in a potentially curable cancer, obviating the need for surgery or radiation in the acute setting.


Gastropleural fistulas are a complication of peptic ulcers in hiatal hernias, trauma, infections, surgical complications, and malignancy. Presenting symptoms may include gastric and chest pain with respiratory failure in the setting of pneumonitis, hydropneumothorax, or tension pneumothorax. We describe a 57-year-old male with a history of transhiatal esophagectomy and esophago-gastroanastomosis who presented in the setting of dyspnea and dark orogastric tube output. Upper endoscopy revealed multiple gastric ulcers with a dominant ulceration communicating with an adjacent space, and a fistulous tract was demonstrated on computed tomography chest, confirming a gastropleural fistula, a rare life-threatening condition. Gastropleural fistulas (GPFs) are a rare and dangerous complication of different pathologies including peptic ulcer disease, trauma, infections, surgical complications, and malignancy . PresentThe patient was a 57-year-old male with a history of COPD and transhiatal esophagectomy with esophago-gastroanastomosis and J-tube placement (2005) secondary to high-grade dysplasia within a segment of Barrett's esophagus. Since 2005, he had numerous hospitalizations for dysphagia, regurgitation, and aspiration with subsequent balloon dilation without significant relief. Several records indicate a significant history of leaving against medical advice (AMA) and noncompliance with tobacco abuse and eating solid foods leading to multiple EGD with subsequent removal of retained food products. Also noted on previous endoscopies were findings suggesting functional gastric outlet obstruction and multiple Forrest class III gastric ulcers thought to be from ischemia and stasis.Prior to this admission, the patient ingested solid food which induced abdominal pain, nausea and nonbloody, nonbilious emesis. In the ER, the patient became dyspneic requiring intubation for airway protection. OGT suctioned approximately 1200 cc of dark-colored fluid. Pertinent physical findings included cachectic appearance with right-sided coarse lung sounds. The abdomen was nondistended, soft, and nontender, and without organomegaly or masses. The J-tube was in place without discharge or erythema.Chest X-ray showed right middle and lower lobe infiltrate. Labs were significant for leukocytosis (WBC 22.9 K/cmm), anemia (hemoglobin of 8.8 g/dL), thrombocytosis (platelets greater than 1000 K/cmm), and lactic acid of 9.92 mmol/L. Electrolyte abnormalities included sodium of 129 mmol/L, chloride of 75 mmol/L, and hyperglycemia with glucose at 248 mg/dL. Arterial blood gas showed hypercapnia without significant hypoxia, and EKG showed sinus tachycardia without ischemic changes. He received a 30 mL/kg bolus of intravenous fluids, antibiotics including vancomycin/cefepime with the addition of clindamycin for aspiration coverage, and 80 mg IV pantoprazole and was admitted to the Intensive Care Unit (ICU). Due to worsening hypoxia and the suspicion of aspiration, Critical Care performed bronchoscopy which showed a normal-appearing trachea as well as clear bilateral lung lobes. Secretions were suctioned and the right lung wash-out specimens were sent for pathology; however, there were no reported findings of food material or bleeding. Gastroenterology was consulted for concern of upper GI bleed with dark liquid from OGT and a decrease in Hgb from 8.8 to 6.2 g/dL requiring 2 units of packed red blood cells (pRBCs).EGD discovered 1.5 L of dark gastric content that was suctioned revealing 5 clean-based gastric ulcers. One such ulcer measured approximately 2 × 2.5 cm in diameter and approximately 1 cm deep at 44 cm from the incisors, along the greater curvature. At the base of this ulcer appeared an external communication with a questionable fistula . BiopsieFirst described in 1960 by Markowtts and Herter, GPF can be an acquired complication from peptic ulcerations via hiatal hernia, trauma, and perforation with abscess formation –6. As thPrognosis of this rare complication depends on the time of diagnosis and intervention with prolonged use of antibiotics, acid-suppressing medications, and strict NPO with total parenteral nutrition which can result in successful outcomes. In the absence of universal guidelines, there are conflicting reports on the proper care of GPF. A majority of reports agree that the laparoscopic approach and surgical interventions are definitive treatments as data indicate higher mortality with conservative management –9. Meta-This case provides an excellent example of septic shock in the setting of an extremely rare surgical and traumatic complication. GPF are infrequent and life-threatening with insidious onset and nonspecific symptoms. This diagnosis has been historically difficult to establish and is often misdiagnosed, as is noted in several reviews and case reports , 5–7. A


Hydatid cyst is a disease caused by the larvae of Echinococcus spp. The larvae often reside in the liver, lungs, and brain. Occasionally, a primary isolated thoracic vertebral hydatid cyst is reported to cause severe complications. Various diseases may lead to the development of progressive compressive myelopathy. Herein, we report a rare case of a primary isolated thoracic vertebral hydatid cyst with compressive myelopathy.A 57-year-old female had numbness and weakness in the lower limbs for a span of 3-months.Thoracic magnetic resonance imaging (MRI) showed that an isolated mass was observed in the T5 vertebral body, which compressed the spinal cord. The diagnosis was confirmed after surgical excision, and Echinococcus granulosus was found to be the etiologic factor.The patient underwent laminectomy with no complications.After surgical decompression, the patient made slow and measurable progress. While relatively rare in the non-pastoral area, the primary isolated thoracic vertebral column hydatid cyst may be considered as a possible etiology of atypical extradural spinal compression.This case illustrates the complexity of spinal echinococcosis manifestations and the necessity of an interdisciplinary approach. Hydatid disease is known to be a fatal zoonosis that could endanger human health worldwide, particularly in pastoral areas, semi agricultural, and semi pastoral areas where animal husbandry has been developed. Hydatid cyst is potentially fatal, with a mortality rate from 0.9% to 3.6%.E. granulosus sensu stricto, G4 genotype as E. equinus, G5 genotype as E. ortleppi, G6-G10 genotype as E. canadensis, and E. granulosus lion strain as E. felidis.,3The two primary forms of human infection with Echinococcosis are cystic echinococcosis and alveolar echinococcosis. Cystic echinococcosis is more common, which is known as Echinococcus granulosus. By analyzing the phylogenetic relationship of Echinococcus granulosus, the G1-G3 genotype of Echinococcus granulosus was classified as After human infection with Echinococcus granulosus, one or more echinococci can develop in the body, primarily in the liver and lungs, and often in kidneys, spleen, muscles, central nervous system, and eyes, occasionally in the bones (2% to 5%). We report a case of T-5 vertebral body and T-5-6 level extradural Echinococcus granulosus causing lower-limb paralysis in the absence of clinical history and exposure. The disease has only been diagnosed after histopathological examination. Imaging and clinical manifestations can accumulate more experience and improve the understanding of Echinococcus granulosus vertebral lesions.2A 57-year-old female without a remarkable history was admitted to our hospital on September 8th, 2019. She had a three-month history of numbness and weakness of the lower limbs, severely aggravated by walking. Subsequently, she developed progressive difficulty with ambulation and bladder dysfunction issues.The general physical examination was found to be normal. The neurological assessment showed grade 0/5 strength of the lower limbs by the Medical Research Council, including a sensory loss below the xiphisternum. The patient is an urban resident in northeast China who works as a civilian in a private company. The patient has been stable in the past and has denied any history of tuberculosis, tumor, diabetes, and surgery. The patient had no experience in living pastoral areas and raising livestock. She has no history of smoking and consuming alcohol. No tumors in the nervous system and autoimmune diseases have been traced in her family.Thoracic magnetic resonance imaging (MRI) results showed that an isolated extradural cystic mass was embedded in the T-5 vertebral body . The lesion was hypointense by T-l-weighted imaging and hyperintense by T-2-weighted imaging. Furthermore, the lesion had spread to the spinal canal by compressing the spinal cord from the right side, causing cord compression. Besides, a few thin septa have been found in the cystic lesion. Peripheral and septum enhancement of contrast T-1-weighted imaging was recorded, whereas an abnormal signal has been noticed in the spinal cord , thus confirming the diagnosis of infection with E. granulosus.Histopathological Fig.  and paraFour days after the surgery, her lower limb muscle strength improved to Grade 3/5 . However, bladder dysfunction continued until 10 days after surgery . A CT scan  have not been obtained.MRI helps to assess the hybrid cyst of the vertebral body,Signs and symptoms, including myelopathy, radiculopathy, or cauda equina syndrome, may depend on the location, spinal level, lesion size, and the presence of inflammation caused by cyst degeneration. The present case had progressive compressive myelopathy that was induced by isolated T-5 vertebral body hydatidosis. Hence, surgical excision of the cystic mass with excision of the involved vertebral segments is the preferred treatment for the vertebral hydatidosis for decompressing the neural structures. Surgery is the preferred treatment for spinal echinococcosis. Laminectomy is widely used in the treatment of spinal hydatid cysts.,11 The primary purpose of this operation is to remove hydatid cyst completely and effectively relieve the compression of spinal cord. However, the uncertain anatomical boundary and the existing neural structure make radical resection almost impossible. We considered it necessary to wash off the cystic lumen and the surrounding surgical region with hypertonic saline. The condition of the patient we reported improved after the decompression treatment. Multiple recurrences are a major problem in vertebral hydatid disease.,16 This leads to a poor prognosis for this disease. As Echinococcus granulosus is a systemic disease with locally focal symptoms, a course of anti-echinococcosis therapy in the postoperative period is recommended. In some cases, the definition of cyst inactivity and the evaluation of therapeutic effect are not easy.Even if spinal echinococcosis is suspected, biopsy or aspiration of the cyst is not recommended because of the risk of anaphylaxis and diffusion. Unfortunately, patients who take anti- echinococcosis drugs for life still cannot avoid relapse. In many patients with spinal echinococcosis, especially in the case of cyst rupture which is unavoidable during surgical treatment, “lifelong medical treatment and follow-up” is inevitable. Echinococcosis has brought high expenses and economic losses to the medical and veterinary medicine. Hence, vaccination could be the most successful strategy for preventing hydatidosis.The use of albendazole is recommended to be limited to six months to one year to prevent the recurrence of hydatid cysts. Our patient received albendazole for 6 months and was followed up for 2 years (including CT and MR imaging).While there are some imaging features of the primary cystic echinococcosis of the spine, including cystic mass, accompanied by spinal cord compression and other symptoms, its imaging and clinical features often lack precision. Correct and comprehensive understanding of the clinical and imaging manifestations of spinal cystic echinococcosis is beneficial for early diagnosis and effective development of a suitable treatment plan.Resources: Li Zhang, Hongli Zhou, Junwei Tian.Writing – original draft: Bei Zhang.Writing – review & editing: Jiping Wang.


Diagnosis and treatment of neurosurgical pathology present unique challenges in underserved areas, and many conditions may go undiagnosed, misdiagnosed, or untreated for prolonged periods. The development of an unusual complication, seemingly unrelated to an area of neurosurgical intervention, may be particularly perplexing to non-neurosurgical providers, particularly in areas where neurosurgical procedures have not historically been available. A 44-year-old male presented with a giant meningioma which was successfully resected. A nasal encephalocele was noted preoperatively but was not addressed due to lack of associated symptoms and distance from the tumor. The patient lived on a remote island and was lost to follow-up. He developed delayed cerebral spinal fluid (CSF) rhinorrhea three months after surgery, which was diagnosed and treated by local providers as allergic rhinitis for 11 months until he presented with new-onset seizure. Imaging demonstrated descent of the lateral ventricle into the encephalocele. The encephalocele was amputated and the skull base defect was repaired successfully. The alteration of ventricular anatomy and CSF fluid dynamics following tumor resection appears to have created an environment where a non-traumatic CSF leak could develop where it had previously shown no signs of developing. It may be prudent to treat skull base defects prophylactically to prevent this type of complication, particularly in patients of remote regions where regular follow-up is difficult. Meningiomas are benign intracranial lesions commonly encountered by neurosurgeons, comprising approximately 25%-37% of all primary intracranial neoplasms diagnosed in the United States -3. WorldGiant meningiomas (>5cm in diameter) are less common but may be more likely to be encountered in medically underserved areas due to lack of available resources for diagnosis or treatment, and a general trend of cultural mistrust of medical providers. Studies of racial, ethnic, and socioeconomic disparities within the United States found that minority populations and those in rural areas presented more often with advanced stages of tumor progression ,7 or a lNasal encephaloceles are estimated to occur in between 1 in 5,000 and 1 in 40,000 live births ,12, the We report here a patient who had an asymptomatic ethmoidal encephalocele prior to resection of a giant meningioma, who then developed a delayed cerebral spinal fluid (CSF) leak from the encephalocele months after tumor resection.A 44-year-old male presented in the emergency department with a three-year history of progressively worsening ataxia, apathy, and aphasia. Computed tomography (CT) scan had revealed a large left frontal lesion a year prior, but the patient had declined surgical intervention at that time. A CT scan performed three years prior had shown no evidence of lesion, demonstrating explosive growth within a three-year period. Records from the earliest scan are not available and the patient does not recall the indication for the scan. On examination, he had profound expressive aphasia. For receptive language, he required some translation by family members, who reported that he had previously been fluent in English, but was now limited to Chamorro, his first language. He had right-sided hyperreflexia and ataxia.Magnetic resonance imaging (MRI) showed a 7.7 x 6.3 x 6.2cm lesion consistent with a giant meningioma Figure . A nasalLeft frontal craniotomy was performed, and a gross total resection was achieved. The patient’s preoperative ataxia resolved quickly, and he was ambulatory without assistive devices by the time staples were removed. His aphasia similarly improved. No formal cognitive testing was performed, but he appeared to still have some residual cognitive impairment. The patient's family confirmed that he had returned to his cognitive baseline. He had two months of follow up, but then returned to his home island of Saipan where he was lost to follow up despite multiple attempts to reach him. Tumor pathology was determined to be WHO grade I meningioma.Fourteen months postoperatively, he was readmitted to the emergency room in Saipan with seizures and persistent rhinorrhea; he had developed the rhinorrhea approximately three months after surgery and had been prescribed a nasal steroid spray for allergic rhinitis. Clinical testing revealed a robust flow of clear fluid from his left nostril. CT and subsequent MRI scans demonstrated descent of the left lateral ventricle into the known osseous defect in the cribriform plate Figures , 3. VentGiven the lack of endoscopic equipment, the skull base defect was repaired via open craniotomy. The multi-pedunculated encephalocele was found to be emerging through 2-3mm dural defects. These were suspected to be nonfunctional based on the miniscule pedicles from which they emerged and were hence amputated. The defect was packed with fat graft, and a titanium mesh was secured over the ethmoidal defect. The frontal sinus was cranialized, stripped of mucosa and packed with fat. A vascularized pericranial flap was secured over the mesh. A lumbar drain was placed and, given the risk of meningitis from such a prolonged CSF leak, CSF was sampled for laboratory testing and culture. The patient was kept on broad-spectrum antibiotics until labs were available. Remarkably, no signs of infection were found and the patient showed no clinical signs of meningitis. A clamp trial was attempted, but a slight leak persisted, and hence the drain was re-opened. The patient accidentally removed his lumbar drain seven days postoperatively, but no further leak was observed and the drain was not replaced. Given his previous disappearance to follow up, he was kept on island in a hotel until three months post-operatively. At that time, he was doing well with no evidence of CSF leak and was released to return to Saipan. Pathology of the encephalocele showed no neoplasm or acute inflammation. Postoperative imaging following skull base defect repair showed that the previously descended ventricle had ballooned up into the resection cavity (Figure Giant meningiomas and frontoethmoidal encephaloceles, while certainly recognized throughout the world, are each relatively uncommon entities in their own right -3,11,12.These explanations are of course speculative, and our case illustrates elements which might suggest an alternative hypothesis. First, the CSF leak was delayed by several months postoperatively, which speaks against an immediate injury such as traction. Second, immediately after the meningioma resection, the operative cavity collapsed. It did not balloon out, as would be expected if intracranial pressure were high. As the ventricle descended into the encephalocele, the large resection cavity did not dilate, as would be predicted if it were a purely pressure-related phenomenon. One might anticipate that the cavity would initially fill with CSF and then the encephalocele might present a point of weakness as the previously compressed brain tissue expanded and the resection cavity collapsed, but the order of events suggests a more complex phenomenon: the resection cavity immediately collapsed; the ventricle slowly descended into the encephalocele without dilating the resection cavity; the resection cavity then ballooned out when the encephalocele was repaired, without any other clinical or radiographic signs of hydrocephalus.CSF leaks not related to a traumatic event are rare occurrences, but have been related to the presence of neoplasms, especially skull-base tumors, inflammation, basal encephaloceles, benign intracranial hypertension or pseudotumor cerebri ,18. In tThis case highlights some of the difficulties in treating neurosurgical patients in remote regions of the world. The “standards of care” can vary depending on the availability of trained medical personnel and adequate facilities. This patient had a substantially delayed complication which went unrecognized due to the absence of neurosurgical resources in his area. For patients with asymptomatic skull base defects, it is important to take into account the potential alteration of CSF fluid dynamics when performing other, seemingly unrelated procedures. Compliance with follow-up may also be problematic when it requires travel to a remote care facility, and may prevent timely diagnosis of complications, as in this case. The presence of skull base defects should prompt aggressive counseling about the importance of follow-up. In extreme cases where patients may be unable to schedule a regular follow-up, it may be appropriate to treat such lesions prophylactically, even when they seem asymptomatic and unrelated to the primary lesion at hand.


Intraductal tubulopapillary neoplasm (ITPN) is a recently described rare tumor of the pancreas. Diagnostic approach and treatment are based on relatively few cases.Here we report a case of a 68-year-old male presenting with an ampullary adenoma with high grade dysplasia who underwent pancreaticoduodenectomy and was incidentally found to have an ITPN at the pancreatic resection margin with areas of microinvasion throughout the resected specimen. He went on to rapidly develop an invasive adenocarcinoma arising in association with recurrent ITPN in the remnant pancreas requiring a completion total pancreatectomy.Patients with ITPN present with non-specific symptoms and diagnosis can be challenging. Radiographic evaluation will reveal tumor ingrowth into the main pancreatic duct and distal duct dilatation without upstream dilation or mucinous engorgement. ITPNs are treated with formal resection given that determination of an invasive component can be difficult and the risk of malignant transformation. Following resection, recurrences are infrequent and 5-year survival is over 70 % even with microinvasion.ITPNs can follow a variable clinical course but hold the potential for malignant transformation. When ITPN is incidentally found at a pancreatic resection margin, we recommend completion resection due to the risk of local recurrence. ITPNs a2A 68-year-old male initially presented with recurrent pancreatitis, initially attributed to cholelithiasis for which he underwent a laparoscopic cholecystectomy. Postoperatively, he continued to have episodes of pancreatitis which prompted an MRCP that demonstrated dilation of the common bile duct and pancreatic duct to 8 mm and 4 mm respectively A with a Following a negative diagnostic laparoscopy, a pancreaticoduodenectomy was performed by a faculty surgeon A. FrozenPathology findings were discussed at multidisciplinary tumor board and with the patient. Given that there was only a single small (<2 mm) focus of ITPN without high-grade dysplasia the decision was made to continue close monitoring. Surveillance CT scan three months following his operation demonstrated a normal remnant pancreas. Eight months after his index operation, however, an MRCP was performed that demonstrated multicystic appearance throughout the gland with a dominant pancreatic body cystic lesion measuring 2.6 cm with enhancing mural nodularity, felt to represent either pancreatitis with developing pseudocysts or possibly evolving malignancy A. Given Three months later, he underwent repeat MRCP which demonstrated progression of the previously noted multicystic appearance of the remnant pancreas with diffusion restriction on delayed imaging B. Given Fifteen months after his index operation the patient, was taken back to the operative room for a completion total pancreatectomy and splenectomy. He had an uncomplicated hospital course and was discharged home on post-operative day 6 when a stable insulin regimen was determined. The completion pancreatectomy and fragment of small intestine underwent pathologic examination. Opening of the small intestine demonstrated detached friable tumor within the lumen. Serial sectioning of the specimen demonstrated diffuse heterogeneity of the remnant gland with evident distension of residual duct spaces with tumor A. Histol3ITPN is a recently described rare entity that remains challenging to identify preoperatively . Here weDue to the limited number of cases, the diagnosis of ITPN can be challenging. Forty-five to 59 % of ITPNs are located in the pancreatic head which comprises the most common location ,3,5,7. AMotosugi et al. identified the 2-tone duct sign on cross-sectional imaging and EUS and the cork-of-wine bottle sign on cholangiopancreatography as associated with ITPN in a study of 10 biopsy proven cases . These sOn pathologic examination, the majority of ITPNs are predominantly solid or polypoid lesions within the duct system; however, tumors may appear cystic in nature that demonstrate intraluminal growth without intraluminal mucin secretion . MicroscGiven the challenging nature of determining an invasive component and the risk of malignant degeneration, ITPNs are treated with formal pancreatic resection. Consistent with patterns of tumor location, the most common resection performed is a pancreaticoduodenectomy followed by a distal pancreatectomy and total pancreatectomy ,3,5. In 4ITPN remains a rare entity, and as such the diagnostic approach and treatment recommendations are based on a limited number of cases. Resection of ITPN with an invasive component confers a more optimistic long-term prognosis when compared with pancreatic adenocarcinoma. Although future studies are necessary, based on our clinical experience, we would advocate for completion total pancreatectomy when ITPN with microinvasion is present at the resection margin, regardless of the degree of dysplasia that is present based on the rapid progression in this case.The authors report no declarations of interest.We have no funding sources to declare.This is an exempted study per the Lifespan Institutional Review Board.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.JTC: Study concept, data collection, writing the paper.MHC: Data collection, writing the paper.ALA: Data collection, writing the paper.EYW: Data collection, writing the paper.PNP: Study concept, writing the paper.REB: Study concept, data collection, writing the paper.Rachel E. Beard.Not commissioned, externally peer-reviewed.


A 55-year-old male presented to our hospital with shortness of breath and leg swelling. Imaging studies revealed deep vein thrombosis and bilateral pulmonary embolism. The patient was placed on anticoagulation. A palpable umbilical nodule consistent with the appearance of Sister Mary Joseph's nodule (SMJN) raised the possibility of an underlying gastrointestinal malignancy. The patient also had significant ascites and underwent paracentesis with cytology, upper gastrointestinal and lower gastrointestinal endoscopy with the biopsy. Gastric lesion histology revealed gastric adenocarcinoma with peritoneal and colonic metastases. The patient was started on chemotherapy with 5‐fluorouracil, leucovorin, oxaliplatin (FOLFOX) for disseminated gastric malignancy. SMJN is a rare cutaneous metastatic manifestation which needs to be considered as a differential diagnosis of an umbilical tumor for prompt diagnosis and initiation of treatment. Sister Mary Joseph's nodule (SMJN) is a rare and peculiar physical sign found in 1%-3% of patients with an intrabdominal or pelvic malignancy . It is aA 55-year-old African American male with a past medical history of hypertension presented to the emergency department at our hospital with a chief complaint of generalized weakness. He experienced an acute onset of shortness of breath that started a couple of days prior to his presentation. He was working in his back yard and suddenly became dizzy and felt like he could not catch his breath. He attributed this episode to an anxiety attack due to ongoing stress factors. He endorsed some level of stress due to a bout of persistent diarrhea he had been experiencing for one month. The diarrhea started off with a few soft bowel movements per day and increased to up to six or seven bowel movements daily. The bowel movements were watery, without blood or mucus. These episodes of diarrhea were associated with mild, dull abdominal aches, and progressive abdominal distension. He also reported a 40-pound weight loss in the past year which he attributed to dietary changes that he had made for his gastroesophageal reflux disease (GERD). He also endorsed a decreased appetite for four weeks. In addition, two weeks ago, he noticed swelling in his right calf and thigh, followed by the swelling of his left leg.He was hypotensive on arrival at the emergency department with low blood pressure (BP) and a systolic blood pressure (SBP) in the 80s and diastolic blood pressure (DBP) in the 50s (mmHg) and tachycardic with a heart rate of 110 beats per minute (bpm). He was alert and oriented times four. Pertinent physical examination findings were of lower extremity swelling up to the mid-thigh level (pitting). Distended, non-tender abdomen with fluid thrills were suggestive of ascites. Further abdominal exam revealed a 5 x 4 cm palpable umbilical nodule which was purplish, non-tender and suggestive of possible SMJN  was 7.5 putting him at high risk of PE. He had computed tomography angiography (CTA) of the chest, Doppler ultrasound scan of the bilateral lower extremities (venous) and an urgent 2D transthoracic echocardiography (2D TTE) due to his hemodynamic instability. Upon further history taking, he denied any recent travel, trauma, surgery, and any family history of thrombophilia or other blood disorders. Our initial lab work revealed white blood cells (WBC) of 17.000, haemoglobin (Hgb) of 12.5 mg/dL, normal electrolytes except for hypokalemia with a potassium level of 2.8 mEq/L. His troponin-I was high with an initial value of 0.166 ng/mL, and D-dimer level was 7 mg/L. His prothrombin time (PT) and partial thromboplastin time (PTT) were normal. His lactate dehydrogenase (LDH) was elevated to 215. CTA of the chest revealed bilateral lobar, segmental and sub segmental pulmonary thromboembolism (PTE) some of which were occlusive, and numerous others were predominantly non-occlusive with patent pulmonary artery. There was straightening of the interventricular septum and bowing into the left ventricle which suggested a pattern of right heart strain. Further extension of the computed tomography (CT) to the abdomen and pelvis revealed large volume ascites, diffuse thickening of the stomach, small bowel, and colon likely secondary to edema. Gastroesophageal varices were also seen Figure . PulmonaOn further evaluation, the patient remained asymptomatic. His oxygen saturation remained normal. He was hemodynamically stabilized with volume resuscitation. Initially, thrombolytic option was considered, but was ultimately deferred since he had esophageal varices on CT in addition to a positive fecal occult blood test. He was started on high-dose heparin (weight based protocol). Doppler ultrasound scan of bilateral extremities revealed occlusive deep vein thrombosis in his left superficial femoral vein, popliteal, and posterior tibial veins. Once stabilized, further work up for the primary source was initiated. A diagnostic paracentesis was performed due to his large volume ascites with drainage of three liters of yellow. Fluid was sent to pathology for cytology and immunohistochemical tests. Peritoneal fluid revealed presence of malignant cells compatible with peritoneal involvement of gastric adenocarcinoma. He underwent colonoscopy and an upper endoscopy with biopsies. Biopsies of the stomach showed invasive poorly differentiated adenocarcinoma, diffuse type, with associated ulceration; immune histochemical stain confirmed epithelial origin of infiltrating neoplastic cells Figures -5. The cAbnormal umbilical pathologies are rare and can be classified as benign and malignant. Benign conditions are umbilical hernia, granuloma, abscess, eczema, and mycosis. Malignant tumors can be either primary umbilical origin or metastatic . On physWhen an umbilical nodule is found, it is necessary to make an accurate histological diagnosis between the primary and metastatic lesion and the easiest method is to do a fine needle aspiration cytology and core biopsy. This is a feasible and inexpensive diagnostic tool . Biopsy The identification of cutaneous metastases heralds a poor prognosis . Upon inOur patient presented with shortness of breath and leg swelling. Imaging studies revealed deep vein thrombosis and bilateral pulmonary embolism. A palpable umbilical nodule consistent with the appearance of SMJN raised the possibility of an underlying gastrointestinal malignancy. Endoscopic findings revealed gastric adenocarcinoma with colonic metastases. The patient was started on chemotherapy with FOLFOX for disseminated gastric malignancy. The presence of SMJN is a rare and a poor prognostic sign of a disseminated malignancy. Presentation of comorbidities with visceral advanced malignancy and pulmonary thromboembolism indicates poor prognosis; however, a timely diagnosis and urge of initiation of treatment will facilitate treatment for improved quality of life.


Although capillary hemangiomas, common lesions involving the proliferation of small capillary vessels and a single layer of endothelial cells, can arise in any organ, they are rarely reported in the greater or lesser omentum. Here in, we report a case of capillary hemangioma arising from the lesser omentum in an adult with interesting diagnostic imaging findings, including changes in tumor size over time on computed tomography (CT), that was resected using laparoscopic surgery. To our knowledge, this is the first English report to describe a capillary hemangioma arising from the lesser omentum.A 63-year-old Japanese man received hemodialysis for chronic renal failure due to diabetic nephropathy, and a small, gradually enlarging tissue mass was found near the lesser curvature of the stomach on plain CT performed annually, without any associated complaints. Diagnostic imaging revealed an 18 × 15-mm tumor with a homogenous, highly enhanced effect in the early phase that was attenuated but prolonged in the delayed phase. Magnetic resonance imaging showed a mass with low signal intensity on T1-weighted imaging and relatively high signal intensity on T2-weighted imaging.The patient was diagnosed with capillary hemangioma arising from the lesser omentum according to the pathological and immunohistological findings.The patient underwent laparoscopy for excision of the tumor from the lesser omentum.At the 1 year follow-up, the patient had no recurrence of the tumor.We describe the first case worldwide of capillary hemangioma that was a true vascular tumor arising from the lesser omentum. Although capillary hemangioma arising from the lesser omentum is extremely rare, it should be considered in the differential diagnosis of patients presenting with a highly enhanced lesser omental tumor, and laparoscopy can be safely applied for the excision of this tumor. These anomalies have been reviewed from such viewpoints as constitution, natural evolution and treatment, and attempts have been made to stratify them into lesions with a proliferative component  and relatively static lesions . Despite long-standing efforts to achieve a uniform classification, the nomenclature of vascular anomalies continues to be confusing. The key features of capillary hemangiomas remain under debate. The pathological examination of capillary hemangiomas reveals the proliferation of capillary-sized vessels and a single layer of endothelial cells. These tumors can be found in any organ, although a higher prevalence in the skin and subcutaneous tissue has been reported. They are very rare in the gastrointestinal tract–5 and even rarer in the mesentery or omentum.–12 To our knowledge, the English-language reports of hemangiomas that occurred in the greater or lesser omentum to date include only 6 cases.–12 The 5 vascular tumors arising from the greater omentum consisted of 2 cases of capillary hemangioma, 1 hemangiopericytoma, 1 epithelioid hemangioendothelioma and 1 malignant hemangioendothelioma. The 1 case of a lesser omental tumor was a cavernous hemangioma. The 2 reported cases of capillary hemangioma were in infants.Until recently, hemangiomas were thought to be due to hamartoma lesions originating from the embryonic sequestration of mesodermal tissue in any organ.The purpose of this article was to highlight the diagnostic imaging, including the characteristic features on computed tomography (CT), the chronological changes in tumor size, and the histopathological and immunohistochemical findings of a small capillary hemangioma arising from the lesser omentum used to determine whether it was a remnant tumor of an infantile hemangioma or a newly developed tumor. To our knowledge, this report is the first to describe a capillary hemangioma arising from the lesser omentum.2123I metaiodobenzylguanidine (123I-MIBG ) scintigraphy at the University of Fukui Hospital, but abnormal accumulation of the isotope was not detected. He was referred to our department for surgical resection of the enlarging tumor and a definitive diagnosis. The patient had a medical history of diabetic nephropathy and chronic renal failure. On physical examination, the patient had a height of 170 cm, a body weight of 61.5 kg, a pulse of 65 beats/min, a blood pressure of 117/77 mm Hg, and a body temperature of 36.8°C. Anemia, jaundice, edema, and malnutrition were not found. No abnormalities were detected in the skin or subcutaneous tissue. Superficial lymph nodes were not detected on palpation. His abdomen was flat and soft, and no mass was detected on palpation. Routine laboratory tests on the day after dialysis showed leukopenia , anemia  and renal dysfunction . The plasma levels of vanillylmandelic acid  and homovanillic acid  were elevated. Considering the imaging results and the chronic enlargement of the tissue mass, we made a preoperative diagnosis of paraganglioma in the lesser omentum as the most likely diagnosis even though we were not able to reach a definitive diagnosis. The patient underwent laparoscopic surgery in February 2018  showed a contrast density that was similar to that in the aorta during the arterial phase, as observed in the present case. Kim et al found that small hemangiomas (<2 cm) had marked enhancement in the early phase on enhanced CT, but this effect dissipated in the delayed phase. Findings similar to those described by Kim et al were observed in our case. In contrast to tumors in the lesser omentum, a paraganglioma shows a well-demarcated, solid, cystic, heterogeneously enhanced mass on enhanced CT.,20 However, no CT features unique to paraganglioma have been found, despite the tendency for smaller tumors to be more homogenous and have more well-defined margins than larger tumors. The laboratory tests showed elevated plasma levels of VMA and HVA despite the final diagnosis of capillary hemangioma. Iwamoto et al showed that the plasma VMA level was elevated in patients on hemodialysis both with and without pheochromocytoma. In patients on hemodialysis, it seems that the plasma level of catecholamine metabolites is not appropriate as an index for the diagnosis of paraganglioma. Although 123I-MIBG scintigraphy in our case did not show isotope accumulation, close attention should be paid to 123I-MIBG-negative cases, as this result is found rather frequently in cases of metastatic pheochromocytoma.The reported characteristic findings of a hemangioma on dynamic enhanced CT include peripheral enhancement during the dynamic bolus phase and complete isoattenuated fill-in during the delayed phase for up to 60 minutes after the administration of contrast medium.,12 Characteristic T2WI findings would be useful for discriminating between capillary hemangiomas and other venous anomalies, and the findings in our case are consistent with a capillary hemangioma. Pathologically, hemangiomas have previously been classified according to channel size into 3 types, namely, cavernous, capillary, or mixed, and the cavernous type is the most common. Hemangiomas can be classified by different parameters:Although the MRI findings in our case, such as low SI on T1WI and relatively high SI on T2WI, did not allow a specific diagnosis to be made, it has been reported that venous malformations typically show uniformly high SI on T2WI, whereas capillary hemangiomas tend to be less intense because of shrinkage due to different degrees of fibrosis and hemosiderin deposition.(1)Vessel type ;(2)Location ;(3)Characteristic cell type ;(4)Neoplastic status ; and(5)Patient age . Unlike the 2 previously reported cases of capillary hemangioma in the omentum, our case occurred in adulthood. Furthermore, a gradual increase in size occurred over 4 years, and there was negative immunostaining for GLUT-1, which is a downstream target of hypoxia-inducible factor 1 alpha. There was also negative immunohistochemical reactivity for VEGF-A and IGF-2, which are sensitive markers for the diagnosis of infantile hemangioma. These findings suggest that the tumor was not a residual anomaly of a congenital hemangioma but rather that it developed in adulthood. The MIB-1 index of the tumor suggests that it was a neoplastic lesion.Capillary hemangiomas are commonly considered congenital, true vascular tumors that usually tend to involute during childhood, and only a minority persist throughout adulthood.Surgical excision should be considered for the treatment of capillary hemangiomas in the lesser omentum, and pathological examination of the resected tissue can be used to obtain a definitive diagnosis. Furthermore, laparoscopic surgery seems to be effective for the excision of hemangiomas in the lesser omentum following thorough preoperative evaluations of the diagnostic images to assess the tumor size, anatomical location, extent of invasion into the gastric wall and laparoscopic surgical field, as this technique has the advantages of minimal surgical invasiveness, reduced wound healing time, reduced postoperative pain, reduced blood loss, reduced wound-related complications and improved cosmetic outcomes.4We report an extremely rare case of capillary hemangioma arising from the lesser omentum in adulthood. Although capillary hemangioma of the lesser omentum is an extremely rare disease, it should be considered in the differential diagnosis of patients presenting with a highly enhanced lesser omental tumor, and laparoscopic surgery is a good treatment option for excision of this tumor.Conceptualization: Hideki NaganoData curation: Hideki Nagano, Seiichi Taguchi, Takayoshi TsubakiInvestigation: Seiichi Taguchi, Takayoshi Tsubaki, Toshikuni Tsuchiyama, Hidemasa Uematsu, Sakon NorikiMethodology: Hideki NaganoProject administration: Hideki Nagano, Seiichi TaguchiSupervision: Takanori GoiData curation: Hidemasa Uematsu, Sakon Noriki.Supervision: Takanori Goi, Seiichi Taguchi, Takayoshi Tsubaki, Toshikuni Tsuchiyama, Hidemasa Uematsu, Sakon Noriki.Validation: Takanori Goi, Sakon Noriki.Writing – original draft: Hideki Nagano.Hideki Nagano orcid: 0000-0003-0524-2247.


Bile duct cysts are congenital malformations characterized by bile duct dilatation with intra and/or extrahepatic localization. About 80% of cases are diagnosed in childhood, so their presentation in adults is rare and repeatedly associated with complications.A 38-year-old female patient complained of severe abdominal pain for one month, with no associated cholestatic signs. She underwent abdominal ultrasonography that showed cholelithiasis with extrahepatic bile duct dilatation and underwent cholangiography (MRI), which revealed fusiform dilatation of the proximal portion of the hepatocholedochal, compatible with choledochal cyst.In adult patients, clinical manifestations are usually nonspecific, and the diagnosis is established by medical imaging. Magnetic resonance cholangiopancreatography (MRCP) is the gold standard diagnostic today. Treatment with better prognosis consists of total cyst resection with hepaticojejunal anastomosis and intestinal transit reconstruction with Roux-en-Y.Bile duct cysts are a rare condition in adults. Currently, increasing access to more accurate medical imaging enables early diagnosis, contributing to the use of therapeutic methods in appropriate time, avoiding development of complications. There are incidences of 1:100.000 to 150.000 live births in the West and 1:1.000 in Asia, being most prevailing in female at a ration from 3 to 4:1 . Around 2A 38-year-old female patient from Parintins, Manaus, Amazonas presented with the chief complaint of “belly pain”. She reported that it began a month ago with mild to severe epigastric pain of the colic type, which radiated to the right hypochondrium and back, beginning after feeding, which persisted even after home analgesia. She denies fever or chills, choluria, jaundice, fecal alcoholism, or recent weight loss. Physical examination was without semiological changes. She underwent abdominal US that revealed cholelithiasis with extrahepatic bile duct dilatation and thus requested MRI and referred to the general surgery outpatient clinic. She denied any other complaints, such as comorbidities, medication use, allergies or previous surgery.Imaging (MRI) revealed normal-walled gallbladder with multiple thin septations within it, notably in the body and fundus, as well as fusiform dilation of the proximal portion of the hepatocholedocal duct, measuring up to 2.5 cm, compatible with cyst of Todani type I choledococcus .Fig. 1MRAfter two months, the patient underwent cholecystectomy and complete surgical resection of the extrahepatic bile duct cyst segment to about 1 cm below the bifurcation of the hepatic ducts, followed by Roux-en-Y end-lateral hepatic jejunostomy . The pos3Reports of bile duct cysts have been gone up in recent years, with more than half of the cases occurring in Japan. A report from Finland estimates that the incidence of bile duct cysts has been increased from 1:128.000 to 1:38.000 for the past 40 years; and although most cases were reported in children in the past, some studies have registered similar data in adults and children . DespiteRegarding classification, Alonso-Lej and colleagues elaborated the first system in 1959, which encompassed 4 types of biliary cysts (type I to IV) . In 2003Concerning the clinical presentation, there is no consensus in the literature about the most common one. Some authors affirm that jaundice is the most observed symptom , whereasThe diagnosis of bile duct cysts is defined by the identification of bile duct dilation after exclusion from other etiologies  . This coThe standard treatment from bile duct cysts is surgical with specific approaches for each type according to its classification. Those classified as type I, II or IV are generally undergone resection surgery due to the risk of neoplastic development. The types I and V are completely resected with cholecystectomy completion and biliary reconstruction by Roux-en-Y hepaticojejunostomy, being required a partial hepatectomy on IVA type ,11. The 4Bile duct cysts are rare pathology in adults, happening to be more common in the pediatric age group, especially in Asian population. Currently, improvements in health care and the increase in accessing more accurate medical imaging enable early diagnosis, contributing to use of therapeutic methods in appropriate time, avoiding development complication. The classic triad signs and symptoms are rarely present, as such vital the awareness of the disease to include it in diagnostic hypothesis and make use of optimal screening tests with a favourable cost-benefit, hence ultrasound to be the most used in the initial approach. The main treatment is consisted in surgical resection from cyst(s) when feasible, due to the risk of future malignancy.We do not have any funding source, this manuscript is just a case report, not a research.As the manuscript is not a research study, we only have the patient consent for writing and others forms of publication. Also, the ethical approval for this case report has been exempted by our institution.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Zuriel Seixas and Ketlen Sousa made contributions to conception and design. collected the patient details and wrote the paper. Frank Macedo made contributions to patient management. Victor Albuquerque and Rubem Silva Junior critically revised the article. All authors read and approved the final manuscript.The manuscript is a case report, not considered a formal research involving participants.Dr. Victor Albuquerque.None.Editorially reviewed, not externally peer-reviewed.The authors declare that there are no conflicts of interest.


The diagnosis of craniofacial osteosarcoma can be quite challenging, and the condition often goes unrecognized for a considerable period of time. In this report, we discuss the case of a 21-year old woman who presented with a one-year history of a small swelling over the left maxillary alveolar ridge. Upon further investigation, the histopathological examination showed high-grade chondroblastic osteosarcoma. The option of four cycles of neoadjuvant chemotherapy regimen preoperatively was chosen, and left inferior maxillectomy was performed along with reconstruction with obturator prosthesis. This case highlights the difficulties encountered in such rare cases of craniofacial osteosarcomas both in terms of the delay in the establishment of the diagnosis as well as management protocol. A high index of suspicion is required in cases of craniofacial osteosarcoma and early surgical resection with adequate safety margins is warranted. Osteosarcoma, also called osteogenic sarcoma, is a rare, aggressive, and malignant form of tumor, and it is considered to be the most common primary malignant bone tumor . OsteosaA tumor is defined as craniofacial if it occurs in the jaw, maxilla, or other unspecified regions . CraniofThe modern treatment protocol for osteosarcoma is multimodal, consisting of preoperative chemotherapy followed by extensive surgery and postoperative chemotherapy . With reOur objective in preparing this case report was to highlight the diagnostic difficulties encountered in a patient with osteosarcoma that presented as small mass over the left maxillary alveolar ridge.A 21-year-old female, a Saudi student, presented to the Otolaryngology Department as an outpatient complaining of a left upper maxillary alveolar progressive recurrent mass of six months. The mass had started as a small swelling over the left maxillary alveolar ridge where excision had been done accordingly in the dental office. One month later, the mass had reappeared with rapid growth and had never regressed until a facial bulge was visible. On further history, the patient did not mention any problems with swallowing or respiration, and her past medical history was insignificant with no chronic illness and no prior history of exposure to radiation or chemotherapy and no history of benign bone conditions. Regarding her family history, there was no genetic predisposition or other malignancies. Furthermore, surgical and dental history was insignificant with no pharmacological medications and no history of allergies or smoking.On physical examination, an irregular mass on the left anterior maxillary alveolar ridge measuring around 2 x 2 cm Figure  with a mThe recurrence of the mass warranted further investigation as an initial differential of osteomyelitis or other mixed lesions. After obtaining the patient’s written consent, an incisional biopsy was performed under local anaesthesia where the histopathology showed high-grade (G3), chondroblastic osteosarcoma Figure .When we discussed the surgical options and treatment with the patient, she was reluctant to follow the treatment protocol and chose to seek a second opinion. After a few months, she presented with a slight increase in mass size (3 x 2 cm).A CT scan of the neck done on the same day showed a maxillary lesion with small growth 3 mm). The lesion was seen in the alveolar process of the left maxilla and appeared to be mostly lytic with no material or sunburst changes, while the lesion itself was seen between the left incisor and the left canine; meanwhile, the left lateral incisor was not seen  Figure  and negaOsteosarcomas are a heterogeneous group of malignancies characterized by the production of osteoid or neoplastic ossification . OsteosaRegarding surgical resection, due to the complex anatomy of the maxillofacial region, getting negative margins in surgical resection as well as reconstruction is quite challenging . MoreoveThere are various histopathological classifications, and the subtypes are classified according to the predominant matrix produced by the tumor: osteoblastic (predominantly bone matrix), chondroblastic (predominantly cartilaginous matrix), or minimal matrix in fibroblastic . SpecialThe diagnosis of craniofacial osteosarcoma is quite challenging and needs a high index of suspicion. The mainstay of treatment in osteosarcoma cases is radical surgical resection with obtaining negative margins alongside chemotherapy, which increases early survival rates considerably, with the multimodal therapy being the most effective in increasing long-term survival rates.


Heterotopia is the presence of normal physiologic tissue in an atypical location. Gastric heterotopia has been described in various locations throughout the gastrointestinal tract, including the small intestine. Gastric heterotopia of the small intestine typically is asymptomatic but may present in several ways with symptoms of obstruction, bleeding, perforation, intussusception, or pain. However, gastric heterotopia is rare beyond the duodenum except for its frequent association with Meckel's diverticulum. This entity should be considered in the differential diagnosis of polypoid lesions presenting with symptoms of bleeding or obstruction especially in younger patients. We present a case of gastric heterotopia of the jejunum in a patient with a prior history of Meckel's diverticulectomy after he presented with obstructive symptoms. His symptoms improved following resection of two jejunal polyps via antegrade double-balloon assisted enteroscopy with fluoroscopy. On histopathlogical examination, findings were consistent with gastric heterotopia. This case highlights the importance of considering gastric heterotopia in the differential diagnosis of polypoid lesions located beyond the ligament of Treitz in younger patients presenting with obstructive symptoms. Heterotopia is the presence of mature physiologic tissue in an atypical location. Gastric heterotopia has been described in several locations throughout the gastrointestinal tract such as the esophagus, duodenum, gallbladder, Meckel's diverticulum, and other areas within the small bowel and rectum. Gastric heterotopia of the small intestine can be asymptomatic or present in various ways with symptoms of obstruction, ulceration or bleeding, perforation, intussusception, or pain . GastricA 33-year-old gentleman presented with lower abdominal pain, occasional nausea, emesis, and inability to pass stool or flatus. He did not have any fever or chills. His past medical history was notable for Meckel's diverticulectomy secondary to gastrointestinal bleeding which ultimately required ileal resection and stapled anastomosis ten years previously. The pathology following operative intervention did not demonstrate any ectopic gastric mucosa. He did not have any additional pertinent past medical, family, or social history.His vital signs were within normal limits. Physical examination was notable for a minimally distended abdomen with tympany on percussion. Laboratory investigations were unremarkable. Coronal and axial computed tomography enterography of the abdomen and pelvis with contrast  demonstrHeterotopia is defined as the presence of normal physiologic tissue in an anatomic location where it is not normally found. Gastric heterotopia is not an uncommon lesion and can be found in several areas throughout the gastrointestinal tract. However, gastric heterotopia is rare beyond the duodenum except for its frequent association with Meckel's diverticulum . GastricIt is important to differentiate gastric heterotopia from gastric metaplasia. Gastric metaplasia is an acquired lesion and is usually seen in association with chronic inflammatory conditions such as inflammatory bowel disease. Unlike heterotopia which is a macroscopic lesion seen on radiographic imaging or endoscopy, ultimately requiring confirmation by biopsy, metaplasia is a microscopic lesion .The typical treatment for gastric heterotopia includes endoscopic or surgical resection to prevent complications. This case is unique in that the patient had two foci of gastric tissue outside of the stomach with a prior history of Meckel's diverticulectomy and prior pathology at the time of small bowel resection without any evidence of ectopic gastric mucosa.It is important to recognize the variety of presentations associated with gastric heterotopia. A timely diagnosis of an enlarged polyp can prevent complications such as obstruction, intussusception, bleeding, or perforation. The combination of clinical presentation, radiologic imaging, and endoscopic and pathologic evaluation is helpful in making the diagnosis .In most reported cases of gastric heterotopia involving the jejunum , patientGastric heterotopia can present in various ways including masslike lesions with symptoms of obstruction, pain, or bleeding, or may even remain asymptomatic. Gastric heterotopia beyond the ligament of Treitz is a rare entity. This case highlights the importance of considering gastric heterotopia in the differential diagnosis of polypoid lesions located beyond the ligament of Treitz in younger patients presenting with obstructive symptoms .


Pancreatic ductal adenocarcinoma (PDAC) is among the most aggressive malignancies. The prognosis for recurrence after surgery is extremely unfavorable, and liver metastasis of PDAC confers poor prognosis despite resection.A 51-year-old man was admitted to our hospital for further examination and treatment, including surgery for a pancreatic tumor. On close inspection, he was suspected to have pancreatic head cancer without enlarged lymph nodes or distant metastasis, and pancreatoduodenectomy with D2 lymph node dissection was performed. A postoperative pathological examination revealed well-differentiated invasive ductal adenocarcinoma with lymph node metastasis . Postoperatively, he received adjuvant chemotherapy containing gemcitabine for 1 year. Eight years after the radical surgery, his serum carbohydrate antigen 19-9 level was elevated, and computed tomography (CT) and magnetic resonance imaging revealed a well-circumscribed 10-mm mass in liver segment 5. Positron emission tomography/CT also revealed high fluorine-18-fluorodeoxyglucose uptake only in this hepatic tumor. Accordingly, the patient was diagnosed with a solitary liver metastasis of PDAC. As the liver metastasis was isolated and identified long after the initial surgery, we decided to resect it using laparoscopic partial hepatectomy of segment 5. Histopathological examination confirmed liver metastasis of PDAC and the patient received adjuvant chemotherapy containing S-1. No evidence of recurrence has been seen for 11 years since the pancreatoduodenectomy and 3 years since the hepatic resection.Cases of metachronous liver metastasis of PDAC after radical surgery, in which patients exhibit long-term survival without recurrence after hepatectomy, are extremely rare. Hepatectomy may confer long-term survival, and the time to postoperative recurrence and the number of liver metastases may be useful criteria for deciding whether to perform hepatic resection. Pancreatic ductal adenocarcinoma (PDAC) is among the most aggressive malignancies. Despite recent progress in diagnostic imaging modalities, surgical procedures, and chemotherapy, its prognosis remains poor , 2. Here2) on days 1, 8, and 15 every 4 weeks for 1 year. Thereafter, the carbohydrate antigen 19-9 (CA19-9) level was within the normal range and a follow-up CT revealed no local recurrence or distant metastasis. However, at 8 years after the first surgery, the serum CA19-9 level was elevated (130.3 U/mL). Additionally, CT identified a 10-mm low-density area in liver segment 5  and computed tomography (CT). He had no history of malignancy, and physical and laboratory examinations, including tumor markers, revealed no specific findings. Contrast-enhanced CT and gadolinium-ethoxybenzyl-diethylenetriamine pantaacetic acid (Gd-EOB-DPTA)–enhanced magnetic resonance imaging (MRI) revealed a nodule that was gradually contrasted in the pancreatic head Fig. . A postot 5 Fig. a, b witht 5 Fig. . Based op = 0.038). Furthermore, in a retrospective analysis, overall survival was significantly prolonged in patients with resected liver metastasis of PDAC  [The prognosis of postoperative recurrence of PDAC is extremely poor . A recen< 0.001) . These f< 0.001) . TherefoPDAC involves the early capability of cells to migrate to distant organs, and many patients have metastatic disease at the time of PDAC diagnosis . For thiHere, we reported an extremely rare case of metachronous liver metastasis of PDAC after radical surgery in a patient who survived without a recurrence for 11 years after PD and 3 years after hepatectomy. The present findings suggested that hepatectomy may confer long-term survival in selected patients. The time to postoperative recurrence and the number of liver metastases may be useful criteria for deciding whether to perform hepatic resection.


Anaplastic ganglioglioma (AGG) is a rare and aggressive counterpart of the more benign and frequently encountered glioma. Herein, we present a 21-year-old female who presented with episodes of total amnesia and complex partial seizures, which led to the diagnosis of AGG localized to the medial temporal lobe. She subsequently underwent surgical cytoreduction of the tumor three times with adjuvant chemoradiotherapy. The extent of resection throughout the surgeries was hindered by the extension of the tumor to critical neurovascular structures; during the last surgery, invasion into the pons was noted, which posed a significant clinical challenge. Gangliogliomas are uncommon mixed neuronal-glial tumors, representing 1.3% of brain tumors and 0.4% of central nervous system neoplasms . In rareWe present a case report of a young female with a recurrent primary anaplastic intracranial ganglioglioma, which was repeatedly treated with surgical cytoreduction and adjuvant therapy. As neurosurgeons are unlikely to see more than just a few cases during their practice, we hope to detail what type of outcome trajectory can be expected in the circumstances when gross-total resection (GTR) of an AGG is not feasible. We further conducted a literature review to describe patient characteristics and survival outcomes in patients with AGG and brainstem involvement.A 21-year-old female patient was hospitalized in our neurosurgical department with episodes of total amnesia and complex partial seizures, lasting two to three minutes and occurring as often as seven times per day. Throughout the seizures, the patient didn’t lose consciousness and the onset of symptoms occurred gradually over two months. Further investigations revealed an expansive mass in the right medial temporal lobe - resection was attempted, but somewhat hindered due to adhesion of the superior pole of the tumor to the branches of the posterior cerebral artery (PCA). Histopathologic analysis of the resected specimen demonstrated dysplastic glial proliferation, with abnormal appearing ganglion cells  gangliogliomas .A central theme of high-grade gangliogliomas is their tendency to recur. In a large retrospective series of 184 supratentorial gangliogliomas, the recurrence rate of first, second, and third-grade gangliogliomas was 1%, 18%, and 50%, respectively . TreatmeDue to the rarity of AGG, evaluation of the Surveillance, Epidemiology, and End Results (SEER) cancer registry has been invaluable in detailing epidemiology and prognostic factors. In a large series of 58 tumors and a median follow-up period of 52 months, Selvanathan et al. found the median OS and five-year survival rates for AGG to be 28.5 months and 63%, respectively [The French Brain Tumor Database study by Terrier et al. attempted to further understand the biological behavior of AGG by providing more information regarding imaging characteristics, treatment strategy, and recurrence statistics . A totalTo further explore AGG brainstem involvement, a search of the PubMed and Scopus databases was done using the keywords “anaplastic ganglioglioma,” “brainstem,” “mesencephalon,” “pons,” “medulla,” and “midbrain” to identify relevant case reports. The keywords were identified as either Medical Subject Heading (MeSH) terms or within the title/abstract. A total of 29 studies were found, 22 studies were excluded due to being unrelated, resulting in six case reports and one study being included in this review. The results are presented in Table 2) and radiotherapy, ranging from 30-60 Gy. In our patient, bevacizumab was added, as it was shown to have some activity in patients with recurrent high-grade gliomas. Of eight patients reviewed, only one case was amenable to GTR. The risk of tumor recurrence or poor response to adjuvant therapy, expectedly, remains a crucial contributing factor that makes survival past eight months recherché.Of the eight patients reviewed, the average age was 27.8 years (range 9-64), with five patients being female and three male. The clinical presentation included classic symptoms of intracranial neoplasms; however, symptom duration was strikingly variable, ranging from two years to several days. In five cases, brainstem involvement was noted on the right side, two left-sided cases, and one case with bilateral brainstem invasion. The average dimensions of the tumor were 46.0x41.6x50.7 mm, although it must be noted that only three cases reported tumor size and from this small sample size, it is difficult to identify if AGG has a predilection for a certain region of the brainstem. In one case, the authors noted atypical neurocytoma to act as a tumor precursor to the AGG . The staThere are currently no studies in the literature that examine the efficacy of mitogen-activating protein kinase pathway (MAPK) inhibitors (or their downstream counterparts: B-Raf and mitogen-activated protein kinase). MAPK inhibitors represent a promising systemic treatment modality for AGG and, more recently, case reports began to emerge detailing their potential utility as an adjuvant -19. The Subtotal resection of AGG portends a poor relapse-free survival. The propensity for the recurrence of AGG must be carefully taken into consideration if surgical cytoreduction of the tumor will be undertaken. In rare cases, AGG may extend into the brainstem or other critical neurovascular structures, thus precluding GTR. In spite of limited data from the literature, attempting maximal safe resection followed by vigilant surveillance from disease progression is an accetable therapeutic strategy. The survival benefit posed by adjuvant chemoradiotherapy for patients who underwent subtotal resection is unclear, despite being widely used.


Metastatic germ cell cancer of the testis is characterized by favorable prognosis since effective treatment methods are available even in cases of extensive disease. Retroperitoneal masses frequently encroach major blood vessels requiring a vascular intervention usually performed in association with the post-chemotherapy retroperitoneal lymph node dissection (RPLND). Reported clinical case describes a successful pre-treatment endovascular surgery for abdominal aortic rupture allowing for full-dose systemic chemotherapy administration, and subsequent radical surgical intervention at primary tumor site as well as metastatic retroperitoneal lymph node dissection including the reconstruction of inferior caval vein.Patient presented with left-sided testicular tumor and voluminous retroperitoneal mass with vascular involvement. Soon after the patient had been admitted for the first cycle of cisplatin-based chemotherapy, computed tomographic angiography (CTA) revealed a dorsal aortic wall rupture with active extravasation and irregular pseudoaneurysmatic dilatation of the aorta below the leak area. Retroperitoneal intratumoral hemorrhage associated with the bilateral iliac venous thrombosis required an endovascular repair procedure of infrarenal abdominal aorta.Following the successful endovascular aortic repair 3 cycles of BEP  regimen were administered with subsequent delayed left radical orchiectomy and RPLND associated with vena cava inferior (VCI) resection. Reconstruction of VCI was originally not deemed necessary as collateral blood flow appeared sufficient, however, intraoperative complications resulted in the need for unilateral VCI reconstruction, using the interposed bypass between right common iliac vein and infrarenal segment of VCI. Histopathologic examination of the attained specimen detected no vital cancer structures. The patient remains disease-free 18 months after the RPLND. Testicular germ cell tumors often present with advanced stages including extensive retroperitoneal disease which might encroach the vital vasculature. Cisplatin-based chemotherapy combined with radical surgery provides excellent disease control  while inVascular reconstructive surgery is often a part of planned surgical intervention in the management of residual masses in the post-chemotherapy period , 4. The Herein we present a patient with newly diagnosed lefd-sided testicular tumor, bulky retroperitoneal tumor involving the major blood vessels along with the aortic wall rupture and active leakage what urged a vascular intervention prior to systemic therapy. of Endurant abdominal stent-grafts were implanted via right common femoral artery and the intervention was performed by specialists at the National Institute of Cardiovascular Diseases in Bratislava. Suprarenal anchoring with not covered stent-like part of abdominal endograft  was used, as standard approach for better fixation and sealing, prevention of endoleaks, shifting, or late migration, in case of tubular or bifurcation stentgraft fixation to infrarenal part of abdominal aorta. Dual antithrombotic therapy  was initiated immediately after stentgraft insertion. Postoperative course was uneventful and patient’s performance status allowed for administration of 1. cycle of BEP regimen, 6 days after the endovascular repair surgery.A 48-year-old patient with personal history of arterial hypertension and toxic hepatopathy was admitted to the National Cancer Institute in Bratislava, presenting with a newly diagnosed left-sided testicular tumor and a 215 × 145 × 170 mm  (CCxLLxAP) heterogeneous retroperitoneal mass, compressing the inferior caval vein and left ureter, with bilateral iliac vein thrombosis. The patient started anticoagulation therapy with low molecular weight heparin . Initial tumor markers were: beta-choriogonadotropin 46.8 µg/l, alfa-fetoprotein 3.1 U/ml, lactate dehydrogenase (LD) 76.8 µkat/l. Systemic therapy with bleomycin, etoposide and cisplatin (BEP) was commenced (day 1) when the patient’s complaint about acute abdominal pain urged the radiographic re-examination. CTA scan detected mass enlargement in the retroperitoneum, suspecting a rupture of dorsal aortic wall 38 mm above the inferior mesenteric artery origin, with active extravasation and irregular pseudoaneurysmatic dilatation of the aorta below the leak area  and circulatory collapse. CTA scan refuted the suspicion of intratumoral bleeding and further examination including the upper gastro-intestinal tract (GIT) endoscopy identified the source of bleeding – 2 duodenal ulcers . Hemostyptic, antiulcer and vasopressor agents were applied along with the hemo substitution and volume therapy, with positive effect. Within following weeks, two more cycles of BEP were administered until the re-staging CT examination was performed. The retroperitoneal tumor mass shrank to 140 × 110 × 125 mm (CCxLLxAP), still compressing the inferior caval vein (VCI), encasing the aorta and deviating nearby intestinal loops as well as the left kidney. Tumor markers were normalized. Subsequently, the patient underwent left inguinal radical orchiectomy and epididymectomy . Histologically no vital cancer cells were identified and all specimens consisted of completely necrotic testicular tumor tissue. The patient had been closely observed during a 6-month period after the radical surgery and repeatedly underwent the radiologic evaluation which reaffirmed the ongoing reduction of retroperitoneal formation to 120 × 80 mm. At this point, retroperitoneal lymph node dissection (RPLND) was suggested and performed, associated with the resection of VCI and the left pannephrectomy. Ten days before elective surgery, antithrombotic therapy was changed to low molecular weight heparin in cooperation with hematologists. Doppler ultrasound examination of deep vein system of lower extremities prior to RPLND showed good blood flow in femoral as well as external iliac veins, no signs of thrombosis were present. Bilateral iliac vein thrombosis diagnosed at first presentation of patient resolved completely, and total infrarenal occlusion of inferior caval vein was caused by tumor itself before RPLND. Collateral blood flow was sufficient and therefore, we planned to do no reconstruction after resection. During resection, anesthesiologist referred hypotension short time after clamping of infrarenal caval vein so peroperatively we made the decision to do reconstruction to improve central venous inflow. As there was severe fibrosis on the left side with multiple collaterals, we decided to do just unilateral reconstruction on the right side. Inferior caval vein was reconstructed by using ringed rifampicin-tinctured polytetrafluoroethylene graft (Gore-Tex)—a 10 mm prosthesis was placed between the right iliac vein and VCI. Postoperatively no lower limb edema was present with patent femoral veins and external iliac veins on both sides confirmed by Doppler ultrasound. During surgery heparin at dose 100 units per kilogram was administered, followed by continual intravenous infusion with aPTT levels between 2–3, then changed for full anticoagulant therapy with low molecular weight heparin on postoperative day 10 and 28 days after surgery, dual antithrombotic therapy was restarted.Surgery duration was 6.5 h, total blood loss estimates 1500 ml, early complications included cardiovascular instability, oliguria and fever. On postoperative day 2, bloody drain output urged an acute CTA scan revealing an extensive perisplenic hematoma, and as the patient became hemodynamically unstable, second look surgery was performed . While it failed to identify the origin of bleeding, further postoperative course was uneventful and led to full recovery. Once again, histopathological examination detected no viable cancer structures in the samples acquired from the RPLND, VCI resection and nephrectomy. To this date, 18 months post the final surgical procedure the patient remains disease-free, asymptomatic with maintained patency of the aortic stent graft Fig. .Fig. 3ReRepresenting the most frequent malignancy in young men (aged 15–34 years), testicular germ cell cancer patients are fortunate to have a good prognosis . Most ofA number of additional procedures may be required to reach the complete resection which is an inevitable condition to optimize the clinical outcome. Either anticipated or unexpected secondary surgeries involve mainly caval vein reconstructions , nephrectomy, splenectomy and thrombectomy. Pancreatic and hepatic resections, bowel resection with or without ostomy, thoracotomy and aortic reconstruction (repair) may also be undertaken as part of RPLND .Arterial reconstructions are usually performed by primary anastomosis, reinsertion, or synthetic prostheses, expanded polytetrafluoroethylene (ePTFE) or Dacron. Autologous vein is not routinely used for arterial repair. Aortic resection adjunctive to the post-chemotherapy RPLND is uncommon and is performed either for a nonrepairable injury of the vessel or on behalf of its inclusion in the tumor mass , 15. HerLocalized defects of inferior caval vein are usually repaired by venoplasty or venous (synthetic) patch while circular damage needs to be reconstructed by either a primary anastomosis, limited by low mobility of remaining ends, or more routinely performed ePTFE prostheses. Veins occluded by a thrombotic process are treated by ligation of their proximal and distal stumps , 15. In Tumor infiltration is a rare cause of aortic rupture. Besides the germ cell testicular cancer , abdominThe patient in this report presented with left-sided testicular tumor and bulky retroperitoneal disease compressing the inferior caval vein as well as the left ureter and was indicated for first line chemotherapy treatment when CTA scan revealed an active leak from dorsal aortic wall. Acute endovascular repair was performed in order to assure the feasibility of oncological treatment. Successful upfront endovascular aortic repair in our patient enabled the delivery of systemic oncologic cisplatin-based chemotherapy with good treatment response followed by radical orchiectomy in seronegative disease and subsequent RPLND along with the nephrectomy and caval vein reconstruction.Aortic infiltration and rupture secondary to germ cell testicular tumor is a rare condition. With respect to the literature, vascular intervention on aorta is rather a part of elective postchemotherapy RPLND. Timely recognition of active intratumoral hemorrhage in metastatic retroperitoneal disease of testicular origin owing to the abdominal aortic rupture enables an appropriate surgical intervention followed by adequate systemic oncologic treatment, and thereby dramatically improves patients’ outlook.


Calcifying fibrous tumor (CFT) is a rare, benign proliferation of fibroblasts and inflammatory cells that is non-invasive and usually arises in deep tissue structures. Due to its overall paucity, no accurate incidence has been reported yet. We understand this disease via a handful of case studies published in the medical literature, first of them being from 1988. Earlier known as 'pseudotumor', it was recently given its name due to the potential of recurrence and multifocal involvement. We describe the case of a 43-year-old Hispanic male who presented with a large symptomatic pleural-based mass which turned out to be CFT and was later complicated by empyema. Our aim is to increase awareness about this rare disease and throw light upon its benign nature, despite an alarming and suspicious appearance on imaging. Due to the large size of our patient’s mass (largest reported yet), he needed extensive chest wall reconstruction, leading to complications requiring additional invasive procedures. This underscores the importance of early diagnosis and treatment which can reduce the need for aggressive surgical manipulation and avoid postoperative complications, thereby providing high-value care. Treating physicians should be mindful of this, in order to prompt early recognition to ensure effective patient care. Calcifying fibrous tumor (CFT) is a rare, benign proliferation of fibroblasts comprising of dense collagen, lymphoplasmacytic infiltrate, and psammomatous or dystrophic calcifications . An accuA 43-year-old Hispanic male with active smoking history presented with new-onset right-sided scapular pain. Past medical history was negative for cardiopulmonary disease. Physical examination revealed expiratory and inspiratory wheezing over the right upper lung zone. Routine laboratory studies were unremarkable. Chest x-ray (CXR) showed a right middle lobe pleural-based mass with stippled calcification, measuring 12 x 7 cm (Figure This was followed by CT of the chest showing a large irregularly shaped mass in the posterior right hemithorax, which appeared to involve the middle and lower lobes of the lung, abutting the intercostal space between ribs 5 through 8 Figure . AdditioCT-guided core needle biopsy revealed tumor fragments with partial translucent and opaque white flecks and pink areas in a rubbery to firm consistency. Histopathological examination showed dense collagenous matrix scattered with areas of psammomatous calcifications, nests of plasma cells, few mast cells, eosinophils, and lymphocytes Figures , 4. No aThe patient underwent thoracotomy. The tumor was firmly attached to the medial aspect of the aforementioned ribs in close proximity to the vertebral transverse process, almost completely decompressing adjoining lung. A 15 x 15 x 10 cm lobulated unencapsulated mass with a smooth, hard white surface was removed along with wedge resection of the involved portion of right upper and lower lobes and ribs 5 through 8 followed by chest wall reconstruction. Microscopically, the tumor was adherent to pulmonary tissue in an intimate relationship with ribs, but without local invasion. The benign tumor cells were notably intermingled with normal rib tissue structures; however, no evidence of cellular invasion was seen (Figure CT chest showed a large empyema in the right lower lobe requiring multiple invasive procedures for intercostal drainage Figure . The patDue to its rarity, no randomized controlled trials exist to understand CFT. The limited evidence of its clinical presentation and pathogenesis comes from a few case reports and systematic reviews available in the medical literature. Owing to the abundance of inflammatory cells reported almost universally on histopathological examination, it was previously thought to be the result of a reactive process, representing end-stage inflammatory myofibroblastic tumor. However, later clinicopathological studies showed that the relationship between these two tumors was weak . ConsequAccording to the largest systematic review which studied 157 cases of CFT, this tumor presents in a bimodal age distribution with one peak at 0-4 years and another between 25 and 34 years with a mild female preponderance .Through this case, our aim is to increase awareness about this rare disease which should be kept in the differential diagnosis while evaluating lung masses. Although radiologically such large masses may appear highly suspicious for malignancy, biopsy is needed to prove their benign nature. Generally being non-aggressive, it does not invade the neighboring tissues, even while growing in size. Some cases have described entrapment of nerve bundles noted on microscopic examination, however without signs of invasion, once again suggesting its benign nature ,10. NotaPrompt treatment is required and might involve surgical excision, with laparoscopic approach employed for smaller lesions. It is important to highlight that to our knowledge, our patient’s mass was the largest among reported CFTs in the literature and required extensive chest wall reconstruction including resection of the involved lung parenchyma. This led to complications like empyema, requiring additional invasive procedures, and further complicating the postoperative course of our patient. This underscores the importance of early diagnosis and treatment. Although difficult due to its asymptomatic presentation and incidental diagnosis in most cases, it is quintessential to prevent the sequela of rapid tumor growth and resulting compression of adjacent organs. Treating physicians should be cautious of these potentially life-threatening complications to provide efficient postoperative management and ensure patient safety. Recurrence is rare, with only a few reports documenting it on long-term follow-up .CFT is a rare entity that should be kept in the differential diagnosis of lung masses. It is important for medical professionals to be aware of its clinical presentation and course to allow early recognition and prompt treatment. Although generally benign, it can have deleterious effects when grown to exponential sizes, leading to aggressive surgeries and related post-operative complications, which can be avoided if caught early. Thus, providing high-value care for patients.