Mandible lesion biopsy was not conclusive. Biochemical workup was consistent with hypercalcaemic primary hyperparathyroidism (HPH)  and vitamin D deficiency (7.8 ng/ml). Neck ultrasound (US) performed at our Unit showed a hypoechoic area under the left thyroid lobe suspected for enlarged parathyroid. This lesion was also identified after CT images re-evaluation (Fig. Two patients presenting with mandible-jaw neoformations were referred to us for endocrine evaluation. A 51-year-old male presented with painful T9 vertebral fracture and multiple osteolytic areas in spine, pelvis, and right mandible branch Fig. . Metastaion Fig. . Patholoion Fig. . Left inThe second case was a 68-year-old woman complaining of mildly painful swelling in the left maxillary branch. Dental CT showed a hypodense lesion at 25, 26, and 27 alveolar regions and small collateral calcifications in the right pharyngeal wall Fig. . Lesion 18F-FDG-PET. These two clinical cases underline the importance of evaluating calcium homeostasis in patients presenting with osteolytic facial bone mass suspected for BT. Once HPH is biochemically detected, US is very useful to identify neck masses consistent with enlarged parathyroids that could be confirmed by subsequent scintigraphy [Available HPH guidelines do not provide clear indications concerning BT presentation and management . BT appetigraphy . Literattigraphy .In conclusion, our cases underline the importance of suspecting HPH in patients with low-to-mild aching bone masses in skeletal regions with major cortical components clinically and radiologically consistent with BT. Patients should undergo imaging workup as well as clinical and biochemical investigation. Multidisciplinary management is advisable.


In younger patients of LCH, we should consider that the effectiveness of follow‐up without aggressive treatment for SS‐type LCH in the oral and maxillofacial bone. However, there are very rare case in which an SS‐type LCH recurred after showing a healing tendency. Regular follow‐up must be performed even after healing. In younger patients of LCH, we should consider that the effectiveness of follow‐up without aggressive treatment for SS‐type LCH in the oral and maxillofacial bone. However, there are very rare case in which an SS‐type LCH recurred after showing a healing tendency. Regular follow‐up must be performed even after healing LCH treatment options include surgery, chemotherapy, and radiation therapy. In some cases, healing may occur spontaneously. We describe a case of LCH in the mandible of a child who experienced spontaneous regression after biopsy.Langerhans cell histiocytosis (LCH) is a disease in which one type of antigen‐presenting cells, the Langerhans cells, aggregates and proliferates in various organs such as the bone, skin, liver, spleen, or lung, resulting in tissue damage,2A 4‐year‐old boy was seen by nearby pediatricians for painful swelling of his left cheek. Computed tomography (CT) and contrast‐enhanced magnetic resonance imaging (MRI) showed osteolysis and mass formation in the left angle of mandible, and he was referred to our hospital with suspected ameloblastoma.At the patient's first visit, his left cheek showed painful swelling, and the submandibular lymph nodes were palpable, tender, and mobile Figure . The pat18F‐fluorodeoxyglucose (FDG) (SUVmax = 4.90) only in the left mandible (Figure CT imaging showed osteolysis with indistinct borders in the left mandibular ramus, and rupture was observed in the buccolingual cortical bone Figure . A perioe Figure . These iTo establish a diagnosis, an incisional biopsy was performed under general anesthesia Figure . SpecifiSurgical intervention was initially planned; however, because the patient was a child, factors such as jaw growth and tooth formation had to be taken into consideration. On the other hand, there have been reports of cases in which spontaneous remission can be expected in SS‐type LCH at the bone limit.3LCH is a condition that results from the monoclonal expansion of immunophenotypically and functionally immature Langerhans cells.The clinical manifestations of LCH in the jawbone include bone pain and swelling, trismus, tooth sway, pathological fractures, and bone deformities.The choice of treatment for LCH depends on the type of disease. For the SM and MM types, combination chemotherapy is often selected. However, the disease can progress rapidly and lead to a fatal progress in refractory and recurrent cases, and the definitive establishment of a treatment method for such cases is much desired. The treatment of SS‐type bone lesions, on the other hand, has been performed empirically and has included surgical curettage, local administration of steroids, combination chemotherapy, and radiation therapy; there is no consensus on treatment to date. It has been reported that a quarter of SS‐type bone lesions are expected to regress spontaneously.Various hypotheses have been proposed for spontaneous regression of LCH, including arrested lesion growth due to the disappearance of local inflammation,The authors declare that they have no conflicts of interest regarding the publication of this paper.KO, TO, SI and TN: gathered the patient data, performed a literature review, and wrote the manuscript. YK, KO, MM and SR: reviewed, corrected patient data, and revised the manuscript. AS: was involved in overall supervision of the paper. All authors: read and approved the final manuscript.Written consent was obtained from the patient's parents.This case report did not receive any funding. Authors have access to all source data for this case report. All procedures performed in this study were in accordance with the ethical standards of Okayama University Research Ethics Committee and with the 1964 Helsinki Declaration and its later amendments.


Optic nerve sheath meningiomas (ONSMs) are rare and benign tumors that affect the optic nerve. Although surgical decompression may be used for large tumors that cause mass effect on the surrounding structures, the mainstay of treatment is radiotherapy. We report the case of a 54-year-old female patient who presented with progressive vision loss due to a recurrent right ONSM despite fractionated radiotherapy eight years prior and the subsequent interval regression of the tumor. The optical coherence tomography at the time of recurrence revealed thinning of the right retinal nerve fiber layer. She underwent salvage stereotactic radiosurgery using a marginal dose of 15 Gy. At six months post-radiosurgery, the patient had a dramatic improvement in visual acuity and visual fields despite persistent thinning of the retinal nerve fiber layer. This case illustrates how salvage radiosurgery can be a useful treatment modality in these challenging situations. This tumor’s exophytic growth and the steep dose fall-off of Gamma Knife radiosurgery might favorably affect visual recovery. However, the outcomes of single-session radiosurgery for ONSMs should be further evaluated. Primary optic nerve sheath meningiomas (ONSMs) are rare benign tumors comprising approximately 2% of all orbital tumors and representing less than 1-2% of all meningiomas .Radiographically, ONSM appears as a contrast-enhancing mass encasing the optic nerve which is visualized as a “tram-tracking” sign on axial post-gadolinium T1-weighted magnetic resonance imaging (MRI). Because these clinical and radiographic features are distinctive, surgical biopsies are currently considered unnecessary given the associated risk of visual morbidities . ManagemAlthough radiotherapy for ONSMs generally provides good tumor control, tumors may recur, and treatment strategies for recurrent ONSMs have not been well established. Here, we present the case of a patient presenting with decreasing visual function and radiographic progression of a tumor toward the chiasm five years after IMRT using 50.4 Gy in 28 daily fractions. Since salvage with single-fraction stereotactic radiosurgery (SRS), she has maintained tumor control and has improved visual function.History and examinationA 54-year-old woman presented with subjective blurry vision and decreased visual acuity (20/150 OD) on visual examination eight years prior and was diagnosed with a primary ONSM. She underwent fractionated IMRT receiving 50.4 Gy in 28 fractions. Her vision improved (20/20 OD) post-therapy and she returned to her normal daily activities. Follow-up imaging demonstrated a decrease in tumor volume on MRI five years later. However, two years later, seven years after completing radiotherapy, the patient began to note further right-sided visual loss, and imaging demonstrated progression of the tumor consistent with late radiation failure. It was at this point she presented to our clinic. She was found to have a progressive ONSM with severe right-sided visual loss with hand motion vision only.Investigation3  is the primary concern following radiotherapy for lesions close to the optic apparatus and is thought to be secondary to ischemia-related vascular injury resulting in optic nerve atrophy and injury to the neuronal elements ,11. In rOn the other hand, regarding tumor control, SRS has an excellent track record ,15. ThisWe do not have a clear explanation for the excellent visual outcome obtained in the present case. Although there is no good standard for pretreatment evaluation for prognosis of visual function -19, goodMilano et al. reviewed 34 studies and 1,578 patients and found that prior resection in 76% of the patients did not correlate with any increased risk of RION but prior irradiation, seen in 6% of patients in this review, demonstrated a crude 10-fold increased risk of RION . This inManaging previously radiated tumors poses a unique challenge. This case highlights some of the more challenging aspects of managing an ONSM that had previously undergone fractionated radiotherapy with good visual improvement and now underwent salvage SRS with a dramatic recovery of vision. Particularly for tumors that are adjacent to the optic apparatus or encompass it, as in this case, understanding and planning therapeutic options for patients who have already received a significant dose of radiation to the optic apparatus limits how aggressive one can be. Understanding some of the nuances of ONSMs and their presentation may allow for the use of salvage radiosurgery in cases that have been previously radiated with poor vision. In addition, this case highlights that OCT and visual fields alone may not always be predictive of RION, or the chance of recovery and salvage therapy should still be considered.


Cerebrospinal fluid over-drainage is a common complication of ventriculoperitoneal devices . In terms of hemorrhage, subdural hematomas are usually more frequent lesions than epidural hematomas, which, more rarely, may also be seen after ventricular shunt procedures and may lead to rapid neurological decline and even death unless a surgical procedure can be promptly performed. In our study we report the case of a 5 years old boy with history of congenital obstructive hydrocephalus treated with a ventriculoperitoneal shunt insertion when he was 8 months old. The patient was admitted with sudden deterioration of level of consciousness secondary to tri-ventricular hydrocephalus. He underwent a shunt revision. Two weeks later, he developed a loss of consciousness with a large left extradural hematoma contralateral to the side of ventriculoperitoneal shunt. He underwent an evacuation of the hematoma with a good postoperative outcome. Epidural hematoma, especially controlateral to Ventriculoperitoneal shunt, is extremely rare. The pathophysiology and the possible use of a programmable valve to prevent these lesions are briefly discussed. •A 5 years old boy treated for congenital hydrocephalus since he was 8 months old with a fixed pressure Ventriculoperitoneal shunt on the right side. He was brought to our emergency for intracranial hypertension syndrome with disorders of consciousness. Patient's consciousness level was gradually improved after shunt revision. After 2 weeks, he was admitted with sudden deterioration of consciousness.•The cerebral CT scan showed a massive left-sided epidural hematoma and was located in the right parietal and occipital aera, causing an important mass effect.•The prognosis is good if the diagnosis is made quickly. The advent of programmable shunts (PS) theoretically enables a better way to avoid the over-shunting and its consequences. However, this procedure is also beset with a considerable number of complications, which may be related to shunt obstruction, infection and excessive drainage of cerebrospinal fluid (CSF) , such as2A 5 years old boy treated for congenital hydrocephalus since he was 8 months old with a fixed pressure VPS on the right side. He was brought by his mother to our emergency for intracranial hypertension syndrome with disorders of consciousness. The CT scan showed active triventricular hydrocephalus.Patient's consciousness level was gradually improved after shunt revision and was discharged 3 days later. After 2 weeks, he was admitted with sudden deterioration of consciousness. At admission, his GCS scores were 13/15 E3V4M5.Neurological examination revealed muscle power was 3/5 on right side, 5/5 on left side without sensitive trouble. The cerebral CT scan showed a massive left-sided epidural hematoma and was located in the right parietal and occipital aera, causing an important mass effect .Fig. 1ThThe intervention was performed by our chief resident under general anesthesia a wide frontoparietal craniotomy was performed we proceeded to the evacuation of the epidural hematoma and tacked the dura up to the skull to prevent recurrence without shunt management .Fig. 2A/The follow up was good, he regains consciousness without any complication. At control CT scan we showed the absence of the epidural hematoma with normal aspect of the ventricles. The patient was discharged after 10 days.the patient was seen after 1 month then 6 months and 1 year and the clinical examination was well without any anomaly confirmed by radiological controls.This case has been reported in line with the 2020 SCARE guidelines {5}.3The use of ventricular shunts to treat hydrocephalus is one of the most common neurosurgical procedures worldwide. The advent of such devices, despite many inconveniences and complications attributed to the use of a prosthesis, enables a fast and efficient treatment of hydrocephalus. Complications such as obstruction of the device and infection (up to 60%) are more frequent than bleeding .Epidural hematomas are an unusual complication of ventriculoperitoneal shunt in the treatment of chronic hydrocephalus and are much less common than post shunting subdural hematomas . The firThe mechanism of epidural hematoma formation is related to the rapid reduction in intracranial pressure as a result of overdrainage of the CSF, resulting in separation of the dura mater from the skull and tearing of small dural vessels ,7,11,12.The most common location of epidural hematoma is on fronto-parietal lobes. This may be due to lose fixation of the dura to the cranial vault at this region . EpiduraThe occurrence of epidural hematomas acutely (few days) after shunting, as in the case reported herein, is a rare condition and, in the authors opinion, demands immediate evacuation of the lesion as well as the prompt replace or, in the case of flow-controlled shunts, the reprogramming of the system pressure to higher levels.The largest cases series of epidural hematoma after ventricular decompression, from Odake and Matsumoto , includeThere are two distinct approaches for treatment of epidural hematomas in these cases; conservative and surgical, the choice for or against of which depend on clinical setting, age, time of bleeding, size on CT-scan, extent and thickness of the hematoma and the presence of midline shifting on CT. Any rapid accumulation in children and younger adults must be treated surgically.Dominic Power, farouk ali-khan and Martin Drage report a historical case of a contralateral epidural hematoma after a ventricular shunt. They supposed that it was sudden decompression of the dilated ventricles that stripped the dura from the bone of the skull vault and caused the acute epidural hemorrhage . The devThe particularity of our observation is that the epidural hematoma arose contralateral to the shunt without recent head trauma. We suppose that the mechanism appears to be caused by a combination of some factors: a likely over-functioning of the device was responsible for the hyper drainage with a sudden decompression of the dilated ventricles, stripping the dura from the bone and leading to the contralateral acute epidural hematoma. He only benefited from the evacuation of the hematoma which was compressive. As for the valve, we did not change it.The advent of programmable shunts (PS) theoretically enables a better way to avoid the over-shunting and its consequences, once the width of the ventricles and the cerebrospinal fluid hydrodynamics can be strictly controlled by adaptation of the draining pressure. In fact, Carmel et al.  and Decq4Controlateral epidural hematoma as a complication of ventriculoperitoneal shunt is a rare but rapidly evolving and often catastrophic entity. Maybe third ventriculocisternostomy or the use of programmable shunts and a longer hospital stay could change the evolution and outcome of these cases.The authors declared that this study has received no financial support.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.None.Provenance and peer review Not commissioned, externally peer-reviewed.Yassine TAHRIR: writing the paper and Corresponding author. Said HILMANI: Correcting the paper. Abdelhakim LAKHDAR: Correcting the paper.TAHRIR YASSINE.The authors of this article have no conflict or competing interests. All of the authors approved the final version of the manuscript.


Bladder paragangliomas (PGLs) are extremely rare catecholamine-producing neuroendocrine tumors. They arise more frequently in the trigone and have unpredictable depth and behavior. Most cases typically present with a well-defined set of symptoms triggered by micturition or bladder overdistension. Besides long-term follow-up, they are usually managed by either transurethral resection (TUR) or partial cystectomy (PC). However, about 25% of all documented cases do not manifest clinically, raising both diagnosis and management challenges. This report describes an unusual case of a misdiagnosed, functional PGL arising on the bladder dome, which was fortuitously detected in a 21-year-old female during a fertility workup. Owing to its hypervascular nature and submucosal location, bladder PGL was suspected on ultrasound and CT findings and successfully diagnosed before surgery through biochemical confirmation. It was managed by cystoscopy-guided laparoscopic partial cystectomy (LPC) with good oncological and urinary outcomes, as well as no compromise of her fertility potential. To our best knowledge, this is the first case to be incidentally detected on transvaginal ultrasound during evaluation for infertility. This case also stresses the importance of considering PGL in the differential diagnosis of atypical bladder tumors, as well as conservative management through simultaneous laparoscopy and cystoscopy, when approaching young patients with large functional PGL. Bladder paragangliomas (PGLs) are very rare neuroendocrine tumors. Since the description of the first case in 1953, its incidence is less than 0.05% of all bladder tumors and less than 1% of all pheochromocytomas . Among aA 21-year-old female, who has been trying to get pregnant for over two years, was referred by her gynecologist to our urology outpatient clinic because of incidental transvaginal scan (TVS) finding of a bladder intramural mass during routine infertility workup Figure .She was a nonsmoker and not exposed to any solvents or chemicals. She denied any history of gross hematuria, LUTS or pelvic pain, weight loss, or appetite disturbance. Nine months prior, she had consulted her general practitioner for intermittent hypertensive attacks during micturition accompanied by headache, sweating, nausea, fatigue, and palpitations of 12 months’ duration. Prior to this clinical scenario, she described herself as healthy with unknown comorbidities. Her doctor had prescribed a calcium channel blocker (10 mg/day of amlodipine) and referred her to a cardiologist for a further workup to assess the potential causes of secondary hypertension. However, she did not show up as she had felt better.On admission, she had an uncontrolled hypertension of 139/100 mmHg and a heart rate of 82 bpm with no fever. Her physical examination was unremarkable. Transabdominal ultrasound revealed a 7-cm solid mass along the dome and the right lateral wall of the urinary bladder, with hypervascularity on color Doppler Figure . UrinalyChest-abdomen-pelvis CT scan demonstrated a localized bladder mass of 70.7 x 33.2 x 47.3 mm along the dome of the urinary bladder, exhibiting significant homogeneous post contrast enhancement Figure . Given tThe patient consented to undergo laparoscopic partial cystectomy (LPC) and subsequently switched on oral alpha-blocker agent for 15 days before, along with high-salt diet and increased water intake. Under general anesthesia and invasive blood pressure monitoring, she was placed in Trendelenburg modified lithotomy position to allow simultaneous cystoscopy, which initially revealed a yellow submucosal mass on the dome and right lateral wall of the bladder. Given the tumor’s location, a ureteral catheter was inserted on the right side. With a transperitoneal approach, laparoscopy showed hypervascular mass on the bladder dome Figure .The specimen was then dissected extravesically under simultaneous cystoscopic guidance and resected completely Figure , with suThe ureteral catheter was removed after two days, and the patient was discharged with urinary catheter removal scheduled on the 10th day. One month later, she become normotensive and had normal levels of urine metanephrines. She was told that her tumor is most likely sporadic and benign; nonetheless, both genetic testing and long-term follow-up were strongly advised. However, she did refuse the genetic screening, concerned about its impact on her relatives and pregnancy planning.At six months, she denied any hypertensive attacks or voiding difficulties, with no residual tumor on CT scan. At her three-year follow-up, she achieved successful natural pregnancy and had no evidence of local recurrence, distant metastasis, or multifocal disease.Bladder PGLs may be fortuitously discovered by pelvic ultrasound, CT, and magnetic resonance imaging (MRI) . To our The endoscopic appearance of bladder PGLs is helpful when endocrine testing or imaging is inconclusive or when TUR of bladder tumor (TURBT) was done incidentally. On cystoscopy, PGL should be considered in the presence of yellowish submucosal mass, especially if the blood pressure shows fluctuations during the procedure -4. HowevPathologically, a PGL consists of polygonal cells harboring abundant cytoplasm and surrounded by a prominent vascular network, with a pathognomonic nested “Zellballen” pattern and staining positive for chromogranin, synaptophysin, and S100 but negative for cytokeratin ,3,5. HowCurrently, more than 50% of all PGLs and 35% of apparently sporadic cases occur in the setting of inherited susceptibility mutations involved in several pheochromocytoma-associated syndromes. It includes multiple endocrine neoplasia type 2, von Hippel-Lindau disease, neurofibromatosis type 1, and hereditary pheochromocytoma-paraganglioma syndromes . Hence, Had PGL not been incidentally imaged, preoperatively diagnosed, and successfully treated, our patient would have been at risk of lethal paroxysm, cardiac disorders, and malignant potential ,9.There are currently no guidelines on the surgical management of bladder PGL. Treatment changes according to the tumor’s functionality, size, location, and invasiveness. It can include TUR of bladder tumor (TURBT) and partial and total cystectomy -4,6. TheIn this case, unlike TURBT, the laparoscopic approach allowed less disruption and en bloc excision of the tumor, resulting in lower intraoperative morbidity and better disease assessment and control . SimultaIncidental TVS finding of a bladder PGL during fertility workup is uncommon, yet it should be considered in the differential diagnosis of atypical highly vascular and/or submucosal bladder tumors. Endoscopic management can be used in selected cases. Nevertheless, LPC is the standard treatment option in this condition. This case also favors the notion that a hybrid approach using simultaneous cystoscopy may be superior to pure laparoscopy, providing efficient, secure, and effective procedure, when approaching young patients with large functional PGL on the dome of the urinary bladder.


Extramedullary involvement of acute myeloid leukemia (AML) is infrequent, and ascitic infiltration is even more unusual. We present a case of a 48-year-old woman diagnosed with NPM1-mutated AML that debuted with ascites, for which morphological studies of the ascitic fluid did not detect leukemic infiltration, maybe due to technical problems in the sample preparation. Multiparameter flow cytometry (MFC) detected a blast population compatible with AML, and allele-specific PCR detected NPM1-mutated transcripts. Body fluid infiltrations are an infrequent initial manifestation or sign of progression in AML. As far as we know, this is the first reported case of an NPM1-mutated AML that debuted with ascites, and also the first description of the utilization of molecular techniques to detect the leukemic origin of the ascites. This case highlights that, given that allele-specific PCR and MFC increase the sensitivity of morphological studies, these techniques should be routinely applied in the study of any kind of effusion detected in an AML patient. NPM1) is currently considered a definitive entity in the revised WHO classification of 2017  karyotype was found and the translocation t(q24q22) Tumor markers were requested and a CA125 elevation was evidenced (191.9 U/mL). Parasite stool tests, quantyferon determination and viral serology were negative. A thoraco-abdominopelvic computed tomography (CT) scan revealed the presence of abundant ascites and both pericardial and bilateral pleural effusion. The echocardiogram showed 60% left-ventricular ejective function (LVEF), without hemodynamic compromise. The CT scan also described thickening of the long segment of the ileum, infradiaphragmatic lymphadenopathies, signs of peritoneal carcinomatosis, and heterogeneous changes in the left ovary. Gynecologic magnetic resonance imaging showed bilateral hemorrhagic cysts and a myomatous uterus.3 with 670 neutrophils/mm3 and 3665 monocyte-looking cells/mm3. Cytologic evaluation of the ascitic fluid manifested infiltration by mixed inflammatory cellularity, predominantly lymphocytic and scarce mesothelial epithelial cellularity, suggestive of an inflammatory origin. MFC distinguished the following cell groups: neutrophils (11.53%), monocytes/histiocytes (<1%), lymphocytes (8.33%), and a population of blasts that constituted 80% of the cellularity analysed, whose abnormal phenotype has been described in BM ; we reached complete remission (CR) with minimal residual disease (MRD) by MFC of 0.10% and a ratio of 0.56% by allele-specific PCR in the BM (with ABL employed as control gene). However, the ascites, diarrhea and abdominal pain persisted. A gastroscopy, ileoscopy and colonoscopy were performed, with biopsies confirming no underlying malignant or infiltrative digestive pathology. A new study of the ascitic fluid was performed after the administration of the induction chemotherapy, showing persistence of 8.7% of myeloid blasts with an immunophenotype similar to that described at diagnosis, and a ratio of 16.3% by NPM1 mutation A allele-specific PCR was detected  < 1.1 g/dL, a leukocyte count of 4470 cells/mmed in BM . Once thdetected . TherefoNPM1 ratio of 686%. Due to her poor performance status , she initiated non-intensive treatment with 5-azacitidine, of which she only received one cycle. A few weeks later, an increase in leukocyte and blast count was documented in the peripheral blood, and a rapid increase in ascites was noted. The patient finally died from upper gastrointestinal bleeding.A second echocardiogram revealed severe ventricular dysfunction (LVEF of 35%) and persistence of moderate pericardial effusion, so we decided to avoid anthracyclines and treated the patient with a cycle of high dose cytarabine (HiDAC). She reached complete cytological remission (1% blasts) in BM with MRD by MFC of 0.95%, and an NPM1. Cytogenetic analysis showed no alterations, and additional mutations detected by NGS did not modify the prognosis according to European LeukamiaNet (ELN) 2017 guidelines [FLT3-ITD mutations modify the prognosis of AML with mutated NPM1 [FLT3-ITD mutation but there are additional molecular and immunophenotypic features that, although not included in ELN classification, seems to have prognostic value. A recent study reports decreased overall survival (OS) in patients with the triple combination of NPM1, DNMT3A and IDH1 or IDH2 mutations, as in our case [NPM1+ AML has been described as presenting negativity for CD34 and HLA-DR, resembling the typical immunophenotype of acute promyelocytic leukemia, as in the case reported herein. The prognostic implications of this specific immunophenotypic profile are not well known, although some studies show a greater overall survival in these patients [NPM1 quantitation in BM performed after the second cycle of chemotherapy, given that these measurements seem to be very concordant [Our patient was diagnosed with AML with mutated idelines . Therefoted NPM1 . Our patpatients . We havencordant .The study of ascitic fluid through paracentesis is necessary in all patients with AML, since more frequent causes of ascites should be ruled out such as liver cirrhosis, infections, hypoalbuminemia or venous thrombosis. Ascites related to malignant disease have been reported mainly in solid tumors, and as a complication of some lymphomas . ExtrameIn our case, the origin of ascites could be mixed . Morphological studies, historically employed in the diagnosis of effusions, may have a lack of sensitivity to detect malignant origin and flow cytometry, and molecular techniques should be employed. Nevertheless, the lack of detection in our case seems to be due to a technical problem with the preparation of the sample, given that ascitic fluid showed a massive leukemic infiltration by MFC. We did not perform molecular analysis of the ascitic fluid at diagnosis because we had not fully characterized the AML at that time, but we should have extracted and stored the DNA. Nevertheless, we could detect NPM1-mutated transcripts in the ascitic fluid after the first cycle of induction. Additionally, MFC and molecular techniques can provide a quantifiable parameter for assessing minimal residual disease, which could help to individualize the postremission therapy of the patients.NPM1-mutated AML that debuted with ascites, and also the first description of the utilization of molecular techniques to detect the leukemic origin of the ascites. This case report highlights the importance of ruling out a leukemic origin by sensitive techniques of any effusion that patients with AML present at diagnosis, or during disease evolution.As far as we know, this is the first reported case of an


To present the importance of considering conjunctival myxoid stromal tumors in the differential when evaluating eyelid lesions as these tumors could indicate undetected systemic syndromes including Zollinger-Ellison Syndrome, Carney complex, and other endocrine disorders.We present the case of a 56-year-old Caucasian female who was evaluated for a solid cyst-like structure of the palpebral conjunctiva just temporal to, but not involving, the left lower eyelid punctum. The lesion was removed with histopathologic examination of the specimen revealing the lesion to be a myxoid spindle cell tumor, consistent with conjunctival myxoid stromal tumor.Myxoid tumors are an abnormal proliferation of mesenchymal cells. These are most commonly found in the heart and less commonly in the bone, skin, and skeletal muscle. Myxoid tumors of the conjunctiva are a very rare reported finding and most reported cases involving the conjunctiva occur on the bulbar conjunctiva. Our patient was found to have a conjunctival myxoid stromal tumor of the palpebral conjunctiva. As these are rare lesions, we believe that considering this as a differential when evaluating eyelid margin lesions is important due to the association of these tumors with systemic conditions including Zollinger-Ellison Syndrome, Carney complex, and other Endocrine disorders. The patient stated that the lesion had been present for greater than one year and that during this time, it fluctuated in size, sometimes improving in size with warm compresses. The patient's past ocular history was notable for blepharitis and dry eye syndrome; however, she had no history of prior eye surgery. Patient's visual acuity was 20/20 in both eyes. External examination was remarkable for an elevated cyst of the left inferior eyelid margin, just temporal to the punctum with telangiectasias but no feeder blood vessels . The cliThe patient requested excision of the lesion as she was experiencing significant irritation. Due to the proximity of the lesion to the punctum, the patient was taken to the operating room for excision of the lesion. Wedge resection was considered, but ultimately not performed given the benign appearance of the lesion and the lesion location close to the lacrimal punctum. For the procedure, a 15-blade scalpel was used to perform a shave biopsy and was sent for histopathological evaluation. The tissue sample was fixed in formalin, embedded in paraffin, and 6-μm sections were obtained for routine Hematoxylin and Eosin stain and immunostaining. Histologic analysis revealed conjunctival mucosa containing proliferation of a singular spindle and round cells within a myxoid stroma  A, B, C.3,,,,,A myxoma is a benign proliferation of cells of mesenchymal origin.On review of the literature, the most common location of conjunctival myxoid tumors is on the bulbar conjunctiva. Qin et al. reported on ten patients with conjunctival myxoid stromal tumors with all ten tumors occurring on the bulbar conjunctiva.,,,,Oftentimes, conjunctival myxoid tumors are clinically misdiagnosed as cysts. Other differentials for these lesions include apocrine mixed tumor, nevus, amelanotic melanoma, fibrous histiocytoma, lymphangioma, myxoid neurofibroma, spindle cell lipoma, solitary fibrous tumor, rhabdomyosarcoma, pseudotumor, fibromyxoma, superficial angiomyxoma, cutaneous benign mixed tumor, and myxoid liposarcoma.4This case report of a conjunctival myxoid stromal tumor of the palpebral conjunctiva presents an uncommon scenario as 93% of reported conjunctival myxomas occur on the bulbar conjunctiva.Written consent to publish this case has not been obtained. This report does not contain any personal identifying information.No funding or grant supportAll authors attest that they meet the current ICMJE criteria for Authorship.The following authors have no financial interests to disclose: PSM, CDR, AF, GE.


Xanthogranulomatous endometritis is a rare benign pathology mimicking endometrial carcinoma. Xanthogranulomatous endometritis is a rare benign pathology mimicking endometrial carcinoma. We report a case of 87‐year‐old woman with an enlarged uterus without other clinical signs. After hysteroscopy and evacuation of 500 mL of lactescent fluid, the relapse of the collection led to a hysterectomy.Xanthogranulomatous inflammation (XGI) is an exceptional condition implicating the presence of foamy histiocytes, and other inflammation cells affecting various organs, mainly the kidney and gallbladder.2A 87‐year‐old postmenopausal woman consulted as part of her annual gynecological follow‐up. Her medical‐surgical history includes bilateral breast in‐situ cancer treated by bilateral mastectomy, permanent atrial fibrillation, and a transient ischemic attack. She also carries an aortic bioprothesis. She had no history of endometriosis, pelvic inflammatory disease or use of intrauterine device. She denied any symptom of pelvic pain, fever, vaginal bleeding, or weight loss. Gynecological examination reveals an enlarged uterus for which a pelvic ultrasound is requested. The pelvic ultrasound Figure  shows anAn operative hysteroscopy is performed under general anesthesia and after dilatation of the cervix visualizes an intrauterine lactescent liquid, which suggests pyometra. This liquid is odorless and could in fact correspond to noninfected blood in extreme degradation. The intervention enables the evacuation of 500 mL of liquid with a surgical aspiration device. Hysteroscopy shows no signs of malignancy, and the endometrium is not hypertrophic. An endometrial curettage is performed, and the histopathological analysis is normal. Bacteriological analysis of the fluid finds multisensitive Pseudomonas aeruginosa for which an antibiotic treatment (ceftazidime intravenous then ciprofloxacine per‐os) is administered. Biologically, there is no inflammatory syndrome and there is no argument for a bacterial infection.At the postoperative visit one month later, the pelvic ultrasound showed a reconstitution of the intrauterine fluid measuring 70 mm long. A total hysterectomy and bilateral salpingooophorectomy are performed. Standard histological technique was performed. The part was fixed in 4% buffered formalin (with a fixation time of 24 hours). The area of interest was included as a block. The latter underwent dehydration in successive alcohol baths of increasing concentration. The sample was then embedded in paraffin. Three μm sections with a microtome were made, then spread on a slide, and stained with hematoxylin‐eosine‐saffron. We observed an entirely destroyed endometrium, replaced by a polymorphic inflammatory infiltrate containing mainly plasma cells, as well as lymphocytes and some neutrophils. There are clusters of large cells with clear cytoplasm corresponding to foamy histiocytes. Some rare giant cells organized around cholesterol crystals are visualized Figure . The his3Xanthogranulomatous endometritis is a rare disease characterized by chronic inflammation of the endometrium. The first case of XGE was reported by Barua et al in 1978,The pathogenesis of XGE is still incompletely understood. The cervical stenosis, as in our case, lead to the accumulation of endometrial secretions, infection, and tissue necrosis, and further release of cholesterol and other lipids which were phagocytozed by macrophages resulting in the formation of foam cells and XGE.Malignancy forms are an important differential diagnosis of this lesion. XGE can be mistaken for malignancy on clinical and radiological appearance,Recovery after antibiotic treatment or spontaneous resolution is the most frequent outcomes,4We report a case of recurrent xanthogranulomatous endometritis with a major intrauterine retention of infected fluid. Since XGE may mimic cervical or endometrial carcinoma clinically and pathologically, knowledge of this unusual and rare inflammatory disease is important for gynecologists, radiologists, and pathologists.The authors report no conflicts of interest in relation to the present study.Philippe Merviel: substantially contributed to the conception, design of the work, the acquisition, analysis, and interpretation of clinical, ultrasound, and MRI data, and have drafted the work or substantively revised it. Pandora James: acquired the clinical data and translated the manuscript. Marianne Carlier: acquired the clinical data. Isabelle Thomas‐Kergastel: served as the head of the Women's Radiology department. Marine Guilloique: acquired the data on MRI. Virginie Conan‐Charlet: served as the Pathologist referent in this case. Clara Bastard: served as the Pathologist in this case. Pascale Marcorelles: served as the head of the Histopathology department. Yannick Jobic: acquired the clinical data. Pierre‐François Dupré: substantially contributed to the conception, the acquisition, analysis, and interpretation of clinical data.The consent of the woman is available in the medical record and with the corresponding author.


Sarcomas of the uterine corpus are rare malignant neoplasms, which are further classified into mesenchymal tumors, and mixed  tumors. The main issues concerning these neoplasms are the small number of clinical trials, insufficient data from evidence-based medicine, insignificant interest from the pharmaceutical industry, all of which close a vicious circle. The low frequency of these malignancies implies insufficient experience in the diagnosis, hence incomplete surgical and complex treatment. Additionally, the rarity of these sarcomas makes it very difficult to develop clinical practice guidelines. Preoperative diagnosis, neoadjuvant and adjuvant chemoradiation, target and hormone therapies still raise many controversies. Disagreements about the role and type of surgical treatment are also often observed in medical literature. There are still insufficient data about the role of pelvic lymph node dissection and fertility-sparing surgery. Pathologists’ experience is of paramount importance for an accurate diagnosis. Additionally, genetics examinations become part of diagnosis in some sarcomas of the uterine corpus. Some gene mutations observed in uterine sarcomas are associated with different outcomes. Therefore, a development of molecular classification of uterine sarcomas should be considered in the future. In this review, we focus on the epidemiology, pathogenesis, pathology, diagnosis and treatment of the following sarcomas of the uterine corpus: leiomyosarcoma, low- and high-grade endometrial stromal sarcomas, undifferentiated sarcoma and adenosarcoma. Uterine carcinosarcomas are excluded as they represent an epithelial tumor rather than a true sarcoma. Sarcomas of the uterine corpus are malignant neoplasms, which are further classified into mesenchymal tumors and mixed  tumors ,2. The mULMS is a malignant mesenchymal tumor of myometrial smooth-muscle derivation, which represents 60% of all uterine sarcomas and accounts for 1–2% of uterine malignancies. Its annual incidence rate is approximately 0.8 per 100,000 women. It is the most common type of uterine sarcomas originating from the myometrium ,4,5,6. IThe most common presenting symptoms for women with ULMS include enlarged uterus, abnormal vaginal bleeding, abdominal pain, vaginal discharge, urinary frequency, anaemia  and constipation ,7,8. An Generally, ULMS arises from a solitary lesion, though multiple lesions are also documented . There iGrossly, ULMS often appears greyish yellow or pink, and with a soft or fleshy consistency  4]. ULM. ULM4]. ULMS is not graded, as there is no correlation between high-grade histology and overall survival . DiffereMicroscopically, ULMS includes three subtypes: spindle cell/conventional, epithelioid, and myxoid .Spindle cell-type/conventional ULMS—high mitotic index (>10 mitoses/10 high power fields and tumor cell necrosis), spindled/fascicular cells with moderate-to-severe pleomorphism, infiltrative border and the possible presence of multinucleated cells, myometrial invasion  are showEpithelioid and myxoid ULMS are rare subtypes, which may have mild or focal nuclear pleomorphism and a lesser degree of mitotic activity than what is seen in the typical ULMS . An abunImmunohistochemical staining with smooth muscle actin, desmin and h-caldesmon shows positive reaction in tumor cells, and p53, p16, Progesterone and Ki-67 help to differentiate ULMS from leiomyoma ,13,14,15There is mounting interest in the preoperative diagnosis of ULMS. Currently, no clinical symptom, laboratory test or imaging study can provide effective preoperative diagnostic modalities for ULMS ,16. FurtOn vaginal or abdominal sonography, large heterogeneous masses with areas with central vascularity or necrosis could be observed in cases of ULMS  Figure . ComputeIn our opinion, there is no imaging modality which can differentiate ULMS from uterine leiomyoma, as both have similar findings on imaging. However, in cases with a newly diagnosed or enlarged pelvic mass, the patients should undergo a pelvic ultrasound by an experienced sonographer as an initial imaging study.Some studies have shown that levels of serum lactate dehydrogenase (LDH) total activity and its isoenzymes may be relevant in preoperative diagnosis of ULMS ,20,21. NIn conclusion, LDH total activity and its isoenzymes may play a role in the preoperative evaluation of suspect uterine mass. However, further studies are necessary to determine its actual reliability.Endometrial biopsy or curettage may detect uterine ULMS in a substantial proportion of cases—approximately in 25% to 50% of cases ,8. ThereThe majority of prognostic factors of ULMS are summarized in FIGO staging of uterine ULMS and endometrial stromal sarcomas is shown in In cases of ULMS confined to the uterus, total abdominal hysterectomy (TAH) without lymph node dissection (LND) is recommended. The procedure could be performed by open surgery or minimally invasive techniques. The incidence of lymph node metastases is approximately 5% to 11% and lymphadenectomy does not impact overall survival (OS). Moreover, hematogenous metastases are typically presented in patients with ULMS—the most common site of initial metastasis is the lung, followed by the peritoneal cavity, liver parenchyma, brain, and bone ,7,8,9. HART does not improve OS but may improve local control. It is recommended in cases of locally advanced tumors confined to the pelvis. A combination of ART and adjuvant chemotherapy impact disease-free survival for 3 years, but do not affect overall 3–5-year survival rate ,7,8. GenThere is no proven benefit of ACT . There aThe first-line treatment is monotherapy with doxorubicin as it is well tolerated and less toxic than other chemotherapy agents. Recommended combination regimens include doxorubicin/ifosfamide and doxorubicin/dacarbazine. Ifosfamide monotherapy treatment is under investigation. Gemcitabine treatment alone or a combination of gemcitabine/dacarbazine and gemcitabine/vinorelbine are other recommended regimes ,5,6,7,8.In advanced stage of the disease, target therapies could be initiated . Trabectedin treatment may improve the quality of life, and it leads to long-term stabilization of the tumor. Pazopanib significantly increased median progression-free survival (PFS), but not OS. Bevacizumab, a monoclonal antibody against VEGF, does not improve OS and PFS ,5,6,7,8.If it is feasible, secondary cytoreduction is the first option in cases of metastatic sarcoma. Better outcomes may be expected in cases of prolonged time to recurrence (>12 months) and an isolated site of recurrence ,5,6,7,8.Doxorubicin is a preferable single agent for the treatment of metastatic ULMS, as it is less toxic than combination regimes . GemcitaA retrospective multicenter study of the Spanish ovarian cancer research group concluded that trabectedin could be used as a second- or third-line treatment such as ULMS. Authors stated that trabectedin showed clinical benefit in patients with recurrent/metastatic ULMS and it could be given after failure to an anthracycline-based regimen . In 2017Pelvic radiation could be utilised in cases of unresectable loco-regional relapse. Radiation therapy may be considered after pelvic metastasectomy for better local control, but there will be no benefit for OS.Suggested treatment of ULMS is shown in Endometrial stromal tumors are of endometrial stromal origin and account for less than 2% of uterine tumors. In accordance with the current World Health Organization (WHO) classification 2020, endometrial stromal tumors include endometrial stromal nodule, low-grade endometrial stromal sarcoma (LGESS), high-grade endometrial stromal sarcoma (HGESS) and undifferentiated uterine sarcoma (UUS) ,41. EndoLGESS, also previously known as endolymphatic stromal myosis, is the second most common type of uterine sarcomas (second to ULMS) ,44. AlthDifferent chromosomal aberrations have been detected in LGESS of which JAZF1-SUZ12 genes fusion is the most common, followed by JAZF1-PHF1, EPC1-PHF1 and MEAF6-PHF1 ,53.Grossly, the LGESS are soft tan to yellow, smooth-surfaced polyps or nodules  that could be partly infarcted and hemorrhagic . Although it is usually with ill-defined borders and worm-like permeation within the myometrium and blood vessels, some tumors might appear relatively circumscribed ,44,47,48Typical histological features of LGESS are the “tongue-like” patterns of myometrial and lymphovascular invasion . CytomorThis may exhibit sex cord-like, epithelioid and glandular differentiation. Other features helpful in the diagnosis include an arborizing vascular pattern, foam cells with necrosis and rope-like collagen ,45,46,47It is important to notice that distinguishing LGESS and endometrial stromal nodule on curettage specimen is not possible . ImmunohUnfavorable prognostic factors include: advanced stage, increased age of the patient, non-surgical treatment, morcellation ,44,45,46TAHBSO is the standardized treatment modality in the treatment of patients with LGESS . OptimalIn 2014, the WHO officially reintroduced HGESS based on the identification of YWHAE-NUTM2A/B gene fusion . The fusion of these two genes causes activation of 14-3-3 oncoprotein as a recurrent event in this more malignant subgroup of tumors ,58,59. TGrossly, HGESS is a pink-to-yellow-tan polypoid friable tumor with extensive invasion through the myometrium . Cases oHistological features include tongue-like permeation, high mitotic activity (>20–30 mitoses/10 HP fields), variable micronecrosis, lymphovascular invasion, mild-to-severe nuclear pleomorphism  60,61].,61.60,61The single most significant prognostic factor is tumor stage. Other prognostic factors are morcellation, age, surgery, chemotherapy and radiotherapy ,66. UnliCurrently, due to the rarity of the disease, there are many controversies about preferable treatment options. Therefore, treatment options should be stated as suggestions. TAHBSO is the standard surgical procedure. Optimal cytoreduction in the advanced stage is recommended . A studyUUS is an extremely rare and aggressive tumor with no specific line of differentiation that constitutes a diagnosis of exclusion ,68,69,70Genes mutations of ULMS, LGESS, HGESS and UUS are summarized in UAS is a rare biphasic tumor composed of benign glandular epithelium and malignant stromal elements ,75. UAS Clinical features include abnormal vaginal discharge or vaginal bleeding, sometimes accompanied by an enlarged uterus. At advanced stages, women have abdominal pain and distension ,75,76.Grossly, UAS typically presents as a polypoid mass ranging from 1 to 20 cm. Cases of multiple polyps have been described . The tumCurrently, Müllerian adenosarcomas are not graded histologically as 50% of patients with the previously described low-grade UAS will develop disease recurrence. Therefore, UAS should not be considered as a tumor of low malignant potential ,83. -Broad leaf-like appearance, formed by the malignant stroma compressing the benign epithelium.-The stroma is typically cellular, atypical, with periglandular stromal cuffing, a feature that distinguishes these tumors from endometrial or adenomatous polyps.-Spindled/round cells are located around the glandular components and form peri-glandular cuffs.-Mitotic activity ≥2 mitoses/10 HPFs (most noticeable in areas of peri-glandular cuffing).Low-power microscopic examination shows a biphasic tumor with admixed glands and prominent stroma throughout . MorpholAlthough mucinous, serous, and squamous epithelium components have been described, the epithelial component is usually endometrioid and metaplasia (squamous and mucinous are most common) is often observed ,82. HomoThere are no immunohistochemical markers pathognomonic for adenosarcoma . The mosPreoperative radiological and pathological findings are not specific enough to provide accurate diagnosis and seem to be difficult for radiologists and pathologists ,77. CT mPrognostic factors are summarized in The most significant prognostic factors are: age, sarcomatous overgrowth, myometrial invasion, lymphovascular invasion, and lymph node involvement. Tumor necrosis and cellular atypia (asterisk in the table) could not be independent risk factors, as they are associated with sarcomatous overgrowth, high mitotic rate, myometrial invasion or necrosis ,82,83,84FIGO staging of UAS is shown in TAHBSO is recommended for the majority of patients with UAS . AdnexesThe first treatment option is surgical resection of local recurrence and distant metastases. They occur in approximately 22 to 42% and 27 to 45% of patients, respectively. Distant metastases is most commonly present in the liver and lungs. Radiation therapy is initiated for palliative situation for non-resectable recurrences ,77. IfosUterine sarcomas raise many controversies in oncogynecological practice. Effective preoperative diagnostic modalities for these sarcomas have not been estimated yet. Molecular studies are more often used as an indispensable part of uterine sarcoma diagnosis and prognosis. Therefore, a development of molecular classification of uterine sarcomas should be consider in the future. There are sufficient data about best optimal treatment regimes, as the role of AHT and ART is controversial. It is supposed that target therapies could play a potential role in uterine sarcomas treatment, but the effects of such therapies have not yet been fully elucidated. Multidisciplinary team management is mandatory in order to increase OS, PFS and DFS of patients with uterine sarcomas.


Actinomycosis is an uncommon cause of central nervous system infection. A case of skull bone osteomyelitis with epidural empyema is presented. A 44-year-old man presented with chronic osteomyelitis of skull vault with epidural and subgaleal collection diagnosed by histopathology as actinomycosis. He had similar lesion at the same site 10 years ago, which was excised completely. Recurrent Actinomycosis of the skull vault is uncommon in literature. This case highlights the importance of considering actinomycosis as a differential diagnosis of tumorous growths and stresses on the importance of tissue histopathology for diagnosis and need for surgery to control the disease. Treatment is prolonged, therefore compliance with the long-term antibiotic duration is essential to prevent complications and avoid recurrence. Actinomyces species are normal inhabitants of the oral cavity, the gastrointestinal and pelvic tracts and are not found existing freely in nature. Cervicofacial, thoracic and abdominal are the most common forms of actinomycosis. Central nervous system (CNS) actinomycosis are rare and usually result from direct extension of a neighboring focus or from hematogenous spread from a distant focus. Brain abscess are the most reported CNS manifestations. Calvarial and epidural abscess are extremely rare, and few cases have been reported in the literature. We herein, present a case of recurrent frontal skull bone osteomyelitis with epidural subgaleal collections caused by actinomycosis and review the literature.Actinomycosis is a subacute bacterial infection characterized by tissue fibrosis, abscess formation and draining sinuses. A 44-year-old Sudanese man with no significant comorbidities was referred to neurosurgery outpatient clinic with the complaint of left frontoparietal scalp mass lesion, progressively increasing in size over 1 year. Patient did not seek medical advice until he started to experience unremitting hemi-cranial headache and cosmetic disturbance. He did not have fever or seizures. There was no history of trauma or dental problems.On examination he appeared healthy and not in distress, with normal vital signs. He was alert oriented, and cranial nerves were intact. There was no motor or sensory or cerebellar function deficit. Ear, nose, mouth, and throat examinations did not show any focus of infection. He never smoked or consumed alcohol. He has been working as a Shepherd for more than 20 years.His past history was remarkable for a similar lesion in the same location 10 years ago. The lesion appeared spontaneously over a period of 6 months. There was no antecedent history of trauma or surgery in the scalp or oro-maxillo-facial area. He had no history of head, neck, or oral infections. CT scan of head at that time showed left frontal mass of 4.5 cm width, homogeneous soft tissue mass, which appeared adherent to the underlying bone with a well-defined erosion confined to the outer table with medullary sclerosis and intact inner table. His routine laboratory tests at that time were within normal limits. No evidence of immunosuppression was reported. The mass was excised, and histopathology revealed the presence of micro abscesses with vague granuloma formation surrounding filamentous bacteria consistent with the diagnosis of actinomycosis. Oral amoxicillin was started, and he was asked to follow up in infectious diseases clinic; however, he was not compliant to his treatment and did not follow up. In the current presentation, his initial work up showed Hemoglobin 15.5 g/dl, White Blood Cell count 9500/microL , C Reactive Protein 12 mg/dL, procalcitonin 0.03 ng/mL. HIV test non-reactive, and normal blood glucose, renal and liver functions.Skull x-ray  revealedTypes of CNS actinomycotic lesions include brain abscess, meningitis, subdural empyema, actinomycoma, and epidural abscess; with brain abscess accounting for 75 % of the cases . CNS inv13 years old Indian boy presented with healing vertex ulcer, seizure and features of raised intracranial pressure [40 years old Spanish-American was admitted in 1967 with left hemiparesis and recurrent generalized seizures [35 years old Indian woman admitted with left hemiparesis following few months of headache and scalp swelling [21 years old Irish man presented with left frontal scalp abscess . IncisioA case of left parietal calvarial osteomyelitis due to actinomycosis in a 32 years old previously healthy woman is described . She preOur case is unique for both the site of occurrence and the relapse after 11 years. In the first episode in 2010 he had osteomyelitis involvement of only the outer table of calvarium. Adequate surgical excision was done, however, he did not comply with antimicrobial therapy and lost from follow up. The second episode was in the same site which suggests that it was a relapse rather than reinfection. This time lesion penetrated the inner table with extension to epidural space. Fortunately, there was no brain parenchymal involvement. No other focus of involvement with actinomycosis infection was identified in our patient.In the case series reported by Smego et al., out of 70 patient with CNS actinomycosis only one patient had relapse . This paIn conclusion, Actinomycosis is chronic indolent locally spreading infection caused mainly by breach of mucosa. We present a rare case of actinomycosis of frontal skull bone which recurred 11years after successful surgery. Keeping a high index of suspicion is essential to avoid delayed diagnosis and to start appropriate treatment. This case demonstrates that prolonged antibacterial treatment is of paramount importance to prevent relapse even after achieving adequate surgical excision.None.


Cerebral cavernous malformation (CCM) is a developmental abnormality of blood vessels that supply the brain. It is composed of large, adjacent capillaries which contain little or no neural tissue. They mostly occur in the supratentorial region. However, the occurrence of these vascular lesions can be seen at different sites of the central nervous system (CNS). The prevalence of CCM is estimated to be 0.4% in the general population and among the affected patients, 18.7% have multiple lesions. However, about 30-50% of CCM cases are asymptomatic and are found incidentally. Here we report a case of an eight-year-old girl with a massive hemorrhagic presentation of a left parietooccipital CCM. Developmental cerebrovascular malformations are types of brain vascular anomalies, which consist of endothelial-lined caverns. Any defect on endothelial tight junctions will make this vascular malformation more susceptible to leak or bleed. The prevalence of this vascular anomaly is estimated to be between 0.4% and 0.8%; however, it has a rare occurrence in the pediatric age group  and few An eight-year-old Bahraini girl, with no known medical illness, was presented to the ED by her parents following a sudden severe headache associated with persistent vomiting. The parents did not report any history of trauma or seizures. She had no significant past medical or surgical history. There was no family history of similar problems or any other neurological defects. On general inspection, she was conscious with a Glasgow Coma Scale (GCS) of 15 out of 15, alert and oriented to time, person, and place. The vitals were as follows: pulse rate 110 beats per minute, blood pressure 110/60 mmHg, O2 saturation 99%, and the temperature was 37.6°C. Neurological examinations such as extraocular movements, pupillary reflex, and cranial nerves assessment were normal. Her higher mental functions were also normal. No papilloedema was noted. All limb motor functions, as well as cerebellar functions and gait, were normal. Examination of other systems was also unremarkable except for a mild abdominal tenderness in the upper part. A brain CT scan was obtained. The pre-contrast CT demonstrated a large round well-defined mixed high-density area in the left parietooccipital region suggesting hemorrhage surrounded by a rim of edema causing adjacent sulci effacement  was done the next day and no vascular anomaly was seen. Subsequently, the patient was planned for surgical evacuation of the hematoma and exploration for any lesion. She underwent left parietal craniotomy and evacuation of the hematoma and lesion under general anesthesia with the assistance of a NeuroNavigator. Operatively, the gross appearance of the lesion was consistent with venous cavernoma (mulberry appearance with gliotic changes around it) filled with clots Figure .Biopsy was taken to the histopathology laboratory. The report explained that the sections of the entire specimen illustrated blood clots and dilated thin-walled blood vessels, some of which are thrombosed, while others reveal a distinct layer of foamy histiocytes/siderophages within and around. Patchy calcification is observed in some of these blood clots. No specific granulomatous inflammation or any atypical changes detected. Immunohistochemistry findings revealed positive cluster differentiation 68 (CD68) in the foamy histiocytes and negative glial fibrillary acidic protein Figure .Postoperatively, she was extubated, kept in the pediatric intensive care unit (PICU) for two days, discharged to the general ward, stayed for three days, and discharged home. On the follow-up a week later, she was asymptomatic, conscious, alert, and oriented with no focal neurological deficits (FNDs). She was advised to continue on anticonvulsant and planned to repeat MRI six months later Figure .The incidence of CCMs is mainly investigated in adults. However, some data has been published regarding their incidence and prevalence in the pediatric age group. The development of CCMs appears to increase with age and reaching to its plateau at the age of 18 to 20. Collecting data regarding sex distribution from 13 independent studies indicate a male/female ratio of 1.3:1. A total of 359 (57%) of the cases were male out of a total number of 629 pediatric patients with CCMs .Cavernous malformations  are capillary malformations in the brain that can happen sporadically or in a familial pattern. These lesions have an evolutionary nature based on serial MRI studies. There are three genetic loci  responsible for the familial cavernomas that varies among different ethnic groups ClinicalThe diagnosis of CM is made by MRI scan. Brain MRI is the most sensitive and specific imaging modality to reach to definite diagnosis of CM. Recommended sequences include standard T1, T2, susceptibility-weighted imaging (SWI), or gradient recall echo images (if SWI not available). A T1 sequence with gadolinium contrast may be useful in determining if there is an associated developmental venous anomaly (DVA) and to rule out the other forms of masses in the brain. With regard to the diagnosis, angiography may not be helpful and that is because blood flow in the cavernoma lesions is relatively poor. MRI is a better diagnostic tool in this setting. MRI can show a “popcorn” appearance on both T1- and T2-weighted images and the hemosiderin ring around the lesion is best seen on T2 images, indicative of a distant hemorrhage. Once the CM is identified, a contrast-enhanced MRI should be obtained to rule out the presence of DVAs . CT scanStudies have indicated that the size of the lesion cannot predict the risk of repeat hemorrhages , 9. In aA decision on how to plan treatment for CCM patients depends on a multitude of factors that are basically related to their symptoms and the risk of secondary seizure or hemorrhage. Stereotactic radiosurgery, microsurgical resection, and conservative therapy are the three methods of treatment for CM lesions. The indications for microsurgical resection of CM are basically characterized by: multiple hemorrhages in eloquent areas or a single hemorrhage in a non-eloquent area that is associated with FND and severe cardiac or respiratory symptoms that may be attributed to patients with CCMs. On the other hand, patients with no signs or symptoms who are diagnosed on MRI should be managed conservatively .AccordiCerebral cavernoma mostly presents itself with seizures, headache, and focal deficits. Emergency neurosurgical intervention is rare in pediatric CM cases because they are less likely to cause life-threatening hemorrhages. However, physicians must be prepared for any unforeseen events in cases AVM is suspected. Surgery still remains the gold standard for intracranial cavernoma. Conservative therapy is appropriate for cavernomas that are asymptomatic and for those who had one non-devastating episode of hemorrhage. Complications can occur and extensive informed consent should be obtained. Careful radiological studies and close clinical follow-up should be planned for the patient to monitor the residual or recurrence of the disease.


Glomus tumors (GT) are rare mesenchymal tumors that develop in the subungual digital region. Extradigital GTs are very rare, with atypical clinical features. We report the case of a 63-year-old male with a five-year history of intermittent shoulder pain, where an excisional biopsy confirmed the diagnosis of a glomus tumor. Glomus tumors (GT) are rare neoplasms, representing less than 2% of all soft tissue tumors . GTs oriGTs are typically located in the upper extremities, with almost 75% of them developing in the subungual region of the digits ,5. TypicAlthough extradigital GTs have been reported in the literature, they are considered quite infrequent, with atypical clinical features, resulting to markedly therapeutic delays . More spHerein, we report a rare case of an extradigital GT, located in the left suprascapular area, and manifesting as chronic shoulder pain.2, while his past medical history included hypertension, coronary heart disease, diabetes, COPD, and hyperuricemia.A 63-year-old male presented to the outpatient department of our institution with a five-year history of pain in the left supra-scapular area. The symptoms were paroxysmal and became incapacitating during the last year. The patient noted that even the application of cold water and minimal direct stimulation triggered the onset of pain. He previously sought treatment with local steroidal ointments, oral non-steroidal anti-inflammatory drugs (NSAIDs) and physiotherapy with little to no success. His BMI was 38 kg/mPhysical examination revealed a small reddish nodule in the left supra-scapular area. The lesion was immobile, well-defined and with no associated inflammatory signs. Upon palpation, pin-point tenderness was recorded. There were no pathological findings from both neurological and vascular clinical examination. Similarly, no palpable axillary and cervical lymph nodes were noted.An excisional biopsy, with wide resection margins was performed, revealing a tumor of a size about 1.6 × 1 × 0.6 cm. The histopathologic examination a revealed a circumscribed mass consisting of uniform cells in haematoxylin eosin stain . A negatOn the seventh post-operative day, wound dehiscence was noted, treated with regular wound dressings care. A complete secondary intention wound healing was achieved twenty days after excision. At six months follow-up, a total remission of symptoms was confirmed, with no signs of local or distant recurrence.Glomus tumors are rare mesenchymal neoplasms arising from the glomus body . GTs typGlomus apparatus is a specialized neuro-myo-arterial unit located in the dermo-hypodermic junction . It consThe exact pathogenetic origin of extradigital GTs though, is, still, unclear . SeveralAlthough GT is considered as a benign neoplasia, cases of malignant transformation and distal metastases have been reported . MalignaTypical manifestation of GTs is a solitary tumor associated with intermittent pain crises, triggered by alterations of temperature and pressure gradient . The paiHowever, only a small percentage of extradigital GTs present with the typical clinical triad ; in factThe atypical presenting symptoms and the low diagnostic accuracy of the clinical tests, often leads to misdiagnoses and delays in the delivery of the optimal treatment . LiteratImaging has an eminent role as a diagnostic adjunct. Magnetic resonance imaging (MRI) is the most sensitive imaging study for GT with a sensitivity and specificity of 90%, and 50%, respectively . HoweverSurgical excision remains the standard of care resulting to the immediate resolution of the symptoms and the minimization of recurrence risk . PreviouThe present study reported the rare case of a suprascapular GT presenting with chronic shoulder pain symptoms. Increased clinical suspicion regarding extradigital GTs is required to reduce misdiagnosis rates, and the physical and mental consequences of delayed diagnoses.


There is no report of the application of intraoperative computed tomography to the extremities, and its usefulness is not mentioned.We present a case of a patient with the elbow pain and loss of the forearm rotation due to the prominent bicipital tuberosity of the radius, which was diagnosed as enthesopathy. Surgical treatment to excise the prominent part of the bicipital tuberosity of the radius was recommended. However, it is difficult to perform the appropriate excision of the abnormal prominent part because of complications such as bicipital tendon rupture. The patient was successfully treated by surgical resection under the control of intraoperative computed tomography.Intraoperative computed tomography scan is a useful tool to assess the remaining volume of the abnormal bones. Enthesopathy of the bicipital tuberosity of the radius has been reported to be the enthesis of the insertion site of the bicipital tendon into the radius which causes limitation of the range of forearm rotation motion . ResectiA 77-year-old, right-handed female was referred to us with right elbow pain that began a few years ago. She had a restricted range of motion with 60° of supination and 30° of pronation with painful crepitus of her right elbow during forearm rotation. She had a full range of motion, with 145° of flexion and 0° of extension. Tenderness existed on the tuberosity of the radius without redness, swelling, muscle atrophy, or neurological deficits. Blood test results were within normal limits. She had no history of trauma or disease. Radiographs of the elbow showed an irregular prominence of the bicipital tuberosity of the radius (Fig. First, conservative treatment was tried, with daily activity instruction and oral medication nonsteroidal anti-inflammatory drugs for 3 months. The conservative treatment was not effective and the patient desired the operation. A skin incision was made on the anterior site of the proximal forearm. The bicipital tuberosity of the radius was exposed between the ulnar side of the brachioradialis muscle and the radial side of the biceps tendon Fig. . The irrAt the latest follow-up 1 year after the operation, the patient had no pain and no motor weakness or limitation in forearm rotation Fig. . CT scanEnthesopathy of the bicipital tuberosity of the radius has previously been reported –4. PatieIt is sometimes difficult to recognize which part of the tuberosity is to be resected because of the deformity of the irregular prominence of the bicipital tuberosity. To prevent the detachment of the bicipital tendon, resection is sometimes insufficient. In the current case, the intraoperative CT scan showed that the resection was insufficient to flatten the prominence and prevent impingement between the radius and the ulna. After assessment of the volume of remaining bone, we resected the abnormal prominence. At the postoperative follow-up, the patient’s pain and forearm rotation range of motion had improved. Intraoperative CT scan is a useful tool to assess the remaining volume and shape of abnormal bones during a bone resection. However, adequate identification of abnormal bones with a proper surgical exposure is easy for a skilled surgeon who can identify them without intraoperative CT scan. Intraoperative CT scan is not widely used, and current intraoperative fluoroscopy in proper position is also helpful when the surgical exposure is difficult.In conclusion, enthesopathy of the bicipital tuberosity of the radius can be successfully treated by surgical resection, in which intraoperative CT scan is a useful tool to assess the remaining volume of the abnormal bones.


Retinal vein occlusion (RVO) is a differential diagnosis for Coats’ disease due to retinal arterial Leber’s aneurysms. Occasionally, RVO shows a Coats-like appearance. The differential diagnosis between Coats’ disease and RVO is essential for clinical therapy, especially for those obsolete RVOs with collateral vessels and without retinal hemorrhage. In this case report, we describe and discuss the imaging characteristics of bilateral RVO-simulated Coats’ disease with tortuous retinal arterioles and its prognosis after anti-vascular endothelial growth factor therapy, which will be beneficial for its definite diagnosis and aid further investigation. Coats’ disease was first reported in 1908 as an ocular disorder with retinal telangiectasis and massive intraretinal and subretinal exudation . RetinalA 52-year-old woman was presented to the ophthalmology department of First Hospital of China Medical University complaining of blurred vision in her left eye for two months. She had amedical history of hypertension and no diabetes. The best-corrected visual acuity was 60/60 in her right eye and 15/60 in her left, and the intraocular pressure was 17mmHg in both eyes. Slight cataracts were found in both eyes, and other anterior segment examinations were normal. Wide-field fundus imaging showed bilateral tortuous retinal arterioles with retinal hemorrhage, as well as tortuous and dilated retinal veins in the supratemporal quadrant. Heavy hard exudates were found in the macula of the left eye. The middle phase of fluorescein angiography revealed numerous miliary aneurysms in both eyes and patchy, blocked fluorescence in the left eye. Peripheral fluorescein angiography demonstrated the “fishing net” appearance of retinal capillaries and non-perfusion regions in both eyes . Multico4 and Schatz et al [Herein, we described a bilateral branch RVO with a Coats-like appearance and tortuous retinal arterioles. Hypertension may be a risk factorfor RVO. In contrast to reports by Scimecaetaltz et al , no secotz et al , which iFamilial retinal arteriolar tortuosity is characterized by tortuosity of the second- and third-order retinal arterioles . Khan etIn conclusion, this was the first report of bilateral RVO-simulated Coats’ disease with tortuous retinal arterioles. A definitive diagnosis is crucial for further investigation and treatment.


Testicular cancer is one of the most curable cancers. However, the course of the disease largely depends on the clinical stage at diagnosis, and there are still cases where the tumor size is large, which makes surgical treatment challenging.A 30-year-old man presented with painless, extremely enlarged scrotum. A CT scan revealed a tumor of the right testis of 21.5 × 15 × 18cm in size. The patient underwent a right orchiectomy and histologic examination revealed teratocarcinoma.Suspicion of hydrocele testis should prompt meticulous differential diagnosis including malignancies. There is a strong need to increase public awareness in terms of symptoms of testicular cancer. •In the presence of a giant hydrocele, palpation of the testis might prove impossible.•Suspicion of hydrocele testis should prompt meticulous differential diagnosis.•Ultrasound scan may underestimate the tumor size.•In a case of giant testicular tumor inguinal - scrotal approach should be considered.•Despite the tumor size, the patient had no metastases beyond the lymphatic system. In the presence of a giant hydrocele, thorough palpation of the testis might prove impossible; therefore ultrasonographic examination is essential to rule out other abnormalities.A hydrocele may coexist with testicular cancer and in up to 9%, it is the reason behind diagnostic delay.2A 30-year-old man was admitted to the Urology Department in February 2020 for diagnosis and treatment of painless scrotal swelling of an undetermined cause. He first noticed the swelling three weeks before admission; the lesion continued to grow causing walking difficulties. The patient had no history of urological disease. On examination, the lesion was huge, soft and tightened the overlying skin. Palpation of the right testis was impossible; transillumination showed accumulation of clear fluid around the testicle.Complete blood count performed on the day of admission revealed leukocytosis (10.52 × 10^3/μl), low hematocrit level (38.5%) and thrombocytosis (419 × 10^3/μl). Urine tests showed protein (25 mg/dl) and leukocytes (25/μl). The patient underwent an ultrasound examination (US) of the scrotum, abdomen and retroperitoneum. Two fluid collections (of 77 and 25 mm in size) were found in the scrotum, and a hydrocele was suspected A and B. The patient was diagnosed with a right testicular tumor and scheduled for right orchiectomy. Due to the enormous size of the tumor, two separate inguinal and scrotal incisions were made A and B. 3Nowadays, due to broad access to healthcare and education, mortality rates in testicular cancer are low, however the prognosis varies significantly depending on the stage and type of cancer.,The main reasons for delay in the diagnosis of testicular cancer are insufficient knowledge and symptom neglect by the patient as well as incorrect initial diagnosis.Nowadays, testicular tumors of the size presented in this report are extremely rare. The average size of a testicular tumor is between 4 and 5.4 cm. Only a few giant testicular tumors have been described so far, with dimensions up to 32 × 28 cm.In this case, the testicular tumor was associated with the accumulation of a large amount of fluid, which hampered palpation. Initially, a hydrocele testis was suspected, which should always alert doctors to consider a neoplastic disease. Testicular ultrasound should not be delayed as it allows detection of neoplastic lesions with almost 100% sensitivity.,The recommended treatment of testicular cancer remains high inguinal orchiectomy, which, however, might prove inadequate to deliver a very large tumor. In such a case, inguinal - scrotal approach should be considered. Separate inguinal and scrotal incisions or one continuous incision are then made.4A primary diagnosis of hydrocele testis necessitates a differential diagnosis to determine the cause of the pathology, including a neoplastic process. Retroperitoneal lymphadenopathy should also prompt a physician to perform scrotal ultrasound to rule out testicular pathologies. The presented case evidences a still unsatisfactory level of society's awareness about testicular cancer. Therefore, efforts should be made to increase knowledge about this cancer, its signs and symptoms, prevention and the importance of early diagnosis.Written informed consent was obtained from the patient for publication of this case report and any accompanying images.This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors. Only the APC fee was financed under the agreement between Elsevier and Polish Consortium.Authors do not declare any conflict of interest.


An angiolipoma is a benign tumor, and a primary mediastinal angiolipoma is extremely rare. Herein, we describe the presentation and management of a posterior mediastinal angiolipoma in a woman with loss of consciousness. Chest computed tomography (CT) revealed a contrast-enhancing mass in the right posterior mediastinum, with intercostal arterial blood supply identified on three-dimensional reconstruction CT (3D-CT). Magnetic resonance imaging revealed a fatty component. Pre-operative embolization of the supplying intercostal artery was performed to reduce intraoperative bleeding. Mass resection was performed using video-assisted thoracic surgery. Histopathology confirmed angiolipoma diagnosis. Although rare, a posterior mediastinum angiolipoma should be considered a possibility; 3D-CT and pre-operative embolization may be useful in the surgical treatment of hypervascular mediastinal tumors, such as angiolipomas. Angiolipomas are rare, benign, soft tissue tumors comprising mature fatty tissue and blood vessels, occurring in the subcutaneous tissue and extremity muscles . PrimaryA 48-year-old woman was brought to our hospital owing to loss of consciousness; she had no major disease history, an unremarkable physical examination and normal laboratory findings. Contrast-enhanced chest CT revealed a mass 35 mm in diameter, with heterogeneous contrast, in the right posterior mediastinum at the thoracic T7–T8 level . The masThe patient provided informed consent for the publication of this report.et al. reported a radiologically lipid-poor posterior mediastinal angiolipoma [An angiolipoma is a rare, benign, mesenchymal tumor, accounting for 5–17% of all lipomas . Previouiolipoma ; thus, cNo evidence of tumor invasion into the surrounding tissues indicated a benign tumor. The hypervascularity and location of the mass preoperatively suggested a hemangioma or neurogenic tumor. Retrospectively, the preoperative MRI suggested a lipoma, liposarcoma or angiolipoma owing to mixed fat components. Although extremely rare, the differential diagnosis of mediastinal tumors should include angiolipoma.et al. reported a case of angiolipoma with massive intra-operative bleeding in excess of 3000 mL [Surgical resection was necessary; however, massive bleeding during surgery was expected because the tumor was hypervascular, and a feeding vessel arising from the intercostal artery was detected on 3D-CT. Fujiu  3000 mL . Massive 3000 mL . Pre-ope 3000 mL . To the In conclusion, this is the second reported posterior mediastinal angiolipoma treated with pre-operative embolization followed by resection. Although rare, an angiolipoma should be considered in the differential diagnosis of posterior mediastinal tumors. Moreover, 3D-CT might be useful to identify feeding vessels to the posterior mediastinal tumor, thus assisting in treatment planning. Preoperative embolization for hypervascular tumors is useful in reducing intra-operative blood loss, with subsequent successful resection using VATS.


The incidence of immature teratomas is highest in young adults aged 18 to 39. The prognosis heavily depends on the International Federation of Gynecology and Obstetrics (FIGO)We report a 36-year-old female who presented with a 17.6cm x 10.5cm x 24.2cm intraabdominal mass and ascites. Upon tumor resection, she was found to have a stage IIIa, grade 2 immature teratoma of the left ovary, with glial tissue being the metastatic cell type. Disease progression continued despite treatment with BEP. She was then treated experimentally with six months of bevacizumab and temozolomide, given its rarity and targeted therapy for glial tissue. Despite monoclonal antibody therapy, the tumor progressed again and was treated with docetaxel and gemcitabine. A repeat CT of the chest, abdomen, and pelvis demonstrated scattered peritoneal implants that were increasing in size. Chromosome analysis was performed and revealed somatic mutations of MLH1, MSH2, MSH6, and PD-L1. The patient has requested a break from chemotherapy but will be treated with direct immunotherapy when she restarts.This case’s importance lies in its rarity because fewer than 10 cases of immature teratomas with metastatic glial tissue are noted in the world’s literature. Furthermore, this is the first reported case of this cell type being treated with immunotherapy in the world literature. Teratoma originates from the Greek word “teras,” which signifies monster. Immature teratomas are malignant tumors of the ovary and represent 1% to 3% of all germ cell tumors and 20% of malignant ovarian germ cell tumors [We report a 36-year-old female with a medical history of Hashimoto’s thyroiditis on levothyroxine who presented with complaints of progressively worsening abdominal distention and clothes fitting tighter over the past two months. She also endorsed left lower quadrant abdominal and suprapubic pain. She reported her symptoms were associated with early satiety, constipation, nausea without emesis, and not being relieved by prescription probiotics. These symptoms prompted her to visit the emergency department. On physical examination, she had a distended abdomen with tenderness to palpation and a low-grade fever. Complete blood count was significant for anemia with a hemoglobin of 8.4 g/dL and hematocrit of 26.9%. Complete metabolic panel noted hypocalcemia of 7.5 mg/dL. Computed tomography (CT) of the abdomen and pelvis was significant for a bulky mass arising in the anterior pelvis and extending cephalad above the level of the umbilicus. The mass measured 17.6 cm transversely, 10.5 cm in the anteroposterior dimension, and 24.2 cm in the craniocaudal dimension. The lesion consisted of fluid and solid components with some calcifications and fat present. Furthermore, there was small to moderate free fluid in the posterior pelvis and lower abdomen bilaterally. The patient agreed to exploratory laparotomy and underwent left salpingo-oophorectomy, appendectomy, omentectomy, left pelvic and aortic lymph nodes sampling, and tumor debulking. Operative findings showed a large left-sided ovarian mass that was fleshy in nature. Frozen sections noted possible teratoma with stromal components concerning for malignancy; therefore, the patient was staged. Pathology results of specimens collected during the surgery showed grade 2 immature teratoma of the ovary, acute appendicitis, gliomatosis peritonei, peritoneal tumor and omentum, aortic and pelvic lymph nodes negative for metastatic tumor. The clinical stage showed the International Federation of Gynecology and Obstetrics (FIGO) stage IIIA1(i) calculated as stage IIIA2 . The pathology results of the left ovary were notable for grade 2 immature teratoma of the ovary. The right pelvic peritoneum, omentum, abdominal fluid, and peritoneal tumor were remarkable for the presence of immature glial tissue. The left fallopian tube, appendix, left pelvic, and aortic lymph nodes were negative for tumor. Immunohistochemical stains were positive for glial fibrillary acidic protein, which is consistent with immature teratoma. In addition, mesothelial cells were positive for pankeratin, calretinin, and vimentin. As seen in Figures She was started on apixaban for deep venous thrombosis (DVT) prophylaxis and oxycodone as needed for pain. Additionally, she was started on bleomycin, etoposide, and cisplatin chemotherapy regimen every 21 days for four cycles. Repeat CT scan showed progression of disease with multiple soft tissue masses seen within the mesentery representing recurrence of the patient’s disease at three months post exploratory laparotomy and start of chemotherapy. It also noted ill-defined density within the pelvis posterior to the uterus, also indicating disease recurrence. To obtain a target, molecular testing was ordered. Subsequently, molecular testing and chromosome analysis were done to direct treatment. Approval was obtained, and the patient was started on temozolomide. Results showed that immunohistochemistry of the mass was positive for MLH1, MSH2, MSH6, and PD-L1 and variation of undetermined significance of BRCA1 and AXIN2. The patient was started on immunotherapy with bevacizumab through port-a-cath. At this point, she reported symptoms of shortness of breath accompanied by chest pain both at rest and with exertion, as well as bilateral pedal swelling. She also developed chemotherapy-induced hypertension. Following cardiology recommendations, the patient was started on carvedilol, amlodipine, and as-needed Lasix for bilateral pedal edema. An echocardiogram was also ordered as bevacizumab is known to cause hypertension and left ventricular dysfunction toxicity. The transthoracic echocardiogram showed mitral and tricuspid valve regurgitation. It also noted a normal left ventricular systolic function with an ejection fraction of 55%-60%. The patient’s current chemotherapy regimen, which included IV bevacizumab and temozolomide, was stopped, and she was started on gemcitabine and docetaxel. The patient was also informed of the palliative nature of the treatment. Three months later, the patient decided she would like to take a break from the chemotherapy, and the treatment was stopped. Immature teratomas are malignant tumors that occur in the first few decades of life. Tumor grading of immature teratomas is based on the amount of immature neural tissue present. The prognosis of immature teratomas depends mainly on the staging system provided by the International Federation of Gynecology and Obstetrics (FIGO) . The othAccording to Iavazzo et al., fertility-sparing management is suggested as the standard of care of immature teratomas in young female patients . Young fThe patient was first started on bleomycin, etoposide, and cisplatin chemotherapeutic regimen. Etoposide inhibits DNA synthesis by interfering with the activity of topoisomerase II. It is predominantly active against cells in the late S and G-2 phases of the cell cycle. Cisplatin is an alkylating agent that acts in a cell-cycle nonspecific manner. Bleomycin is an antineoplastic antibiotic that binds to DNA and causes scissions of DNA strands . Tumor pUnlike the previous treatment, bevacizumab is immunotherapy aiming to prevent pathologic angiogenesis. According to Wilson, pathologic angiogenesis enhances the growth of solid tumors and causes chronic inflammation, cartilage damage, atherosclerotic plaque formation, and scarring of the eye . Wilson Tumor progression continued despite bevacizumab, and the patient was started on gemcitabine and docetaxel. As reported by de Sousa Cavalcante et al., the most common mechanism of action of gemcitabine is by inhibiting DNA synthesis . Per FarImmature teratomas mainly affect young female patients. Prognosis worsens with delay in diagnosis of disease at later stages. The fertility-sparing approach is the standard of care, and various treatment options should be thoroughly discussed with patients. These options commonly involve surgery and chemotherapy. Immunotherapy with drugs such as Bevacizumab is promising and most effective when initiated early. The five-year survival rate is excellent for stage I immature teratomas, but this survival rate decreases drastically for grade 1, 2, and 3 tumors.