Disorders of sexual differentiation are rare congenital conditions in which thechromosomal, anatomic or gonadal sex development is atypical. In some of thesepatients, chromosomal sex is inconsistent with phenotypic sex; in other cases,the phenotype is not classifiable as either male or female, resulting in acondition known as ambiguous genitalia. These are very complex cases in whichdiagnostic certainty is not always possible. A multidisciplinary team includinggeneticists, pediatricians, radiologists is certainly needed to approach thesepatients. We present the case of an 18-year-old boy with an XY karyotype,ambiguous genitalia, uterus and blind-ending vaginal pouch. The patient had notbeen previously diagnosed with a disorder of sex development. The patientunderwent a panel of genetic analyses and diagnostic imaging investigations.Magnetic resonance imaging was decisive for the identification of the internalgenital organs, especially the uterus. At the end of investigations, the patientwas diagnosed with 46,XY disorder of sex development. Our purpose is tounderline the role of imaging in the diagnosis and management of congenitaldisorders of sex differentiation. Disorders of sex development (DSD) are congenital conditions in which thechromosomal, anatomic or gonadal sex development is atypical.DSD can be classified as disorders of chromosomal, gonadal, or phenotypic sexorigin.Today, DSD is divided into three major categories based on patient karyotype: sexchromosome DSD; 46,XY DSD; and 46,XX DSD. The etiology of DSD is multifacetedand and may include genetic and environmental factors  development (true hermaphroditism)Disorders of androgen synthesis or action:- Defects in androgen biosynthesis - Defects in androgen action (e.g.: complete androgeninsensitivity syndrome)- LH receptor defects - Disorders of Anti-Müllerian hormone (AMH) and AMHreceptor (Persistent Müllerian Duct Syndrome)- Other  ; imaging (abdomin-pelvic ultrasoundUS and Magnetic Resonance Imaging)  and the difficulty offering a precise prognosis forindividual patients  at various ages and, in some instances, pubertalsuppression. Pubertal induction is usually performed at ages 10-12 in girls and11-13 in boys, depending on maturity, desire, and informed consent of patientsand parents. Options for hormonal treatment for patients with DSD are limited bypractical considerations, such as pharmacokinetic properties and effectivenessof steroid hormone preparations, and availability  and FSH 53.66 Mui/ml (v.n. mans 0.95-11.95). Estradiol and testosteronelevels were normal  (Tanner stage II).Lower abdominal and testicular ultrasound images did not show a prostate or uterusand ovaries; the testicles were on site and were smaller than average (left 15x8mmand right 10x6mm) with asymmetric vascularity, reduced to the right. Epididymiscould not be evaluated . A pelviThe imaging reports described the following: a vagina ending in a blind pouch in theperineum between the urethra and rectum; uterine remnants between the rectum and thebladder ; startinGenetic counseling is required. After informing the patient, the decision was made toperform karyotype analysis. Peripheral blood was collected and analyzed withshort-term lymphocyte culture (48-72 hours). The result was a normal male karyotype46,XY . The folSOX9, NR0B1(DAX1), WNT4, NR5A1(SF1);Analysis by Multiplex Ligation-dependent Probe Amplification (MLPA) to search forgene duplications/deletions Next-Generation Sequencing (NGS) of the coding exons and related junctions of the 27genes involved in sexual differentiation.No anomaly was found.Pelvic ultrasonography (US) is the most frequently used imaging method in the initialassessment of DSD in patients of all ages.Magnetic resonance imaging (MRI) and US are considered equally sensitive in theevaluation of intrapelvic structures, although MRI is more sensitive than US in theevaluation of the gonads ,followed by mosaicism (As reported in the literature, in our case ectopic gonads had intermediate signalintensity on T1-weighted images and intermediate signal intensity on T2-weightedimages. Streak gonads are difficult to detect and can be seen as low intensitystripes on T2-weighted images of the gonads (The external genitalia presented as a penis-like clitoris; for We have not found any disorders in androgen synthesis or action. No pathogeneticvariants of genes implicated in DSD were found. Thus, the most probable diagnosis,although not certain, is true hermaphroditism. True hermaphroditism is a DSD definedas the presence of ovarian tissue and testicular tissue in the same individual.Definitive diagnosis requires an exploratory laparoscopy, an invasive procedure notalways accepted by patients. It is not uncommon for these patients to remain withouta definitive diagnosis; in fact, some studies have shown that only 50% of 46,XYchildren with DSD will receive a definitive diagnosis (The management of DSDs is complex and requires a multidisciplinary medical approach.Medical decisions for patients with DSD still lack evidence-based principles, and anearly and correct diagnosis of DSD carries important clinical implications forpatient upbringing, the reduction of potential risk of malignancy, and the decisionon the correct timing for a gonadectomy.It is important that a child with ambiguous genitalia be evaluate by amultidisciplinary team using a coordinated approach to arrive at a timely diagnosis,so that proper gender assignment can be made early in life. Imaging plays animportant role in demonstrating the anatomy and potential effects on other organs,in particular when a pathognomonic genetic alteration is not identified, as in ourcase.US is the first line exam for the evaluation of DSD. Our case underlines that MRI isneeded in most cases for a correct diagnosis of gonadal morphological features,especially when a pathognomonic genetic alteration is not identified. MRI does notinvolve exposure to ionizing radiation, allows multiplanar image reconstruction, andprovides excellent soft tissue contrast resolution.Radiologists play an essential role in diagnosis and follow-up, considering the highrisk of tumors developing in these patients. US every six months or MRI every yearis recommended to patients with dysgenetic gonads.


Paratesticular liposarcoma (PLS) causes scrotal mass changes, rarely in the urinary system. Before surgery, PLS causes scrotal mass changes that are difficult to distinguish from other causes. There has been a report of a giant paratestis liposarcoma resection and refusal to undergo orchiectomy. A 65-year-old man presented with finding the left scrotal mass after 2 years. Physical examination showed that the left scrotal mass was obviously difficult to retract. Pelvic CT showed that the left scrotal mass and flaky fat density shadow accompanied with left inguinal hernia. During surgery, laparoscopic exploration was performed to rule out inguinal hernia, and a scrotal exploration was also performed concurrently. The intraoperative frozen pathology considered lipogenic tumor, and the patient’s wife refused to undergo simultaneous left radical orchiectomy. Later the mass was completely removed, and postoperative pathology confirmed paratestis liposarcoma. During a 15-month routine follow-up, the tumor did not recur locally or metastasize distantly. PLS should be focused on early diagnosis and treatment, preoperative examinations and postoperative pathology should be combined, and highly personalized treatment will be implemented. Liposarcoma (LPS) is a malignant tumor of adipocyte differentiation, approximately 20% of all soft tissue sarcomas are liposarcomas . LiposarApproximately 7% of all scrotal tumors are paratesticular liposarcomas (PLS).Only about 272 cases have been reported worldwide . Pelvic CT plain scan and enhancement showed a left inguinal hernia, with content being a small amount of mesenteric fat tissue in the left inguinal region., a mass-like fatty density lesion was seen in the left testicle, which was considered to be a vascular muscle adipose tumor or a fatty tumor , depth of infiltration and histopathology classification . As the In conclusion, rarely diagnosed giant PLS results in delayed treatment due to misdiagnosis, there is no consensus on its management currently. The diagnosis may be challenging but a thorough history, exam and image  with histopathological findings will provide a clear diagnosis. Radical orchiectomy with extensive local excision and high ligature of the sperm cord provides the best results when a preoperative diagnosis or high suspicion is present. The value of adjuvant treatment with radiotherapy and chemotherapy has yet to be determined because there have been no large-scale PLS clinical trials conducted yet. Long-term monitoring is required as local recurrence and distant metastases are possible.The original data that supports the conclusions of the article will be provided by the authors without any improper reservation.Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article. Written informed consent was obtained from the participant/patient(s) for the publication of this case report.QZ compose the manuscripts. SG provide literature review. SW and YL study supervision. QH and SL formal analysis. CG review and editing. All authors contributed to the article and approved the submitted version.


Spinal subarachnoid hemorrhage (SSAH) is a rare condition that can cause spinal cord or nerve root compression and permanent neurologic damage. The reported etiologies include trauma, vascular malformations or aneurysms, coagulopathies, neoplasms, autoimmune disease, and spontaneous hemorrhage. If there is evidence of neurologic deterioration, it is commonly managed as a surgical emergency, but cases of conservative management have also been reported. In this case series, we present three patients who suffered from SSAH. The first was a spontaneous cervical SSAH that occurred following cardiac catheterization, the second was a spontaneous thoracolumbar SSAH in a patient with a known history of coagulopathy, and the third was a thoracolumbar SSAH that was caused by a dural arteriovenous fistula (dAVF). All three patients exhibited neurologic deficits and thus underwent emergent decompression and hematoma evacuation. The patient with the dAVF also required open ligation of the fistula. Following surgical intervention, all three patients regained at least partial neurologic function, but one patient developed symptomatic arachnoid cysts that required further intervention. The presented case series highlights the importance and time-sensitivity of surgical decompression in patients experiencing neurologic deficits from SSAH. These cases underscore the urgency of timely neurosurgical intervention to mitigate neurologic impairment and add insights to the existing literature on this rare condition. Spinal subarachnoid hemorrhage (SSAH) is a rare but serious disease with a multitude of etiologies. A large review article and meta-analysis published in 2003 reported only 96 cases of SSAH between 1826 and 1996, displaying the disease's historical rarity [The clinical presentation of SSAH is usually a neurologic deficit associated with spinal cord or nerve root compression that varies depending on the level of the lesion and associated pathology. Most patients that present with symptoms of mass effect on spinal neural elements present acutely with a complete or incomplete spinal injury, and less than 15% present in a subacute or chronic manner ,3. MagneSSAH is typically managed as a surgical emergency if there are signs of acute neurologic deterioration, and patient recovery hinges upon preoperative clinical status as well as early intervention ,2,4-6. TCase oneA 60-year-old otherwise healthy female who had three weeks of syncope with exertion and lightheadedness when extending her neck underwent an elective heart catheterization as part of her syncopal work-up. The procedure was uneventful, aside from coronary artery spasm. Two hours after waking from anesthesia, she reported an acute onset of severe headache that radiated into her neck and bilateral shoulders. At that time, her headache was associated with weakness in her right arm and bilateral lower extremities, as well as hypoesthesia and paresthesia in her entire body below her chest. Non-contrast CT head and CT angiography (CTA) head/neck did not show any evidence of intracranial hemorrhage or vascular abnormality. The next day, the hypoesthesia and paresthesia had ascended to affect her arms, and she was found to be incontinent. Contrast-enhanced MRI brain, cervical spine, and thoracic spine demonstrated non-enhancing, lobulated, intradural extramedullary lesions, which were consistent with intradural hematomas Figure . She wasTwo months postoperatively, she was taken back to the operating room for a catheter cerebrospinal angiogram, which did not show any vascular pathology. Follow-up MRIs with and without contrast were done three months postoperatively, which showed myelomalacia in her cervical spinal cord extending from C5 through T1 without any target for surgical decompression. At that time, the strength in her left leg was 4/5, and the strength in her right leg was 2/5. Case twoA 45-year-old female with a history of hypertension, as well as multiple deep vein thromboses (DVTs) and pulmonary embolisms (PEs) that she had previously been taking warfarin for, presented to the emergency department with several days of abdominal pain, nausea/vomiting, and diarrhea. She was admitted to the hospital with pneumonia and possible myocardial infarction, so she was given aspirin 325 mg) and started on therapeutic anticoagulation. On the evening of her admission, she reported that her legs had been weak for four days, and she had been experiencing urinary retention and incontinence for two days. On neurologic exam, she had a thoracolumbar sensory level  and objective weakness in her bilateral lower extremities . She had normal reflexes in her bilateral lower extremities with no clonus, and her rectal tone was intact. CT of her lumbar spine did not show any acute pathology, but follow-up MRIs were done given her neurologic exam, and it revealed an intradural hematoma that spanned from the mid-thoracic region through her entire lumbar spine  presented to an outside facility with three days of progressively worsening severe low back pain to the point that he was unable to walk. A CT of his lumbar spine was done, which was concerning for epidural hematoma, so he was transferred to our institution for a higher level of care. On arrival here, he also reported new onset urinary retention. On neurologic exam, his strength was severely pain-limited but near full strength in his left lower extremity and right ankle. Patellar and ankle reflexes were 3+ bilaterally, and his sensation was intact. MRIs of his thoracic and lumbar spine were done on arrival at our institution, which showed an intradural extramedullary hematoma, possible infarct at the conus given T2 hyperintense signal, and abnormal enhancement surrounding the spinal cord near T11 concerning for a dural arteriovenous fistula  throughout her thoracic spine. SAC was first observed in 1943 in a patient that developed lower extremity weakness and had a history of cerebral subarachnoid hemorrhage as a result of an underlying coagulopathy . SACs arThe third case presented in this article of a 65-year-old male on full dose aspirin for a history of myocardial infarction is an example of a vascular malformation (dAVF) leading to SSAH. Dural AVF is one of the more common spinal vascular malformations, but other vascular causes include arteriovenous malformations, spinal aneurysms, coarctation of the aorta, and Moyamoya disease ,15. SpinThis case series highlights the diversity in presentation, etiologies, and outcomes of SSAH. All three cases have unique clinical aspects and possible associated risk factors for SSAH. We acknowledge that our case series has a limited follow-up interval of the patients, and further research is necessary to determine long-term outcomes of various treatment modalities.SSAH is a rare pathology with a wide range of etiologies, including vascular malformations, tumor-associated hemorrhages, trauma, and underlying systemic disease. While the diagnosis of an intradural hematoma has become more accessible due to the widespread availability of advanced imaging, determining the underlying cause and best treatment remains a challenging clinical scenario for clinicians. In this case series, we highlight the diagnostic challenges, the various possible etiologies, and the outcomes of early surgical evacuation of the hematoma in the setting of neurologic compromise. We also report a potential long-term complication of the inflammatory response associated with subarachnoid blood - the formation of clinically significant arachnoid cysts. Given the limited literature available on SSAH, this case series assumes clinical importance as it contributes valuable insights to the existing body of knowledge.


Intravascular leiomyomatosis (IVL) is a histologically benign smooth muscle tumor arising from the uterus that can spread through the pelvic veins and, on rare occasions, extend as far as the heart via the inferior vena cava. Despite its benign characteristics, it can behave like a malignant tumor leading to significant morbidity and even mortality if left untreated.The patient is a 42-year-old woman with a past medical history of uterine leiomyomas. She presented with heavy bleeding and frequent spotting; therefore, she went to her gynecologist. After further evaluation, a mass within the uterus that expanded into the pelvic veins, inferior vena cava, and right atrium was discovered. After the complete removal of the mass, the patient underwent full recovery. IVL with cardiac extension was the final diagnosis.Although IVL is rare, it must be considered in women who underwent previous hysterectomies or myomectomies and present with symptoms of right heart failure. The ideal therapy will need the aid of a multidisciplinary team and will depend on the patient’s symptoms, previous operative history, the tumor’s extension, and resectability. Intravenous leiomyomatosis is a rare benign uterine tumor that can proliferate throughout the venous system without invading it . Its treWe present the case of a 42-year-old woman with a uterine mass that expanded into the pelvic veins, inferior vena cava, and right atrium. After the complete removal of the mass, the patient underwent full recovery. IVL with cardiac extension was the final diagnosis.Patient is a 42-year-old woman with a past medical history of uterine leiomyomas. She underwent several fertility treatments in her youth, including ovarian stimulation, hormonal therapies (GnRH agonist), and myomectomies (two were performed at ages 30 and 33 and measured 2 cm in diameter) overall; nevertheless, all failed. She got tired of doctors and treatments so she stopped going to follow-up visits.In recent years, her leiomyomas had grown to a worrisome size, and in the last year, she began to experience heavy bleeding, frequent spotting, and anemia, so she went to her gynecologist.On clinical examination, an otherwise healthy patient with no signs of right-sided heart failure symptoms, including swelling and shortness of breath, was encountered; her vital signs were normal however, on her lower abdomen, a palpable mass 10 × 10 cm mass was discovered, along with some mild discomfort in the lower abdomen and pelvis. No other masses or lymph nodes were found at that time. Due to this, complementary exams were needed, Complete blood count showed hemoglobin of 11 mg/dl, EKG was normal and a contrast-enhanced computed tomography revealed a 17.6 × 7.2 × 10.3 cm uterus. It had multiple soft tissue density fibroids with central calcifications, the largest one measured 4 × 5 × 2 cm and was in close contact to the parametrial vessels.Upon reviewing the venous phase of the CT, we were surprised to discover that within the lumen of the right internal iliac vein, the right common iliac vein, and the inferior vena cava (IVC), there was a mass causing a heterogeneous filling defect that extended from the uterus into the right atrium. All other organs were normal at that time.With these findings, a transesophageal echocardiogram (TEE) was done, revealing that the 3 × 1.5 cm mass moved freely into the cardiac chambers without compromising the tricuspid valve or any other structure  were initiated to begin the thoracic approach. Once a deep hypothermic circulatory arrest was achieved, a veno-cavotomy was done in the infrarenal IVC, which allowed the complete removal of the 5 × 1.5 cm tan-cream luminal mass from the IVC and iliac veins. The venacavatomy was closed in a one-layer fashion, and then attention was placed on the heart. The right atrium was exposed, and the 1.5 × 14 cm whiteish solid mass was removed from the right atrium and inferior vena cava under TEE guidance. Once the tumor was removed, CPB was re-established. Fig. A The heaThe excised masses consisted of 2 pieces each measuring 5 × 1.5 cm and 14 × 1.5 cm.  on postoperative day three and admitted to the general ward. After she tolerated a full diet, she was discharged under strict surveillance and with long-term nonsteroidal estrogen receptor antagonist therapy.Two years after the initial surgery, the patient is doing well without signs of recurrence.Intravascular leiomyomatosis (IVL) is an extremely rare mesodermal cell tumor . Since tThis disease can grow over many years from the uterus into the pelvic vein, renal vein, vena cava, and finally into the right atrium , 3. In aThe clinical manifestations of IVL are related to the extent of involvement , 3. WhenMacroscopically, intravenous leiomyomatosis is a nodular growth with wormlike extensions into the uterine veins , 7. HistUltrasound, doppler, CT, and Magnetic resonance imaging are needed for diagnosis and confirm disease extension , 6. ThesSurgery is the most effective treatment, but it depends on whether the tumor can be excised entirely , 4. SincComplete removal is the only way to ensure a favorable prognosis, yet, close surveillance is needed as tumor recurrence (22.2 to 30%) can occur up to 15 years after the surgery.  Postoperative hormonal therapy with antiestrogens, aromatase inhibitors, hysterectomy, or bilateral salpingo-oophorectomy is recommended to prevent this complication.  As it was done with our patient.IVL is a rare, aggressive condition that, due to its indolent course, can range from being entirely asymptomatic to experiencing symptoms of right heart failure. Therefore, early detection and close surveillance are vital to ensure a good recovery.Although IVL is rare, it needs to be in our differential. Especially on women who underwent previous hysterectomies or myomectomies. The ideal treatment will require the joint efforts of a multidisciplinary team and will depend on the patient’s symptoms, previous operative history, the tumor’s extension, and resectability. If complete resection is achieved, recurrence is rare, yet if complete removal is not achieved, recurrence can appear in up to a quarter of patients.


Mixed epithelial-stromal tumours (MESTs) are a rare biphasic tumour that frequently arise in women from the renal and urogenital tract. They are also seen in men but are exceptionally uncommon with only few cases reported to originate from the seminal vesicles. Malignant transformation of its epithelial or stromal components is possible; however, by in large, these tumours are benign in nature. We report the case of a 48-year-old man with no remarkable medical or surgical history who presented with a huge expanding pelvic and intra-abdominal mass that required extensive surgical management including a pelvic exenteration. Histopathological analysis concluded the diagnosis of benign MEST originating from the seminal vesicles with no malignant features. No further systemic therapy was recommended for our patient. Given the technical intricacy in the operative resection of this tumour, we aim to present our findings and surgical management of this complex MEST. Mixed epithelial-stromal tumours (MESTs) are a rare and mostly benign neoplasm that arise commonly from the urogenital tract . MESTs aA 48-year-old man with 6 months of worsening abdominal distension was referred with a diagnostic abdominal Computed Tomography (CT) scan revealing a large mass in his abdomen and pelvis. He reported no urinary or faecal obstructive symptoms, vomiting, loss of appetite, or any positional or exertional dyspnoea. There was no relevant personal or familial medial history and no previous abdominal surgeries. On examination, his abdomen was markedly distended  with a pHis full blood count, biochemistry, inflammatory, and tumour markers were within the normal range. The CT scan revealed a large pelvic extraperitoneal mass displacing bowels with heterogenous enhancing solid and non-enhancing cystic components . MRI evaAn incisional biopsy was performed via a mini-laparotomy, which reported an initial diagnosis of angiomyofibroblastoma.The patient underwent an elective laparotomy, which showcased a very large abdominal mass . InitialThe resected tumour measured 65 cm × 36 cm × 9 cm and weighed 8.08 kg. There was a well demarcated outer capsule . The tumHistopathology reported a biphasic tumour comprised an epithelial surface  with cysImmunohistochemistry of the stroma showed positive staining for oestrogen, progesterone, desmin, and smooth muscle actin. The glandular component was cytokeratin positive with intact basal cells and stained negative for prostate markers .Post-surgery the patient remained well and was discharged 2 weeks later with planned radiological follow-up.MESTs are a rare soft-tissue tumour that commonly arise from the urogenital tract with biological behaviours ranging from benign to malignant .MEST typically affects perimenopausal women with a preference to involve renal parenchyma. In males, it is exceptionally rare and is associated with a history of hormonal therapy (androgen deprivation/oestrogen therapy) often coupled with managing prostate cancer .et al. [et al. [et al. [MEST occurrence in the SV is very rare. A recent systematic review by Babar et al.  reported [et al.  who repo [et al. , 8. Comp [et al. , the Wor [et al.  proposinMalignant MESTs have been described using a three-tiered grading system (low/intermediate/high) based on stromal atypia, mitotic activity, pleomorphism, and necrosis , 12. WitMEST diagnosis heavily depends on histopathology as tumours of the pelvic/urogenital tract can have multiple aetiologies and, even with sophisticated imaging modalities, clear differentiation can be difficult , 14.Interestingly, our initial biopsy of the tumour was favoured to represent a myofibroblastic lesion such as an angiomyofibroblastoma or cellular angiofibroma due to the biopsy only containing stromal component. However, upon reviewing the entire resected specimen, both stromal and epithelial components were present, which fits well with MEST of SV.Differentiating benign from malignant MEST is important, with the former commonly exhibiting stromal blandness and the absence of mitotic activity . HistoloTherapeutic management is centred on complete surgical resection of tumour-burdened viscera with a systematic review describing resection using open (57%), laparoscopic 26%), or robotic (17%) techniques %, or rob, 15.However, the extensive size and involvement of pelvic organs, as seen in our case, has not been reported and highlights the need for adequate imaging and preparation for extensive pelvic organ dissection, and where required, as we found, resection of those organs to obtain a complete resection of the tumour.We report a very rare benign tumour of the seminal vesicles that presented with a massive abdominal component emanating from the locally advanced pelvic component. To achieve complete resection, the man required a pelvic exenteration. The prognosis from this extensive benign tumour should be excellent.


This ChatGPT-driven case report describes a unique presentation of neurosarcoidosis. The patient, a 58-year-old female, initially presented with hoarseness and was found to have bilateral jugular foramen tumors and thoracic lymphadenopathy. Imaging revealed significant enlargement and thickening of the vagus nerve and a separate mass of the cervical sympathetic trunk. The patient was referred for an ultrasound-guided biopsy of the abnormal neck masses to establish a pathologic diagnosis. The patient subsequently underwent neck dissection for exposure of the vagus nerve and isolation of the great vessels in preparation for a transmastoid approach to the skull base. The presence of multifocal tumors prompted the need for a biopsy, which ultimately revealed sarcoid granulomas in the nervous system. The patient was diagnosed with neurosarcoidosis. This case highlights the potential for sarcoidosis to affect the nervous system, with multiple cranial nerve involvement, seizures, and cognitive impairment. It also emphasizes the need for a combination of clinical, radiological, and pathological findings for an accurate diagnosis of neurosarcoidosis. Additionally, this case highlights the utility of natural language processing (NLP), as the entire case report was written using ChatGPT. This report serves as a comparison of the quality of case reports generated by humans versus NLP algorithms. The original case report can be found in the references. Neurosarcoidosis is a rare and complex disorder that affects the central and peripheral nervous systems. It is a form of sarcoidosis, a chronic inflammatory disease that can affect multiple organs in the body. The prevalence of neurosarcoidosis is unknown, but it is estimated to affect between 0.5% and 5% of patients with sarcoidosis. The exact cause of neurosarcoidosis is not well understood, but it is believed to be an immune-mediated disorder. The symptoms of neurosarcoidosis can vary widely and can include neurological symptoms such as seizures, headaches, and cognitive impairment, as well as visual disturbances and spinal cord involvement. The diagnosis of neurosarcoidosis can be challenging, as it requires a combination of clinical, radiological, and pathological findings .Radiological findings in neurosarcoidosis include abnormalities in the brain and spinal cord, such as lesions, nodules, and inflammatory changes. These lesions are typically non-specific and can mimic other neurological disorders such as multiple sclerosis or brain tumors. Magnetic resonance imaging (MRI) is often used to detect these lesions and can show high signal intensity on T2-weighted images. Computed tomography (CT) scans can also be used to detect lesions in the brain and spinal cord, but they are less sensitive than MRI.Histologic findings in neurosarcoidosis include the presence of non-caseating granulomas in the nervous system tissue. These granulomas are composed of inflammatory cells and are characteristic of sarcoidosis. However, a biopsy is not always necessary for the diagnosis of neurosarcoidosis and the diagnosis can be made based on the combination of clinical, radiological, and laboratory findings ,2.Bilateral vocal fold paralysis (BVFP) is a condition in which both vocal folds are unable to move normally. It is a rare disorder that can cause symptoms, including hoarseness, breathiness, and difficulty speaking. The anatomic basis of BVFP is complex and involves the interaction of several structures, including the vagus nerve and the muscles of the larynx .The patient was a 58-year-old female who first presented to a clinic in March 2019 with a complaint of hoarseness. The hoarseness began in 2015 and was initially thought to be related to her gastroesophageal reflux disease. Imaging obtained by her laryngologist showed bilateral jugular foramen tumors and thoracic lymphadenopathy  signal hyperintensity in the bilateral deep cerebral white matter, which is most likely an atypical manifestation of mild chronic small vessel white matter ischemic disease. The meninges and extra-axial spaces were normal, major and arterial venous flow voids were normal, and the sinuses and mastoids were normal, but there was a chronic defect in the medial left orbital wall.The operative report describes a surgical procedure performed on a patient to investigate and sample a suspected vagal schwannoma, a benign tumor that develops on the vagus nerve. The procedure was performed under general endotracheal anesthesia and the patient was placed in a supine position. The surgical team used a hockey stick incision to gain access to the neck area, and nerve monitoring electrodes were placed in the palate, trapezius, and face for neuromonitoring. The surgical team then dissected the various layers of tissue in the neck, carefully identifying and preserving the external and internal carotid artery, the internal jugular vein, the hypoglossal nerve, and the spinal accessory nerve.As the surgical team entered the carotid sheath and began to trace the vagus nerve proximally, they observed that the nerve appeared normal in appearance above the level of the carotid bifurcation, but that it was enlarged and fibrous in appearance below the bifurcation, consistent with a schwannoma Figure . The nerGiven the presence of multifocal tumors in multiple nerves and the bilateral nature of the patient's disease, the decision was made to not attempt a complete resection of the schwannoma at this time. Instead, the surgical team took samples of the vagus nerve tumor for pathology and sent them for frozen pathology examination. The frozen pathology confirmed the presence of a schwannoma.The surgical team then closed the neck and the patient was transferred to the post-anesthesia care unit in stable condition with no complications reported. The patient was extubated to room air and the postoperative plan was to keep the patient stable in the post-anesthesia care unit.The pathology report described the results of a biopsy and resection of a lymph node and nerve in the left neck area. The lymph node was described as enlarged and having mild reactive changes. The nerve was described as having granulomatous inflammation, which is characterized by the presence of granulomas, which are clusters of inflammatory cells. The report notes that special stains have been ordered to investigate the possibility of an infectious etiology, but the results are not yet available.The addendum to the report states that special stains and immunohistochemistry were performed on the nerve tissue and that these tests did not reveal any discernible organisms. However, the S100 immunohistochemistry did highlight bundles of nerve twigs running through the dense fibrous tissue and the giant cells Figure . Based oThis case of BVFP caused by neurosarcoidosis is novel in that it is the first case of its kind. The patient, a 58-year-old female, initially presented with hoarseness, which was initially thought to be related to her gastroesophageal reflux disease. Imaging showed bilateral jugular foramen tumors and thoracic lymphadenopathy, leading to a referral to an otology/neurotology specialist. The patient underwent a neck dissection, which revealed an extreme enlargement and thickening of the vagus nerve and a separate mass of the cervical sympathetic trunk. Biopsies were sent for frozen pathology, which was suspicious for a schwannoma. The patient was referred to a colleague in neuroradiology for an ultrasound-guided or CT-guided biopsy of the abnormal neck masses to establish a pathologic diagnosis. CT scan revealed heterogenous left and right parotid glands, symmetrically prominent right and left submandibular glands, an unremarkable thyroid gland, symmetrically prominent pharyngeal tonsils, and a patent airway. MRI of the brain showed bilateral rounded enhancing cerebellopontine angle mass lesions and non-enhancing scattered foci of abnormal T2-FLAIR signal hyperintensity in the bilateral deep cerebral white matter.Neurosarcoidosis is a rare complication of sarcoidosis, a chronic inflammatory disease of unknown cause that can affect multiple organs. In neurosarcoidosis, sarcoid granulomas (clumps of inflammatory cells) form in the nervous system, typically in the brain and spinal cord. The exact mechanisms of neurosarcoidosis are not well understood, but it is believed that the granulomas cause inflammation and damage to the affected nerves and tissues ,2,4.This case is unique in its findings in that it is a patient with an unusually high number of neurological symptoms, including multiple cranial nerve involvement, seizures, and cognitive impairment. This combination of symptoms is uncommon and highlights the potential for sarcoidosis to have a severe impact on the nervous system .Lastly, as this report is a fully ChatGPT/NLP-generated case report, it can be compared against the original article, which serves as the benchmark for case report quality . VariousIn conclusion, this case is the first known case of BVFP caused by neurosarcoidosis. The patient presented with hoarseness and despite the initial diagnosis, further investigation revealed additional abnormalities. This case highlights the importance of considering a wide range of differential diagnoses when evaluating patients with hoarseness and the need for a thorough diagnostic workup.


Sections revealed a multilocular cyst with trabeculated walls and immunohistochemical staining positive for cytokeratin (AE1/AE3) consistent with BMPM. One year post operatively he remains asymptomatic; however, his interval computed tomography (CT) scan revealed several sub centimeter nodules that either represents small splenules or neoplastic implants. These will be followed with close interval imaging.Multicystic peritoneal mesothelioma (BMPM) is a rare, usually benign tumor that arises from peritoneal mesothelial cells that most commonly occurs in women of reproductive age. Pathogenesis of these tumors is thought to come from chronic inflammation from prior surgery, endometriosis, trauma, or recurrent peritonitis. Here we report a case of primary splenic BMPM in a 20-year-old male with no past medical or surgical history and without any typical risk factors for this condition. He underwent an open splenectomy without complication. Pathology revealed an 18 × 4 × 11 cm Multicystic peritoneal mesothelioma (BMPM) is a rare, usually benign tumor that arises from peritoneal mesothelial cells, with just over 200 cases reported in the literature . BMPM us3 complex multilobulated septated cystic lesion in the upper pole of the spleen. He had initially presented to his primary care physician with 4 months of upper abdominal pain with no other symptoms such as nausea, vomiting, fevers, or shortness of breath. He denied any history of trauma, recent illness, or sick contacts. He had never traveled outside of the country and had been working in heating, ventilation, and air conditioning for 1 year.A 20-year-old male with no major past medical or surgical history presented for surgical evaluation after an abdominal computed tomography (CT) scan showed a 12.0 × 10.3 × 9.1 cmAs part of his workup, he underwent an abdominal ultrasound showing a 12 cm cystic or hemorrhagic lesion, likely splenic in origin, with an adjacent 5.6 cm complex septated lesion within the spleen. Subsequent abdominal CT showed a 12 cm in greatest diameter complex cystic lesion within the spleen with peripheral and septal calcification with no solid enhancing component . His bloGiven the size and unclear etiology of the lesion, he was scheduled for an open splenectomy. He was referred to infectious disease preoperatively for workup and for consideration of infectious etiologies, where he was started on albendazole for potential hydatid disease. Before surgery, he met with his primary care physician where he received the Menveo, Hib, and Prevnar 13 vaccines.He was taken to the operating room and underwent an upper midline laparotomy. After inspection of the abdominal cavity and confirmation of no additional peritoneal lesions or significant adhesions to the spleen, the splenic vessels were controlled with 0-silk ties. The spleen was then mobilized and removed without violation of the splenic capsule and the abdomen closed . He had 3 spleen weighing 855 g, with a 10.5 cm in greatest dimension cyst occupying 75% of the splenic surface. Sections revealed a multilocular cyst with copious cloudy yellow fluid and trabeculated walls. Immunohistochemical staining of the cyst wall was positive for cytokeratin (AE1/AE3) consistent with a benign multilocular mesothelial cyst without malignancy.Pathology revealed an 18.0 × 4.0 × 11.0 cmHe was seen at 1, 6, and 12 months following surgery without complaints. A 1-year interval abdominal CT  showed sMulticystic peritoneal mesothelioma is a rare condition with very few reported cases in men , 11, 12.As BMPM is a benign process, symptoms usually occur once the tumor has grown sufficiently to cause mass effect on the surrounding organs or viscera, with non-specific abdominal complaints representing the most common presentation. Treatment includes complete surgical resection. Due to the estimated recurrence rate of 50% in women and 33% in men, some centers have elected to treat BMPM with complete cytoreduction followed by hyperthermic intraperitoneal chemotherapy (HIPEC) , 16, 17.Presented here is a rare case of a splenic benign multicystic peritoneal mesothelioma in a 20-year-old male. With no prior surgical or medical history, the cause of BMPM in this patient does not fit the traditional etiological theory of chronic inflammation, highlighting a gap in our current understanding of the pathogenesis of BMPM. He was treated with an open splenectomy without HIPEC and at 1 year has recovered well with several sub-centimeter nodules in the splenectomy bed on CT that will be followed with an interval CT scan in 1 year.


Mullerian duct anomalies are prevalent in 4-7% of the female population and come in many different shapes and forms. A lot of effort has already gone into trying to classify these anomalies, and some are still found that do not match any of the subcategories. We report a 49-year-old patient, presenting with abdominal pressure and recent onset of abnormal vaginal bleeding. A laparoscopic hysterectomy was performed, which revealed a U3a-C(?)-V2 mullerian anomaly with three cervical ostia. The origin of the third ostium remains unclear. Early and correct diagnosis of Mullerian anomalies is of the utmost importance to provide individually tailored care and to avoid unnecessary surgeries. Mullerian duct anomalies are defined as congenital malformations of the female genital tract stemming from embryological maldevelopment of the Mullerian ducts. They have a prevalence of 4-7% and are as such a rather common benign affliction. They consist of a myriad of conditions, depending on the level and severity of maldevelopment. Symptoms can vary from asymptomatic to extreme dysmenorrhea and fertility problems. Diagnosis of these malformations is mostly made by clinical examination and ultrasound, sometimes aided by hysterosalpingogram, hysteroscopy, MRI or laparoscopy. In most cases there is no need for therapeutic interventions, except to preserve fertility or to alleviate symptoms in the case of obstructive malformations .We present a rare case of a 49-year-old patient, complaining of abdominal pressure and abnormal vaginal bleeding. Diagnostic work-up did not reveal a clear cause. Following a hysterectomy, the pathology report revealed a Mullerian anomaly, which we classified as U3a-C(?)-V2 with three cervical ostia. Two of these ostia lead to two separate endometrial cavities; the third merely consisted of a rudimentary cervical canal opening into a rudimentary obstructed vaginal canal, which is extremely rare.The patient was a 49-year-old female complaining of abdominal pressure and recent onset of abnormal uterine bleeding.Surgical history included a cholecystectomy, 3 Caesarean sections and a tubal ligation. Previous pelvic ultrasound had raised the possibility of a bicornuate uterus. A laparoscopy performed in 1985 had also suggested endometriosis. The patient also had a history of pulmonary embolism, and she was being treated with rivaroxaban due to a recent recurrence. Unfortunately, no operative reports were available from previous surgeries, nor any pre-operative ultrasound images.The patient presented to the clinic complaining of abdominal pressure and abnormal uterine bleeding for 6 months, having previously being amenorrhoeic on the progestogen-only pill.Clinical examination revealed a normal cervix and vaginal canal. Pelvic ultrasound revealed a suspected bicornuate uterus with a thin endometrium in both horns, no evidence of myomas and uterine measurements of 76mm x 34mm x 70mm. Only one ovary could be visualised. On the right side there was a multilocular cystic mass of unclear origin, measuring 77mm x 50mm x 51mm. There was no evidence of ascites on the pelvic ultrasound.Abdominal CT scan revealed no signs of diverticulitis or other gastrointestinal pathology. No pathologically enlarged lymph nodes were observed.An MRI was performed which revealed a Mullerian duct anomaly described as a bicornuate uterus and absent right kidney. The mass was described as a cystic or tubular structure on the right of the uterus, containing blood products. Suggested differential diagnoses included a dilated right fallopian tube containing blood, or a rudimentary right ureter. The right ovary could not be visualised.After counselling, the patient consented to a laparoscopic total hysterectomy, with investigation and removal of the unknown mass.During surgery, a bicornuate uterus, possibly bicorporeal was noted. The right ovary was present and normal in appearance, albeit very small. The left ovary was absent, presumably resected during earlier endometriosis surgery. The right ureter could not be visualised. Upon first inspection of the abdomen, no evidence of the unknown mass was found.While performing a standard colpotomy, there was a sudden evacuation of a moderate amount of brown mucous fluid. Upon further inspection, we found a rudimentary closed off vaginal canal, which was also resected.Inspection of the uterus directly after transvaginal extraction revealed a U3a-C(?)-V2 Mullerian anomaly with 3 cervical ostia.Careful probing of the ostia with a hysterometer revealed that the left and right ostia led to an endometrial cavity. The middle cervical canal measured only 3 cm.The pathology report confirmed the presence of two separate uterine cavities, each with their own cervical canal and cervix. The two hemi-uteri were fused. The report also confirmed the presence of an extra, intermediate cervical ostium, ending in a small, dilated cervical canal. Pathological analysis of the vaginal canal revealed signs of prolonged pressure and chronic inflammation. Findings were also consistent with uterine adenomyosis.There were no immediate postoperative complications. At 4 and 8 weeks post-surgery, the patient reported relief of her preoperative symptoms. Permission to publish this case was obtained.Preoperatively, we believed this to be a bicornuate uterus. However, after surgery our diagnosis had to be adjusted. Final diagnosis in this patient was a U3a-C(?)-V2 Mullerian anomaly with three cervical ostia and an obstructed hemivagina next to a normal vaginal canal. This is a rare form of OHVIRA syndrome . Typically, OHVIRA is associated with a complete bicorporal uterus, though in this case there was only a partial divide.The absence of symptoms or reproductive problems resulted in a late diagnosis at age 49, normally these kinds of anomalies are usually detected at an earlier stage of life. It is worth noting that the patient received a diagnosis of endometriosis at a very young age (15 years old), which could have been an indicator of retrograde menstrual bleeding, caused by occlusion of one hemi-uterus. No haematometra was ever observed in this patient, but the partial bicorporeal uterus could have resulted in a connection at the isthmic level, allowing drainage of both hemi-uteri. Renal agenesis was undiagnosed until presentation and imaging, and the extent of the patient’s Mullerian anomaly was only fully diagnosed during surgery. The reason for the onset of symptoms at a more advanced age is unclear.To the best of our knowledge, this is the first time that a case with possible triple cervices has been described. There are, however, several limitations to this case report. First and foremost, the specimen was not examined by a pathologist experienced in Mullerian anomalies.Secondly, the absence of previous surgical reports is unfortunate. Lastly, our full diagnosis was obtained during surgery, so no hysteroscopy or hysterosalpingography could be performed. The extent of the Mullerian anomaly was possibly underestimated in this case, due to the fact that only one cervix was seen during the preoperative examination.This patient was diagnosed with right renal agenesis and a right obstructed hemi-vagina, which is also known as OHVIRA syndrome. This syndrome represents 7.1% of all female genital tract malformations. Differential diagnosis of the mass includes a Gartner’s cyst or ectopic ureter. However, given the uterine anomaly, renal agenesis, and the absence of urinary incontinence, OHVIRA syndrome is most likely. A higher prevalence of endometriosis in these patients has been described, which was indeed also present in this patient’s history. . StudiesA differential diagnosis is more difficult to establish for the third cervical ostium. A fistula following previous Caesarean section seems less likely, considering that the cervical canal did not connect to either of the endometrial cavities or the abdominal cavity. Furthermore, the third ostium and endocervical canal matched the histological properties of the two other normal cervices. A Gartner’s pseudocyst could also be considered, but this would mean that one cervix was atretic, with no connection to the endometrial cavity. However, this patient had two different, distinct cervices with a clear connection to their respective endometrial cavities. Another very rare possibility is duplication of Mullerian tract tissue at the level of the cervix . An atreNo literature references were found for a triple cervical canal therefore it is not possible to estimate the prevalence of this Mullerian anomaly.It is not even possible to provide a full classification of this specimen using the ESHRE (European Society of Human Reproduction and Embryology). The uterus can best be categorised as class U3, as it is a uterus with an abnormal fundal outline. Yet, our pathology report described the specimen as a uterus didelphys: two separate cavities with fusion of the hemi-uteri, making this a class U3b. Macroscopically however, the divide seemed to be above the level of the cervices, so we would classify this as U3a. The vaginal septum was longitudinal and obstructing, which would be sub-class V2. The cervices, however, don’t fit any of the subclasses. Using the ESHRE classification, this would be U3aC?V2 .The ASRM (American Society for Reproductive Medicine) developed a new classification system in 2021, based on the 1988 AFS (American Fertility Society) system. In this new system, they stress that not all possible anomalies are included. Given the variety and complexity of Mullerian anomalies, it is likely that more will have to be added in the future. They describe possible variations on the uterine malformations with obstructed hemi-vagina, none of which suggest the existence of triple cervices. They do, however, make note of the association between ipsilateral renal agenesis and obstructing longitudinal vaginal septum. It is possible that only one cervix will be observed during clinical examination due to this septum. Imaging options include ultrasound, MRI and HSG, but in our case the third cervical canal was not observed on ultrasound or MRI. However, further inspection of the MRI images after the surgical findings, reveals that a triple canal cervix can be visualised .The clinical impact of a possible triple cervix is unclear. In this case, the patient had no problem conceiving. She did, however, experience symptoms of endometriosis at a very early age, and delivered her children through Caesarean section, though the reason for this was unknown.Early and correct diagnosis of these types of Mullerian anomalies is of the greatest importance to identify other cases, and to reduce the risk of severe endometriosis. Renal agenesis should always raise the suspicion of Mullerian anomalies and instigate further examination. MRI imaging, hysteroscopy and HSG could shed more light on the extent and severity of the suspected anomaly. Using this approach, perhaps symptoms can be alleviated using minimally invasive surgery .Our patient specifically requested a hysterectomy and did not want to continue exploring non-surgical options. If this had been a patient desiring to get pregnant, a transvaginal resection of the vaginal septum could have been considered .In conclusion, firstly, this is a unique case where there are three suspected cervical canals. This could raise questions about the embryological development of the cervix. Given the absence of fertility problems and lack of symptoms in earlier stages of life, this phenomenon may be under reported. The need for therapeutic intervention is unclear.Secondly, in a patient with unilateral renal anomaly or agenesis, one must always consider the possibility of an associated Mullerian anomaly. Correct preoperative diagnosis could contribute to avoiding the need for major surgery.


Ganglioglioma (GG) is a WHO-grade 1 glioneuronal neoplasm. It is well differentiated with a slow-growing pattern and is composed of a combination of neoplastic ganglion and glial cells. Anaplastic ganglioglioma (AGG) is an extremely rare malignant variant of ganglioglioma, which is not included in the new WHO classification; however, the term is used to talk about gangliogliomas with data of malignancy. AGGs usually occur in children and young adults and are associated with high recurrence and mortality.The authors describe the case of a 62-year-old woman with AGG. She presented with cacosmia, vertigo, nausea, and focal-onset seizures with secondary generalization. Magnetic resonance imaging (MRI) revealed an intra-axial lesion in the left temporal lobe. She underwent microsurgical resection guided by electrocorticography (ECoG), and a diagnosis of AGG based on microscopic morphology and immunohistochemical analysis was obtained. She was discharged a few days after surgery with subtotal resection of the lesion, no additional neurological deficit, and adequate seizure control.AGG is a very rare and poorly studied entity. It is currently a controversial term used to refer to gangliogliomas with signs of malignancy. It occurs mainly in children and young adults with temporal lobe epilepsy. Total resection is the best prognostic factor, given the unknown efficacy of radiotherapy and chemotherapy. In our case, the patient was an adult woman with a subtotal resection followed by concomitant radiotherapy and chemotherapy, obtaining a mean survival similar to that reported in the literature, so it can be thought that there is a benefit obtained with chemotherapy and radiotherapy despite having performed a subtotal resection of the lesion. Further studies are needed to establish clear diagnostic criteria for AGG, and a multicenter database of AGGs is necessary for a better understanding of the pathology and to offer the best treatment and prognosis. Gangliogliomas (GGs) are very rare mixed glio-neural tumors that represent approximately 1.3% of all primary brain tumors and 0.4% of central nervous system neoplasms .Anaplastic ganglioglioma (AGG) is a rare malignant variant that is poorly characterized and, right now, is not included in the new WHO classification but is used to talk about gangliogliomas with data of malignancy . It reprA 62-year-old woman with a previous history of systemic arterial hypertension and hyperthyroidism treated with hemithyroidectomy and thiamazole was admitted to our hospital. The patient complained of cacosmia, vertigo, nausea, and focal-onset seizures with secondary generalization that began about one month before admission. Previously, a neurologist prescribed levetiracetam 3 g per day and lamotrigine 50 mg twice daily with inadequate seizure control. Neurological examination exhibited global aphasia with memory preservation and no additional neurological deficit. Serum laboratory analyses were normal. A cranial MRI scan revealed an intra-axial and diffuse lesion in the left temporal lobe measuring 15 mm × 20 mm × 19 mm, associated with perilesional edema to rule out infiltration of the neoplasm. The lesion was isointense in T1-weighted and hyperintense in T2-weighted sequences without contrast enhancement. Spectroscopy showed an elevation of creatine and choline with a decrease of N-acetyl aspartate , a guided resection was performed to achieve a temporal lesionectomy without changes in the ECoG. Mesial structures were not included to avoid memory impairment, and hemostasis was secured, closing the dura in a watertight fashion.Intraoperative pathological analysis reported only a low-grade astrocytoma without another specification.The definitive study was described as a lesion of 3.8 cm × 2.6 cm × 1.8 cm, weighing 12 g, light brown on the external surface, lobed, opaque, of soft consistency, with thin blood vessels; on the inner surface, it is light brown, rough, and soft. Histopathology showed an admixture of neoplastic ganglion and glial cells with microvascular proliferation and mitosis. Ganglion cells showed clustering and cytomegaly; some of them showed binucleated forms; and glial cells showed astrocytic morphology. The immunohistochemical analysis showed positivity for glial fibrillary acidic protein (GFAP), Wilms’ tumor 1 protein (WT1), p53, S100 protein, Ki67 (15%), neurofilament protein (NFP), and synaptophysin . She is being treated with levetiracetam 1 g every 12 hours with a follow-up by the Department of Neurology, and she has had no seizures since surgery. The oncology department treated her with adjuvant radiotherapy (60 GY) in 30 fractions of the residual lesion with concomitant chemotherapy with temozolomide 120 mg daily.Gangliogliomas is a well-differentiated, slow-growing glioneuronal neoplasm composed of a combination of neoplastic ganglion and glial cells that is characterized by genetic alterations that cause MAPK pathway activation. Previously, AGG was included in the WHO classification as a malignant subtype of GG. Currently, this term is excluded in the new 2021 WHO classification; however, it is mentioned that there are malignant variants of ganglioglioma that continue to be called anaplastic ganglioglioma in the absence of a new term. The WHO states that more studies are needed to confirm the existence of AGG through molecular studies and define clear diagnostic criteria. At the moment, since we do not have a clear term to refer to this type of high-grade version of GG, we choose to continue using the term AGG .AGG in adults is an extremely rare entity. In a retrospective study of 326 primary intracranial gangliogliomas, they only found 17 cases that were classified as AGG .The etiology and pathogenesis of this disease remain unclear; however, the cell of origin is noted to be a glioneuronal precursor ,9. The BThe WHO suggests that a molecular diagnosis looks for a BRAF p.V600E mutation or another change in the MAPK pathway, as well as a ganglioglioma methylation profile, ideally with a mutated IDH .Histopathologic analysis can be challenging. GG are biphasic tumors composed of an admixture of neuronal and glial elements; they may be intermixed or geographically separate. Neuronal elements are composed of dysmorphic ganglion cells that may demonstrate abnormal clustering and lack of cytoarchitectural organization, cytomegaly, perimembranous aggregation of Nissl substance, or binucleated forms seen in <50% of cases. The glial component may resemble a fibrillary astrocytoma, an oligodendroglioma, or a pilocytic astrocytoma. They often demonstrate an infiltrative growth pattern microscopically and frequently include dystrophic calcification either within the matrix or as a neuronal or capillary incrustation. Focal cortical dysplasia may be associated with GG .The immunophenotype neuronal markers such as MAP2, neurofilament, chromogranin A, and synaptophysin demonstrate the neuronal component. There is no specific marker to differentiate neoplastic neurons from their normal counterparts. GFAP and OLIG2 highlight the neoplastic glial cell component. Also, CD34 may be expressed in 80% of the GG, and it is not normally expressed outside of vascular endothelial cells in the mature brain. Ki-67 is typically <5% in GG, being higher in malignant variants (as in our patient), and it is limited in the glial component. VE1 antibody identifies BRAF p.600E mutant protein in gangliogliomas with this genetic alteration [The imaging characteristics of gangliogliomas are varied; they can be diffuse, poorly delineated, or solid masses with remarkable heterogeneous enhancement. AGG, such as in this case, tends to be isointense or hypointense on T1-weighted and hyperintense on T2-weighted, and contrast enhancement is generally irregular . MRI speGGs progress locally, and AGGs tend to progress distantly ,14. In oIn this case, the diagnosis was made based on the compatible histopathological and immunohistochemical analysis of a tumor with glial and neuronal components, as well as the patient´s clinical features and imaging study, which were also compatible. Likewise, because the lesion presented characteristics of malignancy both in the histological analysis (microvascular proliferation and mitosis) and in the immunohistochemistry (high Ki-67 index), we decided to diagnose it as an AGG, which is a term that, despite not being found in the current WHO classification, has been used to refer to the malignant variant of GG. It was not possible to perform the molecular analysis due to the lack of resources at the institution; however, we understand the need to perform such analysis to rule out differential diagnoses with other types of neoplasms.Nevertheless, we report a rare case of an older adult woman. AGGs tend to occur at a young age, and most are males under 40 years of age. Patients with AGGs are associated with a poor survival rate (median about 24.7 months). The primary prognostic factor is resectability at presentation, with a median survival of 44 months in those with gross total resection (GTR) , which iAnaplastic gangliogliomas are very rare tumors that can occur at any place in the neuroaxis. It is a poorly characterized entity with multiple differential diagnoses. Diagnosis based on morphological and immunohistochemical analysis, looking for the neuronal and glial components and characteristics of malignancy , is acceptable when no molecular analysis is responsible. The treatment of choice is GTR, or the greatest safe tumor resection possible, with adjuvant radiotherapy and chemotherapy. Follow-up must be carried out in conjunction with the neurology service for the management of epilepsy, as well as surveillance with periodic imaging studies of the neuroaxis. Further investigation is needed to unify the diagnosis and treatment of mixed malignant neuroglial tumors.


Citrobacter diversus was detected in the culture from the mass and any epithelial components were not detected by biopsy. For the diagnosis of primary abdominal wall abscess, the patient underwent surgical drainage because antibiotic treatment was ineffective. The abscess disappeared promptly after the drainage. Thirteen months after the first treatment, another primary abdominal wall abscess was noted in the lower right abdomen. The abscess also promptly disappeared with surgical drainage. Primary abdominal wall abscess is difficult to diagnose because of its rarity. Prompt diagnosis and drainage are important to prevent exacerbation.Primary abdominal wall abscess is extremely rare and difficult to diagnose because abdominal wall abscesses usually occur secondary to malignant tumors or inflammatory diseases. We experienced a case of an 80-year-old man with an asynchronous primary abdominal wall abscess with recurrence. Both abscesses were successfully treated with surgical drainage. A patient without any history of cancer or trauma presented to our department with right upper abdominal pain. His laboratory data showed an abnormal high inflammatory response, and computed tomography revealed a 40 × 30 mm mass formed in the rectus abdominis muscle of the upper right abdomen. The mass had no continuity with the surgical scar after cholecystectomy or intra-abdominal organs.  Most abdominal wall abscesses occur secondary to malignant tumors, inflammatory diseases, surgical site infections, and so on ,2. SolitHere, we present a case of asynchronous multiple recurrent PAWA, which was successfully managed through surgical drainage. The first drainage was delayed due to the diagnostic difficulty, but the second drainage was performed immediately and the patient recovered sooner. Additionally, we reviewed cases in the literature to discuss the clinical features of this rare condition.An 80-year-old man presented to our department in July 2020 with a complaint of right upper abdominal pain for about a month. He had atrial fibrillation for which he was taking anticoagulant agents, chronic renal failure, and diabetes mellitus controlled by subcutaneous insulin injections; he underwent open cholecystectomy five years ago; and had no history of cancers, bruises, or trauma. He did not take any immunosuppressants or steroids. He had no fever but had slight swelling and redness in the upper right abdomen with tenderness. Laboratory studies showed an abnormal white blood cell count (WBC) of 10,900/μL, neutrophils at 81.9 %, C-reactive protein (CRP) at 25.58 mg/dL, blood urea nitrogen at 68.1 mg/dL, and creatinine at 3.10 mg/dL. Serum tumor markers, including carbohydrate antigen 19-9 (CA19-9) and carcinoembryonic antigen, were within the normal ranges. Differential diagnoses were hematoma, tumor (primary or metastasis), or abscess. Computed tomography (CT) revealed a round 40 × 30 mm mass with fluid collection located below the abdominal wall, followed from the rectus abdominis muscle Figure . The masCitrobacter koseri was detected in the culture. None was detected in the blood culture. The patient was hospitalized and treated with antibiotics for over two weeks: tazobactam/piperacillin for a week, cefozopran hydrochloride for a week, and levofloxacin hydrate for five days. However, the abscess did not improve completely, and the laboratory studies showed an abnormal WBC of 10,100/μL and CRP at 10.74 mg/dL. Therefore, surgical drainage was performed 19 days after the first visit. Under local anesthesia, a 5-cm incision was made, and the rectus abdominis muscle was divided to reach the mass. The intra-operative findings showed the abscess cavity located in the rectus abdominis muscle , the T2-weighted image showed a high signal, and the diffusion-weighted image showed a low signal mass Figure . We perfe Figure . The conSubsequently, the patient underwent surgical drainage immediately because conservative treatment with antibiotics had been ineffective in the first treatment. The abscess cavity was located in the rectus abdominis muscle, a large amount of white pus was excreted, and no bacteria were detected. After drainage, the abscess disappeared promptly, and he was discharged after three weeks. He had no postoperative complications, but his discharge was postponed due to the adjustment of the oral dose of warfarin potassium.PAWAs are extremely rare. Muscles have a lesser risk of infection due to abundant blood flow. Inflammation does not easily spread into the muscles, even with sepsis . Most inThe mechanism of PAWA remains unclear. Previous reports have listed muscle damage, diabetes mellitus, and liver cirrhosis as risk factors for PAWA ,4,7. ComGoodman et al. reported that images of abdominal wall abscesses are characterized by cellulitis and fluid collection, with gas in 30% . HoweverThe treatment of muscle abscesses is surgical drainage and antibiotics. Small abscesses could only be treated with antibiotics alone . HoweverA comprehensive diagnostic assessment, including bacterial culture and cytology, is needed for PAWA. Patients with PAWA are sometimes compromised, thus delay of treatment lead to exacerbated infection. For a case with an isolated abdominal wall abscess, PAWA should be considered as one of the diagnoses. Further, early drainage should be considered.


Pilomatrixoma (PMX), also known as calcifying epithelioma of Malherbe, is a rare benign neoplasm that arises from the hair matrix cells, commonly in the head, neck, and upper trunk regions, infrequently affecting upper and lower extremities. It has to two peaks of presentation: under 20 years of age or between 50 and 65 years of age, slightly more common in females. The neoplasm exhibits diverse clinical manifestations and is frequently subject to misdiagnosis with alternative dermatological diseases. We present an atypical case of PMX affecting the upper extremity of a 62-year-old female patient. Surgical removal of the affected tissue under local anesthesia was performed, and subsequent histopathological analysis confirmed the presence of PMX. Based on the literature search we performed, we found out that this pathology is underreported in Jordan, with only one study published describing this tumor in the maxillofacial region. Physicians should be aware of this condition and its different presentations to include it in the differential diagnosis of suspected cases to provide the appropriate management and follow-up. Pilomatrixoma (PMX) is a benign growth primarily observed in the skin, emerging chiefly from the matrix cells of hair follicles situated in the dermal or subcutaneous layers. First identified and recorded by Malherbe and Chenantais in the late 19th century, early interpretations suggested it to be a calcifying epithelioma associated with the sebaceous glands -3.The occurrence of this neoplasm is significantly evident in two specific age groups: individuals aged between 0 and 20 years and a less prominent occurrence in the age group of 50 to 65 years . It typiDiagnosing this tumor can be challenging due to its uncommon clinical features like multiple lesions or locations deep within the skin -10. IdenA significant study done by Moehlenbeck in 1973 reviewed 140,000 samples of skin tumors and highlighted the uncommon nature of PMX, which accounted for only 0.12% of all skin tumor specimens analyzed .Given its benign characteristics and infrequent occurrence, medical professionals need to have a deep understanding of PMX to ensure precise diagnosis and effective management, thus avoiding misidentifying it as other benign or malignant skin lesions .In the Jordanian community, PMX remains notably rare and understudied, with only one documented study examining its clinicopathologic characteristics in the maxillofacial region . There iA 62-year-old female patient, with a known case of controlled hypertension, presented to the general surgery clinic complaining of a small left arm mass for over 30 years, with a recent increase in pain, but no change in the size or color of the overlying skin. There is no history of trauma. No other masses were present. The patient has a negative family history of benign and malignant tumors.On examination, a nodule was seen on the left arm posteriorly, approximately 3 cm proximal to the elbow joint, with no overlying skin discoloration or scar. The mass measured approximately 2.0 cm × 1.0 cm × 5 cm and was superficial, stony-hard in consistency, mildly tender on palpation, and slightly mobile with limited fixation to underlying tissue but not to overlying skin.A left-arm X-ray was requested, and it showed a hyperdense oval lesion, measuring about 2.0 cm × 1.0 cm, approximately 3 cm above the elbow joint with variable calcification , computed tomography (CT), and magnetic resonance imaging (MRI), has been documented in rare cases. These imaging modalities have proven beneficial when the tumor is located in atypical sites, such as the parotid or breast . AlthougA punch or excisional biopsy is the preferred diagnostic method for most cutaneous diseases in cytology. The fine-needle aspiration (FNA) technique is a fast and dependable method for procuring a preoperative diagnosis for specific cutaneous lesions ,27. CertThe histological examination of the affected tissue reveals basaloid cells arranged in either grouped or separated patterns by pink fibrillary material. Additionally, squamous, ghost, and multinucleated giant cell co-occurrence is observed. These histological features indicate the possibility of PMX diagnosis . AccordiThe likelihood of distant metastasis and malignant transformation into pilomatrix cancer is quite low . In mostPMX is an infrequent presentation in general surgery clinics. Upon searching published literature, we found this condition to be underreported in Jordan, which reflects the lack of knowledge and awareness of this pathology. Physicians should be aware of this neoplasm and its possible transformation into malignancy, including it in the differential diagnosis of suspected cases, and ensuring that appropriate treatment and follow-up are provided.


Ovarian vein thrombosis (OVT) is a rare condition most frequently seen in the immediate postpartum period. Typical symptoms include pelvic pain, fever, and abdominal mass. Although a rare diagnosis, OVT can potentially cause fatal complications; thus, early recognition and prompt treatment is important.We present a case of a 30-year-old lady, G4P3A0, previously healthy, who presented at 29+3 weeks to the hospital for preterm premature rupture of membrane. The patient had a Cesarean section (C-section), which was complicated with uterine atony and massive bleeding controlled by emergent intrauterine balloon tamponade and uterine artery embolization. The next day, the patient complained of new-onset right lower quadrant abdominal pain, for which she had an abdominal and pelvic computed tomography (CT) scan that showed OVT. Thus, she was started on anticoagulants, and discharged home.OVT is a rare disorder that has been described as occurring mainly during the postpartum period, after pelvic surgery, or in women with gynecological malignancies. Clinical features of OVT include fever, abdominal pain and tenderness, and a palpable abdominal mass. Diagnosis can be obtained using CT, magnetic resonance (MR), or ultrasound (US) Doppler. Treatment includes a combination of anticoagulants and antibiotics. Mortality is low nowadays. HighlightsOvarian vein thrombosis (OVT) is a rare condition most frequently seen in the immediate postpartum period.Typical symptoms include pelvic pain, fever, and abdominal mass.Diagnosis can be obtained using computed tomography (CT), magnetic resonance (MR), or ultrasound (US) Doppler.Treatment includes a combination of anticoagulants and antibiotics.Mortality is low nowadays.It is a case report study.1. Typical symptoms include pelvic pain, fever, and abdominal mass2. OVT can be associated with gynecological malignancies, pelvic surgeries, pelvic inflammatory disease (PID), and hypercoagulable states2. The incidence of idiopathic OVT is exceedingly rare. Only a few cases with no known etiology have been reported to date in healthy patients. Although a rare diagnosis, OVT can potentially cause fatal complications; thus, early recognition and prompt treatment is important.Ovarian vein thrombosis (OVT) is a rare condition most frequently seen in the immediate postpartum period. OVT has been reported in ~2% of births by Cesarean section (C-section) and in 0.05–0.18% of vaginal birthsA 30-year-old lady, G4P3A0, previously healthy, non-smoker, and non-alcohol user, presented at 29+3 weeks to the hospital for preterm premature rupture of membrane. The patient had early and regular prenatal care; she used to see her health care provider every 4 weeks and denied any history of complicated pregnancy or delivery. Her outpatient medications included prenatal vitamins and iron supplements. Upon presentation, the patient’s temperature was 37°C, heart rate was 89 beats per minute, blood pressure was 105/60 mmHg, respiratory rate was 24 breaths per minute, and oxygen saturation was 98% on room air oxygen. She was clinically and hemodynamically stable with regular heart sounds, clear lungs, soft abdomen, and no lower limb edema. Laboratory tests showed hemoglobin 9.7, hematocrit 30, and a white blood cell count of 10 000 with 90% neutrophils.The patient was started on dexamethasone 6 mg intramuscular injection every 12 h in order to accelerate the maturation of the fetal lungs and rocephine 2 g intravenously once daily. On day 3, the patient had a C-section, which was complicated with uterine atony and massive bleeding; she thus received 4 l of lactate ringer solution, 4 units of packed red blood cells, 2 units of fresh frozen plasma, and 1 pool of platelets with tranexamic acid and calcium gluconate.Hemostasis could not be achieved except after emergent intrauterine balloon tamponade and uterine artery embolization.2 of 97% on room oxygen. The patient was somnolent but conscious and cooperative, oriented to time, place, and persons; pallor was noted with slight jugular venous distension, but otherwise clear lungs on auscultation. The uterine drain showed a small amount (50 cc) of serosanguinous fluid. Laboratory tests revealed hemoglobin 10, hematocrit 31, international normalized ratio (INR) 1.66, partial thromboplastin time (PTT) 39, D-dimer 185 052, and creatinine and electrolytes within the normal range.The patient was then transferred to the intensive care unit (ICU) for observation. Upon arrival in the ICU, the patient’s vitals were stable, with a blood pressure of 90/60 mmHg, pulse of 98, and SpOOn the next day of ICU stay, the patient complained of new-onset right lower quadrant abdominal pain for which she had an abdominal and pelvic computed tomography (CT) scan. The CT scan showed an enlarged uterus without hematoma, pelvic intraperitoneal drainage, and dilated and tortuous right ovarian vein with surrounding amount of fluid suggestive of OVT Fig. . A compl3. However, it has been more frequently detected in recent years due to the widespread use of radiological imaging and its technical improvement3. OVT complicates ~0.01–0.18% of pregnancies 6–8 and is more common after a Cesarean delivery than a vaginal delivery4. Postpartum OVT occurs with a peak around 2–6 days after delivery or miscarriage/abortion, and in 90% of women, it arises within 10 days6. Pregnancy-related OVT is usually diagnosed in women in their early 30s8, while the other types of OVT involve older females with mean age ranging from 40 to 60 years in different studies9. The right ovarian vein is more frequently involved (70–80% of cases), due to the anatomical difference in venous drainage; occasionally, the thrombosis can be bilateral (around 10% of cases)10.OVT is a type of unusual-site venous thromboembolism (VTE) involving the female gonadal veins. OVT is a rare condition, with an incidence of around 60 times less common than lower limb deep vein thrombosis (DVT)8. Finally, a percentage of 4–16% of OVT has no causative factor, and these forms are classified as idiopathic OVT7.The most common risk factors for OVT are pregnancy, recent surgery, pelvic infections, estrogen-containing hormone treatment, and malignancy, although with variable prevalence in different studies. Malignancies associated with the development of OVT include mainly genitourinary cancers (such as uterine or ovarian tumors), followed by gastrointestinal cancers11. The pain may radiate to upper abdomen, the flank, or the groin12. The most typical finding, although reported in only 46% of cases10, is a palpable cord-like abdominal mass, extending from the ovary to the paracolic gutter and corresponding to the thrombosed ovarian vein12. Nonspecific symptoms such as nausea, vomiting, anorexia, malaise, and ileus are reported, but they are usually mild7. The presence of OVT has been evidenced in more than half of the women with puerperal pelvic thrombophlebitis. Asymptomatic OVT is frequent in patients with malignancies, in whom it might be an incidental finding on CT scans performed during follow-up visits13.The most common symptom of OVT is lower quadrant abdominal pain, usually on the side of the thrombosed vein14. US Doppler is usually the first-line imaging, due to its safety, low cost, and wide availability. However, US is an operator-dependent imaging and can often be inconclusive due to obesity or the presence of abdominal meteorism, which interferes with the visualization of the ovarian veins16. CT scan (contrast-enhanced CT or CT venography) can better identify the presence and the extension of the thrombosis and is nowadays the reference imaging16. However, CT scanning involves the use of iodinated contrast medium and ionizing radiation, and contraindications include renal insufficiency, pregnancy, and previous contrast allergy. Specificity of CT has been reported to be 62–99% and sensitivity to be 77–100%17. MR has the benefit of not using iodinated contrast medium or ionizing radiation; however, disadvantages include higher costs, longer execution time, and non-availability for immediate use. Unenhanced MR can allow the direct visualization of the thrombus16. Specificity of MR has been reported to be roughly 100% and sensitivity ~92–100%; thus, MR is indicated when the suspicion of OVT persists and CT scan is inconclusive16.Diagnosis of OVT can be obtained using CT, magnetic resonance (MR), or ultrasound (US) Doppler. However, there is no definite consensus regarding the gold standard test, as only a few studies specifically evaluated sensitivity and specificity18. Furthermore, anticoagulation at a therapeutic dose for 1–3 months is suggested18. The guidelines of the British Committee for Standards in Haematology recommend an anticoagulant treatment duration of 3–6 months for women with postpartum OVT and suggest no treatment for incidentally detected isolated OVT in cancer patients19. Broad-spectrum antibiotics are crucial if there is a concomitant infection17. Anticoagulation follows the principles of the management of usual-site VTE. In the study by Assal et al.8, women with isolated OVT received parenteral anticoagulation in the acute phase, with either low-molecular-weight heparin (LMWH) or unfractionated heparin, and the majority of them were subsequently switched to vitamin K antagonists (VKAs). The novel direct oral anticoagulants (DOACs) were not specifically evaluated in patients with OVT, as unusual-site VTE was excluded from the large phase III randomized controlled trials evaluating their efficacy and safety in DVT and pulmonary embolism (PE)20. Anticoagulant treatment duration was variable in the different studies. Expert opinions suggest a definite anticoagulant treatment duration of 3 months for symptomatic postpartum OVT17. There are only a few studies reporting the risk of bleeding of OVT patients3. There are a few reports regarding the use of thrombolytic drugs 21. However, due to the associated high risk of bleeding, they should be reserved for selected cases of women with massive thrombosis. Retrievable inferior vena cava filters are indicated only when there are absolute contraindications to the anticoagulant treatment 12. Surgical treatments (e.g. ovarian vein excision or ligation) are rarely performed nowadays22.Treatment of OVT includes a combination of anticoagulants and antibiotics. According to the guidelines of the Canadian Society of Obstetricians and Gynaecologists addressing pregnancy-related VTE, parenteral broad-spectrum antibiotics should be commenced as soon as the diagnosis of OVT is confirmed and administered for at least 48 h after fever resolution, considering longer duration in septicemic patients9. In approximately a quarter of patients, the thrombosis might have already extended into other veins at the time of diagnosis7. Rare complications of OVT include ovarian infarction with potential influence on fertility1, and obstruction of the right ureter, which can lead to hydronephrosis and renal failure23. The survival of both OVT and DVT women was decreased when compared with the general population3. Mortality due to OVT is less than 5% nowadays due to the use of antibiotics and anticoagulants23.Possible complications described in patients with OVT include thrombus progression into the left renal vein (for left OVT) or into the inferior vena cava (for right OVT) or PE, recurrent VTE, and pelvic congestion syndromeOur patient was diagnosed with a postpartum OVT. She was initially started on LMWH and later transitioned to oral warfarin therapy. The patient’s abdominal pain subsided soon after starting anticoagulation, and she was discharged with instructions to follow-up with hematology and a plan to repeat imaging in 3 months to determine the need for continuing anticoagulation.24.The work has been reported in line with the SCARE 2020 criteriaOVT is a rare disorder that has been described as occurring mainly during the postpartum period, after pelvic surgery, or in women with gynecological malignancies. Clinical features of OVT include fever, abdominal pain and tenderness, and a palpable abdominal mass, combined with nonspecific gastrointestinal symptoms. Since OVT is one of the causes of puerperal fever, it should be suspected in women with fever and pelvic abdominal pain within a week after delivery, especially if they had a Cesarean section. US Doppler is usually the first-line imaging, as it can be easily performed for the evaluation of puerperal fever or abdominal pain. However, MR or CT scan is suggested to confirm the presence and extension of OVT, because US can often be inconclusive. Due to a treatment combination of anticoagulation and parenteral broad-spectrum antibiotics, mortality is low nowadays. While no anticoagulation has been suggested for incidentally detected cancer-associated OVT, anticoagulant treatment duration of 3–6 months has been recommended for postpartum OVT. Evidence is lacking on the optimal management of other types of OVT.Ethical approval was not applicable.Written informed consent was obtained from the patient’s family for the publication of this case report and any accompanying images. A copy of the written consent is available for review upon request by the Editor-in-Chief of this journal.The authors declare that the research is not funded by any funding agency.Z.E.M. and B.D.: contributed to the management of the case and the conceptualization and planning of the case report; Z.M.: helped in the preparation of the manuscript. Both authors read and approved the final manuscript.The authors declare that there are no conflicts of interest.Not applicable.Zeinab El Mawla.No additional data are available.Not commissioned, externally peer-reviewed.Not applicable.


An odontogenic keratocyst (OKC) was first described by Philipsen in 1956. They are benign cysts of odontogenic origin that behave aggressively and have a high recurrence rate.The present case report describes an unusual presentation of OKC as a multi-locular lesion in the anterior mandible.A 14-year-old male patient was referred to the oral maxillofacial surgery clinic in Tabuk Specialist Dental Centre by his orthodontist to evaluate a radiolucent lesion that had been identified in his lower anterior teeth during an OPG examination. The patient was medically fit and had multiple previous dental restorations. An intraoral examination revealed a small bony expansion in the cystic lesion on the buccal side. The panoramic radiograph showed well-defined multi-locular radiolucencies in the lower left canine area, despite there being no tooth resorption; however, there was a slight divergence noted between the teeth. An excisional biopsy was performed, and the subsequent histopathological examination revealed a cystic lesion diagnosed as an odontogenic keratocyst. The six-month follow-up OPG showed that the site had completely healed without any lesions recurring.OKCs can present at any age, irrespective of gender. The differential diagnosis included a lateral periodontal cyst or a radicular cyst when the tooth was not vital. In this case, the six-month follow-up OPG following surgery revealed no recurrence, although a close follow-up is recommended because of the high recurrence rate. An odontogenic keratocyst (OKC) was first described by Philipsen in 1956 . OKCs prRadiographically, OKCs usually appear as a well-defined unilocular or multilocular radiolucency bounded by corticated margins. Unilocular lesions are the predominant variant, whereas the multilocular variant is seen in approximately 30% of cases and mostly in the mandibular arch ,7. The mIn a systematic review, Johnson et al. showed that the patient treated by enucleation has the highest recurrence rate at approximately 30%, followed by marsupialization alone (approximately 18% recurrence rate) .The present case report describes an unusual presentation of OKC as a multilocular lesion in the anterior mandible. A 14-year-old male patient was referred to an oral maxillofacial surgery clinic in the Tabuk specialist dental center by his orthodontist to evaluate the radiolucent lesion that had been identified in his lower anterior teeth during the panoramic radiographic examination. The patient was medically fit, while their dental history revealed multiple dental restorations. No abnormality was identified in either the physical or additional oral examinations. An intraoral examination identified a little bony expansion in the area of a cystic lesion on the buccal side.The panoramic radiograph illustrated well-defined multi-locular radiolucencies in the lower left canine area, even though no tooth resorption was observed, although a slight divergence was noted between the teeth Figure .The differential diagnosis for this case included a lateral periodontal cyst, an odontogenic keratocyst, a botryoid odontogenic cyst, and an ameloblastoma.After discussing the possible diagnosis with the patient, the patient was referred for oral maxillofacial surgery for a biopsy, where an excisional biopsy was performed to confirm the diagnosis. The complications of the surgical procedure were discussed with the patient, and written informed consent was obtained from the patient. Next, an excisional biopsy was conducted under local anesthesia with 1.8 mL of anesthetic solution (lidocaine HCl 2% and epinephrine 1:100000) administered intraorally via an injection to the inferior alveolar and mental nerves of the left side of the mandible. The cystic lesion was enucleated by the surgeon and submitted for histopathological evaluation , while the conservative treatments include simple cyst excision or marsupialization/decompression, which can be followed by a second surgery. In our case, the choice of treatment was the enucleation of the cystic lesion along with curettage . MoreoveOKCs can be presented in a wide age group with no sex priority. The most common location of OKCs is on the posterior side of the mandible. Conservative surgical excision can be a treatment option for small cysts. Close follow-up was recommended because the OKCs have a high recurrence rate. Our case showed no recurrence after six months of follow-up at the surgical site.


Unilateral nasal obstruction is a common complaint with a broad differential diagnosis that includes anatomic asymmetry, unilateral infective or inflammatory conditions, and benign and malignant sinonasal masses. A rhinolith is an uncommon foreign body in the nose, which serves as a nidus for calcium salt deposition. The foreign body can be endogenous or exogenous in origin and may remain asymptomatic for many years before incidental discovery. When left untreated, stones may cause unilateral nasal obstruction, rhinorrhea, nasal discharge, epistaxis or, in rare cases, progressive destruction leading to septal/palatal perforation or oro-antral fistula. Surgical removal is an effective intervention with limited complications reported.This article describes a 34-year-old male who presented to the emergency department (ED) with unilateral obstructing nasal mass and epistaxis, which was found to be an iatrogenic rhinolith. Successful surgical removal was performed.Epistaxis and nasal obstruction are common presentations to the ED. Rhinolith is an uncommon clinical etiology that if left undiagnosed may lead to progressive destructive disease; it should be included in the differential for any unilateral nasal symptoms of unclear origin. Appropriate work-up for any suspected rhinolith includes computed tomography, as biopsy is risky given the broad differential of unilateral nasal mass. When identified, surgical removal has a high success rate with limited complications reported. Nasal obstruction and epistaxis are common complaints in acute care medicine. Obstruction may be acute or chronic, bilateral or unilateral, and may be anatomical or inflammatory in origin.4A rhinolith is a rare, often overlooked etiology for unilateral nasal obstruction, epistaxis, and nasal drainage that presents similarly to neoplastic nasal masses on physical examination.6We present a case of iatrogenic rhinolith in a neurocognitively intact adult male likely arising from retained packing after an intranasal operation 2.5 years prior in Afghanistan, and we discuss clinical, pathological, and radiological features.A 34-year-old male who had recently immigrated from Afghanistan presented to the emergency department (ED) with several years of right-sided nasal obstruction and several days of intermittent self-limiting right-sided epistaxis. Past medical and surgical history was significant for an intranasal procedure 2.5 years prior while still in Afghanistan. Initial evaluation by the emergency physician revealed a whitish-gray, rock-hard mass in the floor of the right nasal passage, which was biopsied for culture. A CT was also obtained. Biopsy revealed aggregates of filamentous, Gram-positive rods with short, modified acid-fast microorganisms. Computed tomography revealed a 3.5-centimeter calcified mass inferior to the right turbinate .Outpatient referral and subsequent surgical removal was recommended by otolaryngology. Due to physical exam and imaging characteristics, antibiotics were not recommended. The patient ultimately underwent uncomplicated surgical removal. Intraoperatively a large, calcified nasal mass was encountered between the septum and inferior turbinate . This waHe recovered without complication.CPC-EM CapsuleWhat do we already know about this clinical entity?Rhinoliths are benign intranasal calcified foreign bodies that often present with nonspecific symptoms such as epistaxis or nasal obstruction with an indolent course.What makes this presentation of disease reportable?A neurocognitively intact adult presented with epistaxis and intranasal mass. Removal suggested retained surgical material from history of nasal surgeryWhat is the major learning point?There is a broad differential for intranasal masses including benign and malignant etiologies. Imaging and subspecialty consultation are often indicated.How might this improve emergency medicine practice?Rhinoliths are uncommon, difficult to visualize and are nonspecific in presentation. Definitive management is removal. They should remain on the differential for nasal complaints.A rhinolith is a calcification in the nasal passage that originates when a foreign body serves as a nidus for inorganic salt deposition.12Rhinoliths may be found incidentally on anterior rhinoscopy, nasal endoscopy, or imaging studies when asymptomatic.14A common presenting history of exogenous stone formation is of a foreign body placed in childhood that was never removed. Less common is iatrogenic rhinolithiasis from retained nasal packing, sutures, or gauze that facilitate stone formation. Rare reports include concreted topical ointments, debris from nasopharyngeal refluxed emesis, and even stagnant inhaled particulates in the setting of impaired nasociliary clearance.12The vast majority of rhinoliths are observed along the floor of the nasal passage abutting the inferior turbinate; however, they have been described throughout the sinonasal passage.9In this case, an adult male presented to the ED with unilateral nasal obstruction and epistaxis. Computed tomography was appropriately ordered to further classify the mass, and subspecialty referral to otolaryngology was made. Treatment involved surgical removal with no need for antibiotics. Notably, the core of the calcified mass was seen to have a firm, rubbery texture with illegible orange foreign text. Given his history of endoscopic sinus surgery while living in Afghanistan, it is likely this substance represented a retained Penrose drain, which is occasionally used in sinus operations. Rhinolith is an uncommon clinical etiology; however, nasal obstruction and epistaxis are commonly encountered in acute care clinics. Given the broad differential for unilateral obstructing nasal masses, biopsy should be approached with caution.Rhinolithiasis is an uncommon etiology of unilateral nasal symptoms such as obstruction, pain, epistaxis, or purulent drainage. This is a slowly developing condition that is easily overlooked due to similarities in presentation with nasal neoplasms. If left unresolved, there is risk of local destructive change. Work-up of unilateral nasal mass, epistaxis, or purulent drainage without clear etiology should include CT, as biopsy is risky given the broad differential of unilateral mass. Surgical removal has a high success rate with low risk of complication.