Spinal epidural capillary hemangioma is a very rare variety of tumors, usually with a predilection for the thoracic spine.A 16-year-old female complained of hyperacute neurologic deficit progressed within hour, which presented by acute paraplegia, and loss of all sensations from her lower limbs up to her breasts. Neurologic exam revealed paralysis of lower limbs (0/5 on both legs) with a flaccid tone, absence of reflexes, weakness of the trunk with sensory level T4, bilateral flexion of plantar reflexes, and loss of sphincters’ controls. Emergent magnetic resonance imaging showed a dumbbell-shaped epidural mass in the posterior aspect of the spinal canal at the T1–T2 level, measuring approximately 1.1 × 4.5 × 1.5 cm in size. The lesion was isointense on T1-weighted, hyperintense on T2-weighted, and a little enhancement after gadolinium administration. The surgery was obtained nearly 16 hours after paralysis, which eradicated the lesion with good hemostasis. Histological examination showed a well-organized vascular tissue that haphazardly arranged and confirmed the diagnosis of capillary hemangioma. Neurological improvement was quickly observed within days after surgery and further complete recovery was achieved 2 months after discharge.We report an extremely rare case of spinal epidural capillary hemangioma, where acute spontaneous hemorrhage in the lesion resulted in the hyperacute neurologic deficit within an hour. Since these are benign lesions, the immediate surgical intervention results in a very favorable prognosis and is considered the treatment of choice. Also, this case highlighted and rose the question of a better neurologic improvement in younger age patients with spinal cord injury. Spinal vascular tumors are classified as capillary telangiectasias, cavernous hemangiomas, capillary hemangiomas, arteriovenous malformations, or venous malformations.,2 Paravertebral extension of the lesion through the intervertebral neural foramina may frequently occur. Most patients present in their fourth to fifth decades of life, with a median age of 49.2 years. There is a slight predilection for females, with a male-to-female ratio of 9:13.Hemangiomas of the spine are usually lesions of the vertebral bodies, usually located in the posterior part of the epidural space with a predilection for the thoracic spine. Through the literature reports, spinal epidural hemangioma is a very rare variety of tumor, in addition, the capillary type is far rarer than the cavernous type. Approximately 100 cases of cavernous epidural hemangioma compared to only 22 cases of spinal epidural capillary hemangioma (SECH) that were reported previously in the literature.,5Hemangiomas constitute approximately 4% of all epidural tumors and 12% of all intraspinal hemangiomas.,6 Well-circumscribed margins (dumbbell shape) of the lesion and typically locates in the posterior portion of the spinal canal.,6,7 Furthermore, surgical resection is the mainstay treatment for SECH, with a good prognosis, in most cases without recurrence.,8According to the literature, magnetic resonance imaging (MRI) with contrast is the diagnostic modality of choice for all spinal cord lesions, including SECH.Here we report the 23rd case of capillary hemangioma in a 16-years-old female. The great distinction of this case is that symptoms were hyperacute and developed within an hour. Despite the decompression surgery being obtained nearly 16 hours after paralysis, the neurological improvement was quickly observed within days after surgery and further complete recovery was achieved 2 months after discharge.am followed by diffused numbness throughout her lower limbs. Approximately 1 hour thereafter, she could not move her lower limbs, and her back, and she lost all sensations from her lower limbs up to her breasts. She arrived at the hospital after nearly 10 hours after paralysis. There was no traumatic accident and she had no significant past medical history.A 16-year-old female was admitted to our neurology department with acute lower limbs paralysis. She had back pain at 2:00 On examination, blood pressure was 120/80 mm Hg, heart rate was 85/minutes, respiratory rate was 18/minutes, oxygen saturation was 98% in the air room, and the temperature was 37 °C. Neurological examination revealed paralysis of lower limbs (0/5 on both legs) with a flaccid tone, absence of reflexes, weakness of the trunk with sensory level T4, and bilateral flexion of plantar reflexes. Also, there was a loss of sphincters’ controls where an atonic bladder evacuated 1500 mL after catheter placement.Emergent MRI was conducted: axial and sagittal T1- and T2-weighted, and post-gadolinium sequences , showed a hemorrhagic, well-circumscribed, and compressive epidural mass, which was eradicated completely with good hemostasis, and the sample was sent to pathology.We continued the conservative management after the surgery with airbed, continuous mobilization, physiotherapy, infection control, and methylprednisolone (MP) 1 g for 5 days followed by tapering doses. subsequent MRI revealed no mass effect with a drain 24 hours after surgery  for several weeks or months due to the slow-growing nature of SECH.Our patient’s status progressed more severely than in previous reports where she was paralyzed within an hour. Also, there was no previous history of trauma suggesting an acute spontaneous hemorrhage in the lesion that resulted in the patient’s symptoms.,8,12The surgical resection with or without embolization has been advocated, even in the absence of neurological symptoms, to prevent deterioration and permanent neurological deficits if acute spontaneous or post-traumatic hemorrhage into the lesion occurred.,6,7 The lesion typically presents as an iso- or hypointense lesion on T1-weighted images, hyperintense on T2-weighted images, and homogenous enhancement following gadolinium administration.,6 However, these imaging features are not specific to SECH, hence it can be challenging to distinguish from other epidural tumors.,6 Moreover, SECH is often misdiagnosed radiologically as being meningiomas and schwannomas because of post-contrast enhancement demonstration of all these types. Many differential diagnoses might be considered such as nonneoplastic lesions; including disc herniation, epidural hematoma, lipomatosis, and abscess, and neoplastic lesions; including meningioma, lymphoma, hemangioblastoma, sarcoma, and even metastases.,15MRI typically demonstrates epidural well-circumscribed (dumbbell shape) mass that locates in the posterior portion of the spinal canal.,12 The latter showed a large number of sinusoidal channels spread in collagenous tissue. By lateral, capillary hemangiomas are composed of thin and irregular capillary vessels en veloped by fibrous stroma or capsule.,12,17Hence histologic confirmation is necessary in most cases, pathological diagnosis is also necessary to differentiate between hemangiomas and cavernous malformations.Corresponding with previous reports, the SECH was observed in this patient as a dumbbell-shaped epidural mass in the posterior aspect of the spinal canal at the T1–T2 level that compressed the spinal cord. The lesion on MRI was isointense on T1-weighted and hyperintense on T2-weighted with a little enhancement after gadolinium administration. Histological examination aligned with immunostains confirmed haphazardly arranged vascular tissue surrounded by thin capsule.In the surgical field of spinal cord injury (SCI), there is a question about the ideal time after the trauma to perform the surgical procedure. The studies divide the time until surgery after the injury into 2 categories: “early” and “late.” Another systematic review and meta-analysis of 16 studies involving 3977 patients with SCI observed significant improvements in motor scores, light touch scores, and sensitivity in the early group (<24 hours) compared with patients in the late group (>24 hours).A large prospective cohort of the STASCIS  study, found an improvement in the American Spinal Injury scale  in patients undergoing early surgery (<24 hours post-injury) compared to the late surgery (≥24 hours post-injury).Recently, a literature review with a meta-analysis of 9 articles by Iunes et al showed a potential neurological improvement with early or even ultra-early surgical decompression (up to 12 hours) in patients with traumatic cervical SCI. On the other hand, reports about the advantage of early decompression when there is a thoracic injury are scarce.The gap in robust data on the beneficial effect of the intervention within 24 hours can be explained as many studies do not specify the area of SCI. Aarabi et al found no neurological benefit in patients with traumatic cervical SCI undergoing decompression within 12 hours of injury, compared with 12 to 24 hours or >24 hours. Meanwhile, the extent of the intramedullary injury was the only significant predicting factor of the improvement in AIS scale.Another systematic review including 14 studies and 1075 patients by Ter Wengel et al investigated the neurological outcome for injuries of the thoracic and thoracolumbar region (T11–L2) between early and late surgery groups. However, the authors did not observe a significant beneficial effect of 24-hour surgical decompression, the rate of AIS scale improvement in the early surgery group was better than in the late surgery group.,23 Also, Sewell et al reviewed that the early decompression group was associated with shorter ICU stay and a lower complication rate but no significant difference in neurological improvement was observed between the 2 groups. However, the author mentioned that neurological recovery was more noticeable in younger patients.An important limitation of the studies presented is the lack of homogeneity among patients. Fehlings et al and Cengiz et al observed the average age of patients in the early group was lower than that of those in the late group. This was probably because most surgeons opted for earlier surgical operations in younger patients.Our patient had paraplegia for approximately 16 hours so the surgeon was not excited to do the operation and suspected a bad prognosis. However, the young age of the patient and upon the family’s acceptance, the decision was made to obtain the surgery trying to give a chance for at least a few neurological improvements. Fortunately, the complete recovery was quickly observed between 2 to 3 months after surgery. This corresponds with the previous studies that mentioned the benefit of early decompression (within 24 hours) for SCI. Also, this might suggest a further neurologic recovery in younger age patients and rose the question of a better neurologic improvement in younger age patients with SCI mentioned previously by Sewell et al.,26However, the administration of MP was upon the surgeon’s recommendation in our case, there was no rationale for this practice in SCI. Hence, scattered reports of Class III evidence have been published supporting the neuroprotective effect of MP in SCI but these results are likely related to random chance or selection bias.,28 Clinicians considering MP therapy should bear in mind that the drug is not Food and Drug Administration approved for this indication. Also, the pooled evidence existed that high-dose steroids were associated with harmful effects including wound infection, hyperglycemia, and gastrointestinal hemorrhage.Based on the current evidence, high-dose MP treatment does not contribute to better neurologic recoveries and does not demonstrate a significant long-term benefit.Moreover, we referred to the valuable role of supportive care in the treatment, including physiotherapy and continuous mobilization, to help the patient’s recovery which might be more quickly in younger patients as observed in our patient.We report an extremely rare case of SECH that manifested with a hyperacute presentation where acute spontaneous hemorrhage in the lesion resulted in the patient’s symptoms within an hour. Early and prompt diagnosis by MRI is vital and hemangiomas should be considered in the differential diagnosis of epidural lesions with dumbbell-shaped appearance, especially at the thoracic level. Since these are benign lesions, the immediate surgical intervention results in a very favorable prognosis and is considered the treatment of choice. Also, this case highlighted and rose the question of a better neurologic improvement in younger age patients with SCI.Investigation: Marwa Kliea, Mohammad Alsultan, Eyad Chatty, Safaa Qatleesh.Methodology: Marwa Kliea, Mohammad Alsultan.Supervision: Eyad Chatty, Ghassan Hamzeh.Validation: Marwa Kliea, Mohammad Alsultan.Visualization: Marwa Kliea, Mohammad Alsultan.Writing – original draft: Marwa Kliea, Mohammad Alsultan.Writing – review & editing: Marwa Kliea, Mohammad Alsultan, Ghassan Hamzeh.


Gliosarcoma (GS) is a primary central nervous system tumor. It is an unusual type of glioblastoma multiforme (GBM) and rarely invades the skull base. It has a biomorphic tissue pattern with rapid alternation zones of glial and mesenchymal differentiation. We report the case of a 62-year-old male who presented with a one-month history of unsteady gait associated with dizziness. Brain MRI showed a right temporal mass that invaded the skull base with perilesional edema and a significant mass effect on the right lateral ventricle. The patient underwent a right-sided frontotemporal craniotomy with gross total resection. The pathology confirmed the diagnosis of GS. Postoperatively, the patient had an uneventful recovery with no complications and was discharged two days post-surgery. Gliosarcoma (GS) is a rare malignant type of glioblastoma multiforme (GBM) and accounts for 2% to 3% of all cases of GB . The chaHistoryA 62-year-old male patient was referred to the neurosurgery department because of a right temporal lobe ring-enhancing lesion on brain MRI. The patient was complaining of an unsteady gait that was associated with dizziness, increased by leaning forward, for one month. He had no headache, loss of consciousness, seizures, blurred vision, nausea, vomiting, or hearing or speech abnormalities. The patient has no history of malignancy or similar conditions in the family.Physical examinationThe patient was vitally stable with a 15/15 Glasgow coma scale (GCS). Pupils were 2 mm in diameter in bright light with normal eye movements and with no visual field defects. No facial muscle weakness, abnormal sensation, or tongue deviation were found. Also, the patient had normal muscle tone and power. He had normal sensations and reflexes in the upper and lower limbs.ImagingA gadolinium contrast brain MRI showed a 48 x 55 x 57 mm right temporal lobe mass with surrounding edema and a significant mass effect on the right lateral ventricle, midline shift, and right uncal herniation Figure . MoreoveOperationUnder general anesthesia in a supine position, the head was turned to the left, and the operative field was draped in the usual sterile fashion. A question mark incision was created, reflecting the scalp and the temporalis muscle. A fungating mass was found penetrating and eroding the temporal bone and attached to the temporalis muscle at the skull base. The tumor was mildly vascular with a firm consistency. Frontotemporal craniotomy was done dissecting around the fungating mass. The dura was invaded by the lesion, so the dura was opened in a curved linear fashion. A reasonable plane was created between the brain and the mass. A gross total resection of the temporal mass was made including the extracranial components, which was invading through the dura and the bone and its attachment to the temporalis muscle. The intraoperative frozen section confirmed the diagnosis of GS. Hemostasis was obtained, DuraSeal® was used to reconstruct the dura, the bone flap was secured with plates and screws, and the scalp flap was closed in layers. HistopathologyThe specimen illustrated a neoplasm invading dura. The tumor showed a biphasic mixture of gliomatous and sarcomatous tissue. The glial portion is astrocytic in nature with atypical nuclei and eosinophilic cytoplasmic processes. The sarcomatous portion was composed of densely packed long bundles of spindle cells. Mitotic figures, microvascular proliferation, and areas of necrosis were seen Figure .Postoperative follow-upThe procedure was well tolerated. There was no complication, and the patient was sent to the intensive care unit (ICU) for observation and spent one day in ICU in stable condition. Postoperatively, the patient was conscious and oriented and had a GCS score of 15/15. Cranial nerves were intact without any neurological deficit. Postoperative MRI showed subtotal resection of the temporal mass from the right side and the extracranial components Figures -4. On poGliosarcoma is a rare malignancy of the brain that has a biomorphic tissue pattern including glial and mesenchymal differentiation. The exact prevalence of GS is not known. However, it has been reported to account for 2% of all GBM in adults, with a higher incidence in males . Also, iRegarding clinical presentations, headache is considered to be the most reported symptom, followed by unsteady gait -15,17. OThe most common location to metastasize is the temporal lobe ,13. OtheThe management of GS includes surgery in addition to radiotherapy, chemotherapy, or chemoradiotherapy. However, a retrospective study of 75 patients concluded that radiotherapy is superior to temozolomide-based chemoradiation or adjuvant chemotherapy for improving the survival rate .Several factors play a role in overall survival, including age at diagnosis, the extent of surgery, and adjuvant radiotherapy. Fifteen months is the average survival period if the patient is younger than 50 years old, while it would be seven months if the patient is older than 50 years old. Radiotherapy increases survival to 10 months . Gliosarcoma is a rare type of glioblastoma. We highlighted a patient with GS invading the temporal area with the skull base. Skull base involvement and how it affects the prognosis or survival of the patient are not yet known. However, we encourage an aggressive attempt at resection with proper skull base surgical technique to ensure optimal results. Therefore, conducting further research is important to have a better understanding of this condition.


Synovial sarcoma is a subtype of soft tissue sarcoma. Synovial sarcoma in the head and neck region is relatively unusual. Primary synovial sarcoma of the thyroid gland (PSST) is first reported in 2003 by Inako Kikuchi. PSST is extremely rare with only 15 cases documented globally. PSST shows rapid disease progression and a relatively poor prognosis. However, diagnosis and therapy are challenging for clinical surgeons. In this article, we reported the 16th PSST case and reviewed the PSST cases globally for further clinical application.The patient was referred to us because of gradually worsened dyspnea and dysphagia for 20 days. Physical examination showed a 5 × 4 cm mass with a clear boundary and good mobility. Contrast-enhanced ultrasonography (CEUS) and computed tomography (CT) showed a mass in the isthmus of the thyroid gland. The imageology diagnosis tends to be a benign thyroid nodule.in situ hybridization indicated the mass to be primary synovial sarcoma of the thyroid gland with no local and distant metastasis.After surgery, histopathology, immunohistochemistry, and fluorescence, The patient underwent total thyroidectomy and dissected the lymph nodes in the central compartment. This patient received postoperative chemotherapy (a combination of ifosfamide and epirubicin for five cycles). Patients tolerated chemotherapy well. No recurrence was found during the 9-month follow-up.Although PSST is an extremely rare disease, we should raise our awareness when we encounter a rapidly growing, cystic-solid mixed thyroid mass with neck compression symptoms to avoid misdiagnosis. Intraoperatively, surgeons should refine surgical procedures to avoid capsular rupture and tumor local implantation metastasis. Intraoperative frozen section pathology is necessary sometimes, especially when the diagnosis could not be established before surgery. Synovial sarcoma is a kind of histological subtype of soft tissue sarcoma, making up 8–10% of all soft tissue sarcomas. It is a high-grade malignant neoplasm of mesenchymal pluripotent cells, characterized by a specific t p11.2; q11.2) translocation leading to the generation of SYT-SSX fusion gene transcripts ; q11.2 t–13. Due A 59-year-old female patient was admitted to our hospital in June 2022. Her chief complaints were dyspnea and dysphagia for 20 days. She felt dyspnea 20 days before, and the discomfort gradually worsened accompanied by cough and dysphagia. The physical examination showed a palpable hard mass in the center of the neck. The size of the mass was 5 × 4 cm . The bouA Blood test showed no obvious abnormalities. The level of thyroid hormone and calcitonin is in the normal range. No abnormalities were found in the laryngoscopy, chest X-ray, and chest CT. Contrast-enhanced ultrasonography showed a heterogeneous echoic, cystic-solid mixed mass ~48.8 × 37.2 mm in the isthmus of the thyroid gland. The mass is with clear boundary, regular shape. In total, 0.6 ml perflubutane microspheres were injected through the elbow intravenous for CEUS. The isthmus nodule was mostly equally enhanced and partially no enhancement. The peripheral nodule showed annular enhancement. No obvious enlarged lymph nodes were observed in the cervical region. The mass was classified as TI-RADS 3 . CT scanThe patient was intended for partial thyroidectomy. We made a transverse arc-shaped incision in the suprasternal notch, dissociated the subcutaneous tissue, and opened the interval between the bilateral strap muscles in the neck to expose the thyroid mass. The mass was located in the isthmus of the thyroid gland and extended to the back of the sternum. The mass was cystic-solid mixed. The cyst wall was closely adhered to the trachea. Intraoperatively, the mass was difficult to be mobilized and led to a capsular rupture and an effusion of necrotic, hemorrhagic, and gelatinous liquid. When the mass was cut open, chocolate-like contents could be seen in the capsule. The solid section was fish-like, gray-red, and gray-yellow, and hard calcification was visible in some areas. Frozen section pathology showed soft tissue sarcoma, after which we choose to carry out total thyroidectomy and central cervical lymph nodes dissection.Histopathology showed that the thyroid isthmus mass was a mesenchymal tissue-derived spindle cell malignant tumor, with epithelioid differentiation in the focal area. The nucleus was atypical. Lots of mitotic figures could be seen. Invasion of surrounding soft tissues was observed. There are no obvious masses in the left and right lobes of the thyroid gland. There was no metastasis in the central cervical lymph nodes (0/6) .CK(-), EMA(-), CK7(-), CK19(-), CD99(-), Bcl-2(+), WT-1(-), TLE1(+), Ki-67(50%), P53(30%+), Vim(+), Syn, S100(-), SMA(-), MyoD1(-), STAT6(-), and SOX-10(-) .in situ hybridization (FISH), showed an SS18 (SYT) gene rearrangement, highly consistent with the diagnosis of a PSST . The patient tolerated chemotherapy well. The patient received five-cycle chemotherapy. Postoperative adjuvant radiation therapy was recommended by the oncologist, but the patient refused for personal reasons. After 9 months of the surgery, no remarkable recurrence and metastases were detected.In this article, we reported the 16th PSST case globally. Although the mass of this patient was mostly composed of spindle cells, a lack of normal thyroid follicular epithelial cells, the boundary between the mass and the surrounding thyroid tissue was unclear, and a part of the normal thyroid tissue was replaced by the synovial sarcoma. In synovial sarcoma of the head and neck regions originating from other structures, the main tumor foci are usually situated around their local regions instead of the thyroid gland. Therefore, according to a definition in a previous study, this mass was identified to be primary thyroid synovial sarcoma . When suFor further investigation, we searched the PSST case in PubMed and Web of Science. A total of 15 cases were reported previously. We summarized the clinical characteristics of the 15 cases in PSST was usually misdiagnosed with MTC, undifferentiated/anaplastic thyroid carcinoma, or SETTLE. In this part, we would discuss the differential diagnosis of PSST. For PSST, the main differential diagnosis is MTC. An elevated calcitonin level is typical in MTC patients. In a microscopic vision, the MTC tissue usually consists of lympho/plasmacytoid cells, atypical cells, or spindle cells. However, a pure spindle cell pattern MTC is uncommon. Giant cells or multinucleated cells could be seen. The MTC tumor cells are generally positive for calcitonin, thyroglobulin, and neuroendocrine markers . SETTLE Until now, there is no standard and ideal therapeutic regimen for PSST. Surgical resection remains the mainstay of management for localized synovial sarcoma with or without radiation . When thAlthough we tried our best to document this clinical case report in detail, there are still some limitations. First, because the tumor, in this case, was suspected to be benign in the preoperative examination, we did not perform screening for systemic metastases before surgery, but relevant examinations including chest CT, abdominal CT, and MRI of the head and whole-body bone scan were performed before and during chemotherapy. Second, due to capsular rupture and necrotic effusion during the operation, we are pitiful for failing to take photos of the tumor. The necrotic effusion might lead to tumor local implantation metastasis, making postoperative adjuvant therapy necessary and important. Last but not the least, despite the follow-up of the case being less than 1 year, complete treatment for PSST in this patient has been completed, and no recurrence and metastasis have been detected in 9 months postoperatively. We will continue to focus on the case. We will update this case report and literature review if recurrence or death occurs in the current case.1. Synovial sarcoma is a subtype of soft tissue sarcoma, which is extremely rare in the thyroid gland. PSST showed rapid disease progression and a relatively poor prognosis. However, diagnosis and therapy are challenging.2. Complete surgical excision and postoperative adjuvant chemotherapy are usually the typical therapeutic regimen for PSST.1. PSST is difficult to be diagnosed by preoperative evaluation, even FNA. PSST was occasionally misdiagnosed as a benign thyroid tumor. Surgeons should raise their awareness when encountering rapidly growing, cystic-solid mixed thyroid mass with neck compression symptoms.2. The initial surgery of PSST is of vital importance. Surgeons should avoid capsular rupture and effusion of necrotic during surgery. Intraoperative frozen section pathology should be performed when necessary. Postoperative adjuvant chemotherapy should be scheduled for reduced recurrence and prolonged survival time.The original contributions presented in the study are included in the article/supplementary material, further inquiries can be directed to the corresponding authors.Ethical review and approval was not required for the study on human participants in accordance with the local legislation and institutional requirements. The patients/participants provided their written informed consent to participate in this study. Written informed consent was obtained from patient for the publication.JH, YG, FY, and CR performed the surgery. CR, ZC, QJ, and YL collected the data from the patient. CR, XL, and SL drafted the first manuscript. YG and FY revised the manuscript. All authors read and approved the final manuscript.


Tenia solium. Cysticercosis in humans is common in the cerebral tissue but rare in the tongue.Cysticercosis is a condition in which humans are infected by the larval form of the pork tapeworm Here, the authors report a rare case of a 38-year-old male with neurocysticercosis and cysticercosis of the tongue. The patient presented with a complaint of loss of consciousness for 4–5 min. Local examination of his oral cavity revealed a swelling of ~2×2 cm on the tongue. An MRI of the brain showed various stages of neurocysticercosis involving the neuroparenchyma and tongue. For this, he was started on low-dose prednisolone of 50 mg tapered over 6 weeks and levetiracetam of 500 mg BD continued for his seizure episodes. He is responding well with the medications and is planned to start antiparasitic agent only after the perilesional edema decreases.Cysticercosis may involve the central nervous system, muscle, heart, lungs, peritoneum, eye, and subcutaneous tissue. Oral cavity and perioral involvement by cysticercous larva is rare in humans. Radiologic imaging, serology, and tissue biopsy can be used to confirm a diagnosis of cysticercosis. The most common locations for oral cysticercosis are the tongue, buccal mucosa, lower lip, and upper lip.Only 102 cases of oral cysticercosis have been reported based on a PubMed English-language literature search.Oral cysticercosis is a rare event, and it represents a difficulty in clinical diagnosis. But a patient with a mass in the tongue should be considered as a possible case of cysticercosis especially in endemic regions like Nepal. HighlightsCysticercosis in humans is common in the cerebral tissue but rare in the tongue.The most common locations for oral cysticercosis are the tongue, buccal mucosa, lower lip, and upper lip.A Patient with a mass in the tongue should be considered as a possible case of cysticercosis.Tenia solium and results from the ingestion of tapeworm eggs through contaminated food and water or dirty hands, humans acting as the intermediate host. The adult T. solium, or pork tapeworm, lives in the small intestine of man, its definitive host. Humans act as both definitive and intermediate hosts of T. solium1.Cysticercosis is a condition in which humans are infected by the larval form of the pork tapeworm 2. The oral cavity is a rare site of involvement by cysticercosis, even in an endemic area3. In addition, cysticercosis presenting as a nodule or mass in the tongue is even more rare4. Only 102 cases of oral cysticercosis have been reported in the worldwide based on a PubMed English-language literature search. We present a rare case of neurocysticercosis along with cysticercosis of the tongue. This case has been reported following the Surgical CAse REport (SCARE) criteria5.Cysticercosis in humans is common in the cerebral tissue, subcutaneous tissue, muscle, and the eyeA 38-year-old male initially presented to the Medicine Outpatient Department in a tertiary care center of Nepal with a complaint of loss of consciousness (LOC) for 4–5 min. Loss of consciousness was associated with clenching of teeth, up rolling of eyes, vacant stare, and frothing from the mouth, which was suggestive of a seizure. He had experienced similar episodes of symptoms a year back. In addition, he also had painless swelling over the ventral aspect of the anterior 1/3rd of the tongue as shown in Figure There was no history of hoarseness of voice, dysphagia/odynophagia, nonhealing oral ulcers, or weight loss. There was no history of headaches, localized sensory/motor deficits, parasthesias, and personality changes.Local examination of his oral cavity revealed swelling of ~2×2 cm which was firm, mobile, nontender with a diffuse margin, and normal mucosa with unrestricted tongue movement. On chest examination, multiple nontender erythematous lesions were present as shown in Figure His blood reports were within the normal range. A chest radiograph showed multiple grain like lesion present all over the chest muscles as shown in Figure T. solium, a hermaphroditic parasite6. Humans act as both intermediate and definitive hosts, while pigs are intermediate hosts. Eggs ingested by pigs from human feces-contaminated soil then release embryos that penetrate the intestinal wall and develop into larval form (cysticercosis). Humans acquire these larvae by consuming uncooked pork, completing T. solium’s life cycle in the small intestine7. These infected humans can have taeniasis for long periods and contaminate the environment continuously8.Cysticercosis is caused by the larval form of 9. The eggs develop into oncospheres that penetrate the intestine wall and – via lymphatic or vascular circulation – reach a destination, where larvae develop and become the cysticerci or ʻa fluid-filled cystʼ. Once a person becomes the host of cysticercus cellulosae, cysticercosis can develop in various organs and tissues2.Cysticercosis develops when humans ingest tapeworm eggs instead of pigs; humans acting as an intermediate host. This usually happens by consumption of faecally contaminated food and water or by faeco oral contamination as well as by reflux of eggs into the stomach8. Neural cysticercosis has nonspecific signs and symptoms; however, acute symptomatic seizures is the most common presentation. Our case too had presentation of seizure for which levetiracetam of 500 mg BD was prescribed.The most serious involvement is that of the central nervous system called as neurocysticercosis, followed by ocular involvement, usually the only ones which are symptomatic10. Oral cavity and perioral involvement by cysticercous larva is uncommon in humans11. According to a series published by Singh et al.6 in 2018 a total of 89 cases of oral cysticercosis (excluding masseter and parotid) had been reported in PubMed English literature, out of which 50.6% were in the tongue. Since then, 13 more case has been reported, excluding the present case making it total of 102 cases reported till date. The most common locations for oral cysticercosis are the tongue, buccal mucosa, lower lip, and upper lip12.Extraneural cysticercosis, which is also very common, infects muscles, the heart, lung, peritoneum, eye, and subcutaneous tissue13. Lingual cysticercosis can be mistaken for other lesions like fibromas, lipomas, or salivary gland tumors14.While tongue muscle involvement in cysticercosis is common in swine, it is rare in humans. The reason for this is unclear, but some suggest that the high muscular activity and metabolic rate in humans may hinder the cysticercus’ lodging and development in this location7. In most cases diagnostic certainty is not possible; instead a clinical diagnosis is made on the basis of a combination of clinical presentation, radiographic studies, serologic tests, and exposure history15 In contrast to our case, serology and immunology tests for cysticercosis are not available in the center, and when done in other private labs are expensive for the average Nepali (for which the patient has to pay out of pocket). Given the good utility provided by the MRI; the treating team decided further testing would add financial burden to the patient without necessarily providing additional information or influencing the treatment. So the tests were not done.Radiologic imaging, serology, and tissue biopsy can be used to confirm a diagnosis of cysticercosis. Imaging techniques, in particular computed tomography and MRI, are of great value to diagnose cerebral cysticercosis. Histopathological examination makes up a diagnosis of cysticercosis by the detection of a cystic space containing the cysticercus cellulosae16. However, cysticidal drugs may be associated with severe reactions such as local tissue swelling and generalized anaphylactic reaction due to massive antigen release. The use of corticosteroids with or before their administration can help in decreasing the incidence of these complications17 In contrast to this, in our case, the patient was started on low-dose prednisolone of 50 mg tapered over 6 weeks and levetiracetam of 500 mg BD continued for his seizure episodes. He is responding well with the medications and is planned to start antiparasitic agent only after the perilesional edema decreases.The treatment of oral cysticercosis is surgical excision. Praziquantel and albendazole are used to treat cysticercosis, especially in patient with disseminated cysticercosis or where surgical excision is risky or not possible, such as in neurocysticercosisT. solium cysticercosis is linked to the consumption of contaminated food and the practice of coprophagy. Therefore, it is important to inspect pork and ensure proper hygiene and sanitation measures are in place, including the appropriate disposal of human feces18. Health education plays a crucial role in raising awareness about preventing tapeworm infection through adequate pork cooking. Additionally, promoting good personal hygiene and proper fecal disposal can significantly reduce the occurrence of cysticercosis19.The risk of T. solium infection and subsequent cysticercosis.Oral cysticercosis is a rare event, and it represents a difficulty in clinical diagnosis. But a patient with a mass in the tongue should be considered as a possible case of cysticercosis especially in endemic regions like Nepal. This case is about a mixed case of neurocysicercosis and cysticercosis of a tongue managed conservatively. We expect this case could raise the attentions to the control of None. Since this report involves no experiments, the ethical approval is waived.Written informed consent was obtained from the patient for publication of case report. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.None.All authors have contributed to the writing, editing, and preparation of the manuscript and have reviewed it before submission.The authors declare that they have no financial conflicts of interest with regard to the content of this report.1. Name of the registry: not applicable.2. Unique identifying number or registration ID: not applicable.3.Hyperlink to your specific registration (must be publicly accessible and will be checked): not applicable.Bipin Poudel.None.Not commissioned, externally peer-reviewed.


Hutchinson-Gilford progeria syndrome frequently exhibits stunted growth and premature ageing. Notable ocular characteristics can encompass a large number of ocular abnormalities. Here, we report a case of a 17-year-old female who presented with complaints of a red bump not resolving with medications in her right eye for 3 months. On excisional biopsy, the mass was found to be a pyogenic granuloma. The occurrence of pyogenic granuloma in a patient with progeria is extremely rare. This highlights the importance of considering atypical presentations and potential complications in patients with Hutchinson-Gilford progeria syndrome. This information can assist healthcare professionals in the early recognition and appropriate management of ocular conditions in individuals with Hutchinson-Gilford progeria syndrome. The combination of these two conditions is extremely rare and highlights the importance of understanding the specific challenges and considerations in diagnosing and managing HGPS.Hutchinson-Gilford progeria syndrome (HGPS) is a rare hereditary disorder with a prevalence of one in 4-8 million new births.A 17-year-old female from Panauti, presented with complaints of a red bump not resolving with medications in her right eye for 3 months. On examination, her best-corrected visual acuity (BCVA) in both eyes was 6/6 with Snellen's visual acuity chart. External ocular examination showed excessive forehead wrinkling for age, thickened eyelids, pseudo-proptosis, superior sulcus deformity, lagophthalmos, lid lag in downgaze, and loss of eyelashes and eyebrows in both eyes.Systemic examination revealed a typical senile look with craniofacial disproportion, frontal and parietal bossing called pseudo hydrocephaly, beaked nose, mandibular and maxillary hypoplasia, prominent scalp veins, protruding ears, thin lips, thin limbs, prominent and stiff joints, thin and high-pitch voice, generalized alopecia, absence of subcutaneous fat, thin and wrinkled 'sclerodermatous' skin, flecks like hyperpigmentation in sun-exposed areas, prominent superficial veins, and nail dystrophy .Slit-lamp examination revealed a dry and keratinized ocular surface and blocked meibomian gland orifices in both eyes. Cornea was clear and lustrous. There was a small congested mass (3x4 mm in size), not moving with the conjunctiva in the bulbar conjunctiva of the nasal aspect of the right eye .There was grade 1 nuclear sclerosis in both eyes. Intraocular pressure was 15 mmHg and 13 mmHg in the right eye and left eye respectively, measured by air-puff tonometry. Fundus examination under mydriasis was normal in both eyes. Antenatal history was obtained which was uneventful, and delivery was normal at 40 weeks of gestation. Her parents gave no history of consanguinity of marriage. A general physical examination of the patient showed a height of 137 cm and a weight of 34 kg. Her pulse was 72/min, and her blood pressure was 110/70 mmHg. Because of the characteristic clinical presentation, the child was diagnosed with HGPS. An excisional biopsy of the mass was done, the histopathological report showed it to be a pyogenic granuloma .Steroids and tear substitutes were given postoperatively for 3 weeks after excision. On follow-up, the lesion had healed properly with minimal conjunctival scarring and is under regular follow-up in our hospital.3 Progeria can present with a wide range of systemic and ocular manifestations. Our case presented with pyogenic granuloma, a benign vascular proliferation of immature capillaries that usually denotes sequelae of incomplete surgical or traumatic wound healing.8 In a review of ocular pyogenic granulomas, 87 out of 100 cases had predisposing factors, while 13 cases had unknown etiologies.7 A case series showed no specific predisposing risk for pyogenic granuloma.9 Similarly, in our case, the patient did not have any definite clinical history which would indicate the lesion as pyogenic granuloma. These findings suggest that, even though specific etiologies do not exist, the diagnosis of pyogenic granuloma should be considered with its typical clinical presentation of rapid growth and distinctive pink fleshy appearance. In addition to pyogenic granuloma, our case is diagnosed with HGPS, which in itself can present with a multiple arrays of ocular abnormalities.10 Many of the ocular pathologies are based on individual case reports and are absent in our case. Common findings are loss of eyebrows and eyelashes, cataracts, and lipodystrophy of the orbital fat resulting in superior sulcus deformity.Currently, more than 142 patients with classical progeria syndrome i.e. Hutchinson-Gilford progeria syndrome have been reported worldwide.In summary, we have reported a patient with HGPS who demonstrated not only various orbital and ophthalmic manifestations of progeria but also a pyogenic granuloma on the conjunctiva that required an excision. The limitation of this case report is that genetic testing was not performed as it is not readily available and also the diagnosis can be clinical in HGPS. This case report contributes to the medical literature by providing insights into the ocular manifestations and diagnostic challenges associated with Hutchinson-Gilford progeria syndrome. It adds to the existing knowledge base, facilitates early recognition and appropriate management of ocular conditions, and improves the overall understanding of this rare genetic disorder among healthcare professionals.


Epithelioid sarcoma (ES) is a rare soft tissue malignant tumor with an uncertain histogenetic origin. It usually arises in soft tissues of the extremities, while ES in adrenal gland is extremely rare. There is no special clinical manifestation in the early stage, so it may be misdiagnosed and delay the treatment. We reported a 69-year-old male with an adrenal ES. The tumor was completely resected, and two months later, positron emission tomography-computed tomography(PET/CT) noted recurrence at the tumor bed and multiple metastases. The patient has been treated with chemotherapy with good effects. We summarize the radiological findings and immunohistochemical indexes of primary epithelioid sarcoma of adrenal gland, which may be useful to promote disease awareness and help to distinguish among other lesions. Epithelioid sarcoma (ES), a malignant mesenchymal tumor that occurs in extremities of adolescents, was originally described by Enzinger in 1970  identifi1WI and slightly high signals on fat suppression T2WI sequence. A central scar was noted in the lesion, which was hypointense on T1WI and T2WI. Upon enhancement scan, the lesion exhibited a slight to moderate heterogeneous enhancement with no enhancement in the central scar. Visualization of the right adrenal gland was not clear. There were no significant enlarged lymphnodes or metastatic foci. Gene detection and FDG PET/CT were not carried out due to economic reasons.A 69-year-old man with an incidental right adrenal gland mass was admitted to our institution. He had no relevant past medical history, and no family history was identified. Hematological examination did not reveal any abnormal findings. Adrenal function laboratory assay indices were normal, including cortisol, catecholamine and aldosterone. Initial CT scan revealed a round-like, well-defined slightly hypodense lesion in the right adrenal gland region measuring approximately 10.6×6.4×11.0 cm in size, with a CT value of about 34 HU. The right posterior liver, portal vein and upper pole of the right kidney were displayed Figure 1Given the extensive region of focus, laparoscopic right adrenalectomy and intestine adhesion relaxation were performed. Intraoperative findings showed that the right adrenal was occupied by a round-like mass with a diameter of about 10 cm, while the normal adrenal tissue was compressed. Due to the huge size, adhesion of the lesion and liver was obvious and the inferior vena cava was compressed. Pathological examination revealed an incomplete capsule in the adrenal tissue, whose cut surface was gray, reddish-gray and soft. Microscopically, polygonal cells were established to be distributed separately, scattered, in clusters orpatches, with large, vesicular nuclei and prominent nucleoli. Immunohistochemical analysis showed that tumor cells were positive for CK8/18, EMA, vimentin, CD10, Desmin, CD34, SMARCB1/INI-1, SOX-2, SALL4, p53 and Ki-67, while SMARCA4/BRG1, SMARCA 2(BRM), Cr, S-100, HMB-45, Pax-2, Pax-8, WT-1, CK5/6, Melan-A, CK-7, Alpha-inhibin, RCC, MyoD1, Myogenin and SMA were negative Figure 3Epithelioid sarcoma (ES), a malignant soft tissue sarcoma with a certain degree of epithelization, accounts for < 1% of soft-tissue sarcomas. The two pathological subtypes of ES are classical and proximal types. The conventional type is more common and is often located in superficial dermis or subcutaneous tissues of distal limbs of young adults, whereas the proximal type is quite often located in the proximal soft tissue of elderly patients, such as the limb band, trunk and pelvis. The tumor cells of this subtype are higher grade and demonstrate a rhabaoid phenotype, compared to the classical type.Because it is highly rare, accurate diagnosis of the primary ES of solid organs by radiologists is difficult. To elucidate on the disease, we report a case of adrenal ES, which is the fifth reported case of ES arising from the adrenal gland.1WI and T2WI, and homogeneous or heterogeneous enhancement, with no enhancement in the necrotic area. There is infiltration to adjacent tissues or bones in some cases , prone to central necrosis, and 20~30% with calcification. Lesion enhancement scan findings revealed a heterogeneous enhancing mass that descended and attenuated slowly . AngiosaIn previous studies, immunohistochemical analysis of adrenal ES revealed positive expressions of epithelial markers, cytokeratins, vimentin, CD34, and negative expressions of CD31, SMARCB1/INI-1 and S-100. In particular, the loss of SMARCB1/INI-1 is considered to be the most specific aspect of immunohistochemical features for the diagnosis of ES. SMARCB1/INI-1 is one of a component of SW1/SNF chromatin-remodeling complex, which is the main condition factor for regulating gene expression . It is nIn conclusion, we report a case of proximal adrenal ES, which is rare and not commonly suspected in clinical practice. However, due to the high aggressiveness, poor prognosis, and prone to recurrence and metastasis, radiotherapy and chemotherapy after radical surgical resection is the optimal treatment. Currently, definite diagnosis of adrenal ES depends on histopathologic and immunohistochemical characteristics, but imaging examination displays some features and plays a significant role in diagnosis and differential diagnosis, providing the relationship with nearby structures. Therefore, awareness of the above may be useful for clinical diagnosis and treatment.The original contributions presented in the study are included in the article/supplementary material. Further inquiries can be directed to the corresponding author.Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.HY reviewed the literatures and wrote the initial draft of this manuscript. YZ prepared the images and for collected the data of the patient. LW revised the manuscript for English grammar. All authors contributed to the article and approved the submitted version. JZ supervised and reviewed the manuscript.


Leiomyosarcoma with heterologous differentiation is relatively rare. To date, only 19 cases have been reported in the English literature. Heterologous components frequently show histological pleomorphism, while those exhibiting well-differentiated morphology are seldom reported. Here, we report a 34-year-old female diagnosed with leiomyosarcoma and developed abdominal wall recurrence 8 years after primary surgery. The recurrent tumor mainly comprised well-differentiated chondrosarcoma except a single focus of leiomyosarcoma. Due to the rarity and prolonged onset of such a transition, our case provides insight into the understanding of this phenomenon. A 34-year-old woman presented to our hospital with concerns of an abdominal mass with dull pain for several days. Physical examination found a palpable mass measuring 5–6 cm in diameter in the lower abdomen. Computed tomography revealed a multinodular mass measuring 7.6 cm × 4.1 cm × 6.5 cm located in the rectus abdominis muscle with multifocal calcification and uneven enhancement. Serum tumor markers such as CA125 (Cancer Antigen 125), SCC (Squamous Cell Carcinoma Antigen), and CA19-9 (Cancer Antigen 19-9) were all in the normal range. She had a history of “broad ligament leiomyosarcoma” 8 years prior and was treated with radical resection in our hospital. Because recurrence was suspected, she underwent a second surgery for abdominal mass excision. A tough and irregular mass was found beneath the lower abdominal wall during the operation. The mass invaded the subcutaneous fat layer and penetrated through the rectus abdominis muscle. No other intraoperative abnormities were found.Grossly, the tumor had a multinodular appearance in the abdominal muscles below the superficial fascia and intruded into the extraperitoneal adipose tissue. Nodules measuring from 4 to 9 cm were found isolated from each other. All nodules were irregular in shape, with sharp margins and well-demarcated outlines. The cut surfaces were all cartilage-like with a brittle texture, gray to gray‒white color and focal calcification .Microscopically, the vast majority of the neoplasm was composed of well-differentiated cartilaginous islands arranged in lobules . The bouTo clarify the relationship between recurrent and primary tumors, we reviewed the histomorphology of the former surgical specimen. The primary tumor was composed of two types of cells: spindle cells and epithelioid cells. Sarcomatoid-like spindle cells were arranged in the fasciculus or vortex with cigar-shaped nuclei and sparse eosinophilic cytoplasm under high magnification . EpithelThe final diagnosis was leiomyosarcoma with epithelioid differentiation. The patient then received 4 cycles of APDTIC  chemotherapy after radical surgery.The immunoprofiles of primary and recurrent tumor were listed in Taking into account both the overall medical history and the histopathological characteristics, the abdominal lesion was confirmed to be leiomyosarcoma with chondrosarcoma differentiation. The patient was in good condition after 6 cycles of combined chemotherapy .Although leiomyosarcoma is the most common malignant mesenchymal tumor of the uterus, such lesions containing heterologous components are rare, with fewer than 20 cases reported to date –9. The cMDM2 (In our case, the patient was initially diagnosed with leiomyosarcoma with epithelioid differentiation and experienced distant recurrence. The recurrent tumor was composed mainly of well-differentiated chondrosarcoma. Misdiagnosis can easily occur regardless of the immunotype and the patient’s medical history. The differential diagnosis mainly depends on the proportion and histologic type of the heterologous component. Cases in which chondrosarcoma and osteosarcoma are the major components, careful sampling and detailed observations are mandatory to discover the smooth muscle component. For leiomyosarcoma with liposarcoma differentiation, the differential diagnosis from dedifferentiated liposarcoma might be challenging. In such cases, molecular tests can provide helpful evidence, as liposarcoma is characterized by the amplification of MDM2 , 12. WheMDM2 . HoweverMDM2 . An accude novo malignancy can only be established if recurrence is completely excluded.The remote metastasis that occurred in our case, although after a long remission interval, urges us to pay sufficient attention to patients who are clinically stable. Regardless the proportion of the heterologous components, the diagnosis of a


Dieulafoy’s disease of the bronchial tree is a very rare condition. Few cases have been reported in the literature. It can be asymptomatic or manifest with massive hemoptysis. This disease should be considered among heavy smokers when recurrent massive hemoptysis is present amid otherwise normal findings. The treatment can be arterial embolization or surgical intervention. A 16-year-old girl was admitted to the emergency department due to hemoptysis with an unknown lesion in the bronchi. She had suffered massive hemoptysis and respiratory failure one week before admission. Fiberoptic bronchoscopy revealed a lesion in the bronchus of the right lower lobe, which was suspected to be a Dieulafoy lesion. Segmentectomy of the right lower lobe and excision of the lesion was carried out. The outcome for this patient was excellent. Dieulafoy’s disease is a rare vascular anomaly and it is extremely rare in the bronchial tree. In bronchial Dieulafoy’s disease, selective embolization has been suggested as a method for cessation of bleeding. Nevertheless, standard anatomical lung resection is a safe and curative alternative. Dieulafoy’s disease of the bronchial tree is a very rare disease. Few cases have been reported in the literature. It can be asymptomatic or can manifest with massive hemoptysis. This disease should be considered among heavy smokers with recurrent massive hemoptysis.The diagnosis can be confirmed by means of bronchoscopy, which shows aberrant arterial bleeding in the bronchial tree. Imaging, consisting of either normal chest X-ray or chest computed tomography (CT) scan, can be helpful in making the diagnosis, through ruling out other causes of hemoptysis.The treatment usually comprises arterial embolization. If this method is unavailable or unsuccessful, surgery can be another option for achieving a definitive cure.Here, we report a case of Dieulafoy’s disease in a girl who presented with massive hemoptysis, which was diagnosed by means of bronchoscopy and treated through segmentectomy.A 16-year-old nonsmoking girl was referred to our hospital because of an episode of massive hemoptysis. She had been admitted to a local hospital one week earlier because of this symptom and had developed respiratory failure, requiring mechanical ventilation for two days. After extubation and cessation of bleeding, she was referred to our hospital for further evaluation.On admission to the thoracic surgery department, she was conscious and extubated, without respiratory distress, but mildly anxious. Her vital signs were stable and she was afebrile. Physical examination on the head and neck, chest, abdomen and extremities showed that these were normal. Oxygen saturation in the ambient air was 98%. There was no longer any hemoptysis.Laboratory data including white blood cell (WBC) and platelet counts, hemoglobin and hematocrit, prothrombin time, partial thromboplastin time (PTT) and international normalized ratio (INR) were within normal limits. A chest X-ray was normal, while chest CT scans showed some patchy haziness in the right lower lobe and a very small lesion in the distal bronchus intermedius  and 2. TOn the next day, fiberoptic bronchoscopy was performed and this showed a lesion at the beginning of the bronchus of the basal segments of the right lower lobe, without evidence of active bleeding. The lesion originated from the mucosal surface, with a small clot over it. The mucosa surrounding the lesion was absolutely normal . No biopAn intraoperative frozen section study was negative for any malignant condition.The patient had a very smooth and uneventful postoperative course, in which she only presented pain, which could be controlled with ordinary analgesics. She was discharged on the sixth postoperative day. At an outpatient visit one week later, she did not have any serious complaint. Moreover, in the third, sixth and eighteenth months of follow-up, she was still asymptomatic without recurrence of any kind of hemoptysis.Although the diagnosis of this disease was clinical, further pathological studies showed few dilated vessels in the submucosa. This was compatible with a diagnosis of Dieulafoy’s disease .,,,,,,,Dieulafoy’s disease is a rare vascular anomaly consisting of a dysplastic artery in the submucosa. It is mostly seen in the gastrointestinal tract and is extremely rare in the bronchial tree. To the best of our knowledge, there are only a few reports of Dieulafoy’s disease of the bronchial tree in the English-language literature (,The pathogenesis of this disease is also unclear, but most reports state that it occurs in heavy smokers and presents with massive and recurrent hemoptysis. Dieulafoy’s disease of the bronchus may have a congenital origin, arising from either the systemic or the pulmonary circulation.Dieulafoy’s disease can be suspected when there is severe or massive hemoptysis in the absence of any significant abnormality on either chest X-ray or chest CT scan and in the absence of any medical or surgical history, as in our patient’s case. Bronchoscopy, preferably using a fiberoptic when the bleeding is not severe, may be diagnostic. It will usually make it possible to find both the source and the cause of the bleeding.,It has been suggested that, after the diagnosis has been made, angiography and embolization can be the preferred treatmentAlthough the bleeding recurrence rate in patients whose hemorrhaging has stopped spontaneously is not known, physicians cannot take the risk of not initiating any interventions. If selective embolization is unavailable or if it fails, surgery can be lifesaving. Even in patients whose bleeding stops spontaneously, surgery can have a role in prevention of life-threatening hemoptysis.Dieulafoy’s disease is a rare vascular anomaly and is extremely rare in the bronchial tree. It should be considered as a diagnosis when there is severe or massive hemoptysis in an otherwise normal patient who has nearly normal chest imaging. Bronchoscopy is diagnostic. In bronchial Dieulafoy’s disease, selective embolization has been suggested as a method for cessation of bleeding. When angioembolization fails or is unavailable, surgical resection consisting of either segmentectomy or lobectomy can be lifesaving for these patients.


Pleomorphic adenoma affects mostly the parotid gland (85%), submandibular glands (5%), and the minor salivary glands (5%). They are usually referred to as benign mixed tumors because they are composed of both epithelial cells, which are cells from the body's surface, and myoepithelial cells, present inside glands to aid in secretion. An 88-year-old male who had an ulcer for 10 days and a three to four-month-old swelling on his palate arrived in the OPD. On examination, at the intersection of the hard and soft palates, there is a single, well-delineated, nodular exophytic development of small salivary glands left to the median half . For further and confirmatory diagnosis, fine needle aspiration cytology, immunohistochemistry tests, and radiodiagnosis, i.e., X-rays, were performed which revealed periapical abscess and swelling on palate unassociated with pus discharge. Surgical removal of the swelling and extraction of the root stump was done. The patient was asked to stop smoking strictly. Post-operatively, the patient was given suitable medications. The most frequent salivary gland tumor, pleomorphic adenoma, is sometimes referred to as a benign mixed tumor due to its dual genesis from myoepithelial and epithelial components . It makeA few pathological characteristics, such as invasiveness, have also been studied; extracapsular extension and invasion of less than 1.5 mm predict greater mortality and recurrence . AlthougAn 88-year-old male patient came with a chief complaint of swelling on the palate for three to four months with an ulcer for 10 days. Swelling was present left to the median half of the hard palate. It gradually increased its size and was not associated with any pus discharge. History dates back to four months ago when the patient first experienced swelling present left to the median half of the hard palate. Swelling was not associated with pain and gradually increased in size. Ten days ago, a single ulcer was observed associated with a lesion about 1 cm in size and irregular shape. There was no relevant past dental and medical history. The patient has smoked 15 bidis daily for the last 70 years.During the clinical examination, no significant or relevant extra-oral findings were present Figure .On intra-oral examination, swelling of about 1 inch in diameter with ulceration of 1 cm in irregular shape left to median half on the hard palate was observed.On local examination/examination of the lesion, a solitary, well-delineated, nodular exophytic growth of minor salivary glands left to median half on the hard was present, which was about 1 inch in diameter. The swelling was not associated with pus discharge. The surface epithelium of the lesion burst out to give an ulcerated surface of about 1 cm in size, which was irregular in shape and had a sloping edge associated with mild pain Figures -3, off aOn radiological examination, ill-defined radiolucency at 23, 24 with abscess (Figure Following the findings, the patient was informed of his condition and urged to have surgery to completely remove the cyst, which was done successfully.Operative notesThis case was referred to an oral surgeon. The pleomorphic adenoma was successfully removed surgically with sufficient margins, and root stump extraction was also done.The surgery performed was hemimaxillectomy with the reconstruction of the temporalis muscle flap. It was performed under general anesthesia. Submental intubation was done as nasal intubation was not possible. No post-operative complications were there. A complete cessation of smoking was advised.Pleomorphic adenoma is a complex morphological epithelial tumor that is embedded in a mucopolysaccharide stroma and has both epithelium and myoepithelial components organized in different ways. The surrounding salivary parenchyma, which the tumor compresses and causes to fibrose, is what causes the "false capsule" to form.Pleomorphic adenoma of the minor salivary gland of the hard palate is a rare occurrence. It accounts for 5-7% of cases among the various salivary gland tumors. In pathology laboratories, small salivary gland tumors are thought to account for 0.35-1.5% of all biopsies. The palate lip, nasal cavity, pharynx, larynx, and trachea are the most often occurring locations. It usually presents with slow-growing, painless, well-delineated, and nodular exophytic growth. Depending on the location of the tumor, minor salivary gland tumors can cause a range of symptoms, such as dysphagia, hoarseness, dyspnea, trouble chewing, and epistaxis.Finding the right diagnosis can be tough. Fine needle aspiration cytology, MRI, immunohistochemistry, and special stains are used to make the diagnosis. Surgery is the primary form of therapy and has a very good cure rate. High-grade disease, cases of uncertain resection adequacy, lymph nodes, and peri-neural invasion are treated with post-operative radiation. Additionally, patients may be given the choice of receiving both radiation and chemotherapy . On the The patient came with complaints of swelling without pus accompanied by pain. The lesion had a firm consistency, was unilocular, and had well-defined margins. It was also immovable. The mucosa over the lesion was stretched and non-pinchable with a non-bleeding ulcer. All these findings were suggestive of pleomorphic adenoma and chronic generalized periodontitis, and it was confirmed pathologically and through other tests. A surgical hemimaxillectomy with reconstruction of the temporalis muscle was done, and there were no complications post-operative. The patient was given medications and was advised not to smoke ever again.


Pelvic neurofibromas are benign and uncommon retroperitoneal masses. They arise from Schwann cells. One of the most common types of these benign tumors is intraneural neurofibromas, which are solitary, sporadic, and not associated with neurofibromatosis type 1. Here we discuss a case of pelvic neurofibroma in a 20-year-old male who presented with chronic pelvic pain. He had no positive family history of genetic disorders. On physical exam, just a partly firm mass without mobility in the hypogastric region was detected. Ultrasound and Computed tomography scan showed pelvic retroperitoneal mass superior to the urinary bladder with extension to the rectovesical pouch and invasion of the posterior wall and dome of the bladder. The patient underwent laparotomy revealing an infiltrative retroperitoneal mass with the invasion of the posterior wall, dome, and trigone of the bladder. Histopathological findings showed neurofibroma. A 20-year-old male presented to the general hospital and reported 6 months of progressively increasing pelvic pain, fatigue with occasional urinary frequency, and incontinency. He had no history of bowel or genital symptoms or complaint of limb numbness. There was no family history of genetic disorders.In physical examination, the general appearance was normal. Heart rate and respiratory rate were 80 beats/min and 22 breaths/min respectively. The temperature was 37°c and the blood pressure was 145/100 mmHg.No evidence of skin lesion was present. No neurological sign was detected. A partly firm mass without mobility in the hypogastric region was detected on abdominal palpation. The digital rectal examination did not show any abnormality.At admission time, an examination of other systems showed no abnormalities.Lab data revealed an erythrocyte sedimentation rate of 120 mm/h and C-reactive protein of 70 mg/L. The complete blood count test findings were normal. Blood urea nitrogen and serum creatinine were in the normal range.Urinalysis showed microscopic hematuria (Blood 1+ and red blood cell 30-40/HPF).On ultrasound, both kidneys showed normal appearance and size. No evidence of hydronephrosis was present. An infiltrative heterogeneous hypoechoic mass was seen over the urinary bladder dome with extension to the lateral wall of the pelvic cavity and rectovesical pouch, without a sharp margin measuring 125 × 45 × 65 mm. The mentioned mass had a pressure effect on the bladder wall, causing an undulating appearance of its wall. The prostate and seminal vesicles seemed to be normal. Soft tissue sarcoma, nerve sheath tumors, and retroperitoneal fibrosis were considered the differential diagnosis.A CT scan was recommended due to the patient's mass. It showed an enhanced soft tissue density mass in the pelvic cavity, superior to the urinary bladder with extension to the rectovesical pouch without calcification or macroscopic fat density. The mass invades the posterior wall and dome of the bladder. A few pre-aortic lymph nodes with a maximum short axis diameter of 13mm were also seen . Tissue The patient underwent laparotomy with a midline incision. Intraoperative findings showed a large retroperitoneal pelvic mass which was infiltrative and had an invasion of the posterior wall, dome, and trigone of the bladder. In the following, excision of the bladder and seminal vesicles, until the prostate apex was done and sent specimens for frozen pathology which showed the Posterior urethra free of pathologic involvement. Radical cystectomy and orthotopic diversion were done. The patient's recovery was uneventful, and he was discharged 7 days after the drain was removed.Neurofibromas are benign peripheral nerve sheath tumors that are usually solitary and sporadic. They arise from Schwann cells which are a type of glial cells surrounding neurons Neurofibromas subtypes include intraneural, cutaneous , plexiform, and massive diffuse soft tissue The most common type is intraneural neurofibromas, which are solitary and sporadic without association with neurofibromatosis type 1. However, the plexiform subtype of neurofibroma has a strong association with neurofibromatosis type 1. neurofibromas are found in various anatomical sites but rarely in the retroperitoneal location in the pelvis The peak incidence of localized intraneural neurofibromas (in sporadic form) is at 20-30 years old Clinical features depend on which anatomical area is involved in the retroperitoneum. Involving the urinary bladder can cause hematuria, dysuria, recurrent urinary tract infections, irritative symptoms, and pelvic mass; as in our case who presented with urinary frequency and hematuria Imaging modalities including ultrasonography, MRI, and computed tomography (CT) can be helpful in the early detection of these lesions, their extension, and in assessing possible malignant transformation ,8.On histology, exam neurofibroma is composed of collagenous matrix and spindle cells (medium-sized). The spindle cells are mostly positive for S-100 protein on immunostains as seen in our patient. Collagenous matrices are usually positive for Alcian blue. Neurofibromas are negative for cytokeratin and Epithelial membrane antigen Soft tissue sarcoma (low-grade rhabdomyosarcoma and leiomyosarcoma), leiomyoma, and malignant peripheral nerve sheath tumor (low grade) can mimic neurofibroma on histology. Clinical findings and histological and immunohistochemical exams are helpful for a definitive diagnosis The malignant transformation is rare in neurofibromas except for the plexiform subtype, so the prognosis of the majority of cases is very good. Local excision is the commonest treatment reported in the literature. The extent of excision depends on the tumor size and its invasion of adjacent structures Data sharing does not apply to this article as no datasets were generated or analyzed during the current study, but details from the clinical records are available from the corresponding author upon reasonable request.Ethics approval and patient consent were obtained.Written informed consent for the publication of this case report was obtained from the patient.


Large extra cervical-type posterior subserosal leiomyomas originating from the cervix are extremely rare. Our case concerns the surgical treatment of a large posterior pedunculated subserosal extracervical leiomyoma of the uterus with extension to the retroperitoneal space, which was associated with chronic low back pain. A 45-year-old patient, without menstrual disorders and with a medical history of chronic low back pain with sciatica, was referred for gynecological evaluation. The gynecological examination revealed the presence of a large pelvic mass, which occupied the pouch of Douglas. Preoperative imaging confirmed the presence of a solid pelvic mass, but its origin could not be clarified. Neither transvaginal ultrasound nor MRI could establish the diagnosis of extracervical leiomyoma of the uterus. Based on the clinical and imaging findings, surgical management of the patient was decided with laparotomy. Intraoperatively, a large extracervical pedunculated leiomyoma was found, which originated from the posterior wall of the cervix and extended into the retroperitoneal space. An abdominal total hysterectomy and bilateral salpingo-oophorectomy were performed. The procedure had significant surgical difficulties. The postoperative course was uneventful. Three months after surgery, the patient reported relief of symptoms. This paper highlights a brief review of cervical leiomyomas, highlighting the important difficulties regarding the preoperative diagnosis and surgical management of these patients. Fibroids or leiomyomas or myomas are common benign neoplasms of the uterus. Uterine leiomyomas originate from smooth muscle cells of the myometrium, whose growth depends mainly on the levels of endogenous estrogens. They usually occur in premenopausal women (<50 years) and are more common in the black race as compared to the white ,2. LeiomLarge cervical fibroids (>10cm) are even rarer . CervicaThis case report emphasizes the significant surgical difficulties that may arise in the management of extracervical leiomyomas of the uterus with extension to the retroperitoneal space, due to the increased risk of intraoperative bleeding and injury to adjacent organs. At the same time, it is pointed out that, despite their rarity and the considerable difficulties involved in preoperative diagnosis, extracervical leiomyomas of the uterus, particularly those growing in the retroperitoneal space, should be included in the differential diagnosis of chronic low back pain from radiculopathy in order to ensure health and quality of life for these patients.Our case concerns a patient aged 45 years old who was referred from the orthopedic clinic for examination at the gynecology outpatient clinic of the General Hospital of Trikala. The reason for the referral of the patient for gynecological evaluation was the existence of a possible pelvic mass, as it was depicted during the imaging of the lumbar spine with an MRI. An MRI revealed no evidence of bone damage or intervertebral disc disease in the lumbar spine. The patient complained of symptoms of persistent low back pain with accompanying radicular pain in the left leg for the last two years. As reported by the patient herself, the symptoms did not completely resolve after the analgesic medication that she occasionally received. The patient had a regular menstrual cycle and had given birth to three children by vaginal delivery. No previous surgeries were mentioned in the personal medical history. Medical disorders such as hypothyroidism, arterial hypertension, and anxiety disorders were reported and well-regulated with appropriate medication.During the gynecological examination, a large palpable pelvic mass was found, but it could not be determined whether it was a uterine mass or an adnexal mass. The cervix of the uterus was observed to have smooth, squamous epithelium but appeared to be displaced. Transvaginal ultrasound revealed a large solid echogenic mass (maximum diameter of about 11 cm) occupying the pouch of Douglas, but it was not possible to clarify the exact origin of the pelvic mass  were within the normal range.Based on the clinical and imaging findings, it was considered that there is a strong possibility that the symptom of persistent low back pain with concomitant sciatica was due to compression on the lumbar nerve roots from the large pelvic mass that occupied the pouch of Douglas. After consulting the patient about the available treatment options, it was decided to perform a diagnostic laparotomy, with the possible necessity of performing an abdominal total hysterectomy. Intraoperatively, a large extracervical leiomyoma was found, which, through a wide-based pedicle, emerged from the posterior wall of the cervix and which, with retroperitoneal extension, occupied the entire pouch of Douglas Figure .A marked displacement of the anatomical structures in the pelvic area was noted. Careful dissection of the distal ureters up to their entry into the bladder was performed while attempting to preserve their vascularization and avoid injury to the iliac vessels and their branches passing through the anatomical area. In this way, the separation of the cervical leiomyoma from the adjacent structures was safely completed, and it was possible to extract it with the uterus and adnexa Figure .Surgery was completed without significant blood loss. Histological examination of the surgical specimen confirmed the diagnosis of extracervical leiomyoma of the uterus. Microscopic examination of the tumor revealed mild to moderate cellularity, with the absence of mitoses, necrosis, and atypia. In addition, vitrification of the stroma and vascular congestion, probably due to compression, were found in some places Figures -6.After a smooth postoperative course, the patient was discharged from the clinic on the fourth postoperative day, with consultation for re-examination at the gynecological outpatient clinic. Three months postoperatively, the patient reported complete relief of symptoms of chronic lower back pain with accompanying radicular pain.The preoperative diagnosis and surgical management of patients with extracervical leiomyomas are estimated to be much more challenging compared to the management of those patients in whom fibroids are located in the uterine corpus . UrinaryThe diagnostic value of ultrasonography and MRI, or a combination of the two, is considered to be important in the preoperative diagnostic evaluation of cervical leiomyomas of the uterus. Recently, in 2021, Ferrari et al. showed that the diagnosis of cervical leiomyoma can be made by ultrasound in about half of the patients (48.6%) and by MRI in 39% of cases . NeverthThe treatment of cervical leiomyomas of the uterus is mainly surgical. Hysterectomy with laparotomy or laparoscopy is recommended as the primary treatment option for older women. Myomectomy and trachelectomy should be offered to patients who wish to preserve fertility and achieve a future pregnancy. Myomectomy is advisable to be limited to fibroids smaller than 10 cm. In addition to the increased risk of intraoperative bleeding, the risk of recurrence and the need to perform a hysterectomy should be included among the common complications associated with cervical leiomyoma resection . AdditioRegardless of the type of surgery  chosen to treat cervical leiomyoma, the operation is expected to be difficult. A well-organized preoperative evaluation of the patient and thorough knowledge by the surgical team of the pelvic anatomy, which is usually deformed by the cervical leiomyoma, especially when it extends between the broad ligaments or in the retroperitoneal space, are essential for a good outcome of the surgery. In addition, the surgical skills of the team of gynecologists and a well-organized operating room should be included in the requirements for the successful and safe treatment of cervical leiomyomas of the uterus. Significant intraoperative blood loss due to increased cervical blood perfusion, inadequate surgical plan, and increased risk of bladder, rectal, or ureteral injury are complications that often occur in the surgical treatment of cervical leiomyomas ,14. UretIn our patient, intraoperatively, a remarkable extension of the extracervical posterior pedunculated subserosal leiomyoma (>10cm) in the retroperitoneal space and a significant degree of displacement of adjacent anatomical structures, including the ureters, were detected. This condition required careful dissection of the ureters from the adjacent tissues, particularly the right ureter, which had greater misplacement, in order to preserve sufficient blood supply and avoid injury. Furthermore, avoiding injury to the great blood vessels that pass through the anatomical area was of primary importance in order to achieve a safe dissection of the cervical leiomyoma and to exclude it along with the uterus. In our patient, no ureteral stents were placed preoperatively because there was no such possibility in our hospital. This fact further increased the degree of difficulty of the surgery.Large cervical leiomyomas are rare. Early diagnosis of chronic low back pain due to radiculopathy-associated extracervical leiomyoma is critical for the optimal surgical treatment to be applied immediately. The preoperative diagnosis and surgical treatment of large extracervical posterior cervical leiomyomas of the uterus is a challenge today in routine clinical practice. Hysterectomy, myomectomy, or trachelectomy, depending on the age of the patient and her desire or not to preserve fertility, in well-organized medical centers by experienced surgeons, is considered essential for the treatment of symptomatic cervical leiomyomas of the uterus in order to ensure the health and quality of life of these patients.


Lymphangiomas are rare, painless, benign tumors in infancy or early childhood resulting from a congenital malformation of the lymphatic vessels with variable clinical appearance. We report the case of a six-year-old male child who presented with a micronodular surface of the tongue, a burning sensation, difficulties during swallowing and mastication, as well as speech disturbances. Histological examination of the excised tongue specimen showed an angiomatous lesion of the lamina propria that comprised many wide thin-walled spaces. MRI examination revealed an area of disparate T2 signal maximum diameter of 2-3 cm with cystic texture in the middle and frontal left part of the tongue. The lesion was diagnosed as a lymphangioma and was managed through pre-operative embolization and surgery. Tongue lymphangiomas may lead to aesthetic problems, functional issues , psychological disturbances, poor oral hygiene, and occasional bleeding associated with oral trauma. A major fraction of patients also experience infections, often leading to a significant increase in lesion size. A combination of preoperative embolization and surgical excision could be chosen considering its large size and the age of the patient, and to further eliminate the possibility of recurrence. Early diagnosis and radical treatment are critical in its management.  Lymphangiomas consist of rare developmental lesions of the lymphatic vessels, mainly found in children under five years of age. They are present as micro-cysts or lobulated masses and are commonly located in the head or neck area. They were described by Redenbacher in 1828 for the first time and in 1854, Virchow described the first tongue lymphangiomas .Sabin et al., in 1902, proposed that the lymphatic system is derived from primordial endothelial buds originating from the developing venous system starting at 6.5 weeks of gestation. These buds coalesce to form plexi at the neck. Peripheral malformations arise from abnormal plexus networks in the periphery of the body . There aGenerally, there is no sex predilection, but cases of small-sized lymphangiomas (< 1 cm) of the alveolar mandible ridge show a 2:1 male-to-female ratio . The freAccording to De Serres et al., lymphatic malformations can be classified into five groups ranging from single superficial microcystic lymphatic malformation of the tongue (stage I) to extensive microcystic lymphatic malformations involving larger areas of the tongue, floor of the mouth, and further cervical structures (stage V) . The large extension of lymphangiomas may lead to aesthetic, functional, occlusal, psychological disturbances, poor oral hygiene, and bleeding from the tongue associated with oral trauma. About 70-80% of patients with lymphatic malformation experience infections, which are often associated with a significant increase in the size of the lesions . Early dA six-year-old male patient presented with a micronodular, enlarged, surface of the tongue, burning sensation, difficulties during mastication and swallowing, difficulties in speech, irregular bleeding of the tongue, and quite poor oral hygiene. According to his parents, the tongue was apparently normal until the age of three years, after which a swelling, that was slowly growing, was observed. In the beginning, it appeared as a painless soft mass that gradually enlarged and then remained stable for a long period. Neither facial asymmetry nor other anatomic head and neck malformation was observed. All these symptoms had a big impact on the patient’s quality of life. The size of the tongue was normal, about 8 cm. There was no relevant family history. The clinical examination revealed a hyperplastic overgrowth extending to the anterior two-thirds of the dorsal surface of the tongue characterized by small soft irregular vesicles with a red-brownish-yellow color. The child was in good general condition, without signs of obstruction of the superior respiratory tract.The differential diagnosis included hemangioma, lymphoepithelial cyst, mesenchymal neoplasms, vascular malformations, and lipoma. A visceral skull MRI was performed with a 1.5T MRI . Incisions were made with the burden of T1 and T2 time before and after the intravenous administration of the paramagnetic contrast medium. The MRI examination revealed an area of disparate T2 signal maximum diameter of 2.3 cm with cystic texture in the middle and frontal left part of the tongue. These findings could be likely related to lymphangioma and less likely to hemangioma. The major salivary glands and the base of the oral cavity appeared to be normal. No pathological signs such as enlargement of lymph nodes of the neck were observed.The treatment options were surgical removal, laser ablation with long-pulsed neodymium-doped yttrium aluminum garnet (Nd-YAG) or carbon-dioxide CO2) laser, embolization, radiofrequency or sclerotherapy, interferon, and corticosteroids. Cryotherapy and laser can be considered for superficial and localized lymphangiomas. Due to the lesion's large size and the location of the damage, surgical excision with preoperative embolization was chosen as a treatment option. The decision was made after carrying out the necessary blood tests and the MRI. No immediate interventions were under consideration. Analytically, the immediate preoperative direct embolization with the sclerosing agent, Sodium tetradecyl sulfate (the most common sclerosing agent), was performed Figure . Afterwa laser, eHistological examination (on hematoxylin & eosin-stained sections) of the tongue excision specimen showed multiple vascular lesions of the lamina propria that comprised many wide thin-walled spaces, lined by a row of endothelial cells without atypia. Some of them were filled with red blood cells. On immunohistochemical stain for antigen D2-40, the endothelial cells were positive. In epithelium, the vascular spaces aggregated in clusters and were separated by delicate fibrous bands (septa). A few vascular spaces extended to the subjacent striated muscle fibers. Mild inflammatory cells infiltrating the stroma, consisting of lymphocytes and plasma cells, were also found. The combined clinical, imaging, and microscopic findings settled the final diagnosis of an oral (tongue) lymphangioma . The subsequent follow-up plan was to see the patient every year after the operation until a teenager.The rarity of lymphangiomas and their preference for the head/neck area and oral cavity is characteristic -5,10-13.Lymphangioma of the tongue reveals a granular appearance on the surface, microcysts with a thin wall containing lymph, microscopically. Their color ranges from translucent to red. Tongue lesions are mostly slow-growing but some develop quickly, as in our case . OccasioDespite the large size of the lesion in the current case, the tongue was only superficially involved. Overall, the capillary lymphangioma consists of small vessels, in contrast to the cavernous lymphangioma with larger lymph channels and the cystic lymphangioma with large macroscopic cystic spaces. The lymphatic endothelium reveals positive staining for marker D2-40. Small blood vessels positively stained for CD31 are also present in the lamina propria ,14,15. PThe clinicopathologic differential diagnosis for lymphangioma includes vascular malformations, hemangioma, amyloidosis, lipoma, congenital hypothyroidism, neurofibromatosis, papillary hyperplasia of the palate, lymphoepithelial cyst, lingual thyroid, pyogenic granuloma, other mesenchymal neoplasms, and oral papillomatosis. In contrast to hemangiomas, spontaneous regression of lymphangiomas is rare, but it usually presents as a slow progressive enlargement. Hemangiomas are vascular tumors that can appear similar to lymphangiomas, but they are composed of blood vessels rather than lymphatic vessels. Vascular malformations are abnormal collections of veins that can sometimes be confused with lymphangiomas especially when they include deeper tissues. Cystic hygromas are fluid-filled sacs that typically occur in the head and neck region. Lipomas are fatty tumors sometimes similar to soft tissue lymphangiomas, characterized by painless and slow growth. Both benign and malignant tumors can manifest similarly to lymphangiomas ,16. The Total removal may not be possible in all cases, depending on the size or involvement of vital structures. No treatment for non-enlarging lymphangiomas of the tongue has been also proposed due to the high recurrence rate. USG, CT, and MRI can be used to define the location of the lesion in relation to the neighboring structures involving the muscle . SurgicaTongue lymphangiomas may lead to aesthetic problems, functional issues, psychological disturbances, poor oral hygiene, and occasional bleeding. A combination of preoperative embolization and surgical excision could be chosen in younger patients with large lesions. In the present report, preoperative embolization and surgical excision gave ideal results. Although lymphangiomas are relatively common, there is a need for more literature about the efficacy of embolization in young patients. Long-term follow-up is necessary for early detection of possible relapse. Early diagnosis and radical treatment are effective. Also due to variability in clinical presentation and the potential overlap of symptoms with other conditions, it is crucial for the healthcare provider to perform a thorough evaluation which may include imaging studies MRI, CT scans, and biopsy to accurate diagnoses and determine the appropriate treatment plan for patients. Consulting with specialists such as oral maxillofacial surgeons and radiologists can be beneficial in complicated cases.


We present the case of a 73-year-old woman who was incidentally found to have a malignant Brenner tumor (MBT) of the ovary during an evaluation for deep vein thrombosis (DVT). The patient presented with swelling in her left leg, non-healing ulcers, weakness, and numbness in her lower limbs. Imaging studies revealed a large multiloculated cystic mass with areas of calcification in the left adnexa extending to the upper abdomen toward the gallbladder fossa. The patient underwent exploratory laparotomy with removal of the ovarian cyst, later diagnosed as a focal MBT in a background of borderline Brenner tumor. Brenner tumors of the ovary are a rare subtype of ovarian neoplasm that accounts for less than 2% of all ovarian tumors. MBTs are even rarer, comprising less than 5% of all Brenner tumors. To our knowledge, this is the first reported case of an MBT incidentally found in a patient with DVT. Malignant Brenner tumors (MBT) are an uncommon and aggressive type of ovarian neoplasms that make up less than 5% of all Brenner tumors and less than 1% of all ovarian malignancies. Brenner tumors are solid, usually unilateral ovarian tumors made up of abundant stroma, which contain nests of transitional-type epithelium similar to that of the urinary tract. These nests may occasionally be cystic and lined with columnar mucus-secreting cells. Brenner tumors are categorized into benign, borderline or proliferative, and malignant subtypes, with MBT being particularly rare and characterized by stromal invasion under the microscope.MBT usually affects women during the perimenopausal and postmenopausal periods, and symptoms such as abdominal distention, pelvic discomfort, or postmenopausal bleeding may be present. Surgical excision and pathological evaluation are necessary to diagnose MBT because it lacks specific imaging features. Treatment consists of surgical resection, and the prognosis of MBT is generally favorable -4.A 73-year-old African American woman with a medical history of hypertension, hyperlipidemia, type 2 diabetes, and four cerebrovascular accidents presented to the hospital with a recent diagnosis of left lower extremity deep vein thrombosis (DVT). The patient reported experiencing swelling in her left leg for the past few months, along with non-healing ulcers in both legs and weakness and numbness in her lower limbs.On physical examination, the patient had 2+ pitting edema and small ulcers in her lower extremities. Dorsalis pedis pulses and posterior tibial pulses were bilaterally absent. An abdominal examination revealed present bowel sounds and a soft, non-tender, and distended abdomen. Venous and arterial Doppler of bilateral lower extremities confirmed the diagnosis of DVT. The patient was already on anticoagulation, and a venogram and thrombectomy were planned due to the edema and pain.A week later, the patient was brought to the hospital due to sudden onset tachycardia. She had a pulse rate of 140 s and was admitted for cardiovascular event evaluation. The patient's organs and systems were reviewed, and she was negative for chills or fever, vision disturbances, cough, and shortness of breath. However, she did have bilateral leg edema, left leg pain, urinary frequency, and incontinence. A computed tomography (CT) scan of the chest was obtained to ensure there was no pulmonary embolism. Although there was no evidence of acute pulmonary embolism, an 8.8 cm cystic area lesion was visualized in the anterior upper abdomen. A CT scan of the abdomen and pelvis revealed left mild hydronephrosis and a large multiloculated cystic mass with areas of calcification within and multiple septations extending from the left adnexa to the upper abdomen toward the gallbladder fossa, measuring 12 cm anterior-posterior, 21 cm transverse, and at least 20 cm in height , another aggressive high-grade neoplasm with similar histologic features, can be challenging. The histological features of TCC include a high mitotic rate, severe nuclear atypia, and extensive necrosis. Immunohistochemical markers, such as CK7, p16, and p63, can aid in the differentiation of MBT and TCC [There are other types of ovarian tumors, such as serous, mucinous, endometrioid, and clear cell carcinomas, as well as benign Brenner tumors, teratomas, and metastases from urinary tract tumors, which should be considered in the differential diagnosis of MBT. Metastasis from other organs, such as the urinary bladder or colorectum, should also be ruled out .Patients with MBT typically experience symptoms similar to those of other epithelial ovarian carcinomas, such as abdominal pain, distension, and urinary urgency. Imaging studies can assist in the preoperative differential diagnosis of malignant and benign Brenner tumors, and MBTs usually contain both solid and cystic components. Most MBTs are unilateral, high-grade tumors with a median size of 10 cm, and most patients present with localized disease ,6.Surgery is the standard of care for all epithelial ovarian tumors, including MBT. Although the prognosis for MBT is generally good, there is currently no standardized chemotherapy regimen for the treatment of MBT due to the limited number of reported cases ,2.Thorough diagnostic evaluation is crucial in patients who present with symptoms related to DVT, as emphasized in this case report. The incidental finding of a large ovarian cystic mass during the review of the patient's cardiovascular symptoms ultimately led to the diagnosis of MBT, a rare subtype of ovarian cancer. The successful removal of the tumor through surgery underscores the importance of early detection and prompt treatment.