Meningiomas are the most common central nervous system tumor. They are typically benign neoplasms but may produce neurological symptoms due to mass effect. Meningiomas may also extend to extradural locations; however, these account for only a small percentage of all meningiomas. Most extradural meningiomas arise in intraosseous locations, usually within the cranial bones or vertebrae. However, this is a rare case of extradural extension of an asymptomatic intracranial meningioma to the proximal humerus in the absence of any musculoskeletal symptoms. To the best of our knowledge, this presentation of an extradural intraosseous meningioma has not previously been reported in the literature. We present a case of an incidental intraosseous meningioma in a 66-year-old man. This patient was initially being screened for metastasis of stage IA1 adenocarcinoma of the lung, and a positron emission tomography (PET) scan revealed a focus of activity in the proximal diaphysis of the right humerus suspicious for malignancy. The upper extremity magnetic resonance imaging (MRI) demonstrated an indeterminate lesion. Curettage of the humeral lesion revealed an intraosseous psammomatous meningioma without evidence of metastatic lung carcinoma. Our case report aims to illustrate the importance of considering alternative metastatic sources, such as intracranial meningioma, during the investigation of an indeterminate bony lesion. This is the first case to illustrate asymptomatic intraosseous meningioma in an appendicular skeletal location, highlighting the need for thorough source investigation. Meningiomas are the most common tumor of the central nervous system (CNS) and originate from the arachnoid cells of the meningeal layers covering the brain and spinal cord . MeningiContrasting to meningiomas of the axial skeleton, intraosseous meningiomas of the appendicular skeleton are reported with much less frequency. To our knowledge, there are only a sparse number of reports of intraosseous meningiomas within the appendicular skeleton -8. AdditCase reportThe patient, a 66-year-old male, was scheduled for a positron emission tomography (PET) scan following a continued interval increase in the size of a solid irregular lung nodule seen on computerized tomography (CT) measuring 9 mm x 8 mm in the right upper lobe. Biopsy of the pulmonary nodule revealed stage IA1 adenocarcinoma with strong expression of thyroid transcription factor-1 (TTF-1), supporting lung origin. At the time of the PET scan, a suspicious focus of activity was noted in the proximal diaphysis of the right humerus Figures , 1b. RigThe patient was referred to orthopedic surgery for further evaluation and biopsy of the indeterminate humeral lesion. On initial presentation to the orthopedic oncology clinic, it was noted that the patient had a history of adenocarcinoma of the right lung, seropositive rheumatoid arthritis, hyperlipidemia, and obesity. The patient also had a remote history of smoking, less than 10-pack-years. His physical exam was unremarkable. Notably, an examination of bilateral shoulders revealed a full range of movement without pain. There was no evidence of tenderness overlying the region of the proximal humerus, nor was there evidence of distal neurovascular deficit. No apparent abnormality was initially noted on the x-ray Figures , 4b. HowThe patient underwent surgery to obtain an open biopsy specimen for pathological analysis by curettage under general endotracheal anesthesia. Ancef was used as preoperative antibiotic prophylaxis, and a formal timeout was conducted. An incision was made on the anterior humerus in line with the deltopectoral interval. The basilar vein was retracted with the deltoid, and the proximal humerus was exposed. The lesion was confirmed with fluoroscopy, and an oblong window in the bone was created with bur and osteotomes. The underlying tissue was curetted and sent for intraoperative frozen section consultation. The sample appeared to be adequate; however, a definitive diagnosis was difficult to obtain immediately. A thorough curettage was performed and submitted to pathology. The entire cavity was treated with phenol for three minutes, followed by alcohol. This cycle was repeated three times, followed by thorough irrigation. HydroSet was mixed, and 10 mL was injected inside the cavity. Final x-rays were obtained. Once HydroSet was adequately set, the bone window was cleaned with a curette and infiltrated with phenol and alcohol, and it was replaced back on the outer cortex using a tamp. Closure was completed in standard fashion; sterile dressing was applied; and the patient was shifted out of the operating room.Postoperatively, the patient was allowed the motion of the shoulder and use of the arm as tolerated with a lifting restriction of 15 pounds. He was seen for postoperative follow-up where imaging revealed bony remodeling after surgery and good position of the bone graft substitute Figures , 5b.Tissue pathologyA curettage (2 cm x 2 cm x 0.6 cm aggregate) of bony tissue was submitted to pathology in formalin. Sections of the mass demonstrated an intraosseous psammomatous meningioma without evidence of metastatic carcinoma. Plump spindle and polyhedral cells with oval to elongate dark staining nuclei focally arranged in whorls were revealed within the mass Figures , 6b. NumMeningiomas are benign slow-growing tumors originating from meningothelial cells and are the most frequently occurring primary tumor of the CNS, accounting for 34%-37% of all primary intracranial tumors ,10-12. OIn this case, incidental discovery of the humeral intraosseous meningioma was secondary to screening for carcinoma of the lung via PET scan which revealed fluorodeoxyglucose (FDG) uptake at the proximal humerus Figure . A folloDistant metastasis is most commonly a feature of high-grade meningiomas  grade 3) and is estimated to occur in only ~0.76% of all cases . MetastaIt is estimated that up to 56.2% of metastatic meningiomas originate from benign meningiomas and that 31.3% of metastatic meningiomas are found incidentally . This stAt the time of writing this report, a review of the literature revealed no cases of clinically asymptomatic intraosseous meningiomas of the appendicular skeleton. In the present case, the patient was asymptomatic in that there was no pain or tenderness in the region overlying the proximal humerus, no abnormalities with shoulder range of motion, and no neurovascular deficits. Further, no evidence of focal neurological complaints was present, and thus, suspicion for an intraosseous meningioma was low until a screening MRI of the brain revealed an intracranial meningioma at the right cerebellopontine angle. Interestingly, metastatic meningiomas are reported to be clinically silent in ~31% of cases. However, accurate estimates of the frequency of clinically silent intraosseous meningiomas are not available in the literature. This does not exclude the fact that they may occur in similar frequency as painful appendicular intraosseous meningiomas, but rather that the number of painless metastatic intraosseous meningiomas may be underreported due to the lack of screening in the absence of symptoms. While there are no formal recommendations for routine screening following meningioma resection, providers may consider a surveillance protocol for patients who have WHO grade 3 meningiomas or tumors with characteristic high mitotic activity as determined by the Ki-67 proliferation index. Further, as lower-grade meningiomas are also known to metastasize, it would be warranted to include a diagnosis of intraosseous meningiomas when investigating indeterminate bony lesions.To the best of our knowledge, this is the first report of an asymptomatic intraosseous meningioma in a location of the appendicular skeleton. Although metastatic meningioma to intraosseous locations is reported to be rare, its actual incidence is difficult to quantify due to an unknown number of asymptomatic patients.


Uma mulher de 71 anos apresentou fadiga, dispneia e anorexia. O exame físico revelou um sopro diastólico grave grau III no foco mitral. O ecocardiograma transtorácico (ETT) mostrou uma massa oval no interior do átrio esquerdo (AE) estendendo-se de seu teto até a válvula mitral , causanA paciente foi encaminhada para retirada cirúrgica da massa . No exa A apresentação clínica depende da localização, da dimensão e da mobilidade da massa. Os sarcomas representam a maioria dos tumores cardíacos malignos primários. Eles são geralmente agressivos e o prognóstico geral é ruim, com uma sobrevida mediana de cerca de 9 meses. As técnicas de imagem não invasivas têm um papel crucial na investigação diagnóstica, permitindo a localização e a caracterização tecidual da massa, bem como a avaliação do comprometimento funcional e do envolvimento das estruturas circundantes. Todas essas características contribuem para um melhor planejamento cirúrgico. No entanto, elas não diferenciam entre os subtipos histológicos. Atualmente, o único tratamento com benefício de sobrevida é a ressecção cirúrgica.Tumores cardíacos são raros e, na maioria das vezes, benignos. A 71-year-old woman was admitted to our center, complaining of fatigue, dyspnea, and anorexia. Physical examination revealed a grade III low-pitched diastolic murmur at the mitral focus. Transthoracic echocardiogram (TTE) showed an oval mass inside the left atrium (LA) extending from its roof to the mitral valve , causinthpost-operative day due to septic shock secondary to a nosocomial respiratory tract infection.The patient was referred for the surgical removal of the mass . Upon hClinical presentation depends on the location, dimension, and mobility of the mass. Sarcomas account for most primary malignant heart tumors. These are usually aggressive and overall prognosis is poor,with a median survival of about 9 months.Noninvasive imaging techniques play a crucial role in the diagnostic workup, enabling location and tissue characterization of the mass, as well as an evaluation of the functional impairment and involvement of surrounding structures. All of these features contribute to a better surgical planning. However, these do not differentiate between histological subtypes.Currently, the only treatment with survival benefit is surgical resection.Cardiac tumors are rare and most often benign.


Paragangliomas are slow growing, benign tumours arising from embryonal neural crest cells. These neuroendocrine tumours, mostly originate from the adrenal glands in 90% cases in the form of Pheochromocytoma. Only 5–10% of these tumours are extra-adrenal and they can be located anywhere between neck and pelvic region along the distribution of sympathetic nervous system.A 15 year old boy visited our centre with a history of left sided nasal obstruction gradually progressive over a period of two months with recurrent history of bleeding from left nostril. There was no history of bleeding from any other sites or easy bruisability. On anterior rhinoscopy a single smooth surfaced fleshy mass was seen filling the left nasal cavity reaching up to anterior end of inferior turbinate deviating the septum to the right side. The mass was firm, sensitive to touch and pain, bleeding on touch. Posterior rhinoscopy did not reveal any choanal extension. Rigid nasal endoscopy was performed and the mass was seen attached to the postero superior part of the left side of the nasal septum.Complete blood count, biochemistry parameters and coagulation profile were all within normal limits. Computed Tomography (CT) scan of paranasal sinuses showed a homogenous soft tissue lesion measuring 41 mm × 32 mm × 24 mm in left nasal cavity. All the sinuses were clear. No bony erosion was seen on CT scan. On contrast study, there was heterogeneous enhancement indicating a vascular tumour .Figure 1Given the vascularity of the tumour on CT scan and considering the patient's age and gender nasal angiofibroma was suspected and hence biopsy was not taken and the patient was posted for endoscopic excision of the nasal mass under general anaesthesia after taking informed consent. The mass was removed completely by a regular endoscopic approach with removal of a cuff of septal perichondrium . The opeZellballen patterns which are separated by vascularised connective tissue stroma. There were no mitotic figures and cellular atypia (Histopathology of the excised specimen showed round to oval shaped tumour cells with moderate pale cytoplasm with round vesicular nuclei with dispersed chromatin. The tumour cells were arranged in nest or classical r atypia . The tumPheochromocytoma is usually a functional tumour and patients present with symptoms of catecholamine excess but paragangliomas of head and neck region are non-functional or non-chromaffin tumour.The origin of nasal paragangliomas is a controversial entity. Talbot et al. described paraganglionic cells at the distal maxillary artery in stillborn children supporting the theory of persistence of paraganglionic tissues along cranial arteries which could lead to development of head and neck paragangliomas.Nose and paranasal sinuses are the least common sites for this neoplasm in head and neck region. Nasal cavity is more commonly affected by the tumour than the sinuses. Lateral nasal wall, middle turbinate and ethmoid sinus are the commonly involved areas.Nasal paragangliomas are slow growing tumours with usual interval between the onset of symptoms and final diagnosis of two years or more. The tumours tend to be bilateral and multicentric and their incidence is 3% and it goes up to 26% among patients with positive family history which corroborates with genetic predisposition.Clinically patients present with mild to profuse recurrent epistaxis, nasal obstruction, rhinorrhoea and facial oedema. The tumours have been reported in patients aged between 8 to 89 years with middle aged females being most commonly affected. Anterior rhinoscopy reveals a reddish polypoidal mass in nasal cavity extending into the sinuses. CT scan reveals a soft tissue density expansile mass seen in nasal cavity and para-nasal sinuses with heterogenous contrast enhancement. Bony erosion is seen in case of malignant paragangliomas.Zellballen pattern which is surrounded by a rich capillary network of reticulin. Electron microscopy reveals the presence of cytoplasmic neurosecretory granules. The neoplastic cells exhibit affinity for chromic acid.Macroscopically paragangliomas are firm to hard, encapsulated tumours, greyish or rosy in colour. Microscopically the tumour consists of epithelioid cells also called chief cells with round nuclei and eosinophilic cytoplasm forming a nest like pattern known as On immunohistochemistry, paragangliomas express numerous markers and Chromogranin is ubiquitous in most of the paragangliomas as it is present in the secretory granules. Other markers are Synaptophysin, neuron specific Enolase and S-100 protein, the last one stains the peripheral sustentacular cells.Head and neck paragangliomas can be malignant in 4–19% cases and metastasis occur in 9% cases. Lymphnodes, lungs and bones are the prone sites for metastasis.We present a case of paraganglioma of nasal septum in a 15 year old boy. Paraganglioma of nasal cavity is an exceptionally rare entity and there have only been three previous reports of this pathology arising from the nasal septum. We conclude that paraganglioma should be included in the differential diagnosis of unilateral vascular nasal mass and nasal septum should be considered as one of the potential yet rare sites for this tumour. Complete surgical excision is not associated with recurrence. Histopathology and immunohistochemistry should be corroborated in doubtful cases.The authors declare no conflicts of interest.


Since the first description of paediatric intervertebral disc calcification (IDC) by Báron in 1924, only approximately 400 cases have been reported in the literature. Paediatric IDC combined with ossification of the posterior longitudinal ligament (OPLL) is an even rarer condition, with only 8 cases described in detail to date. In this paper, we present a review of the disease characteristics described in the relevant English language literature and discuss the possible mechanisms of lesion enhancement in contrast-enhanced magnetic resonance imaging (MRI).In May 2020, a 6-year-old Han nationality girl presented with the chief complaint of neck pain that had lasted for a week. She did not report a history of trauma or a past illness. On admission, there was no personal and family history, congenital diseases, or non-specific infections such as tuberculosis, among others. Further physical examination revealed that the movement of her cervical spine was limited. Computed tomography (CT) and MRI revealed ossification of the intervertebral discs and posterior longitudinal ligament (PLL) at the C4/5 levels and an absence of obvious spinal cord compression. When contrast-enhanced MRI was performed, significant enhancement was observed in the intervertebral discs and PLL at the C4/5 level. We adopted a non-interventional approach and performed an imaging re-examination 8 months later. Both the plain and contrast-enhanced MRI scans indicated swelling in the C4/5 intervertebral discs and disappearance of the previously observed enhancement in the nucleus pulposus (NP) and PLL at the corresponding levels; CT examination revealed that the ossified lesions had been completely resorbed.Obvious lesion enhancement in contrast-enhanced MRI is an extremely rare manifestation of paediatric IDC combined with OPLL. However, the exact mechanisms of this phenomenon remain unclear. We surmise that it may be caused by a series of biophysical changes related to vertebral endplate injury and repair, but further research will be required for in-depth investigation. Paediatric 0–18 years) or juvenile intervertebral disc calcification (IDC) is a rare, typically self-limiting disease of an unknown origin. Since the first report of pain in the thoracic vertebrae, pyrexia, and scoliosis in a 12-year-old boy by Báron in 1924, only approximately 400 cases in the 0–20-year age group have been reported in the literature 8 years o. OssificIn May 2020, a 6-year-old Han nationality girl presented with the chief complaint of neck pain that had lasted for a week. She didn’t take any medical treatment, but 3 days after the initial onset, her symptoms did not improve. She did not report a history of trauma, infection, or past illness. On admission, there was no personal and family history, congenital diseases, or non-specific infections such as tuberculosis, among others. Physical examination revealed that the movement of her cervical spine was limited. No torticollis nor palpable mass was found. Further neurological examination didn’t reveal apparently motor dysfunction or myodynamia deficit. A plain MRI scan revealed a localised reduction of the T2-weighted signal and soft tissue swelling in the PLL Fig. A at the Considering that the disease was benign and self-limited, and the patient’s symptoms were not very severe, we adopted a non-interventional approach. At 8-month-follow-up visit, the patient’s symptoms greatly improved and we performed an imaging re-examination. Both the plain and contrast-enhanced MRI scans indicated swelling in the C4/5 intervertebral discs and disappearance of the previously observed enhancement in the NP and PLL at the corresponding levels Fig. D and E. IDC is a rare disease that affects the intervertebral discs and the structures of surrounding vertebrae, muscles, and ligaments. It usually results in local pain or sensorimotor disorders. The potential aetiologies and pathophysiological mechanisms of the disease remain unclear, but IDC is typically self-limiting, with clinical symptoms disappearing spontaneously within several weeks or months . IDC hasn = 5, 22.7%), followed by the C3/4 level , C5/6 level , C2/3 level , C6/7 level , T7/8 level , and C6 level . Lesions mainly involved the cervical spine; a possible explanation for this phenomenon offered by Zhu et al. [Combined analysis of the relevant literature (8 cases + the current case study) revealed that the median age of patients with IDC combined with OPLL was 8 years (6–11 years) and the ratio of boys to girls was 3:6. The condition was not significantly correlated with trauma, with only 2 of the 9 patients having a definite history of trauma. The vast majority of patients exhibited non-specific clinical symptoms and were only diagnosed by chance through imaging examinations . White bl n = 3, .6%, C6/7u et al. . ExcessiWe summarized the imaging manifestations of IDC combined with OPLL in children in previous relevant research and our case Table . A revieNerlich et al.  stated tWhen all cases of IDC combined with OPLL in the literature were retrospectively analysed, it was found that OPLL occurred in the ligament connecting two vertebral bodies at the site of IDC in almost all cases, including the present case. Such OPLL belongs to the localised subtype, and there is a need to investigate the cause of this phenomenon. Chen et al.  held theIDC combined with OPLL is extremely rare in children with an unknown origin. MRI revealed obvious lesion enhancement in contrast-enhanced MRI, an extremely rare manifestation of paediatric IDC combined with OPLL. However, we must be aware of this possibility to avoid unnecessary excessive treatment or even surgery for the patient. The exact mechanisms of the enhancement remain unclear. We surmise that it may be caused by a series of biophysical changes related to vertebral endplate injury and repair, but further research will be required for in-depth investigation.


Conditions on pleura cover a broad range of pathology ranging from benign to malignant, which may potentially carry a poor prognosis and lead to high morbidity and mortality. Radiology plays a pivotal role in the diagnosis of pleural malignancy; however, the diagnostic endeavor can be challenging because of overlapping radiological appearances of one condition to another. This case report presents a 61-year-old male with worsening chronic shortness of breath. Despite early imaging resulting in highly suggested mesothelioma, subsequent biopsy proved the malignancy to be pulmonary adenocarcinoma. The patient underwent Pemetrexed-Cisplatin protocol in accordance with the biopsy result, and follow-up imaging depicted a marked improvement of the pleural condition. This case is a prime example of the challenge radiologists have to face regarding pleural tumors and dictates the necessity of a specialized multidisciplinary team to improve the patient's outcome. Pleural diseases compose a wide range of pathology from benign to malignant, primary or secondary, which may appear with various appearances, whether solitary or diffuse, with multiple involvements. The most common primary malignancy in pleura is mesothelioma, while the metastatic process is the most recurrent secondary malignancy Radiology plays a pivotal role in the diagnosis of pleural tumors. The chest X-ray is usually the first modality for patients with thoracic complaints In some exceptional cases, imaging findings of the pleural tumor might be specific. However, in most other cases, the appearance might not be as specific A 61-year-old male presented in the outpatient clinic with chronic shortness of breath that had worsened in the past 2 weeks. The patient denied prolonged cough and nocturnal perspiration. He was a carpenter for over 30 years, quit 3 years ago, and he was a non-smoker with no history of cancer. Vital signs were normal; nevertheless, the physical examination revealed apparent respiratory effort with lagging right chest movement. The complete blood test did not indicate a significant abnormality.Initial chest X-ray  showed rSubsequent contrast-enhanced chest CT scan  demonstrNo evidence of a distant metastatic process was observed from the bone survey and abdominal ultrasound. Pleural nodules , constituting approximately 40% of NSCLC With the absence of discrete mass on visualized lungs, another minor possibility to consider is primary pleural adenocarcinoma. However, in our literature study, we were unable to find any report of primary pleural adenocarcinoma. The closest condition that has been reported is primary pleural squamous cell carcinoma (SCC) ,15, consRecently, there has been a development of targeted lung cancer therapy due to the discovery of EGFR mutation found in lung cancer. Nevertheless, in our case, the EGFR mutation status was confirmed to be wild-type. Many studies have been conducted to predict the EGFR mutation status of lung cancer based on imaging, stipulating that the EGFR mutation is associated with females and non-smokers. Radiological features associated with EGFR mutation are ground-glass attenuation of the tumor, the presence of air bronchogram, vascular convergence, spiculated margin, and pleural retraction Pleural mass remains a significant challenge for radiologists. A wide range of pathologies may arise in pleura, and their radiological appearance may overlap one another. A specialized multidisciplinary team is necessary to establish the diagnosis and plan the best course of action for patients' outcomes.This study has met the ethical principle and has already obtained approval from Research Ethics Committee at Dr. Soetomo General Hospital, Surabaya.Informed consent for patient information to be published in this article was obtained. Appropriate informed consent was obtained for the publication of this case report and accompanying images. This report has been approved by The Ethical Committee of Dr. Soetomo General Academic Hospital.


Glioblastoma multiforme (GBM) is a high-grade glioma that may be a rare complication of radiotherapy. We report a case of a patient who was treated for medulloblastoma (MB) of the posterior fossa at the age of 27 years. Twenty-nine years later, at the age of 56 years, he presented with a double-location tumor: supratentorial and in the posterior fossa. Imaging features of the supratentorial location were very suggestive of a meningioma. We operated on the posterior fossa location, which revealed a glioblastoma. Histologically, the tumor cells exhibited characteristics of both GBM and rhabdoid tumor cells. Literature reports of cases of GBM following MB at the same place are very rare, and presenting rhabdoid characteristics is even rarer. This is the first case of MB and GBM at ages 27 and 56 years, respectively. The double-location supratentorial probable meningioma and GBM of the posterior fossa 32 years after MB is the only case reported in the literature. What to do in this case remains a topic of debate, and there are no clear recommendations in the literature. The occurrence of a secondary tumor, which is a very rare complication, has increased with the improvement of the survival of patients with brain tumors due to the advances in radiotherapy or intensive chemotherapy .Radiation-induced high-grade glioma may arise following several other tumors, such as glioblastoma multiforme (GBM), medulloblastoma (MB), germinomas, Burkitt’s lymphoma, and treated acute lymphocytic leukemia –4. GliobRhabdoid glioblastoma (RGBM) is rare, but is the most malignant among astrocytic tumors. Accumulating evidence indicates its highly aggressive nature and distinct histopathological features .Radiation-induced meningioma is the most common brain neoplasm known to be caused by ionizing radiation , 15. TypHere, we report a case of double-location GBM with rhabdoid characteristics following treated MB 29 years later in the posterior fossa and the supratentorial location evoking a probable meningioma.Our patient is a 56-year-old Moroccan man living in Casablanca, married, father of two children, a taxi driver on sick leave for a year, with low socioeconomic status, and is right-handed.The medical history of the patient was marked by a vitiligo evolving since the age of 13 years, diabetes managed with insulin therapy, a high blood pressure with poor adherence to therapy, and an untreated dyslipidemia. His personal habits were principally alcohol and tobacco addiction. He had no particular family history. He does not present a genetic disease, as well as the members of his family.He was operated in Lyon, France, in 1992 at the age of 27 for a tumor of the posterior fossa. It was revealed by cerebellar syndrome and intracranial hypertension. The diagnosis of MB was retained after anatomopathological examination. The patient and his sister, who is a doctor, reported having received several sessions of radiotherapy and chemotherapy. We did not have details of the doses or the number and duration of treatments, and the files were destroyed at the Radiotherapy Center according to the French law on the management of medical archives. Physical examination revealed tattoo marks on the scalp and back for craniospinal radiotherapy. The patient has evolved well postoperatively, and he has since lived asymptomatic.The patient presented 4 months before the admission with a frontal headache, pulsatile, which increased in frequency and intensity over time, and visual disorders marked by a permanent bilateral visual loss and double vision. He also presented with an imbalance and slurring of speech with a discreet onset and very progressive evolution. The symptoms were worsened by the appearance of hiccups and vomiting outside of meals relieving headaches. He had no seizures or other deficits. During the course of his disease, he exhibited moderate weight loss and apyrexia.Physical examination found a conscious patient, hemodynamically stable, and his performance status was evaluated at 1. The neurological examination found a cerebellar syndrome: cerebellar ataxia with gait ataxia and a predominance of truncal ataxia and dysarthria. A slight strabismus was also noted during the examination. The rest of the neurological examination found no other relevant symptoms. Physical examination of the skin revealed multiple typical lesions of the vitiligo. Examination of the other organ systems found no abnormalities.The initial cerebral CT scan and a cerebral MRI revealed the double location of the tumors. In the posterior fossa, the tumor was in the cerebellar vermis and the right cerebellar hemisphere, grossly limited and irregularly well enhanced after injection. The second is an extra-axial tumor, a dural-based mass, at the right paramedian level in contact with the superior sagittal sinus, which was enhanced vividly on both MRI and CT with some calcifications and scalloping appearance on the cranial vault  outflow pathways. Medullary MRI did not reveal abnormalities. The second supratentorial lesion was probably a radiation-induced parasagittal sinus meningioma, based on suggestive imaging features of a patient undergoing radiotherapy for a long time.We decided to operate on the lesion of the posterior fossa because it showed worse prognosis than did the supratentorial lesion. It was also the symptomatic lesion. The same surgical median suboccipital approach was used in the prone position by incising on the occipito-cervical scar. Intraoperatively, the tumor was grayish, friable, and very hemorrhagic. The follow-up was simple. The patient preserved some gait ataxia.The anatomopathological study showed a diffuse tumor proliferation, poor differentiation, and rich vascularization. Immunohistochemistry showed a focal positivity to anti-GFAP  and negativity of tumor cells to anti-IDH1, concluding in glioblastoma with rhabdoid characteristics  is 1.7%–2.7% at 15 years after radiation therapy. Glioblastoma is relatively more frequent following some treated tumors, such as acute lymphoblastic leukemia/lymphoma: 81% within 10 years after radiotherapy, 59% following other tumors, and 18% with benign conditions .A review of the literature on RIG following MB at the same site in the posterior fossa revealed six cases . The complications of chemotherapy were well supported with medical management. We did not use radiotherapy for our patient. The prognosis was well explained to the patient and his family, and he was aware of the very short survival described in the literature. The therapeutic plan was discussed with the patient, and he showed very good treatment compliance. Our patient died after 11 months. The cause of death is still unknown, and probably is not in relation to the glioblastoma.A review of the literature . It occurs in younger populations: the mean age at presentation has been reported as 29.2–37.9 years in patients exposed to high-dose radiation and as 45–58 years in those who received low-dose treatment, whereas SM generally arises in the fifth or sixth decade of life . It is aSurgical management is based on an aggressive resection of the dura mater, with a large resection margin. This is completed by removal of the bone if there is suspicion of osseous invasion with acrylic replacement .This clinical case illustrated the need for the prolonged monitoring of patients who received treatment with radiotherapy for brain tumors, thus allowing early and adequate treatment of any abnormalities discovered during monitoring.In the literature, the main therapeutic methods used are principally surgical resection followed by chemotherapy only without radiotherapy, or re-irradiation combined with chemotherapy.Temozolomide retains its place in chemotherapy for radiation-induced glioblastoma.The original contributions presented in the study are included in the article/supplementary material. Further inquiries can be directed to the corresponding author.Ethical review and approval were not required for the study on human participants in accordance with the local legislation and institutional requirements. The patients/participants provided written informed consent to participate in this study. Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.TM and AL conceived and designed the study. TM drafted the manuscript. AL critically revised the manuscript for content. All authors contributed to the article and approved the submitted version.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


Case Description. In this report, an 87-year-old female with a history of previous open cystectomy and ileal conduit formation, presented with right upper quadrant pain and worsening parastomal swelling. A computed tomography scan showed a parastomal herniation of the gallbladder, cholelithiasis, and possible early acute cholecystitis. Within 12 hours of admission, bile staining skin changes developed around her urostomy site which raised our suspicion of a perforated gallbladder. Biliary peritonitis was confirmed on laparoscopy the same day. We proceeded with an open midline cholecystectomy without hernia repair. The patient was well at her last follow-up. Discussion. A literature review found 14 published cases of incarcerated gallbladder hernia. This uncommon condition mainly affects elderly females. Ten cases were managed operatively, and four cases were managed nonoperatively, with good outcomes. Currently, no consensus on treatment guidelines exists. In a frail elderly patient, a nonoperative approach may be suitable. Operative management was indicated in this case due to clinical concerns of perforation due to developing skin changes. This cutaneous sign has not been previously documented in the literature.  Parastomal hernia is a common complication following an enterostomy. Gallbladder herniation into parastomal hernia is rare and may become symptomatic and inflamed and very rarely can lead to gallbladder perforation. We present the first case of gallbladder perforation inside a parastomal hernia with a unique skin change.  While rare, the gallbladder can herniate and become incarcerated inside a parastomal hernia. Bile staining of the skin should raise clinical suspicion of perforation. Management options depend on patient and pathology factors and can be nonoperative or operative, with or without hernia repair. A parastomal hernia refers to protrusion of abdominal contents through an abdominal wall defect created during ostomy formation. The reported rates of parastomal hernias in the literature are as high as 65%, with high recurrence rates post repair . Rates aAn 87-year-old female presented with a 1-day history of right upper quadrant abdominal pain, nausea, and worsening distension of her existing parastomal hernia. She was not constipated, obstipated, or in urinary retention. Her past surgical history of note includes an open cystectomy, total hysterectomy, and formation of ileal conduit for bladder cancer seven years prior. This was followed by a repair of parastomal hernia with prolene onlay mesh three years later. Her parastomal hernia recurred, but she was advised for conservative treatment. Other medical history includes a large head of pancreas lesion and chronic obstructive pulmonary disease.On examination, her vital signs were within normal limits. A lower midline laparotomy scar was present. Her abdomen was distended with no skin changes, and the right parastomal hernia was incarcerated, measuring 20 by 20 cm, with focal tenderness.9/L, C-reactive protein 14 mg/L, lipase 1349 U/L, bilirubin 9 umol/L, and deranged liver enzymes. Computed tomography (CT) demonstrated a parastomal hernia containing a portion of nonobstructed transverse colon and the gallbladder with a thickened fundus, pericholecystic fluid, and impacted stone (Her haemoglobin was 111 g/L, white cell count 14.5 × 10ed stone . The knoed stone . Given hOn review that afternoon, her pain worsened and new bile staining surrounding the urostomy site was noted . This raThe patient, therefore, was taken for emergency operative intervention without further imaging. This commenced with a diagnostic laparoscopy via a left lower quadrant cut down. This demonstrated extensive adhesions and bilious fluid in the right upper quadrant, making proceeding with laparoscopic management unsafe. An upper midline laparotomy was performed superior to the previous midline scar. An oedematous, perforated gallbladder with biliary peritonitis and spilled yellow gallstones was encountered. The gallbladder was reduced from the hernia, and full-thickness perforation of the fundus was noted. The transverse colon appeared viable, and it was not reduced. Previous mesh was not appreciated. Cholecystectomy was performed, and the parastomal hernia sac was lavaged. A 19 French Blake drain was placed in the right upper quadrant. The operative time was 80 minutes. Postoperatively, she was transferred to the Intensive Care Unit for ongoing care. Her postoperative course was complicated by myocardial infarction, after which she recovered uneventfully. She was discharged 1 month later after a period of inpatient rehabilitation. The skin changes secondary to bile staining completely resolved . HistopaGallbladder herniation into a parastomal hernia is rare. The majority of cases occur in elderly female patients . PotentiIn all but two published cases , 8, CT iOne case decompressed the gallbladder intraoperatively with a 14-gauge needle for source control of the intraluminal infection and to facilitate with reduction of the hernia . For nonThe decision to repair the hernia defect is challenging given the high recurrence rate. In an emergency situation, direct fascial repair is simpler but has associated high recurrence rates. Stoma relocation has relatively lower recurrences; however, this prolongs surgery time and risks putting both sites at risk of future hernias and adds potential complications to the ileal conduit. The gold standard repair is with mesh due to its lower recurrences and favourable safety profile . In our Here, we present a unique case where a gallbladder-containing parastomal hernia progressed to cholecystitis and perforation. Of note, a rare finding of extensive bile staining of the skin was observed and guided the need for urgent operative intervention. Management options depend on multiple factors and can be nonoperative or operative, with or without repair of the hernia. In the present case, evident perforation of the gallbladder within the hernia prompted surgical exploration.


Leiomyosarcoma (LMS) is a mesenchymal malignancy of smooth muscle, which accounts for 1%–4% of head and neck sarcomas.4Clinically, sarcomas of the oral cavity show nonspecific signs and symptoms and can appear as primary tumors, radiation-associated tumors or metastatic tumors.5The aim of this study was to report a case of maxillary LMS and to perform a review of literature including papers describing LMS of the oral cavity.An electronic search in PubMed  was performed including the previous reported cases of primary oral leiomyosarcoma from the year 2000 to March 2021. The terms “oral leiomyosarcoma” (295 results), “leiomyosarcoma tongue” (28 results), “leiomyosarcoma maxilla” (23 results), “leiomyosarcoma mandible” (31 results) and “leiomyosarcoma buccal” (13 results) were used. Cases were considered as “oral” if they primarily arose in mandible, maxillary bone, gingiva, buccal mucosa, lips, palate, tongue, and floor of the mouth. Only the article in English language were included in the final analysis. We excluded the articles which did not report complete immunohistochemical data related to LMS diagnosis. We included only the cases of primary oral LMS which stated, within the abstract or the manuscript, the immunohistochemical positivity for Smooth Muscle Actin (SMA) or at least for two of these markers: muscle specific actin (HHF35), desmin, h-caldesmon.A 67-year-old man presented at the oral medicine department for oral growing-mass, which caused difficult in feeding and speaking. The patient reported the first occurrence of a small swelling on the premaxilla’s palatal side approximately three months before, but he did not seek medical care due to the COVID-19 pandemic lockdown. He reported sporadic bleeding episodes and progressive increasing of lesion’s dimensions up to our observation. The clinical history was negative for systemic diseases and drug intake.max 15.8), and few areas, putatively inflammatory, localized at the esophagus and mediastinum (SUVmax 2.2)  . Becausemax 2.2) . After smax 2.2) .Figure 1th decades, with similar gender incidence rate .3Leiomyosarcoma is a malignant tumor of smooth muscle derivation which account for 5%–10% of all soft tissue sarcomas.ate M:F, :9.11 CliDue to its rarity, oral LMS can be misdiagnosed with other common benign lesions before biopsy.13Surgical resection with tumour-free margins is widely accepted as the treatment of choice for oral LMS.The prognosis for oral LMS is poor, associated with a high rate of recurrence (42% for high-grade LMS within 2 years from surgery) and regional or distant metastasis.11In the case presented above, the delayed diagnosis is associated with an increased size of the tumor and a subsequent more aggressive surgical intervention; it is not possible to establish how much the delayed diagnosis could have influenced the prognosis for the patient. However, on our experience during the first months of COVID pandemic, patients were often more scared of contracting COVID-infection in the hospital rather than their neoplastic disease.Even if extremely rare, LMS should be considered in the differential diagnosis of oral growing-mass. Early diagnosis and treatment are essential to reduce the risk of recurrence and guarantee better prognosis. Periodic follow-up examinations are mandatory to identify early any signs of recurrence.Institution’s ethics committee approval is not required for the case report.The study was conducted in compliance with the recognized international standards, including the principles of the Declaration of Helsinki.Data and samples were collected under patient’s informed written consent, guaranteeing anonymity.The authors received no specific funding for this work. No funds, grants, or other support were received.The authors declare no conflicts of interest.


Gout is one of the most common inflammatory arthropathies in the developed world. However, involvement of the spine is relatively rare, and other sinister differential diagnoses will need to be considered. We describe an unusual case of gouty tophi deposition within the spine in an elderly patient presenting with signs and symptoms of acute cord compression. Important differential diagnoses that need to be excluded include bony metastases from underlying malignancy and other infective/inflammatory causes. Early recognition of imaging findings can avoid delayed or inappropriate medical treatment. A 70-year-old male with longstanding history of multiple joint pain and soft tissue swelling, and multiple other co-morbidities including diabetes, hypertension and chronic renal failure, presents with sudden onset of upper and lower limb weakness. Physical examination was worrisome for spinal cord compression.A lateral view of the cervical spine showed partial collapse of the C5 and C6 vertebral bodies with complete loss of the C5/6 disc space .Contrast-enhanced computed tomography (CT) of the cervical and thoracic spine was performed with images obtained in pre-contrast, post contrast and bone window to further delineate the pathology. On the axial images in bone window, a bulging soft tissue mass causing narrowing of spinal canal and obliteration of bilateral neural foramina at the C5 level T1-weighted and T2-weighted intermediate signal lesion centered at the intervening disc space  or pseudogout, can present in a similar fashion as gout, but can be differentiated radiologically by the presence of chondrocalcinosis commonly in the hands, wrist or knees. CPPD also seldomly has a soft tissue component as seen in goutA CT-guided bone biopsy of T10 and T11 vertebral bodies was performed and histology showed foreign body granulomatous reaction towards eosinophilic fibrillary material, suggestive of tophaceous gout. Patient was referred to the rheumatologist and neurosurgeon for further management during this in-patient stay, but unfortunately succumbed to hospital acquired pneumonia before further treatment for his gout and cord compression could be administered.Gouty arthropathy is an asymmetrical polyarticular disease, with the foot being the most common in location and in particular the first metatarsophalangeal joint. The next most common regions are the hands and wrists, followed by the elbow and knee joints. Michael et al has reported that most cases (80%) involved only one region of the spine, and around 25% affected the cervical spine only. To our knowledge, only around 10 or so cervical gout cases with images have been reported so far.There can be involvement of any segment of the spine, including the vertebral bodies, pedicles, lamina, ligaments, epidural, and intradural spaces. Lumbar spinal involvement is most commonly affected in the spine. Tophi are predominantly juxta-articular in location. Occasionally, it may arise from within the bone, giving rise to lytic, expansile, well-defined lesions with thin sclerotic margins.Gouty tophi may vary considerably in radiological manifestation, depending on their histological composition. They commonly appear as round or oval ill-defined hyperdense mass lesions in comparison with normal soft tissue. Small lesions can also be radiographically occult if less than 10 mm in size. The principle and value of DECT is its ability to differentiate densities based on their relative absorption of X-rays at different photon energy levels (commonly at 80 and 140 kVp), which enables quantification of gouty deposits and demonstration of non-crystalline tophus components with greater resolution.Dual-energy computed tomography (DECT) is an increasingly common investigation for evaluating tophaceous gout deposition.T1-weighted intermediate signal and homogeneous enhancement post-contrast. Differences in its calcium concentration can give rise to variable and T2-weighted signal. Bone weakening can lead to pathological fractures and locoregional mass effect/compression by the tophi, as demonstrated in this caseGouty tophi on MRI typically shows  Disease presentation in atypical locations may warrant a bone or soft tissue biopsy to exclude other sinister causes.Histological diagnosis is confirmed with microscopic monosodium urate crystals in synovial fluid or within the tophi.Conservative medical treatment can be considered in patients without concomitant neurological deficits, after excluding spinal infection or early reversible causes. Nonsteroidal antiinflammatory agents and steroids have been proven to be effective for acute pain control. Surgery may be considered when there is a need for relief of neurologic symptoms, but long-term management still relies on adequate medical therapy.This case illustrates a common disease entity presenting in an atypical location and presentation with complications.Involvement of the vertebral column is uncommon in gouty arthropathy, and causing acute cord compression is even rarer. Important common differential diagnoses that needs to be excluded are bony metastases from underlying malignancy, and other infective/inflammatory changes which warrant more urgent management.


We report a case of osteolytic polyarthritis and atlanto‐axial lesion revealing multiple myeloma‐associated amyloid arthropathy in a 56‐year‐old man. Only histological exam of the salivary gland biopsy demonstrating the presence of amyloid deposits stained with congo red could confirm the diagnosis. We report an unusual case of multiple myeloma‐associated amyloid arthropathy revealed by a osteolytic polyarthritis and atlanto‐axial lesion. Amyloid deposits can occur in a variety of organs, with involvement of the heart, kidney, gastrointestinal tract, and nervous system. Musculoskeletal manifestations of amyloidosis include myopathy, arthropathy, and osteopathy.This rare location is of interest because of its functional and vital impact. We report here a case of osteolytic polyarthritis and atlantoaxial lesion revealing AL amyloidosis associated with multiple myeloma.Written patient’s consent was obtained for publication.2A 56‐year‐old man with non‐insulin‐dependent diabetes (for 7 years) and kidney failure presented with worsening swelling and pain in both wrists, small joints of the hands, elbows, shoulders, and knees since 4 months. He had been on regular hemodialysis for 5 months. He also noted fatigue, weight loss, and loss of appetite. He complained of painful numbness and weakness in the legs. He had been unable to walk independently for about 1 month.On admission, he was astenic. Physical examination showed an active limitation in shoulders, symmetrical swelling with tenderness in knees, and multiple joints of extremities, particularly in wrists, metacarpo‐phalangeal (MCP), and proximal interphalangeal (PIP) joints. There were no abnormal findings in his chest or abdomen suggestive of involvement of visceral organs. Neurologic examination revealed normal mental status and cranial nerves. He had weakness in distal limbs. He was areflexic. Sensation was absent to all modalities in the lower extremities. Passive movement of the neck was painful.3, MCH: 68%), hypercalcemia , elevated ESR (74 mm), CRP (25 mg/L), and hypogammaglobulinemia (1.8 g/L). Serum immunofixation electrophoresis identified an Ig G Kappa‐type monoclonal gammapathy. Examination of a bone marrow aspirate found 60% of dystrophic plasma cells.Laboratory analysis showed anemia microcytic and hypochromic  is a revealing sign of this monoclonal gammapathy.It is most commonly described as a seronegative nonerosive polyarthritis, mimicking RA.Ultrasonography could be a helpful tool in the diagnosis. The findings include thickening of supraspinatus and biceps tendons, synovial thickening, the presence of hypoechoic masses around tendons, bursas synovial enlargement, and effusion within the bursa. The power Doppler signal is always negative.Radiologically, counter to RA, MAA is rarely erosive. The classical X‐ray is usually normal. The joint space is preserved or even widened. Some bone cysts may be encountered mainly at the carpus, inferior extremity of the radius, superior extremity of the humerus, or the femoral neck.The atlantoaxial articulation may also be affected. Our patient complained about neck pain and stiffness. The cervical scan showed an osteolysis of the odontoid process. Only one case of amyloid C1–C2 synovitis with subluxation of atlantoaxial joint was reported by Fautrel et al.Once the cervical spine affected, symptoms may vary from common cervicalgia to spinal cord compression. Usually, magnetic resonance imaging reveals a low signal on T1 and T2‐weighted images with a peripheral enhancement after gadolinium injection. In case of neurological complication, surgical excision completed by stabilization may be required. Outcome seems to be excellent.An osteolysis in synovial fold zone of the two sternoclavicular joints with periarticular soft tissues mass syndrome was identified in our patient. Goffin et al found amyloid deposits in the sternoclavicular fibrocartilage.4Myeloma‐associated amyloid arthropathy should be one of the differential diagnoses of RA. Both MAA and RA can be responsible of symmetrical erosive polyarthritis, cervical spine, and sternoclavicular lesions. In this case, only the histological exam of the biopsied tissues demonstrating the presence of amyloid deposits stained with congo red can confirm the diagnosis.Ines Mahmoud: Writing. Leila Rouached: Writing. Meriem Chammakhi: collecting information. Aicha Ben Tekaya: writing. Selma Bouden: analyses. Aycha Ben Miled: interpretation of figures. Rawdha Tekaya: collecting information. Olfa Saidane: correction. Leila Abdelmoula: supervision.The authors have no conflict of interest to declare.Our case report complies with the Declaration of Helsinki.Informed consent has been obtained from the patient.


Supernumerary testis (SNT), or polyorchidism, is a rare congenital anomaly of the genitourinary tract, described as the presence of more than two testicles. Testicular torsion (TT) in unilateral SNT is extremely rare. We report a case of unilateral SNT in a 16-year-old boy accompanied by TT, which was diagnosed preoperatively based on the outcomes of physical examination and ultrasound and confirmed intraoperatively. We opted for orchiectomy of this SNT because of the long-time hypoperfusion. And as for the normal testis, orchidopexy was performed. His clinical presentation subsided after surgery, and the patient was discharged 3 days later. Supernumerary testis or polyorchidism is a rare congenital anomaly of the genitourinary tract, described as the presence of more than two testicles. The majority of the patients do not have any clinical symptoms or are detected by imaging or during surgical explorations accidentally . TesticuA 16-year-old boy with a history of polyorchidism presented to our hospital with a 2-day history of left testicular and inguinal pain. He denied a history of scrotal trauma, and the patient did not feel any discomfort previously with the history of polyorchidism. On physical examination, the left side of the scrotum was markedly red and swollen, and two masses were identified in the left hemi-scrotum. The higher mass was painful, hard on palpation, and elevated compared to the right testicle. The lower mass was palpable in the left hemi-scrotum. The left cremasteric reflex was significantly diminished. After admission, his blood work was completed: leukocytes , hemoglobin (16 g/dl), platelets , LDH (173 co/L), HCG (1.2 mIU/ml), and AFP (2.15 ng/ml).An ultrasonography scan of the scrotum revealed two hypoechoic masses, both with testicular-like textures. The higher mass, measuring 3.1 × 2.0 cm, did not have visible vascular flow inside on color Doppler examination ; the lowDuring operation, we found that two testicles were lying within the single tunica vaginalis. The upper left testicle was black and twisted 360° clockwise. The lower left testicle was normal in its color. On gross examination, the two testicles share the same vas and two separate epididymites . DetorsiPolyorchidism is a rare congenital anomaly of the genitourinary tract. Around 200 cases have been reported up to the present after the first cases reported by Blasius in 1670, and triorchidism is the most common type .The precise etiology of polyorchidism is unknown. A literature review showed that the testes occur early in the sixth week of ovulation age and originate from the primitive genital ridge. However, the vas deferens and epididymis develop from the mesonephric duct 2 weeks later. The most popular theory is a transverse division of the genital ridge that can clarify all of the forms of supernumerary testis –3. BasedType 2 is the most common of the four types  cases have been reported in the literature until the present % cases h. CurrentIn our case, intravaginal torsion occurred in the supernumerary testis. Beiko et al.  believe In our case, the supernumerary testis has separate epididymis and shares the same vas with the normal testis in the hemi-scrotum. The fertility potential was considered, but we opted for orchiectomy for this supernumerary testis because of the nonvital testicle. Feher et al.  found thSupernumerary testis is a rare congenital anomaly of the genitourinary tract, but it is a high-risk factor of testicular torsion. Active surveillance is necessary; if testicular torsion occurs, early and accurate diagnosis can reduce the chance of orchiectomy for nonvital testicles.The original contributions presented in the study are included in the article/Written informed consent was obtained from the individual(s) and minor(s)' legal guardian/next of kin, for the publication of any potentially identifiable images or data included in this article.LX: writing—original draft preparation. ZB and LX: writing—review and editing. All authors have read and agreed to the published version of the manuscript.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


The Buschke-Löwenstein tumor (BLT), also known as giant condylomata acuminata (GCA), is a pseudo-epithelial proliferation engendered by the human papillomavirus (HPV). Interestingly, its location at the anal margin, or perianal skin, is rare. The authors present the case of a gentleman who became unstable while standing, stating that his ears were ringing. His emergency presentation, clinical course, and imaging findings are discussed. The patient presented with signs of condyloma acuminata and BLT. This can be excised through surgery and removed with the help of adjuvant treatments, but there is still much to learn about this disease. First described by Buschke and Löwenstein in 1925, the Buschke-Löwenstein tumor (BLT) is a sexually transmitted infection that is primarily caused by the human papillomavirus (HPV). This infectious disease is relatively rare but is often identifiable by a precedent case of condylomata acuminata. Because condylomata acuminata always predates the BLT, this infectious disease is preventable with early diagnosis, hygiene, and wider control of sexually transmitted infections. The BLT can affect both sexes; however, it is most commonly found in males [This is the case of a 42-year-old male with no significant past medical history besides dyslipidemia and anal warts, reporting three months history of feeling off-balance. He also complained of ringing in both ears and reported worsening symptoms with standing. He denied syncope, chest pain, shortness of breath, headache, focal weakness, incoordination, double vision, melena, hematochezia, hematemesis, fever, chills, or neck pain. The patient also reported genital anal warts since age 19, and that area around warts had been oozing blood for some time. He was referred from his primary care physician for outpatient head computerized tomography (CT) scan, lab work, and cardiology consultation but symptoms worsened, and he came for evaluation. The patient’s vital signs were within normal limits. Electrocardiogram revealed normal sinus rhythm. Physical examination was significant for mildly decreased breath sounds. Rectal examination showed a patent anus. CT of the head showed no abnormal acute findings. Laboratory evaluation revealed hemoglobin 6.1, white blood cell count 8.26, mean corpuscular volume 68.2, red cell distribution width (RDW) 15.6. The metabolic profile was without significant findings and was troponin negative. Human immunodeficiency virus test negative. The urinalysis was unremarkable. The patient was transfused with two units of packed red blood cells and admitted with gastroenterology consultation. The patient had surgery and biopsy with results consistent with condyloma acuminata with features of BLT and areas suspicious for invasive squamous cell carcinoma (Figure BLT is a rare condition, and its pathogenesis and natural history are not well understood. Tumors have a high proliferation rate and increased risk of transformation to malignancy. Our patient also had a p16 immunohistochemical stain which was reported as negative. The diagnosis remained unchanged.The BLT clinically appears as a larger mass with budding similar to the form of a cauliflower. The excessive size poses danger to nearby organs and structures, as it can extend beyond tens of centimeters . In our Even though the incidence of the BLT has somewhat increased for the past 10 years, physicians still have a difficult time diagnosing the disease due to its similarities with ordinary condylomas or well-differentiated squamous cell carcinomas . LesionsDue to its rare nature, physicians do not have a general consensus concerning the treatment of the BLT. Even though there has been potential for several therapies, surgery is designated as the gold standard of treatment ,5. UnforSeveral problems may appear after treatment due to reappearance or reinfection at the site of surgery. The high recurrence rate can prove to be an issue in regard to surgical treatment. Patients with cases presenting involvement of the bones or pelvis may require abdominoperineal resection. Additionally, complications can occur secondary to contamination of the wound in the perianal area by feces .Our patient presented with the commonly reported symptoms associated with condyloma acuminata and BLT. There is still much to learn about BLT in regard to treatment because of its rarity. Surgical excision with plastic reconstruction of skin defects appears to be one of the more promising options. Adjuvant treatments such as chemo- and radiotherapy may also have a role.


Nodulocystic basal cell carcinoma (BCC) is a cystic variant of BCC, which can easily be misdiagnosed. We report a case of a 52-year-old man with a nodulocystic BCC that appeared at the site of a previously excised BCC 9 years prior. It examined as a benign cyst with a radiological signature of a vascular malformation. It was histologically confirmed on fine needle aspirate (FNA) and excisional biopsy to be a nodulocystic BCC. BCC is one of the most common paraneoplastic neoplasms affecting photo-exposed areas and displaying many variants. Nodulocystic BCC is a rarer variant that may have more than one dermoscopic face and can appear macroscopically benign. Given its malignant potential, it is imperative that it is accurately diagnosed. We highlighted that nodular cystic BCC can easily be misdiagnosed. Careful history and FNA are key differentiators to establish the correct diagnosis. Recurrence of basal cell carcinoma (BCC) after initial treatment is not uncommon. Having its origin from follicular germinative cells, its histologic appearance may vary considerably depending on the direction and degree of differentiation of the cells . We descA 52-year-old man was referred by his general practitioner with a mass arising from his dorsal distal left forearm overlying the radius. On examination, he had a 4 × 3 cm, mobile, cystic mass . PertineUltrasound of the forearm revealed a 2.8 × 2.3 × 6 mm hypoechoic fusiform mass within the subcutaneous adipose tissue . It was A subsequent MRI, showed a hyper intense 23 × 5 × 15 mm subcutaneous mass, with prominent adjacent feeding vessels, overlying the muscle bellies of abductor pollicis longus and extensor pollicis brevis . RadioloGiven the diagnostic dilemma, a fine needle aspirate biopsy was performed and showed the specimen to contain irregular groups of hyperchromatoid basaloid cells with peripheral palisading surrounding eosinophilic stroma in a background of myxoid matrix. Morphologically, the features were consistent with a recurrence of BCC.The patient then proceeded to have a full thickness excision of the cystic lesion with margins down to the extensor muscles .Sectioning showed a 30 × 30 × 8 mm cream multiloculated mucoid nodule. On microscopy, the sections show Nodulocystic and focally infiltrative BCC that invades the subcutis with clear excision margin. No peripheral or lymphovascular invasion was seen .The patient made a full recovery and was discharged from our clinic with recommendation of regular full skin check with his general practitioner.Clinical and morphological subtypes of BCC vary with the classic type being nodular. Tumoral necrosis in nodular BCC may lead to cavity formation and result in its cystic form .Nodulocystic variant is not innocuous and has been described to have metastatic potential . HencefoThe initial evaluation of BCC includes clinical examination, biopsy and imaging for staging. Imaging characteristic in this case was a red herring. Literature review in ultrasound and MRI characteristics of nodulocystic BCC yielded no directly relevant article. We presumed that this is because of the retrospective nature of cases with nodulocystic BCC.The key differentiator in this case was the classical cytomorphological feature of BCC on fine needle aspiration cytology (FNAC). Cytological diagnosis with FNAC is reliable with a meta-analysis showing a pooled sensitivity of 97%  94–99) and pooled specificity of 86% (95% CI 80–91) [BCC has many histologic variants with a rare variant of nodular BCC being its cystic form. The importance of making a correct diagnosis lies in the necessity to differentiate this tumour from a benign cyst given its malignant potential. The knowledge of cytological features of the cyst is helpful to prevent any misdiagnosis.None declared.This research did not receive any specific grant from funding agencies in the public, commercial or not-for-profit sectors.


Infantile hepatic hemangioendothelioma/hemangioma is the most common benign hepatic vascular tumor of infancy, comprising approximately 1% of all childhood tumors. The tumor can present during the fetal or neonatal period as a hepatic mass. Common presentations include abdominal distension and a palpable hepatic mass. Clinico-radio-pathological correlation is essential for a definite diagnosis. Frequent complications such as congestive heart failure, thrombocytopenia, anemia, and Kasabach-Merritt syndrome should be investigated. Chemotherapy has been reported as an effective treatment option. Surgical resection has an essential role for symptomatic patients with medical treatment failure or other certain conditions such as refusal to take medication. Furthermore, prenatal diagnosis is essential for better patient outcomes due to prompt treatment in the neonatal period. We report a case of a female infant at 39 weeks of gestation who was delivered from a 32-year-old mother. The infant was in utero diagnosed by ultrasonography with a hepatic mass, most likely hemangioma. The mass was resected after birth and it was diagnosed as infantile hepatic hemangioendothelioma type II. The course of the disease was excellent and the patient was cured after treatment. Infantile hepatic hemangioendothelioma/hemangioma (IHH) is the most common hepatic tumor of mesenchymal origin . The firA second child, a female neonate at 39 weeks gestation, with a bodyweight of 3,335 g, was born to a 32-year-old mother with vaginal birth. The pregnancy progressed successfully with no obstetric complications. However, patient history indicated that the mother had 10 years of underlying chronic hepatitis B virus (HBV) infection. She was previously treated with antiviral medication, but she decided to abandon the treatment three years before her first pregnancy. Further antenatal care laboratory tests showed positive HBsAg, positive HBeAg, and a high HBV viral load of 43,525,558 IU/mL. The mother also received tenofovir to prevent perinatal HBV transmission at 27 weeks gestation. Prenatal tests, according to the Thai national antenatal care program, are negative for syphilis and HIV. Prenatal ultrasonography for anomaly screening in the second trimester period was uneventful. However, at the gestational age of 36 weeks, a heterogeneous mass (2.7 x 2.3 x 2.5 cm) in the left lobe of the fetal liver was incidentally discovered by ultrasonography during maternal antenatal care. The lesion revealed a well-circumscribed peripheral vascularization accompanied by a low internal flow, giving the impression of a liver mass, most likely hemangioma.Physical examinationThe neonate weighed 3,335 g with normal activity, and mild pallor without jaundice was observed. The heart sounds were normal without murmurs, and the chest movement was symmetrical. A physical examination of the abdomen indicated no distention with active bowel sounds and non-palpated liver and spleen. No other deformities such as orofacial clefts were identified. In company with a physical examination, no neural tube defects were detected. Lastly, the skin had no cutaneous hemangioma, petechiae, or purpura.Imaging study and laboratory findingsWhen the patient was seven days old, abdominal ultrasonography was performed to evaluate the lesion in the left lobe liver. It showed a normal-sized liver with a well-circumscribed oval-shaped heterogeneously hypoechoic mass measuring 3.75 x 2.82 cm in the left lobe liver. Cystic spaces and tiny calcifications were found in the mass. An increased peripheral vascular flow was discovered. The celiac trunk, hepatic artery, hepatic veins, and portal veins were normal in size. No bile duct dilations were uncovered. All findings indicated that infantile hepatic hemangioendothelioma was possible Figure .A multidetector computed tomography (CT) of the upper abdomen was performed using plain CT and dual-phase contrast-enhancement CT when the patient was 16 days old. CT was performed to fully characterize the mass before treatment decision. A mass was present in the lateral segment of the left lobe of the liver, measuring 3.7 x 4 x 3.2 cm. Stippled calcifications were also noted in the mass. It had avid nodular peripheral arterial enhancement with incomplete centripetal fill-in in the portovenous phase. The celiac trunk and hepatic artery were normal in size. There was no change in the aortic caliber above and below the celiac origin. The attenuation of the enhancing portion equaled that of the aorta in both arterial and venous phases. These findings were compatible with infantile hepatic hemangioendothelioma  is a neonate’s common hepatic mesenchymal tumor. Generally, at the point of diagnosis, 90% of patients are less than six months old . The tumPatients with IHH sometimes show variation in clinical presentation depending on the tumor size and location. Usually, abdominal distension or palpable hepatic mass is present. Further, cardiac complications such as congestive heart failure and hematologic complications such as thrombocytopenia, anemia, and Kasabach-Merritt syndrome are observed ,7. In adImaging studies of IHH show a space-occupying lesion in the liver. By computed tomographic scan, infantile hemangioendothelioma presents a well-defined border mass alongside decreased attenuation compared to the adjacent non-neoplastic liver tissue . The areHistologically, IHH is classified into two types. Type I IHH shows the proliferation of small, capillary-like vascular spaces lined by bland or plump endothelial cells. The vascular channels are separated by connective tissue interspersed with small bile ducts. The mitotic figures are either rare or absent. Malignant spindle cell components are not present. Type II IHH shows disorganized branching vascular channels lined by endothelial cells, which reveal pleomorphism, nuclear hyperchromatism, and frequent mitoses . The endThe differential diagnosis of infantile hepatic hemangioendothelioma is a cavernous hemangioma, angiosarcoma, hepatoblastoma, and mesenchymal hamartoma . The cavIHH is classified as a benign vascular tumor with spontaneous regression in some patients ,20. HowePerinatal diagnosis plays a vital role in postpartum care . In thisInfantile hepatic hemangioendothelioma is one of the causes of liver mass in neonates. Physical examination, radiographic findings, and pathological findings are essential for the definite diagnosis and differentiation from other hepatic masses. Kasabach-Merritt syndrome and congestive heart failure are probable complications and should be investigated. It is important to note that surgery plays an important role in the case presenting with complications, failure of supportive treatment, or refusal of medical therapy. Prenatal diagnosis is also essential in early diagnosis, with prompt treatment after birth leading to an excellent outcome.


Lymphangiomatosis is an extremely rare disease with potential soft tissue, bone, and spleen involvement, which can be characterized by lymphangioma. Only a few cases of colon and mesenteric lymphangiomatosis have been reported. We report a case presenting with fatigue, periumbilical pain, and intermittent bloody stools. This patient underwent a series of examinations. Exploratory laparoscopy, in particular, yielded very valuable images and videos for this disease, which can provide evidence for the diagnosis of this disease.The current patient had fatigue, periumbilical pain, and intermittent bloody stools. Colonoscopy indicated numerous variable-sized hyaline cysts in the colon. Submucosal puncture was performed during colonoscopy. The patient was readmitted to the hospital due to periumbilical pain. B-ultrasound and abdominal CT showed multiple hypoechoic nodules in the mesenteric area. Exploratory laparoscopy was performed, and histopathology revealed that D2-40 was positive. Based on auxiliary examination and laparoscopic biopsy, surgeons and pathologists reached the diagnosis of mesenteric lymphangiomatosis.Clinicians need to comprehensively improve their knowledge of lymphangiomatosis, and the combination of clinical symptoms, histological characteristics, and colonoscopy biopsy findings should be considered to improve lymphangiomatosis diagnosis, thereby reducing misdiagnosis.Colon and mesenteric lymphangiomatosis is an extremely uncommon benign condition of unknown etiology and pathogenesis in adult patients. We report a case of mesenteric lymphangiomatosis in a 37-year-old woman who presented with fatigue, periumbilical pain, and intermittent bloody stools, as well as lesions in the kidney, spleen, and bones. This case provides new insights into the diagnosis and treatment of this disease. Colon and mesenteric lymphangiomatosis is an extremely uncommon benign condition of unknown etiology and pathogenesis, which could lead to compression symptoms caused by mechanical pressure. Here, we report a case of abdominal lymphangiomatosis evaluated by laparoscopy, which has not been previously reported.2) were found in the retroperitoneum  for 3 months, and follow-up colonoscopy was performed. Colonoscopy indicated decreased number and size of cysts compared with the condition observed 6 months before  was proposed by the International Society for the Study of Vascular Anomalies (ISSVA) as a new term for lymphangiomatosis  can be found in the article/XW and YY contributed to the study concept and design. YY generated the literature strategy and filtered through the identified studies. YY evaluated study quality and wrote the manuscript. RW provided critical feedback on the manuscript. All authors contributed to the article and approved the submitted version.National Natural Science Foundation of China, Grant Number: 82072732, 81572930; The National Key Research and Development Program of China, Grant Number: 2016YFC0905303; Beijing Science and Technology Plan, Grant Number: D171100002617004.We thank Dr. Hulin Ma for data acquisition and clinical assessment and thank pathologist Quan Zhou for her contribution in the diagnosis of mesenteric lymphangiomatosis.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


Acute tumour embolism to the popliteal artery resulting in limb-threatening ischemia is a rare complication of neoplastic disease. Generally, tumors embolize to the pulmonary circulation via the venous system. In this case, the originating tumor was a lung cancer of a large size and advanced stage that had invaded the left atrium of the heart and disseminated in the systemic circulation. The tumor likely fragmented, resulting in showering to the right popliteal artery, superior mesenteric artery, and left renal artery, which is a unique presentation of tumor embolism.We present a case of a 62-year-old Caucasian gentleman with a large left lower lobe squamous cell lung cancer that had invaded into the left atrium via the pulmonary veins. He presented with acute limb threatening ischemia. A computed tomographic angiogram revealed an occlusion of the left popliteal artery as well as embolization to the superior mesenteric artery and the right renal artery. He was started on intravenous heparin and underwent an emergency popliteal embolectomy and calf fasciotomies, which was limb saving. His fasciotomy wounds were closed after 1 week and he was discharged on anticoagulation.This is a rare case of tumor embolism resulting in both an embolectomy and calf fasciotomies. In the light of such cases, we suggest that tumors invading the bloodstream should be considered high risk for embolization and hypothesize that prophylactic antithrombotic therapy may avoid major morbidity. Acute limb ischemia secondary to tumor embolism is a rare complication of primary neoplasm. An analysis of 877 cases of arterial embolization showed that only three cases (0.34%) were caused by tumor embolism . The majThe showering of tumor emboli has been known to cause acute occlusions at various sites including the mesenteric, brachial, common femoral, and popliteal arteries . These aThese patients invariably have a poor prognosis as tumor embolization indicates advanced cancer, which is the primary cause of mortality, even if the acute treatment of embolization is successful .We present a case of a large primary lung cancer, which had invaded into the left atrium. This resulted in tumor embolization at multiple arterial sites and acute limb-threatening occlusion of the left popliteal artery requiring embolectomy and fasciotomy. The popliteal artery generally has a diameter of 7–8 mm in men. The completeness and severity of popliteal arterial occlusion indicates that large amounts of tumor fragments were released into the bloodstream with concomitant secondary thrombosis. This is a unique presentation of tumor embolism, in which surgical intervention and fasciotomy was required to prevent limb loss.A 62-year-old Caucasian gentleman presented to respiratory clinic with persistent cough, hemoptysis, and recurrent chest infection. He was unwell for 6 months with increased breathlessness, mild hoarseness of voice, fatigue, reduced appetite, and weight loss. A chest X-ray showed left-sided opacification and a small effusion.Computed tomography (CT) of the thorax showed a large 7 × 9 cm complex centrally necrotic lesion of the left lower lobe with distal collapse and consolidation Fig. . This waHe had a background of osteoarthritis, hyperlipidemia, and benign prostatic hyperplasia. He was an ex-smoker with 25–30 cigarettes/day until recently.After 6 months, he presented to vascular surgery with sudden onset left leg pain. On examination, the left leg was cold, mottled, pale, and lacking sensation but he was able to move his toes. He had a palpable femoral pulse but an absent left popliteal pulse. He was diagnosed with acute limb ischemia and was started on intravenous heparin.The CT angiogram showed an occlusive thrombus in the mid superior mesenteric artery (SMA). The inferior mesenteric artery was patent and iliofemoral arteries were patent with no opacification of the arteries from mid-thigh onward. He was recalled for delayed phase imaging, which showed a large popliteal artery occlusion at the bifurcation with no run-off distally Fig. .Fig. 2LeIn addition, the large necrotic tumor was noted and was causing complete collapse of the left lower lobe. The tumor extended along the inferior pulmonary vein into the left atrium and appearance had worsened since previous imaging. The lower pole of the right kidney was infarcted Fig. . A diagnHe underwent a left popliteal embolectomy along with calf fasciotomies. This involved open surgical exposure of the popliteal artery using a medial approach below the knee. The popliteal artery was carefully dissected free. Proximal and distal vessel control was obtained and a transverse arteriotomy was made. A Fogarty balloon catheter was used to perform the embolectomy and adequate blood flow was seen. Polypropylene suture was used to close the arteriotomy in an interrupted manner. Four-compartment fasciotomies were then performed.Postoperative arterial duplex did not identify flow in the tibio–peroneal trunk, the posterior tibial artery, and the distal peroneal artery. There was monophasic flow in the anterior tibial artery. However, the foot was warm and well perfused. Postoperatively, he was anticoagulated with subcutaneous therapeutic dalteparin injections, 12,500 units/day. The fasciotomy wounds were closed a week later, and the patient was discharged.The embolectomy specimen sent to the lab contained three pieces of arterial tissue and multiple fragments of gray tissue, which measured 15 × 25 × 8 mm in aggregate. Microscopic examination showed that all pieces of tissue had histological features of a recent thrombus. The pieces of gray tissue macroscopically resembled lung tissue. Immunohistochemical staining was not done to formally identify tumor cells in the specimen.He subsequently underwent a bronchoscopy and biopsy, which confirmed squamous cell lung cancer, characterized by sheets of large, polygonal, malignant cells containing keratin. This was treated palliatively with chemotherapy and radiotherapy.et al. described a case of pulmonary tumor embolism due to a gastric carcinoma and Murugan et al. reported a case of invasive ductal breast cancer with embolization to the pulmonary artery [et al. described a case of pulmonary tumor embolism due to a myxoid sarcoma originating from the thyroid gland [et al. who reported a case of urothelial carcinoma with neoplastic embolization to the pulmonary artery resulting in pulmonary hypertension due to pulmonary thrombotic microangiopathy [This is a unique case of a lung cancer embolizing to the systemic circulation via the arterial system. Generally, the literature describes tumor embolization to the pulmonary circulation via the venous system, often with an underlying gastrointestinal, breast, soft-tissue, or urological malignancy. For example, Aiyappan y artery , 6. Simiid gland . A furthgiopathy .et al. reported a case of non-small cell lung cancer with cardiac metastases that embolized to the infra-renal aorta and bilateral common iliac arteries resulting in bilateral limb ischemia [et al. in 2018 reported a case of squamous cell lung cancer with left atrial invasion that embolized to the right superficial femoral artery and the superior mesenteric artery, leading to acute limb ischemia and acute mesenteric ischemia, respectively [Lung cancer embolization into the systemic circulation has also been reported in the literature. In 2018, Cheema ischemia . Surgicaectively . This paIn contrast to the above cases, our patient had a successful outcome and survived to discharge with his limb preserved. This was due to early recognition and immediate surgical intervention. Popliteal embolectomy restored the blood supply in a timely manner and fasciotomy prevented reperfusion injury and compartment syndrome. We were fortunate that the limb ischemia was unilateral, rather than bilateral, and the occlusion site was more distal that is popliteal artery rather than femoral or common iliac artery. There was also no concomitant bowel ischemia in our case.et al. in 2012, when they reported a lung adenocarcinoma that had invaded into the left atrium and embolized to the left external artery and right popliteal artery causing bilateral limb ischemia, which was also associated with left frontal lobe cortical infarcts, bilateral renal infarcts, and splenic infarcts [Cases of lung cancer tumor embolism that cause limb ischemia severe enough to warrant fasciotomy are extremely rare. The only other such case was published by Schreffler infarcts . This paAs seen from the above cases, lung cancer invasion into the left atrium via the pulmonary vein is a well-described mechanism of tumor embolism . EmbolizThis explains why lung cancer and atrial myxoma are the most common tumors to embolize, given their proximity to the heart . The tumIn this case, the diagnosis of tumor embolus was primarily clinical and radiological as the histology did not comment on tumor burden. Given the underlying lung malignancy and presence of thrombosis at three separate arterial sites  on CT scan, the embolus is likely to represent tumor showering. The macroscopic appearance of the embolectomy specimen was also more consistent with lung tissue, as opposed to a fibrin blood clot.Acute tumor embolism is a physical process and should be differentiated from the hypercoagulability associated with malignancy. Cancer induces a thrombotic state by activating the coagulation cascade. It causes increased secretion of procoagulant molecules and inflammatory cytokines . Other mFactors predisposing to tumor embolism include the size of the tumor, rate of growth of tumor, number of tumor cells, malignant potential, and poor differentiation of tumor cells  In our cAlthough we managed to salvage our patient’s limb, he underwent major surgery with a high morbidity. He was in the later stages of limb ischemia and required a fasciotomy to prevent compartment syndrome. We were also fortunate that embolization to the SMA did not cause acute mesenteric ischemia, which has a high mortality. Therefore, it may be more beneficial to focus efforts on prevention of acute tumor embolism through risk stratification and prophylactic antithrombotic therapy in high-risk cases.Current American Society of Hematology guidelines recommend anticoagulation treatment to treat venous thromboembolism in cancer patients . It is aGenerally, acute tumor embolus has a poor prognosis due to the advanced stage of malignancy. Often, it is regarded as a preterminal event and palliative treatment may be considered more appropriate. Surgical intervention does not change the overall prognosis for the patient, which is determined by the stage and type of cancer.This patient required emergency surgical embolectomy to save his limb. Catheter-directed thrombolysis was not appropriate in this case because the mechanism of action of thrombolytic agents involves binding to fibrin to activate tissue plasminogen to form plasmin, which is a fibrinolytic enzyme that dissolves the fibrin blood clot. As the embolus in this case was due to tumor, rather than a fibrin clot, thrombolysis would have been ineffective.This case shows that surgical embolectomy can be immediately limb saving and prevent major morbidity for patients with acute tumor embolism causing limb ischemia. It can prevent complications such as compartment syndrome, rhabdomyolysis, and acute kidney injury. Therefore, it should be considered in carefully selected patients as it can greatly improve the patient’s quality of life in the terminal stages of disease.Tumor embolism resulting in popliteal occlusion is a rare occurrence and can cause loss of limb and death. Lung cancer invading into the pulmonary veins is associated with embolization to the systemic circulation. The clinician needs to be cognizant to the possibility of tumor embolism in such cases, as limb salvage depends on early diagnosis and surgical intervention.Further research is needed to explore the role of prophylactic antithrombotic therapy in preventing tumor embolization. It would also be useful to compare prognostic outcomes with and without surgical intervention in patients with acute tumor embolism.


Eccrine spiradenocarcinoma (SC), also known as malignant eccrine spiradenoma, is a rare malignant cutaneous adnexal neoplasm arising from long-standing benign eccrine spiradenoma. Malignant skin tumors rarely show direct intracranial invasion. However, once the intracranial structure is infiltrated, curative excision with sufficient margins can become extremely difficult, particularly when the venous sinuses are involved. No effective adjuvant therapies have yet been established. Here, we report an extremely rare case of scalp eccrine SC with direct intracranial invasion, which does not appear to have been reported previously.An 81-year-old woman presented with a large swelling on the parietal scalp 12 years after resection of spiradenoma from the same site. The tumor showed intracranial invasion with involvement of the superior sagittal sinus and repeated recurrences after four surgeries with preservation of the sinus. The histopathological diagnosis was eccrine SC. Adjuvant high-precision external beam radiotherapy (EBRT) proved effective after the third surgery, achieving remission of the residual tumor. The patient died 7 years after the first surgery for SC.Scalp SC with direct intracranial invasion is extremely rare. Radical resection with tumor-free margins is the mainstay of treatment, but the involvement of venous sinuses makes this unfeasible. High-precision EBRT in combination with maximal resection preserving the venous sinuses could be a treatment option for local tumor control. Eccrine spiradenocarcinoma (SC), also known as malignant eccrine spiradenoma, is a rare malignant cutaneous adnexal neoplasm arising from long-standing benign spiradenoma . A histoMalignant skin tumors rarely show direct intracranial invasion. However, once the intracranial structure is infiltrated, wide local excision with sufficient margins can become unavailable, particularly when the venous sinuses are involved. Radical treatment can thus become difficult without effective adjuvant therapy. Here, we report an extremely rare case of scalp eccrine SC that showed direct intracranial invasion, repeated recurrences after surgery, and the effectiveness of adjuvant high-precision external beam radiotherapy (EBRT), which does not appear to have been reported previously.An 81-year-old woman, who had a history of resection of a benign eccrine spiradenoma from the parietal scalp 12 years earlier, presented with a large swelling on the same region that had been growing over the preceding 5 years without any neurological deficit. Radiological examinations demonstrated that the lesion was a single, large cystic mass invading bilateral parietal bones and attached to but not occluding the superior sagittal sinus (SSS)  with 42.2 Gy in 10 fractions to the margin of the residual cyst wall and the tumor involving the SSS  to 20 months (after the third surgery). Notably, recurrence after the EBRT was limited to around the SSS, where unresectable tumor had been left, even though the tumor was seen to show broad attachment to the dura at the third operation. Immediate adjuvant EBRT after maximal resections like the first or second surgeries might have yielded longer, better tumor control , which wWe have reported a rare case of scalp SC with direct intracranial invasion. Radical resection with tumor-free margins represents the mainstay of treatment, but involvement of the venous sinuses makes this method unfeasible. High-precision EBRT with sufficient tumor dose in combination with maximal resection preserving the venous sinuses could be a treatment option for longer tumor control.