There is very limited experience regarding the interventional radiological treatment of adrenocortical cancer (ACC). We present the case of a 57-year-old female patient with a large, potentially unresectable left-sided ACC and two hepatic metastases. Both liver tumors were effectively treated by trans-arterial embolization (TAE), followed by TAE of the bulky primary tumor as a life-saving intervention necessitated by severe intratumoral bleeding. Surgical removal of the primary tumor revealed complete necrosis. The patient is considered tumor free after 3.5 years. To the best of our knowledge, this is the first report to show that even a primary ACC may be completely ablated by selective embolization, and the fourth to prove the curative potential of liver TAE for ACC metastases. This case highlights the potential of selective embolization in ACC treatment. Adrenocortical cancer (ACC) is a rare malignancy with mostly bad prognosis. Most cases are diagnosed late, in the presence of distant metastases –8. The mThe use of ablative methods other than surgery for ACC are poorly documented in the literature. Here we present a case, where intraarterial embolization was effectively used both for liver metastases and a bulky primary tumor leading to complete necrosis and effective surgical removal. To the best of our knowledge, this is the first case to show that an ACC primary tumor may be completely ablated by selective embolization, together with liver metastases.2, serum albumin 35.5 g/L ). On physical examination, a large palpable left upper quadrant non-tender mass was found. The patient was afebrile and had normal cardiopulmonary parameters. There were no preexisting chronic diseases in the medical history, nor were there any potential inherited genetic disorders in the family. The patient did not take any medications. Abdominal contrast-enhanced computed tomography (CT) showed a 20 cm subdiaphragmatic retroperitoneal tumor together with two metastases in segment 7 (29x21 mm) and segment 8 (52x43 mm) of the liver : 19 kg/mThe timeline of the case with the most important therapeutic and diagnostic procedures is shown in 2, albumin 26.5 g/L). By this time, the otherwise non-tender abdominal mass that was still present on physical examination started to obviously cause difficulties in eating normal amounts of solid diet due to local gastric compression. The patient had severe concerns about a potential second bleeding but did not feel ready for a second operative exploration, which was mentioned to and discussed with her after the embolization. High caloric fluid supplementation diet was administered and prescribed before discharging her with stabile vital parameters. She was again referred to the multidisciplinary board of the university after a control CT in February 2017.Adjuvant mitotane was initiated, but target level could not be reached because of serious, mostly gastrointestinal intolerance symptoms including nausea, severe malaise and diarrhea. EDP chemotherapy was initiated. The patient was treated with therapeutic LMWH (low molecular weight heparin) because of CT-documented portal vein thrombosis. After 4 cycles of EDP, staging CT in August 2016 showed significant growth in the S8 liver metastasis (d=7 cm) with unchanged primary. To affect tumor growth, palliative superselective trans-arterial embolization of both liver metastases was performed with 2 mL 0.1mm PVA particles through the replaced right hepatic artery arising from the superior mesenteric artery  score 4:The CT documented a large necrotic mass occupying the tumor region Figure 4The patient has been regularly followed-up since June 2017 (CT or MRI every 3-6 months) without proof of viable residuum or tumor recurrence. The small S8 scar in the liver is not growing and considered non-viable. There is no sign of novel metastasis. The laboratory parameters including kidney function, liver enzymes and blood count are in the normal range. The patient gained weight (BMI 20) and feels completely healthy. Adjuvant therapy has not been initiated any more. At present, the patient can be regarded as tumor free.The biological nature of adrenocortical cancers is heterogeneous. Known risk factors are age, Ki-67-index, early presence of metastases, number of affected organs and R0 resection , 7. TherThe authors are unsure, whether the primary tumor would have been really unresectable, if the first attempt was done in a more experienced center; however, this first unsuccessful surgical exploration was a key factor in this series of partly unpredictable events showing that selective embolization of a primary ACC may lead to complete tumor necrosis. It is also very probable, that the patient would have later undergone liver resection, had she been referred to the university multidisciplinary team at the beginning. Going here through a very different path, a theoretically palliative method was proved to be curative.Transarterial embolization of liver tumors, mainly hepatocellular cancers , colorecThe number of reported primary or locally recurrent ACCs treated with embolization is 6 in the English literature. O’Keeffe et al.  reportedTreatment of ACC liver metastases should anecdotally have better results, but such cases are rarely reported. Koh et al.  documentThe variable arterial supply of the adrenal gland may limit the effect of its embolization: each adrenal gland may be supplied from 3 major arteries arising from the aorta/celiac trunk, renal artery, subphrenic artery to form a strong plexus under the capsule , but mosThe necrotic areas affected by TAE might become infected. These events are considered to be rare (0-1%) in terms of liver tumors, where there is much more documented experience available , 29. NecAbout two-thirds of patients develop local recurrence or distant metastases, and most of these tumors are diagnosed in the first two postoperative years –6. The mIn conclusion, we presented that not just liver metastases, but even a large primary ACC may be effectively treated by arterial embolization facilitating R0 resection. Based on the many times proved fact, that R0 resection is the key to survival in ACC, this example suggests a potentially more important role for preoperative embolization in ACC not just as a technical adjunct, but also as a potential method to improve oncological efficacy: transarterial embolization may represent an effective treatment modality in adrenocortical cancer.The original contributions presented in the study are included in the article/supplementary material. Further inquiries can be directed to the corresponding author.Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.GH: operating surgeon, surgeon of endocrine-oncology multidisciplinary team, concept of work and manuscript. AD: interventional radiologist. KF: ward surgeon and critical review. LK: head of surgical department and critical review. PR: ward endocrinologist and critical review. JT: multidisciplinary team endocrinologist and critical review. MT: endocrine-oncologist, head of multidisciplinary team, and critical discussion of case. PI: endocrinologist, outpatient care and follow-up, revision of manuscript, and treatment supervision. All authors contributed to the article and approved the submitted version.Hungarian National Research, Development and Innovation Office (NKFIH) grant K134215 to PI. The study was also financed by the Higher Education Institutional Excellence Program to Semmelweis University by the Ministry of Human Capacities in Hungary.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.


Ewing’s sarcoma (ES) is a rare and highly aggressive tumor belonging to a family of neoplasms of neuroectodermal origin, which primarily affects the bones or soft tissues. ES originating from lung parenchyma without chest wall involvement is extremely rare with less than 40 cases reported in the English literature. A 41-year-old man admitted to the thoracic surgery department presenting with intermittent non-productive cough, dyspnea, left-sided chest pain for two months for further evaluation and treatment with a preliminary diagnosis of pulmonary mass. Contrast-enhanced thorax CT and MRI revealed a large heterogeneous soft-tissue mass in the left lower lobe with no distant metastases or occult primary tumor. Following the percutaneous transthoracic biopsy, histopathological and immunohistochemical results were consistent with primary pulmonary ES. Though rare, primary pulmonary ES should be considered in the differential diagnosis of young patients presenting with a large heterogeneous soft tissue mass in the lung. This case report highlights the diagnosis, radiologic and pathologic findings, and management of primary pulmonary ES. The Ewing’s sarcoma family tumor (ESFT) are rare malignant tumors originating from neuroectodermal cells that commonly affect the long bones, such as arms, legs, ribs, vertebral column, and pelvis. ESFT occurs mainly in pediatric patients and young adults. Osseous Ewing’s sarcoma (ES), extraosseous ES, peripheral primitive neuroectodermal tumor (PNET), and malignant neuroectodermal tumor of the chest wall (Askin tumor) are all considered members of the ESFT. Approximately 16% of all ESFT are extraosseous . PrimaryA 41-year-old man admitted to thoracic surgery department with of intermittent non-productive cough, dyspnea and chest pain of two months. The past history was not contributory and there was no family history of any cancer. The laboratory tests, which included assessment of tumor markers  were completely normal. Physical examination of the lungs revealed absent breath sounds at the left lung base. A chest radiograph confirmed a large pleural effusion and total atelectasis of the left lower lobe. A diagnostic thoracentesis of pleural effusion was performed that was cytologically negative. A chest tube was then inserted in the left side posterior pleural cavity. Due to continuity of the opacity on subsequent radiograph, the patient was investigated with contrast-enhanced chest multidetector computed tomography (CT) and contrast-enhanced chest magnetic resonance imaging (MRI) for suspicion of mass. CT scan was performed using 64-slice CT scanner  after intravenous contrast administration . And the patient was also scanned with 3T MRI scanner  with intravenous gadolinium contrast administration . Contrast-enhanced CT and MRI revealed a large, relatively well-defined, noncalcified heterogenous soft tissue mass measuring approximately 15x15x14 cm within the left lower lobe that compressed and displaced the left hilum, mediastinum and left atrium and ventricule  in the left lower lobe parenchyma, with no additional increased activity to suggest an occult primary or metastatic disease. Ultrasound-guided tru-cut needle lung biopsy was performed. Histologically, the tumor was composed of uniform small round cells in sheets with scant clear cytoplasm and round nuclei containing fine chromatin  and radiation therapy. Adjuvant and/or neoadjuvant chemotherapy may affect the overall survival and improve the treatment results in the future. Some studies have been reported that ESFT can respond dramatically to initial therapy, with robust initial responses predicting a better outcome ,7.Despite being a rarity, pulmonary ES should always be considered in the differential diagnosis in young patients with pulmonary soft tissue tumors. Diagnostic imaging has an increasingly important role in accurate diagnosis, management and follow-up. The definitive diagnosis of ESFT is made based on histomorphologic immunohistochemical and molecular genetic features.


Adrenal cysts are a rare entity that is usually nonfunctional and asymptomatic. Their association with adrenal neoplasms was rarely described in the literature. We report a unique case of a 40 -year-old woman who was referred for evaluation of a left adrenal incidentaloma with subclinical Cushing's syndrome. The tumor was suspicious for malignancy regarding computed tomography scan (CT scan) features. Therefore, a laparoscopic left partial adrenalectomy was performed. Pathology examination showed multilocular spaces lined by endothelial cells which are compatible with endothelial adrenal cyst, associated to an adrenocortical adenoma. We further discuss the management of adrenal cyst with review of the literature. Adrenal cysts are rare cystic masses that arise from the adrenal gland. They are usually nonfunctional and asymptomatic measuring less than 10 cm. These lesions may occur simultaneously with other adrenal tumors associated with hormonal hypersecretion. Only few papers describe the association between an adrenal cyst and adrenocortical adenoma.We report one of the rare cases of a functioning adrenal cyst incidentally discovered.A 40-year-old woman was referred to our endocrinology department to evaluate a left adrenal incidentaloma discovered when a CT scan was performed because of her history of ureteral stones revealing a left adrenal mass of 34 × 27 mm. She had also hypertension treated with amlodipine 10 mg 2 months ago and did not complain neither of headache nor diaphoresis or palpitations.2, and waist size 105 cm). But, her blood pressure levels were not well controlled under amlodipine 10 mg.No clinical features of Cushing's syndrome were noticed on physical examination regardless of moderate obesity with android distribution  besides the low-dose dexamethasone suppression test (DST) showed an absence of cortisol suppression, which was consistent with the European Society of Endocrinology criteria of autonomous cortisol secretion [Her plasma levels of midnight cortisol of two different days were 5, 2–4, and 2 ug/dL , urine free cortisol values of three different days were 36.3, 42.8, and 3.25 ug/day , and serum dehydroepiandrosterone sulfate level was 119 ug/dL . However, her plasma adrenocorticotropic hormone (ACTH) concentration was at the low limit: 10.4 ng/L , and the overnight 1 mg dexamethasone suppression test  and triglyceride: 3.21 g/l  and decreased level of HDL cholesterol: 0.47 g/L . Fasting blood sugar level was 0.85 g/L (N: 0.70–1.10 g/L) and hemoglobin A1c level (HbA1c) was 5.2%. The 75 g oral glucose tolerance test noted an impaired glucose tolerance. Bone density was normal.The CT scan revealed a rounded well-defined hypodense lesion of the left adrenal. Its size increased to 44 × 29 mm. Spontaneous density was 22 Hounsfield unit (HU) , and absAccording to the clinical, laboratory, and imaging findings, the left adrenal mass was suspicious. Laparoscopic retroperitoneal cystic resection and left partial adrenalectomy were performed. It is the elective surgical procedure adopted by our urologist, for numerous advantages, especially for a shorter surgery duration and a minor intraoperative bleeding. Hydrocortisone replacement was not administered after the surgery because the patient did not have signs or symptoms of adrenal insufficiency.At three months after surgery, her weight turned to 88 kg and her hypertension was stabilized under amlodipine 10 mg. Plasma 8 am ACTH concentration increased to 28 pg/mL, and plasma cortisol concentration decreased compared with her preoperative levels . Moreove∗ 2.3 ∗ 1.7 cm mass weighing 13 g with a partially yellowish cystic appearance.Pathological analysis of the resected tumor reported a 4 Histological examination of the different specimens showed a cystic cavity with a fibrous wall and constituted of multilocular spaces lined by endothelial cells and filled with red blood cells  without Adrenal cysts are generally rare, and around 600 cases have been documented in the literature so far , 4. The Clinical symptoms are related to the position and size of these benign cysts, although abdominal pain, gastrointestinal disorders, dyspnea, and palpable mass can be noted in large cysts. Besides, intracystic hemorrhage, infection, and hypertension have been also reported . Our patIn 1966, Foster  classifiEndothelial cysts, also known as “simple cysts,” are usually thin-walled and multilocular measuring less than 2 cm. They are defined by their smooth and flattened endothelial lining, in addition to their yellow-tinged serous fluid . They caEndothelial cysts are typically described at CT imaging as unilateral, with different sizes, thin walls (<3.5 mm), smooth borders, and multilocular structure besides septal calcifications . They haSince functioning adrenal cysts have been described in the literature , all patOnly a few case series and reports of endothelial cysts have been documented in the literature, and their association with other adrenal neoplasms is even uncommon. In fact, Erickson et al. reported only one case of endothelial cyst associated with pheochromocytoma , and Nigβ-hydroxysteroid dehydrogenase (3βHSD), P450c17 α-hydroxylase (P450c17) and P450 21 hydroxylase (P450c21)1, and DHEA-sulfotransferase (DHEA-ST). This exploration was carried out in case 1 and case 2; however, it was not made in our case because of economical issues.Our case supported what was previously described, and the gender was the same in the three cases except for the third one. The masses were incidentally discovered in the CT scan. Clinically, our patient had poorly controlled hypertension with obesity just as the case reported by Nigawara et al. . BiologiThe pathogenesis of an adrenal endothelial cyst formation within an adrenocortical adenoma is still a mysterious issue. Several theories suggested that the cyst may arise from a preexisting vascular hamartoma , 14 or lUp to now, no guidelines have been established on the management and treatment of cystic adrenal lesions because of the low incidence of these lesions and the difficulty in achieving a definitive preoperative diagnosis. These particular cases require a multidisciplinary team when malignancy is suspected, a hormone secretion excess has been established, and when surgery is envisaged . IndicatTwo procedures of laparoscopic adrenalectomy have been described in the literature. The transperitoneal approach is more appropriate to resect large tumors because it allows evaluation of the peritoneal space. However, the retroperitoneal technique provides good visualization of the retroperitonal anatomy, direct access to the adrenal tumor, and short duration of surgery and minimizes operative bleeding. Several studies have shown that the outcomes of the retroperitonal approach were superior to those of transperitoneal approches regarding adrenal tumors resection. Nevertheless, there is a lack of evidence regarding the effectiveness of the two approaches' comparison in patients .Different methods have been suggested, and complete adrenalectomy was recommended by most authors , 24. AccIn our case, a small cystic tumor discovered at the time of the surgical exploration allowed for partial adrenalectomy.Open surgery should be advocated either in complicated cases, larger cyst size, or preoperative evidence of massive compression or infiltration of adjacent structures to avoid mini-invasive approaches' complication .To sum up, this is a case of adrenal cyst presenting with subclinical Cushing's syndrome. Preoperative diagnosis was made by CT scan, and laparoscopic partial adrenalectomy was performed without complications. The pathology was consistent with an adrenal endothelial cyst associated with adrenocortical adenoma. As far as we know, this is the fourth reported case of adrenal endothelial cyst associated with adrenocortical adenoma. Although it was regarded as a benign tumor in our particular case, the potential risk of malignancy when managing adrenal cyst should be taken into consideration before any surgery.


This case report describes a rare presentation of superficial mucocele along the midline of the hard palate in a 30-year-old male patient, which was histopathologically confirmed post-surgical excision; the patient is currently disease free with no recurrence for six months.Superficial mucoceles are benign, small, translucent vesicles occurring in any part of the oral cavity, due to extravasation of saliva due to ruptured minor salivary gland ducts. This distinct entity presents as single or multiple asymptomatic vesicles. The etiology is unclear; however, these are not associated with a history of trauma, unlike the conventional mucoceles. These lesions tend to be recurrent and are occasionally associated with discomfort to the patients. Since they clinically mimic various vesiculobullous lesions such as pemphigoid, bullous lichen planus, or herpes virus infection, they are often misdiagnosed. Asymptomatic superficial mucoceles or lesions in multiple locations do not require treatment. Nevertheless, the treatment for lesions causing discomfort includes surgical excision, cryosurgery, and carbon dioxide (CO Superficial mucoceles are reported as translucent vesicles occurring in any part of the oral cavity where there is presence of minor salivary glands . Eveson,A 30-year-old male patient presented to the oral medicine outpatient department with a chief complaint of blisters in the roof of the mouth for one month. He claimed that the swelling ruptured upon slight trauma with history of recurrence subsequently. Intraoral soft tissue examination revealed evidence of two dome shaped, vesicles of size 3 x 3 mm in the left posterior region of the hard palate, along the midline. On palpation, the vesicles were firm in consistency, tender with no evidence of any discharge Figure .No tooth related abnormality was detected clinically; intraoral periapical radiograph and occlusal radiograph did not reveal any pathology. Correlating the history and clinical features, a provisional diagnosis of superficial mucocele in the hard palate was given. Differential diagnosis of vesiculobullous lesions such as mucous membrane pemphigoid, bullous lichen planus, and herpes infection was considered. The hemoglobin levels were with in normal limits and excisional biopsy of the swelling was performed. The histopathology revealed the presence of extravasated mucin containing mucinophages from adjacent minor salivary glands, extravasated RBCs and few inflammatory cells, with peripheral para-keratinized stratified squamous epithelium, suggestive of mucocele Figure .The healing was satisfactory post-surgical excision and there was no recurrence at six months follow up.Superficial mucoceles are distinct entities representing sub-epithelial extravasation of sialomucin at the interface of the epithelium and connective tissue . It has 2) [It has been suggested that superficial mucoceles are self-limiting; no biopsy or treatment is necessary, especially for asymptomatic cases ,3,6. Xu 2) , Neodymi2)  have bee2) . A case 2) . In the Superficial mucoceles are a relatively rare and distinct entity. Identification of such benign lesions by clinicians is usually through proper history taking and thorough intra oral examination, which helps to arrive at an accurate diagnosis. Though such lesions pose a diagnostic and therapeutic challenge to the clinician because of close resemblance of clinical presentation of lesions with different etiologies such as reactional inflammatory processes, infections, immune and iatrogenic responses, systematic clinical examination aided with histopathological evaluation will avoid misdiagnosis and enable better management of patients with superficial mucoceles.


Background: The central nervous system is an unusual location of sarcoidosis, which commonly affects the cranial nerves, meninges, hypothalamus, and pituitary gland. Involvement of the pineal region is extremely rare. This systematic review focused on the diagnosis and management of pineal region sarcoidosis, dorsal mesencephalon, and periaqueductal region.Objectives: This study aimed to discuss diagnostic modalities and best management tools of the aforementioned pathology.Methods: ScienceDirect, PubMed, and Google Scholar databases were searched for English or French articles about sarcoidosis of the pineal region, dorsal mesencephalon, and periaqueductal region. The clinical case of a patient managed at our department that we believe is directly relevant to this review is also presented. Patients’ demographics, clinical presentations, presence of hydrocephalus, other sarcoidosis locations in the central nervous system, and medical treatment were collected. Surgical management, surgical approach, and outcomes and complications of each procedure were also obtained. This study was conducted in agreement with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses statement.Results: Fifteen cases were examined. The study sample consisted of nine (60%) male and six (40%) female, and the mean age was 32 years. Eight (53%) patients had hydrocephalus, and the predominant clinical presentations were signs of increasing intracranial pressure . Six (40%) patients had diplopia, and convergence–retraction nystagmus was noted in three (20%) patients. Argyll Robertson sign was present in one patient and suspected in another patient (13%). Medical treatment consisted mainly of steroids (93% of cases). Open surgery on the pineal region was performed in five patients, and four of them reported to have serious complications . Endoscopic management was performed in two patients without complications.Conclusion: To treat hydrocephalus, brain imaging is mandatory in patients with sarcoidosis if intracranial hypertension is suspected. In pineal region sarcoidosis, management of hydrocephalus is the priority, followed by medical treatment of the lesion. Open surgery of any approach presents a high risk of complications; thus, an endoscopic approach is the preferred management, as it treats hydrocephalus and makes biopsy possible with minimal risk. Pineal region sarcoidosis (PRS) is challenging to diagnose and manage. This study is considered a wider update about PRS and thus aimed to clarify the best diagnosis and management methods for this pathology.Sarcoidosis is a systemic condition of unknown originThe patient was a 51-year-old female nurse with a medical history of high blood pressure, type I diabetes, and cholecystectomy. The patient was followed for systemic sarcoidosis under steroids, colchicine, and chloroquine. The patient presented with a sudden diplopia accompanied with intermittent headaches.Neurological examination did not find any objective deficit. The ophthalmological examination found bilateral sixth nerve palsy in the Lancaster test , her visAn endoscopic third ventriculostomy and biopsy of the lesion were performed. Postoperatively, the patient's condition improved clinically and diplopia disappeared . The int3 The protocol for this systematic review was not registered.ScienceDirect, PubMed, and Google Scholar databases were searched for journal articles about sarcoidosis of the pineal region, dorsal mesencephalon, and periaqueductal region. The following keywords were used: “pineal,” “tectal,” “tectum,” “quadrigeminal,” “mesencephalon,” “aqueduct,” “splenium,” “tentorium,” “tentorial,” “obstructive hydrocephalus,” “noncommunicating hydrocephalus,” and “triventricular hydrocephalus.” Articles with title containing one of the keywords and “sarcoidosis” were reviewed. The study included English (n = 13) and French (n = 1) articles, and there was no publication date limitation. The Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines were applied to this review.The following articles were excluded from the review: three articles in Japanese, three articles where the lesions were strictly located in the anterior mesencephalon, and one article where the full text was not available (abstract only without case presentation). Patients’ demographics, clinical presentations related to the pineal region location of the pathology, presence of hydrocephalus, other neurosarcoidosis locations, and medical treatment were collected. The study focused on the surgical management: whether surgery was performed, surgical approach, and outcomes and complications of each procedure. In one case report, the corresponding author was contacted  but could not give all previous data.The numbers of studies screened, assessed for eligibility, and included in the review are summarized in 2 Many authors have suggested that sarcoidosis granuloma is a result of an exaggerated immunologic response to an unknown antigen, causing inappropriate T lymphocyte reaction after acquiring a cellular immunity. This inappropriate response could have a genetic origin; in fact, many cases of familial sarcoidosis have been reported, but a causative gene has not been identified.2 An environmental influence has been suggested, as the pathology commonly affects some professionals such as firefighters and nurses.2 Nervous system involvement accounted for 5%–13% of all sarcoidosis cases14; cranial nerves, meninges, hypothalamus, pituitary gland, spinal cord, and peripheral nerves are commonly affected.14 Pineal region involvement is extremely rare.16 Similar to other neurosarcoidosis, PRS affects young adults2; only two patients in the present series were over 50 years old . Although neurosarcoidosis is more common in the female population according to most studies,2 pineal region involvement is more common in the male population . In 2019, Saban et al., reported 20 cases of hydrocephalus caused by neurosarcoidosis, and 60% presented with obstructive hydrocephalus.18 This case was also included in the present review; in fact, the major feature of PRS is the position of the lesion in the outflow of the cerebrospinal fluid (CSF), which could cause obstructive ventricular dilation. Hydrocephalus in neurosarcoidosis could also be nonobstructive (communicating); in this case, it is caused by granulomatous leptomeningitis in basal cisterns that reduces absorption of the CSF in the arachnoidal villi.23 Including our case, eight cases from the presented series had hydrocephalus, which is a serious complication. On forensic papers, several cases of sudden death were considered caused by obstructive hydrocephalus in neurosarcoidosis.21 Thus, brain imaging should be a routine, and conservative management of hydrocephalus should not be considered. In the present review, the clinical presentation of PRS was dominated by signs of intracranial hypertension in patients with active hydrocephalus, presented mainly with headaches, vomiting, and papilledema. Moreover, diplopia is very common; including our case, six (40%) patients had diplopia caused by sixth nerve palsy (6 was present in one case and suspected in another (13%). Other presentations are related to other lesion locations  clinical syndrome and exclusion of other origins. The diagnosis is probable if the patient presents a clinical syndrome involving sarcoidosis, with other examinations supporting the diagnosis, such as MRI or laboratory tests suggest a CNS inflammation  as well as positive systemic sarcoidosis , with no evidence of alternative disease. Finally, the diagnosis is possible if a clinical syndrome is present, lesions from other origins were not found, and previous criteria are not met.24 Our PRS management depends on whether the diagnosis of sarcoidosis is already made and on the presence or absence of hydrocephalus and its type , followed by measurement of tumor markers and cytological analysis of the CSF. The surgeon could also perform a biopsy in visu of the lesion. Finally, shunt-related complications are not possible.25 In the present series, most patients who underwent open surgery experienced complications postoperatively. The most serious complications related to open surgery are oculomotor deficit,11 hemiparesis,9 cerebellar syndrome,9 cephalic tremor,9 and hemianopsia.11 Our case and another case in the review were managed by endoscopic biopsy and ETV, which both remarkably improved postoperatively. An external ventricular drain appears to be not a good choice anymore; it gives a temporary solution for the hydrocephalus and increases the patient's risk of meningitis. Herein, it was used in two patients: replaced in one by a VP shunt15 and removed in the other case on postoperative day 7 without need for any shunt.10 In this review, PRS is often accompanied with lesions located in other parts of the CNS (40%); thus, the physician should be aware and predict those locations before the onset of major signs. In PRS management, complications are related to invasive procedure aiming at aggressive removal; otherwise, the outcome is generally good with total recovery of most patients, and the prognosis is related to other systemic complications of the disease.The pineal region is the part of the brain limited superiorly by the splenium, anteriorly by the posterior wall of the third ventricle, inferiorly by the tectal plate, laterally by the thalamus, and posteriorly by the falcotentorial angle, splenium, and vermis. Sarcoidosis is a systemic granulomatosis of unknown origin.ocations . Brain cits type . If no vBrain imaging is mandatory in patients with sarcoidosis if intracranial hypertension is suspected to eliminate the presence of hydrocephalus. In case of PRS, the management of hydrocephalus is the priority, followed by medical treatment. If diagnosis is already known, medical treatment could be totally efficient. Biopsy is indicated in the absence of clear diagnosis. Open surgery of any approach presents a high risk of complications; thus, endoscopy is the preferred management, as it treats the hydrocephalus and makes biopsy possible with minimal risk.


Malignant peritoneal mesothelioma is a local-regional disease process that requires a comprehensive treatment plan including complete cytoreductive surgery and regional chemotherapy.Treatments used in our patient began with a complete cytoreductive surgery. This required visceral resections, parietal peritonectomy, peritonectomy of the small bowel and its mesentery, and a peritoneal resection of the colonic mesentery with sparing of the major vasculature of the large bowel.Peritoneal resection of the colonic mesentery and other treatments were performed in the absence of major complications. A 20-day hospitalization was required. The patient shows no internal hernias and no evidence of disease by CT follow-up at 4 years postoperatively. Her quality of life is excellent.Malignant peritoneal mesothelioma was in the past a disease of limited survival without effective treatment options. Peritoneal resection of the colonic mesentery may be required for complete cytoreduction. A sequence of cytoreductive surgical procedures and regional chemotherapy treatments has made long-term survival possible. •Malignant peritoneal mesothelioma has treatment options that combined surgery and regional chemotherapy.•A major surgical challenge is complete mesenteric peritonectomy.•Complete cytoreduction of the large bowel mesentery requires its resection with sparing major vasculature.•Long-term survival is possible with low perioperative morbidity and mortality. This peritonectomy involves a resection of mesentery and peritoneum surrounding the vasculature of the colon and rectum with a sparing of the vessels. A patient who received this total colonic mesenterectomy is presented.2www.researchregistry.com website with UIN 6986.Data on this patient was prospectively recorded and then retrospectively reviewed at an academic institution. This research work has been reported in line with the SCARE 2020 criteria 2.1H. pylori which was treated with antibiotics. Colonoscopy was normal. Abdominal and pelvic MRI was read as normal and symptoms were attributed to menopause.Fall of 2014: This 51-year-old woman noted a lack of energy and increasing fatigue. Abdominal distention was first noted in mid-2015. Upper endoscopy was performed which showed November 2016: A persistent thrombocytosis was noted. An abdominal ultrasound January of 2017 was abnormal.January 29, 2017: CT of abdomen and pelvis showed ascites. Fluid and enhancing nodules were depicted in the right upper quadrant. The greater omentum had a nodular infiltrate. Small bowel regions were interpreted to be class 1 February 20, 2017: An ultrasound-guided biopsy revealed diffuse malignant epithelial mesothelioma. CA125 tumor marker was normal.Family history and review of systems was non-contributory.March 21, 2017: Physical examination was performed. Chest and abdominal exams were normal except for a 1.5 cm nodule within the umbilicus. Pelvic examination was unremarkable.The treatment plan that was presented to the patient involved cytoreductive surgery, hyperthermic intraperitoneal chemotherapy (HIPEC), early postoperative intraperitoneal chemotherapy (EPIC), placement of an intraperitoneal port and six cycles of combined normothermic intraperitoneal and systemic chemotherapy (NIPEC) March 31, 2017: A nine-hour cytoreductive surgical procedure with HIPEC was performed. A midline abdominal incision was made with skin tabs so that the umbilicus could be reconstructed The Thompson retractor was inserted. We could explore the abdomen. There was tumor layered out approximately 2 cm thick beneath the right hemidiaphragm. The left hemidiaphragm was clear. The right retrohepatic space was also layered by mesothelioma. The gallbladder was involved, but the stomach was not. The greater and lesser omentum were infiltrated by tumor. The tumor was densely adherent to the transverse colon, especially at the hepatic and splenic flexure. Both pericolic sulci were layered by tumor. The small bowel contained approximately 1000 tumor nodules on its mesenteric surface . The utebloc with the pelvic peritoneum and uterus, tubes and ovaries  was infused intravenously as rapidly as possible 15 min prior to the initiation of HIPEC and 4 h and 8 h later The cytoreductive surgery was followed by HIPEC and EPIC. A pair of curled peritoneal dialysis catheters  were used to infuse the chemotherapy solution and four outflow catheters to drain the chemotherapy solution to allow recirculation through the heat pump. Two drugs were administered intraperitoneally in 1.5 l/m2.1.22 was administered intraperitoneally. The carrier solution for the paclitaxel was 6% hetastarch solution  administered through an infusion pump at 1000 ml/h 2. A 20-day hospitalization with the patient on total parenteral nutrition through a peripheral intravenous central catheter (PICC) was necessary before return of bowel function.The Tenckhoff catheter and closed-suction drains were maintained after the 90 min of intraoperative chemotherapy. EPIC administration was initiated on the first postoperative day. A 1-liter chemotherapy solution containing paclitaxel at 20 mg/m2.1.32 in 1 l of 1.5% dextrose peritoneal dialysis solution infused at 1000 ml over 1 h. Following intraperitoneal pemetrexed administration, cisplatin at 75 mg/m2 was infused intravenously in 250 ml of normal saline over 120 min. These treatments were repeated for a total of 6 cycles with 3 weeks between each treatment adding approximately 6 months of intensive postoperative management Prior to closure of the abdomen, an intraperitoneal port  was implanted 2.1.4CT was performed every 6 months with maximal oral and intravenous contrast. At 4 years, the patient remains disease-free, fully ambulatory with normal bladder and bowel function.33.1Peritoneal mesothelioma has two unique features as compared to other cancers that occur within the abdomen and pelvis. First, this is a malignancy that lacks a primary site. All other cancers start at some specific location or within a specific organ. For example gastric cancer, colon and rectal cancer, ovarian cancer and retroperitoneal sarcoma. Peritoneal mesothelioma arises from the peritoneum but is not limited to any specific site within the abdomen and pelvis. It also progresses on both parietal and visceral peritoneal surfaces. A second unique feature is the progression limited to within the peritoneal spaces. Even as the disease progresses to a terminal state, malignant peritoneal mesothelioma remains confined within the abdomen and pelvis. Direct extension through the tendinous mid-portion of the right or left hemidiaphragm to involve the pleura does sometimes occur, but distant metastases through the blood is very unusual.It is this second unique feature of peritoneal mesothelioma that causes aggressive local-regional treatment strategies to be successful. Remarkable improvement in survival has occurred as cytoreductive surgery combined with HIPEC has become standard of care for selected patients with epithelial peritoneal mesothelioma 3.2Multiple studies have documented that the long-term survival of patients with malignant peritoneal mesothelioma is dependent on the resection of all visible disease from the abdomen and pelvis. Since peritoneal metastases is a disease of the peritoneum itself, this structure must be extensively removed in the majority of patients. Parietal peritonectomy is usually performed without difficulty because of the generous tissues supporting this structure. The parietal peritoneal specimen in need of stripping remains intact so that a complete removal with a margin of heat necrosis provided by high-voltage electrosurgery is possible In contrast, the visceral peritoneum does not have a fibrous tissue support that allows for the stripping of an intact specimen. In selected patients the tumor itself holds the peritoneal layer intact to allow the mesenteric peritonectomy to be performed 3.3Peritonectomy of the mesocolon of ascending colon, transverse colon and descending colon presents the surgeon with a dilemma. A formal mesenteric peritonectomy is not possible because there is no mesenteric adipose tissue beneath as in the small bowel mesentery. Scissor dissection will invariably result in multiple perforations through the mesocolon. The technology used successfully on the patient presented is the total resection of the mesocolon with a sparing of its vasculature. The resection of the avascular sections of the mesocolon is illustrated in No closure of the open spaces between colonic vasculature is possible. The mesenteric defect between ileocolic vessels and middle colic vessels is at risk for an internal hernia but the spaces created beyond the middle colic vessels is immediately adjacent to the retroperitoneum with small bowel superficial to its posterior position. No internal hernia has occurred in the patient presented.4This patient population illustrates that long-term survival is possible in patients with diffuse large volume epithelial type malignant peritoneal mesothelioma. Success begins with cytoreductive surgery that requires visceral resections, mesenteric peritonectomy of the small bowel and its mesentery. Clearing of the mesentery of the colon requires resection of the mesocolon with sparing of the ileocolic and middle colic vessels. HIPEC, EPIC and long-term bidirectional intravenous cisplatin and intraperitoneal pemetrexed follows with access to the peritoneal space by an intraperitoneal port.Foundation for Applied Research in Gastrointestinal Oncology.Data management and secretarial support provided by MedStar Health Institutional Review Board has determined that a case report of less than three Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.www.researchregistry.com website with UIN 6986.This study was registered as a case report on the Paul H. Sugarbaker, MD.Not commissioned, externally peer-reviewed.Paul H. Sugarbaker: study concept or design, data collection, data analysis or interpretation, writing the paper.Paul H. Sugarbaker has no conflicts of interest to declare.


To discuss the clinical application value of contrast-enhanced ultrasound (CEUS) in testicular occupied lesions.  Nine conventional-ultrasound-found testicular occupied lesions which underwent CEUS meantime were analyzed retrospectively. The CEUS perfusion pattern was compared with the surgical pathological result or follow-up findings.  Among all the 9 testicular occupied lesions, there were 5 testicular malignant tumors, 1 testicular benign tumor, 1 testicular tuberculosis, and 2 testicular hematomas. CEUS diagnosed 6 testicular malignant tumors, 1 testicular benign tumor, and 2 testicular hematomas, and its diagnostic accuracy was about 88.9%.  CEUS has high clinical application value in the differential diagnoses of benign and malignant testicular occupied lesions. Testicular tumors are relatively rare, typically speaking, which accounts for only 1% of all sorts of malignant tumors of the whole body, but they are the most common solid tumors in young men aging from 20 to 40 years old, almost all of which are malignant . In mostDue to its convenience and noninvasive advantages, ultrasound examination has been widely used in the diagnosis of testicular occupied lesions, and it has become an important method of distinguishing benign and malignant testicular tumors. Contrast-enhanced ultrasound (CEUS) is a safe, relatively cheap, and widely available imaging technology . It usesCollecting testicular occupied lesion cases during November 2013 and September 2020, which were detected by routine ultrasound examination in our hospital, we had 9 patients in all. Five cases had lesions in the right testicles and 4 cases in the left ones. Besides these, 2 cases had a history of testicular trauma. Patients were aged from 30 to 59 years old, 38 years old on average. The size of lesions was 52 mm × 25 mm × 25 mm maximum and 8 mm × 3 mm × 7 mm minimum. CEUS was performed in all 9 cases.Aplio 500  ultrasonic scanner with a 5~14 MHz linear transducer was used for an ultrasound examination. It was also equipped with color Doppler flow imaging (CDFI) and CEUS imaging software. The patients lay supine on the couch, fully exposing the scrotums. We scanned the scrotums from multiple sections and angles. First, the testicular scan was performed with conventional ultrasound, focusing on the observation of the location, size, number, morphology, boundary, and internal echo of the lesions. CDFI was then used to observe the distribution of blood flow in and around the lesions. After that, we switched it to CEUS mode. SonoVue was used as the contrast agent. 5 ml of normal saline (NS) was injected before use, and 2.4-3.0 ml of the mixture was extracted after shaking. After windowing the best image of the lesion, the extracted mixture was injected rapidly through the cubital vein, followed by 5 ml of NS, and a timer was set simultaneously. After contrast agent injection, real-time images were observed continuously for 3-5 mins. All images were archived for offline analysis. Two to three experienced senior sonographers repeatedly reviewed the playback clips, so as to dynamically observe the changing trend of contrast agent; summarized the perfusion pattern of contrast agent in terms of time and echo intensity; and analyzed the CEUS results. Finally, the medical history and pathological data were followed up for comparative analysis.CEUS descriptions: (1) the enhancement intensity of the mass was divided into hyperenhancement, isoenhancement, hypoenhancement, and no enhancement; (2) the homogeneity of enhancement was divided into homogeneous enhancement and heterogeneous enhancement; (3) compared with normal testicular tissues, the entering time and washout time of the contrast agent were observed, and the enhancement phase of mass was divided into fast-in and fast-out, fast-in and slow-out, slow-in and fast-out, slow-in and slow-out, etc.Testicular occupied lesions showed hypoechoic or heterogeneous echogenic regions in the testis on gray scale ultrasonography and weak or rich blood flow signals in some cases on CDFI . There wAll 9 patients with testicular occupied lesions were followed up or surgically treated.Six cases were suggested as malignant tumors by CEUS, and 5 of them were confirmed correct by surgical pathology (including 3 seminoma cases and 2 mixed germ cell tumor cases). The other one was testicular tuberculosis confirmed surgically. CEUS showed a benign tumor, and the surgical pathology was an adenomatoid tumor of the testis. Two cases of testicular hematoma were confirmed by follow-up observation. The CEUS diagnostic accordance rate of testicular occupied lesions was approximately 88.9% (8/9).Testicular tumors are classified into germ cell and nongerm cell types. Germ cell testicular tumors account for the majority of them, of which about 95% are malignant, and seminoma is the most common type . NongermEarly detection, diagnosis, and treatment are the keys to improve the cure rate of testicular tumors . UltrasoAmong the CEUS manifestations of all the 5 testicular malignant tumor cases, fast-in and fast-out enhancement was seen in 2 seminomas and 2 mixed germ cell tumors, and fast-in and slow-out enhancement was seen in 1 seminoma. Heterogeneous hyperenhancement was seen at their peak time. One testicular adenomatoid tumor presented fast-in and fast-out enhancement and slightly heterogeneous hyperenhancement at the peak time, which was similar to a testicular malignant tumor, indicating abundant blood supply inside. A testicular adenomatoid tumor is a kind of benign tumor without typical clinical manifestations. Substantially occupied lesions on gray scale ultrasound can usually be seen in the regional part of the testicle, which is with clear boundaries, homogeneous internal echo, and hypoecho or isoecho compared with the surrounding tissues . Since tThe reason why 1 case of testicular tuberculosis was misdiagnosed as a malignant testicular tumor in this study was that it had similar CEUS characteristics to testicular malignant tumors, which brought certain difficulties to the differential diagnosis. So, when we meet patients with diffuse nodules, details of TB history should be inquired about, because the exact clinical history data plays a suggestive role in the diagnosis of TB .Ultrasonic contrast agent SonoVue is the ideal contrast agent; its characteristics are listed as follows: (1) nontoxic; (2) possible to intravenously inject; (3) circulation through the lungs, heart, and capillaries; and (4) stability of recirculation. Ultrasound contrast agents can increase the reflective difference between the tumor and normal tissue and enhance the Doppler blood flow signals inside lesions. The vascular morphology, distribution, and blood supply status of tumors can be displayed more completely. It is possible to effectively improve the detection of tumor vessels, so as to evaluate them more accurately. When coming to combined diagnosis with CDFI, CEUS is hopeful to elevate the diagnosis rate and differential diagnosis rate .This study has limitations. The number of research cases is limited, and we call for further in-depth research with a larger sample size to improve the diagnostic accuracy of testicular space-occupying lesions. In future studies, I will collect more clinical samples for further verification. We will study the clinical application value of CEUS in the diagnosis, staging, and follow-up of testicular space-occupying lesions.In conclusion, as it is noninvasive, nonradiative, and possible to observe the real-time blood perfusion of testicular occupied lesions dynamically, CEUS has a high clinical application value of the diagnosis and differential diagnosis concerning benign and malignant testicular occupied lesions. We reported for the first time the application value of CEUS as a benign and malignant diagnosis and differentiation of testicular space-occupying lesions. It has important value in the diagnosis of testicular space-occupying lesions.


Gossypiboma is a mass of foreign body with cotton matrix accidentally left inside the body after a surgical procedure. It is a surgeon’s nightmare and has a varied presentation ranging from asymptomatic cases to the formation of an abscess, mass, intestinal obstruction/perforation, malabsorption, gastrointestinal hemorrhage, and various internal and external fistulization. Genital tract fistulas are one of the most distressing conditions for women of reproductive age that not only hamper their day-to-day work but also impair their social life and psychological state. Colouterine fistula is a rare pathology and has been mainly reported as a complication of diverticulitis in the elderly. We present here a case of gossypiboma presenting as colouterine fistula in a young lady following lower segment cesarean section. The case highlights a rare complication of gossypiboma, probably the first of its kind, and the diagnostic challenges that it presents. Gossypiboma  is described as a mass of a foreign body with a cotton matrix left inside the body cavity during a surgical procedure. The reported incidence of this misevent is between one and 1000-1500 abdominal surgeries . It has A 28-year-old woman presented with complaints of pain and progressively increasing swelling in the left side of the lower abdomen for three months. She also complained of fecal discharge with the periodic involuntary escape of gas through the introitus. She had undergone emergency lower segment cesarean section for obstructed labor four months back with intraoperative hemorrhage managed successfully with blood transfusion and pressure application.Abdominal examination revealed a firm lump of size 15 x 10 cm in the left lower abdomen with the lower border going into the pelvis. Per-speculum and per-vaginal examination showed the vaginal cavity filled with pus-mixed fecal content with a bimanually palpable mass in the left fornix.Ultrasonography (USG) of the pelvis revealed air foci in the endometrial cavity with multiple surrounding loculated abscesses and a hyperechoic mass with posterior acoustic shadowing in the left parauterine space Figure . MagnetiThe patient was planned for exploration with consent for stoma and hysterectomy. Laparotomy revealed a thick-walled abscess cavity in the lower abdomen surrounding a surgical sponge in the left parauterine space Figure . It had Gossypiboma often becomes a differential diagnosis, by exclusion, of soft tissue masses or localized abdominal pain in a patient with a history of prior operation. The well-known risk factors are emergency surgery, change in surgical team, obesity, hemorrhage, and a high count of sponge/instruments used . In our Clinical presentation depends upon the location of the foreign body and the type of inflammatory response. Gauze and cotton pads can cause aseptic fibrous or exudative responses . The fibDiagnosis of gossypiboma is not straightforward because the cotton can simulate hematoma, granulomatous process, abscess formation, cystic masses, or neoplasm . AlthougAmong the various treatment options for colouterine fistula, Hartman’s procedure, transverse colostomy, closure of the fistula without bowel resection, and en bloc resection of the uterus and sigmoid colon are several available surgical treatment options ,7-8. AltGossypiboma should be included in the differential diagnosis of soft tissue masses or localized abdominal pain in a patient with a history of prior operation. The diagnosis is often difficult to make. Fecal discharge per vaginum can be a presentation of a rare pathology like colouterine fistula. Counting gauges, sponges, and instruments before and after the surgery; inspection of the operative field thoroughly before closing; using radio-opaque markers and X-rays of the abdomen before and after fascial closure; avoiding a change of surgical team/scrub nurse in between the procedure; avoiding communication errors and implementing the WHO Surgical Safety Check List in all surgical procedures are some of the proven methods for prevention of a retained foreign body.


Headache is a common cause of emergency department (ED) visits. Migraine is a prevalent neurological disorder that is encountered by emergency physicians in day-to-day practice. However, patients with a known history of migraines should be carefully evaluated when presenting with headaches and serious pathologies of headache should be ruled out. We report the case of a 43-year-old woman, with a known history of classic migraine, who presented to the ED with a severe headache. She described the headache as persistent generalized pain. The headache was worse on awakening and bending. The headache did not improve with the use of oral sumatriptan. She reported that the current episode of headache is more severe than her usual migraine headaches. The patient underwent a cranial CT scan which demonstrated a homogenously hyperdense well-defined round lesion located in the midline at the approximate location of the foramen of Monro with prominent lateral ventricles, conferring the diagnosis of the colloid cyst. The patient underwent a right craniotomy with resection of the cyst using the transcallosal approach. Recognition of this important diagnosis is crucial to prevent serious neurological complications by having timely management. Headache is one of the most common presentations to the emergency department (ED). It accounts for up to 4% of all emergency visits . The preMigraine is a prevalent neurological disorder that is encountered by emergency physicians in day-to-day practice. It is characterized by recurrent attacks of headaches. However, patients with a known history of migraines should be carefully evaluated when presenting with headaches and serious pathologies of headache should be ruled out. We present the case of a middle-aged woman, with a history of migraines, who presented with a severe headache due to a colloid cyst, which is a potentially fatal condition if its management was delayed or missed.We present the case of a 43-year-old woman, with a known history of classic migraine for the last 20 years, who presented to the ED with a severe headache. Her pain started four days prior to her presentation and progressively worsened in severity. She described the headache as persistent generalized pain, compared with the pulsatile nature of her previous migraine headaches. The headache was worse on awakening and bending. Unlike her usual episodes of migraine, the headache did not improve with the use of oral sumatriptan. She scored the pain as eight out of 10 in severity and it was awakening the patient from sleep. The headache was associated with nausea and recurrent episodes of vomiting. There was no history of preceding head trauma. The patient did not report any history of photophobia, fever, weakness, or loss of consciousness. The patient visited several outpatient clinics who managed her as having tension headache and offered symptomatic treatment only.She reported that the current episode of headache was different in character and severe than her usual episodes of migraine headaches. Of note, the patient was using sumatriptan for the last eight years. Her past medical history is not significant for any health condition other than migraine. She has not undergone any surgical operation in the past. Her family history is significant for classic migraines in her mother and sister. She is a non-smoker and does not drink alcohol.Upon examination, the patient looked in pain and distress. She was drowsy but was fully oriented. Her vital signs on presentation were as follows: pulse rate of 114 bpm, blood pressure of 132/78 mmHg, respiratory rate of 18 bpm, temperature of 36.7℃, and oxygen saturation of 99% on room air. Neurological examination revealed grossly intact cranial nerves, no focal motor or sensory deficits, and normal cerebellar examinations. Funduscopic examination showed normal findings. The cardiorespiratory examination was unremarkable. There was no neck stiffness and the meningeal signs were not present. Her laboratory findings revealed a hemoglobin level of 13.9 g/dL, a white blood cell count of 6,500/µL, and a platelet count of 420,000/µL. Other biochemical investigations, including hepatic and renal profiles, were within the normal range.In view of the aforementioned clinical features, the patient underwent CT of the brain to rule out any space-occupying lesion. The CT demonstrated a homogenously hyperdense well-defined round lesion located in the midline at the approximate location of the foramen of Monro with prominent lateral ventricles Figure . This fiThe patient was prepared for an emergency neurosurgical operation for excision of the cyst. She had a right craniotomy with resection of the cyst using the transcallosal approach. The patient tolerated the procedure well without any complications. Histopathological examination of the resected cyst showed a ciliated pseudostratified epithelium lining of the cyst Figure . She hadWe presented the case of a middle-aged woman, with a history of migraines, who presented with progressive headache that was diagnosed as having colloid cyst near the foramen of Monro. The colloid cyst is a rare benign tumor accounting for less than 2% of all primary brain tumors [The majority of colloid cysts are asymptomatic and are detected incidentally on imaging. The clinical manifestations of colloid cysts include headache, visual disturbances, vertigo, and even sudden death. Such presentations are related to the obstruction of the flow of the cerebrospinal fluid which precipitates the development of hydrocephalus . In the The pathogenesis of colloid cysts remains unclear. It is postulated that the cyst could be a paraphysis remnant, which is an embryonic structure at the diencephalon located between the telencephalon hemispheres. However, this theory does not provide an explanation for the colloid cysts which may be found in the frontal lobe and cerebellum .The diagnosis of colloid cysts can readily be made by CT head or MRI brain. On CT imaging, the cyst appears as a rounded hyperdense lesion near the foramen of Monro . The cysThe management approaches for colloid cyst excision include craniotomy using transcallosal or transcortical approaches, endoscopic removal, and stereotactic aspiration. The prognosis is good and the recurrence is very rare -4.A colloid cyst is a rare benign primary brain tumor. Recognition of this important diagnosis is crucial to prevent serious neurological complications by having timely management. Physicians should have a high index of suspicion for secondary causes of headaches in patients with a migraine so that they do not falsely attribute all the episodes of a headache to migraine.


Tyrosine kinase inhibitors (TKIs) are nowadays a valuable treatment of desmoid tumors, a rare mesenchymal neoplasm. Although many side effects of imatinib and pazopanib, commonly or rarely occurring, have been described, reactional lymphadenopathy has not yet been reported. In this publication, we report two cases of patients with desmoid tumors, treated with pazopanib and imatinib, who developed reactional lymphadenopathy. As this side effect is presented as a newly formed mass, it can result in new diagnostic questions and added imaging tests and can even lead to discontinuation of the treatment. This report may help the clinicians facing similar problems adopt a “watch and wait” approach. Multitargeted TKIs of vascular endothelial growth factor receptors are effective therapy for DT.We report on two patients with reactional adenopathy induced by imatinib or pazopanib, mimicking tumor progression or other primary tumors.Desmoid tumors (DTs) are mesenchymal neoplasms considered to be of intermediate malignancy due to their non-metastasizing but locally invasive behavior –3. Their®), a synthetic indazolylpyrimidine (®), a 2-phenylamino-pyrimidine, is also a multitargeted TKI, and it aims at PEGFR, SCFR (c-KIT gene product), and BCR-ABL fusion protein (Tyrosine kinase inhibitors (TKIs) are orally administered anticancer drugs that inhibit tyrosine kinase proteins, a class of proteins that participate in various cellular processes . Pazopanrimidine , is a smrimidine –9. Commorimidine . Imatini protein , 11. Com protein .We are reporting the case of two patients with DTs treated with pazopanib in our department who presented reactionary lymphadenopathy after imatinib or pazopanib administration. The lymphadenopathy occurred as a new clinically palpable mass next to the known desmoid primary. MRI scans as well as biopsies were needed in order to exclude other possible diagnoses.Our first patient is female, aged 20 (born 2001), with no previous medical history. She was diagnosed with a right retroscapular DT located between the clavicle and the scapula with a p.T41A mutation of CTNNB1 gene (beta-catenin). She underwent surgical resection on November 2, 2011, and on December 6, 2012, a local recurrence was confirmed by biopsy. Due to rapidly symptomatic progression, chemotherapy according to the POG 9650 protocol (vinblastine i.v. and methotrexate i.v.) was introduced with partial remission of the disease. Four years later, on January 3, 2018, a new tumor progression appeared associated with local pain, and treatment by imatinib was started without any clinical and radiological benefit after 3 months.In this context, treatment was switched for pazopanib on August 12, 2018, at an initial dose of 800 mg qd but a dose reduction to 600 mg qd due to abdominal pain. An MRI in November 2019 revealed the appearance of a nodular lesion, located between the teres minor and infraspinatus muscles, misdiagnosed as a new DT lesion. In March 2020, pazopanib treatment was stopped due to persistent abdominal pain. A follow-up MRI of the shoulder on December 3, 2020, revealed a tendency for progression of previous smaller lymph nodes, especially of the right lower axillary lymph nodes, compared to the two previous examinations as well as an increase of the nodular lesion. At that time and because of the dubious nature of this nodular lesion, we decided to do further investigations to rule out a possible disease progression. On January 14, 2021, the patient underwent a percutaneous biopsy and cryoablation of the right retroscapular polylobed target lesion with the question of a possible disease progression. Pathological examination of the retroscapular mass showed fragments of a lymph node of preserved architecture and with slight sinus histiocytosis; no sign of malignancy within sampling limits was found, proving thus that it was only reactionary lymphadenopathy Figure 1Our second patient is female, aged 45 (born in 1976), known for obesity, obstructive sleep apnea syndrome, and moderate depressive episodes. She was diagnosed on November 4, 2020, with a sporadic rapidly increased right laterocervical DT of 4 × 10 × 5.8 cm with the presence of a c.121A>G (p.Thr41Ala) mutation in exon 3 of CTNNB1 gene.Pazopanib treatment was introduced on December 4, 2020, at a daily dose of 800 mg qd. By the end of December 2020, she presented B symptoms (night sweats and fever); and at the clinical exam, we noted the appearance of palpable cervical lymph nodes on the right side. A PET-CT scan was demanded with the question of a possible second primary tumor (low-grade lymphoma?). It was performed on January 25, 2021, and it showed the known laterocervical mass as well as discrete hypermetabolism of peri-centimetric lymph node formations of the cervical area. Unable to exclude the presence of a low-grade lymphoma based on radiological imaging, we decided to perform a surgical biopsy. Our main motivation was the clinical image of the patient who was presenting persistent B symptoms. The surgical excision of a cervical lymph node  took place on February 22, 2021, and the pathological examination revealed benign lymphadenopathy associating follicular and paracortical lymphoid hyperplasia, sinus histiocytosis, moderate polytypic plasmacytosis, and hemorrhagic lysis in places. No metastatic tumor infiltration was found. A follow-up MRI on April 6, 2021, revealed a discrete decrease in the size of the primary tumor but a marked decrease in signal intensity and enhancement as well as a decrease in dimensions of the cervical lymph nodes Figure 3DTs is a rare disease , 2, 13 aWe have summarized above the most common side effects of pazopanib and imatinib, some of which can result in dose reduction or even discontinuation of the treatment. There are also reports for more rare complications, such as pancreatitis, posterior reversible encephalopathy, or periosteal reaction .As far as we know, lymphadenopathy has not been reported as pazopanib- or imatinib-induced reaction. As mentioned above, reactional lymphadenopathy progressed in our two patients after the introduction of TKI treatment and was presented as a new nearby mass. This mass could at first be a second primary tumor, a progression of the known DT, a low-grade lymphoma , and of course reactional lymphadenopathy since DTs do not spread to lymph nodes. This can perhaps lead to new diagnostic questions or even lead to treatment discontinuation.In conclusion, TKIs are a valuable medication for DTs, and possible lymphadenopathy induced by the medication could lead to extra cost, operational dangers for the patient, and even treatment changes. We think that this two-case report can help clinicians facing similar problems to be less concerned with this kind of reaction and perhaps choose monitoring versus interventional acts. Nevertheless, more data are needed not only to validate the reactional lymphadenopathy as a side effect of TKI but also to determine the chance of its occurring and the possible relation to DT treatment.The original contributions presented in the study are included in the article/supplementary material. Further inquiries can be directed to the corresponding author.The studies involving human participants were reviewed and approved by CHUV. The patients/participants provided their written informed consent to participate in this study.ADi contributed to the conception and design of the study. SP wrote the first draft of the manuscript. PK wrote sections of the manuscript. All authors contributed to manuscript revision and read 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.


Sacrococcygeal myxopapillary ependymoma (MPE) is an uncommon type I glial tumor detected most frequently in the lumbosacral area of adolescents and children. It is usually presented as an intradural ependymal tumor that originates from the filum terminale and other locations within the ventricular system along the craniospinal axis. In rare cases, however, MPE may develop as a primary subcutaneous tumor in the sacrococcygeal area. Tumors can also appear as a dorsal sacrococcygeal growth or subcutaneous nodule. In this case report, we describe a rare case presenting as a subcutaneous sacrococcygeal mass in an elderly female that was subsequently resected and confirmed as subcutaneous MPE. The current standard treatment for MPE is maximal surgical resection with or without postoperative radiotherapy based on the locoregional extent and histological grading. However, there is limited evidence that radiotherapy for oligometastatic foci improves longevity or extends the time to recurrence. In addition to this case report, we provide a comprehensive review of similar cases and case series in the medical literature. Prospective studies evaluating the efficacy of resection and/or radiotherapy are required for improved management of extradural MPE. BackgroundEpendymomas are slowly growing glial cancers of the central nervous system (CNS) . While tExtraspinal ependymomas arising in the sacrococcygeal region are also observed, as either presacral or postsacral masses. While rare, these show a stronger propensity to spread systemically through lymphovascular dissemination ,4,5. PreThe grade and location of an ependymoma determine its management. Metastasis occurs more frequently 18%) in the very uncommon posterior extraspinal ependymomas, but rarely in the anterior extrathecal  ependymomas (7%). Metastatic sacrococcygeal MPE deposits have been observed in regional lymph nodes, liver, lungs, and bone % in the ,9-11. HeCase report2. On examination, the swelling was soft and had a baggy feeling on palpation. Despite its size, the mass was painless and associated with no neurosensory deficits. CT scan revealed a large, well-defined, hyperintense lesion around 13 × 10 cm2 in size over the sacrococcygeal region in the subcutaneous plane. The anterior aspect abutted the sacrum, coccyx, vertebral bone but without invasion, while the inferior aspect compressed the bilateral ischiorectal fossa fat pad. There was no extension into the anal canal. The lesion had a heterogeneous enhancement on post-contrast images but with multiple non-enhancing areas  in the conus medullaris or the filum terminale . The cocSitesAs mentioned, MPEs mainly affect the caudal spinal cord (cauda equina and conus medullaris). Extraspinal  ependymomas are thought to arise from heterotopic ependymal cell rests such as those found in the coccygeal region Figure  13]. Po. Po13]. Postsacral extraspinal ependymomas can also arise following surgical excision, from distant metastases, or from the direct expansion of a primary CNS tumor , such asAge of presentationSubcutaneous sacrococcygeal MPEs develop gradually and hence are usually large by clinical presentation. In one series, the average age at presentation was 17 years but ranged widely from 10 months to 47 years . This agRadiographic findingsThe imaging features of MPEs are not always unique . On radiMRI also frequently reveals a lobular mass that appears hypo- or isointense to muscle on T1-weighted (T1W) images and hyperintense on T2-weighted (T2W) images, with scattered hypointense fibrovascular septa and covered by a hypointense fibrous capsule. On T1W images, MPEs show homogeneous low signal strength within the mass, while intravenous gadolinium administration results in the appearance of focal nodules at the periphery with streaks of enhancement extending through most of the mass or in stronger homogeneous enhancement. On T2W images, MPEs present a strong signal (with intensity slightly lower than adipose tissue) inside a well-circumscribed mass. The majority of MPEs are circumscribed, but some may have invaded the sacral bone or adjacent soft tissue by a presentation .This heterogeneous contrast enhancement on T2W images is due to the association of the perivascular mucinous stroma with fibrous tissue. The mass frequently also has a peripheral rim of low signal due to local deposition of hemosiderin. In addition, necrosis and hemorrhagic areas may appear, and such lesions are more likely to be cancerous . SubcutaClassificationIn light of frequent recurrences, MPE has been upgraded to WHO grade 2 as per the WHO classification of tumors of the nervous system . Over 15-35% of MPEs can spread hematogenously to the pleura, lungs, local subcutaneous soft tissues, and bone .PathogenesisThe ependymal cell rests found in the filum terminale are thought to be the primary source of MPEs. Light microscopic findings reveal that aggregation of myxoid content in perivascular spaces distorts the typical ependymal architecture. Direct release of myxoid material by tumor cells and vascular leakage through endothelial fenestrations have been suggested as potential pathways for deposition . HoweverGross structure of MPEsThe pathologic presentations of 32 first subcutaneous sacrococcygeal MPE cases were reviewed by Helwig and Stern . The aveMacroscopic appearanceMacroscopically, cauda equina MPEs appear as enlarged, sausage-shaped tumors with a smooth lobulated surface, ranging in size from 10 cm to more than 40 cm in the largest dimension . Large tHistopathologyThe word "myxopapillary" refers to the proclivity of these neoplasms to produce mucin and form papillae as a result of arborizing vasculature, thereby markedly altering the architecture of the filum terminale .On histological sections, MPEs are distinguished by a dense, slender, micropapillary bundle covered with cuboidal to elongated tumor cells organized in papillary structures surrounding hyalinized fibrovascular cores. Cell nuclei are usually rounded, with fragile, transparent Figure  chromatiImmunohistochemistryCells are typically immunopositive for vimentin, GFAP, S-100 protein, and immunohistochemical cancer markers, while cytokeratin immunoreactivity is relatively uncommon Figure  27]..27].Electron microscopyCilia, complex interdigitations, and abundant basement membrane material have been observed under electron microscopy. In addition, microtubule aggregation within rough endoplasmic reticulum complexes is a distinguishing feature of some cases . OccasioMolecular pathology and cytogeneticsThere have been only a few reported cases of MPE with distinct genetic and cytogenetic abnormalities. In one such case, chromosome 1p rearrangement was noted. In another, tumor tissue sequencing revealed truncation of CTNNA1, which encodes the cadherin family cytoskeleton-associated protein alpha-catenin. This truncation undermines tumor suppression in epithelial tumors   and leadMolecular characterizationMolecular characterization has revealed several MPE subtypes, and so may facilitate improved clinical management. Histopathologic criteria have been used to classify ependymomas into grade I MPE (subependymoma), grade II or III ependymoma, RELA fusion-positive grade II ependymoma, and grade III anaplastic ependymoma . A recenThe highest frequency of genomic variation among ependymomas, specifically, depletion of 22q, has been observed in SP-EPN tumors  . HoweverApproach to a Case of Subcutaneous EpendymomaPathologists working with sacrococcygeal surgical specimens should be aware that these neoplasms may emerge from ependymal cells remaining within the coccygeal medullary vestige and manifest as subcutaneous masses of varying size. As a result, diagnosing these rare tumors necessitates a high clinical index of skepticism[A12]. Furthermore, when diagnosing these lesions in excision biopsies, fine-needle aspiration cytopathology (FNAC) samples, and core biopsies, a suitable histopathologic diagnostic procedure must be used. The clinical strategy for diagnosis of any sacrococcygeal mass should start with an examination of associated neurological defects and somatic symptoms. Sphincter dysfunction, lower back pain, and fatigue in the lower limbs are all common symptoms of filum terminale and cauda equine tumors. Furthermore, radiologic and clinical evidence of intraspinal contact suggests possible spinal dysraphism, which must be ruled out. If there are no neurological deficits, SE should be added to the list of possible diagnoses.Differentiating Subcutaneous Ependymoma From Ependymal Cell RestsThe most important consideration when verifying an ependymomatous lesion is to distinguish between neoplastic ependymoma and remaining exuberant ependymal cell rests. Careful consideration of diagnostic factors can help distinguish between these growths Table .Myxopapillary ependymal cells remaining in the subcutaneous tissue are benign neuroglial heterotopias originating from the filum terminale. They may appear as small masses or dimples, so this structure should be considered when diagnosing presacral acrochordons. Neurologic imaging must be conducted before biopsy when a mass appears in the centerline of the sacral region to assess CNS involvement. It is also vital to differentiate between ER and SME based on histology . A myxopCytomorphologic Approach to a Case of Subcutaneous EpendymomaThe FNAC of MPE is distinguished by an excess of mucinous material (myxomatous as the name suggests). However, mucinous metastases , bone tumors, and myxomatous primary soft tissue tumors  are linked to the sacrum and parasacral tissues. These lesions constitute a difficult differential diagnosis list due to variable cytomorphologic and histologic characteristics within the type and shared features between types, particularly the defined myxomatous or mucinous history ,33. In aTinctorial qualities of the background extracellular substance and architectural features often allow accurate diagnosis. When cytomorphologic characteristics are considered appropriately, MPE can be identified and other closely related lesions ruled out. The most helpful features for distinguishing neoplasms within the differential diagnosis list are goblet cells, physaliphorous cells, rosette-like structures, round stromal globules, cytoplasmic processes, signet ring cells, and chondroid fragments with lacunae . The preHistomorphologic Approaches for Diagnosis of the Current Ependymoma CaseThe histomorphologic approach to subcutaneous ependymoma diagnosis requires the elimination of other morphologically similar neoplasms. These include a group of neoplasms with villopapillary structure Table  37,38],,38,37,38Myxopapillary ependymoma, chordoma, adenocarcinoma, and myxoid chondrosarcoma can be distinguished using immunohistochemistry. However, immunoexpression levels of GFAP, cytokeratin (CK), S-100, and epithelial membrane antigen , human chorionic gonadotropin (HCG), and lactate dehydrogenase (LDH).However, there are a few instances where the growth pattern of MPEs is more solid, resulting in perivascular epithelioid cell aggregates showing cytoplasmic clearing and occasional cribriform glandular spaces. In these lesions, the vascular centers of papillae critical for diagnosis are seen only focally ,18 or noClinicopathologic correlationSpinal MPEs generally show a propensity for local recurrence and sluggish progressive growth. However, intracranial multifocality, local spinal dissemination, and extraneural metastasis have been reported. Metastasis to the bone, liver, regional lymph nodes, and lungs occurs in approximately 20% of subcutaneous sacrococcygeal MPE cases, often 10-20 years after the first appearance . Helwig In a study of 32 subcutaneous sacrococcygeal MPEs, Helwig and Stern found 17% cases of metastasis, with the majority occurring in the lung and inguinal lymph nodes . MetastaDifferential diagnosisExtradural MPE should be considered in the differential diagnosis of any patient with a slowly growing soft tissue mass in the pre-or postsacral area. Thus, any lesion, particularly near the spine midline, should warn the clinician of a possible underlying ependymal rest or spinal dysraphism as the etiology for abnormalities within the extradural tissue, particularly in young patients. Neurogenic tumor, sacrococcygeal teratoma, pilonidal cyst, lipoma, teratoma, chordoma, metastatic mucinous carcinoma, myxoid chondrosarcoma, metastatic carcinoid, and myxoid soft tissue tumors are among the differential diagnoses of subcutaneous MPE .Sacrococcygeal teratomas can appear as cystic or solid, but they are most frequently cystic. While a solid teratoma may be misdiagnosed as sacrococcygeal MPE, most sacrococcygeal teratomas are found in subcutaneous sacrococcygeal MPEs . CongeniTreatmentSurgery, Either En Bloc or Gross Total ResectionExtradural MPE has a propensity for spreading to the lungs, lymph nodes, and liver . As a rePostoperative ComplicationsA variety of postoperative complications have been reported across studies. For instance, Chakraborti et al.  reportedRole of RadiotherapyRetrospective evidence indicates that salvage radiotherapy (RT) may slow disease progression in cases of deteriorated capsular integrity following partial MPE resection . AdolescPatients who received GTR demonstrated a recurrence rate of approximately 10%, substantially lower than the 19% of patients receiving subtotal or piecemeal resection . FurtherAdjuvant RT is being used more often to prevent recurrence after resection. A statistically significant increase in local maintenance and progression-free survival (PFS) was noted in a study of 35 patients receiving RT and surgery compared to patients receiving only surgery . A separThis conclusion, however, is based on a series of retrospective studies conducted on spinal MPE patients rather than extradural MPE patients, and none of the aforementioned studies found a substantial difference in overall survival when adjuvant RT was added . In addiRole of ChemotherapyChemotherapy has been used in only a few MPE cases, including for young children with ependymomas, for treatment of tumors highly resistant to RT , and in Primary sacrococcygeal MPE is slowly growing and usually treatable, but may recur and spread to the lungs, lymph nodes, or spine. After the formation of metastases, the disease progresses slowly but steadily. Long-term monitoring is important because distant and locoregional recurrence can develop years after removal of the primary tumor. The current case-patient emphasizes the significance of careful case reporting for choosing the best possible treatment plan for patients with unusual or rare diseases. This case report and appended review also add to our body of knowledge on ependymomas by presenting evidence-based treatment and follow-up approaches. Following full or near-complete resection, MPEs have a positive prognosis, with greater than 10 years of survival. Subcutaneous sacrococcygeal MPEs still show substantial rates of recurrence after a long delay and a tendency for remote metastasis.


Bone choristoma is a benign tumor with normal histology and ectopic location. This paper aims to report a rare case of bone choristoma in the maxillary gingival location.The authors report a case of a 39-year-old woman, with a history of maxillary full edentulousness, who consulted for a slight pain evolved for about six months, triggered by movements on the lesion. Clinical examination found growth at the level of the right maxillary gingival alveolar ridge. Surgical biopsy was indicated and performed under local anesthesia. The histological examination of the excisional specimen concluded at a bone choristoma. The patient had a good evolution after the surgical removal.Choristoma is a rare and benign condition. The management is surgical. •Choristoma is a rare and benign condition.•Bone choristoma is a benign tumor that can occur regardless of the patient's age.•The symptomatology is variable depending on the location and size of the choristoma.•The treatment is surgical and the absence of recurrence is the rule. It is a heterotopy consisting of the localization of histologically normal tissue in an ectopic position . In mostThis case has been reported in line with the SCARE criteria .2A 39-year-old female with a history of maxillary edentulism consulted for moderate pain reported by the patient during masticatory movements. Clinical examination found a right maxillary gingival overgrowth that had been evolving for about six months . This gr3A bone choristoma is rare and corresponds to a proliferation of normal tissue in an ectopic position [ 1]. Bone choristoma was introduced by Krolls et al. He described a tumor-like growth of mature, normal lamellar bone occurring in the soft tissues of the oral cavity . Bone chThe CT scan and panoramic radiograph of the maxilla were normal. Histologically, a well-organized central lamellar bone surrounded by periosteum and embedded in fibroadenoma tissue has been described . Osteocy4Bone choristoma is a benign tumor that can occur regardless of the patient's age. The symptomatology is variable depending on the location and size of the choristoma. It is a heterotopia whose diagnosis is histological. The treatment is surgical and the absence of recurrence is the rule.5Not commissioned, externally peer reviewed.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.The authors declared that this study has received no financial support.Belem Ousmane: Corresponding author writing the paper.Rachid Aloua: writing the paper.Ouassime kerdoud: writing the paper.Savadogo sayouba: writing the paper.Faiçal Slimani: Correction of the paper.Authors of this article have no conflict or competing interests. All of the authors approved the final version of the manuscript.1. Name of the registry:2. Unique Identifying number or registration ID:3. Hyperlink to your specific registration (must be publicly accessible and will be checked):Belem Ousmane.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.


Dislocation of mandibular condyle into the middle cranial fossa is rare but diagnosis and treatment timely is very important due to significant complications. In this paper, we present a very rare case of asymptomatic intrusion of the mandibular condyle into the middle cranial fossa after orthognathic surgery in a 23 year old man from Iran.  The most common cause is traffic accident 5. According to our knowledge, this complication is not reported yet after orthognathic surgery. Intrusion of the mandibular condyle into the middle cranial fossa is a rare complication of mandibular trauma2, 4.Clinical examination, computerized tomography (CT)-scan and magnetic resonance imaging (MRI) can be useful for diagnosisIn this paper we present a case of asymptomatic intrusion of mandibular condyle into the middle cranial fossa after orthognathic surgery discovered accidentally during follow up of patient.We present a 23 yr old man with chief complaint of facial asymmetry. He had past medical history of left hemifacial lymphatic malformation that underwent surgery in childhood. During surgery buccal branch of left facial nerve was injured. After surgery gradually patient developed dentofacial deformity. After puberty patient underwent prolonged orthodontic treatment (pre and post-op), bimaxillary surgery, genioplasty, mandibular body and angle implants. In clinical examination his facial countur was not optimal and there was no mouth opening limitation and his occlusion was acceptable with slight deviation of the mandible to the left. Totally patient was asymptomatic. We requested maxillofacial CT-scan. CT-scan revealed intrusion of left mandibular condyle into middle cranial fossa . BecauseInformed consent was obtained from the patient. .2, 6-8. Intrusion of mandibular condyle in to middle cranial fossa is a rare form of condylar dislocation especially as a complication of orthognathic surgery. The most common cause of this injury is traffic accident5, 9. One of the factors that increases the risk of this complication is the shape and morphology of the glenoid fossa and head of mandibular condyle. Because the middle part of the roof of the glenoid fossa is thinner than the lateral parts, a small and round head of mandibular condyle can penetrate into middle cranial fossa more easily than the normal scroll-shaped condyle1, 7. Other predisposing factors for this type of dislocation include increased pneumatization of the temporal bone, lack of posterior occlusion or open mouth position when the impact occurs and the presence of a defect in the roof of the glenoid fossa structure2-4, 10. Mandibular condyle is located inside a cavity within the temporal bone called glenoid fossa. The anterior wall of the glenoid fossa is built by the articular eminence of squamous part of temporal bone and its posterior wall built by the tympanic plate of temporal bone which also forms the anterior wall of external acoustic meatus. The roof of the glenoid fossa is a thin and translucent bone. The anterior and posterior walls of this fossa are well developed compared to its roof In our case in fact we believe that there may have been a defect in the structure of the glenoid fossa that was related to the left hemifacial lymphatic malformation which weakened the glenoid fossa. 1, 3, 4, 6, 10. Because these symptoms are non-specific and can be seen in other mandibular condyle fracture we need to use other diagnostic tools such as CT-scan and MRI2, 4. However, the inferior part of the temporal lobe is a relatively silent area of the brain and epilepsy and other cerebral deficits are uncommon in this region but patient should be evaluated for neurosurgical complications such as cerebrospinal fluid leakage, extradural hematoma and meningitis because the roof of the glenoid fossa is floor of the cranial base and placement of the middle meningeal artery in the floor of the middle cranial fossa in close proximity of the penetration3, 4, 11.There are several clinical findings that help for diagnosis of this type of condylar dislocation such as: restriction of mandibular mobility, unstable occlusion, deviation of the jaw toward the side of injury, posterior open bite on the opposite side of injury, cerebrospinal leakage or external auditory canal hemorrhage, facial asymmetry, reduce height of ramus on the side of injury and empty glenoid fossa in examination of temporomandibular jointIn our case, the patient complained of facial asymmetry. At the time of referral there was no history of limitation in the mandibular mobility or trauma to the mandible. His occlusion was acceptable and he had no neurologic symptoms. After clinical and radiographic evaluation we concluded that the main reason for the facial asymmetry was the intrusion of the mandibular condyle into the middle cranial fossa although previous buccal nerve injury caused slight asymmetry. 1, 3, 9. We informed patient his main problem and after talking with him and his parents, we did not do any surgical intervention. We decided to follow him regularly because many years had passed since his orthognathic surgery and he was asymptomatic. In the similar studies, different treatments have been recommended according to the existing conditions such as osteotomy of the mandibular condyle and remaining of the condylar head in the middle cranial fossa, condylectomy through the craniotomy, closed reduction and open reduction with removal of the condylar head as well as reconstruction of the glenoid fossaAt the time of writing this paper, the patient had been followed for 2 years. At recent follow up appointment his maximum mouth opening was 40 mm with slight deviation of the mandible to the left. To our knowledge this is the first case of the asymptomatic intrusion of the mandibular condyle into the middle cranial fossa as a complication of orthognathic surgery that is reported.Although the prevalence of intrusion of mandibular condyle into middle cranial fossa as one of the complications of orthognathic surgery is very rare but it should be considered .The authors declare no conflict of interest.


Teaching Point: Aggressive peptic ulcer disease can be indistinguishable from malignancy on imaging, even presenting invasion of adjacent organs, which often implies histological characterization. The patient underwent a blood transfusion and upper digestive endoscopy (UDE), which revealed a large ulcer next to the antrum, with pearly bottom and congestive edges. Biopsies were done. The patient underwent abdominopelvic computed tomography (CT) after intravenous and oral contrasts, which revealed an irregular thickening of the antral/inferior region of the lesser gastric curvature , with a 1.6 × 1.9 cm gastric wall defect communicating to the liver . A large (>10 cm) hepatic collection was also identified, in continuity with the gastric lumen, predominantly containing liquid, multiple internal gas bubbles and oral contrast (asterisk on 2). These aspects were compatible with a large liver abscess due to penetration from a gastric ulcer. Absence of signs of peritoneal carcinomatosis and metastases favored benign etiology. The patient underwent subtotal gastrectomy with Billroth II reconstruction and liver abscess drainage. Pathological findings were of a benign peptic ulcer, with chronic atrophic antral gastritis and intestinal metaplasia, without evidence of dysplasia.A 72-year-old male presented to the emergency department due to rectorrhagia on the night before. He reported dark stools over the previous month. His past medical history was relevant for peptic ulcer disease (PUD) and atrioventricular block. Physical examination revealed normal vital signs and the rectal examination showed neither bleeding, fissure, nor hemorrhoidal pathology. On blood analysis, severe anemia (5.8 g/dL), leukocytosis  or mucosal enhancement discontinuity and luminal sacculation (direct signs) as in the present case. Acute complications of PUD such as hemorrhage and pneumoperitoneum (perforation) may equally be diagnosed on CT. Perforation may also lead to collection or fistulization/penetration to adjacent organs, which requires prompt diagnosis. PUD also increases the risk of gastric malignancies. Thus, although CT is not the first-line diagnostic technique, it has an essential role in the initial assessment and correct diagnosis, of both uncomplicated PUD and its complications [PUD remains a highly prevalent disease, affecting 10% of the world population. Its main causes are intake of nonsteroidal anti-inflammatory drugs and