Angiolipoma in the region of the hypopharynx-esophageal introitus is a rare occurrence. Surgical treatment was performed in the few cases reported in the literature. Endoscopic submucosal dissection (ESD) is a minimally invasive treatment for hypopharyngeal and esophageal lesions. Our objective was to evaluate the feasibility, safety, and efficacy of ESD for treatment of angiolipoma at the hypopharynx-esophageal introitus. The patients with submucosal tumors at the hypopharynx-esophageal introitus were diagnosed as angiolipoma by preoperative evaluation with endoscopy, endoscopic ultrasonography, and computed tomography (CT). The patients who were diagnosed with angiolipoma agreed to undergo endoscopic submucosal dissection. Under general anesthesia and endotracheal intubation, ESD was used to remove the lesions. Preoperative, intraoperative, and postoperative data were collected and analyzed to evaluate the feasibility, safety, and effectiveness of endoscopic submucosal dissection. From January 2013 to December 2018, 6 cases of angiolipoma were treated with ESD with a success rate of 100%. The average operation time was 107.0 ± 69.4 minutes. Intraoperative blood loss is the main risk. Endoscopic thermocoagulation successfully stopped bleeding in all cases. Pharyngeal pain and painful swallowing were the main clinical signs. There was no stricture at the hypopharynx-esophageal introitus after the operation. ESD treatment of angiolipoma at hypopharynx-esophageal introitus is feasible, safe, and effective. Angiolipoma is one of the most common benign soft tissue tumors. In a few cases, familial heredity was observed . It commIn total, 6 patients with angiolipoma located at the hypopharynx-esophageal introitus in patients were treated in the Department of Gastroenterology, the Fourth Affiliated Hospital of China Medical University between July 2013 and January 2018. The average age of the patients was 52.7 years (27-85 years). There were four males and two females. The main clinical manifestations were dysphagia in 4 cases and intermittent chest pain in 2 cases. Six patients failed to complete gastroscopy in the other hospitals. In one case, the gastroscopy failed to pass through the hypopharynx, while in 3 cases, the gastroscopy entered the esophageal introitus but could not progress. In the remaining 2 cases, the gastroscope was progressed to the middle third of the esophagus before the evaluation was terminated. Routine gastroscopy was successfully performed in these 6 patients in our outpatient department with the esophageal tumor duly characterized in each case. The tumor was considered to have originated from the hypopharynx. The mean tumor size was 5.63 cm (3.8-30 cm).The patients underwent preoperative gastroscopy, ultrasonic gastroscopy, and CT examination with a presumptive diagnosis of lipoma at hypopharynx-esophageal introitus made based on clinical experience. After analysis in each separate case, endoscopic mucosal dissection was selected for these patients. All patients were informed of the possible complications of the operation. Additionally, they were informed about the possibility of additional procedures to manage complications or postoperative pathologic findings.Preoperative instruments included Olympus CV 260sl, Olympus GIF Q260J, and Distal Attachment Cap .All ESD procedures were performed by the same surgeon (Zhao ZF). Patients underwent endotracheal intubation under intravenous general anesthesia.ESD: after submucosal injection of normal saline containing methylene blue and hyaluronic acid at the pedicle of the tumor, the mucosa and submucosa layers were gradually dissected with incision knife and the tumor was completely dissected. Intraoperative blood vessel bleeding was stopped by means of thermocoagulation forceps, and hemostasis of the dissected wound surface was achieved by thermocoagulation. All wounds were left open without suturing .In total, 6 patients with angiolipoma originated from the hypopharynx-esophageal introitus region were treated in our hospital, including 4 cases with the tumor pedicle at the hypopharynx  and 2 caWe initially performed radial scanning and linear endoscopic ultrasonography, all of which failed. Among them, 4 cases had severe pain when entering the esophageal introitus, so the examination was canceled. In the remaining 2 cases, entry through the esophageal introitus was challenging. All patients successfully completed routine gastroscopy combined with microprobe endoscopic ultrasonography (EUS). EUS  revealedAll the 6 cases underwent imaging by noncontrast CT . CT reveCombining the imaging and gastroscopy results, we concluded on the diagnosis of angiolipoma at the hypopharynx-esophageal introitus, and after much deliberation, chose to perform endoscopic submucosal dissection. Hemorrhage occurred in all 6 cases, mainly during injection and incision. During the operation, 2 cases suffered large blood vessel hemorrhage due to increased vascularity on the tumor surface. Thermocoagulation forceps-assisted hemostasis was successful, and the intraoperative blood loss was about 100 ml in these two cases. Two cases (case 1 and case 2) with large tumors were treated with a snare to cut the tumor into blocks. About one-fifth of the tumor was removed for examination. The repeated attempts to take out the resected tumor failed, and the attempt to push the whole tumor into the stomach also failed. Therefore, we chose to use incision knife and snare to cut the tumor into blocks and pushing the parts into the stomach, so that the tumor was exposed under the action of gastric acid and then corroded. The tumors were removed after segmental incision in 2 cases (cases 4 and 5). The whole tumors were completely removed in 2 cases (cases 3 and 6). Surgical duration was 50-240 minutes (107.0 ± 69.4 minutes). The wound was left open about 1-1.5 cm. All the tumors were resected successfully. During the reexamination using gastroscopy, the exposed tumors were excreted from the body; no residues were found in the stomach .Immunohistochemical staining was performed. As shown in The patients were observed for 1-week after ESD in hospital, then followed up at two months and one year after ESD. During the 1-week observation, we used the 0-10 Numeric Pain Rating Scale (NPRS) to measure the pain intensity—0: no pain, 1-3: mild pain, 4-6: moderate pain, and 7-10: severe pain. Two cases showed postoperative pharyngeal pain; a further two cases showed pain when drinking water; all pain levels were tolerable (pain scores < 3). One year after the operation, scar formation at the esophageal introitus was found by gastroscopy in six patients. . No striAngiolipoma is a distinct morphological variation of a lipoma, accounting for 5-7% of lipomas , 3. Its Angiolipoma occurring at the hypopharynx-esophageal introitus is extremely rare in clinical practice. A literature search of “hypopharynx-esophagus lipoma” and “hypopharyngeal lipoma” revealed 1 case of pharyngeal lipoma , 1 case Angiolipomas at the hypopharynx-esophageal introitus often deform the hypopharynx by traction and occupy the esophageal lumen. Gastroscopy is generally difficult. All six patients failed to complete gastroscopy in the other hospitals, the gastroscopy failed to pass through the hypopharynx in 1 case, the gastroscopy entered esophageal introitus but could not progress in 3 cases, and the gastroscopy arrived the middle third of the esophagus and terminated the examination in 2 cases. The routine gastroscopic examination in our hospital is also difficult, but it was successful in all 6 patients. All gastroscopes succeeded in entering the esophagus along the right side of the hypopharynx. The left side of hypopharynx is difficult to enter, and the right side of the hypopharynx is easy to enter, which is closely related to the distortion of the hypopharynx-esophageal introitus caused by the traction of the prolapsed tumor. Because the tumor completely occupies the esophageal cavity, it is difficult to progress in the esophagus, which is mainly manifested in a small gap, high resistance, and distortion of the esophagus. Distal attachment cap-assisted examination is helpful for the observation of the origin, pedicle range, and endoscopic observing of the tumor. The endoscopic manifestations of the tumors mainly include the following aspects: (1) The hypopharynx is pulled and shifted, and the hypopharynx mucosa was pulling the mucosal bridge, and with the traction directed towards the right side in all cases. (2) The traction change of esophageal introitus by the pedicle of the tumor was seen, the pedicles were located at the left and anterior walls of the esophagus, and the surface of the tumors showed a large number of dilated veins. (3) The esophageal lumen is filled with a tumor; the esophagus is enlarged; the gastroscopy can squeeze out the gap between the tumor and the esophageal wall. (4) The mucosa on the tumor surface is smooth and slightly yellowish-white, and the esophageal wall has no abnormal changes. (5) The tumors were with a firm texture and can be squeezed to slightly deform. (6) In the case that the tumor prolapsed into the stomach, the tumor surface was accompanied by rupture, ulceration, and bleeding, and coffee-like old bleeding was seen in the stomach cavity. The presurgical diagnosis and differential diagnosis of angiolipoma mainly rely on endoscopic manifestations, ultrasonic endoscopic changes, CT, and postoperative pathology. Because angiolipoma is rich in blood supplies and it is difficult to get the tissue from the biopsy at the mucosal surface, hence endoscopic biopsy is not recommended , so no bEUS can preoperatively judge the nature of the tumor by evaluating the components of the tumor, which is of great significance to diagnosis and treatment. The results of this case series demonstrated that radial scanning and linear endoscopic ultrasonography were difficult to perform. Among the 6 cases of unsuccessful radial scanning endoscopic ultrasonography, 4 cases were terminated due to severe pain, and it was difficult to enter the esophageal introitus in 2 cases. The main reason for the failure of endoscopy progression is that the deformation of hypopharynx due to traction and tumor occupying the esophageal lumen, which results in the difficulty for radial scanning and linear endoscopy entering the esophagus. The main cause of pain in patients during endoscopy progression is that the tumor pulls the tissue of esophageal introitus, and the endoscopy operation can significantly increase the pain caused by the downward traction of the tumor. Compared with radial scanning and linear endoscopic ultrasonography, the advantage of microprobe endoscopic ultrasonography is, the microprobe can be introduced using routine gastroscope, it can be used to examine the stenosis region due to its small size, not tending to cause pain and bleeding. All patients successfully completed routine gastroscopy combined with microprobe endoscopic ultrasonography; the internal echo signals of the tumors were effectively evaluated in all cases. However, microprobe EUS images were seriously attenuated and the image quality was low. This is mainly due to the internal tissue of the tumor being mainly fat, so the ultrasonic attenuation is obvious. Microprobe endoscopic ultrasonography often showed good mucosal layer, an irregular submucosal muscle-like area, and slightly hyperechoic areas.CT and multiplanar reconstruction (MPR) are important methods to evaluate the lesion and have the advantage of convenience and pain-free. CT scan of all cases showed that the density of the tumor was similar to that of fat, and the density of the tumor was heterogeneous and mixed with other ingredients. CT multiplanar reconstruction images can clearly show the size and extent of the lesions. The disadvantage of unenhanced CT is that it cannot distinguish the origin of the lesion. Contrast-enhanced CT was not performed in all six cases, so the role of contrast-enhanced CT in locating the origin of the lesions has not yet been clarified.Angiolipomas of the gastrointestinal tract were previously treated by complete resection and could be removed by endoscopy or surgery depending on the location and urgency of the lesion. Surgical excision is preferable to endoscopic excision because of the increased risk of bleeding and perforation for tumor with wider pedicle . Our casESD was successfully performed in all 6 patients, which proved that the procedure was safe and reliable. Hemorrhage occurred in all 6 cases, mainly during injection and incision. Hemostasis through forceps-assisted thermocoagulation was successful in all cases; no uncontrollable bleeding occurred. Postoperative wound pain was the main symptom without other postoperative complications. The advantages of ESD include safety, less injury, quick recovery, and no serious complications. The main reason for intraoperative blood loss is that angiolipoma is usually large and rich in blood vessels, especially the vessels at the pedicle. It is easy to damage blood vessels and cause hemorrhage. The best way to prevent and reduce bleeding is more submucosal injection, to increase the distance between blood vessels, to widen the tissue gap, to cut layer by layer, and to use more thermocoagulation forceps to prevent hemostasis. Postoperative pharyngeal pain is considered due to two aspects: First, the wound is located at the pharynx and esophageal introitus, thus often causes traction pain. Second, the wound was stimulated by the digestive juice secreted by the oropharyngeal cavity to produce irritating pain.Endoscopic mucosal dissection excision for angiolipoma at hypopharynx and esophagus is very difficult. It is mainly due to: First, the blood vessels of the lesions are rich and easy to bleed. Second, it is difficult to control the endoscopic field of vision. Third, the tumor is too big to be taken out. Fourth, endoscopic instruments are difficult to operate. The tumors were not taken out in 2 cases. Instead, they were removed, cut into pieces and moved to the stomach. The two patients whose tumors were not taken out had no postoperative complications such as intestinal obstruction. The remaining tumor parts were excreted through feces.The largest wound in these patients did not exceed 1/2 of the circumference of the esophageal introitus, which may be related to the absence of postoperative stricture.It can be seen from our treatment results that large angiolipoma is prone to have large blood vessels. It tends to cause intraoperative bleeding, difficulty in removing the angiolipoma, increased difficulty of operation, and prolonged operation duration. But there was no obvious difference in postoperative recovery. Therefore, early detection, diagnosis, and treatment are recommended to reduce the risk of endoscopic surgery.Angiolipoma occurring at the hypopharynx-esophageal introitus is a rare clinical disease, with intermittent chest pain and dysphagia as the main clinical manifestations. Gastroscopy, CT imaging, and microprobe endoscopic ultrasonography are the main methods of preoperative diagnosis. ESD can be an important, minimally invasive, safe, and effective treatment option for angiolipoma at hypopharynx-esophageal introitus.


Cavernous hemangiomas are one of the most common benign primary orbital lesions. These tumors are insidious in onset, slowly progressive and present more often in middle aged women. Multiple orbital cavernous hemangiomas are extremely rare, and only a few cases have been reported in the published literature.Here, we report the diagnosis and treatment of multiple cavernous hemangiomas in the right orbit of a female patient with impaired visual acuity and proptosis of the eye for more than 10 years.Magnetic resonance imaging of the orbit showed a giant and irregular soft mass filling the intraconal and extraconal space of the right orbit, compressing the right optic nerve. After tumor resection, histopathological examination confirmed the diagnosis of cavernous hemangioma.A lateral orbitotomy was performed and a total of 13 tumors were excised, with the largest tumor measuring approximately 2.5 × 3.0 cm.The visual acuity of the patient was preserved, with only a slightly dilated pupil of the right eye. The follow-up period was 6 months with no signs of recurrence.Multiple cavernous hemangiomas in the orbit is rare and should be excised surgically as soon as possible. Radiological examinations, such as computerized tomography (CT) and magnetic resonance imaging, have a fundamental role in its diagnosis. Most of the tumors are unilateral or solitary, and multiple or bilateral lesions within the orbit are rarely reported.,4 Here, we report a case of multiple cavernous hemangiomas in the right orbit of a middle-aged woman who had a history of proptosis for more than a decade and experienced an acute progression within 6 months.Cavernous hemangioma is the most common benign orbital tumor in adults, which includes approximately 10% of orbital tumors and occurs often in the intraconal space as well-circumscribed lesions.2A 47-year-old female patient was admitted to the hospital with a complaint of painless progressive proptosis of the right eye for more than 10 years and a significant decrease in visual acuity for 6 months. On physical examination, the best-corrected visual acuity (BCVA) for the right eye was 3/10 and 10/10 for the left eye. There was 10 mm of right axial proptosis. The upper eyelid of the right eye had mild prolapse, covering the upper corneal margin by approximately 3 mm. A 3.0 × 3.0 cm soft mass was visualized and palpated in the lower eyelid Fig. A. Histop3 In the present study, the patient diagnosed with orbital cavernous hemangioma had multiple tumors within the same orbit, with a total of 13 tumors of different sizes, which is seen very rarely in clinical practice.Cavernous hemangiomas are the most common benign primary tumor of the orbit, which are vascular malformations consisting of dilated blood vessels separated by fibrous tissue and most frequently found in middle-aged women.,3,5–17 Among those cases, one was that of bilateral intraorbital hemangiomas with two tumors in the right orbit and three tumors in the left orbit. Twelve patients were female, and three were male. The mean age was 41.9 years, ranging from 7 to 63 years. The right orbit was involved in 10 patients, and left orbit was involved in 5 patients. The average number of tumors excised with surgery was 5.3, with a maximum 15 tumors in 2 cases. There were 3 cases with recurrence of the tumor during the follow-up period: the first case reported 15 years after the complete excision of the tumors; the second case recurred 8 years after surgery. The third case took 6 year for the first recurrence and 24 years for the second relapse.To our knowledge, only 15 cases of multiple cavernous hemangiomas occurring within the same orbit have been reported (summarized in Table  reported that in 9 incomplete excised cases, 3 cases experienced recurrence. Orbital cavernous hemangioma expresses tumor growth factor bFGF, which can stimulate the growth of endothelial cells and vascular smooth muscle cells and may participate in the growth of tumor. Even after complete excision of the tumor, the secreted and remaining bFGF may still have the potential to stimulate the growth of cavernous hemangioma. In addition, the progesterone receptor is present in the orbital cavernous hemangioma, implying a hormone-dependent mechanism in tumor growth. Undeniably, we cannot exclude the remnants or small hidden tumors within the orbit, even after complete excision. The general therapeutic outcomes of the surgery were excellent for the majority of reported cases, and the recurrence rate was very low.In large case series, there were no reports of tumor recurrence if the lesions were excised completely. However, if the tumors were incompletely excised, recurrence may occur. Yan and Wu Interestingly, in all the reported cases, the symptoms were unilateral involving only the orbit harboring the larger mass. However, surgical management is the same option as for unilateral cavernous hemangioma and should be limited to symptomatic lesions.Multiple cavernous lesions can also present in a bilateral form. To date, 13 cases of bilateral cavernous hemangiomas have been reported.These patients usually had a long course of disease, and the tumor filled the orbital cavity, which compressed the optic nerve and extraocular muscle, resulting in irreversible visual impairment, suggesting that such patients should undergo surgical excision as soon as possible.Although extremely rare, cavernous hemangiomas should be considered in the differential diagnosis of patients with multiple orbital mass lesions. In the presence of multiple cavernous hemangiomas, systemic evaluation of the patients should be done to rule out multi-centric lesions. The tumors should be completely excised by surgery and a very long follow-up of these patients is recommended to monitor recurrence.We thank the patient for granting permission to publish this information.Conceptualization: Chaohua Deng, Weikun Hu.Resources: Weikun Hu.Supervision: Weikun Hu.Writing – original draft: Chaohua Deng, Weikun Hu.Writing – reviewing & editing: Chaohua Deng, Weikun Hu.


This case report describes a unique nasal mass that was difficult to diagnose clinically and histologically. The patient was a middle-aged man employed as a metalworker, and he presented with a unilateral nasal obstruction and a mass arising from the right middle meatus. After a series of investigations, he underwent right-sided sphenoethmoidectomy with excision of a nasal lesion. The surgical specimen presented a major diagnostic challenge for the pathologists and clinicians involved. A series of discussions amongst two different head and neck expert teams combined with detailed clinicopathological correlation resulted in a diagnosis of a granulomatous lesion or pseudotumour related to the ingestion of water-soluble cutting oils, or “Suds oil,” as they are more commonly called. Although occupational exposures to certain inhalants, such as wood dust and formaldehyde, are well-known risk factors for sinonasal lesions, here we present a rare association between a sinonasal lesion and another inhalant, Suds oil, that has not been previously reported in the literature. The paranasal sinuses and nasal cavity occupy a relatively small anatomical space. Lesions arising from these two regions are typically rare and represent a histologically diverse group of tumours .A number of occupational risk factors have been associated with sinonasal lesions, including exposure to wood, nickel, and formaldehyde inhalants . The preA 63-year-old man presented to the clinic with a two-year history of right-sided facial discomfort, medial canthal swelling, and nasal obstruction. There was no history of epistaxis or visual deficit, but he reported pain and persistent epiphora on the right side. His past medical history included ischaemic heart disease, hypertension, and diabetes. He had been taking regular analgesia for management of right-sided frontotemporal headaches. This patient was a nonsmoker and had worked as an engineer with open machines for construction of pins for 25 years.On physical examination, there was a medial canthal swelling. Flexible nasal endoscopy revealed a large mass arising from the right middle meatal opening and a clear nasal cavity on the contralateral side. His magnetic resonance imaging (MRI) scans showed thinning of the medial wall of the right orbit, with evidence of a discrete mass arising from the right ethmoid sinus, extending into the right nasal cavity with likely impingement on the nasolacrimal duct . SubsequThe resected specimen was sent for histological examination which was inconclusive in the first round. The case was sent for external review to a second specialist centre for head and neck pathology in London. The second report favoured a benign process as there was no cytological atypia or marked variation in cellularity and no inflammatory cell infiltrate. The report noted numerous foamy macrophages containing eosinophilic material of uncertain aetiology. Periodic acid-Schiff (PAS)/Alcian blue special stain was negative for mucin, but the stain highlighted the eosinophilic material . The repSix months after the determination of this aetiology, a follow-up was conducted and an MRI was performed which showed no lesion residuum or recurrence . The patSinonasal tumours are rare, with literature reporting rates of 0.2–1% of all neoplasms and 4-5% of those arising in the head and neck regions , 4. AlthThe actual incidence of benign sinonasal tumours is difficult to determine, as most of these tumours are asymptomatic . They caIn this case, the histology was unusual and difficult to diagnose even for specialist head and neck pathologists in tertiary referral centres. The pathologist described a fibromyxoid proliferation and a range of differentials offered—from benign nasal glial heterotopia, chordoid meningioma, and sinonasal myxoma/fibromyxoma to low-grade malignant sarcoma and chordoma. Special histochemical stains, including Gram, PAS, and Grocott, helped exclude an infective process. A wide panel of immunostains proved nonspecific. Initially, a benign mesenchymal neoplasm with some fibroblastic/myofibroblastic differentiation was the preferred diagnosis. The supplementary report by the external experts also favoured a benign process that was not consistent with schwannoma, as suggested on imaging. The histiocytic proliferation and associated eosinophilic material led to the suggestion that the lesion represented a granulomatous reaction or pseudotumour as the most likely diagnosis among the differentials. The clinicopathological correlation was essential to support the diagnosis and for patient management.Apart from its rare and challenging histological features, this case was associated with a potentially unrecognised occupational hazard. The association between sinonasal lesions and other occupational hazards, including exposure to wood dust and leather dust, has been widely documented , 7, 8. TSuds oil, also called “cutting” or “straight” oil, is a petroleum-based class of metal-working fluids. They are widely used in a variety of industrial metal-working operations to improve the surface integrity of the workpiece by lubricating and reducing the friction between the tool and metal .International Journal of Occupational Safety reports threefold incidence of allergic and irritant contact dermatitis in people who work closely with MWFs [There is strong evidence that metal-working fluids (MWFs), including Suds oils, can cause skin irritation. For instance, the ith MWFs . The othith MWFs . ClinicaConsidering the close daily contact with Suds oils in absence of any other historical causalities or risk factors, the consultant surgeon in charge of the case suggested this metal-working fluid as the most probable foreign body causing the granulomatous lesion described above.We hereby present, to the best of our knowledge, the first case report of frontoethmoid pseudotumour associated with metal-working fluids. We propose a case-control study in future which measures the exposure to this potential risk factor across patients with sinonasal lesions and to examine the relationship between MWFs and the disease in more detail.Sinonasal lesions are rare and histologically diverse. This case demonstrates the challenges often faced by clinicians in multidisciplinary teams managing rare sinonasal lesions. Diagnosis required numerous investigation modalities and surgical interventions and was eventually reached by consensus after careful clinicopathological correlation. This case highlights the importance of reporting challenging cases, reviewing the literature, and continuing research examining occupational hazards related to sinonasal neoplasms.


During pregnancy, the discovery of adnexal masses remains frequent. Such masses are mostly benign. Ovarian endometrioma is a rare etiology. The diagnosis may be difficult in some situations, such as decidualization. It may be asymptomatic or result in complications for which magnetic resonance imaging is needed.We describe an unusual case of decidualization of an ovarian endometrioma complicated by a sigmoid fistula during a 7-week, 1-day pregnancy in a Arabic patient aged 38 years who developed acute pelvic pain with fever. She had a medical history of unexplored secondary dysmenorrhea. The diagnosis was suspected on the basis of magnetic resonance imaging findings. The management was based on surgery, during which exploration revealed a mass at the expense of the left ovary being very adherent and fistulized to the sigmoid. We performed adnexectomy followed by digestive ostomy. The result of pathological study with immunohistochemistry led to a diagnosis of decidualization of an ovarian endometrioma altered by infection.Decidualization of an ovarian endometrioma can lead sometimes to unexpected complications. The decision to provide surgery must be made with caution without delaying treatment in the event of a deep suspicion of malignancy and/or complication. The particular and exceptional complication discovered in our patient is the fistulization to the sigmoid. Access to ultrasound at the beginning of pregnancy makes the association between pregnancy and adnexal mass an increasingly frequent situation . EndometOur patient was a 38-year-old pauciparous Arabic woman with a pregnancy of 7 weeks, 1 day gestation. She had consulted for acute pelvic pain of increasing intensity with fever without metrorrhagia and any other digestive or urinary signs. She had no specific medical or family history except a notion of unexplored secondary dysmenorrhea.On examination, the patient was hemodynamically stable and conscious, feverish at 38.6 °C, with tenderness of the left iliac fossa without defense or abdominal contracture. Her uterus was enlarged with tenderness and filling of the left lateral cul de sac.3, hemoglobin at 11.5 g/dl, platelet count at 241,000/mm3, a very high C-reactive protein (CRP) at 254 mg/l, and positive leukocyturia with a negative urine culture result.Her biological assessment objectified hyperleukocytosis at 15,200 elements/mmPelvic ultrasound described an enlarged uterus that was the seat of a toned gestational sac containing an embryo of 7 weeks, 1 day with cardiac activity and yolk sac. The patient’s right ovary was without visible abnormality, and her left ovary contained two contiguous cystic images of 68 and 35 mm with thickened wall in places not vascularized on the basis of Doppler imaging, with finely echogenic content without vegetation or endocystic septum with posterior strengthening of the echoes, without effusion. We mentioned the diagnosis of hemorrhagic corpus luteum or decidualized endometrioma, given the notion of dysmenorrhea.The patient received pain relievers for her pelvic pain and antibiotics. Due to the persistence of the symptomatology (pelvic pain and fever) and the increase of CRP (294 mg/l vs 254 mg/l) after 1 week of treatment, an ultrasound control was performed, which objectified a progressive intrauterine pregnancy with persistence on the left side, of the two cysts that appeared as heterogeneous echogenic images with a thickened wall in places, pseudovegetations not taking the Doppler, and the presence of a liquid and thin effusion in the cul de sac of the pouch of Douglas  was performed to complete the exploration , with the following findings: two cystic hemorrhagic formations of the left ovary in T1 and T2 hyperintensity of 87 mm × 78 mm and 25 mm × 25 mm with fluid levels without endocystic vegetation and increasing at the periphery after injection of gadolinium. The largest cyst was in intimate contact with the sigmoid with a possible rupture of its wall at this level Figs. . PresencOn exploration, discovery was of a minimal serohematic effusion, a uterus at 8 weeks of gestation, and the right appendix without particularity. A mass was present at the expense of the left ovary, smooth-walled, about 10 cm and adherent to the left fallopian tube, the posterior surface of the uterus, and the sigmoid. This mass was very adherent and fistulized to the sigmoid, and there had been an accidental rupture during adhesiolysis and outpouring of a chocolate liquid. We performed a left adnexectomy and resection of the sigmoid by removing the fistula and then placing a Hartmann-type ostomy. The cytologic specimen returned in favor of an inflammatory hemorrhagic fluid without any tumor cell. Histology revealed that the cystic wall was fibrous with large foci of suppuration on the periphery. The colic wall had an ulcerated epithelium in places with broad suppurative areas at the level of the chorion. Immunohistochemistry highlighted glandular structures at the level of the intestinal wall that were well underlined by the anticytokeratin antibody and expressed estrogen and progesterone receptors with an expression of CD10 around the glands. Moreover, tumor cells were absent. Thus, the final histological study concluded the diagnosis of a decidualized and reshaped endometriotic cyst.The postoperative follow-up and monitoring of the pregnancy were simple with full-term vaginal delivery without incident. The restoration of intestinal continuity was performed by laparotomy 6 weeks after the delivery.The discovery of an adnexal mass during pregnancy poses a problem of etiological diagnosis and often leads wrongly to surgical management . Only 1–In ultrasound, the diagnosis of decidualization is all the more difficult because the image sometimes leads to confusion with a malignant tumor, regardless of the digestive, ovarian , or bladIn our patient’s case, ultrasound had objectified two finely echogenic cystic images with a wall thickened in places but not taking the Doppler and then at the control imaging, 1 week later, the appearance of an intracystic fluid level evoking hemorrhage associated with pseudovegetations.In patients with known endometriosis, the diagnostic orientation is quickly established. Otherwise, it is the bilaterality, the multifocality, and/or the presence of peritoneal lesions that will evoke the diagnosis .However, in certain cases, in particular those with complications or doubt about the malignancy, MRI is necessary, which was the case in our patient. It has been reported that these situations must always have an MRI performed to better guide the diagnosis , 7.Even if it was preferred previously from the second trimester, MRI is now authorized by some American and European authors, regardless of the patient’s gestation, if the maternal benefit is significant .The signal characteristics are those of an endometrioma with vegetation-like formations on the wall in T1 hyposignal and characteristic T2 hypersignal. The gadolinium injection will not make the difference between a borderline tumor or a decidualization [In our patient’s case, MRI was performed with gadolinium injection and objectified two cystic formations of the left ovary in hyperintensity T1 and T2. We did not objectify a vegetative formation on the cystic wall, but it increased after injection of gadolinium. In addition, one of the cysts was in close contact with the sigmoid with suspicion of a rupture of the wall at this level.Macroscopically, the endometrioma contains a thick chocolate liquid with adhesions to neighboring structures . Some auIn histology, although difficult to differentiate it from the hemorrhagic cyst, it is reported by many authors that the wall of the endometrioma is so laminated that it contains only a simple cubic epithelium surrounded by fibrosis .In our patient, the cyst actually sat at the level of the left ovary with a macroscopic aspect of endometrioma, and the final histology of the wall as well as immunohistochemistry were compatible with what the literature reports.We asked ourselves the question whether the ultrasound characteristics and those of MRI in our patient’s case have not been modified by alterations linked to infection. The pathologic examination answers this question by specifying the changes undergone by the cyst.Abstention is the attitude recommended by large numbers of authors, with regular ultrasound monitoring for ectopic deciduosis and masses whose characterization on imaging was sufficient for diagnosis –15. HoweApart from any complications and after delivery, there is a total regression of the changes in the wall of the endometrioma, which takes the appearance of a simple endometrioma , 5.A decidualized ovarian endometrioma can lead sometimes to unexpected complications. The decision to perform surgery must be made with caution without delaying treatment in the event of a strong suspicion of malignancy and/or a complication. The particular and exceptional complication discovered in our patient is the fistulization to the sigmoid, which explains the persistence of the clinical symptomatology. The infection was probably a cause of the ultrasound and MRI changes. Histology was key to the diagnosis despite changes due to the infection.


Abdominal epilepsy (AE) is a very rare diagnosis; it is considered to be a category of temporal lobe epilepsies and is more commonly a diagnosis of exclusion. Demographic presentation of AE is usually in the pediatric age group. However, there is recorded documentation of its occurrence even in adults. AE can present with unexplained, relentless, and recurrent gastrointestinal symptoms such as paroxysmal pain, nausea, bloating, and diarrhoea that improve with antiepileptic therapy. It is commonly linked with electroencephalography (EEG) changes in the temporal lobes along with symptoms that reflect the involvement of the central nervous system (CNS) such as altered consciousness, confusion, and lethargy. Due to the vague nature of these symptoms, there is a high chance of misdiagnosing a patient. We present the case of a 20-year-old man with AE who was misdiagnosed with psychogenic abdominal pain after undergoing multiple investigations with various hospital departments. Abdominal epilepsy (AE) is a rare syndrome, even rarer when seen in adults and presents with paroxysmal symptoms favouring an abdominal pathology that result from seizure activity . The speThe patient is a 20-year-old right-handed male. He presented to the General Surgery Clinic with recurrent episodes of severe "piercing" epigastric abdominal pain for six months. These episodes were associated with non-projectile vomiting followed by lethargy or loss of consciousness which lasted for a "few minutes" after each episode. They were neither associated with food intake nor had any aggravating or relieving factors. Each episode of abdominal pain lasted for 8-12 minutes and occurred three to five times per day. They were sudden in onset and resolved spontaneously. His birth, developmental, and past medical histories were unremarkable.General and local examinations of the abdomen and nervous system did not reveal any significant findings. He was prescribed analgesics and proton pump inhibitors and he was reviewed after one week. He did not report any significant improvement in his symptoms. His complete blood picture, stool for ova and parasites, 24 hr Holter ECG, ultrasound abdomen and CECT abdomen were unremarkable.Following this visit, he was put in for a gastroenterology referral; upper gastrointestinal (GI) endoscopy and barium swallow studies were found to be normal after which he was referred to the psychiatrist in view of psychogenic abdominal pain. There was no psychiatric intervention and he was referred to the neurology department for a second opinion.MRI brain was found to be normal. At this point, an epileptic aetiology was suspected. As in many Indian settings, the facilities for an ambulatory-EEG or video-EEG are either unavailable or beyond the affordability of most patients, a 30-minute awake EEG was ordered which revealed an abnormality in the form of repetitive sharp waves in the right temporal leads which were highly suggestive of an epileptogenic focus arising from the right temporal lobe as seen in Figure  Then, the conclusive diagnosis of AE was made and he was started on oxcarbazepine 450mg BID. He was scheduled with regular monthly follow-up visits and was compliant with his medication. A follow-up over a 12-month period showed a progressive decrease in seizure frequency and has been asymptomatic for the past four months. AE is a rare disorder and considering the vague nature of its symptoms, it is usually unconsidered and often misdiagnosed or missed from being diagnosed. It commonly occurs in the pediatric age group, but there is also documentation of its occurrence in adults . It is cAE has a variety of presenting symptoms. GI symptoms include paroxysmal pain, nausea, bloating, and diarrhoea, whereas the CNS symptoms can comprise of dizziness, lethargy, headache, confusion, syncope, and transient blindness ,7. As AEAlthough many mechanisms have been outlined, the cause of AE still remains unclear. We hypothesise that the Sylvian fissure and insular cortex lying right beneath it could be the origin of the seizure as they coincide with the locations of the abdomen on the Sensory homunculus. Additionally, the M2 portion of the middle cerebral artery courses through the Sylvian fissure . Any patRecurrent abdominal pain may also be seen in visceral hyperalgesia, peptic ulcer disease and abdominal migraine ,15. The Patients with AE may have the following possible variations in the EEGs:(1) Patients may have normal EEG patters in the inter-ictal periods and diagnosis must not be purely based on an EEG .(2) Extra-temporal origins of epileptic foci; secondary generalisation.(3) There are strong suggestions that EEG conducted after the first 24 hours after the epileptic episode can detect abnormalities to a greater extent .This case clearly exhibits how coming to a conclusive diagnosis of AE can be challenging and time-consuming as the symptomatology is indistinct and as the suspicion is low especially in the case of adults. As AE might often be overlooked or misdiagnosed, it should be taken into account in patients presenting with episodic, recurrent and paroxysmal GI complaints along with symptoms suggestive of CNS disturbance which do not improve with standard treatment modalities and after thoroughly excluding the more common causes in order to provide and an accurate and timely diagnosis. We must also be aware of the stigma that revolves around the diagnosis of a somatoform disorder in these scenarios and quickly consider getting an EEG, preferably a video-EEG if the facilities are available for such patients so that their symptoms are not falsely labelled as "psychogenic" or "functional". If the EEG findings are abnormal, the treatment involves the initiation of antiepileptic drugs with regular follow-up. 450mg oxcarbazepine BID was very effective in the management of AE for our patient.


Isolated gastric varices (IGV) are gastric varices in the absence of esophageal varices. IGV is one of the rare causes of gastrointestinal bleeding and an uncommon complication of pancreatic neuroendocrine tumors (PNET). The gold standard diagnostic tool of varices is esophagogastroduodenoscopy (EGD). IGV tend to bleed with lesser portal pressure compared to esophageal varices. Initial treatment is similar to the gastroesophageal varices. The intervention options include endoscopic, radiological, and surgical approach. Isolated gastric varices (IGV) can develop with or without the formation of esophageal varices. IGV occurs less frequently than esophageal varices; however, it has a higher risk of rebleeding, a rapid progression, and higher mortality . Portal A 71-year-old Hispanic woman with a past medical history of hypertension, hyperlipidemia, and diabetes mellitus presented to the hospital with an episode of hematemesis, generalized weakness, and epigastric abdominal pain for two days duration. Laboratory studies on admission were remarkable for normocytic anemia and a positive fecal occult blood test. The patient was admitted to the hospital and started on intravenous (IV) Protonix® (pantoprazole sodium) . Her hemoglobin continued to plummet, and as a result, she required multiple blood transfusions. The gastrointestinal (GI) service evaluated the patient. An upper endoscopy was performed which revealed a bleeding ulcer in the gastric fundus that was subsequently treated successfully with Hemospray®  therapy. Computed tomography (CT) of the abdomen/pelvis with contrast demonstrated an infiltrating mass involving the pancreatic tail and the spleen, causing splenic vein thrombosis and large gastric fundal varices Figures . A left hepatic lobe mass was also identified on imaging and the pathologic specimen from a percutaneous biopsy. The pathology sections showed focal benign hepatic parenchyma and nests of tumor cells with papillary architecture and nuclear pleomorphism. No mitotic figures were present in 10 high-power field count; tumor cells were positive for pankeratin, chromogranin, synaptophysin, and cluster of differentiation (CD)56. Alpha-1-antichymotrypsin (A-ACT) and CD10 were equivocal with high background staining, negative for arginase 1 and leukocyte common antigen (LCA), beta creatinine did not show nuclear staining, and Ki-67 was ~3%. These findings favored a well-differentiated neuroendocrine tumor. After an extensive multidisciplinary meeting, the patient did not opt for aggressive treatment. In the setting of substantial tumor burden, the patient underwent a short course of radiation therapy and was started on octreotide infusion to prevent any further upper GI bleeding. The patient was then discharged and a follow-up outpatient visit with the oncology service was arranged.Pancreatic neuroendocrine tumors (PNET) are sporadic and diagnostically challenging tumors with a wide variety of clinical presentations . IGV is Our patient presented with acute upper gastrointestinal bleeding secondary to metastatic PNET. An abdominal CT scan demonstrated an infiltrative mass involving the pancreatic tail and spleen, causing splenic vein thrombosis and regional portal hypertension, leading to type 1 IGV. In this case, IGV1 resulted from the redirection of blood flow through the short gastric veins . IGV1 coThe most common cause of IGV is portal hypertension secondary to liver cirrhosis after the obliteration of esophageal varices. Bleeding IGVs usually occur secondary to the isolated splenic vein obstruction due to pancreatic, gastric, colon, and, rarely, renal carcinoma.Esophagogastroduodenoscopy (EGD) is the gold standard for diagnosing gastroesophageal varices . In our Other options include transarterial embolization of the splenic artery, balloon-occluded retrograde transvenous obliteration (BRTO), or any of its variations, and splenic resection. The other variations of BRTO include coil-assisted retrograde transvenous obliteration (CARTO) and plug-assisted retrograde transvenous obliteration (PARTO) , 12. ThePNET presenting with bleeding IGV is uncommon. In patients presenting with IGV without risk factors or history of liver cirrhosis, it is crucial to consider other causes of regional portal hypertension, such as splenic vein thrombosis and obstruction, and the possibility of underlying malignancy.


Solid pseudopapillary tumors (SPTs) constitute 0.2 to 2.7% of non-endocrine primary tumors of the pancreas and comprise the majority (70%) of pediatric pancreatic neoplasms. These tumors are of unclear pathogenesis, low malignancy and favorable prognosis. Surgical resection offers an excellent chance for longterm survival, even in the presence of distant metastasis. The objective of this study is to review our experience in the management of SPT in a 12 years old girl at the pediatric hospital of the University hospital of Casablanca, in Morocco and provide an update on current management in pediatric population. Solid pseudopapillary tumors (SPTs) are rare neoplasms accounting for 2% to 3% of pancreatic tumors and 0.9% to 2.7% of exocrine pancreatic neoplasms . The firWe present our experience in the management of SPT in a 12 years old girl with no past medical history. The story of her illness began 2 months previously with abdominal pain without other associated signs. Abdominal examination revealed a palpable mass in the left hypochondrium. Ultrasound demonstrated a heterogeneous mass, having a contact with the tail of the pancreas and the spleen . On compPathologic results, combined with immunohistochemical study were consistent with the diagnosis of SPT , Table 1Epidemiology: SPTs comprise the majority of pediatric pancreatic neoplasms [eoplasms . It is aeoplasms ,7. The feoplasms . About 2eoplasms . The oriClinical features: Lee et al. demonstrated that SPTs in children presented with a palpable mass (60%), followed by abdominal pain (33.3%) [ (33.3%) . SPTs ar (33.3%) .Biologic features: pancreatic enzymes and common serum tumor markers  are consistently negative, as was seen in our patient [ patient ,5. There patient .Imaging features: SPTs may appear in any part of the pancreas [pancreas . Tumor lpancreas . SPTs arpancreas . Ultrasopancreas . The fibpancreas . The mospancreas . It helppancreas ,10. CT dpancreas . A delaypancreas . Enhancepancreas .et al. reported that solid pseudopapillary carcinoma may be differentiated from benign SPTs by its aggressive behavior, such as pancreatic duct dilation and vessel invasion, either with or without metastases. If such imaging features are present, aggressive surgical approach is mandatory and intensive follow-up is recommended [Fluid-debris levels are seen in up to 20% of tumors. Less than one-third demonstrate internal septations. Duct dilatation is uncommon even with large masses in the head of the pancreas because SPTs often compress adjacent structures rather than invading them . Solid pommended . Preoperommended ,8,9.Pathology: SPTs are a rare epithelial solid tumor of the pancreas that invariably develop significant cystic degeneration, acquiring the characteristic solid-cystic appearance.Gross appearance: macroscopically, SPTs are usually large, well circumscribed and have mixed solid whitish brown areas with foci of necrosis and hemorrhage and cystic areas containing necrotic fragments. As they increase in size, the areas of cystic-hemorrhagic degeneration grow until they get simular to a pseudocyst [eudocyst ,9.Histologic appearance: histologically, SPTs have a very characteristic appearance of solid areas composed with of poorly cohesive cells that form pseudopapillae around thin blood vessels. These polygonal epithelioid cells have medium-size, soft appearance, basophilic cytoplasms, large rounded or ovoid nuclei, with reinforced nuclear membrane and thin chromatin [hromatin ,9. FoamiImmunohistochemical: immunohistochemical study is very useful, revealing a diffuse positivity for certain histochemical markers like neuron-specific enolase (NSE), vimentin, CD-10 and béta-catenin, which is not specific [specific . α1-antispecific . Immunosspecific . Notoharspecific . Cells aspecific . Accordispecific . To datespecific .Treatment and prognosis: the current recommendations concur that children with pancreatic SPTs should undergo complete surgical resection that is dictated by tumor location and remains the treatment of choice [f choice . Distal f choice ,9. Invasf choice . Chemothf choice . Metastaf choice . There if choice . Althougf choice .Differential diagnosis: despite the technological advances, preoperative diagnosis is difficult because of the similarity of findings among other pancreatic tumors [c tumors . In chilc tumors ,6. The ic tumors .SPTs constitute the majority (70%) of pediatric pancreatic neoplasms. It is a rare exocrine pancreatic tumor that typically have the form of a well-encapsulated mass with solid and cystic components due to varying degrees of internal hemorrhage and necrosis. These tumors are of unclear pathogenesis, low malignancy and surgical resection offers an excellent chance for longterm survival, even in the presence of distant metastasis. Although surgical resection is generally curative, a close follow-up is advised in order to diagnose a local recurrence or distant metastasis and choose the proper therapeutic option for the patient.The authors declare no competing interests.


Lipomas are common benign mesenchymal tumors that appear in the head and neck region in approximately 25% of cases where they are noted. Lipomas of the airway region are exceedingly rare, accounting for less than 1% of airway obstruction tumors. Correlation of radiographic findings from cone beam computed tomography (CBCT), multi-detector computed tomography (MDCT), and magnetic resonance imaging (MRI) of a rare retropharyngeal lipoma has not been previously reported. CBCT studies acquired for implant and/or other diagnostic purposes may be the first line of detection as an incidental finding.A 66-year-old female presented for a pre-implant CBCT with no history of other complaints or signs/symptoms. CBCT imaging depicts a large, well-defined, low-attenuation/soft tissue density lesion with an undulating appearance extending from the posterior left pharyngeal wall and occluding two-thirds of the airway from C2 to C4. The lesion extends laterally into the left parapharyngeal space and inferiorly beyond the field of view of the study. Evidence of faint internal septations was noted. The patient was immediately referred for an ENT consult. Laryngoscopy, MRI, and contrast-enhanced MDCT imaging were conducted to determine the full extent and nature of the lesion, as well as to potentially plan for biopsy and/or surgical resection. Removal of the lesion was successful, and histopathologic evaluation confirmed lipoma. Periodic follow-up was recommended to monitor for possible recurrence.The slower growth pattern of some benign lesions may obscure any symptoms as changes the patient may normally notice take place over an extended period. Furthermore, soft tissue lesions and especially those in the posterior midline, such as in this case, may not be easily visible on routine panoramic imaging or clinical exam, allowing for substantially large growth before detection. While the soft tissue contrast of the CBCT volume is poor, enough information was present to establish an initial differential diagnosis and the need for more advanced imaging modalities. With the growing popularity and adoption of CBCT in maxillofacial imaging, a thorough understanding of the appearance of hard and soft tissue lesions, as well as a strong understanding of the baseline appearance of normal anatomy, is important to ensure no incidental pathoses go undiagnosed. Lipomas are relatively common benign mesenchymal tumors that are usually found in the torso and extremities. Lipomas in the head and neck region are more uncommon, accounting for 25% of all lipomas . LipomasAdditionally, the cases reported previously demonstrate the appearance of retropharyngeal lipomas in contrast-enhanced MDCT (multi-detector computed tomography) and MRI (magnetic resonance imaging) –7. HowevThis case report aims to demonstrate the characteristics of a retropharyngeal lipoma on CBCT, MDCT, and MRI and to demonstrate the importance of reading the CBCT examination in full so that appropriate intervention can be instituted. Furthermore, it highlights the rather insidious growth potential of tumors in the retropharyngeal space without pronounced symptoms. Additionally, this report serves to emphasize that a thorough understanding of the radiographic appearance of anatomic structures as seen on CBCT  is imperative to identifying critical incidental findings with potential serious consequences if left untreated such as an airway obstruction.A 66-year-old female was seen for potential implant treatment. Clinical examination revealed missing posterior mandibular teeth intra-orally with no significant extra-oral findings. The patient’s chief complaint was her partial edentulism which she wanted to address via implants. The previous panoramic radiograph taken 15 months prior was reviewed with no significant abnormalities noted  extending from the level of the dens to that of C4 with the following scan parameters: 120 kVp, 5 mA, voxel size 0.3 mm, scan time 8.9 s, and field of view 16 × 6 cm. The CBCT volume  will help prevent missed diagnosis or misdiagnoses of incidental pathoses. Additionally, recommendations for advanced complex imaging can be provided by an OMFR.Airway lesions require prompt diagnosis and attention in addition to subsequent coordination with multidisciplinary care providers to ensure appropriate patient management under the circumstances. Many lesions may have specific imaging characteristics that strongly indicate a diagnosis; however, advanced imaging and/or histopathologic evaluation via biopsy provides the most definitive diagnosis in addition to assisting with treatment planning. In addition, if the CBCT study does not include the entire lesion (similar to this case as noted in Fig.


Odontogenic pathological lesions can generally be divided into cysts and tumors. Calcifying odontogenic cyst (COC) is a non-aggressive odontogenic cyst, however, in a few occasions, COC can show excessive growth and destruction. This study describes an uncommon aggressive presentation of a large COC and discusses the treatment strategies used to achieve a favorable result.A 11-year-old child sought treatment for a painless growth on his mandible that caused facial asymmetry. On clinical and imaging examination, a large radiolucent lesion was observed in a panoramic x-ray involving the right side of the mandible. Radiopaque areas were observed within the lesion. Treatment comprised of an initial marsupialization followed by surgical removal of the lesion. Follow up extended for more than 10 years and no signs of recurrence were observed.Initial marsupialization was successfully employed in order to decrease the lesion, inducing bone formation, and reducing the risk of mandibular fracture and inferior alveolar nerve damage. Four months after marsupialization, the lesion substantially reduced its volume and enucleation with peripheral ostectomy was carried out. This approach allowed a safer surgical removal of the lesion and achieve an excellent result.Although some authors consider marsupialization as an unnecessary step that delays final treatment, when well indicated, it facilitates surgical treatment, promotes and preserves normal tissues, reduces the necessity of surgical reconstruction and costs, yet showing excellent results. AlthougOdontogenic lesions can achieve large dimensions , becauseMarsupialization was first described by Partsch as a treatment of cystic lesions . It consThis study aimed to show the importance of using conservative techniques in the surgical management of a large odontogenic cyst in a young patient. This case demonstrate that performing a few invasive procedures  can be a2An 11-year-old male patient presented to the oral diagnostic service of our institute, complaining of swelling inside the mouth on the right-hand side of the face, that showed evidence of a slow and asymptomatic growth.Physical examination showed facial asymmetry due to a painless growth on the right mandibular region A, which Panoramic radiography revealed a well-defined, homogeneous multilocular radiolucent lesion extending from the right lower first molar region to the coronoid process, with the presence of calcified areas and inclusion of tooth germ 47 A. On sagAn incisional biopsy A was perThe patient remained under clinical radiographic follow-up for four months, during that time, a progressive decrease in lesion size and new bone formation were observed. After this period, cyst reduction slowed and changes in lesion size and bone formation have established. At this moment, surgical enucleation with peripheral osteotomy was carried out under general anesthesia A–C. TootOne year after surgery, the patient had no sensorial deficits or any signs of recurrence of the lesion. There was also a major improvement in facial asymmetry A, with n3Initially described by Gorlin et al. in 1962 . Most COCOC lesions defined as tumors have a more locally destructive behavior, containing a proliferation of odontogenic epithelial islands, and their connective tissue has varying amounts of dentinoid. As a result, treatment modalities for these lesions involve a more aggressive surgical approach. Depending on the size of the lesion, this approach may be in the form of peripheral ostectomy or segmental resection .The lesion described in this current case had histological, radiographic and clinical characteristics of a cyst variant of COC. The gold standard treatment for COC is enucleation and curettage. However, because it was a large lesion and involved a young patient, a conservative approach using an initial marsupialization followed by total enucleation was chosen .Marsupialization has as its main objective the reduction of lesion size, and therefore, reducing the need for a more extensive and radical surgery . As a reSuccessful treatment of large odontogenic cysts using initial marsupialization and a second phase surgery with enucleation and curettage has been previously reported . When le4Marsupialization prior to total enucleation of a large COC proved to be a favorable and effective treatment choice. Since this surgical maneuver promoted a significant reduction of the lesion, damage to anatomical structures was insignificant and bone repair was facilitated. Together, this approach reduces the overall morbidity and costs, doesn´t require surgical reconstruction and results in an excellent outcome.This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.We declare that our institution does not require ethical approval of clinical case reports.Written informed consent was obtained from the patient’s parent 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.JJVP and ALRR contributed in conceptualisation, ATLM, HAS, contributed in study concept and design, JJVP and ATLM contributed in writing the paper.None.The guarantor of this work, Joao de Jesus Viana Pinheiro, accept full responsibility for the study and the conduct of the study, had access to the data, and controlled the decision to publish.Not commissioned, externally peer-reviewed.All authors declare no conflict of interest in formulating this article.


Purpose: To present an extremely rare case of corneal melanoma.Method: An 84-year-old female patient presented to our department with a pigmented corneal lesion in her right eye (OD), 6x4 mm, complaining of mild pain and inability of complete eyelid closure. Tumor growth had been noted the previous year. She had undergone cataract surgery in her right eye three years before, followed by an unspecified postoperative complication. Her visual acuity was 3/10 OD and 9/10 OS. Ophthalmic evaluation and ultrasonography (A- and B-scan) did not reveal any other pathology. The pigmented lesion was surgically removed and the patient underwent a protocol therapy of topical chemotherapy .Results: The surgical removal of the lesion was uncomplicated, as was the postoperative period. The patient’s visual acuity improved to 6/10 three months postoperatively. The histologic examination revealed malignant melanoma.Conclusions: Despite its rarity, primary melanoma of the cornea is an existing entity. Treatment of corneal melanoma consists of surgical removal and postoperative topical chemotherapy. Postoperative follow-up is mandatory. Primary melanoma of the cornea is an extremely rare clinical entity, which clearly differs from conjunctival melanoma extending onto the cornea. The first case of corneal melanoma was reported in 1892 , 4], , , 7], , , 11], , , 13], , , 15], , . FurtherIn December 2015, an 84-year-old woman presented to the 1st Department of Ophthalmology, Aristotle University of Thessaloniki, AHEPA Hospital, Greece, complaining of gradual vision deterioration, pain, and inability of complete eyelid closure  was 3/10 OD, with the head tilted to the right and 9/10 OS. Slit-lamp examination of her right eye revealed a circular corneal pigmented lesion, measuring 4x6 mm, clearly demarcated and surrounded by clear cornea of over 1 mm. Two feeder vessels passed through the limbus onto the lesion Bone scanDifferential diagnosis included tumors of the ocular surface, with corneal melanoma being the most prevalent based on strong clinical suspicion and published data regarding tumor atypia , 18]..18].PapillomaNaevusSquamous cell carcinomaPrimary acquired melanosis (PAM)Malignant melanomaIrregular tumor boundariesChange and/or color, shape, size variationPresence of epithelial defects or ulcers on the surfaceDilated feeder blood vessels leading to the tumorDisplacement and/or infiltration of adjacent tissuesDiagnostic and therapeutic management included surgical excision. Following cauterization of the feeder vessels, the lesion was removed on healthy boundaries via excision and scraping of the corneal epithelium . The patHistopathologic analysis confirmed our clinical suspicion. The corneal lesion specimen examined by the Pathology Department measured 0.5x0.4x0.3 cm and had gray-black colour. Hematoxylin- and eosin-stained sections revealed malignant corneal tissue involving the junction of the epithelium and the lamina propria, as well as the underlying connective tissue in its full thickness , September 2016, Copenhagen, Denmark.Dr. Panagiotou received a bursary from the ESCRS for attending the Congress and presenting this case.We express sincere thanks to Prof. Leonidas Zografos for his advisory guidance.A- & B-scan and UBM images courtesy of Dr. Miltos Balidis.Histologic images courtesy of Prof. Prodromos Chitiroglou.


Intrapulmonary teratoma (IPT) is a rare type of extra gonadal teratoma which often presents with non-specific symptoms and can be misdiagnosed as other diseases. Here we report a patient with IPT which was initially misdiagnosed as lung hydatid cyst versus abscess.We report an intrapulmonary teratoma in a 27-year-old female presenting with persistent chest pain and dyspnea since a few years prior to her admission with associated symptoms of cough and fever. Chest x-ray only showed left side massive pleural effusion and computed tomography scan of the lungs was suggestive of hydatid cyst or a lung abscess. She underwent lobectomy and postoperative histopathological study revealed IPT as the final diagnosis.Due to the non-specific symptoms and rarity, IPT can be easily misdiagnosed at first. It is essential that physicians take into account the possibility of IPT when approaching a new case of lung mass. Teratomas are benign germ cell tumors that are mostly found in the gonads with a low malignant transformation potency –3. ExtraA 27-year-old female visited our clinic with an unremarkable past medical history, with the chief complaint of progressive dyspnea and chest pain since 2 weeks prior to admission, which was recently accompanied by non-productive cough, chills, fever, and orthopnea. She also reported a mild, episodic, and occasionally pleuritic chest pain that radiated to back and left upper extremity. She denied any nausea, vomiting, rashes, joint pain, weight loss, or a history of smoking. She also reported a previous admission a few years ago due to dyspnea and chest pain in which after normal cardiac evaluation, was discharged with no established diagnosis.3). On the suspicion of rheumatologic disorders, rheumatoid factors were evaluated which were all normal. Subsequently, a chest computed tomography (CT) scan also revealed pleural effusion, which along with previous findings, provided us with the impression of empyema. Therefore, a pleural drainage needle catheter was inserted but due to insufficient drainage, it was replaced with a chest tube.On physical examination, the patient was febrile  and breathing sound was decreased in lower two-third of the left lung. Other systemic exams were unremarkable. The patient underwent radiological chest evaluation, in which chest X-ray revealed massive left side pleural effusion with no apparent focal opacities. On admission, routine blood investigations including renal and liver function tests were within normal limits, apart from white blood cell count that showed leukocytosis  and Albendazole , however, the symptoms did not alleviate and due to the possibility of hydatid cyst, intensive procedures such as aspiration and biopsy was avoided and surgical interventions to remove the lesion was suggested for treatment. Furthermore, abdominopelvic ultrasonography was done to rule out possible liver, retroperitoneal, and gonadal mass, which no significant findings were detected.The patient was operated under general anesthesia. Due to the severe adhesions caused by the recurrent previous infections, the operation changed from up position thoracoscopy to posterolateral thoracotomy. While exploring the pleural cavity, a grayish multi-lobulated firm intrapulmonary cystic mass (13 × 11 × 4 cm) was detected in the left upper lobe which was filled with hair and keratinized material. Based on the severe involvement and the retraction of the left upper lobe, left upper lobectomy was carried out.The specimen was collected from the lesion and the pleura for microscopic pathological evaluation. The gross examination of the operated specimen showed the upper lobe of the lung attached to a lobulated grayish cystic mass measuring 13 × 11 × 4 cm. Cut sections of the mass revealed unilocular cystic lesion filled with hair and waxy material. Microscopic sections showed the cystic lesion consisted of endodermal, ectodermal, and mesodermal components. Pancreatic tissue, mucinous epithelium, respiratory epithelium, epidermal tissue with sebaceous glands, adipose tissue, smooth muscle, and cartilage were identified in multiple microscopic sections Figs. , 3. Non-Based on the patients’ surgical and histopathological findings, a final diagnosis of intrapulmonary mature cystic teratoma was achieved and the patient was discharged after 8 days with an uneventful post-op course. Follow up during the next years showed no sign of recurrence and a normal chest x-ray Fig. .Fig. 4FIn this study, we presented a rare clinical case of IPT in a 27-year-old female involving the left upper lobe of the lung which presented with chest pain, dyspnea, low-grade fever, and dry cough. Teratomas are germinal cell tumors that are mostly present in gonads but can also be seen in extragonadal tissues . RegardiTeratomas are usually considered benign tumors but in ovaries in 2% of the cases, they can undergo a malignant transformation . until 2Previous studies have proved that there is an equal distribution of IPT by sex for men and women . Due to In our case, the patient presented with chest pain, dyspnea, fever, and dry cough. Similar studies have shown the common presenting clinical symptoms of IPT include chest pain as the most common presentation, fever, cough, dyspnea, and also hallmarks of pneumonia or bronchiectasis . TrichopSalmonella enteritidis [Reports from other studies have shown that laboratory tests are usually within normal limits , 13, whieritidis . Due to Based on radiological findings, in the majority of the cases, cystic lesions often with focal calcification are reported  but in sThe preoperative diagnosis in our case was in favor of empyema along with the possibility of hydatid cyst or lung abscess. Based on the nature of the tumor and symptoms, other possible differential diagnoses include ruptured hydatid cysts, fungal masses, lung abscess, pulmonary hamartoma, bronchogenic cyst, adenomatoid cystic malformation, intrapulmonary cystic lymphangioma, mediastinal teratoma, and pulmonary leiomyoma could also be considered , 16–19. Table Rupture of the tumor, hemoptysis, airway compression, and malignant transformation are the complications of IPT if remains untreated , 24. Sur. Therefore, prompt diagnosis and suitable treatment should be immediately performed for these patients to avoid significant and life-threatening complications.In conclusion, the preoperative diagnosis of IPT is not always possible and is usually misdiagnosed at first because of its rarity, non-specific and vague symptoms, normal laboratory results, and indistinguishable chest radiography findings. Initial diagnosis can be established based on a CT scan which can demonstrate calcification, cavitation, and peripheral translucency. Complete resection and surgery are considered as its gold standard curative treatment modality to avoid complications and malignant transformation


Teaching point: This case emphasizes the importance of the “tail sign” to sort the differential diagnosis of soft tissue tumors by highly suggesting schwannoma. A 50-year-old patient presented with a lump in the distal posterior arm, painful upon compression. Ultrasound showed an intramuscular fusiform mass of 9 × 12 mm in the distal triceps muscle. The tumor , hypothesis of a neurogenic tumor should be contemplated. The “split fat” sign that refers to the presence of fat at the upper and lower poles of a lesion as shown in this case (arrowheads in Figure This case emphasizes the usefulness of the “tail sign” on all imaging modalities to sort the differential diagnosis of soft-tissue masses by suggesting peripheral nerve tumor.


The foreign body reaction caused by oil contrast medium, Lipiodol, is rare. We present a rare case of inflammatory granuloma in the inguinal hernia sac after hysterosalpingography with lipiodol.A 30-year-old woman who had left inguinal growing mass for 7 months after hysterosalpingography with Lipiodol for examination of infertile. About 3 cm soft mass was palpable on the left inguinal region, and plane CT scan showed a multifocal cystic mass with metal concentration. Thus, laparoscopic herniorrhaphy was performed to repair the inguinal hernia and to confirm the contents of cystic lesion. The hernia sac filled with mucinous contents. Pathological examination revealed that inflammatory granuloma due to numerous lipid droplets and phagocytic images of leukocytes in the hernia capsule.Although oil contrast medium is safety agent, there is rear complication which need the surgical treatment. Complete resection of granuloma, including hernia sac, is important to prevent recurrence. Especially, improvements of fertility after hysterosalpingography using lipiodol were reported in the field of obstetrics and gynecology . HoweverWe reported a rear case of inflammatory granulomatous alteration in the inguinal hernia sac by accumulation of Lipiodol after hysterosalpingography, who was diagnosed and treated by laparoscopic surgery. This article has been written according to SCARE criteria .2A 30-year-old woman with left inguinal growing mass consulted our hospital.She was examined by hysterosalpingography with Lipiodol about 8 months before consult because of infertile at another hospital. She recognized left inguinal mass 7 months after the examination. About 3 cm of soft mass was palpable on the left inguinal region in a standing position, and this mass was disappeared in a spine position. The patient was afebrile and the blood tests revealed neither neutrophilic leukocytosis nor raised level of inflammatory C-reactive protein. Plane CT scan showed a multilocular mass in the left inguinal region A and B. Trans vaginal ultrasonography showed bilateral ovaries were present in the pelvis and these were normal. Some aberrations such as endometriosis, hydrocele of the canal of Nuck, etc. were considered as differential diagnosis. Thus, we decided to perform a surgical treatment, laparoscopic hernia repair with hernia sac resection, was mandatory.3About 2 cm of skin incision was made on the umbilicus, and a camera port was inserted by the open laparotomy method. Bilateral indirect inguinal hernias (I-1 of The Japan Hernia Society (JHS) Classification for groin hernia) were recognized A and B. 4The hernia sac was generally fibrously thickened cyst, and the increase of fibroblasts and the collection area of foam cells were observed . It was 5Hysterosalpingography is a basical examination method of infertility, a method of injecting a contrast medium from the vagina side into the lumen of the uterus, observing the shape of the uterine cavity and checking the passability of the fallopian tubes. There are two type of contrast medium, oily and aqueous. And oily contrast agents are widely used because of their superiority of rendering properties and the high possibility of pregnancy after treatment. Lipiodol, an oily contrast agent, is more widely used as a contrast agent than aqueous contrast because of its less complication rate, such as systemic reaction and peritoneal irritation. However, there is a few reports that oily contrast medium is hardly absorbed in the body and remains in the long term, resulting in inflammatory granuloma due to foreign body reaction .Inflammatory granuloma is one of the chronic inflammation caused by accumulation of inflammatory cells, such as macrophage, phagocytosing lipids, epithelial cells, multinucleated giant cells. Many papers reported that inflammatory granuloma is easily caused in the kidney or gall bladder . HoweverIn this case, the patient had no symptom of groin hernia before hysterosalpingography, and it takes about 7 months when the patient recognized groin mass on the left inguinal region. Thus, it is speculated that Lipiodol which remain in hernia sac for a long time cause foreign body reaction, resulting in inflammatory granuloma.Although there are many reports that surgical resection was performed as a diagnostic treatment for the inflammatory granuloma, no standard therapy was established. In this case we underwent inguinal hernia repair by the transabdominal preperitoneal approach (TAPP). There are two reasons, 1) the form and properties of cystic lesions can be confirmed from intraabdominal cavity, 2) whole abdomen, especially pelvic cavity, and bilateral inguinal hernia are able to be observed. Some papers reported that the cystic formation with inflammatory granuloma on the pelvis, diaphragm and intestinal tract after uterine oviducture contrast examination caused stricture of the ureter, bile duct and intestine . Thus, oWe performed herniorrhaphy by LPEC for the contra lateral inguinal hernia which was no symptom in this case, because there was a possibility that the intra-abdominal pressure will be rise when she will pregnant, and contralateral inguinal hernia will be occurred. Although regarding the adequacy of LPEC for adults was controversial, Because the mechanism of occurrence of young adult inguinal hernia is often the same as in childhood, treatment with high - level ligation method can be considered. Recently, there are some papers reported that LPEC is effective for a inguinal hernia of a young woman without vulnerability of the abdominal wall . Thus, L6We reported the rare case of inguinal hernia due to inflammatory granuloma by oil contrast medium. Although oil contrast medium is safety agent, there is rear complication which need the surgical treatment. Complete resection of granuloma, including hernia sac, is important to prevent recurrence.All authors declare no conflicts of interest associated with this manuscript.This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.The study was reviewed and approved by the Shimane University Institutional Review Board. The reference number is 20200127-1.Informed consent was obtained from the patient.Contributors YT was responsible for the organisation and coordination of this study. TY was the chief investigator and also responsible for the data analysis. RH, TT, TH and NH planned this study. All authors contributed to the writing of the final manuscript. All authors contributed equally to the conception, design, literature review, analysis, drafting, critical revision, and editing and approved the final version of this study.http://www.researchregistry.com. The identifying number is researchregistry5607.This case report was registered at Tetsu Yamamoto and Yoshitsugu Tajima.The study was reviewed and approved by the Shimane University Institutional Review Board.Not commissioned, externally peer-reviewed.


Schwannomas of the seminal vesicles are extremely rare, and only cases of single seminal vesicle schwannomas have been reported. Here, we report a case of multiple schwannoma of the seminal vesicle.We report a rare case of multiple schwannoma of the seminal vesicle that occurred in a 48-year-old man during physical examination. Multiple mixed masses in the left region of the seminal vesicle were documented with transrectal ultrasonography and magnetic resonance imaging. The patient presented no clinical symptoms, no family history of the disease and no history of genetic disease.Postoperative pathology revealed a diagnosis of seminal vesical schwannoma.The patient underwent robotic-assisted laparoscopic surgery to remove the mass.The patient recovered rapidly and the length of hospitalization was 6 days after operation. At present, there is no recurrence in 10 month follow up.Whether benign or malignant, single or multiple, schwannomas still need to be diagnosed by pathology because of the limitations of examination methods. Surgical resection is still the preferred treatment. Only 10 cases of single seminal vesicle schwannomas have been reported.–11 Multiple schwannomas are still rarer in the seminal vesicle, and they are only reported in the penis of the urinary system. Here, we report a rare case of multiple schwannoma of the seminal vesicle and its related clinical features.Neurilemmoma, also called schwannoma, is usually a benign tumor with slow growth, a complete capsule and occasional malignant changes.2A 48-year-old married man who underwent magnetic resonance examination in a local hospital was found to have a mixed mass in the left seminal vesicle. In order to seek further treatment to our hospital, the patient denied any related clinical symptoms, family history of the disease, or history of genetic disease. Routine blood examination, coagulation function, prostate-specific antigen, and tumor markers were all within the normal ranges. We observed 4 independent but closely adjacent cystic-solid mixed masses in the left seminal vesicle through a scan provided by the patient Fig. , the lefGeneral appearance Fig. : The sec3 At present, there were only 10 cases of single seminal vesicle schwannomas have been reported in the literature. Andrea et al reported a 2.6-cm single schwannoma of the seminal vesicle in a 43-year-old man with lower urinary tract symptoms. Arun et al reported a 13.5-cm single schwannoma of the seminal vesicle in a 50-year-old man who was found to have left abdominal pain that lasted for 1 month. Elmer-DeWitt et al reported that a 5.3-cm single schwannoma was found in the seminal vesicle adjacent to the rectum in a 62-year-old man with rectal pain, and this case was the first to report removal of the tumor by robotic surgery. Huang et al reported a 5.1-cm single schwannoma of the left seminal vesicle in a 55-year-old man after physical examination. According to these existing reports, schwannoma of the seminal vesicle usually does not cause clinical symptoms; however, when the surrounding tissues are compressed, there are some nonspecific symptoms, such as hemospermia, pain, irritation, obstructive lower urinary tract symptoms and infertility.,14Schwannomas are relatively common benign tumors of peripheral nerves. Schwannomas can occur spontaneously or in the context of a familial tumor syndrome such as neurofibromatosis type 2 (NF2), schwannomatosis or Carney complex. The reason why the number of lesions was not observed in this case was that the lesions had similar intervals or overlaps, and echoes were similar, and it is difficult to display the spatial relationship of the mass by two-dimensional ultrasound. Transrectal ultrasound could only be used to describe the general echoes of the lesions as well as the boundary and blood flow. However, because of its convenience and low cost, transrectal ultrasound can still be used as the preferred method for primary screening and follow-up after operation. CT and MRI can evaluate the nature of a mass and the degree of infiltration surround the mass, providing a basis for the choice of operation method. Neurilemmoma tumor cells generally consist of Antoni A and Antoni B regions as assessed by microscopy, and its imaging characteristics are different because of the different distribution of Antonia A and Antonib B in different tissues. Hideyuki et al by observing the imaging and pathological features of 162 cases of peripheral neurilemmoma, found that the area of Antoni A is situated in the central portion and the area of Antoni B is in the peripheral portion. On T2-weighted images, the area of Antoni B  with edematous stroma shows relatively higher intensity than the area of Antoni A ; on Gd-enhanced T1-weighted images, focal enhancement in the area of Antoni A with rich capillaries shows higher intensity than focal enhancement in the area of Antoni B. The author holds that the target sign should be defined as the characteristic central/peripheral biphasic pattern seen on T2-weighted images and Gd-enhanced T1-weighted images, MRI is the best way to connect macroscopic and microscopic findings. However, these characteristic imaging findings are not found in neurofibroma and other soft tissue tumors that are difficult to distinguish. We report a case of multiple seminal vesicle schwannoma, in which the cystic component (Antoni B region) showed higher signal in the sagittal T2 lipid-pressing sequence, and the cyst wall and solid portion (Antoni A region) of the T1 lipid-pressing enhancement sequence showed progressive enhancement. Because of more cystic degeneration, the biphasic pattern on MRI is not typical, which is difficult to diagnose. However, the imaging feature of the bipolar pattern of schwannoma on MRI provides a diagnostic method for similar cases in the future. In the left seminal vesicle of the patient, the boundary of the lesion was smooth and clear. Although the adjacent tissues were compressed, there was no sign of infiltration, suggesting a benign lesion. Benign schwannoma of the seminal vesicle should be differentiated from cystadenoma and low-grade epithelial-stromal tumors. Cystadenoma of the seminal vesicle is rare and can manifest as a septal multilocular neoplasm. The histological findings demonstrated that the tumors were composed of glands and cysts that were lined predominantly by cuboidal and columnar epithelium surrounded by fibrous stroma. Reikie et al proposed that the term seminal vesicle “mixed epithelial-stromal tumor” should be used to designate tumors of the seminal vesicle containing epithelial and stromal components. According to their criterion, cystadenoma of the seminal vesicle can be considered a low-grade mixed epithelial-stromal tumor.Ultrasonography, computed tomography (CT) and magnetic resonance imaging (MRI) are the common imaging methods used to examination urologic neoplasms. Transrectal ultrasonography is the first-line method to detect urogenital tumors, and it can dynamically visualize the lesions.For benign or malignant, single or multiple, schwannomas still need to be diagnosed by pathology because of the limitations of examination methods. Surgical resection is the first choice of treatment. In principle, the tumor should be thoroughly removed, and the surrounding organs, large vessels, and nerves should be maintained. There are various operative methods that can be chosen. The individual conditions of patients should be considered comprehensively, and the most suitable individual surgical scheme should be administered to patients. Traditional open surgery is time-consuming and involves intraoperative blood loss and trauma, and it easy to damage the surrounding organs such as the rectum with this method. Laparoscopy can reveal the deep seminal vesicles and surrounding tissues, and this procedure is relatively simple, safe and minimally invasive. Robot-assisted laparoscopic mass resection was used in this case. The patient was discharged on the sixth day after the operation and recovered quickly. The patient did not show any signs of recurrence at the 10-month follow-up. No further immunohistochemistry was performed in this case, continued vigilance is necessary for the possibility of recurrence, and this patient should be followed up for a long period of time.The authors would like to thank the patient and all members of the study team.Conceptualization: Yan Zhang, Jing Zhao, Pan Xu, Qi Qi.Data curation: Yan Zhang, Qi Qi.Resources: Pan Xu.Validation: Pan Xu, Qi Qi.Writing – original draft: Yan Zhang, Jing Zhao.Writing – review & editing: Pan Xu.