The nodulus and ventral uvula (NU) of the cerebellum play a major role in vestibular function in humans and experimental animals; however, there is almost no information about NU function in the veterinary clinical literature. In this report, we describe three canine cases diagnosed with presumptive NU hypoplasia. Of them, one adult dog presented with cervical intervertebral disk disease, and two juvenile dogs presented with signs of central vestibular disease. Interestingly, an unusual and possibly overlooked neurological sign that we called “positioning head tilt” was observed in these dogs. The dogs were able to turn freely in any direction at will. The head was in a level position when static or when the dog walked in a straight line. However, the head was tilted to the opposite side when the dog turned. Veterinary clinicians should be aware of this neurological sign that has not been reported previously, and its application in lesion localization in dogs. The vestibulo-cerebellum is described in the veterinary literature as consisting of the flocculonodular lobe (flocculus and nodulus) and the fastigial nuclei and as being principally responsible for maintenance of equilibrium and coordination of head and eye movements –5. In huIn this report, we present three cases of canines with presumptive NU hypoplasia and resultant head tilting in response to head movement caused by lack of inhibition of the vestibular nuclei by the NU. We name this symptom “positioning head tilt.”Dirofilaria immitis was negative. Thoracic and abdominal radiographs revealed no abnormality. Magnetic resonance imaging (MRI) and radiography of the cervical spine performed on the initial presentation day revealed that the tetraparesis was caused by C3–4 intervertebral disk disease that was treated surgically using a ventral slot decompression procedure on the same day. After the tetraparesis had resolved a week after surgery, the head tilt was still present and reevaluated during observation of free walking. The dog could walk freely in any direction at will. The head was in a level position when static or when the dog was walking in a straight line. However, the head was consistently tilted to the right when the dog turned to the left, and to the left when the dog turned to the right . An antigen test for A 5-month-old female Shiba dog presented to Bingo Pet Clinic  in 2015 with a history of circling. Since the owner had acquired the dog at 2 months of age, it had exhibited throwing of the head back and forth when looking into the distance. Peracute onset of rolling to the left was observed before the dog was brought to the clinic, but this symptom disappeared within several minutes. Although abnormal rolling or circling was not observed at presentation, slight ataxia and head tilt to either side were identified, and the dog was referred to Tamura Animal Clinic 10 days after the initial consultation. Positioning head tilt was observed during observation of free walking. Most of the time, but not always, the head was tilted in response to head movement. Positioning head tilt had been observed by the owner since acquiring the dog, but the owner had considered this head tilt a peculiarity. Ataxia of the whole body was observed when the dog shook her head or scratched her ear with a hind limb  was performed on the initial presentation day. T2W  sagittal, coronal, and transverse images; FLAIR  sagittal and transverse images; and T1W  sagittal and transverse images were acquired. Post-contrast T1W sagittal and transverse images were acquired after intravenous administration of gadopentetate dimeglumine . Absence of the NU and hypoplasia of the caudal fossa were revealed. There were no other abnormalities, and no abnormal contrast enhancement was observed in the brain  –16. It iThe positioning head tilt observed in our cases may be explained by the following hypothesis. When the head is static or when the subject walks in a straight line, bilateral vestibular nuclei are equally stimulated by both vestibular organs, and the head is maintained in a level position. Vestibular nuclear projections to the spinal cord are situated in the vestibulospinal tracts. The fibers terminate on interneurons that facilitate ipsilateral flexors and decussate to inhibit contralateral extensor muscle activity. The vestibulospinal tracts facilitate spinal reflexes, especially those involved in maintaining posture and the antigravity/extensor muscles. Turning the head to the left causes a shift in the distribution of body mass to the left. This shift in mass is supported by increased extension on the left side, which is reflexively induced by both myotatic and vestibular reflexes. This also reduces extension on the right side, and thereby minimizes weight transfer to the left . The NU A congenital origin for the abnormal NU is more likely than a lesion secondary to infarction of a branch of the bilateral caudal cerebellar arteries because of the presence of hypoplasia of the caudal fossa and no history of the dog having had any episode of a suspected cerebellar vascular accident in Case 1, and positioning head tilt had been observed before the peracute vestibular episode in Cases 2 and 3.The causes of the ataxia and decreased menace response observed in Case 2 and peracute episodic vestibular signs observed in Cases 2 and 3 were undetermined. Another disease process  cannot be ruled out because of the lack of CSF analysis. However, these signs might have been specific to the acute phase of NU dysfunction, because they were seen in the juvenile dogs and later disappeared. The partial hypoplasia of the left cerebellar hemisphere in Case 2 might also have contributed to these signs. It is interesting that the direction of the vestibular signs differed between the 2 episodes in Case 3. This may have been due to bilateral NU lesions.Some genetic mutations have been identified in humans with cerebellar vermis hypoplasia . There iClinical findings in humans with lesions affecting the NU have been reported previously  and inclLimitations of this study include the small number of cases and the lack of pathological diagnoses in all cases. However, the locations of focal lesions were clearly identified on MRI in all dogs, so it is appropriate to discuss the relationship between neurological signs and lesion localization. Detailed observation of the clinical signs and course in large numbers of cases with similar characteristics to our cases, as well as pathological diagnosis, is warranted.This novel information about the neurological signs in canine NU disorder may help veterinary clinicians to localize the lesion in dogs with this condition, as well as neurophysiologists to acquire further understanding of the functions of the NU.Consent procedure is not applicable, as this article is a retrospective review of clinical cases.All the authors met the criteria for authorship. ST, YN, TU, and YT participated in clinical case management. ST drafted the manuscript. ST and YN reviewed the neuroimaging studies. ST, YN, and YT participated in the review and the editing of the manuscript.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


The calcifying odontogenic cyst (COC) is a benign odontogenic cyst that occurs in the gnathic bones. This cyst is part of a spectrum of lesions characterized by odontogenic epithelium containing “ghost cells,” which may undergo calcification. Areas of an eosinophilic matrix material compatible dentinoid also may present adjacent to the epithelial component. However, these areas of dentinoid commonly do not appear so abundant in COCs. In this study, we report a case of intraosseous COC with extensive areas of dentinoid and perform an update regarding the clinical, radiographical, histopathological, and differential diagnosis, treatment, and prognosis of this cystic lesion. The calcifying odontogenic cyst (COC) was first described by Gorlin and colleagues in 1962 .Recently, this lesion was reclassified as a cystic lesion again by the World Health Organization (WHO) . It was COCs represent a heterogeneous group of lesions that show a variety of clinicopathologic and behavioral features . IntraosThe distinct feature of COC is a cystic lining demonstrating “ghost” epithelial cells with a propensity to calcify. This cyst most commonly occurs as a well-defined cystic lesion with a fibrous capsule and a lining of odontogenic epithelium with Ameloblastomatous appearance , 11. AreAn 82-year-old male patient presented in the Oral and Maxillofacial Surgery Service of the Federal University of Rio Grande do Norte for evaluation of a swelling on the alveolar ridge of the anterior mandible, which had been previously identified in routine radiographic examination with prosthesis purposes. During anamnesis, the patient reported to be hypertensive and diabetic, as well as having chronic heart disease. At the clinical examination, no volume could be observed in the anterior region of the mandible, and the patient reported no painful symptomatology.Radiographical examination revealed an unilocular and irregular radiolucent lesion, measuring approximately 3 cm in diameter . PreoperThe COC is a rare entity of uncertain pathogenesis; its clinical and radiographic characteristics are not pathognomonic, being characterized mainly by its histopathological characteristics . This cySince its recognition and description by Gorlin et al. 1962) [962 [1], Approximately 65% of all cases of COC are reported in the anterior region of the mandible . This stThe histopathological findings of the COC include basal layer of the epithelial lining presenting a columnar or cuboid appearance with a similar appearance to ameloblasts. It is also possible to observe a cellular arrangement that resembles the stellate reticulum of the enamel organ in the suprabasal layers , 14. A sIn the present case, it was possible to observe a prominent and extensive presence of dentinoid in the fibrous capsule. There are different theories regarding the etiology of this material; while some authors believe that it may be a result of an inductive effect by the odontogenic epithelium on the adjacent mesenchymal tissue , 11, othIntraosseous COC is mainly treated by enucleation, with a low rate of recurrence, occurring in <5% of cases . HoweverIn conclusion, the present case reinforces the importance of knowledge by the pathologists of the histopathological features of the intraosseous COC in order to achieve a correct diagnosis and, consequently, a favorable outcome for the patient. Furthermore, the literature shows that these areas of dentinoid commonly do not appear so extensive in COCs, as seen in the current case.


Emphysematous epididymo-orchitis is a rare cause of acute scrotum pain characterized by gas formation within the tissue. Diabetes mellitus and recto-seminal fistula secondary to sigmoid diverticulitis are generally accepted as being responsible for this disease. However, prostate invasion secondary to rectal cancer may be considered to be a newly identified pathogenetic mechanism. Herein, we report this rare case and illustrate the pathogenesis. A 69-year-old man arrived at our emergency department presenting with sepsis and acute scrotal pain. Emphysematous epididymo-orchitis was diagnosed by scrotal sonography initially; however, advanced rectal cancer with prostate invasion was diagnosed by CT after a recurrent episode. An exploratory laparotomy with abdominoperineal resection and radical prostectomy were performed after neoadjuvant chemoradiotherapy. Histopathologic analysis confirmed the previous diagnosis. Emphysematous epididymo-orchitis caused by advanced rectal cancer is very rare, and our case is the first to be reported according to a literature search. Neoadjuvant chemoradiotherapy plus extended surgery can achieve a good oncological outcome.This case indicated that the very rare presentation as emphysematous epididymo-orchitis caused by locally advanced colorectal cancer. However, the true pathogenesis is unclear owing to the small number of cases. Herein, we describe a case of emphysematous epididymo-orchitis with initial treatment failure. Computed tomography (CT) of the pelvis demonstrated a rectal mass, which was found to have directly invaded into the prostate after further evaluation. Pathologic analysis revealed adenocarcinoma of the rectum with prostate invasion. We assumed that anatomic change of the invasive tumor induced microfistula formation between the rectum and prostate. Furthermore, intestinal bacteria induced prostate abscess formation and extended from the ejaculatory duct to the epididymis. Finally, the bacteria with gas production resulted in the clinical emphysematous epididymo-orchitis. Interference in prostatic intraductal drainage by the bulging rectal tumor also exacerbated the bacterial retrograde infection. This hypothesis provides a more complete explanation of the pathogenesis of emphysematous epididymo-orchitis.Acute epididymo-orchitis is an inflammation of the epididymis, often involving the testis, caused by bacterial infection. Extension of infection from the urethra or bladder is a well-known pathogenesis of acute epididymo-orchitis. Rarely, emphysematous epididymo-orchitis may be characterized by the presence of air within the tissue, presenting a rare cause of acute scrotum pain.2Bacteroides fragilis and Clostridium spp. The patient was discharged after adequate antibiotics with ceftriaxone and metronidazole at stable wound condition. However, recurrent right side scrotal pain was noted with purulent discharge from the previous surgical wound after 1 month, which pathogen proved as mixed organisms including B fragilis, Clostridium spp., and Klebsiella pneumonia. Digital rectum examination demonstrated moderate enlarged prostate with induration, without palpable rectal mass. Due to inexplicable clinical features, CT of the pelvis and scrotum was performed, which demonstrated a heterogeneous density of the prostate with central low attenuation, whole wall-thickening of the rectum, and enlargement of lymph nodes  level was 14.55 mg/dL. Scrotum sonography demonstrated some bright spots and hypoechoic areas (Fig. Written informed consent to publish the case report was provided by the patient, and the consent procedure was approved by the Ethics Committee of Tri-Service General Hospital.3–3 Acute scrotal pain, swelling of the scrotum, and fever are most common symptoms. Imaging findings of ultrasound and CT have demonstrated air within the testis to be the main characteristic in the few reported cases.–3 Unlike extended infection, which is a well-known mechanism of acute epididymitis, the pathogenesis of emphysematous epididymo-orchitis remains unclear. Diabetes mellitus and recto-seminal fistula secondary to sigmoid diverticulitis have been reported to be pathogenetic mechanisms. However, only 1 colorectal cancer patient has been reported to be related to the epididymo-orchitis in the literature.Emphysematous epididymo-orchitis is a very rare disease, and only 3 cases have been reported. Urinary tract involvement in cases of colorectal cancer is by means of direct invasion or fistula formation.–8 In addition, colon cancer can induce Fournier gangrene.,10 Therefore, the urinary tract can be affected by direct tumor invasion or tumor-associated inflammatory processes.Advanced colorectal cancer invading the urinary tract is not uncommon, and owing to the close anatomic relationship, approximately 5% of primary colorectal cancers are locally advanced to the urinary system. Colo-urinary tract fistula complicating colonic diverticulitis has been reported. A fistula between the colon and urinary tract is most common among other types, and urinary tract infection is the main symptom. Colorectal cancer also can induce the same inflammatory process and fistula formation. Bruketa et al ever concluded that the perforated rectal cancer could induce Fournier gangrene with perineum fascia extension and absence of testis involvement. However our case presented as annular rectal cancer with prostate invasion, which hypothesized to process the development of emphysematous epididymo-orchitis through the vas deferens via backflow of gas-producing bacteria. With the observation that the emphysematous epididymo-orchitis without perineum fascia extension was noted in our case, we postulated that there existed a fistula between rectum and prostate. Further, intestinal bacteria induced prostate abscess and infection extending from the ejaculatory duct to the epididymis. The pathogenesis of our presented case is quite different from perforated rectal cancer inducing Fournier gangrene. In our case, the abscess and infection were evidenced by recurrent infection and bacterial culture analysis. Moreover the inflamed prostate obstructed the prostatic urethra, causing poor prostatic intraductal drainage and backflow of urine. This is also an aggravating factor in emphysematous epididymo-orchitis. In conclusion, the hypothesized pathogenesis was illustrated in the schematic diagram (Fig. Emphysematous epididymo-orchitis can be an unusual presentation of sigmoid diverticulitis.ram Fig. .–16; however, a bladder-sparing procedure allows conservative surgical treatment to be administered in selected patients. In addition, neoadjuvant chemoradiotherapy with capecitabine before total pelvic exenteration is effective for the treatment of locally advanced sigmoid colon cancer. In a recent review article, neoadjuvant chemoradiotherapy was reported to result in a higher R0 rate than surgery alone, and a longer overall survival of 85% to 75% within 3 to 5 years was achieved. In our case, we performed suprapubic cystostomy for urinary diversion and T-loop colostomy for infection control first. The patient subsequently received neoadjuvant chemoradiotherapy and an exploratory laparotomy with abdominoperineal resection. Postoperative adjuvant chemotherapy is currently in progress. No distant metastasis or recurrence has been observed. Therefore, multimodal therapy and extended surgery result in a good prognosis in patients with locally advanced colorectal cancer.Total pelvic exenteration is the gold-standard treatment for locally advanced colorectal cancer4Emphysematous epididymo-orchitis caused by locally advanced colorectal cancer is very rare, and our case was the first to be reported according to a search of the literature. A fistula between the urinary tract and colon in addition to an extended bacterial infection is the most reasonable pathogenetic mechanism of emphysematous epididymo-orchitis. Neoadjuvant chemoradiotherapy with total pelvic exenteration can achieve a successful oncological outcome.


Mediastinal solitary fibrous tumors (SFTs) are rare mesenchymal neoplasms. Complete resection is considered as the effective treatment and the prognosis is quite good. Rapid metastasis after surgery is extremely rare.In this case report we describe a 42-year-old man who present with a mediastinal malignant SFTs. Enhanced computed tomography of chest revealed a 4.5 × 4.0-cm mass in the anterior mediastinum.The tumor is composed of massive proliferation of atypical spindle cells. Immunohistochemical staining for cluster of differentiation (CD) 34, CD99, and vimentin were strongly positive.Due to the possibility that the tumor was malignant, a standard median sternotomy was performed under general anesthesia. The mediastinal tumor and the affected part of the pericardium and right upper lobe of the lung were completely resected.The patient underwent surgery and recovered uneventfully. After 2 months follow-up postoperation, there was recurrence in the chest wall and right middle lung. The patient refused any treatment and was dead after 2 months.The present cases indicate that mediastinal SFTs should always be kept in mind for rapid metastasis. Once found, surgical intervention should be performed promptly. Due to the rapid metastasis, radiotherapy, and chemotherapy may be needed after surgery and long-term follow-up is required to monitor the metastasis of this type of tumor. To our knowledge, mediastinal SFTs with lung and pericardium invasion are extremely rare and mostly grows slowly and usually found incidentally. After that, due to the tumor growth, such symptoms occur: cough, chest pain, and chest tightness. Complete resection is considered as the most effective and successful treatment of mediastinal SFTs and the prognosis is good. The present study presents a rare case of rapid metastasis in mediastinal malignant SFT with lung and pericardium invasion in a 42-year-old man, and reviews the previously reported cases in the literature.Solitary fibrous tumors (SFTs) are rare nonepithelial neoplasms that most often involve the visceral pleura, which were initially described by Klemperer and Rabin in 1931.212/L ; white blood cell count, 7200 cells/mL ; platelet count, 1330 cells/mL ], serum electrolyte , glucose level [4.38 mmol/L ] coagulation function , liver function , renal function , electrocardiogram , were all within normal limits. Distant metastasis or dissemination was not found during ultrasound (US) of the abdomen, radionuclide bone scanning, and magnetic resonance imaging (MRI) of the head. Due to the possibility that the tumor was malignant, a standard median sternotomy was performed under general anesthesia. The mediastinal tumor and the affected part of the pericardium and right upper lobe of the lung were completely resected. On macroscopic examination, the tumor was firm and measured 6.0 × 4.5 × 3.5 cm  scan  of the chest revealed a 4.5 × 4.0 cm solid mass involving the anterior mediastinum .SFTs are usually found incidentally and observed in middle-aged adults between 20 and 70 years with no sex predilection. The criteria for the diagnosis of malignant SFTs should include: high cellularity with crowded or overlapping nuclei; nuclear pleomorphism; high mitotic activity, with >4 mitotic figures per 10 high-power fields; and pleomorphic giant cells and abnormal mitotic activity. The differential diagnosis of mediastinal SFTs includes numerous malignant and benign tumors, including malignant mesothelioma, peripheral nerve sheath tumors, spindle cell thymoma, sarcomatoid carcinoma, inflammatory myofibroblastic tumor, and various sarcomas. Imaging examinations, including US, CT, and magnetic resonance (MR) are used for assessing mediastinal SFTs. It is not possible to determine whether a mass is benign or malignant by imaging examination. However, The CT findings can reveal the extent of tumor, regional invasion, and vascular encasement calcification. To our best knowledge, Positron emission tomography (PET)-CT was a new tool for assessing the SFTs. Suehisa et al reported a case of mediastinal SFT that assessed with PET-CT, it showed weak uptake by the tumor, with a maximum standardized uptake value (max SUV) of 1.85. Definite diagnosis is base on immunohistochemistry. CD34, which is a hematopoietic progenitor antigen, plays a substantial role in supporting the diagnosis of SFTs. Recently, some studies have shown positivity for Bcl-2, CD99, and vimentin are important markers for the diagnosis of an SFT, but it not considered specific.Because of the lack of specific imaging features, mediastinal SFTs are relatively difficult to distinguish from other tumors before surgery. According to the study of England et al. In the past, the majority of mediastinal tumor excisions have been approached via standard median sternotomy. With the improvement of thoracoscopic surgical skills, surgery is increasing being performed by thoracoscope. The present patient was not considered to be a suitable candidate for video-assisted thoracic surgery (VATS) because the tumor was malignant and huge.The most successful management of mediastinal SFTs is by surgical resection, which can be either radical or staged. The prognosis for patients with mediastinal malignant is unknown. Xue et al reported a rare case of mediastinal SFT invading the lung and diaphragm. The patient remains alive with no sign of recurrence more than 18 months after her operation. Zhang et al reported 13 cases of mediastinal SFT that managed by surgery. The patients had no recurrence or metastasis, except one died of cerebral hemorrhage 2 months after the surgery. But in our present study, there was metastasis in the chest wall and right middle lung after 2 months follow-up postoperation, and patient was dead after 4 months.Mediastinal malignant SFTs can recur after complete resection. With the local recurrence, second surgery is remains considered the best method.In conclusion, we described a rare case of 42-year-old male who presented with a mediastinal malignant SFT invading the lung and pericardium, and received tumor complete excision. The patient recovered uneventful and discharged on day 13. After 2 months follow-up postoperation, there was metastasis in the chest wall and right middle lung. The patient refused any treatment and was dead after 4 months. Due to the high mortality, new strategy is needed to monitor the rapid metastasis of this type of tumor.We would like to acknowledge the patient for allowing this case to be published.


Leiomyoma is a rare esophageal tumor that constitutes less than 1% of esophageal malignancies. It should be removed when diagnosed even if asymptomatic. This study presents two cases of esophageal leiomyoma in 38 and 62 years old men who presented with dysphagia and evaluated for diagnosis and location of related tumors. Patients were clinically examined and upper gastrointestinal endoscopy, chest computerized tomography scan, barium swallow and endoscopic ultrasonography were performed. The masses were diagnosed and both patients underwent 3-port right videothoracoscopic enucleation of esophageal leiomyoma with sleeve calibration tube (SCT) assistance. SCT was used to protrude out the mass from surrounding tissues. Also immunohistochemistry was done after mass enucleation. Two patients were operated routinely without any unpredictable events by help of SCT assistance. Mass size was 3.5×2.5×1 cm in one case and 1.5×1×0.5 cm in another patient. Positive smooth muscle antigen, desmin and ki67 and negative CD34, CD117 and S100 in both cases were obtained in immunohistochemistry. Patients were followed after surgery for 3 months and no complications were detected in none of them. Thoracoscopic enucleation of esophageal leiomyoma is a safe and feasible procedure which can decrease hospitalization and operation time. Based on our findings, the use of esophageal SCT help to detect smaller tumor without need of intra-operative endoscopy, facilitates separation of the tumor mass from both esophageal mucosal and muscular layers, and may prevent perforation. Finally, use of SCT makes the operation faster and safer. It is, however, a rare condition with an overall incidence of 8-43 per 10000 in autopsy series.3,4 The leiomyoma was found in an intramural position within the esophageal wall (97%), true polypoid lesions (1%) and extending extra-esophageal as mediastinal out growths (2%). It is estimated that 10% of intramural leiomyoma was reported to have grown in a circumferential way, almost surrounding the esophageal lumen.3,5Morgagni described leiomyoma in 1761 and then Munro was reported esophageal leiomyoma in 1797.5 They vary in size and shape from few mm to near 30 cm in diameter.6 Approximately half of the patients are asymptomatic, but when symptoms occur they comprise dysphagia, retrosternal pain, heartburn and weight loss.3,4 Diagnosis is usually made on esophagogram, chest X-ray, computerized tomography (CT) scanning and magnetic resonance imaging (MRI). Recently transesophageal echo probe has been widely used for pre-operative diagnosis.3,4,7About half of the patients with leiomyoma are asymptomatic; this may be more so in cases of lesions found incidentally at autopsy. The location was in lower third of the esophagus (56%), in the middle third (33%), in the upper third (11%) and small percentage at the gastro-esophageal junction (GEJ).1,3 Here, we describe two cases of symptomatic leiomyoma of the mid esophagus, resected after right videothoracoscopic enucleation with sleeve calibration tube (SCT) assistance.Leiomyoma should be removed unless there are specific contraindication. The majority can be removed by simple enucleation. The mortality rate associated with enucleation is low and success in relieving the dysphagia is near 100%. Large lesions or those involving the GEJ may require esophageal resection.The characteristic features of two patients are shown in The esophageal muscular layer was opened. The tumor was dissected away from the esophageal mucosa by blunt dissection without use of cautery (in order to avoid the risk of perforation due to a delayed burn injury). At this point, the tumor was completely enucleated by SCT assistance and anchoring of mass by silk stitch in case 1. In this case, we were not successful to pass the SCT in early operation before mass excision but was passed it after mass enucleation. The esophageal mucosa of case 1 was perforated during tumor enucleation and repaired by polydioxanone (PDS) 4-0. Next, the esophageal wall and mediastinal pleura were repaired with separate stitches of PDS 4-0 suture. Leakage test confirmed by insufflating methylene blue into the esophageal lumen through the SCT while distal site of operation was obstructed. st and 3rd days post-operation in case 2 and 1, respectively, which were normal (The chest tube (No. 28) was placed along the mediastinal pleura over the reconstruction site. The lung was inflated to ensure complete expansion and the drain was secured to the skin with a silk stitch. Closure was performed with interrupt nylon 3-0 sutures and dressing was applied. Patients had an uneventful post-operative recovery. The naso-gastric tubes (NGT) were removed post-operation. In case 1 due to esophageal perforation fluids were started on day 4 post-operation. Case 2 was operated, identical to procedure of case 1, but mass was detected in middle thoracic esophagus, after passing of SCT 40 F and no esophageal perforation occurred. The patient had good post-operative recovery without problems. Barium swallow was done on 1e normal . Both pa6 They are usually solitary, but multiple tumors have been found in approximately 50% of patients.5,6,8 It has been reported that the male to female ratio of incidence is 2:1 and the age incidence is between 30 to 50 years old.1,3,5 Both our cases were male but age of one of them was out of this reported range. Most symptomatic patients have some degree of dysphagia, pain, pyrosis, weight loss, and weakness.1-6 Differential diagnoses include foreign body, malignant tumors of esophagus include squamous or adenomatous carcinomas or leiomyosarcoma and other benign tumors such as cysts and gastrointestinal stromal tumor (GIST).5Leiomyoma is the most common benign neoplasm of the oesophagus accounting for 1% of all oesophageal tumors.5 Diagnostic work up begins with an upper GI endoscopy which shows a submucosal swelling with normal mucosa over it. Endoscopic ultrasonography (EUS), barium swallow, CT scan can help to differential diagnosis.5,6,8 Surgical enucleation is the accepted treatment for leiomyoma.1 The location of the lesions determines the side of the thoracoscopy: a left one is used for the lower third, whereas for lesions of the middle and upper thirds, a right thoracoscopy gives the best exposure and is widely used.5 The enucleation of leiomyoma through thoracoscopy was reported for the first time by Ohsawa in 1933.2,5Leiomyoma is positive for desmin and actin and negative for CD34 and CD117 which oppositely occurs in GIST.2,5 Everitt and colleagues and Bardini et al., published the first reports of the thoracoscopic approach independently in 1992.2,5 The first published literature comparing open and minimally invasive surgery was presented by Von Rahden.9 The port sites and the number of used trocars are controversial issues. The thoracoscopic approach has been accomplished with seven ,10 six,11 five,12 four,13 and three trocars2,14,15 reported. However a 3-thoracic port technique which was deemed a safe procedure. Moreover, a thoracoscopy in the prone position has also been reported.16Until 1992, the enucleation of esophageal leiomyoma was traditionally performed via thoracoscopy.17 and a balloon dilator,17,18 have been employed and were found to be useful for facilitating the separation of the tumor by promoting progressive expulsion of the lesion from the esophageal wall. Izumi et al. expressed the use of a balloon-mounted esophagoscope as a new technique which called the balloon push-out method.18 This technique was suggested instead of pulling the tumor, which be hard to grasp due to its delicate nature and it was pushed out of the esophageal wall.19Several different techniques have been described to assist extra mucosal enucleation using intraluminal tools. For example, esophageal SCT20 but we never needed intraoperative endoscopy in this method. Indeed, for better exposure, dissection and detect the small mass we recommend use of esophageal SCT 40 F. For leakage test, we used injection of methylene blue by SCT instead NGT. It seems SCT is better than NGT due to large size of SCT. We performed this surgery technique by using 3-ports without problems. Indeed protruding of mass by SCT assistance and hanging of mass by stitch help to reduced port, better dissection and faster operation. Although a successful procedure for large esophageal leiomyoma, which was removed by a thoracoscopic-endoscopic combined submucosal tunneling method was reported recently,5In case 1, we could not pass SCT at first due to large mass. Therefore, esophageal perforation occurred during dissection. If SCT was passed prior to dissection, perforation may not occur. The results of surgical treatment of esophageal leiomyoma have been generally excellent. Mortality rate after enucleation is low and success in relieving the dysphagia is about 100%. Few deaths, were reported following esophageal resection. The morbidity included some instances of either hemothorax or postoperative infection.Conclusively, the described technique is a safe, effective and feasible procedure with decreased hospital stay, operative trauma, reduced postoperative pain, quick recovery, and minute skin scars. Also, we used 3-port technique which is safe and effective. We describe here in a new videothoracoscopic technique to enucleate an esophageal leiomyoma with SCT assistance. The use of the SCT facilitates separation of the tumor mass interfering layers of esophagus, and may help to prevent perforation, and may help to detect of small mass which was not be detectable and operation was faster and more safely.


Adenocarcinoma arising in an ileal diverticulum are very rare. A 66-year-old man was recognized to have high serum CEA level and periappendiceal polycystic tumor in CT findings. Colonoscopy showed no abnormality in the ileocecal mucosa. However, the patient was suspected of appendiceal adenocarcinoma by PET/CT, which revealed FDG uptake with SUVmax of 3.9 in the tumor, and underwent radial surgery by single-incision laparoscopic surgery. Intraoperative findings showed the mass in the mesenterium of the terminal ileum but not the abnormality of the appendix. The resected specimen revealed a cystic tumor of 45 mm on the back side of the intestinal tract. Pathological findings showed that the tumor lesion mainly consisted of mucinous adenocarcinoma was developing from the base of the ileal diverticula. The postoperative process was going well, and the patient left the hospital 14 days after the operation. The recurrence has not been evident 10 months after the operation. Adenocarcinoma arising in an ileal diverticulum has been little reported, since not only small intestinal diverticulum (SID) but also adenocarcinoma in the small intestine are relatively rare . In the A 66-year-old man had been under surveillance for diabetes, hypertension, and dyslipidemia. The patient was recognized to have high serum carcinoembryonic antigen (CEA) level, which let us suppose some digestive malignancies. On physical examination, no mass was palpable in his abdomen, and the patient had no symptoms like abdominal pain, vomiting, and nausea. Computed tomography (CT) revealed periappendiceal polycystic tumor of 40 mm and lymph node swelling of 9 mm neighboring the tumor Fig. . ColonosThus, the patient was suspected to have appendiceal adenocarcinoma and underwent ileocecal resection. We selected SILS. Intraoperative findings showed the mass invading to the testicular artery and vein in the mesenterium of the terminal ileum and that the appendix stayed intact, but not showed the distinct metastasis, peritoneal dissemination, and ascites.We achieved combined resection of the ileocecum and testicular artery and vein by SILS without tumor residual. The operation took a total of 195 min.The resected specimen did not reveal any abnormal lesion on the ileocecal mucosa but a cystic tumor of 45 mm on the back side of the intestinal tract Fig. , b and cSID excluding Meckel’s diverticula is only a few of all gastrointestinal diverticula , and theSmall intestinal adenocarcinoma is also rare, constituting 1 to 2% of all primary gastrointestinal cancers , 11. AdeRecently, advances of examination such as imaging and endoscopy are increasing small intestinal adenocarcinoma in a proper diagnostic rate. However, that is still difficult to diagnose because of absence of specific symptoms and signs and limited method of access to this organ. Among primary malignant neoplasms of the small intestine, carcinoid tumors are the most common histological types (41%), followed by adenocarcinomas (24%), lymphomas (22%), and sarcomas (11%) .For accurate preoperative diagnosis of adenocarcinoma arising in the ileal diverticulum, strict examinations are important. Tsujii et al.  describeThe current authors encountered a case in which adenocarcinoma arising in an ileal diverticulum was performed in operation. Since it is very rare and mostly an asymptomatic disease, early detection and definite diagnosis are so difficult. There is a possibility that a high CEA level becomes a clue to diagnose; however, it needs accumulation of cases to reveal the possibility.


The sellar/juxtasellar region comprises the bone component of the sella turcica,pituitary gland, cavernous sinus, and suprasellar cistern. Abnormalities in thisregion can be attributed to underproduction or overproduction of hormones or tothe neurological signs and symptoms resulting from the compression of adjacentstructures. Magnetic resonance imaging (MRI) is currently the imaging method ofchoice, having supplanted computed tomography. The aim of this study was todemonstrate the common and uncommon imaging aspects of sellar and juxtasellarchanges, which could facilitate the differential diagnosis. We retrospectivelyevaluated the MRI scans of 70 patients with sellar/juxtasellar abnormalitiesfrom didactic files, and report those with more unusual changes, where MRIplayed an important role in diagnosis. All cases were confirmed histologicallyor clinical laboratory. Abnormalities in this region can beattributed to the underproduction or overproduction of hormones or to theneurological signs and symptoms resulting from the compression of adjacentstructures. Virtually any of those factors can lead to disease, ranging from theinnocuous to the severeAlthough pituitary adenomas are the most common lesions in the sellar/juxtasellarregion, lesions originating in other structures can also affect the region,complicating the diagnosis. We retrospectively evaluated 70 cases ofsellar/juxtasellar abnormalities, confirmed by histopathology or on the basis ofclinical and biochemical findings, in order to describe the common and unusualaspects of these alterations that aid in the differential diagnosis.(6).Pituitary adenomas represent the most common sellar lesions. They are classified, bysize, as macroadenomas (≥ 10 mm) or microadenomas (< 10 mm) and, byhormone production, as secreting or nonfunctioning(9).On MRI, adenomas typically present hypointense signals in T1-weighted sequences andsignals of variable intensity in T2-weighted sequences. One important aspect isthat, due to those hypointense signals, the majority of adenomas can be detected inT1-weighted sequences, which makes such sequences the most important of the MRIprotocol for the evaluation of this alteration, given that it allows the diagnosiseven without contrast administration. Another important aspect is that most adenomaspresent post-contrast enhancement slower than does the normal parenchyma , making (6).Among secreting adenomas, the most common are prolactin producers (prolactinomas). Inmost cases, prolactinomas can be treated exclusively with dopaminergic agonists,although such treatment can result in alterations to the imaging aspects , which m(5). Large adenomas areusually heterogeneous, containing cystic areas resulting from cystic degeneration ornecrosis, and can occasionally develop infarction or hemorrhage, due to poorvascular supply(8).Macroadenomas sometimes extend beyond the boundaries of the sellar region, invadingthe cavernous sinus, sphenoid sinus, or clivus, as well as compressing the opticchiasm and enveloping the internal carotid artery . On MRI,(9).Craniopharyngiomas are slow-growing epithelial neoplasms that originate from theremnant of the craniopharyngeal duct and account for 3-5% of intracranial neoplasms.Their incidence shows two peaks, the first occurring between 10 and 14 years of ageand the second between the fourth and sixth decade of life. Althoughcraniopharyngiomas are suprasellar in origin, approximately 50% extend into thesellar region. The typical appearance includes solid-cystic components andcalcifications(8).The classic, adamantinomatous, type of craniopharyngioma has a cystic appearance andcontains heterogeneous nodules. The least common, squamous papillary, type has apredominant solid component. In T2-weighted MRI sequences, the cystic componentshows a hyperintense signal, whereas the solid components show heterogeneoussignals. After contrast administration, the solid portions show intenseheterogeneous enhancement and there is enhancement of the cystic walls .Although macroadenomas with pituitary apoplexy  and Rath(6).Rathke's cleft cysts are benign, often asymptomatic, lesions of the sellar region,most often being intrasellar. On MRI, they usually show a hyperintense signal inT2-weighted sequences, whereas they can show hyperintense or hypointense signals onT1-weighted sequences, depending on their protein content . The dif(8).Sellar meningiomas account for 20-30% of all intracranial meningiomas. On MRI, sellarmeningiomas show an isointense signal in T1-weighted sequences and an isointense orhyperintense in T2-weighted sequences, as well as early enhancement, usuallyaccompanied by the dural tail sign .When th(5).Aneurysms of the sellar region typically originate from the cavernous or supraclinoidportion of the internal carotid artery, accounting for up to 10% of all cerebralaneurysms. Their diagnosis is made more easily with MRI than with CT, because theformer can reveal a flow void, due to the rapid luminal flow, and heterogeneoussignal intensity in areas with slow, turbulent flow . HoweverFigure11(9).Hypothalamic hamartomas consist of ectopic foci of neural tissue (gray matter),typically located in the tuber cinereum and mammillary bodies. They typicallymanifest as an increase in the size of the tuber cinereum. On MRI, hypothalamichamartomas present signals that are, in comparison with that of the gray matter,isointense in T1-weighted MRI sequences  and isoi(10).Hemangiomas constitute vascular malformations found in various organ systems,including the central nervous system. When extracerebral, they can originate fromthe cavernous sinus or from the adjacent tissues. Like hepatic hemangiomas,hemangiomas in the sellar region manifest on MRI as well-defined masses withhypointense or isointense signals in T1-weighted sequences and markedly hyperintensesignals in T2-weighted sequences ,initial(12).Inflammation of the pituitary gland, or hypophysitis, comprises a complex group ofdiseases, with two main histological forms: lymphocytic ; and granulomatous . Because it is practically impossible to distinguishbetween the two forms on the basis of the radiological findings, the clinicalhistory has great value in the differential diagnosis. On MRI, hypophysitis presentsas thickening of the pituitary gland in combination with intense contrastenhancement, as shown in Figure16(14).Normally, the neurohypophysis is located within the sella turcica, posterior to theadenohypophysis. It consists of the terminal axons of neurons projected from thehypothalamus, differentiated to store oxytocin and the antidiuretic hormone. Ectopicneurohypophysis occurs in three situations: when there is compression of thepituitary stalk by an expansile lesion ; when a The great number of lesions that can affect the sellar/juxtasellar region requiresthat radiologists not only possess knowledge of the anatomy and the contents of thisregion but also familiarize themselves with the various possible aspects of suchlesions. In most cases, the application of such knowledge can lead to an accurateetiological diagnosis.


A 74-year-old woman with a history of membranous glomerulonephritis and a recentdiagnosis of mediastinal adenopathy was admitted to the emergency department with acuteheart failure. She complained of progressive dyspnea and weakness in the last week.Physical examination revealed hypotension, tachypnea, jugular vein distention, anddesaturation. The most relevant laboratory findings were: anemia, lymphocytopenia,lactic acidosis, and increased lactate dehydrogenase. An electrocardiogram showed rapidatrial fibrillation and low-voltage QRS complexes. An echocardiogram revealed severepericardial effusion and diffuse heterogeneous thickening of the ventricular and atrialwalls. The patient required mechanical ventilation and inotropic support. Therapeuticpericardiocentesis was performed without clinical improvement. Cardiovascular magneticresonance imaging (CMR) showed septal bounce (compatible with restrictive physiology)and a heterogeneous isointense mass surrounding the ventricular and atrial walls withlate gadolinium enhancement of the myocardium and hypoenhancement of the tumor , compatiSecondary involvement of the myocardium in patients with systemic lymphoma is relativelyfrequent (around 30% in disseminated non-Hodgkin lymphoma) whereas primary cardiaclymphoma is rare (1-2%). We present a case of acute heart failure with restrictivephysiology secondary to cardiac lymphoma. In our experience, CMR was key to the finaldiagnosis.


Thymic tumors are rare mediastinal tumors that can present with a wide variety of symptoms. They can cause distant manifestations and are frequently associated with paraneoplastic syndromes. In our case, we describe the evolution of a 68-year-old male whose first manifestation was thrombosis of the inferior vena cava and renal veins. Thrombosis of large abdominal veins is rare, especially without being associated with any other comorbidity or risk factors. Thymic tumors are rare mediastinal tumors that are considered orphan diseases due to their low prevalence. In the United States, they have an overall incidence of 0.13 cases per 100,000 person-year . Thymic Thymic tumors are often associated with paraneoplastic syndromes, with myasthenia gravis being the most frequent , 4. The When a patient is diagnosed with a mediastinal mass, the differential diagnosis must include thymoma, lymphoma, thymic carcinoma, thymolipoma, germ cell tumors, and lung metastases but also thymic cysts, lymphangiomas, and aortic aneurysms , 10.We present the case of a 68-year-old male who is admitted to the regional hospital reporting pain in the upper abdomen.The patient had no known history of any illness or any surgical intervention and did not take any medication at home. He was an ex-smoker (20 packs/year) but had quit smoking almost 25 years ago. His complaints started approximately a week before and progressively worsened until he decided to go to the hospital.N = 25–100 U/L) and lipase = 97 U/L (N = 0–60 U/L). EKG and chest radiography showed no modifications; however, the abdominal ultrasound revealed a right renal mass with modifications of the renal parenchyma and also modification of the pancreas ecostructure. The patient is transferred to our clinic with suspicion of right renal mass and pancreatitis.The initial bloodwork showed slightly enhanced amylase = 174 U/L  and 11.1 g/dl of hemoglobin (N = 12–16 g/dl). He also had hypoalbuminemia, 3.1 g/dl (N = 3.5–5.1 g/dl), and proteinuria of 2.28 g/24 h.The patient'sThe CT scan of the abdomen and pelvis proved to be an indispensable tool for the diagnosis. It showed thrombosis of the left and right renal veins and of the portal vein  with enlµg), present PDF, and a protein S deficiency (45%). However, the protein S deficiency alone is not enough to cause the massive vein thrombosis. We also tested for a genetic hypercoagulability state, which included tests for factors II, V, MTHFR, and PAI genes; all tests came back negative.The abdominal CT excluded the suspicion of a renal tumor. Having ruled out the most likely suspect that can cause renal vein thrombosis, we started to look for more unusual causes. All the tumor markers came back negative. But the extended coagulation testes revealed elevated D-dimers . The operation was uneventful and the patient remained under observation for almost two weeks.The patient was scheduled for surgical intervention. Sternotomy was performed followed by tumorectomy of the anterior mediastinal mass and thymomectomy of the remaining thymus tissue. The thymic tumor mass was 7/5/3 cm in size and was encapsulated being well differentiated from the surrounding tissue. Histopathology showed anWhen he returned a month later for a follow-up, he was symptom-free and was feeling like a new man. The dyspnea and abdominal pain were gone and he was no longer feeling fatigued. An abdominal ultrasound revealed partial regression of the inferior vena cava thrombosis. The patient remained on oral anticoagulants and was followed up every 3 months. After one year, a control CT scan showed complete regression of the inferior vena cava and renal vein thrombosis . He was Thymic tumors are rare mediastinal tumors and, most often, they are discovered by using imaging techniques (CT) because of nonspecific symptoms. However, in our case, thrombosis of the inferior vena cava and of both renal veins as the first manifestation of such tumors has never been described in the literature. The nonspecific symptoms of the patient  were what determined us to closely investigate the patient and perform an echography and, afterwards, a CT scan. But to our surprise, the cause of his symptomatology was neither abdominal nor pulmonary but mediastinal. The thymic carcinoma manifested itself as a paraneoplastic syndrome which induced a hypercoagulability state that led to the thrombosis of the inferior vena cava and renal veins. The modifications in the patient's blood can be attributed to the thrombosis which affected the connected organs , except for the anemia and the thrombopenia which might have been another paraneoplastic manifestation. The patient's hereditary thrombophilia (the protein S deficiency) also played an important part and was probably the reason why the first paraneoplastic manifestation presented as a thrombosis of major veins.The initial therapeutic attitude after the diagnosis of inferior vena cava and renal vein thrombosis was the correct one. Firstly, heparin anticoagulation was initiated and was maintained until oral anticoagulants could be administered and had taken effect. This prevented the thrombosis from advancing and even managed to slightly regress in its spread. The thoracic surgeon performed ideal tumorectomy and according to the Masaoka-Koga staging system (1994), the thymic tumor was of stage I  . For thiOne month after the excision of the tumor, the thrombosis of the inferior vena cava and of the renal veins had decreased (on Doppler echography). The symptomatology had completely disappeared and the patient remained on oral anticoagulants. He was followed up every 3 months and was scheduled for a CT scan after one year. When the follow-up CT scan was performed it showed complete regression of the inferior vena cava and renal vein thrombosis. Being symptom-free and with no sign of thrombosis, the patient was taken of oral anticoagulants.Thrombosis of the inferior vena cava and renal veins is a rare manifestation that can be caused by a handful of diseases. The list is short, with the most frequent being renal cancer. In our case, it was a manifestation of a paraneoplastic syndrome caused by thymic carcinoma. Although rare, thymic carcinomas can cause distant manifestations and are discovered when nonspecific symptoms start to emerge. In our case, the inferior vena cava and renal vein thrombosis without any local or apparent cause was what determined us to search further and investigate distant sites and rarer causes for this manifestation.


Atlantooccipital assimilation is a partial or complete congenital fusion between the atlas and the base of the occiput. Most patients with atlas assimilation are asymptomatic, but some may present with neurological problems such as myelopathy. We present the case of a 37-year-old woman who presented with neck and occipital pain, episodic neck stiffness, and dizziness. Medical imaging revealed complete atlantooccipital assimilation associated with basilar invagination, atlantoaxial subluxation, and predisposing anterior spinal cord compression. The patient was treated non-operatively with medications, cervical interferential therapy, and a rigid cervical orthosis. The craniovertebral junction (CVJ) comprises of the occiput, atlas, and axis . OcciputA 37-year-old lady presented to our clinic with dull, aching pain in the neck and occiput since a fortnight, which began after lifting a heavy object. The pain was sudden in onset, dull-aching in character and was associated with episodic neck stiffness. The pain radiated to both shoulders. A general examination of the patient was unremarkable except for a low hairline and a short neck. There were no signs of anterior (negative Babinski and Hoffman signs) or posterior spinal cord compression . Her cranial nerve function was normal. Her sensory and motor examinations were normal in both upper and lower extremities. Deep tendon reflexes were physiological and symmetrical. Ambulation was normal. A routine radiograph was obtained which was initially falsely interpreted as “complete absence of atlas” Figure . This prompted further medical imaging. A multiplanar, multiecho magnetic resonance imaging (MRI) scan and a computed tomography (CT) scan with 3D reconstruction were performed. The imaging revealed complete atlantooccipital assimilation - complete fusion of the lateral masses of the atlas with the occipital condyles Figures -4.A sagittal CT revealed an increased anterior atlanto-dens interval measuring 7 mm suggestive of atlantoaxial instability Figure .The odontoid process of the axis and the anterior arch of the atlas were located above the Chamberlain line, drawn between the posterior pole of the hard palate and opisthion, suggestive of basilar invagination Figure .The Welcher basal angle was normal measuring 134 degrees. There was a reduction in the craniovertebral angle (136 degrees), suggestive of predisposition to anterior spinal cord compression, which was seen as a hyperintense signal intensity in the T2 weighted sagittal and axial MRI at the cervicomedullary junction, at the lower border of the C2 vertebra Figures -9.Since signs of cervical myelopathy and cord compression were absent, the patient was offered non-surgical management with medications, cervical interferential therapy, and a hard cervical collar, Miami- J Collar . All symptoms resolved within 10 days of treatment. At eight months follow-up, the patient continues to remain asymptomatic and uses the hard cervical collar most of the time, as advised.Atlas assimilation or occipitalisation is a partial or complete congenital fusion between the atlas and the occipital bone, ranging from a complete bony fusion to a bony bridge, or a fibrous band uniting one small area of the atlas to the base of the occiput . The incWe propose that all patients with atlantooccipital assimilation , who do not present with signs of cervical cord compression, must be offered an unbiased non-operative treatment plan before considering surgical intervention.


Maxillary cysts, including the cysts lined by respiratory epithelium, can present a diagnostic challenge. We report an unusual case of a maxillary cyst on an endodontically treated tooth #16, in which the cavity was totally lined by a respiratory epithelium. The patient, a 35-year-old male, presented with a generalized chronic periodontitis and complained of a pain in the tooth #16 region. A periodontal pocket extending to the root apices with pus coming out from the gingival was found. A combined endodontic periodontal was observed on a panoramic radiography. CBCT-scan revealed a well-circumscribed radiolucent lesion at the apices of the distobuccal root of the 16. A communication with the right maxillary sinus cavity and a maxillary and ethmoidal sinusitis were also observed. The lesion was removed and histological examination revealed a cyst lined exclusively by respiratory epithelium. Ciliated and rare mucous cells were also observed. The diagnosis could evoke a surgical ciliated cyst mimicking the radicular cyst but the patient has no previous history of trauma or surgery in the maxillofacial region. It could also be an unusual radicular cyst in which the stratified squamous epithelium was destroyed by inflammation and replaced by a respiratory epithelium of the maxillary sinus. The maxillary cysts lined by pseudostratified columnar epithelium (respiratory epithelium) can show different pathologic conditions and hinder the diagnosis, thus challenging the clinician and pathologist. These lesions include mucocele of the maxillary sinus and surgical ciliated cyst. The latter develops after a surgical procedure, such as maxillary sinus surgery , orthognathic surgery, and trauma caused by dental extraction –3. In soWe report the case of an unusual cyst on the maxillary right first molar (tooth #16) region, in which the cavity was totally lined by respiratory epithelium. Interestingly, no previous history of surgical treatment of the sinus, trauma, or dental extraction was observed.We report the case of a 35-year-old healthy male who consulted our Oral Surgery, Implantology and Pathology Emergency Department with a chief complaint of pain in the posterior maxillary right region. He reported no history of trauma or surgery in the maxillofacial region and was not known for recurrent sinusitis. The clinical examination revealed a generalized periodontitis. Tooth #16 presented a periodontal pocket extending to the root apices with pus coming out from the gingival sulcus. The mobility of the teeth was grade 3, the vitality was negative, and the percussion was positive. The patient was not swollen and did not have any systemic symptomatology. A severe generalized horizontal bone loss associated with local vertical lesions and furcation involvement in the first quadrant was seen on the panoramic radiography. The diagnosis of a combined endodontic periodontal lesion was inferred. The Cone-Beam Computed Tomography (CBCT) revealed a communication of 6 mm in the anteroposterior axis, of the apices of the root of tooth #16 with the right maxillary sinus cavity. A well-circumscribed lesion that protrudes in the maxillary sinus is described at the apices of the distobuccal root of tooth #16. The right maxillary and anterior ethmoidal sinus are opacified and the peripheral mucosa thickened. A deviation of the nasal septum on the right is also outlined Figures . Tooth #In this particular case, the differential diagnosis of the maxillary cyst in which the cavity is fully lined by respiratory epithelium should include the surgical ciliated cyst or an unusual radicular cyst. In both cases, a resorption of the maxillary sinus floor can happen and cause the communication between the cyst and the maxillary sinus cavity.The surgical ciliated cyst is a cystic lesion which develops after a surgical procedure such as maxillary sinus surgery , orthognathic surgery, and trauma caused by dental extraction. They are locally aggressive and may resemble a tumour. Bone expansion, pain, or discomfort in the maxillary region and fistulization in some cases can be present. Radiographically, they can be unilocular or multilocular radiolucent or radiopaque with a well-defined margin and surrounding sclerotic edge in the floor of the maxillary sinus, with or without bony perforation –3. In soAmong the odontogenic cysts of the jaws, the radicular cyst  is the most common. Radiographically, it shows a well-defined radiolucent area around the apex of a tooth root. Although it is usually asymptomatic, the cyst can cause pain after a secondary infection. The cyst arises from an inflammation at the root apices of a tooth secondary to pulp necrosis, which stimulates cell proliferation from the remains of odontogenic epithelial rests of Malassez. Caries and trauma can be responsible for radicular cyst formation  HistologNevertheless, the cystic cavity can in some cases be partially or totally lined by a respiratory epithelium characterized by ciliated pseudostratified columnar epithelium with mucous cells –7. The eWe reported a cyst case on the maxillary right first molar (tooth #16), in which the cavity was totally lined by respiratory epithelium. This lesion could correspond to a special surgical ciliated cyst mimicking a radicular cyst  but the


Pheochromocytomas are rare catecholamine-producing neuroendocrine tumors. Hypertension secondary to pheochromocytoma is often paroxysmal, and patients occasionally present with sudden attacks of alternating hypertension and hypotension. Spontaneous, extensive necrosis within the tumor that is associated with catecholamine crisis is an infrequent complication of adrenal pheochromocytoma, but its pathogenesis remains unclear.A 69-year-old Japanese man developed acute-onset episodic headaches, palpitations, and chest pains. During the episodes, both marked fluctuations in blood pressure (ranging from 40/25 to 300/160 mmHg) and high plasma levels of catecholamines were found simultaneously. Radiological findings indicated a 4-cm left adrenal pheochromocytoma. These episodic symptoms disappeared within 2 weeks with normalization of plasma catecholamine levels. Two months later, the patient underwent adrenalectomy. Microscopic examinations revealed pheocromocytoma with a large central area of coagulative necrosis. The necrotic material was immunohistochemically positive for chromogranin A. Granulation tissue was adjacent to the necrotic area, accompanied by numerous hemosiderin-laden macrophages and histiocytes with vascular proliferation. Viable tumor cells, detected along the periphery of the tumor, demonstrated pyknosis, and the Ki-67 labeling index was 2 % in the hot spot. No embolus or thrombus formation was found in the resected specimen harboring the whole tumor. The Pheochromocytoma of the Adrenal gland Scaled Score was 2 out of 20. The patient’s postoperative course was unremarkable for > 7 years.Presumed causal factors for the extensive necrosis of adrenal pheochromocytoma in previously reported cases include hemorrhage into the tumor, hypotension induced by a phentolamine administration, embolic infarction, high intracapsular pressure due to malignant growth of the tumor, and catecholamine-induced vasoconstriction. In the present case, histopathological and clinical findings suggest that under conditions of chronic ischemia due to catecholamine-induced vasoconstriction, an acute infarction occurred after sudden attacks of alternating hypertension and hypotension. Over the subsequent 2 weeks, repetitive massive release of catecholamines from the infarcts into circulation likely accelerated infarction progression by causing repeated attacks of alternating hypertension and hypotension and resulted in the large necrosis. This case highlights the need for physicians to consider acute spontaneous tumor infarction accompanying episodic catecholamine crisis as a rare but severe complication of pheochromocytoma. Pheochromocytomas are rare catecholamine-producing neuroendocrine tumors that arise from chromaffin cells of the adrenal medulla or extra-adrenal paraganglia . The cliSpontaneous, extensive necrosis within the tumor that is associated with acute and severe clinical manifestations of massive catecholamine release, including acute abdomen, chest pain, or even shock, is an infrequent complication of adrenal pheochromocytoma –13. HoweHere, we report a case of a patient who exhibited a large coagulative necrosis of an adrenal pheochromocytoma associated with acute attacks of alternating hypertension and hypotension, on which a detailed histopathological analysis was performed.A 69-year-old Japanese man was admitted to our hospital in July 2009 because of 1 week of episodic headaches, palpitations, and chest pains. He had a family history of type 2 diabetes mellitus (T2DM) in his mother. The patient drank 360 mL/day of sake. He had a smoking history of one pack of cigarettes/day from 20 to 60 years old. Our patient was diagnosed with T2DM at the age of 60 years at our hospital and started treatment with diet therapy . He was started with the oral antidiabetic drug nateglinide (90 mg/day) at the age of 63 years, and subsequently had fair glycemic control with a mean glycated hemoglobin (HbA1c) of 7 % (reference range: 4.6–6.2 %) with no diabetic retinopathy. He had never had hypertension and had normal blood pressure (BP), with a systolic and diastolic BP of around 120 mmHg and 70 mmHg, respectively. He showed acute-onset episodic headaches, palpitations, and chest pains at rest in June 2009, which lasted for 10 to 20 minutes and occurred several times per day, and was admitted to our hospital.On physical examination performed at presentation with no symptoms, he was 161 cm tall, weighed 55 kg, and his body temperature and blood pressure were 36.2 °C and 106/58 mmHg, respectively. No thyroid struma, chest rales, heart murmurs, abdominal tenderness or mass, or peripheral edema was detected. An electrocardiogram showed a normal sinus rhythm, with a heart rate (HR) of 65 beats/minute and no abnormal wave form. Chest X-ray showed no abnormalities with a cardiothoracic ratio of 46 %. Laboratory findings showed a normal complete blood count, normal liver and renal functions, normal balance of serum electrolytes, and high HbA1c value (7.6 %).On day 2 of admission, our patient again experienced episodic headache, palpitation, and chest pain. During the episode, an electrocardiogram showed nonsustained ventricular tachycardia. His symptoms resolved within 20 minutes after sublingual administration of nitroglycerin (0.3 mg), which was administered as diagnostic therapy for ischemic heart disease, but it was unclear whether the drug was responsible for the observed improvement. Although an electrocardiogram did not show significant changes in the ST-T segment, the possibility of acute coronary syndrome remained because of his noticeable chest symptoms, and our patient received cardiac catheterization on the same day. Coronary arteriography showed no significant coronary artery lesion, and left ventriculography showed normal ventricular function with a left ventricular ejection fraction of 69 %. However, during the test, he again experienced episodic headache, palpitation, and chest pain accompanied by marked fluctuations of systemic BP (ranging from 40/25 mmHg to 300/160 mmHg). Catecholamine levels  in his plasma taken during the attacks of alternating hypertension and hypotension were high. Neck, chest, and abdominal computed tomography (CT) performed following cardiac catheterization showed a 4-cm left adrenal tumor without any other mass lesion. He was believed to have possible pheochromocytoma, and treatment with an alpha 1 adrenergic blocker, oral doxazosin, (2 mg/day) was begun.Urinary excretion of catecholamines  and metanephrines  in a 24-hour urine sample collected from days 2 through 3 of admission were high. An iodine-123 metaiodobenzylguanidine (MIBG) scan detected no pathological accumulation in the patient’s whole body, including the region of the left adrenal gland. Gadolinium-enhanced T1-weighted magnetic resonance imaging (MRI) indicated altered intensity in a 4-cm left adrenal tumor, and an area of high intensity was found along the periphery of the tumor, which persisted until the delayed phase Fig. , b. TakeOur patient underwent laparoscopic left adrenalectomy in September 2009. The histopathological features of the tumor were consistent with those of intra-adrenal paraganglioma (pheochromocytoma)  The patient discontinued oral doxazosin just after surgery and had no recurrent episodic headache, chest symptoms, or labile BP. His T2DM was treated with continued oral nateglinide, with a mean HbA1c of 7 %. His postoperative course during close follow-up for > 7 years has been uneventful without local or distant recurrence of pheochromocytoma.An elderly Japanese patient exhibited spontaneous large necrosis of adrenal pheochromocytoma that manifested as 2 weeks of episodic headaches, palpitations, chest pains, and rapid fluctuations of hypertension and hypotension. Our patient underwent laparoscopic adrenalectomy 2 months after the episodes, and a diagnosis of pheochromocytoma was confirmed by immunohistochemistry. Light microscopic examination revealed a large coagulative necrosis in the tumor (Fig. Presumed causal factors for the extensive necrosis of adrenal pheochromocytoma in previously reported cases include hemorrhage into the tumor , 12, 13,Many previously reported cases of extensive necrosis in adrenal pheochromocytoma showed spontaneous remission of catecholamine crisis even before they underwent surgical removal of the tumor , 12, 13.Patients with extensive necrosis of adrenal pheochromocytoma often present with abdominal pain , 13, 17.Most previously reported cases of spontaneous large necrosis of adrenal pheochromocytoma without rupture were shown to be benign , 7–13, bIodine-MIBG scintigraphy, in combination with anatomical imaging such as CT or MRI, has been used as a diagnostic tool for detecting chromaffin cell tumors in patients with biochemically confirmed pheochromocytoma . The senHistopathological and clinical findings suggest that under the conditions of chronic ischemia within the tumor due to catecholamine-induced vasoconstriction, an acute infarction occurred after sudden attacks of alternating hypertension and hypotension in our patient. Over the subsequent 2 weeks, a repeated massive release of catecholamines from the infarcts into the circulation probably accelerated the progression of infarction by causing episodic attacks of alternating hypertension and hypotension, resulting in the extensive necrosis of adrenal pheochromocytoma. This case highlights the need for physicians to consider acute spontaneous tumor infarction accompanying episodic catecholamine crisis as a rare but severe complication of pheochromocytoma.


CT imaging. This case also highlights the need for immunohistochemical analysis of unusual tumor deposits facilitating appropriate treatment.Colonic metastases from lung cancer are rare . Presentation of an abdominal mass in the setting of a new lung cancer diagnosis should prompt complete evaluation including endoscopic and  CT imaging confirmed a colonic lesion during staging investigations of a 56‐year‐old female smoker with lung adenocarcinoma and mediastinal lymphadenopathy Fig. . ImmunohGF: drafted submission and submitted case report for publication. EO: performed literature review and assisted with drafting of submission. MF: obtained and prepared radiology and endoscopic images for case report. CR: is respiratory physician responsible for diagnosing and managing patient's lung adenocarcinoma. FB: is pathologist who reported on colonic biopsy samples and who obtained pathology slides contained in this case report. KB: is general surgeon responsible for investigating patient's colonic lesion through performing colonoscopy and obtaining colonic biopsy samples, assisted with drafting of submission and literature review.None declared.


Dear Editor,A 68-year-old man presented with a complaint of dyspnea on moderate exertion, andphysical examination revealed stridor. The patient reported having previously beentreated for chronic obstructive pulmonary disease and adenocarcinoma of the prostate,the latter having been treated with 39 radiotherapy sessions. He was a former smokerwith a smoking history of 150 pack-years (3 packs/day for 50 years), having quit 4 yearsprior. We performed contrast-enhanced computed tomography (CT) of the neck and chest,which showed an expansive, well-defined nodular mass in the distal trachea, near thecarina, without enhancement or signs of invasion of the tracheal walls  and 2. B-4. Less common still are tracheal tumors, which account for only 1-2% ofall respiratory tract tumors,6, affecting mainly the lower third ofthe tract. Such tumors can belocally invasive, adenoid cysticcarcinoma and squamous cell carcinoma being the malignant tumors most often affectingthe trachea,6,8. The most commonsymptoms are related to airway obstruction, dyspnea being the most common, and becomemore evident when the tracheal lumen is narrowed by more than 75%,9. Other symptoms include cough, dysphonia, hoarseness, hemoptysis,stridor, dysphagia, nasal obstruction, epistaxis, rhinorrhea, ear pain, weight loss, andcyanosis.Diseases involving the trachea or the main bronchi are not common,8,10. Involvement of thelarynx, hypopharynx, cervical glands, esophagus, cervical lymph nodes, middle ear, andmastoid is rare, and trachealinvolvement is even rarer,11-13, occurring in only 3% of all extramedullaryplasmacytomas. As of 2005,only 15 cases of solitary extramedullary plasmacytoma of the trachea had been reportedin the medical literature. Itprimarily affects men between 50 and 60 years of age, with a male/female ratio rangingfrom 3:1 to 5:1,8. Progression to multiple myeloma is considerably lessfrequent than is solitary plasmacytoma of the bone.Extramedullary plasmacytoma of the trachea is a rare plasma cell malignancy (accountingfor only 4% of plasma cell tumors), having been described in soft tissues outside thebone marrow, involving the submucosal lymphoid tissue, and occurring at differentlocations, especially in the upper airways, most often in the paranasal sinuses ornose. A CT scanallows the lumen, airway wall, and mediastinal structures to be evaluated. Multiplanarreconstructions are useful for assessing the type, degree, and longitudinal extent ofthe airway narrowing as well as the location of the tumor and its distance from thecricoid cartilage and carina,7. Bronchoscopy correlates well with CTand can be used in order to resect the lesion,7. The diagnosis is madethrough histological and immunohistochemical studies,14. There was onereported case in which the tracheal plasmacytoma was identified as an incidental findingon positron emission tomography/CT.The treatment can be surgical resection alone, radiotherapy alone, requiring annualmonitoring, or a combination of the two,13,14. There is local recurrence in approximately 30% ofcases and metastasis in 15-40%.In ultrasound of the neck, tracheal lesions, especially those located anteriorly, can bevisualized clearly


Background. Solitary fibrous tumor (SFT) is a fibroblastic mesenchymal tumor that was initially described from the pleura but currently arises at almost every anatomic site. It is usually benign, and surgical resection is curative. SFT involving the perineum is extremely rare. This is the third case report of a perineal SFT in the literature. Case Presentation. We reported an uncommon case of a 64-year-old man presenting with a huge perineal mass that started growing 3 years before his arrival in our service. He was asymptomatic. A contrast-enhanced CT scan revealed a heterogeneous well-circumscribed perineal mass with soft-tissue density. Invasion of the surrounding organs, distal metastasis, and lymph node swelling were absent. The complete resection of mass was done successfully. The specimen was a 23.0 × 14.0 × 8.0 cm encapsulated tumor. Mass weight was 1,170 g. After pathological analysis, we confirmed that the mass was a solitary fibrous tumor. The diagnosis was based on clinical findings and histological morphology and immunohistochemistry study. Conclusion. SFTs are usually indolent tumors with a favorable prognosis. The perineal location is extremely rare. Complete resection of the mass is the treatment of choice. Solitary fibrous tumor (SFT) is a rare fibroblastic mesenchymal tumor initially described in the pleura and lately documented at almost every anatomic site and organ , includiSFTs accounted for less than 2 percent of all soft-tissue tumors cases presenting to a large referral center in one series, demonstrating how rare this neoplasm is [About 30 percent of SFTs arise in the peritoneal cavity, retroperitoneal soft tissue, or pelvis  and they constitute the largest site-related group in most series of extrapleural SFTs [The most common presentation of soft-tissue SFT is a painless mass , 4. If tThere are few cases of this tumor arising in perineum reported in the literature. We present here a case of a large perineal mass revealed to represent a SFT.A 64-year-old man presented in our urological service complaining of a huge mass in his perineum. He described a slow-growing, painless mass, and he noticed it the first time 3 years ago . He had The CT scan showed a bulky, lobulated, hypervascularized, and heterogenous mass in soft tissue of the perineum measuring 18.0 × 12.0 × 7.7 cm (860 cc) . CT of tHe was submitted to excision of the mass Figures –5. The tAfter the surgery, the patient was sent to ICU prophylactically due to his medical past of myocardial infarction, where he remained for only one day. No blood transfusion was necessary. The patient presented no complaints during the length of stay. No kind of peri- or postoperative complication occurred. He had a length of hospital stay of 4 days.Macroscopic examination of the tumor demonstrated a 23.0 × 14.0 × 8.0 cm, well-defined, firm mass without necrosis and with a pale cut surface.Pathological findings were of a solitary fibrous tumor which had variable cellularity, moderate to high, with a morphological pattern predominantly fusocellular without formation of bundles, in a haemangiopericytic arrangement, with sclerotic stroma. Microscopically, the mass was completely encapsulated. The neoplasm was composed of small, tightly packed, ovoid to spindled cells and ill-defined eosinophilic cytoplasm. Tumor cell nuclei showed open chromatin, giving a vesicular appearance. The tumor cells were arranged in a haphazard pattern around thin walled ramifying blood vessels in a fibrous stroma. There were mastocytes along the tumor. In some areas, the tumor showed atypia and pleomorphism. Mitotic activity was 1-2 mitosis per 10 HPF. Necrosis was not present.The tumor showed strong and diffuse staining for CD34, CD99, bcl-2, and STAT- 6; other markers, such as pan-cytokeratins, desmin, alpha-smooth muscle actin, DOG-1, C-Kit, Glut-1, and S-100 protein, were negative and had a low Ki67 (positive in 15% of neoplastic cells). EMA was focally positive. Based on morphological and immunohistochemical findings, a diagnosis of solitary fibrous tumor was determined Figures –11.We have one year of follow-up after surgery. He was evaluated in outpatient setting and he was very satisfied, without any complaints. A control CT scan was done one year after resection showing no recurrence . The extSFTs are rare mesenchymal spindle cell neoplasms and important differential diagnosis in all anatomical locations. The first cases of primary SFT were published by Klemperer and Rabin in 1931 . They haPerineal location is extremely rare. The case described here is the third one reported in the literature. The first one was described by Suster et al.  in a casLesions tend to present as slowly growing mass or nodule as we could see in our case, or it can present with symptoms due to mass or pressure effects on adjacent structures. Intra-abdominal SFTs can present as a palpable mass, accompanied by pain and weight loss. Sometimes, SFTs occur in urinary tract and patients may complain of urinary symptoms including dysuria, urinary retention, hematuria, and nocturia motivating them to go to a urological clinic . GastroiRarely, SFTs can develop paraneoplastic syndromes, most commonly hypoglycemia. It can happen with SFTs arising in all sites. Hypoglycemia can occur due to tumor secretion of large insulin-like growth factor II (IGF2) .CT or MRI can help to identify SFTs and often reveals proliferation of fibrous tissues as well as tumor and adjacent tissue details to facilitate decisions for surgical tumor removal. Radiologically, SFTs are often large, well-defined, lobulated, solid, and vascular masses, often with prominent feeding vessels or sometimes a visible fatty component, which displace adjacent structures . RadioloIn our case, bleeding during surgery was unremarkable; no red blood cells transfusion was necessary; but there are some cases reported in the literature of massive hemorrhage during surgical resection due to the hypervascular nature of SFTs .Complete resection is required for full histopathologic evaluation. Fine needle aspiration biopsies and core biopsies are inadequate. Diagnosis is based upon recognition of typical morphologic features in conjunction with a characteristic immunophenotype.Grossly, tumors are well circumscribed, with a fibrous pseudocapsule or serosal lining. Large tumors may present hemorrhage, necrosis, or calcification . In the Immunohistochemical analysis is paramount during differential diagnosis with other soft-tissue tumors. Markers such as CD34, CD99, BCL2, and LSD1 are commonly applied, although they are not sufficiently sensitive or specific. But new markers such as STAT6, used in our case, are more accurate in the diagnosis of SFT. Ouladan et al.  showed iAgain, the majority of SFTs have an indolent course with low risk of local recurrence or metastasis. A minority of tumors recur locally or metastasize to distant sites. Some histologic features have been used to attempt to discover malignant SFTs: high mitotic activity; presence of necrosis or hemorrhage; increased cellularity; nuclear pleomorphism; stromal infiltration beyond pseudocapsule or vascular invasion. en bloc surgical resection to negative margins is the mainstay of therapy for all localized SFTs [Completezed SFTs . R0 reseThere is no consensus about SFTs follow-up, but continued long-term surveillance is advised because of the indolent natural history and possibility of late recurrence up to 20 years after initial treatment .Solitary fibrous tumors have usually an indolent course with a favorable prognosis. The perineal location is extremely rare. Complete resection of the mass is the treatment of choice and it is associated with high rates of success and low recurrence.


Ameloblastomas are rarely found in pregnant women, with only two cases reported in the scientific literature. We report the first case of ameloblastoma in a pregnant woman in Brazil.A 27-year-old white woman, 12-weeks pregnant, presented with a large mass in her right posterior mandible. Panoramic radiography revealed a lesion involving her mandibular right first molar with displacement of her mandibular right third molar and impairment of the mandibular bone base. The results of an incisional biopsy led to a diagnosis of acanthomatous ameloblastoma. We fixed Erich arch bars to both dental arches and performed an en-bloc resection surgery under general anesthesia for tumor removal. She was then treated by maxillomandibular rigid fixation with the installation of a 2.7 mm non-locking reconstruction plate. So far, she has presented no motor deficits, chewing difficulties, or relevant asymmetries. The tumor showed no recurrence after the first year (pregnancy period) and post-surgery radiographic follow-up revealed a reduction in the surgical area after osseous growth in the margins of the lesion. Although she displayed no systemic comorbidities that affected pregnancy, the fetus was born with alobar holoprosencephaly.The possible influence of pregnancy hormones on the growth and development of tumors in general and ameloblastoma in particular, is still not explained in the literature. However, evidence reveals that the issue should be further studied. Although en-bloc resection surgery is considered a radical method of treatment, it is an effective alternative in ameloblastoma removal, presenting low rates of recurrence. Ameloblastoma is an aggressive infiltrating odontogenic tumor with high recurrence rates. It represents 11 % of all odontogenic tumors and less than 1 % of all tumors affecting the jaws, with a rare ability to metastasize . The disAmeloblastoma radiology usually presents a unilocular or multilocular radiolucency; the latter has a soap bubble appearance, indicating that it might be divided into several bone spaces by trabeculae. Ameloblastomas are often associated with the presence of unerupted teeth. The teeth associated with ameloblastoma are vital, presenting in some cases with migration, mobility, and root resorption .Surgical treatment for this type of tumor extends from conservative forms, such as curettage, enucleation, and cryosurgery, to more radical forms, such as marginal resection, en-bloc resection, or segmental/hemiresection . RecurreAmeloblastoma cases during pregnancy are still scarce in the literature. Here we present a rare clinical case of ameloblastoma in a 27-year-old white pregnant woman, the first case reported in the Brazilian population.A descriptive case study conducted at Hospital Regional de Sobradinho (HRS) after Federal District health ethics committee approval (CEP/SESDF) on 3 August 2015, CAAE - 47521015.6.0000.5553, number: 1,167,855.These data were recorded in the free informed consent form signed by the patient. The case was selected due to the rarity of ameloblastoma occurrence in pregnant women.A 27-year-old white woman, 12-weeks pregnant, presented with increased mandibular volume in the right posterior region of her mandible. She had been complaining of pain and bleeding for months, feeling uncomfortable on her face when sleeping on her right side. She described initial observation of the pain in the middle of the second month of pregnancy. The pain increased in the third month, which led her to search for public dental care. She was observed until week 22 because surgery was recommended only after the first trimester of pregnancy.According to hospital records, she was a non-cigarette smoking and non-alcoholic primipara reporting no relevant systemic comorbidities. She had attended 16 prenatal appointments due to the diagnosis of alobar holoprosencephaly at week 30 of pregnancy. She reported no pregnancy complications and exhibited normal values in blood tests.An intraoral examination showed mandibular/buccal expansion in the right alveolar process, close to her molars, with deviation of her right mandibular third molar. Mucosa was ulcerated and bleeding due to trauma caused by the maxillary teeth cusps, which was the source of the pain. She had had the right mandibular second molar extracted on an unknown date due to extensive tooth decay and pain. Imaging examination of the lesion region dating back to 2005 revealed no abnormalities from the surgery.A panoramic X-ray showed a large unilocular osteolytic lesion extending from her right mandibular first molar to the ascending ramus. Images also showed displacement of her right mandibular third molar and involvement of the mandibular bone base Fig. .Fig. 1InAn incisional biopsy revealed acanthomatous ameloblastoma with nests of cords and islands of basaloid odontogenic palisading epithelial cells. The columnar cells showed elongated nuclei with inconspicuous nucleoli and exhibited inverted polarization opposite to the basement membrane Fig. . The cenShe was admitted to our surgical center to be submitted to en-bloc resection surgery and fixation of a reconstructive non-locking plate under general anesthesia; during surgery she was in a supine position, and she was monitored and pre-oxygenated. Anesthesia was induced by rapid sequence of Sellick’s maneuver with fentanyl 200 mg, propofol 150 mg, Quelicin (succinylcholine) 60 mg and nasal intubation. Maintenance was performed with oxygen, nitrous oxide, and sevoflurane.Surgical access was provided with Erich arch bar applications in both dental arches and through intramuscular incision in the right cervical region. Prior to tumor removal, we adapted a 2.7 mm reconstructive non-locking plate by boring holes in it. The plate was removed and the maxillomandibular fixation was released for intrasulcular incisions and gingiva detachment.Osteotomy was performed from the mesial of her second premolar to half of the ascending ramus. Maxillomandibular fixation was redone and the 2.7 mm reconstructive non-locking plate was re-installed. Extraoral suturing, fixation release and intraoral suturing were performed in sequence. At the end of the surgery, we used elastics for occlusal maintenance.We submitted the surgical specimen to a new histopathological analysis, which confirmed the previous diagnosis of ameloblastoma Fig. .Fig. 3FrShe was then monitored for 24 months at the Sobradinho Regional Hospital dental care unit, having evolved well without any signs of recurrence. She presented no motor deficits, chewing difficulties, or relevant asymmetries. She even reported improvement in chin skin sensitivity. The tumor showed no recurrence after the first year (pregnancy period) and post-surgery radiographic follow-up revealed a reduction of surgical area after osseous growth in the margins of the lesion. In the second year, radiography revealed a suggestive image formation and osseous growth without tumor recurrence , the risk of mental abnormalities due to dental radiology is considered nonexistent . Soet al. reported a case of adenomatoid odontogenic tumor (AOT) in which estrogen receptors (located in tumor cell nuclei) were positive for B-cell lymphoma 2 (Bcl-2), suggesting that the survival of tumor cells was facilitated by Bcl-2 upon continuous stimulation of estrogen during pregnancy [Studies show conflicting results on the development of odontogenic tumors during pregnancy. Sekiya regnancy . Howeverregnancy .Only two cases of ameloblastoma tumors during pregnancy have been reported in the literature , 23. HerAnother study reported by Gordy et al.  suggesteIn the cases reported in the literature, the women were 7-weeks  and 36-wBoth studies , 23 consIn the case of not treating the ameloblastoma, the patient may run the risk of a pathologic fracture of the lower jaw base , large lAcanthomatous-type ameloblastomas have a recurrence rate of 16.2 %. Resection of unicystic ameloblastoma is described to have the lowest recurrence rate (3.6 %) if bone margins are removed appropriately. A conservative method of treatment resulted in an 18 % recurrence rate. The aim of the conservative method is to reduce the size of unicystic ameloblastoma; it is not a popular method, but it is more beneficial for severely ill patients or those with a huge lesion .Regarding surgical risk, due to the fact that a unicystic ameloblastoma has a lower recurrence rate and is less aggressive, removal can be delayed. However, the possibility of pathologic fracture of the jaw and increased tumor volume were the main reasons we performed the surgical procedure after our patient completed the first trimester of pregnancy, when the maturation of the vital structures of her fetus had already been completed. Sevoflurane has excellent safety ratings, although there are rare single case reports of severe acute liver injury similar to halothane hepatitis, and it is the preferred agent because it reduces mucous membrane irritation . StudiesThe possible influence of pregnancy hormones on the growth and development of tumors, particularly ameloblastoma, is not explained in the literature. However, evidence suggests that the issue should be better explored by further studies. En-bloc resection surgery, although a radical method of treatment, is an effective alternative for ameloblastoma removal, presenting lower recurrence rates.I write the following to provide assistance to the case report written about my disease and care. I have no medical or odontological knowledge, so I only write from my own perspective and experience.I sought the dental service that serves my community because I started to feel uncomfortable on my face when I slept on the right side. I was in the third month of pregnancy and, fearing a toothache during that period, I looked for a dentist. The dentist examined my mouth and noticed something different on my face. One side was slightly higher than the other. She asked my doctor’s opinion about having a CT scan to identify the reason for the growth. The doctor, however, said that since I was pregnant, a CT scan was not suitable because of the radiation. An X-ray was taken instead.After the results of the examination-ray, the dentist told me I had an injury on the bone of the right mandible and referred me to the center for dental specialties at Sobradinho Regional Hospital for a consultation with a specialist. The specialist dentist said it was an injury involving the right side of my jaw and that surgery would be necessary to remove it. He explained that the lesion was large and it would be removed with a part of the injured bone and teeth involved. After removal, he would use a metal bar to join the ends. I was worried about the procedure because I thought it could affect my baby somehow. The dentist explained the risks of not removing the lesion and told me what the best time during pregnancy to do the procedure was. Pre-surgical tests were done to check my health and I obtained authorization from my prenatal doctor for the surgery.The surgery was performed at the hospital’s surgical center on 17 March 2014. I stayed in the hospital for a day for observation and my bite was restrained for rapid healing. The greatest difficulty after surgery was the inability to eat solid food in the first few weeks. Another thing that bothered me was sleeping because of the post-surgical pain. After the surgery, I was accompanied by the professionals who treated me and had no problems during pregnancy. I go to the dentist regularly for routine screening to see if everything is still okay. The scar was along the skin folds of my neck, so it does not appear on my face.