Background: Perforation of the gall bladder with cholecystohepatic communication is a rare cause of liver abscess. We are reporting here six rare cases of gall bladder perforation with variable clinical presentations.Materials and Methods: Most patients presented with right hypochondrium pain and fever but two patients presented with only pain in the abdomen. Ultrasonography (USG) and Computed Tomography (CT) were used for diagnosis. The patients were also successfully treated.Results: There was a gall bladder perforation with cholecystohepatic communication, leading to liver abscess formation in most cases on USG and CT. The final diagnosis was confirmed on surgery.Conclusion: The perforation of the gall bladder which leads to liver abscess is a rare complication of acute, chronic or empyema gall bladder. USG and CT scans are the most important diagnostic tool in diagnosing this rare complication. In the set up, where advanced options are not available, the only treatment of choice is the conservative one or surgery, according to the status of the patients. In 1934, Neimeir proposed a classification of the acute perforation of the gall bladder based on his findings and therapeutic approaches (Table 1) [4].The perforation of the gall bladder with cholecystohepatic communication is a rare cause of liver abscess [6]. Ultrasonography (USG) is the initial imaging modality of choice for the evaluation of the suspected acute gall bladder disorders, and it is often sufficient for a correct diagnosis. Contrast enhanced computed tomography (CECT) also plays a vital role in the evaluation of acute gall bladder pathology. CT is particularly useful in situations where ultrasound findings are equivocal. CT is also extremely valuable in the assessment of suspected complications of acute cholecystitis, particularly emphysematous cholecystitis, hemorrhagic cholecystitis, and gall bladder perforation, diagnoses which are often very difficult to establish at sonography [7].He concluded that this rare condition required early recognition and treatment to decrease the mortality rate. There are no classical symptoms and signs associated with gall bladder perforations (GBP). Intrahepatic perforation of the gall bladder, with liver abscess and cholecystohepatic communication, is even rarer. Only a few adult cases have been reported in the English literature .In 1934, Niemeier classified GBF into three types: type 1, chronic perforation with the presence of a fistulous communication between the gall bladder and some other viscus; type 2, subacute perforation where the perforated gall bladder is surrounded by an abscess walled off by adhesions from the general peritoneal cavity; and type 3, acute perforation of the gall bladder into the free peritoneal cavity without protective adhesions (5] reported 31 patients with GBF. Nine patients (29%) had type I perforation, presented with complaints of generalized abdominal pain. Seven patients had history of pain, which was initially localized to the right hypochondrium before becoming generalized. Three of these were already diagnosed to have gallstone disease and were waiting for elective cholecystectomy. Seven (78%) patients had presented with shock. Features of generalized peritonitis were seen in six (67%) patients. Type II perforation was present in 14 patients (45%) who presented with complaints of right hypochondrial pain, which ranged in duration from 3 days to 15 days. All except for one patient described a history of such complaints even though they may have been milder. Type III perforation was present in eight patients (26%) and presented with acute cholecystitis. However, these patients had symptoms of longer duration and were recurrent. Fever was noted in 15 (48%) patients, all of whom had either type I or type II perforations. An abdominal mass was present in 11 patients (36%) while three (10%) patients have presented with jaundice.Somasekhar RM et al [1]. Elevated liver enzymes, especially alkaline phosphatase levels are commonly observed [3]. A sudden decrease in pain intensity may occur due to perforation which causes relief of high intracholecystic pressure [3].GBF can be due to cholelithiasis, infection, malignancy, trauma, corticosteroid therapy, diabetes mellitus, impaired vascular supply, old age and male sex. Patients may present with right upper quadrant pain, fever, palpable right upper quadrant mass and tenderness may herald an acute onset. However, patients may present with weakness, malaise, anorexia and a palpable right upper quadrant mass in malignancy [3]. USG may show gall bladder wall thickening >3mm, gall bladder distention (largest diameter >3.5–4.0 cm), gallstones, coarse intracholecystic echogenic debris and bile duct dilatation including halo sign, which can help in diagnosis, as diagnosed in our case [15]. The earliest signs of impending gall bladder perforation detectable on sonography may be distended gall bladder and edema of its walls along with liver abscess, raise the suspicion of intrahepatic perforation [16]. Computed tomography can demonstrate either calculi outside the gall bladder or a ruptured segment of the gall bladder wall, which are the direct indicators of perforation according to Pedrosa et al [17].The demonstration of an abscess outside the gall bladder, presence of gallstones and thickening of gall bladder wall are the indirect indicators. Similar to our reported case we also diagnosed a hole sign between the gall bladder and the liver abscess, which matched to other reported studies [16].Fischer [18] reported 17 patients with hepatic abscess, out of which two were associated with acute cholecystitis. These hepatic abscesses were found in surgery to be the result of direct extension from an acutely inflamed gall bladder.The treatment modality, which has not been fully established for GBF with cholecystohepatic communication, leads to liver abscesses, which are rare [19] reported 5 cases of hepatic abscess secondary to acute cholecystitis , aged between 46 and 78 years. They presented with fever, abdominal pain and, one, with jaundice. Liver abscess were diagnosed by USG and CT scan. In acute phase, all the patients were treated with percutaneous drainage and subsequently, four of them had elective cholecystectomy. CT may have an important role in preoperative diagnosis of complicated acute cholecystitis and in the surgical management of patients with this condition, if ultrasound examination fails to establish the diagnosis or cannot be done for technical reasons [17]. Similar to our cases, CT and USG were the helpful investigations in the treatment of the perforation. The increase in incidence may be attributed to better diagnostic modalities like ultrasound and computerized tomography, which have since become available and have been used in the diagnostic evaluation of these patients [20].Zerman [21].With CT, a pyogenic liver abscess typically appears as a well–defined, round, hypodense mass with a central density that is between 0–45 Hounsfield units. The peripheral wall usually enhances with contrast administration and internal septa may be present. Small microabscesses may coalesce to form a larger abscess. At sonography, a pyogenic abscess usually appears round or ovoid with variable internal echogenicity [27] as an uncommon surgical emergency exclusively in the seventh and eighth decade, sparingly occuring in younger patients. The gallstone intermittently obstructs the bowel before impaction, leading to tumbling obstruction [28]. In a review of 458 cases, only two cases of perforation were cited [29]. The perforation occurs either at the site of impaction of gallstone, or at previous sites of obstruction and is present because of pressure necrosis of jejunal wall [30].Type III patients, present with features similar to those of chronic cholecystitis are so are difficult to identify preoperatively, unless they have obstructive symptoms. This has been the experience of other authors as well.Gallstone ileus was first described by Bartolin in 1654 [31]. Though ultrasound could not specifically identify type I perforations, it was helpful in determining the need for surgical intervention, as it could identify the presence of free fluid, the appearance of which, on guided tap, left no doubts about the pathology. The results of ultrasound and computerized tomography in our patients are similar to those quoted in various studies comparing efficacy of ultrasound and CECT in detecting gall bladder perforation [24].In the past, clinical and radiological aids were insufficient to clinch the diagnosis, however advent of CT and MRI has made it easier [32] have reported mortality rate to be between 12 and 16%. They have suggested that spontaneous gall bladder perforation is caused by hypoperfusion of viscera, secondary to systemic disease and also reported that the fundus of the gall bladder is the most common site of perforation in gall bladder. However, other hypotheses include trauma, congenital abnormality, infection, pancreatic secretions, obstructions, calculi and abnormal bile [33].Roslyn and Busutte [34]. It is an adequate procedure for elderly patients, in which subsequent cholecystectomy is not mandatory [36]. But, our 3rd case of bowel resection anastomosis was done due to gangrenous and perforation of the bowel to avoid further mortality. In the 4th case, the patient was treated conservatively and was operated after 3 months and discharged in satisfactory condition.Although one and two stage procedures can be carried out safely in cases of gall stone ileus with optimal survival, enterolithotomy alone is the minimal surgery sufficient in emergency situations [Delay in surgical intervention is the major reason for increased morbidity and mortality associated with GBF. Emergency cholecystectomy should be considered in patients with acute cholecystitis at an early stage to prevent this complication.We presented a rare case of gall bladder perforation which led to formation of hepatic abscess with cholecystohepatic communication. Initially, such cases could be treated conservatively with percutaneous drainage with subsequent cholecystectomy. Early diagnosis of gall bladder perforation and immediate surgical intervention are of prime importance in decreasing morbidity and mortality associated with this condition. In such cases, the intrahepatic nature of the gall bladder can lead to difficulty in laparoscopic cholecystectomy, with a high rate of conversion to open cholecystectomy.


Melanotic neuroectodermal tumors of infancy (MNTIs) are rare fast-growing tumors with high recurrence rates. These tumors, which originate in the neural crest, commonly occur in the anterior maxilla of children under the age of one. Here, we describe an MNTI case in a two-month-old girl with increasing swelling in the left cheek. MNTI was diagnosed in this case following tomography and biopsy. The patient's histological and immunohistochemical profile indicated a remarkable combination of neural, melanocytic, and epithelial cell differentiation. One year following tumor excision, a follow-up examination revealed that the child exhibited no tumor recurrence. Approximately 260 cases of MNTI have been reported since this type of tumor was first described. In the present case, early diagnosis minimized the difficulties and risks associated with treatment and facilitated an optimal outcome. Despite complete surgical excision, careful followup is recommended. In addition, maxillary functional orthopedics and reconstruction may be necessary in cases of MNTI. Melanotic neuroectodermal tumors of infancy (MNTIs) are rare, fast-growing, melanin-containing lesions that commonly occur in the head and neck regions of children under the age of one . MNTIs aMNTIs generally occur in the maxilla 68%–80%), but they can occasionally arise in the skull 10.8%), mandible (5.8%) or brain 4.3%) [0.8%, man8%–80%, b% [0.8%, MNTI lesions are regarded as benign tumors, although they can present locally aggressive behavior, including gradual invasion of the surrounding bone and sinuses. These lesions are characterized by a high recurrence rate that varies between 10% and 60% , 6. In pMNTI poses a challenge to clinicians not only in its clinical diagnosis but also in its treatment. Complete surgical excision is generally the first treatment option , but proIn this report, we present a case of MNTI in a two-month-old baby girl who was treated by complete surgical excision. The clinical, imaging, and histological characteristics of this case are also discussed. Immunohistochemical data regarding MNTI are somewhat inconsistent, so a panel of specific antibodies was performed to correctly identify the different types of tumor cells.A two-month-old girl was referred to the Oral and Maxillofacial Surgery Department of Conjunto Hospitalar do Mandaqui  and presented with a mouth tumefaction with one-month evolution. An extraoral examination revealed facial asymmetry, deletion of the left nasolabial folds, and elevation of the left nasal alar base. Otherwise, the baby appeared to be in a healthy condition and was hydrated.During the intraoral assessment, a left premaxilla tumefaction was observed in the alveolar ridge near the canine region . The oveAn odontogenic tumor was our first diagnostic hypothesis. A surgical excision was performed under general anesthesia. During surgery, an inner brown-colored aspect of the lesion was observed, which raised the suspicion of MNTI. A peripheral ostectomy was performed to assure total tumor excision.The surgical piece indicated a fibrous blackish-brown lesion containing two primary teeth within the tumor mass. A microscopic assessment revealed fragments of tissue characterized by the proliferation of a dual population of cells arranged in solid nests or cords in the middle of dense, well-cellularized connective tissue . The firAt the time of the one-year follow-up appointment, clinical and tomography examinations did not reveal any tumor recurrence Figures . The chiMNTI is an unusual type of neoplasm , 6–8. ThAlthough the recurrence rate of MNTI has been reported to be 15% within the first year of enucleation , no signAlthough MNTI exhibits some local invasive features, it generally follows a benign course. Several studies , 17, 18 Although several authors have reported positive results following surgical treatment and chemotherapy , 15, othMNTIs are biphasic tumors composed of small-cell and large-cell components that are arranged in nest or cord arrangements set in a vascularized fibrous stroma . The nesImmunohistochemical staining in MNTI is somewhat variable and can be used to assist diagnoses –3, 8. PrIn the present case the expression of synaptophysin was more frequently detected in small cells than in larger cells. Chromogranin, another neuroendocrine marker expressed by both cell types, was expressed at a greater intensity in the small neuroblast-like cells. Earlier chromogranin assays were negative in many MNTI studies , 22.Large epithelioid cells typically express cytokeratin, HMB45, and EMA but are rarely positive for S100 protein and are usually negative for melan-A, glial fibrillary acidic protein (GFAP), and alpha-fetoprotein (AFP). S100 is expressed by melanocytes and melanomas. The immunohistochemical profile of MNTI is generally positive for cytokeratin and HMB45 and negative for S100 , 8. In tThe expressions of Ki-67 and CD99 are quite uncommon and might be related to more aggressive tumor growth , 6. Ki-6Ki-67 expression was observed to be elevated in 15% of the histological fields observed in this report, a finding that indicates a slight potential for a more aggressive lesion. Fortunately, the treatment approach employed here, which included a marginal ostectomy, was adequate even despite the high rate of mitosis.Although our data reveal that the risk of recurrence is significantly reduced after one year, a rigorous followup should be performed over a longer period of the child's development. The prospect of local reconstruction has been planned, and maxillary functional orthopedic treatment will likely be required to stimulate adequate facial growth as fibrous healing is a considerable local issue.In view of the rapid growth of MNTIs, their malignant potential, and their high rate of recurrence, it is essential to diagnose this type of tumor at an early stage. Pediatricians and pediatric dentists should be aware of this disease and direct patients to prompt oral surgery treatment to minimize mutilating surgeries.


Prostatic stromal sarcoma is a fairly rare tumor that constitutes approximately 0.1–0.2% of all prostatic cancers. Detailed characteristics of the tumor are still unclear due to its rarity.We describe a case of prostatic stromal sarcoma in a 63 year-old man who suffered from urinary obstructive symptoms. Palliative transuterine resection was performed and the preliminary histopathological diagnosis was neuroendocrine carcinoma. After chemotherapy, total pelvic exenteration was performed. Histopathologically, the tumor was composed of monotonously proliferating small to medium-sized round cells, which existed in compact islands with loose or dense fibrovascular networks. Immunohistochemically, the tumor cells were widely positive for vimentin, CD56, CD99 and focally positive for synaptophysin, CD10, progesterone receptor, desmin and CD34, but negative for EMA, cytokeratin, estrogen receptor, S-100 and myoglobin. Most of the previously reported tumors exhibited positive stainability for CD10 and progesterone receptor. In addition to these markers, expressions of CD56, CD99 and synaptophysin were characteristically detected in our case. To the best of our knowledge, we present the first case of prostatic stromal sarcoma with characteristic immunohistochemical staining properties. Although the biological characteristics of this rare tumor have not yet been elucidated, these findings suggest prostatic stromal sarcoma can potentially show neuroectodermal differentiation.http://www.diagnosticpathology.diagnomx.eu/vs/7291874028051262The virtual slide(s) for this article can be found here: Prostatic stromal sarcoma (PSS) is a fairly rare tumor, constituting approximately 0.1% of all prostatic cancers. ProstatThe patient was a 63 year-old man who presented to the urologist with a one week history of dysuria, pollakiuria and an unrelieved feeling after urination. He had a previous history of diabetes mellitus and asymptomatic multiple brain infarction. He habitually drank alcohol and smoked and his mother died of gastric cancer. Abdominal ultrasonography revealed 343 ml of residual urine volume. Computed tomography revealed a prostatic mass lesion which protruded into the bladder space Figure. GadriniProstatic stromal sarcoma is a rare tumor that constitutes approximately 0.1–0.2% of all prostatic cancers. Except Recent immunohistochemical analysis revealed that both prostatic stromal sarcoma and STUMP usually express CD10, CD34 and the progesterone receptor,8,10-12.de novo tumorigenesis and malignant transformation step by step[PSS is suspiciously derived from mesenchymal pluripotent stem cells in the prostatic stroma. As few cases of STUMP change to PSS in the history, some genetic transformations are considered to be related to PSS,19. Arva by step. However by step-27. Elo  by step. These m by step. Amsterd by step. This knWe describe a rare case of prostatic stromal sarcoma (PSS) presenting with characteristic immunohistochemical staining properties. To our knowledge, this case is the first case of PSS with positive stainability for synaptophysin, as well as CD99 and CD56.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of Diagnostic Pathology.We do not have any competing interests for our manuscript.HY was responsible for data collection and drafted the manuscript. TO, TT, YT, DK and TA made contributions to acquisition of clinical data and performed surgical procedure. HI performed radiological interpretation and helped drafting the manuscript. All authors read and approved the final manuscript.


Gall bladder (GB) may be found in a variety of abnormal positions. Most of them are due to arrested development of embryonic growth at different stages. A 63-year-old female patient was admitted to our radiology unit for magnetic resonance imaging (MRI) of the liver for the lesions identified in abdominal ultrasonography (US) and computed tomography (CT). MRI showed that there was a lobulated heterogenous mass in the left lobe of the liver and a smaller one in the right lobe of the liver with the same appearance. The inferior pole of the liver was located in the pelvic space, and the GB, which contained sludges and stones, was lying down to the upper pelvic space. Hepatic masses were considered to be hemangiomas, and GB was diagnosed as ptotic GB with luminal sludge and stones. In this case, especially, MR imaging helped the surgeon to plan a proper approach to the GB in abnormal localization. GB is located adjacent to the undersurface of the liver at the junction between left and right lobes. It is mostly found in the right upper quadrant but may be seen in any part of the abdomen . AlthougTo our knowledge, ptosis of the GB is a rare very important entity, as it can create a serious clinical confusion. If the location of the GB is away from the peritoneum or retroperitoneal, the typical clinical signs of cholecystitis can be absent, and symptoms of peritoneal irritation may not be seen. A GB lying outside its normal anatomic fossa may be susceptible to torsion and consequent gangrene .If the routine preoperative studies may not detect the aberrant location, an unusual surprise for the surgeons during laparoscopy may occur precedingly .We report here a case with ptotic GB and liver masses. The US, CT, and MRI findings of the patient will be presented.A 63-year-old woman was admitted to the hospital with lower quadrant pain. On physical examination, there was hepatomegaly with soft and mildly tender abdomen in the right lower quadrant without peritoneal signs. There was no Murphy's sign on examination.In the laboratory data, serum bilirubin was normal , ALT and AST were normal with values of 23 IU/L and 17 IU/L, and alkaline phosphatase was normal (56 IU/L). Gama glutamyl transferase and lactate dehydrogenase were elevated with values of 76 IU/L and 255 IU/L. Serum electrolytes were normal . On US examination, the GB with endoluminal sludges and stones was lying down to the upper pelvic space (ptotic GB) . The blaOn MRI, dynamic series were performed to characterize the hepatic masses. The lesion in the left lobe predicted early peripheral nodular enhancement with the increasing central enhancement on delayed images. There was fibrotic scar tissue at the peripheral and in the centre of the lesion. The scar tissue did not enhance all images. With regard to all these findings, the lesion was considered to be a papillary hemangioma . The lesion in the right lobe presented the same enhancement pattern like the one in the left lobe except for the enhancement of central scar tissue and also was considered as a hemangioma . The ptoAn operation for the GB and/or hepatic masses was not planned by the surgeon, but clinical followup in short intervals was recommended. On these circumstances, our radiologic findings were correlated with the clinical findings. An aberrant GB situated under the left lobe of liver, medial to the falciform ligament, was first described by Hochstetter in 1886 and was termed as “left -sided GB” . TranspoThe GB rotation and/or displacement can be caused by hepatic lobe abnormalities, such as aplasia, hypoplasia, hypertrophy and by abnormal mobility of the GB itself .Bender et al.  reportedSaygun et al.  reportedReddy et al.  reportedQureshi and Awad  and MeekAlthough these cases about left-sided GB were present in the literature, to the best of our knowledge, there were just two cases of ptotic GB. Meeker and Lisenby  reportedMaki et al.  reportedWhen a ptotic GB was discovered, the findings indicate that there might be associated anomalies such as ptosis of other abdominal visceral organs. Surgeons should be aware of these anomalies and should initiate the dissection as so close to the GB as possible . The pedIn our case, surgery was not performed, and clinical follow-up was recommended by the surgeon: that was the main limitation of this report.Finally we believe that this is the first reported case of ptotic GB with hepatomegaly, liver masses, and atrophic spleen. Radiologic approaches especially MRI should be performed for detecting the associated anomalies and helping the clinicians to manage the proper approach for all patient's gall bladder problems , 18.


Intranodal palisaded myofibroblastoma is one of the primary mesenchymal tumours. The inguinal region is the commonest site of this rare tumour. As there are only about 55 such cases reported in the literature, the precise aetiology and pathogenesis have yet to be explained adequately. Here we report a case of a 72 year old man presented with incidental finding of intranodal palisaded myofibroblastoma in the retroperitoneal region.A 72-year old man presented with abdominal pain in right upper quadrant with an incidental finding of abdominal mass in the right flank. The computerised tomogram scan of abdomen confirmed acute cholecystitis with a 5 x 5 cm retroperitoneal mass. He underwent cholecystectomy with excision of this mass. He recovered well following his operation and was discharged from the hospital. Histological examination confirmed the diagnosis of intranodal palisaded myofibroblastoma.To our knowledge, this is the first case of intranodal palisaded myofibroblastoma originating from retroperitoneum. Along with the rarity of this case, we also discussed its typical histopathological findings, aetiology and pathogenesis. In comparison to secondary mesenchymal tumours, primary mesenchymal tumours involving lymph nodes are very rare. The most common primary mesenchymal tumours include Kaposi's sarcoma, pseudosarcomatous malignant melanomas and melanocytic naevi . Intrano2 mass was also noted in the right flank. The provisional diagnosis of acute cholecystitis with incidental finding of abdominal mass was made and further investigation in the form of computerised tomography (CT) of his abdomen and pelvis was performed. The scan confirmed the presence of 50 × 55 mm2 mass in the retroperitoneum on the right side of the urinary bladder but separate from the small intestinal loops without any evidence of local infiltration. The radiological differential diagnosis included carcinoid lesion and teratoma of a right undescended testis. However, undescended testis was ruled out with a fully grown testis on the right side of the scrotum. The patient underwent exploratory laparotomy, open cholecystectomy and also excision of the pelvic mass. Intra-operatively, the mass was found to be approximately 8 cms in size, partly haemorrhagic and covered by peritoneum. It was situated lateral to the right ureter and superior to the right iliac vessels. This mass was excised en-block without any intra-operative complications. The patient had an uneventful post-operative recovery and was discharged on the 10th post-operative day. Histological diagnosis of intranodal palisaded myofibroblastoma was confirmed. He did not reveal any signs of recurrence in seven years of follow up. The histopathological findings are described as follows.A 72 year-old man presented in the emergency department with right upper quadrant and epigastric pain, nausea and vomiting. On examination, the patient was sweating and pale with a tender right upper quadrant. A tender 5 × 5 cm3 with surrounding attached fatty tissue measuring up to 100 mms in maximum dimension was noted. On slicing, a well defined nodule with partially firm, white and solid areas, alternating with haemorrhagic areas, was found. At the periphery there was some calcification. The microscopic study was performed on extensively sampled solid and haemorrhagic areas including the resection margins. The gall bladder was 105 mms with a thick wall and a few tiny stones.A Solid mass measuring 80 × 80 × 50 mmThis represented encapsulated, a well defined nodular lesion with variable cellularity. The cells were spindle shaped with slightly wavy nuclei forming short interlacing fascicles, exhibiting areas that were reminiscent of AntonyA/Antony B area figure . No signThe spindle cells were positive for Smooth muscle actin (SMA) Figure , CalponiAmong primary mesenchymal tumours, intranodal palisaded myofibroblastoma is very rare. It was initially described by Deligdish and Katz as neurilemmoma or schwanoma and later classified as palisaded myofibroblastoma by Weiss ,8. It shIPM commonly affects the second to eighth decade, with a peak incidence in the group between 40 and 60 years of age, but occurrence of this tumour in an infant has also been reported ,10. It iIt is essential to differentiate IPM from other soft tissue tumours. These include Schwannoma, Kaposi's sarcoma, intranodal leiomyoma, inflammatory pseudotumour, solitary fibromastocytic tumour, angiomyomatous hamartoma and metastatic spindle cell lesions in the lymph node. The clinical history, examination and typical histological characteristics help in the correct diagnosis of the IPM. Schwannoma is uncommon in the inguinal region and immunohistochemically it is positive for S100 that differentiates it from IPM. Negative immunostaining for HHV 8 along with an absence of EBV DNA and a history of immunocompromised status favours the diagnosis of IPM rather than Kaposi's sarcoma. There were no slit-like vascular channels, no extravasation of red blood cells and no hyaline globules as typically seen in Kaposi's sarcoma. Differentiation of inflammatory myofibroblastic tumour (IMT) from IPM can be made by the presence of inflammatory cells and absence of the amianthoid fibers in IMT . NegativDue to the rarity of the condition the precise aetiology and pathogenesis have yet to be defined. Inguinal lymph nodes have a higher concentration of myofibroblasts compare to other lymph nodes in the body due to increased drainage area and function: this might explain its more common occurrence in the inguinal region. This is favoured by positive staining for actin and vinmentin and negative for desmin in myofibroblasts of the inguinal region compared to fibroblasts elsewhere in the body . HoweverAlthough rare, IPM should be considered as one of the differential diagnosis in patients presenting with an abdominal mass in the retroperitoneal region. This site of origin may provide an opportunity to reconsider the aetiology and pathogenesis of IPM.IMT: Inflammtory Myofibroblastic Tumour, IPM: Intranodal Palisaded MyofibroblastomaWritten informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.JS was involved directly in patient management and preparation of manuscript. AV was involved in preparation of manuscript. HM provided the histological pictures with picture legends. All authors read and approved the final manuscript.The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1472-6890/11/7/prepub


Introduction. Thymic cysts are among the rarest cysts found in the neck. Nests of thymic tissue may be found anywhere along the descent of the thymic primordia from the angle of the mandible to the mediastinum. Mediastinal extension is seen in 50% of cervical thymic cysts. Case Report. We report an uncommon case of a 15-year-old male, who noted a painless, growing mass on left side of his neck of one-year duration. Computerised tomographic scan showed a multiloculated fluid density lesion with enhancing septae in the left parapharyngeal space, extending from the level of mandible up to C7 vertebral level. Here, we discuss the surgical aspect, histopathology, and management of this rare lateral neck swelling. Discussion. Clinically, in most cases, cervical thymic lesions present as a unilateral asymptomatic neck mass, commonly on the left side of the neck, and 75% of patients present before 20 years of age. Conclusion. Thymic cyst should be included as differential diagnosis of cystic neck masses. Greater awareness among the pathologists may decrease misdiagnosis. Cervical thymic cysts are unusual lesions usually presenting in the 1st decade of life . The preWe report a rare case of thymopharyngeal duct cyst in a 15-year-old male, describing CT findings, intraoperative findings and histopathology.A 15-year-old boy presented to our outpatient department with a swelling over the left side of neck of 1-year duration which had gradually increased in size over time with no associated pain, weight loss, or pressure symptoms. There was no significant family history. His general physical examination appeared normal without any obvious deformities or abnormalities. Local examination revealed a 3 × 2 cm oval, nontender, and firm mass on the left side of neck at level III. All his other systems seemed to be normal clinically. CT scan of the neck  showed aThe mass was excised under general anaesthesia with a horizontal incision two finger breadths below the body of the mandible. Surgical exploration revealed that a cystic mass surrounded by fibrous adhesions was found lying between the left lobe of thyroid gland medially, internal jugular vein laterally, and the sternocleidomastoid anterolaterally extending 1 cm below the angle of the mandible to the level of the cricoid cartilage. The cyst was found to have a fibrous cord tracking inferiorly up to the superior mediastinum, for which a stepladder incision was made 2 cm inferior to the upper incision, and the cord was dissected and ligated deep to the clavicle. The mass was completely excised . Histopathological examination revealed multiple cystic spaces lined by cuboidal to squamous epithelium. The cyst contained numerous cholesterol clefts with subepithelium showing fibrocartilagenous tissue, foreign body corpuscles, and Hassall's corpuscles thereby diagnosed as a thymic cyst .A final diagnosis of thymic cyst was made. Postoperative period was uneventful. The patient has been symptom free for more than a year.Differential diagnosis for cystic masses in the neck is done. Cystic masses of the neck are thyroglossal duct cysts, branchial cleft cysts, cystic hygromas, dermoid cysts, epidermoid cysts, thymic cysts, bronchogenic cysts , and laryngoceles. Thymic cysts are very rare representing only 1% of cystic cervical masses .Patients with thymic cysts usually present in the 1st decade of life being more common in males. Patients usually present with slow growing painless mass in the lateral aspect of neck near thoracic inlet either deep or superficial to sternocleidomastoid muscle. The size can vary from 1 cm to 26 cms [Two varieties of thymic cysts are described, congenital and acquired. Persistence of thymopharyngeal tracts and the degeneration of Hassall's corpuscles within ectopic thymic remnants are the two most important etiologies of thymic cysts . The thyThe cervical thymic lesions include the following categories distinguished by anatomic location and the nature of the thymic gland tissue.Accessory Cervical Thymus. Solid cervical thymic tissue is sequestered from the main gland, along the normal descent path, with or without parathyroid. Previous terms include aberrant, ectopic, undescended, persistent, or accessory thymus.Cervical Thymic Cyst. Sequestered cystic cervical thymus is found along a normal path of descent, with or without parathyroid glands. It is a cystic version of accessory cervical thymus and may have fibrous band or a solid thymic cord connection to the pharynx or mediastinum.Undescended Cervical Thymus. This occurs when a solid lobe of thymus fails to descend entirely, with or without a parathyroid complex. It differs from accessory cervical thymus in that only half of the normally blobbed thymus is present in the mediastinum; conceivably, it may also become cystic.Persistent Thymopharyngeal Duct Cyst. This is the same as undescended cervical thymus; however, the thymic duct is cystic. The thymus is solid, with or without parathyroid complex, and probably represents undescended thymus. A variant would be the cervical cystic duct leading to the mediastinal thymus.Persistent Thymic Cord. This is the cervical prolongation of a solid thymic cord which is continuous with the mediastinal thymus. The cystic variant may overlap with the histology and clinical appearance of the cervical thymic cyst if a true connection to mediastinal thymus cannot be documented.Cervical Extension of Mediastinal Thymus. This appears as low midline solid thymus at the thoracic inlet due to incomplete mediastinal descent. It may transiently present with increased intrathoracic pressure.Ectopic Thymus. This is the rare, solid thymic tissue in abnormal locations, for example, in the pharynx, trachea, or base of skull [of skull , 8.On CT, thymic cysts usually have a homogeneous low attenuation (10 to 20 HU) with a thin uniformly smooth wall. Thymic cysts may be unilocular or multilocular. After contrast injection the cyst wall shows smooth regular enhancement. When a cyst becomes infected, its protein content increases, and this is seen on CT as an increase in attenuation. On MR imaging, thymic cysts are homogeneous with a low or intermediate signal on T1-weighted and a high signal on T2-weighted images. Relationship of the cyst with surrounding structures can be clearly demonstrated on CT and MRI.Thymic cysts are unilocular or multilocular containing brownish fluid. The cyst wall lining ranges from flattened squamous or cuboidal cells to multilayered stratified squamous epithelium to even primitive respiratory epithelium. Lobulated lymphoid tissue in the cyst wall contains Hassall's corpuscles . The incWe report this case to highlight the importance of thymic cyst as a differential diagnosis of cystic lateral neck mass, although it is very rare. Also, in our case, cyst was found to have a fibrous cord tracking inferiorly up to the superior mediastinum; hence, we believe it could have arisen from remnant thymopharyngeal duct, which is rarer variety of thymic cyst in the neck. Thymic cysts are unusual causes of cystic cervical masses. However, they should be included as differential diagnosis of cystic neck masses. Imaging, surgical findings, and histopathological correlation play an important role in diagnosing thymic cysts. This paper could be summarized as follows. Thymic cyst is a very rare differential diagnosis of cystic lateral neck swelling. According to the various literatures published, thymic cyst accounts for less than 0.5% of cystic neck swelling. Mediastinal extension is seen in 50% of cervical thymic cysts. Thymic cyst is thought to develop from persistent thymopharyngeal tracts and the degeneration of Hassall's corpuscles within ectopic thymic remnants.


Dear SirAcute appendicitis is one of the frequently seen emergencies in the pediatric hospitals. Nevertheless, its diagnosis and management is often challenging in infants and neonates. The diagnosis is frequently made at operation performed for the complications , 2, 3. WA 2-month-old male infant presented to the surgical emergency with 4-day history of bilious vomiting, abdominal distension, constipation, irritability, and reluctance to feed. Two days prior to presentation he developed moderate intensity fever without rigors. There was no history of diarrhea or respiratory tract infection. General physical examination showed temperature 100F, respiratory rate 30 breaths/min, and pulse of 100 beats/min. On examination, abdomen was distended and mildly tender in the right lower quadrant. No mass or viscera were palpable. A digital rectal examination showed mucous in the rectum.The laboratory investigations  were normal. Abdominal radiograph showed multiple air fluid levels Fig. . UltrasoIt is estimated that about 8% of children who present with abdominal pain are ultimately diagnosed with acute appendicitis. It is very uncommon in infants and neonates. Only 2% patients treated for acute appendicitis are below 2 years of age. The rate of perforation of appendix is 30% in adults as compared to 98% in infants and neonates , 2, 3.The common clinical features in infants and toddlers who present with appendicular perforation are abdominal pain, vomiting, fever, lethargy, and reluctance to feed. These are nonspecific features but a high index of suspicion may lead to the accurate diagnosis. In case of delayed diagnosis, the appendix may perforate and result in localized or generalized peritonitis, appendicular abscess and appendicular mass. Occasionally intestinal loops may adhere to the inflamed and perforated appendix limiting the spread of peritonitis , 3.Source of Support: NilConflict of Interest: None declared


Ewing’s sarcoma uncommonly arises from extraosseous soft tissue or parenchymal organs. Primary adrenal Ewing’s Sarcoma, although very rare, is extremely aggressive and commonly fatal.A 17 year old Pakistani male was referred to the outpatient oncology clinic at our center with a three month history of concomitant pain, swelling and dragging sensation in the right hypochondrium. Abdominal examination revealed a large, firm mass in the right hypochondrium extending into the right lumbar region and epigastrium. His genital exam was unremarkable and there were no stigmata of hepatic or adrenal disease.Computed tomography scans revealed a large peripherally enhancing mass in the hepatorenal area, biopsy of which showed a neoplastic lesion composed of small round blue cells which exhibited abundance of glycogen and stained diffusely positive for CD99 (MIC2 antigen). Fluorescence in situ hybridization demonstrated gene rearrangement at chromosome 22q12 which confirmed the diagnosis of Ewing’s sarcoma. Staging scans revealed pulmonary metastasis and hence he was commenced on systemic chemotherapy.This case report highlights the importance of keeping Ewing’s sarcoma in mind when a young patient presents with a large non-functional adrenal mass. Ewing sarcoma (ES) and peripheral primitive neuroectodermal tumor (PNET) are part of a spectrum of diseases comprising the Ewing sarcoma family of tumors (ESFTs) which most commonly arise from long and flat bones and share similar histologic and immunohistochemical characteristics . ESFTs cAlthough ES/PNET most commonly develops in bone and soft tissues, solid organ primaries have been reported in the past at locations including the paravertebral areas and along the genitourinary tract . The adrWe report the case of a young man with ES/PNET of the adrenal gland. We believe that this is the first reported case of this rare presentation from our center and possibly from the country. The case highlights the importance of having ESFTs in the differential diagnosis of a non-functional adrenal mass in order to make a correct diagnosis, as the management and prognosis of ESFTs is entirely different from other possible causes.A 17 year old Pakistani male was referred to the outpatient oncology clinic at our center with a three month history of swelling in the right upper abdomen with concomitant pain and dragging sensation in the right hypochondrium. An abdominal ultrasonogram ordered by his referring physician had revealed a huge retroperitoneal soft tissue mass of unclear origin.He was uncomfortable due to moderate pain at his first clinic visit and though vitally stable, was obviously pale. In the absence of icterus and pedal edema, abdominal examination revealed a large, firm mass in the right hypochondrium extending into the right lumbar region and epigastrium. His genital exam was unremarkable and there were no stigmata of chronic liver disease or Cushing’s syndrome.Considering his age and presentation, a diagnosis of germ cell tumor was still pursued but relevant tumor markers were non-contributory although serum lactate dehydrogenase (LDH) was mildly elevated 714 IU/L). Computed tomography (CT) scans revealed a large peripherally enhancing and centrally necrotic hypervascular lesion in the hepatorenal area, arising from the right adrenal gland  and ifosfamide and etoposide (IE). He has shown promising clinical response to systemic treatment with a significant reduction in the size of the mass and alleviation of symptoms.ES/PNET occurs more commonly in adolescents with a peak incidence in the second decade of life, has a slight male preponderance , and typMetastatic ES/PNET needs to be considered before a conclusive diagnosis of primary adrenal disease is made. Extensive radiological imaging failed to demonstrate any other site of tumor in our patient’s case and other causes of small round blue cell tumors were excluded by an appropriate panel of immunohistochemical stains.Despite the fact that less than a quarter of patients have overt metastasis at presentation, ESFTs is postulated to be a systemic disease. Relapse rates are as high as 80-90% in patients undergoing local therapy alone and the majority of patients now receive chemotherapy, usually administered prior to and following local treatment . MultimoThe reporting of this case is of paramount importance because it adds to the limited number of documented cases of adrenal ES/PNET in literature and emphasizes upon the consideration of this rare diagnosis when a young patient presents with a large non-functional adrenal mass. It is true that without a high index of suspicion, the diagnosis can easily be missed.Written informed consent was obtained from the patient’s parent for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.MNZ did the literature search and drafted the manuscript. TZA conceived the case report and helped in drafting the manuscript. TM and WM provided the molecular images and the radiological images respectively and provided guidance for the drafting of the manuscript. SP participated in the design and co-ordination and helped draft key areas of the manuscript in addition to providing the histopathological images. All authors read and approved the final manuscript.


Colonic lipomas are rare nonepithelial tumors that are usually detected incidentally during surgery or colonoscopy. Although lipomas generally remain asymptomatic, when they exceed 2 cm of diameter they may cause abdominal pain, obstruction, or intussusception. Here we present a case of an elderly woman referred by her general practitioner to a general hospital of Crete because of acute abdominal pain along with signs of intestinal obstruction and a positive history of appetite loss. Abdominal computed tomography was performed. To marginalise the risk of malignancy, a right hemicolectomy was performed. Histopathological examination of the resected specimen confirmed the diagnosis of cecal lipoma. Colonic lipomas are rare nonepithelial tumors of the large intestine which are usually detected incidentally during surgery or endoscopy . Their iA 68-year-old Caucasian Greek woman was referred to the Surgery Department of the Saint George General Hospital of Chania, Crete by her general practitioner due to a history of intermittent right abdominal pain, meteorism, and constipation lasting for the last 3 days. She also reported loss of appetite during the last month. Her past medical history was unremarkable. The patient did not report any history of anemia or episodes of diarrhea or rectal bleeding. On abdominal examination, palpation revealed slight abdominal tenderness on the right lower quadrant and auscultation increased bowel sounds. Laboratory workup including complete blood count, renal and liver function tests, and blood coagulation was within normal limits except of an elevated level of C-reactive protein . Contrast-enhanced abdominal CT disclosed the presence of a well defined, ovoid mass of 5 × 7 cm in size protruding into the lumen of the cecum . Due to Lipomas of the gastrointestinal tract were first described by Bauer in 1757 , 6. TheyOnly a quarter of the colonic lipomas become symptomatic . Abdomin Abdominal CT represents a valuable noninvasive imaging modality for the diagnosis of colonic lipomas . Barium Management of colonic lipomas includes endoscopy and surgery. Polypectomy through endoscopy is recommended for lipomas that are considered of low complication risk . These aColonic lipomas pose a preoperative diagnostic difficulty, since they may be asymptomatic for years or may be misdiagnosed as malignant tumors . Atypica


We present the case of a dermoid cyst with an oral and a submental component in a 21-year-old Japanese woman who presented with complaints of a mass in the oral cavity and difficulty in chewing and swallowing solid foods for about 2 years. MRI shows a 55 × 65 mm well-circumscribed cystic mass extending from the sublingual area to the mylohyoid muscle. Under general anesthesia and with nasotracheal intubation, the patient underwent surgical removal of the mass. Although the cyst was large and extending mylohyoid muscle, intraoral midline incision was performed through the mucosa overlying the swelling and the cyst was separated from the surrounding tissues with appropriate traction and countertraction and successfully removed without extraoral incision. Oral approach in surgical enucleation is useful procedure to avoid cosmetic problems in large and extending mylohyoid muscle cyst. Sublingual epidermoid and dermoid cysts are benign lesions encountered throughout the body, with 7% occurring in the head and neck area and 1.6% within the oral cavity . They reA 21-year-old Japanese woman was referred to our otolaryngology department with the chief complaint of a swelling below the tongue producing difficulty in chewing and swallowing of solid foods for about 2 years. Examination revealed the presence of a solitary midline swelling in the sublingual region measuring 6 × 5 cm. It was nontender, fluctuant, soft, and nonmobile, and the overlying mucosa showed no secondary changes. There were no inflammatory signs or lymphadenopathy associated with the swelling . Axial aEpidermoid and dermoid cysts of the oral cavity represent less than 0.01% of all oral cavity cysts . HistoloThe differential diagnosis of sublingual lesions includes infectious process, ranula, lymphatic malformation, heterotopic gastrointestinal cyst, and duplication foregut cyst –9. For t Treatment is by enucleation via an intraoral or extraoral approach. An intraoral approach is recommended by most authors for sublingual cysts of small or moderate dimensions (less than 6 cm) above the mylohyoid muscle, whereas an extraoral approach is preferred for larger sublingual cysts (more than 6 cm) , 12. In  Recurrence is very rare with complete excision of the lesion, but a 5% rate of malignant transformation of oral dermoid cysts into the teratoid type has been reported in literature .Appropriate imaging techniques are necessary in the preoperative diagnosis of dermoid cysts of the mouth. Oral approach in surgical enucleation is useful procedure to avoid cosmetic problems in large and entending the mylohyoid muscle cyst.


Spontaneous gastrointestinal neoplasms in non-human primates are commonly seen in aged individuals. Due to genetic similarities between human and non-human primates, scientists have shown increasing interest in terms of comparative oncology studies.Macaca nigra), kept on captivity by Matecaña Zoo, Pereira City, Colombia. The animal had abdominal distension, anorexia, vomiting, diarrhea and behavioral changes. Clinical examination showed an increased volume in the upper right abdominal quadrant caused by a neoplastic mass. The patient died during the surgical procedure. Necropsy revealed several small nodules in the peritoneum with adhesion to different portions of the small and large intestines, liver, stomach and diaphragm. Tissue samples were collected, routinely processed and stained by H&E. Microscopic examination revealed a mesenchymal tumor limited to tunica muscularis, resembling normal smooth muscle cells. Neoplastic cells were positive for alpha-smooth muscle actin and vimentin, and negative for cytokeratin AE1/AE3 by immunohistochemistry. Those morphological and immunohistochemical findings allowed to diagnose the intestinal leiomyoma referred above.The present study is related to a case of an intestinal leiomyoma in a black crested macaque (Neoplastic diseases in primates have multifaceted causes. Their manifestations are understudied, leading to a greater difficulty in detection and measurement of the real impact provides by this disease. According to some surveys, Spontaneous gastrointestinal neoplasm in non-human primates is the most common tumor described in literature, accounting for up to 48.8% of the cases . The disMacaca genus [Intestinal adenocarcinoma is the most prevalent tumor reported in primates belonging to ca genus , constitca genus . Howeverca genus , gastroica genus ,7, leiomca genus  and a poca genus , were alGalagoides demidovii) and cotton-tap tamarins (Saguinus oedipus) [Macaca sp. was not found, up to date.Leiomyoma, after adenoma, is the most common tumor of the small bowel in human ,11, whicoedipus) , a speciThe present study describes clinical signs, biochemical and hematological parameters, necropsy and microscopic findings of a case related to a spontaneous intestinal leiomyoma in a crested black macaque.A 6-year-old female crested black macaque, born on captivity in Matecaña Zoo, Pereira City, Colombia was presented at Veterinary Hospital for clinical evaluation. Reportedly, it was suffering from isolation, apathy, weakness, anorexia, and recurrent episodes of diarrhea, with loose stools, and vomiting. At first, a regular corporal condition of the animal was determined , with a weight of 6 kg. However, some clinical disorders as pale mucous membranes, weak pulse and mild hypothermia were presented. Auscultation revealed rattles at the lower respiratory tract and increased peristalsis. Palpation allowed detecting a marked abdominal distention mainly in the upper right quadrant caused by a firm and irregular intra-abdominal mass. Ultrasound evaluation confirmed the presence of an irregular, multilobulated mass measuring 3.0 x 5.0 x 3.0 cm, attached to internal organs. Other organs were not properly visualized because of an excessive gas accumulation in the small and large bowel .Peritoneal cavity was full-filled with an evident quantity of a hemorrhagic fluid. Peritoneal surface was thickened by scattered multifocal coalescent grayish-white, with firm and well defined nodules of variable sizes. There were multiple adhesions between the serosal surfaces of liver, stomach, peritoneum and diaphragm. A firm, multilobulated, non-encapsulated, poor defined grayish-white mass measuring 3.0x 5.0 cm x 3.0 cm, originated from the final portion of a stenotic ileum, filled the entire upper right quadrant. The exact origin within the ileum was not possible to determine. Stomach, small intestine and large intestine were distended by abundant gas. Pancreas showed small white nodules on surface. Spleen showed several small white dots on cut surface. The mesenteric lymph nodes were increased in size and thickness. Also, their consistency was firm. Samples from all affected organs were fixed in 10% neutral-buffered formalin solution, routinely processed and stained with hematoxylin and eosin (H&E) at the Laboratory of Pathology, Universidad de Caldas, Colombia.tunica muscularis. These cells were fusiform, having both an indistinct cell boundaries and an intensely and homogeneous eosinophilic cytoplasm. Sometimes, they displayed perinuclear clearing. The nuclei were elongated, cigar-shaped with a finely stippled chromatin pattern and a single central nucleolus. Mitotic figures were rarely seen. As a result of these findings, a small bowel leiomyoma was diagnosed. Liver revealed a thickened capsule. Its portal areas were infiltrated by a mild lymphoplasmacytic. Likewise, several intracytoplasmic clear vacuoles in centrilobular hepatocytes were found. Lungs had diffuse congestion, anthracnose and severe multifocal emphysema. Heart had a large amount of fibrillar and intense acidophilic material (fibrin clots) attached to epicardial surface. Kidneys showed a nephropathy, characterized by a decrease of Bowman’s space of some renal corpuscles. Also, dilatation and congestion of the peritubular vessels and a discrete intratubular protein deposit were present.Histopathological examination of small bowel mass tissue sections revealed an expanded, well delimited, non-encapsulated, highly cellular mesenchymal neoplasm, resembling normal smooth muscle cells, which were arranged in multidirectional fascicles and restricted to Immunohistochemistry was performed at Laboratory of Investigative and Comparative Pathology, Universidade Estadual Paulista, Botucatu, São Paulo, Brazil. Briefly, 4 mm histological sections were obtained in positive charged slides (Amitel®), which were deparaffinized in xylene and rehydrated in decreased ethanol concentrations. Tissue sections were exposed to heat in a Pascal chamber (Dako® Cytomation) with citrate buffer solution (pH 7.0) at 125°C for 30 sec in order to induce them to epitope retrieval (HIER). The slides were rinsed 3x by mean of Tris pH7.0. Then, they were incubated (overnight) by using primary antibodies such as vimentin, α-SMA, and cytokeratin AE1/AE3  is an Old World primate that lives in the rainforest of Sulawesi Island, Indonesia. Nowadays, it is considered one of the most endangered species [The crested black macaque ( species .M. mulatta) and 4 common marmosets [Galagoides demidovii) and cotton-tap tamarins (Saguinus oedipus) [Macaca mulatta), one leyomioma in stomach was differenced from 127 gastrointestinal diagnosed tumors [Macaca mulatta), from 61 tumors diagnosed in uterus, 30 of them corresponded to leiomyoma [Aotus nancymae) as a rare benign neoplasm [et al., 2012 [There are several reports regarding intestinal neoplasms in primates. A study on prevalence of spontaneous neoplasm in two colonies of Captive Rhesus Macaques found that the gastrointestinal system was the most commonly affected. It also established adenocarcinoma as the most prevalent identified tumor . Other sarmosets . Regardioedipus) . A resead tumors . Referreeiomyoma ). Finallneoplasm . The epineoplasm , or by tneoplasm . Accordil., 2012 , in humal., 2012 . In relal., 2012 , represel., 2012 . Some anCallithrix jacchus) suffering from small intestinal adenocarcinoma, nine of them had common symptoms related to weight loss and an intractable diarrhea, with variable presence of blood [The most common clinical symptoms associated with small bowel tumor are related to obstruction and bleeding. Obstruction usually manifests as recurrent crampy abdominal pain. The pain is usually periumbilical or epigastric, occurring after meals. Some people may experience associated bloating, nausea, or vomiting . Howeverof blood . Human pRelated to present study, patient´s symptoms such as anorexia, and recurrent episodes of diarrhea, with loose stools, and vomiting, match tract tumor cases reported in previous studies and revisited bibliography. Moreover, abdominal bloating symptom coincides with an accumulation of gas produced by a tumor intestinal obstruction, among other causes, as previously found in human -25. In oHelicobacter sp. bacteria is involved in the development of gastric and colonic carcinoma in human and immunodeficient mouse. Isolation of this species of bacteria in cotton-toptamarins (Saguinus oedipus), suffering from chronic colitis, showed no connection with a potential tumor formation [Lower gastrointestinal tract tumor is uncommon in domestic animal, especially in horse and production animals. However, this tumor has been found frequently in surgical biopsy applied to dogs and cats. Malignant neoplasm is more common than benign tumor. With the exception of lymphosarcoma, carcinomas are the most . In humaormation . On the ormation . Epsteinormation . Non-humormation .In human, there is a genetic predisposition to gastrointestinal cancer due to a defect in repair genes, or DNA mismatch repair genes (MMR), causing hereditary nonpolyposis colorectal cancer (HNPCC). Affected individuals are predisposed to develop neoplasms, and more frequently develop adenocarcinomas of the colon . As a suImmunohistochemical study revealed neoplastic cells negative for cytokeratin, and positive for vimentin and α-smooth muscle actin, which his consistent with an IHC profile of a smooth muscle neoplasm. Despite this, there are other immunohistohemical markers that could be useful in this case such as S-100, CD34 and CD117 (c-KIT). The c-KIT is expressed in gastrointestinal stromal tumor (GIST) ,31 togetThe hematological examination revealed leukocytes, exhibiting prominent neutrophilia and eosinophilia. Neutrophilia is a common indicator for an inflammatory response to an infectious or non-infectious origin. Likewise, eosinophilia is characteristic of allergic inflammation or parasites . Both, nThe serum chemistry parameters reached increased levels of ALT, AST and ALP, which collectively indicate liver injury. This was confirmed by histopathological analysis of the liver that revealed lymphoplasmacytic infiltrate in addition to several intracytoplasmic vacuolation (steatosis). By itself, creatinine is formed in the muscle during the metabolism of creatine. Then, it is subsequently diffuse into the blood, which is transported to the kidney, where is filtered by the glomerulus and, finally, is excreted in the urine . CreatinDevelopment of malignant neoplasms in general is due to a genetic predisposition and exposure to risk factors such environmental carcinogen. However, chronic inflammation and viral or bacterial gene mutations also could play an important role in promoting tumor development in both, human and animal.Neoplastic diseases in primates have multifaceted causes. Their behaviors are understudied leading to a greater difficulty in the detection and measurement of the real impact that this disease provides. The information of the basic factors that influence this disease such dietary habits, nutritional requirements, behavior, normal biochemical values and susceptibility to various agents is poorly understood. The understanding of the disease requires more detailed research efforts in order to understand pathogenesis and to create management plans  to improALT: Alanine aminotransferase; AST: Aspartate aminotransferase; ALP: Alkaline phosphatase; BUN: Blood urea nitrogen; DAB: Chromogen 3,3´-diaminobenzidine; CHV-3: Callitrichine Herpesvirus 3; HNPCC: Causing hereditary nonpolyposis colorectal cancer; CD34: Cluster differentiation; GGT: Gamma glutamyl transferase; H&E: Hematoxylin and eosin; HIER: Heat induced epitope retrieval; IHC: Immunohistochemical; MCV: Mean corpuscular volume; MCHC: Mean corpuscular hemoglobin concentration; MMR: Mismatch repair genes; α-SMA: α smooth muscle actin; S-100: S-100 protein; c-KIT: Tyrosine protein kinase Kit.None of the authors of the present document has conflict of interest for its publication.JMR: Clinical management of the case and elaboration of the preliminary report FPO, MMF, MAA: Analysis and interpretation of data, making and critical review of the manuscript. FG: Imunohistochemical analysis and critical review of the manuscript. All authors read and approved the final manuscript.


The spleen is an infrequent site for metastatic lesions, and solitary splenic metastases from squamous cell carcinoma of the esophagus are very rare: only 4 cases have been reported thus far. These lesions are whitish nodules that are macroscopically and radiologically similar to primary splenic lymphomas. We report a case of metachronous splenic metastases from esophageal cancer and multiple splenic abscesses, which developed nine months after apparently curative esophagectomy without adjuvant chemotherapy. The patient underwent splenectomy dissection followed by adjuvant chemotherapy, but liver and skin metastases developed, and the patient died 9 months later. Splenic metastases from solid malignancies generally occur within a setting of extensive multiorgan involvement. Over half of all patients with metastatic disease involving five or more organs have lesions in the spleen (often microscopic), and these patients represent 2-7% of those who die from end-stage metastatic disease ,2. It isThus far, 93 cases of solitary splenic metastases have been reported in the literature. Most are secondary to colorectal (20 cases), ovarian 18), or lung (10) cancers, and adenocarcinoma primaries are more common than epidermoid tumors ,8. Only , or lungA 59-year-old man was referred to the general surgery department of our hospital for a one-month history of progressive dysphagia for solids, which was not associated with malnutrition or significant weight loss. The patient had recently undergone esophagogastroduodenoscopy in another hospital, which revealed a bleeding, ulcerative lesion in the middle third of the esophagus, but no biopsy had been collected. The medical past history included COPD diagnosed in 1999 and a myocardial infarction in 2002. The patient had smoked approximately 25 cigarettes per day for several years.Physical examination was unremarkable. Computed tomography (CT) of the chest and abdomen revealed stenosis involving a 5-cm segment of the middle third of the esophagus with no other lesions in the thoracic or abdominal organs. Barium studies disclosed a swelling in the esophageal wall 7 cm above the cardia with an ulcerative pattern, which reduced the diameter of the lumen to 5 mm. An endoscopic biopsy of the oesophageal mass demonstrated poorly differentiated (G3) squamous cell carcinoma.Mid-distal esophagectomy was performed with oesophagogastric anastomosis and gastric tube reconstruction. Pathological examination of the surgical specimen confirmed the biopsy diagnosis of poorly differentiated (G3) SCC. The tumor, which measured 3 cm of length, had infiltrated the oesophageal wall and the surrounding paraesophageal fat. Surgical margins were tumor-free, as the seven perigastric limph-nodes dissected (pT3 N0). The postoperative period was quite unremarkable, and a contrast enhanced x-ray obtained on the 9th POD showed normal esophageal and gastric transit. On the 14th POD, the patient was discharged with an oncology referral for routine medical follow-up.Nine months after the operation, CT and esophagogastroduodenoscopy were repeated. The imaging study revealed mild splenomegaly with multiple nonspecific nodules within the organ Figure . The patth December 2007, the patient had transabdominal total splenectomy with splenic and celiac artery lymph node dissection.The patient was referred to our centre for splenectomy, which was performed as a routine procedure to role out, also, a spontaneous rupture of the spleen. On 12th postoperative day, Doppler ultrasonography revealed portal-tree patency with no signs of thrombosis. Ten days later, the patient was discharged with a stable platelet count , Hb 10.9 g/dL, and a WBC count of 16,500/mm3.The postoperative course was uneventful. On the 7Pathological examination of the spleen described multiple nodules containing medium to large-sized cells, some of which were keratinized. The nodules were mostly solid with areas of central necrosis Figure . The finThe spleen is tenth on the list of sites for solid tumor metastases -6,10. MeIsolated splenic metastases are very rare. Sileri et al. recently reported a case of a single splenic metastasis from colon cancer that appeared five years after surgery . SeveralConsensus holds that metastases to the spleen are hematogenous and may be a part of generalized blood-borne dissemination. This assumption is largely based on the hypothesis by Marymount and Gross, who concluded that splenic metastases arise from circulating cancer cells that arrive via the organ's arterial blood supply . Back inThe splenic lesions may be associated with vague non-specific symptoms or with pain or discomfort (like that reported by our patient). More frequently such condition are diagnosed incidentally on imaging studies performed during routine follow-up. The increasing availability and use of high-resolution modalities multi-slice CT and MRI has led to the identification of an increasing number of splenic metastases from different types of solid tumors, which previously escaped detection .Our experience confirms that locoregionally controlled esophageal cancer can be associated with splenic metastases. Our patient's lesions were discovered during a routine follow-up visit after a 9-month disease free interval, and the patient had no clinical evidence of systemic disease at the time. The splenic metastases appeared as nonspecific lesions on abdominal CT and were promptly subjected to FNAC. Space-occupying lesions in this organ are more commonly related to other causes, but when the patient has a history of malignancy, the possibility that the splenic lesions are metastatic must be ruled out.Splenic metastases are not a widely discussed problem. Because they are frequently associated with disseminated metastases, their presence is usually considered a sign of rapidly progressive disease that is no longer eligible for curative treatment. Noteworthy exceptions are cases occurring in patients with hematologic malignancies and those in which the splenic involvement is limited to a solitary lesion. The latter can sometimes be successfully managed with splenectomy, particularly when lesion onset occurs years after potentially curative surgical resection of the primary tumor. Further studies are necessary to define the prognostic impact of these lesions and their potential relation to the original histotype.Esophageal carcinoma is a rare cause of splenic metastases, and only four cases of isolated splenic metachronous secondaries have been reported thus far ,9,10. GiWritten informed consent was obtained from the next of kin of the patient for publication of study on January 2011. A copy of the written consent is available for review by the editor in Chief of this journal.FNAC: fine-needle aspiration cytology; COPD: chronic obstructive pulmonary disease; SSC: squamous cell carcinoma; CT: Computed tomography, MRI: magnetic resonance imaging; POD: post-operative day.The authors declare that they have no competing interests.IB: principle investigator who prepared, organized, wrote, and edited all aspects of the manuscript. JV: General surgeon who performed both operations and supported the work of principle investigator in preparing the manuscript. LC: supported the work of principle investigator in preparing the manuscript. GF: analyzed FNAC of the spleen and supported the work of principle investigator in writing and editing the manuscript. SB and VP: supported the work of principle investigator in writing and editing the manuscript. AV: chose imaging CT scan and supported the work of principle investigator in writing and editing the manuscript. MM and PD: They read, edited, and approved the final version of the manuscript. All authors read and approved the final version of the manuscript


Childhood lymphangiomatous polyp of the palatine tonsil is a very unusual lesion found in the head and neck. Tonsillectomy has been reported to be the curative procedure of choice for this lesion. We report a case of a very young child with unilateral lymphangiomatous polyp of the palatine tonsil. Lymphangioma or lymphatic malformation is a benign congenital tumor of the lymphovascular system that most commonly occurs in the head and neck region . LymphanLymphangiomatous polypoid lesions of the head and neck are very rare and case reports of these tumors arising from the palatine tonsils in the literature are sparse , 8–10.In this paper, we describe a case of a very young child who was found to have a lymphangiomatous polypoid lesion of the palatine tonsil.A 3-year-old girl was seen in the pediatric otolaryngology clinic for evaluation of dysphagia to solid foods. She had no other local or systemic symptoms. Esophagogastroduodenoscopy was arranged to rule out eosinophilic esophagitis and any structural pathologies. Intraoperatively, she was found to have a polypoid lesion arising from the superior pole of the right palatine tonsil . InteresThe endoscopic examination was normal and eosinophilic esophagitis was ruled out. The histopathologic sections of the lesion arising from the tonsil showed a polypoid portion of fibrous connective tissue with nonkeratinizing stratified squamous epithelium at the surface and dense lymphoid tissue at the base . CentralPostoperative followup at one year revealed no evidence of any residual or recurrent polypoid disease. Interestingly, the symptoms of dysphagia gradually improved without any other interventions.Lymphangiomatous polyps are uncommon benign lesions mostly occurring in the lower gastrointestinal tracts, including the colon , 6, the To date, there has been just over 30 cases of tonsillar lymphangiomatous polyps reported in the literature –10. Of tThe incidence of these tumors in the general population is currently unknown but many authors suggest that the true incidence is most likely higher than expected , 3. ThisPrior cases of lymphangiomatous polyp of the palatine tonsil have shown that it mostly occurs unilaterally and does not appear to have a gender predilection. The average age of patients in the aforementioned large case series was 25 years , and mosBenign tumors of the palatine tonsil are less common than malignancies . DiffereThe histopathologic features of a lymphangiomatous polyp that distinguish it from the other processes include dilated vascular channels containing luminal proteinaceous fluid and lymphocyte aggregation, which were present in the biopsied specimen in our case. When compared to other lymphatic malformations in the head and neck, the polypoid lesions differ in that they contain a greater degree of dense connective tissue and the lymphatic channels are not as prominently dilated.The pathogenesis of tonsillar lymphangiomatous polyps remains unclear. Chronic inflammation and associated obstruction of lymphatic channels were previously suggested as a possible mechanism , but thiLymphangiomatous polyps of the palatine tonsils have not been linked to other lymphatic lesions elsewhere in the head and neck region . Yet, onRegarding the management of the polypoid lesions, most authors suggest that a tonsillectomy is the curative procedure of choice , 10. Our


Inflammatory myofibroblastic tumor of the temporal bone is an unusual but distinct disease entity. The most common presenting symptoms are otalgia, otorrhea, hearing loss, facial palsy, and vertigo. We describe here what we believe to be the first reported case of a patient presenting with persistent pulsatile tinnitus. The clinical features, radiological and histopathologic findings, and treatment outcomes of the patient are presented.A 59-year-old woman of Chinese Han origin presented with complaints of left-sided pulsatile tinnitus and progressive hearing loss for several years. Clinical evaluations revealed a reddish mass behind the intact tympanic membrane, and a moderately severe conductive hearing loss in the left ear. The computed tomographic imaging of the temporal bone demonstrated a slightly ill-defined left middle ear soft tissue mass involving the posterior portion of the mesotympanum and epitympanum, and the mastoid antrum. The patient underwent surgical excision of the lesion which subsequently resolved her symptoms. Postoperative pathology was consistent with an inflammatory myofibroblastic tumor.An inflammatory myofibroblastic tumor of the temporal bone can present clinically with pulsatile tinnitus and masquerade as venous hum or vascular tumors of the middle ear; therefore, it should be included in the differential diagnosis of pulsatile tinnitus. It is ach cases . Howeverch cases . Intratech cases -10. ThisIn this article we describe what we believe is the first reported case with an IMFT in a 59-year-old woman presenting with persistent pulsatile tinnitus. The clinical features, radiological and histopathologic findings, and treatment outcomes are presented.A 59-year-old woman of Chinese Han origin presented with a complaint of a left pulsatile tinnitus. She had been extremely depressed for 10 years because this tinnitus was constant and prevented her from sleeping. She had visited the otological clinics at different hospitals several times. At each presentation, the tympanic membrane was carefully examined otoscopically and was found to be normal. A computed tomographic (CT) scan of the temporal bone was also performed 9 years prior to her presentation to our hospital with a negative finding. With the initial diagnosis of venous hum, she received no effective treatment.The patient was referred to us for further evaluation on January 17, 2007, due to the unbearable left tinnitus and a new awareness that her hearing had gradually deteriorated for 2 years. Her medical history was otherwise unremarkable except for hypertension, which was under treatment. On examination, the pulsatile tinnitus could be completely suppressed by digital pressure over the ipsilateral internal jugular vein or when the patient was asked to turn her head towards the side of tinnitus. Cranial, carotid and chest auscultation were negative. Other examinations including carotid ultrasonography, electrocardiography, thyroid function tests, and chest X-ray were normal. Careful otoscopic examination revealed a vague and reddish mass behind the superior-posterior portion of the intact left tympanic membrane. A pure tone audiogram showed a moderately severe conductive hearing loss in the left ear, especially at lower frequencies; middle ear impedance testing ascertained a flat tympanogram of the same ear. High resolution CT scans of the temporal bone showed a slightly ill-defined left middle ear soft tissue mass involving the posterior mesotympanum and epitympanum, and the mastoid antrum with erosive changes of the facial fallopian canal  The patient we present here was a 59-year-old woman. She had a complaint of left-sided pulsatile tinnitus for 10 years, and recently a progressive hearing loss in the same ear for 2 years. Otoscopic examination revealed a reddish middle ear mass behind her left tympanic membrane. CT scans of the temporal bone demonstrated a soft tissue mass around the incus with extension into the posterior portion of the mesotympanum and epitympanum. For that reason, the clinical suggestion was a possible neoplastic or inflammatory lesion, especially a vascular tumor. Histologically, diagnosis of IMFT was a surprise.IMFT or inflammatory pseudotumor is an unusual disease entity. The World Health Organization classification recognizes this disease, but very few cases in the temporal bone area have been published ,4,15. ThA review of the literature suggests that the IMFT of the temporal bone has no distinctive characteristics, both clinically and radiologically. Thus, a diagnosis can only be made with histopathologic appearances and is confirmed by immunohistochemical studies . The difIMFT of the temporal bone is an unusual but distinct disease entity. The most common presenting symptoms are otalgia, otorrhea, hearing loss, facial palsy, and vertigo. An IMFT can also present clinically with pulsatile tinnitus and masquerade as venous hum or vascular tumors of the middle ear, as in our case report. Therefore, an IMFT should be included in the differential diagnosis of pulsatile tinnitus, especially for those cases with a visible retrotympanic mass lesion.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.GX analyzed and interpreted the patient data regarding the ear disease. XZ and TL were major contributors in writing the manuscript. ZC participated in the surgical treatment of the patient. ZZ performed the histological examination of the specimen. All authors read and approved the final manuscript.


Contrary to its name, synovial sarcoma does not arise from the synovial membrane but from multipotent stem cells and can present in any part of the body. Very few cases of vulval synovial sarcoma have been reported in the literature; we report on such a presentation. These tumors can present as painless lumps, which must be completely excised to give the best prognosis. Therefore the diagnosis of synovial sarcoma should always be kept in mind in the management of vulval masses, especially in young patients.We report the case of a 28-year-old Caucasian woman with synovial sarcoma of the vulva. Complete excision was possible in this case.We have presented a rare case of synovial sarcoma of the vulva, which can be easily confused with lipoma of the vulva. The management of this tumor requires referral to a cancer centre, with a multidisciplinary approach. Synovial sarcoma is the fourth most commonly occurring sarcoma, accounting for 7-8% of all sarcomas . It mostThe term synovial sarcoma was coined to denominate tumors arising near tendon sheaths and joint capsules. Despite its name, synovial sarcomas do not appear to arise from synovial membranes, but from unknown multipotent stem cells that are capable of differentiating into mesenchymal and/or epithelial structures  and lackOnly five cases of synovial sarcoma arising from the vulva have been reported in the literature  as summavirgo intacta, was referred to us, presenting with a four-month history of a left vulva swelling, which was becoming increasingly uncomfortable. There was no other gynaecological history to note, and our patient did not report masses elsewhere.A 28-year-old Caucasian woman, Clinical examination revealed a healthy woman with a well-defined deep mobile mass in her left labium majus. She underwent excision of the swelling under general anesthetic and the lump, which did not appear to be attached to adjoining structures, was dissected free from the underlying tissue.Histology demonstrated a large biphasic tumor (measuring 2.2 × 1.5 × 1.0 cm) consisting of epithelial cells and fibroblast-like spindle cells, with the presence of glandular structures and mitoses. There was also a hemangiopericytomatous pattern in some areas, with spindled and rounded cells adjacent to numerous vessels without muscular walls, and myxoid areas together with zones of definite formation of epithelial structures with large lining cells surrounded by a spindle cell matrix. Bone formation was seen focally and there was a retiform component to the tumor  scan of the chest, abdomen and pelvis showed no evidence of metastatic disease. Re-excision by a consultant gynecological oncologist showed no residual tumor on histology. After discussion at our sarcoma multidisciplinary team meeting, it was felt that further adjuvant therapy was not necessary in view of the completeness of excision. We have followed up our patient with clinical examinations and a chest X-ray every three months for the last three years, and there is still no evidence of recurrence.Synovial sarcoma is a soft tissue sarcoma and is divided into three subtypes: biphasic, monophasic and poorly differentiated . The bipIt is difficult to recognize synovial sarcoma on the basis of only its histological appearance. In most cases, it can only be unambiguously identified by immunohistochemical analysis, ultrastructural findings and the demonstration of the specific chromosomal translocation mentioned above.It most commonly presents as a slowly growing, painless mass, with the most common sites of presentation being at the extremities. The lower extremities are more frequently affected than upper extremities, often in the region of the thigh and knee. The peri-articular regions are especially affected, usually in close association with the tendon sheath, bursae and joint capsules, but they rarely involve the articular surface . HoweverA definitive diagnosis can only be established by an adequate tissue biopsy, but radiological investigations may be useful to characterise the tumor. On a CT scan, the tumor usually shows as a heterogenous septate mass with a mixed solid and cystic appearance; calcification can be seen in one third of them. Magnetic resonance imaging (MRI) provides a greater contrast between tumor and normal tissue and can show neurovascular or regional lymphatic involvement . RadioloThe presence of metastases, a tumor size greater than 5 cm, invasiveness, high histological grade, positive surgical margins and poor histological differentiation are all associated with adverse prognostic significance .The complete surgical excision of the tumor is the treatment of choice. Often, synovial sarcoma has a pseudocapsule that allows the tumor to shell out fairly easily, giving a false sense of security that complete removal has occurred . The preRadiotherapy appears to have a role in the treatment of patients newly diagnosed with synovial sarcoma, especially children with minimal primary tumor or following surgery . RadiothFuture areas of investigation should include the role of molecular genetics to evaluate for possible targeted therapy, as well as the role of chemotherapy and radiation in improving prognosis.This is a rare case of synovial sarcoma of the vulva, the appearance of which can be easily mistaken for a lipoma. The overall prognosis for our patient should be good given the complete excision of the tumor. Other favorable factors affecting the prognosis include a tumor size of less than 5 cm and no evidence of metastatic spread.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.VH was involved in pre- and post-operative care of the patient and wrote the manuscript. GVS performed the histopathological examination and made the histological diagnosis. AB performed the surgery and helped in correction of the manuscript. All authors have read, approved and contributed towards the final manuscript.


Primary tracheal tumors occur infrequently, accounting for less than 0.1% of all tumors. Adenoid cystic carcinoma (ACC) is the second most common type of malignancy of the trachea after squamous cell carcinoma (SCC). Little has been reported on the risk factors for tracheal ACC. The purpose of this study is to describe a case of tracheal ACC in a patient who had been exposed to rubber fumes, and to review the relationship between tracheal ACC and rubber fumes.A 48-year-old man who had been experiencing aggravation of dyspnea for several months was diagnosed as having ACC of the trachea on the basis of a pathologic examination of a biopsy specimen obtained via laser microscopy-guided resection. The patient had been exposed to rubber fumes for 10 years at a tire manufacturing factory where he worked until ACC was diagnosed. His job involved preheating and changing rubber molds during the curing process.ACC of both the trachea and the salivary glands show very similar patterns with regard to histopathology and epidemiology and are therefore assumed to have a common etiology. Rubber manufacturing is an occupational risk factor for the development of salivary gland tumors. Further, rubber fumes have been reported to be mutagenic. The exposure level to rubber fumes during the curing process at the patient’s workplace was estimated to be close to or higher than British Occupational Exposure Limits. Therefore, tracheal ACC in this case might have been influenced by occupational exposure to rubber fumes. The incidence of primary tracheal carcinoma is 0.10 to 0.26 per 100,000 persons , accountDue to the low incidence rate, little has been reported on the risk factors for tracheal ACC ,9. The oRubber fumes consist of respirable dust generated during the compounding or curing processes of tire manufacturing. Although the specific carcinogen(s) have not yet been identified, several reports have described the mutagenic potential of rubber fumes -14. The A 48-year-old man first visited an otolaryngology clinic in May 2005 because of exertional dyspnea. His symptoms persisted and deteriorated over several months, and he was found to have a tracheal tumor. At this point, he was transferred to a university hospital. The patient did not have hypertension, diabetes, tuberculosis, viral hepatitis, or HIV infection. Several years earlier, he had undergone an operation for appendicitis. He did not have any known drug allergies. His family did not have a history of respiratory cancer. The patient reported that he had smoked approximately 4 cigarettes a day since he was 23 years old and had drunk 50 mL of Korean distilled spirits per day since he was 20 years old. He had no dyspepsia or resting dyspnea. Visual inspection of the larynx did not reveal any remarkable findings. The Epstein-Barr virus (EBV) antigen/antibody status was not available from the medical records.Computed tomography (CT) of the patient’s neck, conducted on May, 2005, revealed a 3.5-cm polypoid soft-tissue tumor at the inner side of the right posterior wall of the subglottic region, extending to the outer side. The tumor blocked the inner cavity of the trachea. Laser microsurgery was conducted for partial excision of the tumor and frozen section biopsy. The final diagnosis on the basis of the frozen biopsy was tracheal ACC, with no extra-tracheal metastasis. Adjuvant radiation therapy was administered twice during next year. A follow-up medical examination was performed 5 years later, during which, chest radiography revealed multiple metastatic nodules in both lung fields. The patient subsequently received palliative chemotherapy.The patient was discharged from the army after 3 years of service 1977–1980) and worked in rice farming (1981–1994). In 1995, he entered tire manufacturing, and was assigned to the curing process, where he worked for 10 years, until 2005. The curing process involved the use of 72 vulcanizers . His main duty was preheating and changing molds and bladders. Groups of 3 workers were required to perform the mold-changing job. The workers moved the molds and bladders by using a forklift after the molds had been used several times or if the size of the tire being made needed to be changed  determined by the Korean Ministry of Employment and Labor [3) [3, which was less than 10% of the Korean OEL (200 μg/m3) [Available environmental measurement data provided by the company included information from 1997 to 2005, but information for 1999 was missing. The risk factor measured by the company was total dust until 2002. The company then changed to measuring rubber fumes (Table nd Labor . The expabor [3) . Other pabor [3) . N-nitro0 μg/m3) . Tempera0 μg/m3) .There was a possibility of exposure to PAHs vaporized from anti-rust agents applied to molds due to the high temperature. In addition, cleaning the molds with an airbrush might have generated dust from pipe insulation materials. We analyzed a sample of those materials for the presence of asbestos and PAHs. The insulation material was found to contain glass fiber. Major components of the anti-rust agents were found to be mineral oil  and barium sulfate ; however, when these chemicals were exposed to temperatures similar to those used during the mold preheating process (180‒195°C ), PAHs were not detected, as assayed by the gas chromatography–mass spectrometry (GC-MSD) analytical method.Tracheal ACC is known to originate from the salivary glands of the trachea . TherefoThese similarities indicate that tracheal ACC and HNACC may have common risk factors. Because HNACC is one of many subtypes of salivary gland tumors of the head and neck, risk factors for such a specific subtype are not well-known . Therefoet al.[et al.[Salmonella typhimurium strains, which overproduce specific enzymes such as nitroreductase and acetyltransferase in the presence of mutagenic material, and proposed that some components of rubber fumes may have mutagenic potential, and these may include aromatic amines. Kim B et al.[Mancuso et al. suggestel.[et al. used Sal B et al. reportedLee et al[3) [The present case was potentially exposed to carcinogens including PAHs and N-nitrosamines. PAHs are generated by incomplete combustion of organic material and are known to increase the risks of lung cancer and skin cancer . N-nitroLee et al. Indeed,et al[3)  (Table 2This study has some limitations. We aimed to describe a relationship between rubber fume exposure and the development of tracheal ACC. First, for the 10-year exposure period, information regarding the first 6 years was not available. This forced us to rely on estimations based on statements from the patient and measurements from a tire manufacturing company in a previous study. Second, we presumed that the risk factors for head and neck salivary gland tumors and tracheal ACC are similar, which needs more thoughtful consideration. Third, it is known that the chemical composition analysis of rubber fumes is difficult and that data about their harmful effects are scanty , so we cDespite the above limitations, this study is the first case report that suggests a relationship between exposure to rubber fumes and the development of tracheal ACC. However, in order to definitively prove the relationship between rubber fumes and occupational cancer, further epidemiological and experimental studies are necessary.In conclusion, tracheal ACC and salivary gland tumor of head and neck have very similar patterns with regard to their histopathology and epidemiology, and may therefore share a common etiology. Rubber manufacturing is considered to be a risk factor of salivary gland tumors, and rubber fumes have been reported to have mutagenic and toxicological properties. Exposure levels of rubber fumes for this case are estimated to have been close to or higher than the British OEL, but those of other potential carcinogens such as PAHs and N-nitrosamines are not. Although the patient was an ex-smoker, tracheal ACC is not known to be associated with a smoking habit. Besides, this patient was not concluded to have been exposed to other risk factors of salivary gland tumors such as HIV infection or ionizing radiation. Therefore, it is suggested that the development of tracheal ACC in this case may have been influenced by occupational exposure to rubber fumes.Written informed consent was obtained from the patient’s guardian/parent/next of kin for the publication of this report and any accompanying images.The authors declare that they have no competing interests.KDH and KEA conceived and designed the study. KDH, CSY and HYI were involved in conduction of the study. LNR and HYI performed the analysis and estimation of the environmental assessment. PCY and KEA interpretation of data. KEA, KDH, PCY, LNR and HYI were involved in writing the manuscript. KBG and YSD performed the revision the manuscript. All authors read and approved the final manuscript.