Dentigerous cysts (DCs) are benign odontogenic cysts that are associated with the crowns of permanent teeth. The purpose of this study is to describe the management of DCs in four children. Four boys aged between 7 and 9 years were referred to our clinics with the complaints of intraoral alveolar swelling or facial asymmetry on the affected area. The panoramic radiographies showed large, well-defined radiolucent lesions associated with the deciduous teeth and displaced tooth buds. The treatment consisted of the extraction of the involved deciduous tooth and marsupialization of the cyst to allow eruption of the permanent tooth. Permanent teeth displaced by the DCs in three cases erupted spontaneously within one-year period. The case with horizontally displaced permanent tooth was managed by replantation. This is the first time that underlying permanent tooth in a DC case was intentionally replanted. Dentigerous cysts (DC) are benign odontogenic lesions caused by an alteration of reduced enamel epithelium which results in fluid accumulation either between the reduced enamel epithelium and the crown of an unerupted/impacted tooth or within the enamel organ itself. DCs are usually asymptomatic, and they may be detected incidentally via routine radiographic examination or when they are large enough (>2 cm in diameter) to cause facial asymmetry –3. CliniDifferent theories have been proposed to explain the etiology of the DC. The first theory suggests that DCs are developmental in origin and occur in mature teeth usually as a result of impaction. These cysts usually occur in the late second and third decades, predominantly involving mandibular third molars. The second theory advocates that DCs are inflammatory and occur in immature teeth as a result of inflammation from a nonvital deciduous tooth or another source spreading to involve the tooth follicle. These are diagnosed in the first and early part of second decade, predominantly involving mandibular premolars .It is important to emphasize that DCs in children might show a rapid and painless expansion and may result in fractures and deformation of facial bone structures. Early diagnosis and treatment are therefore important. The aim of the present paper is to describe clinical, radiological, and histopathological findings of four additional cases with DCs treated by conservative surgical techniques. To our knowledge, this study constitutes the first reported case of DC in which affected permanent tooth was intentionally replanted to its place after marsupialization.Four boys, aged between 7 and 9 years, were referred to clinics in the Department of Pediatric Dentistry at Istanbul University with the chief complaints of intraoral alveolar swelling or facial asymmetry on the affected area. The patients were evaluated by a comprehensive clinical and radiologic examination, including panoramic radiography and cone beam computed tomography (CBCT). Clinical and radiographic images of these cases are shown in Figures All of the cases were treated by extraction of the deciduous tooth around the cyst and marsupialization of the cyst to allow eruption of the permanent tooth. In all cases, a biopsy tissue sample was taken from the access window of marsupialization for the histopathological examination, and then a silicon tube was installed to drain the intracystic pressure. The histopathologic examination of the lesions confirmed the diagnostic hypothesis of a dentigerous cyst with inflammatory origin.In our study, the histopathologic features of all four cases were similar. Figures Permanent teeth displaced by the DCs in three cases erupted in proper position spontaneously within a one-year period. The other case with a horizontally displaced permanent premolar did not have a chance to erupt spontaneously; thus intentional replantation was chosen as the treatment. After nine months of healing of the cyst cavity, the tooth was extracted and replaced in its proper place in the arch by surgery in a position where the alveolar bone support is highest. Therefore, the crown of the replanted tooth was in a slightly vestibular position at the end of surgery. The position of the tooth was corrected orthodontically. A follow-up visit at twenty-eight months showed complete healing of the surrounding bone and the tooth was asymptomatic.DCs are the third most common odontogenic cysts, after radicular cysts and odontogenic keratocyst, and account for approximately 10.39% of all odontogenic cysts . PatientConsidering that the size of a normal follicular space in radiographs is 3 to 4 mm in diameter, a DC can be suspected when a follicular space measuring more than 5 mm is observed , 10. ConRadicular cysts described in association with the pulpotomized primary molars seem to show similar clinical features with inflammatory dentigerous cysts. Several differences were reported between the radiological features of radicular cysts and dentigerous cysts by various reports –13. BasiDCs are treated via surgical enucleation of the cystic lesion or marsupialization technique. Enucleation involves the complete removal of all tissues involved with the cyst, including removal of the unerupted tooth. Marsupialization or decompression is a technique that attempts to relieve intracystic pressure through the creation of an accessory cavity . The treThe major disadvantage of marsupialization is pathologic tissue left in situ without a thorough histological examination, because of the possibility that a more aggressive lesion may occur in the residual tissue. However, recurrence of DC is rare, especially after complete removal of the cyst or tooth eruption . In the Conservative treatment is a favorable treatment modality for DCs in growing children and adolescents. Considering that the regeneration capacity of the bony structures in children is greater than in adults and the teeth with open apices have greater eruptive potential, efforts should be made to allow the involved tooth to erupt spontaneously. Intentional replantation could also be employed in teeth which have a great strategic value for maintaining the balanced occlusion. It is important to follow up with patients regularly for the possibility of recurrence.


Colonic lipomas are rare benign tumors. Colonic intussusception is an uncommon complication of colonic lipoma. The current study presents an unusual case of a 4-cm symptomatic lipoma of the transverse colon causing colonic intussusception. A 65-year-old female was admitted to Wenzhou Central Hospital  with intermittent pain in the left abdomen that had been present for two weeks. Colonoscopy revealed a 4×5-cm intraluminal spherical mass with erosional mucosa 60 cm above the anal verge, indicating the presence of a malignant gastrointestinal stromal tumor. Contrast-enhanced computed tomography revealed a well-defined fatty tissue mass of 4 cm in diameter in the distal transverse colon proximal to the splenic flexure, with intussusception. The patient underwent segmental resection of the transverse colon and intraoperative frozen sections were obtained. The intraoperative frozen sections revealed a submucosal lipoma of the transverse colon and thus, a conclusive diagnosis was achieved. The patient was followed up for one year and 10 months following the segmental resection of the transverse colon, with a good prognosis. This study may increase clinical awareness with regard to colonic lipomas. Furthermore, open surgery combined with use of intraoperative frozen sections should be recommended for large symptomatic colonic lipomas accompanied by colonic intussusception, thus avoiding unnecessary radical resection and improving patient prognosis. Lipomas of the gastrointestinal tract are benign tumours and were first reported by Bauer in 1757 . General9 cells/l, a neutrophil count of 9.2×109 cells/l (73.2%) and a C-reactive protein level of 10.4 mg/l. Tumor markers were within the normal ranges. Colonoscopy revealed a 4×5-cm intraluminal spherical mass, 60 cm above the anal verge, which prevented further progression of the endoscope. The mass was covered by a 2×3-cm superficial mucosal erosion, indicating the presence of a malignant gastrointestinal stromal tumor  with intermittent pain in the left abdomen that had been present for two weeks. The patient had no past history of cancer and no family history of colorectal cancer. The physical examination was unremarkable. The laboratory results revealed a white blood cell count of 12.5×10al tumor . In addial tumor , with inal tumor  and locaThe patient underwent segmental resection of the transverse colon following the initial diagnosis. The intraoperative frozen section revealed a submucosal lipoma of the transverse colon. No further resection was required. Macroscopic assessment of the resected specimen identified the presence of a yellow, round and broader-based 4×4-cm mass exhibiting the features of a lipoma . HistopaColonic lipomas are rare benign tumors of the gastrointestinal tract and are classified as a type of benign non-epithelial tumor. The incidence of colonic lipoma ranges between 0.035 and 4.4% , and theThe size of colonic lipomas ranges between 2 mm and 30 cm and may mimic colonic malignancies . The preHowever, an intraoperative frozen section may provide an accurate diagnosis to guide surgery . In the In conclusion, colonic lipoma is a relatively rare benign tumor, which as a clinical entity may be easily misdiagnosed as a malignant tumor. The clinical awareness of colonic lipomas must be increased. Open surgery combined with the use of intraoperative frozen sections should be recommended for large symptomatic colonic lipomas accompanied by colonic intussusception, thus avoiding unnecessary radical resection and improving the patient prognosis.


Severe paroxysmal sympathetic overactivity occurs in a subgroup of patients with acquired brain injuries including traumatic brain injury, hypoxia, infection and tumor-related complications. This condition is characterized by sudden increase of heart rate, respiratory rate, blood pressure, body temperature and excessive diaphoresis. The episodes may be induced by external stimulation or may occur spontaneously. Frequent occurrence of this condition could result in secondary morbidities, therefore, should be diagnosed and managed insightfully. These symptoms could be confused with seizures or other medical conditions, leading to unnecessary treatment. Despite clinical significance of paroxysmal sympathetic hyperactivity (PSH), brain tumor-induced PSH has not been studied nearly. In this report, two cases of the PSH in patients with brainstem-compressing benign tumors were introduced. The most useful pharmacologic agents were opioid  in preventing PSH attack, and nonselective β-blocker  in relieving the symptoms. Clinical experiences of the rare cases of benign tumor-induced PSH can be helpful as an essential basis for further research. Episodes of acute sympathetic disturbances may occur after acquired brain injuries, such as traumatic brain injury, hypoxia, infection, and tumor-related complications  and intermittent dystonic movements. In neurologic examination, he demonstrated alert mentality and his pupil was slightly dilated and decreased light reflex in right side. In addition, it is also identified that the decreased swallowing function and dysarthria. A MRI scan showed an 8.1 cm-sized calcified extraaxial mass over left middle and posterior cranial fossa with petrosal apex epicenter. At the same time, the midbrain, pons, and left middle cerebral peduncle was compressed and deviated to right side  were administrated and maintained within therapeutic level of concentration in blood, the paroxysmal sympathetic hyperactivities (PSH) were repeated. In blood chemistry, the level of glucose and electrolytes were within normal ranges. The inflammation markers, such as WBC, ESR, CRP, and lactic acid were also not significant. There were no active lung lesions on chest X-ray and urine analysis was also normal.Initially, the beta antagonist  and diazepam (2 mg/day) were tried under impression of PSH for several days, however, there was no effect in prevention of the PSH. The baclofen (10 mg/tid/day) was added, but not effective. The opioid patch was applied and the occurrence of the PSH was gradually decreased over a week. After 10 days of the opioid patch application, the PSH was not observed, however, abdominal distention and constipation occurred. To relief the constipation, the opioid patch was removed, and then the PSH was recurred at 3 days after cessation of the patch. Therefore, the opioid patch  was reapplied and PSH was subsided again. At same time, stool softner  and bowel motility drugs  were administered and intermittent enema was also performed. The use of the opioid patch was tapered gradually after 1 week of reapplication, and the PSH was not recurred.A 30-year-old man was referred with stupor consciousness from other hospital after primary surgery. He was diagnosed as the huge sized jugular foramen schwannoma with compressing brainstem Fig. a. He alrThe mass was subtotally removed via previous craniotomy site. The firmly adhesion lesion to brainstem was left, because of concerning about brainstem injury. In postoperative CT scan, it was observed the focal hemorrhage and mild edema in brainstem Fig. b. His neAt 2 weeks after surgery, the paroxysmal increase of the heart rate, respiratory rate and blood pressure was observed, as well as extensor posturing, reactive to bathing or repositioning of the body. These symptoms were resolved spontaneously within several minutes. There were no significant findings in EEG, CT scan, and blood chemistry. The diazepam and baclofen were not effective in preventing the PSH, therefore, the opioid patch  was applied. The PSH had been disappeared gradually over a week. The opioid patch was used totally for 2 weeks, the PSH didn’t recur any more.The distinctive clinical features of sudden increase of sympathetic activities responding to non-noxious external stimuli have been described a wide range of labels, dysautonomia, sympathetic storming, hypothalamic-midbrain dysregulation syndrome, diencephalic epilepsy, and paroxysmal sympathetic hyperactivity (PSH)  , followed by hypoxia (9.7 %), stroke (5.4 %), unspecified (1.8 %), hydrocephalus (2.6 %), tumor (0.6 %), hypoglycemia (0.3 %), CNS infection (0.3 %) in precedence factors order Meyer . AlthougUntil now, the postoperative PSH in brainstem compression benign tumors were not extensively reported. In this report, huge petroclival chondrosarcoma (patient 1) and jugular foramen schwannoma (patient 2) were compressing brainstem with edematous changes. In both cases, the tumors were firmly adherent to brainstem, as a result, some of tumors remained at brainstem region. Focal brainstem hemorrhage and edema were observed at brainstem, although, the neurologic status was not changed relative to preoperative condition. After 3–4 weeks after surgery, a paroxysmal sympathetic hyperactivities, increasing HR, RR, BP, BT and extensor posturing were observed responding to non-noxious stimuli. These clinical phenotypes were needed to make a differential diagnosis from several clinical conditions including seizure, hydrocephalus, encephalitis, electrolyte imbalance, hypoglycemia, sepsis, and spinal cord injury. Initially, EEG was monitored to exclude seizure, however, no epileptiform discharges were identified. In blood chemistry, electrolytes, glucose, CO2 levels were within normal range, and there were no evidence for inflammatory condition. A CT scan also showed no hydrocephalus. Numerous researches have proposed lesion locations associated with the PSH, however, the definite lesions induced the PSH were still elusive.Characteristically, a number of previous studies have reported an association between afferent stimuli and sympathetic paroxysms  and hypoxia could cause PSH. Although the pathophysiology of the PSH is still elusive, it appears to be evident that brainstem injury is associated with the PSH. Most cases of the PSH reported to date occurred after TBI and hypoxia. However, the postoperative PSH cases in benign brain tumors compressing brainstem have not been reported extensively. In this report, the postoperative PSH in huge benign brain tumors compressing brainstem was effectively treated with opioid patch. The opioid was effective in preventing the PSH spell, as well as the intensity of it. The constipation was occasionally problematic, however, it could be controlled with intermittent enema and bowel motility drugs.The patient and guardian have consented to the submission of the case report for submission to the SpringerPlus.


Inferior thyroid artery (ITA) rupture is rare and may progress to life-threatening conditions. We present a patient who visited the emergency department after an episode of syncope and dizziness in which he had a mechanical fall that resulted in abrasions and a hematoma to his left forehead. The patient presented with dysphagia and anterior neck swelling that progressed rapidly into airway compromise requiring endotracheal intubation. Emergent computed tomography revealed a large retropharyngeal hematoma, with active arterial extravasation that was thought to be arising from the thyrocervical trunk on the left. The hematoma measured approximately 6.7 cm transversely and 3.2 cm anteroposteriorly and extended from the level of the lower nasopharynx, down the neck into the retropharyngeal and danger space and into the mediastinum posterior to the esophagus, overall approximately 25 cm. The larynx was deviated anteriorly and there was esophageal compression. An emergent arteriogram and catheterization confirmed bleeding from branches of the ITA, and successful embolization was performed. It is important to recognize the ITA rupture as a potential etiology of an acute airway compromise. In emergent situations, while securing an airway is a priority, rapidly initiating diagnostic testing to confirm the diagnosis and arranging for arterial embolization can be life-saving. Aneurysm and rupture of the inferior thyroid artery (ITA) are rare and less than 25 cases have been reported in the literature, the first reported in 1959 –9. A retAn 80-year-old male presented to the emergency department (ED) after an episode of syncope and dizziness in which he awoke on the floor earlier that morning. The patient did not complain of a headache or neck pain but expressed concern with dysphagia. The patient had a cardiac history consisting of controlled response ventricular atrial fibrillation and was removed from his Coumadin two weeks priorly on the recommendation of his cardiologist due to recurrent dizziness, syncope, and frequent falls. His records revealed that his cardiologist felt his history of dizzy and syncopal episodes was not due to a cardiac etiology. He was on sotalol for the atrial fibrillation. Two years priorly the patient had two-vessel coronary artery bypass surgery. Additionally he had a history of hyperlipidemia and gastroesophageal reflux disease and was due for surgery on a 4.8 cm abdominal aortic aneurysm.On examination, his vital signs were temperature 97.7°F, blood pressure 160/94 mmHg, pulse rate 65 beats/minute, respiratory rate 18 breaths/minute, and oxygen saturation 100%. He had a small abrasion and contusion on his left forehead. He was awake, alert, and coherent but appeared uncomfortable with prominent soft tissue in the anterior neck. His heart rate had a controlled ventricular response and his lungs were clear to auscultation. His coagulation laboratory studies were normal.Very rapidly over the next ten-fifteen minutes the patient had more difficulty swallowing and obvious swelling started evolving in the anterior neck region on the left. Within thirty minutes his dysphagia progressed and the patient visibly had increasing fullness on the left side of his neck and anterior chest wall without crepitus. The patient became hoarse and began to posture into the sniff position. Due to airway compromise, endotracheal intubation with mechanical ventilation was performed without complication. An enhanced computed tomography (CT) of the soft tissues of the neck, cervical spine, and abdomen was performed. The chest CT revealed a large retropharyngeal hematoma posterior to the level of the thyroid gland with active arterial extravasation thought to be coming from the thyrocervical trunk. The hematoma measured 6.7 cm transversely and 3.2 cm anteroposteriorly and extended from the level of the lower nasopharynx down the neck into the retropharyngeal space and into the mediastinum posterior to the esophagus, totaling 25 cm. The larynx was deviated anteriorly by the hematoma, in addition to esophageal compression. The cervical spine CT revealed prevertebral soft tissue swelling displacing the endotracheal tube anteriorly. The CT of the abdomen incidentally confirmed the 4.8 cm abdominal aortic aneurysm.An emergency arteriogram and embolization were requested. Selective catheterization of the left thyrocervical trunk revealed evidence of a large hemorrhage coming from branches of the ITA which was subsequently successfully embolized.Patients with an ITA rupture can either be asymptomatic or present with symptoms such as hoarseness, dysphagia, or swelling in the neck –4, 6. ThEmergency medicine physicians are frequently the first point of contact for such symptoms, so correct diagnosis and management are essential in preventing fatal outcomes. In emergent situations, a secure airway needs to be established, an enhanced CT and angiography allow for diagnosis, and arterial embolization is minimally invasive and should be the first choice for stopping the bleeding , 6, 8–10


Swyer syndrome or pure gonadal dysgenesis 46, XY is a medical condition associated with 46 XY karyotype and primary amenorrhea in a phenotypic female. In this syndrome, there is an abnormality in testicular differentiation. Patients with disorders in sexual differentiation have an increased risk for development of genital malignancies. A 14-year-old female admitted with abdominal pain was diagnosed to have Swyer syndrome and a pelvic tumor after clinical and laboratory investigations. She underwent surgery, and the histology report revealed a mixed germ cell tumor in a dysgenetic gonad. She recurred three months later and was successfully treated with chemotherapy and a second surgery to remove the differentiated teratoma. The early diagnosis of patients with Swyer syndrome is important because of the increased risk for the development of malignancy. Early surgical treatment is required. Recurrent and metastatic disease respond well to chemotherapy. Gonadal dysgenesis is defined as an incomplete or defective formation of the gonads, resulting from a disturbed process of migration of the germ cells and/or their correct organization in the fetal gonadal ridge. It is caused by structural or numerical anomalies of the sex chromosomes or mutations in one of the genes involved in the formation of the urogenital ridge and sex determination of the bipotential gonad . NeoplasHere, we report a case of mixed germ cell tumour diagnosed in a dysgenetic gonad in a 14-year-old girl with Swyer syndrome.A 14-year-old phenotypic female, the eldest of three children born out of a non-consanguineous marriage and an uneventful, full-term normal delivery, presented to a local hospital with a one-month history of diffuse abdominal pain, progressive abdominal distention, and vomiting. Her past surgical and medical history was unremarkable. She was premenarchal and had no history of developmental delay. There was no family history of any malignancies or developmental anomalies. On physical examination, she was 155 cm tall and weighed 50 kg. She did not have any secondary sexual characters, and her external genitalia appeared to be that of a normal female phenotype. Her abdominal examination revealed a large pelvic-abdominal mass occupying almost the whole of the abdomen with gross ascites. Systemic examination was within normal limits. Imaging showed a large lobulated heterogeneous mass in the pelvis extending to the abdomen with numerous cystic areas Figure -4. AscitShe was referred to us but reported only three months later with progressive abdominal symptoms and raised levels of serum (AFP), beta-HCG, and LDH. A CT scan of the abdomen showed a huge pelvic recurrence. She received four cycles of combination chemotherapy with bleomycin, etoposide, and cisplatinum. At the end of four cycles, serum markers normalised, but she still had a large residual pelvic mass Figure . She undThe first reported case of Swyer syndrome was in 1955 by Dr G I Swyer who described two women who had a 46, XY karyotype, tall stature, primary amenorrhea, female external genitalia, normal vagina , and cervix [During embryogenesis, without any external influences for or against, the human reproductive system is intrinsically conditioned to give rise to a female reproductive organization. As a result, if a gonad cannot express its sexual identity via its hormones , then the affected person, no matter whether their chromosomes are XY or XX, will develop external female genitalia. Internal female genitalia, primarily the uterus, may or may not be present depending on the etiology of the disorder.The incidence of neoplasia in patients with gonadal dysgenesis merits mention. Slowikowska et al.  reportedThe treatment of such a patient is primarily surgical, including resection of the primary lesion and proper surgical staging. Chemotherapy is administered to patients with recurrent and metastatic disease . The comSwyer syndrome or pure gonadal dysgenesis 46, XY is associated with a significant risk of developing germ cell tumors, and prophylactic gonadectomy is recommended. In any patient with a premenarchal pelvic mass, karyotyping should be performed. With prompt treatment, either surgery alone or surgery with chemotherapy, a good response and favorable survival outcomes can be obtained even in a metastatic or recurrent setting.


Neonatal scrotal hematoma is a rare genitourinary emergency. Some cases have underlining aetiologic factors such as testicular torsion, adrenal hemorrhage, or birth trauma, and others are idiopathic. Previously, immediate scrotal exploration was considered imperative for diagnosis and treatment. With good imaging techniques, some patients are managed nonoperatively. We report a case of idiopathic scrotal hematoma in a neonate. He was managed conservatively with clinical and radiological follow-up. There was complete resolution of hematoma within two months, thus, avoiding unnecessary exploration. Scrotal hematoma in the neonate, though a rare condition, warrants prompt diagnosis and urgent intervention. It commonly results from testicular torsion, adrenal hemorrhage, and birth trauma.However, in some cases no cause may be discernable. Since Putnam recorded his first experience in 1989 [7+3 seamstress in a maternity home. The birth weight was 3 kg. Antenatal care was inadequate. Painless left hemiscrotal swelling was noticed 72 hours after birth by the pediatricians managing him for neonatal jaundice and sepsis. There was no bleeding from any body orifice.A term male neonate with vertex presentation was delivered spontaneously per vaginuum to a 45-year-old PThere was no bruise over the groin. Abdominal examination was normal. External genitalia revealed scrotal asymmetry with a fluctuant, nontender, left hemiscrotal swelling. The ipsilateral testis could not be palpated separate from the swelling. The swelling did not transilluminate and we could get above it. There was no evidence of trauma. An initial suspicion of acute scrotum was made. An emergency scrotal ultrasonography showed both testes were normal in their positions, sizes, and outline. The left tunica revealed fluid collection with high level echoes . The tesThe abdominal ultrasonography was normal. His packed cell volume was 42.6%, and clotting profile and platelet count were within normal limits. Direct bilirubin dropped from 15.2 mg/dL to 12.8 mg/dL after 4 days of phototherapy. The sepsis was treated with antibiotics. He had parenteral vitamin K. The Direct Coombs test was negative. A diagnosis of idiopathic left hemiscrotal hematoma was entertained. The child was managed nonoperatively. He was monitored clinically and radiologically. At two- and ten-month clinic follow-up, the patient was well and weighed 6.9 kg and 9.3 kg, respectively. Follow-up ultrasounds scan revealed complete resolution of the earlier noted left scrotal hematoma. The testicular parenchyma echo pattern was also preserved bilaterally .Acute scrotal hematoma in the neonate is an urgent genitourinary condition. Though its list of differential diagnoses is legion, it is necessary to rule out torsion of spermatic cord which truly warrants emergency surgical exploration . Other cSpontaneous idiopathic hemorrhage of the scrotum, as found in our patient, has also been described –5. This Our patient presented with hemiscrotal swelling which was misdiagnosed as congenital hydrocele. The swelling, however, did not transilluminate as would be ordinarily expected in this age group. There was no clinical feature suggestive of trauma or bleeding diathesis. Scrotal and abdominal ultrasonography excluded the common aetiologies of neonatal hemoscrotum.In most parts of our region where ultrasonography is not easily accessible and affordable, it is better to explore the scrotum so as to save the gonad (testis) in case the cause is testicular torsion . HoweverAlso, radioisotope testicular scanning may be useful in assessing testicular perfusion with high specificity and sensitivity for testicular torsion , 11.Few cases of adrenal hemorrhage induced hemoscrotum have been reported in the literature , 8, 12. In neonates, the suprarenal glands are relatively big and are vulnerable to vascular injury , 14. TheExcept for spermatic cord torsion, hemoscrotum from other causes may be managed nonoperatively. We treated our patient as such. He was monitored clinically and radiologically with good outcome. This modality was documented by other workers . SurgicaNeonatal hemoscrotum is a condition rarely seen in surgical practice. When encountered it is important to speedily exclude testicular torsion which truly warrants emergency scrotal exploration. Urgent scrotal and abdominal ultrasonography is the diagnostic modality of choice. This may prevent unnecessary scrotal exploration.


We present a rare case of probable caseous calcification of the mitral. This pathology is more frequently detected in asymptomatic women older than 70 years. To recognize this image is important because echocardiography is the easiest way to elucidate this diagnosis, and more importantly because this structure could be easily misdiagnosed as tumors, thrombus and vegetations, which are much more common. Normally, it has a benign evolution, and the correct diagnosis is crucial to avoid unnecessary surgical interventions. It often occurs in elderly women (older than 70 years) and is considered an important differential diagnosis for heart tumors, thrombus or vegetationsHospital Israelita Albert Einstein in São Paulo (SP) to undergo a transesophageal echocardiogram in order to investigate the diagnosis of an intracardiac mass visualized through a transthoracic echocardiography in another service. Her clinical history included systemic arterial hypertension, dyslipidemia and obesity degree I (body mass index – BMI: 31.2kg/m2). She reported effort dyspnea, but refused to have other cardiovascular symptoms.An 83-year-old woman  was referred to echocardiography service of the Following the service routine the patient was submitted to transthoracic echocardiography before the transesophageal study. The transthoracic echocardiogram showed important enlargement of both atria and mild increase in the thickness of the heart muscle. Global systolic function of left ventricle was preserved , and no changes in segmental myocardial contractility were seen. The analysis of diastolic function showed diastolic standard of restrictive left ventricular filling . The aortic valve was thickened without restriction to its opening and had a mild reflux. Mitral valve was slightly thickened with normal opening and had mild to moderate regurgitation. Tricuspid regurgitation was moderated and enabled to estimate maximum systolic pressure of pulmonary artery at 106mmHg. Ascending aorta had a mild ecstasy in tubular portion measuring 3.8cm. Pericardium showed normal echocardiographic aspect.In the transthoracic study was possible to observe a mass of heterogeneous content with calcification points, regular edges and more echolucent central portion placed in the posterior mitral valve annulus . The mass did not determine any restriction to the opening of mitral valve cusps .The patient was sedated with midazolam and fentanyl, and received lidocaine 10% local anesthesia spray before the transesophageal echocardiography investigation that enabled to visualize better the mass dimensions and where it was located; in the posterior annulus (adjacent to P2 segment of posterior cusp), however it did not add information with regarding the nature and aspect of the mass. ,3.The patient underwent a nuclear magnetic resonance of the heart that revealed a rounded image with low magnetic signal at the posterior mitral valve annulus compatible with coarse calcification. Although no anatomopathologic study was performed, echocardiographic findings and the nuclear magnetic resonance suggested, based on evidence of the literature, a diagnosis of caseous calcification of the mitral annulus,2,4. It is often represented as an intracardiac mass easily misdiagnosed as tumors or thrombus, which leads to unnecessary surgical interventions. This affection is commonly found in the posterior annulus, medium portions and posterior basal cusp. The more affected population by the disease are women older than 70 years.Caseous calcification of the mitral annulus is rare and accounts for 0.5 to 1% of all calcifications of the mitral annulus. The largest serie in the literature has only 18 cases.The anatomopathologic aspect described in the literature is a periannular calcification composed by calcium, fatty acids and cholesterol. Microscopically its interior content has a typical aspect of “toothpaste”, and the prevalence in necropsies corresponds to 2.7% of calcifications of the mitral annulus.In general patients are asymptomatic. However, when symptoms are presented, dyspnea secondary to mitral insufficiency or to mitral stenosis is the most frequent clinical finding. Embolic phenomena, which are rare, might also occur. In addition, there is an association between this type of calcification with hypertension, coronary artery disease and aortic atheromatous disease. The use of transesophageal study enables to define better the local, consistence and aspect of the mass mainly in patients with limited acoustic window. It is important to identify the possibility of this disease and to differentiate it from tumors, thrombus or abscesses. In addition, the benign evolution of the disease, peripheral calcification, well-defined edges and its typical localization on the posterior annulus help to differentiate it from abscesses . In tumors the central echolucent seen in cases of caseous calcification is not observed-8.The transthoracic echocardiography study is often enough to diagnose the disease, and in most of cases the transesophageal becomes a complement to support the diagnosis. The featured echocardiography image is an echodense mass, rounded and sometimes semilunar with an echolucent area on its interior that is located in the posterior mitral valve annulus.To recognize and differentiate this disease is crucial to avoid unnecessary surgical interventions, especially because caseous calcification presents a benign evolution and good long-term prognosis. Surgical treatment should be restricted to cases of valves lesions (stenosis or insufficiency) with significant repercussionWe reported a case of a woman with a mass found in mitral valve annulus in which was observed the probable etiology of caseous calcification of the annulus.


Fibrous dysplasia is one of many well-known disorders in which there is a defect in the remodeling process of immature bone to mature into lamellar bone, and it often exists in metaphyseal and diaphyseal parts of the long bone. In this report, we describe a rare case where fibrous dysplasia was found only in the proximal part of the epiphysis of the tibia without other bony lesions.A 14-year-old Asian girl was referred to our hospital after slipping down with pain on the left knee. A radiograph showed an abnormal finding of a central radiolucent lesion with a marginal sclerotic border near the proximal tibial spine. A magnetic resonance image showed the lesion at low signal intensity on a T1-weighted image and at high signal intensity on a T2-weighted image. The biopsy results led us to conclude that the lesion was a fibrous dysplasia.If an abnormal lesion on the epiphysis, especially in long bones, is detected on a radiograph, several differential diagnoses can be made. Although fibrous dysplasia is usually not encountered as an epiphyseal lesion, it is important to incorporate all the clinical, radiographic and pathologic features to diagnose monostotic fibrous dysplasia when the lesion is located at the epiphyseal location. FD has a tendency of predominantly unilateral involvement and a prolonged clinical course characterized by pain, deformity and pathologic fracture of the affected bones[The term ed bones. McCune-ed bones. Mazabraed bones. In thisA 14-year-old Asian girl visited our hospital with radiographs taken 2 months earlier at a local clinic she attended for the evaluation of knee pain after slipping down. She had experienced no antecedent pain before slipping down, and the lesion was fortuitously found on radiographs. Anteroposterior and lateral radiographs were obtained. They showed a 1.5×2.0×2.0cm epiphyseal tumor in the proximal tibia. The matrix of the lesion had a clear, ground glass appearance, and the border showed well-defined marginal sclerosis. There was neither cortical disruption nor periosteal reaction. Computed tomography showed a sclerotic margin around the lesion, suggestive of a benign tumor  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.


We report a rare case of resected hepatic AML, which was misdiagnosed as hepatocellular carcinoma in a chronic hepatitis B carrier. A 45-year-old woman who was a carrier of hepatitis B virus infection presented with a hepatic tumor. Her serum alpha-fetoprotein level was normal. Ultrasonography revealed a round and well-circumscribed echogenic hepatic tumor measuring 2.5 cm in the segment VI. On contrast-enhanced computed tomography, a hypervascular tumor was observed in the arterial phase and washing-out of the contrast medium in the portal phase and delayed phase. On MR T1-weighted in-phase images, the mass showed low signal intensity, and on out-of-phase images, the mass showed signal drop and dark signal intensity. On MR T2-weighted images, the mass showed high signal intensity. The mass demonstrated high signal intensity on arterial phase after contrast injection, suggestive of hepatocellular carcinoma. The patient underwent hepatic wedge resection and histopathological diagnosis was a hepatic angiomyolipoma. Angiomyolipoma (AML) typically occurs in the kidney and rarely in liver . HepaticA 45-year-old woman, a chronic hepatitis B carrier, was admitted to our hospital for further evaluation and treatment of a liver mass that had been found on abdominal US at regular medical checkup in November 11, 2011. Clinically, no pathologic findings were observed during physical examination. All routine blood investigations, including liver function tests, were normal. The serologic studies for viral hepatitis B showed only positive hepatitis B surface antigen (HBs Ag) test result. The serologic markers for hepatitis C were nonreactive. The hepatitis B virus DNA titer was 438 IU/mL  and serum alpha-fetoprotein level was 2.01 ng/mL .Abdominal US showed a well-defined, hyperechoic mass, with maximal diameter of 2.5 cm in the segment VI of the liver . On contAs hepatocellular carcinoma was highly suspected from preoperative image studies with her medical history, hepatic wedge-resection with tumor in the segment VI was performed. Wedge resected liver showed a well-demarcated but nonencapsulated soft mass, representing variegated cut surface with yellowish fat tissue, multifocal hemorrhage, and no necrosis . The masAngiomyolipoma (AML) is an uncommon mesenchymal tumor that occurred more frequently in kidney than in liver . Most ofThe definitive diagnostic study remains the histological examination coupled with immunohistochemical stains. Among the components of hepatic AML, HMB-45 positive smooth muscle cell is the only specific and definitive criterions for diagnosis [In the present case, the preoperative radiological image was quite difficult to distinguish from fat-containing hepatocellular carcinoma. Although her serum alpha-fetoprotein was normal, the radiological findings and her clinical history are compatible with hepatocellular carcinoma. Under the above impression, these lesions were resected and the histological examination coupled with immunohistochemical stains made the final diagnosis of hepatic AML. Because the preoperative images showed atypical findings for AML, we did not make a accurate diagnosis of hepatic AML at that time.The management of hepatic AML sometimes remains controversial. Several authors have suggested that this disease can be managed with conservative treatment with followup after fine-needle aspiration biopsy in previous series , 14, 15.In conclusion, preoperative diagnosis of hepatic AML by image is sometimes quite difficult particularly in endemic areas of hepatocellular carcinoma, and in the patients who have risk factors of hepatocellular carcinoma with suggested malignancy by image but showing normal laboratory findings, repeated studies with different diagnostic modalities, such as biopsy or angiography, and careful interpretation are recommended.


Intramuscular myxoma is a rare benign mesenchymal tumor. Myxomas most commonly occur in the heart. They may occur less frequently in aponeurotic tissues, bone, genitourinary tract, subcutaneous tissue and skin.The case described here is a 44-year-old Turkish woman who presented with the complaint of a swelling in her right shoulder. A preoperative magnetic resonance imaging revealed a lobular contoured mass lesion in her deltoid muscle. The mass was marginally excised and pathology revealed intramuscular myxoma.Intramuscular myxoma of the deltoid muscle is a very rare benign tumor. In the differential diagnosis, reactive lesions, myxoid nodular fasciitis and low -grade myxoid sarcomas should be kept in mind, upon which the treatment should be planned. Myxomas have been described as a true neoplasm of low-vascularity composed of undifferentiated satellite cells embedded in myxoid stroma containing collagen and reticular fibers. Myxoma Intramuscular myxoma usually occurs as an isolated lesion. The first case of intramuscular myxoma was described in 1965. IntramuHere we report a case of a 44-year-old woman with myxoma located in her right deltoid muscle and aim to discuss intramuscular myxomas in the light of clinical, radiological and pathological data.A 44-year-old Turkish woman presented to our clinic with the complaint of a 3-year history of a slow growing palpable mass in her right shoulder. She had no other documented or reported condition.Her physical examination revealed a palpable, mobile, painless, partially fluctuating mass, 5×5cm in size, in her deltoid muscle, extending to the lower lateral part of her right shoulder and proximal lateral part of her right arm.Magnetic resonance imaging (MRI) revealed a lobular contoured mass lesion, cystic in nature and measuring 34×23×37mm at its largest that was located caudal to the lateral compartment of her deltoid muscle and an interstitial edema in the adjacent deltoid muscle Figure . A complDuring her surgical operation, the mass was approached via a longitudinal incision over the mass in the deltoid area of her right shoulder, sensory neural branches of the axillary nerve were preserved, and the encapsulated gelatinous mass 4×5cm in size was easily separated from the muscle tissue and then excised. A macroscopic pathological examination of the mass revealed a nodular lesion cream-beige in color, 2.5×2.5×3cm in size and capsular in appearance that was adjacent to the deltoid muscle. The sectioned surface of the mass was pink-beige in color and solid bright myxoid in appearance. The surgical margin was tumor-free.A microscopic examination showed that the mass consisted of a small number of spindle or stellate cells, a small number of vessels, thin collagen fibers and a small number of loose reticular fibers embedded in an abundant myxoid stroma Figure . The celAn immunohistochemical examination of the mass showed diffuse positivity for vimentin in tumor cells Figure , focal aMyxoma is a benign soft tissue tumor of unknown origin. It may Soft tissue myxomas are rare. ExtracaIntramuscular myxomas can occur in the muscles of the thigh, buttocks, shoulder and upper extremities. IntramuOn clinical examination, intramuscular myxoma presents as a painless, palpable mass. The clinical pattern varies depending on the size and site of the mass, 23.A radiograph reveals a nonspecific mass with no calcification. On ultrasonography, it appears as cystic lesions with echogenicity in normal muscle tissue. On compThe differential diagnosis of intramuscular myxoma includes other myxoid neoplasms and proliferative lesions of the soft tissue. Benign Initial diagnosis can be checked by needle biopsy, 24 wherThere are transition zones between hypocellular myxoid areas and fat tissues in myxolipoma. Myxoid liposarcoma includes atypical lipoblasts and characteristic diffuse plexiform capillary network. Myxomas do not contain lipoblasts and have poor vascularity. Myxolipomas and liposarcomas stain positive for S-100.Neurofibroma cells display a greater degree of orientation whereas the vascularity is more prominent and they contain abundant collagen fibers. Some areas show positive staining for S-100. Neurothecomas contain prominent lobules separated by fibrous bands. Lobules consist of myxoid and cellular areas and stain positive for S-100.Myxochondromas contain lobular patterns with more cellular chondroid cell groups among myxoid areas, which usually stain positive for S-100. Myxoid chondrosarcomas present more pleomorphic, cellular and infiltrative growth.Nodular fasciitis stains positive for smooth muscle. In myxomas, there is zonal organization or regional heterogeneity of nodular fasciitis. Myxomas are more hypocellular and have poor vascularity.Low grade myxofibrosarcomas differ from myxomas in being more pleomorphic and ill defined, and in having a characteristic nodular pattern and organized vascular pattern. Low grade fibromyxoid sarcomas are characterized by contrasting myxoid and cellular fibrous areas with a swirling, whorled pattern.Treatment of solitary myxomas is marginal surgical excision, 9, 32. In our case, no needle biopsy was performed and the entire mass was removed with wide margins along with the surrounding muscle tissue and the surgical specimen was sent for pathological examination. Histopathological examination revealed intramuscular myxoma. The patient’s treatment was completed with extensive surgical excision in one session. Currently, she is at the postoperative 6-month follow-up with no complaints or recurrence.In conclusion, intramuscular myxoma of the deltoid muscle is a very rare benign tumor. Even though imaging techniques are helpful, the exact diagnosis is established by histological examination. In the differential diagnosis, reactive lesions, myxoid nodular fasciitis and low grade myxoid sarcomas should be kept in mind, upon which the treatment should be planned.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.


A 2-year-old male, intact Yorkshire terrier presented with a 1-month history of progressive paraparesis. Neurological examination revealed paraplegia with absent deep pain perception, decreased right pelvic limb withdrawal reflex, and lumbar pain consistent with an L4–S2 neurolocalization. Magnetic resonance imaging (MRI) showed a single, well-demarcated, intramedullary mass centered over the L3–4 disk space. A hemilaminectomy was performed, and the mass was removed en bloc. Histopathological evaluation was consistent with a hemangioblastoma. Follow-up MRI 9 months after surgery showed no evidence of tumor recurrence, and the dog was ambulatory paraparetic at that time. This case is consistent with a previous histopathological report of spinal cord hemangioblastoma in a dog and provides additional clinical information regarding diagnosis, treatment, and outcome associated with this tumor type. A 2-year-old male, intact Yorkshire terrier [4.77 kg (10.56 lb)] was referred with a 1-month history of paraparesis starting in the right pelvic limb. Clinical signs progressed despite treatment with prednisone . On neurological examination, the patient was paraplegic with absent deep pain perception in the pelvic limbs. The pelvic limbs had increased extensor tone, normal patellar reflexes, normal withdrawal reflex in the left, and decreased withdrawal reflex in the right. Pain was elicited on palpation of the lumbar spine. Findings were consistent with a lesion of the L4–S2 spinal cord segments.Complete blood count and serum biochemistry panel and electrolyte analysis were unremarkable. Magnetic resonance imaging  of the thoracolumbar spine revealed a slightly right-sided, discrete, intramedullary mass at the level of the L3–4 intervertebral disk space; the mass occupied over 90% of the vertebral canal  was used to perform the initial dissection through the pia as well as the underlying, thin, vascular tumor capsule. Once the pia and capsule were removed, a plane of dissection was easily distinguished, and the mass was removed en bloc. On further gross examination, the mass was firm and appeared noticeably more vascular along the surface than on the inside.The dog was hospitalized and received standard post-operative care, including intravenous fluids and analgesia. At the time of dismissal from the hospital approximately 48 h after surgery, the dog was non-ambulatory paraparetic and was voiding voluntarily.On histopathological evaluation, the spinal mass was composed of mildly pleomorphic spindle cells arranged in loose streams and bundles separated by variable amounts of fibrillar stroma Figure . The spiThe dog was re-evaluated at our institution 9 months after surgery. Neurological examination revealed ambulatory paraparesis and proprioceptive ataxia in the pelvic limbs with the right limb being more severely affected. Postural reactions in the pelvic limbs were normal on the left and delayed to absent on the right. Spinal pain was not appreciated. Follow-up MRI performed 9 months after surgery showed no evidence of tumor recurrence; however, spinal cord atrophy was present at the level of the excised hemangioblastoma. Hypointense material on pre-contrast T1-weighted and post-contrast T1W with Fat Sat images was visible to the right of the atrophied spinal cord and corresponded to an area of mixed intensity on the T2W images. This material may represent CSF and adhesions within the vertebral canal secondary to atrophy of the spinal cord and surgery Figure . The appBest veterinary care was practiced in the clinical and diagnostic evaluation as well as treatment of this patient, and the owner of the patient provided informed consent for all procedures prior to them being performed. As no experimental protocols were utilized, an institutional review was neither required nor performed.Hemangioblastomas are rare, benign, highly vascular central nervous system tumors. They composed <3% of all central nervous system tumors in people and occur most commonly in the caudal fossa, although 7.5–25% of people develop spinal cord lesions , 2. MostTo the best of our knowledge, only two cases of spinal hemangioblastoma (SHB) have been reported in dogs , 7. FurtThe clinical, MRI, surgical, and histopathological features of this tumor closely resemble descriptions of human hemangioblastomas, as well as those of previously reported canine cases. Approximately a quarter to a third of central nervous system hemangioblastomas in people occur as a result of VHL . In peopIn human patients, SHBs are significantly more likely to occur in the cervical or thoracic spinal cord (>90%) , 2, 4. TMany people with VHL-associated SHB do not have any clinical signs referable to the tumor. However, a majority of sporadically occurring SHBs are diagnosed after becoming symptomatic. In people, clinical signs caused by SHB include pain, hypoesthesia, dysesthesia, paresthesia, hyperreflexia, weakness, scoliosis, proprioceptive deficits, spasticity, and bowel and bladder abnormalities , 5.Characteristic MRI features of hemangioblastoma in people include a well circumscribed, nodular to ovoid-shaped mass that is homogenously and intensely contrast enhancing. These tumors are T1W isointense or hypointense and T2W hyperintense to normal spinal cord parenchyma . The preUse of radiation therapy, chemotherapy, and stereotactic radiosurgery has been reported for the treatment of SHB in people, although stereotactic radiosurgery is used primarily for treatment of intracranial hemangioblastoma. Despite these alternative treatment options, microsurgical resection is considered the treatment of choice for SHB in people, and a surgical plane of dissection typically facilitates total resection . MyelotoHistopathological and immunohistochemical findings in this case are consistent with previous veterinary reports of hemangioblastoma , 7, 12. In a review of all reported cases of sporadic SHB in people undergoing surgery, neurological improvement was described in 65% of patients whereas 24% remained unchanged (In conclusion, hemangioblastoma should be considered as a rare differential diagnosis for dogs with intramedullary spinal cord masses. Evidence from human literature as well as experience with this case suggests that en bloc surgical resection is not only possible but may be the treatment of choice in dogs. This case also demonstrates the possibility of clinical improvement and long-term control of the disease with surgical resection alone.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


Cavernous hemangiomas are rare benign bone tumors and those at the level of the cranial bones are even rarer.A 50-year-old woman of Italian ethnicity presented with a frontal mass. A computed tomography scan showed an osteolytic lesion and a magnetic resonance imaging scan revealed a hypointense lesion on the T1-weighted image and a hyperintense lesion on the T2-weighted image. We performed a tailored craniectomy and cranioplasty. Histological examination revealed a cavernous hemangioma.These benign tumors do not have classic radiographic features and so can be misinterpreted as lesions like multiple myeloma or osteosarcoma. Consequently, the diagnosis is most often made during surgical resection. Cavernous hemangiomas are uncommon tumors, accounting for 0.7 to 1 percent of all bone neoplasms .We present the case of a 50-year-old woman of Italian ethnicity who presented with a slow-growing frontal mass, tender to pressure, with spontaneous pain. Her neurological examination was completely normal. There was no history of trauma or other systemic disease. A magnetic resonance imaging (MRI) scan showed a hypointense lesion on the T1-weighted image and a hyperintense lesion on the T2-weighted image. A computed tomography (CT) scan showed an osteolytic lesion with erosion of the tabula externa (Figure en bloc resection. Plain radiography or CT scan reveals this lesion as solitary lytic lesions with a sclerotic rim while MRI shows isointense on T1-weighted images and hyperintense on T2-weighted images, consistent with regions of slow-flowing blood. Sometimes, the classic radiographic appearances are not evident. Consequently, the diagnosis is most often made during surgical resection. These tumors can be misinterpreted as lesions like multiple myeloma or osteosarcoma [en bloc resection of the tumor with the removal of a rim of normal bone, described in 1923 by Cushing [The most commonly involved site of intraosseus hemangiomas is the vertebrae, and hemangiomas of the calvarial bones account for 0.2 percent of all bone neoplasms ,3. Womenosarcoma . The firosarcoma  in 1845  Cushing . Instead Cushing . Cases oCalvarian hemangiomas do not always have typical radiologic features and should always be considered in the differential diagnosis of malignant skull lesions. Histopathological confirmation after surgical resection of the tumor is the definitive method for diagnosis.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.CT: computed tomography; MRI: magnetic resonance imaging.The authors declare they have no competing interests.All authors analyzed and interpreted the patient data and contributed to writing the manuscript. All authors read and approved the final manuscript.


Extralobar pulmonary sequestrations may be located in intrathoracic or extrathoracic areas. Extrathoracic intradiaphragmatic extralobar pulmonary sequestrations are an extremely rare subset of bronchopulmonary sequestrations and there have been very few reported cases until now. We describe a 48-year-old Korean woman found to have left peridiaphragmatic lesion on computed tomography. We performed thoracoscopic surgery and successfully resected the tumor. Based on the histological findings, it was diagnosed as an intradiaphragmatic extralobar pulmonary sequestration. Postoperative course was uneventful. Intradiaphragmatic extralobar pulmonary sequestration in adult is extremely rare, so we report the case with a literature review. Extralobar pulmonary sequestrations are most commonly found within the thoracic cavity, but have been described in extrathoracic areas as so called extrathoracic extralobar pulmonary sequestrations. Sequestrations within the diaphragm  are extremely rare. In this case, the tumor was resected using thoracoscopic surgery, and based on the histological findings, it was diagnosed as an intradiaphragmatic extralobar pulmonary sequestration. We herein report a case of an extrathoracic intradiaphragmatic extralobar pulmonary sequestration in a 48-year-old Korean woman.A 48-year-old Korean female patient presented with an abnormal mass lesion that was detected by abdominal computed tomography in a visit to our hospital. She had experienced intermittent abdominal pain for several months. She had no other specific past medical history and no history of trauma. The patient’s vital signs were stable and laboratory tests were normal. Chest X-ray showed no abnormal findings and the computed tomography of her chest showed a 4-cm-sized round mass with areas of calcification in the left hemidiaphragmatic area Figure . After aPulmonary sequestration was first defined by Pryce in 1949,3. ExtraA case of intradiaphragmatic extralobar pulmonary sequestration is rare. The patients previously reported with such lesions were neonates or younger than two year old. To our knowledge, this is the only case of intradiaphragmatic extralobar pulmonary sequestration in an adult. We report the intradiaphragmatic extralobar pulmonary sequestration and its successful removal by thoracoscopic surgery along with literature reviews.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.We gently appreciate the staffs of Radiology, YeungNam University Medical Center.JH and MJ wrote the draft of the manuscript and obtained the written consent. JH performed the literature review and participated in the manuscript writing and helped to the final writing of the paper and gave final approval of the manuscript. All authors have read and approved the final manuscript.


A case of an unusual unilocular cystic lesion of diameter 7 cm located retroperitoneally in the pelvis in close connection to the right adnexa of a 61 year-old woman is presented. Macroscopically, the lesion had a smooth outer and inner surface and was filled with translucent fluid. Histological examination revealed a fibrous and hyalinized wall which lacked a specific lining. Numerous nerve bundles in the cyst wall constituted the most conspicuous element of its histology possibly with some contribution of perineurial and/or mesothelial components. The morphology and immunohistochemistry speak for an intraneural pseudocyst sometimes called intraneural ganglion cyst which is rare in this location.http://www.diagnosticpathology.diagnomx.eu/vs/1357862917132314The virtual slide(s) for this article can be found here:  Intraneural ganglion cysts or alternatively ganglion cysts involving peripheral nerves - GCPNs -  are rare disorders and present as unilocular cysts filled with translucent mucin . ClinicaIn the patient  with the history of ill-defined discomfort over the right lower abdomen and pelvis, the cystic lesion in the region of the right adnexa has been found in bimanual examination and in ultrasonography. She had no history of either previous operations, cesarean section or any other ailments or diseases. She had regular moderate menses  without complaints and bore 2 children by spontaneous delivery. The transvaginal ultrasonography at the admission to the Clinic of Gynecology and Oncology of the University Hospital in Krakow (Poland) did not reveal any other abnormalities or pathology in the pelvic region (uterus anteroflexed and normoechogenic) apart from the unilocular and oval, lemon-like shaped cyst of diameter 7 cm. The lesion presented with mixed echogenicity and without any abnormal vascularization in Power Doppler (PD) scan  cystic lesion like in our case.During the process of arriving at the diagnosis, several options arose and were evaluated  as reported by Fiori et al. . NoteworOne should also not forget the possibility of a form of endometriotic cysts which not infrequently are devoid of any remnants of endometrium. However, typically they show evidences of previous hemorrhages (deposits of hemosiderin and/or siderophages). Such cysts deserve the term “hemorrhagic cysts of undetermined origin” . In the Last but not least, one may speculate upon an intraneural, un-specific inflammatory process of pathogenesis. Although we have not found any evidences of an active inflammatory reaction (like lymphocytes etc.) one cannot exclude that some parts of S-100 immunopositivity may be due to the presence of dendritic cells, which are involved in autoimmune inflammatory processes within the nerve (autoimmune neuritis for instance) . We may Lastly, one cannot help remarking that though it is very hard to find a more benign and innocuous lesion in human pathology in whichever site of the body as the cyst described here, even such a “simple” lesion demands cautious differential diagnosis. Moreover, a clinician, especially a gynecologist, being aware of the existence of lesions like reported here may be much more convincing trying to dispel any worries of a patient if confronted with a similar case.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.GCPN: Ganglion cyst involving peripheral nerves; EMA: Epithelial membrane antigen; NET: Gastro-entero-pancreatic neuroendocrine tumor.The authors declare that they have no competing interests.DA made the diagnosis and edited the manuscript. CJ prepared a draft of the manuscript and performed critical review of literature. MH contributed to the histopathological examination and methods. RJ supervised the treatment of the patient and contributed to the description of the case. BG performed the physical examination and transvaginal ultrasonography and contributed its description to the manuscript. All authors read and approved the final manuscript.D.A.  is a specialist in pathomorphology and neuropathology and now at the position of Chair of Pathomorphology and Head of the Department of Neuropathology, Medical College, Jagiellonian University, Krakow (Poland). He is also a lecturer of pathology in the Faculty of Medicine of the Jagiellonian University.C.J. (B.Sc.) is a student of biomedicine (master) at the Julius-Maximilians-University of Wuerzburg (Germany) doing an internship at the Chair of Pathomorphology, Jagiellonian University.M.H. (MD) is a postgraduate/intern in pathomorphology at the Chair of Pathomorphology, Jagiellonian University.R.J.  is a specialist in Gynecology and Obstetrics and Gynecology and Oncology, Jagiellonian University. He is also a lecturer in Gynecology at the Medical Faculty of the Jagiellonian University.B.G. (MD) is a postgraduate/intern at Gynecology and a junior assistant.


Extramammary Paget’s disease is a rare histological type of intraepithelial adenocarcinoma that mainly affects apocrine sweat gland-rich areas. Predilection sites include the anogenital region and, less commonly, the axillae. These tumors rarely occur in non-apocrine regions. The aim of this case report is to describe a case of extramammary Paget’s disease in the external auditory canal with extensive temporal bone involvement and skull base invasion.A 40-year-old Caucasian man presented with a progressively growing, vegetating lesion localized in his right-hand external auditory canal. He had peripheral facial nerve paralysis and complete ipsilateral hearing loss. An incisional biopsy suggested extramammary Paget’s disease, and the immunohistochemical analysis confirmed the diagnosis. The tumor was considered inoperable because of the extensive skull base involvement, and he was referred for palliative radiotherapy.We report here the third case of extramammary Paget’s disease affecting the external auditory canal to be described in the literature. In all the three cases, the prognosis was unfavorable despite treatment. Extramammary Paget’s disease (EMPD) is a rare histological type of intraepithelial adenocarcinoma that mainly affects apocrine sweat gland-rich areas. The predilection sites include the anogenital region and, less commonly, the axillae. These tThe patient was a 40-year-old Caucasian man who presented with a 3-month history of a progressively growing, vegetating lesion localized in his right-hand external auditory canal. Upon physical examination, a 2.5cm bleeding, ulcerated lesion was observed to occlude his right external auditory canal Figure . The patThe incisional biopsy suggested EMPD Figure a, b, butThe tumor was considered inoperable because of the extensive skull base involvement, and the patient was referred for palliative radiotherapy. The patient underwent 36 sessions of radiotherapy and was given a total radiation dose of 72Gy. A complete response was achieved at the primary site, but during treatment, diffuse bilateral pulmonary metastases were detected. The patient experienced significant weight loss during treatment and died 4 months after completing radiotherapy.EMPD was first reported by Crocker in 1889 and is aIn its usual presentation in the genital and perianal regions, EMPD is reported as a slow-growing, erythematous, and well-demarcated lesion that forms crusts, scaly edges, and occasional ulcerations. These tumors grow until achieving local invasion and metastasis. Preciseet al.[Cases of EMPD that affect the external auditory canal are extremely rare. Such a case was first published by Kaneko and Fligiel in 1975 et al. in 1998.et al.. Besideset al..et al.[Immunohistochemistry is critical for diagnosis because the histological aspect can resemble amelanotic melanoma or pagetoid Bowen’s disease. Markerset al. publisheCombined surgical resection and adjuvant radiotherapy provides the most effective treatment; howeverThe prognosis of EMPD in its usual presentation is generally favorable and depends on the extent of the lesion, depth of invasion, and lymph node spread. The assIn summary, EMPD very rarely affects the external auditory canal, and only two cases have been previously described in the literature. In this case and the two previous cases, the prognosis was unfavorable despite treatment. If the patient had sought medical attention earlier, the disease could possibly have been diagnosed at an initial stage and treated with radical surgery combined with adjuvant radiotherapy and could possibly have been healed.Written informed consent was obtained from the patient’s next of kin for publication of this case report and the 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.FACBJ cared for the patient and analyzed and interpreted the patient data. WOM wrote the manuscript and was solely responsible for the literature search. IMV performed radiotherapy planning and reviewed the radiotherapy details. AMSC performed the histopathological examination and immunohistochemical study. JCF and FMCJ were involved in the revision of the manuscript. All the authors read and approved the final manuscript.


Eccrine spiradenoma (ES) is a rare, benign adnexal neoplasm that may easily be mistaken for glomus lesions or angioleiomyoma due to its painfulness and florid vascularization. A 44-year-old male with a blue-colored, nodular tumor on the left knee, present for 10 years, was submitted for diagnosis. Dermatological examination was undertaken, followed by surgical excision of the subcutaneous lesion and histopathological examination of the tissue. Subjective symptoms included tenderness upon palpation and routine investigations were within normal limits. Immunohistochemical analysis of the tumor cells demonstrated positive staining for CK5/CK6, CK8/CK18, S100, as well as small vacuole-like positive for EMA, and was therefore diagnosed as ES. The results of the present study suggest that immunohistochemical assays may be helpful to clarify the diagnosis and differentiate ES from other painful subcutaneous tumors exhibiting similar clinical and histological presentations. Eccrine spiradenoma (ES) is a rare, benign adnexal neoplasm that has been historically designated as a tumor of eccrine differentiation. ES is able to be present on any part of the body , with ~1A 44-year-old male presented with a blue intradermal nodule ~1 cm in size localized in the left knee. The tumor was initially observed 10 years previously without any associated pain or pruritus and gradually enlarged thereafter. Dermatological examination revealed a firm, tender and blue nodule with a smooth surface and obscure boundaries . StromalAn excisional biopsy was performed. Histological examination revealed multiple strongly basophilic lobules arranged in sheets in the dermal and subcutaneous tissue. The overlying epidermis was almost intact without connections to the tumor island . The nodES may be easily mistaken for other lesions that characteristically present with localized pain and/or a marked degree of vascularization. These include: i) aggregated lymphatic nodules; in the primary clinical differential diagnosis, the immunohistochemical results are usually clear (tubular differentiation was demonstrated in the present case); ii) glomus tumor, a benign neoplasm characteristically associated with conspicuous vasculature components (poor vasculature was observed in the current case); and iii) angioleiomyoma, a benign tumor arising from the vascular smooth muscle typically expressing SMA (which was negative in the present case).Eccrine spiradenoma (ES), as first described in 1956, is a rare, benign adnexal neoplasm that is able to present on any part of the body, with ~1/5 of cases occurring in the extremities . The preHistologically, ES may present in a variety of ways, including as tumors arranged in sheets, cords or islands, often precluding a straightforward diagnosis. Tumor cells are strongly basophilic, resembling lymph nodes when observed under a low power microscope. In certain cases, lymphocyte infiltration and abundant telangiectasia are observed in the tumor region, with irregular clearance presented around the lumen. A differential diagnosis for glomus tumors should be performed when vascular hyperplasia is statistically significant. Occasionally, a nerve trunk may be observed in the vicinity of the lobules, as identified in the present case.The diagnosis of ES may be elusive given its multiple presentations without a change in the skin surface. Correct diagnosis is critical due to the potential for malignancy. The primarily clinical feature of ES is the presence of pain in the patient  and painA clinical differential diagnosis of glomus tumor/aggregated lymphatic nodule was offered in the current case. The diagnosis could not be confirmed from the clinical and histological investigations and an immunohistochemical assay was performed. A diagnosis of ES was suggested on the basis of ductal differentiation and poor vasculature identified following immunohistochemical staining of the excised tumor mass.ES has been historically designated as a tumor of eccrine lineage, although the current view is that it may arise due to an apocrine process ,10. In tTreatments for ES have not been well established; however, surgical excision is currently the gold standard option, with low rates of recurrence documented . Other t


The cystic lymphangioma of the liver is an extremely rare benign tumor that usually belongs to a systemic lymphangiomatosis. An 8-month-old boy came to our Department due to abdominal distension. On physical examination, a palpable abdominal mass was noticed on the right upper quadrant, with voluntary guarding and no peritoneal irritation. Laboratory tests did not produce any positive findings. Abdominal ultrasound revealed a hepatic cyst measuring 14.9 cm × 10.7 cm × 9.7 cm in the right hepatic lobe. An enhanced abdominal CT scan confirmed a cystic hepatic lesion with no capsule. There were no definite septations or solid elements within the cyst. At laparotomy, a giant unilocular cyst arising from the visceral surface of the right lobe of liver extending into whole abdominal cavity and pelvis was observed. Complete excision of the cyst was performed. The pathology report defined the tumor as a cystic hepatic lymphangioma.


Gossypiboma is a term used to describe a mass that forms around a cotton sponge or abdominal compress accidentally left in a patient during surgery. Transmural migration of an intra-abdominal gossypiboma has been reported to occur in the digestive tract, bladder, vagina and diaphragm. Open surgery is the most common approach in the treatment of gossypiboma. However, gossypibomas can be extracted by endoscopy while migrating into the digestive tract. We report a case of intractable duodenal ulcer caused by transmural migration of gossypiboma successfully treated by duodenorrhaphy. A systemic literature review is provided and a scheme of the therapeutic approach is proposed.A 61-year-old Han Chinese man presented with intermittent epigastric pain for the last 10 months. He had undergone laparoscopic cholecystectomy conversion to open cholecystectomy for acute gangrenous cholecystitis 10 months ago at another hospital. Transmural migration of gossypiboma into the duodenum was found. Endoscopic intervention failed to remove the entire gauze, and duodenal ulcer caused by the gauze persisted. Surgical intervention was performed and the gauze was removed successfully. The penetrated ulcer was repaired with duodenorrhaphy. The postoperative period was uneventful.We systematically reviewed the literature on transmural migration of gossypiboma into duodenum and present an overview of published cases. Our PubMed search yielded seven reports of transmural migration of retained surgical sponge into the duodenum. Surgical interventions were necessary in two patients.Transmural migration of gossypiboma into the duodenum is a rare surgical complication. The treatment strategies include endoscopic extraction and surgical intervention. Prompt surgical intervention should be considered for emergent conditions such as active bleeding, gastrointestinal obstruction, or intra-abdominal sepsis. For non-emergent conditions, surgical intervention could be considered for intractable cases in which endoscopic extraction failed. Gossypiboma is a term used to describe a mass that forms around a cotton sponge or abdominal compress accidentally left in a patient during surgery. Transmural migration of an intra-abdominal gossypiboma has been reported to occur in the stomach, duodenum, ileum, colon, bladder, vagina and diaphragm -3. Open A 61-year-old Han Chinese man presented with intermittent epigastric pain for the last 10 months. The pain was mild and non-radiating, without specific relieving or aggravating factors. The patient had no history of nausea, vomiting, general weakness, poor appetite or body weight loss. He had undergone laparoscopic cholecystectomy conversion to open cholecystectomy for acute gangrenous cholecystitis 10 months ago at another hospital. Gauze retention in the peritoneal cavity with migration into the duodenum was noted after upper gastrointestinal (UGI) endoscopy Figure . AbdominWe searched the PubMed (2000–2013) database for case reports about transmural migration of gossypiboma into the duodenum. The abstracts of all articles published in Dutch, English, French, German, and Spanish were screened. The full texts of articles published in other languages but with an abstract in English were analyzed. Articles were selected for review if they included the following patient data: age, sex, initial surgery, interval, clinical presentation, diagnostic methods, location, and surgical procedures.Our PubMed search yielded seven reports of transmural migration of retained surgical sponge into the duodenum. Relevant data are shown in Table The term “gossypiboma” denotes a cotton sponge that is retained inside a patient during surgery. The reported incidence of gossypiboma varies between 1/100 and 1/3000 for all surgical interventions and from 1/1000 to 1/1500 for intra-abdominal operations -18. TherAs a consequence of gossypiboma, two types of foreign body reactions can occur. The first type is an aseptic fibrous response to the foreign material that creates adhesions and encapsulation. The result is a foreign body granuloma which may take a silent clinical course which dose not produce any clinical symptoms. A gossypiboma may undergo calcification, disruption, partial absorption, and even diffusion. The second type of foreign body reaction is exudative in nature and produces an inflammatory reaction with abscess formation. The body attempts to extrude the foreign material, which may lead to post-surgical complications such as external fistula formation or erosion and perforation into adjacent viscera. This may then result in migration of the foreign body into the gut, intestinal obstruction, or extrusion of the sponge through the rectum. The exudative type of response often causes symptoms in the early postoperative period, but the extrusion process may take years and the clinical symptoms are unspecific -9,19-25.According to the literature, transmural migration of gossypiboma into the duodenum is rare. To date, only 7 cases have been published ,4-8,26. Many risk factors, such as duration and complexity of surgery, excessive blood loss in trauma patients, surgery under emergency conditions, unplanned procedural changes, a change in operating room teams during the course of the operation, and a failure to count surgical instruments and sponges, were identified. The three most important risk factors are emergency surgery, unplanned change in the operation, and body mass index ,15,18,33Nonspecific clinical symptoms may preclude an accurate diagnosis. The clinical presentation of gossypiboma is variable. According to the literature, common symptoms and signs of transmural migration of gossypiboma into the duodenum may include abdominal pain, vomiting, and bleeding ,9,30. ThThe diagnosis of gossypiboma is difficult because the clinical symptoms are nonspecific and the imaging findings are often inconclusive. In imaging studies, they are mostly seen as radio-opaque material, yet radiolucent material like sponges can cause diagnostic problems. However, plain radiography, barium studies, endoscopy, ultrasonography (US), CT, and magnetic resonance imaging (MRI) are useful for diagnosis . Plain rGossypibomas should be removed as soon as diagnosed. Surgery is the preferred method of treatment for gossypiboma. Various techniques, including percutaneous techniques, such as laparoscopy and laparotomy, are used for the removal of gossypiboma, depending on the clinical presentation and medical equipment available ,26,30,46In conclusion, gossypiboma should be considered in the differential diagnosis of any postoperative patient who presents with pain, infection, or a palpable mass. Plain radiography, barium studies, endoscopy, ultrasonography, CT scan, and MRI are useful for diagnosis. Transmural migration of gossypiboma into the duodenum is a rare surgical complication. The treatment strategies include endoscopic removal and surgical intervention. Prompt surgical intervention should be considered for emergent conditions such as active bleeding, GI obstruction, or intra-abdominal sepsis. For non-emergent conditions, surgical intervention could be considered in intractable cases if endoscopic extraction failed.Written informed consent was obtained from the patient for publication of this Case report and any accompanying images. A copy of the written consent form is available for review by the Editor of this journal.UGI: Upper Gastrointestinal; CT: Computed Tomography; US: Ultrasound; MRI: Magnetic resonance imaging; ERCP: Endoscopic retrograde cholangiopancreatography.The authors declare that they have no competing interests.YXL searched the literatures and drafted the manuscript. CCY conceived of the study, participated in its design and coordination, and final revision of the manuscript. CFT participated in the collection of the clinical data and design of the study. CCW participated in the design of the study and critical revision of the manuscript. All authors read and approved the final manuscript.The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1471-2482/14/36/prepub


Head and neck paraganglioma is a rare and predominantly asymptomatic tumor. In the present study, an extremely rare case of asymptomatic paraganglioma located between the left common carotid artery and the left thyroid is described. The clinical presentation, cytomorphology and the immunohistochemical characteristics for the diagnosis of head and neck paraganglioma are described. To the best of our knowledge, only two cases of paraganglioma located between the left common carotid artery and the left thyroid have previously been reported. Paraganglioma is a tumor that develops from the paraganglionic system, which may be classified as a symptomatic or asymptomatic type based on the presence or absence of endogenous hormone secretion, respectively. Head and neck paraganglioma (HNP) is a rare and typically asymptomatic tumor. Germline variations in succinate dehydrogenase (SDH) genes, such as the SDHx mutation, may be significant in sporadic HNP and familial paraganglioma. Positron emission tomography (PET)/computed tomography (CT) have been highlighted in the diagnosis of HNPs; however, immunohistochemical analysis remains the primary diagnostic method. Furthermore, complete resection of the tumor is directly associated with the positive prognosis. In the present study, we report the case of a 50-year-old female presenting with asymptomatic HNP located between the left common carotid artery and the left thyroid. To the best of our knowledge, only two previous cases of HNP located at this site have been reported ,3. PatieA 50-year-old female was admitted to the Department of Endocrinology and Metabolism, Shaoxing People’s Hospital  due to weight loss over a period of nine months. The patient had no previous history of hypertension or diabetes and no family history of paraganglioma. On admission, the patient did not exhibit proptosis. A physical examination revealed palpable thyroid nodules  with a medium texture, clear boundaries and the nodules were moving freely during swallowing. Furthermore, no enlarged neck lymph nodes were palpable. Auxiliary examinations, including a chest X-ray, electrocardiogram, gastroscopy, blood glucose testing, cancer screening panels, thyroid function panels and parathyroid hormone measurements, did not reveal any abnormalities. However, an ultrasound scan identified multiple bilateral thyroid nodules and a hypoechoic mass in the left side of the neck, which was considered to be a neurogenic tumor . Plain aOriginating from the parasympathetic ganglia, paragangliomas consist of either chromaffin or non-chromaffin cells, which are defined by the response of the main cells to chromium salts . The tumet al  are significant in the preoperative diagnosis of paragangliomas  and PET/et al  reportedl (et al  confirmel (et al  has beenThe pathological characteristics of paragangliomas include ovoid, marginally lobulated, elastic masses with a smooth surface, incomplete capsules and local infiltration in the majority of cases. The surgical surface appears to be a gray/brown/red color, with a rich supply of blood vessels. Under a light microscope, the tumor consists of epithelial main cells arranged in a nest-like structure, which are separated by abundant enlarged fibrovascular stroma. Although supporting cells are visible around the nest, nerve fibers are difficult to observe . ImmunohSurgery remains the common treatment method for paragangliomas and complete resection of the lesions is the most critical component for remission. Our patient had an intact tumor capsule without significant adhesions to the surrounding tissues and surgery was successfully completed. Although adjuvant radiotherapy may be included for malignant paragangliomas, overtreatment and undertreatment are common in certain cases as no standard criteria are currently available . As a reIn conclusion, the present report described a case of a paraganglioma in a rare location between the thyroid gland and the left common carotid artery. With the continuous development of immunohistochemical technology and the discovery of unusually located paragangliomas, the clinical data provided in this report will contribute to a greater understanding of this type of tumor.


Leiomyomas are benign tumors extending from smooth muscle cells and only few cases of paraurethral leiomyomas have been described in the literature. They are often seen in the reproductive age and around 50% of the cases are asymptomatic. We describe a 59-year-old woman with a solid mobile tumor below the symphysis revealed at a gynecological examination. Transvaginal ultrasound and MRI confirmed the tumor and excision of the paraurethral tumor was carried out. The histological examination showed a benign paraurethral leiomyoma. The postoperative period was characterized by urethral pain as well as vaginal leakage of urine. Leiomyomas are benign tumors extending from smooth muscle cells. They are the most common type of tumors in the female reproductive organs, but they are rare outside the uterus. Only few cases of paraurethral leiomyomas (PL) have been described in the literature. They are often seen in the reproductive age and around 50% of the cases are asymptomatic.We hereby present the first European case of a paraurethral leiomyoma in a postmenopausal woman.A 59-year-old woman was referred to a gynecological outpatient clinic suspected to have an ovarian cyst. Some months earlier she had been examined at the general surgery department due to abdominal pain.The patient had previously undergone a hysterectomy and was medically treated for diabetes. She was diagnosed with diverticulosis and the suspicion of a cyst in the left ovary. She had experienced constipation problems and pain in the lower left side of the abdomen. The pain increased during movement and when eating. The constipation was improved by lactulose treatment, and as a result she only suffered from abdominal pain during physical activity. Importantly, she also reported occasional incontinence, alternating with difficulties emptying the bladder.A gynecological examination revealed a solid mobile tumor below the symphysis, measuring 3 × 3 cm. A transvaginal ultrasound examination showed two normal ovaries and normal findings in the lower pelvis. In contrast, a tumor measuring 2.8 × 2.3 cm closely related to and intruding into the urethral wall was seen.A magnetic resonance imaging (MRI) revealed a sharply defined tumor, measuring 3 × 3 cm closely related to the vaginal vault . The tumSubsequently, an excision of the paraurethral tumor was carried out in collaboration between urologists and gynecologists. In the beginning of the operation a cystoscopy was performed. A little diverticel was seen in the urethra but there was no observed connection between the paraurethral tumor and the urethra. The operation was performed with only local anesthesia. Lidocaine with adrenalin was injected into submucosa in the anterior wall of vagina. The mucosa was divided lengthwise and the tumor was dissected free. During the dissection a minor lesion of the bladder occurred and lesion was sutured in two layers. Injection with methylene blue fluid confirmed no leakage in the lesion. Finally the pubocervical fascia and the mucosa in vagina were sutured. The patient was catheterized for one week and got antibiotic treatment both preoperatively and postoperatively for one week.The postoperative period was characterized by urethral pain as well as vaginal leakage of urine. A subsequent gynecological examination revealed two vesicovaginal fistulas, which were sutured. Following this the leakage stopped and the patient was free of symptoms.The histological examination showed a benign leiomyoma .PL is rare benign tumors originating from smooth muscle cells , 2. LeioDespite the fact that PLs are benign tumors, most studies recommend that the PL is removed in order to rule out a leiomyosarcoma . Other dWithout an operation the patient might still experience pressure symptoms with risk of deterioration.Although a MRI can give an idea of whether a leiomyoma or a leiomyosarcoma should be suspected, the final diagnosis is histological.Leiomyomas typically appear as round and well-defined structures. There is a low T1 signal intensity and a low to intermediate T2 signal intensity . DegenerIt is believed that ovarian hormones stimulate the development of leiomyomas because they mainly occur in the reproductive age , 8. It iIn our case neither any hormonal studies nor immunohistological chemical examination was performed.In conclusion, we present the first PL case reported in a postmenopausal European woman. Our case demonstrates the importance of a thorough vaginal exploration, including the entire vagina when uncharacteristic pain occurs in the lower pelvis in combination with voiding problems. even in postmenopausal women.