Peripheral neuropathies caused by ganglion cysts are rare. They seldom cause serious complications especially in the lower extremities. The case was a 51-year-old woman referred by her physician to the vascular surgeon with diagnosis including intermittent (vascular) claudication and deep venous thrombosis. Primarily vascular surgeon performed a doppler ultrasound of the lower extremity and calculation of the ankle-brachial index. There were no abnormal pathological findings. Careful physical examination revealed soft swelling and tenderness around the fibular head and neck. Weakness was observed in foot eversion and dorsiflexion. There was pain and tingling in the distribution of the peroneal nerve. and referring the patient to orthopedic surgeon owing to concern for a potential compressive lesion at the right proximal tibiofibular region. Electromyogram studies and physical examination confirmed a diagnosis of compression neuropathy of common peroneal nerve. Magnetic resonance imaging revealed a fluid-filled, lobulated mass indicating a ganglion cyst. One months after decompression, the patient had no complaint. Fast diagnosis and immediate management are essential to regain best possible recovery. Leg pain, loss of ankle dorsiflexion and sensory loss,neurogenic claudication most commonly produced by degenerative disc disease of the lumbar spine. On the other hand, isolated peroneal nerve compression can mimic lumbar disc disease. Differentiation of symptoms of vascular claudication from symptoms of neurogenic claudication is important. Ganglion cyst is the most frequent tumours of the upper extremity. Despite their high incidence, ganglion cyst rarely result in peripheral nerve compression . CompresA 51-year-old female patient presented to her physician with a six-month history of intermittent claudication. She developed aching, cramping, pain and weakness of her right calf. She has radiating pain and hypoesthesia, while motor weakness was less prominent. She was referred to a vascular surgeon. After examination of the case there was nothing that concerns vascular surgery. Tenderness in the area of the right fibular head with gradual development of swelling in the same area was determined. Therefore,case was referred to orthopedic surgeon. A comprehensive physical examination revealed soft tissue swelling in the region around the fibular head and neck. There was slightly weakness in foot eversion and dorsiflexion, especially of the first toe. Inversion was normal. Electromyogram studies of the common peroneal nerve demonstrated significant neuropathic abnormalities. Subsequent magnetic resonance imaging demonstrated a lobulated, multilocular,cystic-appearing mass around the proximal fibular area. It was measured approximately 3 cm × 2 cm × 2 cm (Figure Peripheral nerve lesions owing to ganglionic cysts are infrequent findings. The pathogenesis of these cysts has been the subject of controversy. However, evidence has demonstrated that they are of articular origin ,11. GangWhen a patient presenting with intermittent claudication, compression neuropathy of the peroneal nerve secondary to a ganglion cyst should be kept in mind in aspect of the differential diagnosis. After a complete history and physical examination, electromyogram and magnetic resonance imaging should be performed in terms of the differential diagnosis of a ganglion cyst. Careful preoperative evaluation, and early surgical excision by microsurgical technique in the management of the ganglion cyst should be recommended.Written informed consent was obtained from the patient for publication of this case report and accompanying images.We declare that we have no competing interests.AK is the chief author who deals the patient clinically, BCG draws SKM attention for this case, RO and VU help AK in every aspect and YD take care of this patient preoperatively. All authors have read and approved the final manuscript.Corresponding Author: Raif Ozden M.D. Assistant Professor. Mustafa Kemal University Faculty of Medicine, Department of Orthopaedics and Traumatology, Antakya, Hatay, Turkey.Vedat Uruc M.D. Assistant Professor. Kemal University Faculty of Medicine, Department of Orthopaedics and Traumatology, Antakya, Hatay, Turkey.Aydıner Kalacı M.D. Associate Professor. Mustafa Kemal University Faculty of Medicine, Department of Orthopaedics and Traumatology, Antakya, Hatay, Turkey.Yunus Dogramacı M.D. Associate Professor. Mustafa Kemal University Faculty of Medicine, Department of Orthopaedics and Traumatology, Antakya, Hatay, Turkey.


Pyogenic vertebral osteomyelitis is a rare condition usually associated with endocarditis or spinal surgery. However, it may also occur following abdominal penetrating trauma with associated gastrointestinal perforation. Diagnosis might be challenging and appropriate treatment is essential to ensure a positive outcome. In trans-abdominal trauma, 48 hours of broad-spectrum antibiotics is generally recommended for prophylaxis of secondary infections. A case report of vertebral osteomyelitis complicating trans-colonic injury to the retroperitoneum is presented and clinical management is discussed in the light of literature review. Pyogenic vertebral osteomyelitis is a rare condition usually related to endocarditis or spinal procedures ,2. It maA 21 year-old male was admitted to the emergency department for abdominal penetrating injury by a pointed metal stick . On primary survey, vital signs were normal and clinical examination demonstrated a single penetrating wound at the right inferior abdominal quadrant. No peritoneal free fluid was detected on ultrasound scan. Tetanus prophylaxis was administered. A thoraco-abdominal computed-tomography (CT) scan showed a retroperitoneal hematoma surrounding the sub-hepatic inferior vena cava with no intraperitoneal fluid or other abnormalities  levels and leukocyte count. Abdominal CT scan showed no signs of intra or retroperitoneal abscess; the retroperitoneal hematoma appeared decreased in size. No evidence of chest or urinary tract infections was demonstrated. Eventually, magnetic resonance imaging (MRI) showed osteomyelitis at III and IV lumbar vertebrae with bone erosion and inflammation of disc space; a small collection in the paravertebral tissue at that level was also detected Figure . No vertPyogenic vertebral osteomyelitis is a rare disease that counts for 2-5% of all cases of osteomyelitis, with an annual incidence of 0.4 to 2.4/100′000 among European population . In trauAppropriate prophylaxis for infective complications in trauma patients has been largely investigated. Colon perforation is considered to be a risk factor for developing secondary infections and use of broad-spectrum antibiotic prophylaxis is generally recommended for at least 24–48 hours ,10. HowePyogenic vertebral osteomyelitis is a rare complication that may occur following abdominal penetrating injury, especially when bowel is perforated. Diagnosis can be difficult and should be guided by high clinical suspicion. An accurate management and appropriate treatment are essential to ensure a positive outcome. 48 hours of broad-spectrum antibiotics is suggested for prophylaxis of secondary infections following trans-abdominal trauma.Written informed consent was obtained from the patient for publication of this case report and accompanying images.The authors declare that they have no competing interests.AZ was involved in the clinical management of the patient and drafted the manuscript. PD and PB performed the operation and contributed in conceiving the manuscript. AM admitted the patient and reviewed the manuscript. All authors read and approved the final manuscript.


GIST of anal canal is an extremely rare tumor, accounting for only 3% of all anorectal mesenchymal tumors and 0.1–0.4% of all GIST. GIST with large tumor size and high mitotic activity are highly malignant, but the biological behavior of anorectal GIST is less clear. Abdominoperineal resection (APR) or conservative surgery is the best treatment option. Imatinib mesylate, a tyrosine kinase inhibitor, has shown promising results in its management. We present a case of anorectal GIST diagnosed by computed tomography (CT) scan, magnetic resonance imaging (MRI), and colonoscopy with biopsy. The patient underwent abdominoperineal resection (APR) and was confirmed on histopathology to have anal canal GIST with tumor size more than 5 cm in maximum dimension and mitotic figures more than 5/50 high power field (HPF). The CD117—immunoreactive score—was 3+ in spindled cells. Therefore the patient was put on adjuvant imatinib mesylate 400 mg daily.Gastrointestinal stromal tumors or “GIST” are mesenchymal neoplasms expressing KIT(CD117) tyrosine kinase and showing the presence of activating mutations in KIT or  PDGFRα  [Gastrointestinal stromal tumor or “GIST” was a name given in 1983 to a group of gastrointestinal tumors which were otherwise unclassifiable as being of smooth muscle or neurogenic origin [r alpha) . It is tr alpha) . GIST ofr alpha) , 5. Hower alpha) , 7.A 61-year-old male presented with pain during defecation and occasional bleeding per rectum over 2 months. Pain was nonradiating, dull aching, and persistent. Pain increased with constipation. Bleeding was frank red and came as drops after passage of stool. There was no tenesmus. There was no dizziness, weakness, pica, or weight loss. There was no significant relief with medication.The patient was not a known case of piles/diabetes mellitus/hypertension/tuberculosis/or any other chronic ailment. The patient was a known alcoholic and smoker. He had no urinary complaints. There were no other complaints referable to chest and cardiac or nervous system.He was a heterosexual with no known drug allergies.On examination he was moderately built and of average nourishment. Karnofsky performance scale was more than 80. There was no pallor/icterus/pedal edema/lymphadenopathy. On digital rectal examination, a hard indurated nontender mass was felt from 2 o'clock to 9 o'clock, starting from the anal verge. The mass had a bosselated surface, and the overlying mucosa was tethered. The upper border of the mass could not be reached. No inguinal, iliac, para aortic, or supraclavicular lymph nodes were palpable.His hematological and biochemical parameters were all within normal limits, and he was HIV seronegative. His transrectal ultrasound revealed an enlarged prostate with insignificant postvoid residual urine. There was significant ill-defined mural thickness and hypervascularity along anorectal canal causing indentation of prostate. CT scan of the abdomen and pelvis showed an endo-exophytic soft tissue mass at anorectum suggestive of mitotic pathology. MRI pelvis revealed a lesion involving rectum and anal canal with extension into intersphincteric plane and puborectalis Figures  and 2. HThe patient underwent abdominoperineal resection with end colostomy Figures  and 4. HThe incidence of anal cancer in the western world is between 7 and 9 per million population. It contributes to only 1.5% of all malignancies of the digestive system . Of thesThe anal canal extends from the perianal skin  to the rectal mucosa. An important landmark within the canal is the dentate or pectinate line, which represents the end of the squamous mucosa and the beginning of a zone of transition from squamous to nonsquamous  mucosa. Thus, tumors arising in the anal canal can be either keratinizing or nonkeratinizing depending on their location in relation to the dentate line. Importantly, both keratinizing and nonkeratinizing tumors appear to have similar biology and prognosis . Adenocaα genes are the basis for the diagnosis of this neoplasia and represent the criteria for surgical therapy, expected chemotherapy response, and clinical outcomes [GIST are currently thought to originate from interstitial cells of Cajal. The presence of interstitial Cajal-like cells has been reported in several extraintestinal organs including urinary bladder, prostate gallbladder, omentum, uterus, fallopian tube, and atrial and ventricular myocardium , 12. Thioutcomes .Risk factors for anorectal malignancies include female sex, patients with history of human papilloma virus (HPV) infection, human immunodeficiency virus- (HIV-) positive patients, patients who engage in anal receptive intercourse, presence of sexually transmitted disease, a history of more than 10 sexual partners, and a history of cervical, vulval, or vaginal cancers. Immunosuppressed individuals including renal transplant patients and those on chronic glucocorticoid therapy also appear to be at increased risk. Smoking is also a risk factor .Most patients present with locoregional disease, and less than 20% of patients will present with or develop distant metastases. GIST with large tumor size and high mitotic activity are highly malignant, but the biological behavior of anorectal GIST is less clear , 15. GISGIST are best treated by surgery and are not radio- or chemosensitive. However, controversy exists whether abdominoperineal resection (APR) or conservative surgery is the best alternative . Though Anorectal GIST, though rare, should be considered in the differential diagnosis of tumors in this region, especially if the pre-operative biopsy is equivocal. Gross and histopathological are both important, as prognosis depends on tumor size as well as grade. However, prognosis is usually better than for corresponding carcinomas in the region. Immunohistochemistry is a must, as CD-117 score is not only diagnostic but also guides adjuvant therapy and is an important prognostic marker.Our case was a case of invasive anal canal GIST with tumor size more than 5 cm in maximum dimension. Mitotic figures were more than 5/50 HPF. The CD117—immunoreactive score was—3+ in spindled cells. Hence our patient has been put on adjuvant imatinib mesylate 400 mg daily.


Fusion is a rare occurrence in molar teeth. The purpose of this rare case presentation is to describe the nonsurgical endodontic treatment of maxillary molar. A 28-year-old patient was referred for endodontic treatment of her chronic apical abscess of right maxillary second molar. In the clinical examination, a sinus tract adjacent to involved tooth and a small crown of supernumerary tooth fused to the buccal surface of the molar at gingival margin was observed. Endodontic treatment was decided for the involved molar for functional reason. Recall examination, a year after completion of endodontic and restorative treatments, showed the tooth was clinically asymptomatic and there was no radiographic lucency around the apical region. Dental fusion is defined as the merging of two or more teeth at enamel and/or dentinal level -2. The eLocal metabolic interferences, which occur during differentiation of the tooth germ, may be another cause for dental fusion . Genetica) Dehiscence: laceration resulting from trauma and affecting the crown of a tooth germ;b) Concrescence: the merging of two or more teeth at root cementum;c) Gemination (twinning): attempted division of a tooth germ in two;d) Schizodontia: complete division of a tooth germ in two; ande) Dens in dente: enamel penetration into the pulp chamber.Dental fusion occurring particularly in anterior teeth with apparent equal distribution between the two jaws, and is more common during the deciduous dentition phase. It is very rare in molars. The prevalence of this dental anomaly is estimated at 0.1-2.5% of individuals -9.Fused teeth may present two independent endodontic systems, one pulp chamber dividing into two root canals or, less often, a single root and one or two pulp chambers ,10.The fusion may be partial or total depending on the stage of tooth development at the time of union, including only the tooth crowns or the tooth crowns and roots, respectively . AlthougDental fusion is generally asymptomatic and dose not requires any treatment. However, there could be poor aesthetics, periodontal damage or caries leading to pulp necrosis .This case report describes the endodontic management of a maxillary second molar fused with a supernumerary.A 28-year-old woman was referred for endodontic treatment of an anomalous tooth (maxillary right second molar). The patient complained of draining sinus tract in the gingiva of involved tooth ,Figure . Her medClinical examination revealed that the maxillary right second molar was fused with a supernumerary. There was an extra small crown of abnormal appearance adjacent to the normal tooth. A draining sinus tract was present at the distobuccal of the fused tooth. No caries could be detected. The tooth displayed physiological mobility, but was sensitive to percussion. Thermal and electrical pulp testing didn’t give a normal response while probing revealed no periodontal pocketing around the tooth.Radiographic examination demonstrated mesio- buccal and distobuccal roots fused. A radiolucent area, 5 mm in diameter, with distinct borders was seen around the roots. Radiographic tracing with a gutta-percha cone revealed that the sinus tract led to the radiolucent area ,Figure . A diagnAfter local anesthesia  and rubber dam isolation, working lengths were determined by radiograph  and chemThe recall examination after 1 year revealed that periodontal condition was healthy, the tooth was asymptomatic, and complete bone regeneration was seen on radiograph .As a common opinion, the clinical differentiation between dental fusion and germination is difficult. For the purpose of having better differentiation between these anomalies, it has been suggested that the teeth in the arch be counted with the anomalous crown counted as one. The presence of all teeth indicates gemination, whilst one tooth less than normal indicates fusion . UnfortuIn this case, the history of wisdom tooth extraction, clinical observation of a small abnormal fused crown of a supernumerary tooth, and normal number of remaining teeth led us to established diagnosis of either dental fusion of the second maxillary molar with a supernumerary tooth or gemination of the second molar.A prerequisite for nonsurgical endodontic treatment of anomalous teeth is a careful clinical and radiographic examination. Many clinicians revealed very complex internal anatomy in double tooth and stressed the significance of knowledge with the root canal morphology before starting the treatment -19. DuriSuccessful endodontic treatment depends on careful cleaning, shaping and three-dimensional obturation of the root canal system. Although mechanical debridement of the root canals in double tooth is difficult, but the combination of chemo-mechanical instrumentation and the use of sodium hypochlorite in the present case was sufficient.In our opinion, an individualized treatment plan is required in double tooth, since these cases may or may not require different treatment modalities compared to normal teeth.


Fungal infections are serious and important cause of morbidity and mortality in immunocompromised patients. Angioinvasive aspergillosis causing mycotic aneurysm of major blood vessels is very rare and only a few cases are reported in literature. We hereby report one case of acute lymphoblastic leukaemia presenting with this fatal complication. Mycotic aneurysms of subclavian artery, caused by angioinvasive aspergillosis, in immunocompromised children are very rare. Only one case has been reported previously to the best of our knowledge.We present a case of 6-year-old girl undergoing chemotherapy for acute lymphoblastic leukemia, who presented to us initially with pneumothorax and then rapidly developed a fatal giant intrathoracic pseudoaneurysm of left subclavian artery over a period of 1 month, due to infection with angioinvasive aspergillosis. Serial chest radiographs, computed tomography (CT) and computed tomography angiography were performed to establish the diagnosis and follow the course of the disease which eventually had a fatal outcome.A 6-year-old girl, a diagnosed case of acute lymphoblastic leukemia on chemotherapy in another hospital, was admitted to our hospital on 11 August 2008, with complaints of fever and cough for the last 15 days, not responding to antibiotics.3, platelet count of 41,500/mm3 and Hb of 6.3 g/dl.Her blood examination on the day of admission showed total leukocyte count of 4100/mmChest radiograph  done on High-resolution computed tomography (HRCT) of chest, done 3 days later on 14 August 2008, showed left apical small soft tissue density mass with air crescent inside , invadinHer fever subsided; however, cough and respiratory distress persisted. Serial chest radiographs showed a homogenous left upper zone mass with well-defined lower margin which increased in size to almost completely occupy the left hemithorax. A contrast-enhanced CT (CECT)  done on CT angiography  confirmeShe succumbed to her illness and died while on her way to higher center for endovascular management of aneurysm.Mycotic aneurysms of subclavian artery caused by angioinvasive aspergillosis are very rare and only a few cases are reported in literature.Angioinvasive aspergillosis is a serious infection frequently seen in immunocompromised patients. It is characterized on CT by nodules surrounded by ground glass halo  or pleural based wedge-shaped areas of consolidation. Separation of necrotic fragment from adjacent lung parenchyma results in formation of air crescent sign.The halo sign represents hemorrhage around the central infarctions caused by angioinvasion. Cavitatiet al.[Visrutaratna et al. reportedet al.[Saliou et al. reportedThe various mechanisms postulated in the development of mycotic aneurysms include: embolism of vasa-vasorum, direct wall invasion and erosion of the vessel from an adjacent lesion of the lung or via lymphatic system.Very few cases of aspergillosis induced mycotic aneurysms of sites other than subclavian artery5 in chilIn our case also, the patient was immunocompromised and had typical CT features of angioinvasive aspergillosis. She presented with fever and pneumothorax . Her aneurysm progressed rapidly over a course of 1 month to completely occupy left hemithorax and ultimately proved to be fatal.We report this case for its rarity and to alert the readers that in appropriate clinical setting of immunocompromised state with CT features of aspergillosis, a strict vigil should be kept over the patient regarding development of mycotic aneurysm so that timely intervention can be done.


Melanocytic meningeal tumours are rare extra-axial neoplasms of the nervous system, with only three reported cases in the cavernous sinus. Herein we describe for the first time the association of ocular melanosis and multiple intracranial melanocytic meningeal tumours, with the presenting lesion being in the cavernous sinus. The importance of this association is discussed together with the diagnostic and therapeutic challenges of the case.A 20-year-old man presented with a left sixth cranial nerve deficit; general examination documented only congenital melanosis of the homolateral eye. MRI examination showed a space occupying lesion in the left cavernous sinus, which was followed conservatively for 2 years, until a new space occupying lesion was evident at the level of the right frontal convexity: both lesions presented with neuroradiological characteristics suggestive of melanin content.The frontal convexity lesion was removed: intraoperatively the dura was markedly and diffusely melanotic. Histological examination documented a melanocytic meningeal tumour, with a proliferative index of 3 %. The patient underwent 3D-Conformal Radiation Therapy on the lesion of the cavernous sinus , with initial tumour reduction. Three years later, due to a symptomatic growth, he underwent partial removal of the lesion in the cavernous sinus. Histological examination was unchanged. He then received adjuvant Temozolomide with Low Dose Fractionated Radiation Therapy (LD-FRT). Due to further disease progression cisplatin plus fotemustine were administered, concomitant with LD-FRT: after two cycles MRI documented significant disease regression. After a period of apparent disease control, the patient presented with persistent cough and evidence of multiple thoracic metastases, which lead to his death, seven years after presentation.Intracranial melanocytic meningeal tumours are challenging lesions, both from a diagnostic and therapeutic point of view; though rare, the possible association with ocular melanosis should be recognized and might facilitate an early diagnosis. Surgery remains the best possible option when feasible. In the event of partial resection, this “benign” disease might be clinically aggressive. Melanocytic meningeal tumour is a rare tumour of the nervous system with approximately 110 reported cases . The posHerein we describe the unique case of the association of simple ocular melanosis and multiple intracranial melanocytic meningeal tumours. The diagnostic and therapeutic challenges of the case are presented together with a concise literature review.A 20 year-old Caucasian male presented to our department with incomplete left abducens nerve palsy; neuroradiological examination documented three intracranial space occupying lesions, respectively in the left cavernous sinus, on the right frontal convexity and on the superior aspect of the right petrous pyramid Figure .He presented with a two year history of diplopia, of subacute onset, due to a left sixth cranial nerve palsy. Neurological examination was otherwise normal at the time; he denied any pain and had no fever. Head CT scan documented a small space-occupying lesion in the left cavernous sinus, with well-defined margins and minimal contrast enhancement Figure A. Brain At this stage in his care the patient was referred to our department. Neurological examination documented diplopia only in the extreme left gaze. General examination documented patches of bluish discoloration of the sclera at the level of the left eye, which the mother reported to be congenital Figure ; there wOn haematoxylin and eosin (H&E) staining Figure B and antFive days after surgery the patient developed a marked diplopia, pain in the eye and a third cranial nerve palsy. An urgent CT scan documented a minimal enlargement of the cavernous sinus lesion (not shown). Corticosteroid treatment was initiated and three weeks later there was complete resolution of both pain and cranial nerve palsy. Corticosteroids were subsequently tapered and discontinued. The patient underwent 3D-Conformal Radiation Therapy (3D-CRT) of the lesion of the cavernous sinus , with an initial tumour reduction. Three years later, due to a symptomatic growth, the patient underwent surgical debulking of the lesion with resolution of the pre-operative trigeminal neuralgia and persistence of third cranial nerve palsy. Histological examination of the lesion documented features similar to the previously resected lesion, with a proliferative index of 2 %.He then received four cycles of adjuvant Temozolomide (150–200 mg/mq/die for 5 days q 28); the third and fourth cycle were combined with Low Dose Fractionated Radiation Therapy (LD-FRT) 0.4 Gy twice daily ,12, withMeningeal melanocytic tumour is a rare tumour of the nervous system, with approximately 110 cases reported since 1972 . It origOn computerized tomography (CT) imaging, melanocytic tumors appear as iso- to slightly hyper-dense lesions with homogeneous enhancement after contrast media administration ,18. ThisIntracranial melanocytic non-metastatic tumors can be associated with nevus of Ota or oculodermal melanosis ; to the Complete surgical resection is the best treatment for meningeal melanocytic tumors . If not Radiotherapy improves outcomes and a total dose of 50.4 Gy (1.8 Gy per fraction) appears appropriate for most patients . RadiosuTo the best of our knowledge there is also a lack of data concerning the role of chemotherapy in the treatment of melanocytic tumors . Our decRare cases have been reported with leptomeningeal spread -29 transThis is the first reported case of the association of simple ocular melanosis and intracranial melanocytoma. Awareness of this association and of the radiological characteristics of the lesion might help the pre-operative diagnosis of this rare intracranial tumor. The clinical course of our patient indeed confirms the relatively aggressive behavior of this tumor.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 Series Editor of this journal.The authors have no competing interests.FD conceived the study, reviewed the literature and wrote the first draft; CC conceived the study, provided the radiological data, wrote the draft of the radiological part of the study and critically reviewed the manuscript; LL provided the histological data, and critically reviewed the manuscript; MB provided the data on the radiotherapic treatment, and critically reviewed the manuscript; PDB conceived the study, reviewed the literature and critically reviewed the manuscript; NM reviewed the literature and critically reviewed the manuscript; GZ critically reviewed the drafts and final manuscript; GM critically reviewed the manuscript; RP conceived the study and critically reviewed 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/1471-2407/12/220/prepub


Hemangiopericytoma is an uncommon perivascular tumor. Rectal Hemangiopericytomas are extremely rare. To the best of our knowledge, only two cases have been reported in the literature.We report the case of a 37-year-old Asian woman with an Hemangiopericytoma rising from the anterior wall of her rectum. Abdominopelvic computed tomography showed a 7.4 cm solid mass between her uterus and her rectum. Heterogeneous gradual enhancement after intravenous injection of contrast material was noted with several tortuous vessels around her tumor. Intra-operative findings indicated a capsule and well-circumscribed solid tumor connecting with the anterior wall of her rectum by a small pedicle. With immunohistochemical stains, her tumor cells reacted positive for Bcl-2, CD34, and ki67 and negative for CD10, CD117, S100, and Desmin. Follow-up computed tomography scans have shown no tumor recurrence or metastasis signs.Rectal Hemangiopericytoma is a rare tumor with non-specific imaging findings. Hemangiopericytomas should be included in the differential list when a massive tumor with heterogeneously gradual enhancement in the regions of the rectum is encountered. Hemangiopericytoma (HPC), an uncommon perivascular tumor, accounts for 1% of primary vascular tumors and occurs most frequently in the extremities, pelvis, head and neck, and meninges . It has A 37-year-old Asian woman was referred to our hospital because of lower abdominal pain that began four months earlier. A vaginal palpation revealed a hard, adhering, and painless mass. Another physical examination revealed no abnormalities. The results of laboratory tests, including complete blood count, serum electrolytes, creatinine, and urea, were normal. Our patient underwent an intra-vaginal ultrasonography (US) examination, which revealed a 6.0 × 7.6 × 6.0 cm solid mass between her uterus and rectum Figure . An abdoHPC was first described in 1942, by Stout and Murray . ConsequHPCs have some characteristic clinical features. One of these features is the rate of recurrence, which is as high as 52% of cases   of the rectum, and retroperitoneal tumors. On MRI, non-degenerating uterine myomas show entirely or predominantly low signal intensity on T2-weighted images, and it displays differentiation between uterine myomas and HPCs because HPCs appear as high signal intensity on T2-weighted images. But degenerated uterine myomas may have varied appearances on T2-weighted and contrast-enhanced images according to the hyaline or myxoid degeneration, degree of interstitial edema, cystic degeneration, necrosis, fibrosis, calcification, hemorrhage, carneous degeneration, and fat.Small tumors typically appear as homogeneous soft-tissue masses with moderate contrast enhancement, whereas large tumors often appear to have a heterogeneous density or signal intensity because of ulceration, necrosis, or cavitation. Thus, precise differential diagnosis is very difficult, but GISTs rarely cause lymph node metastasis; if extensive lymph node metastases are found, other diseases should be considered .It is very difficult to differentiate retroperitoneal tumors, such as leiomyosarcoma, liposarcoma, neurogenic tumors, and malignant fibrous histiocytoma (MFH), on the basis of imaging findings. Of these tumors, liposarcoma is one of the most common primary neoplasms in the retropenitoneum. The lipoma-like component may lead to a diagnosis of liposarcoma, although abdominal tumors with fat are not always liposarcomas . NeurogeTumor resection is the mainstay method of treatment of HPCs. Post-operative adjuvant radiotherapy should be offered to all patients, regardless of the degree of resection. The optimal management of local recurrence is indicated by the size of the recurrence and the overall systemic disease burden present at the time of recurrence. Post-operative radiation therapy does not confer any significant protection against the development of distant metastases. For this reason, long-term clinical and radiographic follow-up of these patients is imperative given that recurrence or metastasis or both often take several years to develop.Rectal HPC is a rare tumor with non-specific imaging findings. HPCs should be included in the differential list when a massive tumor with heterogeneously gradual enhancement in the regions of the rectum is encountered.CT: computed tomography; GIST: gastrointestinal stromal tumor; HPC: hemangiopericytoma; HU: Hounsfield units; MFH: malignant fibrous histiocytoma; MRI: magnetic resonance imaging; US: ultrasonography.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.LL gathered the data, performed the literature review, and edited the manuscript. LJZ and CSZ participated in the acquisition and analysis of the literature data and helped to draft the manuscript. GML revised the final manuscript. All authors read and approved the final manuscript.


Angiomyofibroblastoma (AMFB) is a rare benign mesenchymal tumour that occurs almost exclusively in the vulvovaginal region of women but can also occur occasionally in the inguinoscrotal region of men. It is a well-circumscribed lesion that clinically is often thought to represent a Bartholin's gland cyst and usually does not form a pedunculated mass. To our knowledge, only five cases of vulvar AMFB with pedunculated mass have been reported in the English literature and all cases involving the labia majora and middle-aged women. We report the first case of pedunculated AMFB of the vulva occurring in a young woman of 21 years old and involving the left labia minora. After excluding the most common diseases, pedunculated AMFB should be part of differential diagnosis in the workup of any pedunculated vulvar mass even in young women with a lesion involving the labia minora. We reviewed the literature and summarized all reported cases. Angiomyofibroblastoma (AMFB) is a rare benign mesenchymal tumor usually occurring in the vulvovaginal area of middle-aged women –3. HistoA 21-year-old woman came to our observation reporting the presence of a painless vulvar neoformation starting four years before, which had grown during the last months. She ignored the mass, despite its gradual enlargement during her disease course, and the complaint was of discomfort when seated and a localized burning sensation. Her personal history was noncontributory for previous surgery and laboratory data  showed no significant abnormalities. Clinical examination showed a pedunculated bilobulated neoformation of ten centimeters in diameter of tense elastic consistency, at the level of the left labia minora . On gyneThe mass gave a clinical impression of vulvar fibroid, and a simple tumor excision was subsequently performed at the site of the stalk. During surgery, to exclude a malignancy lesion, a rapid intraoperative pathologic diagnosis of the tumor was performed, and it was reported as a benign soft lesion of the vulva. On gross examination, the lesion was well circumscribed, weighed 1230 gr, had a soft to firm consistency, and appeared homogeneously light gray to tan in color on the cut surface. No foci of hemorrhage nor necrosis was detected.α-smooth muscle actin, S-100 protein, cytokeratin, and CD34. All these features were in favour of the diagnosis of AMFB. Surgical margins of the lesion were clear, and the pedicle was not involved by the tumor.Microscopically, the mass consisted of fibroconnective tissue with abundant vessels of various wall thickness Figures . No mitoCurrently the patient is in followup for two years with clinical examination every six months with no evidence of recurrence.This distinctive tumor was first delineated by Fletcher et al. in 1992 who described 10 cases involving the vulva  with onlOn gross examination they are typically well circumscribed and ranged from 0.5 to 12 cm. They have a soft to rubbery consistency and a bulging, pink, and somewhat lobulated sectioned surface . The angImmunohistochemical stains show uniform staining for vimentin and staining of a variable number of cells for desmin in most cases. The tumor cells also show variable expression for muscle actin and smooth muscle actin and are frequently positive for estrogen and progesterone receptors . The expThe tumor usually exists as a sharply circumscribed mass in the subcutaneous tissue of the vulva and usually does not form a pedunculated mass, which represents an exceptional event . To our All cases are reported in α-smooth muscle actin and CD34. Staining for estrogen and progesterone receptors was observed in the nucleus of all tumor cells. Given the data reported in the literature on the pedunculated AMFB of the vulva, our case is the first concerning a young woman of 21 years old and involving the labia minora. The patient underwent simple excision with clear margin of the lesion and currently is in followup performing clinical examination every six months for two years with no recurrence. This paper aims to emphasize that, after excluding the most common vulvar diseases, the appearance of a pedunculated AMFB should be part of differential diagnosis in the workup of any pedunculated vulvar mass even in young women with a lesion involving the labia minora. The treatment of choice is a surgical excision with clear margins, which is resolutive as demonstrated by reported cases in the literature.


Carcinoma of unknown primary origin (CUP) is defined as the histological diagnosis of metastasis without the detection of a primary tumor. The incidence of CUP in all patients with a malignant disease has been reported to be between 3 and 15%. Esophageal cancer is the third most common type of cancer of the digestive tract and the seventh most common cause of cancer-related mortality worldwide. The overall incidence of the disease is highest in males >50 years old. Brain metastases have been reported in only 1.7–3.6% of all patients with different types of esophageal cancer. Brain metastasis as the presenting form of esophageal carcinoma is highly uncommon. The present study reports the case of a patient with an unknown primary tumor who presented with a cerebral metastasis, without extra-neurological symptoms. The CUP was subsequently diagnosed as an esophageal carcinoma. The identification of the primary tumor in patients presenting with cerebral metastasis is mandatory in order to plan a proper treatment strategy. Unfortunately, in certain instances, the primary tumor remains occult even after a extensive and scrupulous diagnostic work-up ,2.Esophageal cancer was well described at the beginning of the 19th century, and the first successful resection was performed by Torek in 1913 ,5, whileA 69-year-old male patient was referred to the Neurological Centre of Latium (NCL) in July 2011 due to rapidly progressing right-hand apraxia and agraphia and recent reoccurring headaches. The patient was a heavy smoker (>40 pack years) and had a history of alcohol abuse. A physical examination indicated right, upper-limb prevailing hemiparesis . Furthermore, right dysmetria, adiadochokinesia and somatesthesic disturbance were present.An imaging work-up included computerized tomography (CT) scanning and brain magnetic resonance imaging (MRI), which indicated a low-density, contrast-enhancing subcortical lesion that was localized in the proximity of the left motor area, with perifocal edema and flattened sulci . Whole-b−) and carcinoembryonic antigen-positive (CEA+)].The patient underwent microsurgical resection of the mass via a trans-sulcal approach. The histology was conclusive for a well-differentiated adenocarcinoma . The staOnce discharged, the patient was scheduled for a complete oncological work-up that included a whole-body positron emission tomography (PET) scan and digestive tract endoscopy. The esophagogastroduodenoscopy revealed a flat, whitish area extending for ~2 cm in the longitudinal axis lying on the lower third of the thoracic esophagus, extending to the submucosa. No evidence of enlarged regional lymph nodes was present (cT2N0M1). The patient refused further surgical procedures and was scheduled for whole-brain and local radiotherapy and chemotherapy treatments. The protocol that was used consisted of a fractionated 50 Gy dose (2 Gy/day) and concurrent chemotherapy, including paclitaxel and cisplatin. An antiblastic therapy protocol was accomplished with a favorable course, despite an episode of non-fatal toxicity occurring subsequent to 2 months (transient leukopenia and pneumonia).At the last follow-up at 14 months post-surgery, the patient was alive and well . A neurological examination revealed amelioration of the motor function of the right hand and arm, with a positive impact on the quality of life physical domain, in spite of the development of a minor depressive syndrome. The patient was alive at 16 months after the initial diagnosis and, to date, no local or distant tumor recurrence has been documented.The diagnosis of a brain metastasis is usually made during the routine follow-up examinations of patients with a known type of cancer. In the case of a CUP presenting with brain metastases, either a neurosurgeon or a neurologist are consulted prior to the oncologist. The incidence of brain metastasis of unknown primary origin is almost equal to that of cerebral metastases where the primary cancer is known ,7. Moreoet al, anaplastic astrocytomas (AAs), metastases and lymphomas, all of which are often characterized by similar conventional CT and MRI findings . Howeveret al suggestePatients with a newly detected brain mass and no history of other tumors, usually undergo extensive and expensive diagnostic testing to identify the primary neoplasm prior to the selection of a biopsy site. In this situation, a neurosurgical procedure may be considered as the most appropriate step to be taken in order to provide a definitive diagnosis, and also to avoid the unnecessary waste of time and resources. By contrast, it should be noted that esophageal cancer often lacks distinctive morphological characteristics leading to the potential unsuccessful identification of the site of origin by routine histological examination . TherefoEsophageal cancer is a highly lethal disease with an extremely poor 5-year survival rate regardless of the stage of the disease. In 2008, esophageal cancer had an estimated annual incidence rate of 19.2/100,000 for males and 4.2/100,000 for females, who are exposed to a lower risk compared with males . At the There was a trend toward a worse survival in patients with liver metastases and patients in recursive partitioning analysis (RPA) class II–III vs. RPA class I. Moreover the reported 5-year survival rate ranges from 20 to 36% subsequent to intentionally curative surgery due to a high rate of either local or distant recurrence. The distant metastasis rate has been reported to be 26% within 20 months after radical surgery. Hematoxylin and eosin staining is one of the most effective predictors of survival in esophageal cancer due to the number of lymph node metastases detected using this technique ,19.The case reported in the present study is exceptional due to the patient’s prolonged, disease-free survival, the excellent response (to date) to non-surgical treatment of the primary disease and the clinical presentation resembling a brain glioma, which made the post-operative search for the primary tumor a challenge. Radical surgery on the brain lesion, even though located in an eloquent area, resulted in improvement to the presenting neurological deficits and represented the basis for a proper oncological assessment and successful management.Esophageal carcinoma rarely presents with an isolated brain metastasis. In such cases, other than a careful assessment aimed to discover the unknown primary origin, removal of the lesion must be considered in order to treat the presenting symptoms and obtain a rapid histological diagnosis. Further studies are warranted in order to assess whether progression of the primary disease may be accelerated by post-surgical stress. This hypothesis should also be investigated against the potential benefits in terms of an improved quality of life and the advantages of a shorter diagnostic time.


Introduction. Lymphoepithelioma-like carcinoma (LELC) of the salivary gland is a rare tumor. Currently, surgery with or without radiotherapy is the recommended treatment for all salivary gland carcinomas. However, in contrast to other high-grade salivary gland carcinomas, LELCs are considered radiosensitive. There are only a few published reports of radiotherapy alone for the treatment of salivary gland LELC. Case. We present two cases of LELC of the salivary gland. One was treated with surgery and postoperative radiotherapy, and the other was given a single cycle of chemotherapy and then radiotherapy. Currently, both patients have no evidence of disease. Conclusion. Radiotherapy as a single modality should be reevaluated. The role of systemic chemotherapy to gain systemic control should be addressed due to noteworthy metastatic disease. Lymphoepithelioma is a term that was used by Regaud and Schmincke in 1921 to describe certain tumors of the nasopharynx . SignifiThe mainstay of treatment for salivary gland tumors is surgical resection. Postoperative radiotherapy is indicated for unfavorable prognostic features, such as close or positive surgical margins, high-grade malignancy, perineural invasion, tumor infiltration of adjacent organs, recurrent disease, and regional metastasis, and has been shown to improve local-regional control and survival, particularly in advanced-stage disease . In contA 19-year-old female with no history of chronic diseases complained of a swelling in the floor of her mouth. Physical examination revealed a 5-6 cm right sublingual swelling. Biopsy from the sublingual lesion was consistent with LELCs, most probably a primary tumor of the sublingual salivary gland, somewhat similar to those of nasopharyngeal origin. No distant metastases were noted. The patient was referred to the Maxillofacial Surgery Department and underwent a resection of the sublingual salivary gland, a marginal mandibulectomy from teeth 33 to 47, partial glossectomy and bilateral cervical dissection, reconstruction of the jaw with titanium plate, prosthetic dental implants, and a myocutaneous free-flap from her left arm. The pathology report indicated LELC with a maximal diameter of 3 cm. The tumor invaded the fat and mucosa with 0.3 cm clear lateral margins and 0.5 cm clear caudal margins. The posterior margins were microscopically involved by the tumor. No lymph node metastases were noted.A week after the surgery, the patient suffered from necrosis of the flap and it was excised. During this procedure, posterior extension of the resection was performed due to positive microscopic margins. The wound was primarily closed. The pathology report showed no residual disease of the new resected specimen. A month after surgery, the patient suffered from a cervicocutaneous fistula that was treated conservatively. She underwent a percutaneous endoscopic gastrostomy and was referred to the Oncology Department for radiotherapy. She received external beam irradiation to the neck and surgical bed, with a total dose of 50 Gy to the bilateral neck and the tumor bed and a boost dose of 16 Gy to the tumor bed. Two and a half years after the diagnosis, the patient has no evidence of disease, although she suffers from severe neck fibrosis.A 62-year-old female was diagnosed with a mass in the left parotid gland and left supraclavicular lymphadenopathy. A biopsy from both masses revealed LELC. Random biopsies from the nasopharynx were negative for malignancy. PET-FDG scan showed pathological uptake in the left parotid gland and left supraclavicular lymph nodes. The patient was given a cycle of chemotherapy with docetaxel (120 mg intravenous), cisplatin (130 mg intravenous), and 5-fluorouracil (continuous drip of 1300 mg/per 22 hours for 5 days). During the treatment, the patient suffered from grade 2 nausea and vomiting. A few days after the first chemotherapy cycle, she was hospitalized for 6 days due to neutropenia and fever. She received antibiotics and daily injections of granulocyte colony-stimulating factor (GCSF). It was decided not to continue chemotherapy, and the patient was referred for radiotherapy. Prior to radiation, she underwent a percutaneous endoscopic gastrostomy. The patient received external beam irradiation; the irradiation field included bilateral neck and left parotid with a total dose of 50 Gy and a boost of 16 Gy to the left parotid gland and involved the left supraclavicular lymph nodes. PET-FDG scan performed three months after the end of the radiation treatment showed no pathological uptakes. Four years after the initial diagnosis of LELC, the patient has no evidence of disease. It is noteworthy that this patient did not undergo surgical treatment of the tumor.Salivary gland malignancies are uncommon and comprise approximately 3% of annual, newly diagnosed head and neck carcinomas in North America , 14. Of The prognosis of salivary gland tumors depends on the histological type, stage, grade, completeness of the surgical resection, and, when the resection is incomplete, whether or not postoperative radiotherapy is administered , 13. TheDubey et al.  retrospeWith the current use of advanced radiotherapy techniques, such as IMRT and IGRT, complication rates are expected to be lower. There was no statistical significance in the 5-year local and regional control rates in the all-patients group and in the radiotherapy-alone group. It was recommended that locoregional treatment strategy for patients with non-nasopharyngeal LELC should include radiotherapy to all primary tumors. Because the main cause of treatment failure was distant metastases and patients with regional lymphadenopathy had relatively high rates of distant metastases, it was recommended that patients who present with regional lymphadenopathy should be treated with chemotherapy as well . In the Two studies of salivary gland LELC in which surgery was the main treatment demonstrated interesting results. In the first study, which included seven cases of LELC of parotid gland origin treated by surgery and postoperative radiotherapy , two patIn the second study , all eigLymphoepithelioma-like carcinoma of the salivary gland is a rare entity, but, despite the poor differentiation and high-grade histological appearance, its clinical course is better than other poorly differentiated tumors. It is considered to be radiosensitive and has similar histopathologic characteristics as lymphoepithelioma of the nasopharynx , in whic


Histiocytic sarcoma is a rare neoplasm with few cases reported in the literature of which some were diagnosed in animals. This neoplasm arises from abnormal reticuloendothelial system cell proliferation of histiocytes and has an aggressive behavior especially if located in the central nervous system. We present the first case of a patient with histiocytic sarcoma that involved the meninges and had a good course after multidisciplinary treatment.Our patient was a 41-year-old Caucasian woman with no previous history of disease who started with systemic symptoms such as headache and chills. Magnetic resonance imaging with gadolinium contrast of the brain suggested a mass 1.5×2cm in diameter in the temporal lobe with a non-uniform vasogenic edema. This lesion was implanted in the meninges and surgery was the first treatment. The histological findings revealed a histiocytic sarcoma. The patient received concomitant chemoradiotherapy after surgery with good tolerance and currently lives without disease.Although histiocytic sarcomas in the brain present an unusual location and have a poorer prognosis, we have identified the first primary leptomeningeal histiocytic sarcoma with a disease-free survival greater than 3 years following multidisciplinary treatment with surgery and chemotherapy and radiotherapy. Histiocytic sarcoma (HS) is a rare neoplasm showing evidence of histiocytic differentiation. It often appears in the skin, lymph nodes, and intestinal tract but central nervous system (CNS) involvement is rare. The standard treatment for these sarcomas is surgery. The best option for treating these tumors is unclear and affected patients have a poor prognosis despite treatment.In June 2009, a 41-year-old Caucasian woman with no previous history of illness went to the Emergency Room with systemic symptoms, including headache, generalized weakness, and chills. The physical examination showed nothing remarkable. The workup included a computed tomography (CT) brain scan without contrast that showed nothing unusual. The patient received empirical treatment for a diagnosis of a suspected viral infection. A week later, she presented with dysphasia and deviation of the right corner of her mouth. Magnetic resonance imaging (MRI) with gadolinium contrast of the brain suggested a mass 1.5×2cm in diameter in the temporal lobe with a non-uniform vasogenic edema. This lesion was implanted in the meninges and indicated possible meningioma (Figure et al. [HS also formerly known as “true” histiocytic lymphoma is a rare neoplasm showing evidence of histiocytic differentiation. It often appears in the skin, lymph nodes, and intestinal tract . Althouget al.  which apHS has often been classified as being similar to diseases such as malignant histiocytosis, histiocytic lymphoma, and reticular sarcoma, based on morphological criteria . HoweverHS is composed of mononuclear tumor cells with large cytoplasmic vacuoles. Some cells present with phagocytosis phenomena. The diagnosis is based on immunohistochemistry markers of histiocytic cells such as positive CD68, lysozyme, α1-antitrypsin and CD163, which is regarded as a specific HS marker . HoweverRadiological findings have not been established due to the low number of cases. The HS image study reported in the literature addressing the brain is MRI , which dThe standard treatment for these sarcomas is surgery. The best option for treating these tumors is unclear. Furthermore, when the tumor is located in the CNS the therapeutic options are reduced. Brain sarcomas have an aggressive course with an overall survival rate of around 4 to 5 months, regardless of treatment. There are no studies where the benefit of radiotherapy or chemotherapy for HS has been established.In our case, the patient received concomitant chemoradiotherapy after surgery. We decided to administer temozolomide with the radiotherapy because the patient was young and she presented with a sarcoma affecting the CNS. It is also known that this type of chemotherapy crosses the blood–brain barrier and it has shown benefit over radiotherapy alone in other aggressive tumors . She hadIn conclusion, HS is a rare neoplasm with a diagnosis based on immunohistochemistry and biological findings. Although HS in the brain present an unusual location and have a poorer prognosis, we have identified the first primary leptomeningeal HS with a disease-free survival greater than 3 years following treatment with surgery and chemotherapy and radiotherapy.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.EPR and AR analyzed and interpreted the patient data regarding the sarcoma histiocytic disease and the treatment. AS performed the histological examination of the brain, and was a major contributor in writing the manuscript. MD and AMS treated the patient. All authors read and approved the final manuscript.


Melanosis coli (MC) is a benign condition found in 1% of general population. It is associated with high intake of anthranoid laxatives [We herein report two cases of elderly patients with MC in whom this condition masked underlying diseases.18FDG-PET-CT examination, which revealed an advanced disease with secondary lesions to liver and peritoneum. The patient refused endoscopic bowel stenting and an Hartmann procedure was performed. The surgical specimen showed MC before and after the colonic stenosis . The lady underwent ileostomy closure 2 months later and she is being followed-up. No adverse events have been recorded up to 1 year after ileostomy closure.M. D. F., a 70-year-old woman came to our observation with an history of irritable bowel syndrome, diagnosed by General Practitioner (GP). She was used to take laxatives in case of constipation. During the last 2 years she had several episodes of diarrhoea, sometimes with emission of blood, lasting for approximately a week. Her GP prescribed suspension of laxatives and rectal enemas of Mesalazine. She underwent sigmoidoscopy, and MC was found. When we visited the lady she was having hematic liquid stool emission since one week, she was pale and weak. Inflammatory indices were high. We performed a pancolonoscopy: MC was not observed; however, an intense mucosal inflammation suggesting an inflammatory bowel disease was found extending cranially from the sigmoid colon up to the proximal transverse colon. The colon was rigid, and a lesion suspect for dysplasia-associated lesion or mass (DALM) was found in the sigmoid colon . Lesion and random bioptic samples were taken. Stool cultures were negative. In the light of clinical, endoscopic and pathological findings, diagnosis of ulcerative colitis was posed. The biopsies of the lesion concluded for a adenocarcinoma. A Agreement exists about the benignity of MC. This condition is due to a mechanical effect of anthraquinones pigment deposition on colonic mucosa, and physiological appearance is promptly restored after laxative withdrawal within some weeks . Even ifHowever, we are reporting these two cases to highlight the importance not to overlook mucosal alterations which can underlie MC.In the patient described in case 1 the adenocarcinoma was likely to have been present at the time of first endoscopy: random biopsies or repetition of the procedure after laxative withdrawal could have allowed for diagnosis of the cancer at an earlier stage, with better outcomes.Patient described in case 2 also was poorly studied before laxatives and anti-inflammatory enemas administration. As expected, MC had promptly disappeared after laxative suspension. The Mesalazine enemas conferred a falsely sane aspect to the mucosa of the distal part of the colon, but when examining the proximal part of the large bowel a severe mucosal inflammation was found. Also, an neoplastic lesion was found. Even if the diagnostic delay did not impact on surgical completeness and functional outcomes, an earlier diagnosis was desirable and achievable in this patient.MC can be regarded as a benign condition, but one should aim to look at the underlying mucosal aspect when MC occurs. Anthranoid laxatives should be proscribed and patients encouraged to withdraw intake of these drugs.Authors have no competing interests to disclose.GP designed the study, and wrote the draft of the manuscript. GS, SDF and GC collected data and participated in the drafting. SDF, AC, RM and FR collected and analyzed data. SC and FS conceived of the study, participated in its design and coordination and helped to draft the manuscript. FS performed surgical procedure in our Unit as operating surgeon. All authors read and approved the final manuscript.


A 54 year old lady presented for routine excision of a scalp lesion thought clinically to represent a sebaceous cyst of the right occiput. 4 years earlier she underwent total abdominal hysterectomy and right salpingo-oophorectomy for 3 large uterine fibroids. Histo-pathological examination of the hysterectomy specimen revealed an incidental low-grade leiomyosarcoma. Staging imaging was negative for metastatic disease. She made an uneventful recovery and was treated further by adjuvant pelvic radiotherapy.She noticed an uncomfortable and unsightly cystic swelling on her occiput four years after hysterectomy and was referred for routine excision of what was believed to be a benign lesion. The lesion was excised and sent for histopathological examination. Microscopic analysis including immuno-histochemistry demonstrated an ER and PR positive metastatic deposit of leiomyosarcoma. The margins of excision were histologically clear of disease.At Multi-Disciplinary Team (MDT) discussion a diagnosis of metastatic scalp deposit from previous uterine leiomyosarcoma was made. Re-staging CT brain, thorax, abdomen and pelvis and MRI brain were negative for local recurrence or distant metastases. She is currently undergoing radiotherapy to the scalp and surrounding tissues and will be followed up closely by the involved teams.To the best of our knowledge, this is the first case described in the worldwide literature of isolated cutaneous metastasis to the scalp of uterine leiomyosarcoma without evidence of disseminated disease at other sites.http://www.diagnosticpathology.diagnomx.eu/vs/1311834987345566.The virtual slide(s) for this article can be found here:  A 54 year old lady was referred to the surgical day ward of our institution for routine excision of a scalp lesion thought clinically to represent a sebaceous cyst of the right occiput. She had presented 4 years earlier with menorrhagia, fatigue and iron-deficiency anaemia and was diagnosed with a 9 cm fibroid uterus on ultrasound examination after inconclusive diagnostic laparoscopy. She underwent total abdominal hysterectomy and right salpingo-oophorectomy for 3 large uterine fibroids. Histo-pathological examination of the hysterectomy specimen revealed an incidental low-grade leiomyosarcoma Figure . StagingShe noticed an uncomfortable and unsightly cystic swelling on her occiput four years after hysterectomy and was referred for routine excision of what was believed to be a benign lesion. Following injection of local anaesthetic, the lesion was excised and the wound uneventfully closed with absorbable sutures. According to routine practice, the lesion was sent for histopathological examination. The wound healed well. Microscopic analysis including immuno-histochemistry demonstrated an ER and PR positive metastatic deposit of leiomyosarcoma Figures , 6 and 7At Multi-Disciplinary Team (MDT) discussion with input from pathologists, medical oncologists, radiation oncologists and surgeons, including review of the original hysterectomy sides, a confirmed diagnosis of metastatic scalp deposit from previous uterine leiomyosarcoma was made. Re-staging CT brain, thorax, abdomen and pelvis and MRI brain were negative for local recurrence or distant metastases. She is currently undergoing radiotherapy to the scalp and surrounding tissues, and may require salvage adjuvant chemotherapy at a future date if other sites of metastatic disease are detected.Leiomyosarcoma is a rare and unpredictable malignancy of smooth muscle. Soft tissue sarcomas account for 0.7% of all malignancies and leiomyosarcoma accounts for 5-10% of these cases . MetastaThe definition of a cutaneous metastasis is “a neoplastic lesion arising from another neoplasm with which there is no longer continuity” . These lPort site metastasis has been described in colorectal , gallblaUterine leiomyosarcoma (ULMS) is an uncommon malignancy accounting for approximately 1% of uterine cancer  with an Cutaneous metastasis of ULMS is exceptionally rare and there is a paucity of information in the literature.To the best of our knowledge only 4 cases of scalp metastasis of ULMS have been described worldwide -31. The One theory postulated by several authors explaining how a uterine leiomyosarcoma could metastasize to the scalp is haematogenous spread via Batson’s Plexus. This plexus was first described by anatomist Oscar Vivian Batson in 1940 and describes spread of tumour cells through the deep pelvic veins to the internal vertebral venous plexuses. Batson’s vertebral venous plexus consists of four interconnected venous networks surrounding the vertebral column. These valveless, thin walled, low pressure veins bypass the pulmonary, caval and portal venous systems, so this could provide an aberrant pathway of haematogenous spread by tumor in the pelvis to the head and neck -34.The diagnostic value of progesterone receptor (PR) and p53 expression in ULMS is still under investigation. PR expression is useful in classifying leiomyosarcoma (LMS) vs. smooth muscle tumors of uncertain malignant potential (STUMP), Leiomyoma (LM) and Atypical Leiomyoma (ALM). A recent study  publisheRegarding treatment of recurrent or metastatic uterine leiomyosarcoma, surgical resection should be strongly considered in patients with localized single foci recurrences, either local or metastatic. In one study of 41 women who underwent resection for recurrent ULMS , a two-year survival of 71% was reported in those patients who had a disease-free interval of 12 months or longer between resection of the primary tumor and the diagnosis of metastatic disease. Optimal surgical resection may provide an opportunity for long-term survival in these patients .Single agent doxorubicin has been shown to be somewhat successful in combating advanced ULMS. There have been two randomized trails showing the efficacy of doxorubicin single agent chemotherapy vs. combination chemotherapy. Doxorubicin proved as effective as a single agent vs. doxorubicin plus cyclphosphamide as response rates, and median overall survival (median 11.6 vs. 10.9 months) were similar in both arms for patients with measurable disease . In anotPostoperative adjuvant radiotherapy (RT) for advanced uterine leiomyosarcoma has been suggested by the European Organization for Research and Treatment of Cancer to have lower rates of local recurrence but no improvement in long term survival . A retroClinically, patients with cutaneous metastasis of leiomyosarcoma present with cyst like structures underneath the skin as in our case, or as cutaneous nodules . In the Written informed consent was obtained from the patient for publication of this Case Report and accompanying images.The authors declare there were no competing interests and the project was not funded.SC drafted the manuscript. SC, AMH, WK and KB were involved in the surgical management of this patient. KB was responsible for conception and design of the case report. TN and FB made the histo-pathological diagnosis and provided images and slides for publication. FJS provided information related to the patient’s radiotherapy, follow up, and contributed valuable intellectual content. All authors read and approved the final manuscript.


Keratoacanthoma centrifugum marginatum (KCM) is a rare variant of keratoacanthoma (KA). It is characterized by a progressive peripheral expansion and central healing leaving atrophic scar. It is sometimes confused with squamous cell carcinoma (SCC) both clinically and histopathologically. We here report a case of KCM over the extensor aspect of the right forearm in a 57-year-old man with an abnormal looking scar. Keratoacanthoma (KA) is a rare benign epidermal tumour, usually diagnosed clinically, and histopathological examination confirms the diagnosis. Most lesions are classical solitary but few unusual rare variants are also described. Keratoacanthoma centrifugum marginatum (KCM) is a rare variant of solitary KA with a clinical course of centrifugal expansion and central restitution or scarring . The cauA 57-year-old male, a farmer by profession, presented in the dermatology OPD with a large plaque extending from below the right elbow onto the extensor aspect of the forearm. The lesion started as an asymptomatic nodule on a normal looking skin and attained its present state over the past 1 year by peripheral spreading and central healing. Nodular ulcerated margin was seen on the proximal and distal aspects in an arciform manner . The whiKeratoacanthoma is a rapidly growing tumour with a tendency to spontaneous regression but it can recur if incompletely excised and if occurring over fingers, hands, lips, and pinnae . There aWe thought of reporting this case because KAs are rare while KCM with a peculiar looking scar is even rarer and also to highlight the simmering controversy of its relationship with SCC. KCM should be a differential diagnosis in skin lesion with central atypical scaring and peripheral spreading, and it should always be differentiated from SCC by histopathological examination.


A 38-year-old man without any symptoms was admitted to our institution because of an abnormal shadow found incidentally on a chest X-ray. Chest computed tomography showed a round mass in the lingular segment of the left upper lobe. Lingular segmentectomy was performed, and the histopathological diagnosis was intrapulmonary schwannoma. Immunohistochemical staining revealed a positive result for S-100 protein and negative results for CD34 and desmin. We report this case of intrapulmonary schwannoma, which is extremely rare. Schwannoma may arise from any peripheral nerve and is often found in the chest wall and posterior mediastinum. However, case reports of intrapulmonary schwannoma are extremely rare. We report a case of primary intrapulmonary schwannoma.A 38-year-old man was referred to our institution because of an abnormal shadow found incidentally on the chest X-ray. His past medical history was insignificant. The plain chest X-ray showed a mass lesion in the left middle zone. The mass was about 3 cm in diameter. Chest computed tomography (CT) showed a round and homogeneous mass 25 × 18 mm in size with a well-defined margin in the lingular segment of the left upper lobe  of the 10 schwannomas ranged from 1.9 to 7.2, and the variation in the SUVmax could be explained by the variation in cellularity [Today, FDG-PET is a useful approach to differentiate malignant solitary pulmonary nodules from benign nodules. Beaulieu et al. reported the maximum standard uptake values  laser resection have been employed –7. BecauIn conclusion, we had reported a case of intrapulmonary schwannoma, what is extremely rare. The symptoms and CT manifestations are nonspecific, therefore preoperative diagnosis is generally difficult.


Aneurysmal bone cyst is a pseudotumoral lesion. Complete resection prior to selective arterial embolization seems to be the treatment of choice for the more extensive and destructive lesions. In these cases maintaining stability of the cervical spine is critical. This can be very challenging in children and adolescents in whom the axial skeleton is still growing. In this case a young girl presented with a voluminous cervical aneurysmal bone cyst encaging both vertebral arteries and spinal cord. The lesion was treated with aggressive surgical resection, followed by cervical vertebral fusion with instrumentation. After nine months the patient referred no pain and no neurological deficit. MRI scans showed an extensive local recurrence. The family of the young girl refused any other therapy and any other followup. The patients returned to our attention after five years with no pain and neurological deficit. Cervical spine radiographs and MRI scans showed a complete regression of the extensive local recurrence. In the literature, the possibility of spontaneous regression of residual part or local recurrence is reported. The case of this young girl provided the chance to attend a spontaneous regression in an extensive recurrence of aneurismal bone cyst. Aneurysmal bone cysts are benign, highly vascular lesions characterized by expansible blood-filled cystic cavities –3. FirstØ ≈ 5 cm). On admission, physical examination revealed a grade 4/5 muscle strength in internal and external rotation of the shoulder, elbow flexion, and wrist extension on left arm. Left brachioradialis reflex was hypovalid. Hypoesthesia at the distribution of the fifth and sixth left cervical nerve roots was present. There was a positive Modified Spurling's Maneuver on left side.A 13 year-old female with a 2-month history of gradually worsening cervical pain was referred to this institution. She reported progressive weakness on left arm and progressive growth of a mass in left supraclavicular fossa  is a pseudotumoural hyperemic-hemorrhagic lesion of unknown etiology –3. The lCommon clinical signs are pain, often worse at night and with recumbency, and swelling; the duration of symptoms prior to diagnosis is usually 4 to 8 months. Rarely, acute vertebral collapse, with or without spinal cord injury, may be the presenting symptom .The combination of standard radiographs, computed tomography (CT), and magnetic resonance imaging (MRI) provide useful information. The radiographic and CT scans typically reveal a characteristic soap-bubble appearance with fluid-fluid levels, which represents a ballooning, multilocular lytic lesion; this is pathognomonic in many cases .Treatment options include simple curettage with or without bone grafting, complete excision, embolization, radiation therapy, or a combination of these modalities, but local recurrences are described with any type of management , 20. TheThe aim of selective arterial embolization in the treatment of ABCs is to reduce the vascularity of the lesion, as well as to decrease intraoperative blood loss as a preoperative procedure , 10. ManRadiation therapy is limited for these lesions and sometimes associated with poor results , 14. RadEn bloc excision, if correctly planned and performed, is the only treatment option that has a 0% of local recurrence [Although curettage and bone grafting have been reported to be successful in the management of aneurysmal bone cysts in the long bones of the extremities, the same does not apply to lesions of the spine , 16. Comcurrence . Howevercurrence , 14.Spinal reconstruction is particularly important in pediatric patients. Extensive fusion and stabilization with instrumentation have often been avoided in children due to problems of future axial skeleton growth . DespiteIn this case a young girl, presented with a voluminous cervical aneurysmal bone cyst encaging both vertebral arteries and spinal cord, associated with initial neurological symptoms. The osteolytic cervical lesion of C5 and C6 was treated with surgical resection, followed by cervical vertebral fusion and instrumentation. This course of treatment resulted in good removal of deeply involved tumor and successful reconstruction of the spine. After nine months the patient referred no pain and no neurological deficit. MRI scans showed an extensive local recurrence. The family of the young girl refused any other therapy and any other followup. The patient returned to our attention after five years with no pain and neurological deficit. Cervical spine radiographs and MRI scans showed a complete regression of the extensive local recurrence and stability of the implant. In the literature the tendency for spontaneous regression of residual part or local recurrence is reported , 14. The


Pseudomyxoma peritonei is a rare but challenging neoplastic disease which is characterized with intraperitoneal mucinous-gelatinous fluid accumulation. It rarely presents as a mass mimicking abdominal wall hernias A recurrent incisional hernia due to pseudomyxoma peritonei is presented here. A 60-year-old female patient had been operated on for a left mucinous ovarian cyst 20 cm in diameter in 1998. Mucinous material had disseminated into interloop spaces through the right subdiaphragmatic region. Total abdominal hysterectomy + bilateral salpingooophorectomy and peritoneal toilet had been performed. She was rehospitalized for abdominal distention and a 4 cm defect over the incision and underwent a hernia repair using polypropylene mesh in 2001. Abdominal distention recurred to give a rise to an incisional hernia in 2006. She was reoperated for decompression and repair, but nothing could be done because of sticky adhesions and the incision were simply closed. The patient was referred to our department for operation. A prosthetic hernia repair with 30 × 30 cm polypropylene mesh was performed. The patient was discharged on the postoperative 5th day following an uneventful recovery. However, she died of disseminated disease after 18 months. Pseudomyxoma peritonei is a rare but challenging neoplastic disease. It is characterized with intraperitoneal mucinous-gelatinous fluid accumulation. The origin is mostly mucinous cysts of the ovary or appendix vermiformis –3. PseudA 60-year-old female patient had been operated on for a left mucinous ovarian cyst 20 cm in diameter in another center in February 1998. Mucinous material had disseminated into interloop spaces through the right subdiaphragmatic region. Total abdominal hysterectomy + bilateral salpingooophorectomy and peritoneal toilet had been performed. She was rehospitalized for abdominal distention and underwent surgery with diagnosis of pseudomyxoma peritonei in December 2001. A 4 cm defect over the incision was observed, and a hernia repair using polypropylene mesh was performed. Abdominal distention recurred by time and gave a rise to an incisional hernia in Pfannenstiel incision in May 2004. She was operated on again for decompression and repair, but the operating team could not achieve this intent because of sticky adhesions and simply closed the incision. As the hernia became gigantic to limit the patient's daily life within the next 2 years, the patient was referred to our department for operation. Her abdominal girth remarkably enlarged. A very large hernia, mostly located on the left side, existed . MRI datThe prognosis of pseudomyxoma peritonei is poor. Five-year survival rate is less than 50% , 10. RecPatients with pseudomyxoma peritonei usually complain with an increase in abdominal girth . HoweverAbdominal pain is the most common complaint of pseudomyxoma peritonei with an incidence of 23% in the initial evaluation, whereas a newly onset hernia is seen in 12% of the cases . In factPseudomyxoma peritonei is a very rare cause of abdominal wall hernias. The results of surgical interventions are not promising, but remain the only option for the patient with poor quality of life.Z. Ergul, E. Olcucuoglu, and H. Kulacoglu joined the second hernia repair. Z. Ergul, E. Olcucuoglu, and H. Kulacoglu did the literature review. H. Kulacoglu wrote the paper. C. Dener performed the first hernia repair. C. Dener got the imaging studies done.The authors declare no conflict of interests.


Occipital artery aneurysms are very rare vascular lesions. Most cases reported in the literature have been post-traumatic pseudoaneurysms of the occipital artery.We report the case of a 14-year-old Caucasian boy presented with a painless non-pulsatile scalp mass that developed rapidly after minor blunt head trauma. The scalp mass was excised six months after the trauma. A pathologic diagnosis of a thrombosed true aneurysm was made. Our patient has had no recurrence of the mass at 15 months follow-up.We present a case of a true aneurysm of the occipital artery following minor head trauma. We review the literature for similar cases and discuss the difficulty of establishing a diagnosis prior to surgical intervention. Aneurysms of the external carotid circulation are rare. Of these aneurysms, scalp aneurysms involving the occipital artery are the rarest. They have been described primarily as a consequence of blunt, penetrating or iatrogenic trauma. There are also cases secondary to infection and autoimmune disease, and cases of spontaneous aneurysms of idiopathic etiology.Pathologic examination of these lesions reveals that the majority are pseudoaneurysms, the dilated walls of which are comprised of only the outer layers of the native vessel wall. These lesions usually present as pulsatile scalp masses and are often painless.Only 10 cases of traumatic and spontaneous aneurysm of the occipital artery have been previously reported in the English literature, with sporadic cases in other languages. Of these previously reported cases, only one was confirmed to be a true aneurysm of the occipital artery, comprising all layers of the arterial wall. We report another case of a true aneurysm of the occipital artery in a pediatric patient.A 14-year-old Caucasian boy presented to our Pediatric Neurosurgery clinic for evaluation of a scalp mass. Our patient was a previously healthy child who had a basketball strike to the back of his head four months prior to presentation. Our patient did not have loss of consciousness and had immediate scalp swelling near the impact site. While initially painful, the pain subsided over the next two days, but a firm mass persisted for months after the incident. He was then sent to our Pediatric Neurosurgery clinic for evaluation of the persistent mass.On examination, our patient was found to have a 2.5cm firm, non-tender, non-pulsatile, mobile mass in the left posterior temporal area approximately 6.5cm superior to the mastoid tip and 4.5cm lateral to the external occipital protuberance. The mass was not bothersome to our patient other than it being unsightly. Upon palpation, no palpable thrill was present, and we did not auscultate the lesion. A skull X-ray showed no bony abnormalities. A magnetic resonance imaging scan showing heterogeneity within the lesion suggested partial thrombosis of the scalp mass or evolving hematoma of differing ages Figure . Due to Our patient was brought to the operating room, and a curvilinear skin incision was made superior to the mass. The firm, smooth, non-pulsatile mass was encountered just under the galea aponeurotica. It was circumferentially dissected and associated with brisk arterial bleeding from vessels proximal and distal to the lesion. We had not anticipated an aneurysm and, therefore, encountered significant bleeding until the proximal and distal arteries were coagulated. The bleeding was easily controlled with bipolar electrocautery and division of the proximal and distal artery. The mass was excised en bloc and sent for pathologic examination. However, we did not section the mass on the operating table to look for intraluminal thrombosis. Our patient tolerated the procedure well and was sent home later the same day.Gross examination of the specimen demonstrated a smooth, fluctuant, intact cyst Figure . SectionOur patient returned to our Pediatric Neurosurgery clinic for follow-up, and at 15 months has had no recurrence of this mass.Aneurysms of the terminal branches of the external carotid artery are rare. They are generally the results of blunt, penetrating or iatrogenic trauma but can also be associated with infections. Traumatic aneurysms usually develop two to six weeks after blunt head trauma. Pseudoaneurysms are more common in the scalp and do not involve all layers of the arterial wall. True aneurysms, however, involve all three vessel layers - the intima, media and adventitia - and represent a localized or diffuse dilatation of the vessel wall.The occipital artery has three segments. From proximal-to-distal-most, they are the digastric, suboccipital and subgaleal segments. At the level of the superior nuchal line, the suboccipital segment of the artery crosses the sagittal plane, intersecting the midpoint of the lambdoid suture on the ipsilateral side . Here, tThere have only been 10 previous reports of traumatic and spontaneous aneurysm of the occipital artery (Table This disorder may not be readily diagnosed based on history and physical examination, especially if the scalp mass is a thrombosed, non-pulsatile vascular lesion. A high degree of clinical suspicion is required to recognize an occipital artery aneurysm or pseudoaneurysm. Differential diagnosis of the occipital scalp mass should include dermoid or epidermoid cyst, eosinophilic granuloma, hematoma, abscess, aneurysm, arteriovenous fistula, encephalocele, lymphoid hyperplasia and sinus pericranii . While, If clinically suspected, other diagnostic tools may be essential for correct diagnosis. Duplex ultrasound shows fusiform dilation and turbulent intraluminal arterial flow in non-thrombosed aneurysms . ComputeAlthough the natural history of this rare lesion is unknown, indications in the treatment of occipital artery aneurysm have included reduced risk of hemorrhage, pain relief and, in most cases, the alleviation of cosmetic disfigurement. In our case, the indication for surgery was for definitive pathologic diagnosis of the scalp mass and cosmesis. Treatment options for occipital aneurysms include simple resection, proximal ligation of the parent artery, trapping of the aneurysm, percutaneous ultrasound-guided thrombosis of the lesion, and endovascular arterial embolization or coil occlusion ,15.Aneurysms of the occipital artery are rare, and the majority of them are pseudoaneurysms. When true aneurysms do occur, they present as painless swelling. Neurological complications are exceedingly rare. The diagnosis may be made preoperatively by careful physical examination if the clinical suspicion is high. In other instances, however, the diagnosis of an aneurysm is not made until pathologic review after surgical resection of the scalp mass. Preoperative diagnosis is especially difficult in the case of thrombosed aneurysms. Surgical resection is generally curative. Care should be taken in very young children due to a potential for blood loss during surgery.Written informed consent was obtained from the patient’s parents for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.VYR and AJ drafted the manuscript for important intellectual content. VYR, SWH, AMA and AJ made substantial editorial revisions to the manuscript. AJ made major contributions to conception and design. All authors read and approved the final manuscript.


Multiple primary malignancies are not rare. While metachronous triple cancers are rare and a triple tumor case involving maxillary sinus and tympanic membrane is exceptionally rare. We present such an extremely rare case with the index tumor of adenoid cystic carcinoma of the maxillary sinus and 14 years later esophageal cancer was observed as a metachronous tumor. One year after esophageal cancer, squamous cell carcinoma arising from tympanic membrane was detected. Before the tumor of tympanic membrane was observed, the patient had received total three radiation courses. Prior radiation therapy is suspected to be playing a role in inducing the squamous cell carcinoma of the tympanic membrane. Multiple primary malignancies are not rare. However a case involving the tympanic membrane is very rare. We present such an extremely rare triple primary tumor case with the index tumor of adenoid cystic carcinoma (ACC) of maxillary sinus and two metachronous tumors of squamous cell carcinomas (SCC) of tympanic membrane and esophagus. A 67-year-old man presented at our Department in September 2008 with a history of purulent ear drainage for three months. Examination revealed a tumor mass arising from tympanic membrane in the right external auditory canal. A computed tomography (CT) examination showed the tympanic cavity and the bone of the external auditory canal were not involved . The his During the postoperative course, no signs of either local tumor recurrence or distant metastases were found on review at twenty-two months. However, it is the second metachronous tumor in the patient. In August 2007, the first metachronous tumor in the patient was confirmed. Because of dysphagia, barium esophagogram was performed and neoplasm located in the thoracic esophagus was detected. Gastroscopic examination was then performed and tumor mass was found. A diagnosis of SCC was confirmed by biopsy. For treatment, the patient received 60 Gy of radiation alone and the tumor completely regressed. During 18 months follow-up, he remained free of symptoms and complaints, and no recurrence or distant metastases were detected. The index tumor was ACC of the maxillary sinus. At the age of 52, total maxillectomy was performed for the diagnosis of ACC of the right maxillary sinus. The patient then received postoperative radiation therapy at the dose of 66 Gy. Six months later, local recurrence was found and the patient underwent 65 Gy radiation treatment. After that, during the 15-year follow-up period, no recurrence or metastases were observed.1 The maxillary sinus is the most common site.2,3 Because of late detection owing to sinusitis-like symptoms and relative surgical inaccessibility owing to important adjacent structures, maxillary ACC is recognized to have a unfavorable prognosis.2,3 Many studies have indicated that surgery with postoperative adjuvant radiation therapy is the main strategy for sinonasal ACC.2-4 However, patients who were treated with surgery and postoperative radiation had a high recurrence rate of 65%.2 In our case, local recurrence six months after initial surgery and radiation treatment was observed. This may be due to perineural invasion in ACC.2,5 Most primary malignant tumors arising in the paranasal sinus are SCC. ACC is the second most common malignancy and accounts for 5% to 15% of all paranasal sinus cancers. Unlike other malignancies in head and neck, to our knowledge, multiple primary malignancies with sinonasal ACC have not been reported before, which may be partly owing to the low incidence of sinonasal ACC. In this patient, maxillary ACC was the index tumor and 14 years later esophageal cancer (EC) was observed as a metachronous tumor. One year after EC, SCC arising from tympanic membrane was detected.6 In our patient, total three radiation courses were performed. It may be a risk factor for malignant transformation. SCC of the tympanic membrane is extremely rare and appearing in the patient with sinonasal ACC has not to our knowledge been reported previously. As the pathological examination indicated malignant change of the papilloma and clinical examination showed no SCC existed elsewhere, we believed that this tumor is independent of the others. However, it may be partly attributed to prior radiation. The tympanic membrane is in the maxillary radiotherapy field in this patient. And radiation therapy is regard as a risk factor of SCC of this site.7 The outcome is related to the stage of the tumor. Local recurrence and distant metastasis were 16% in patients with SCC of middle ear.7 The therapeutic strategy for these patients is a complete removal of the tumor with free margins followed by postoperative adjuvant radiation.8,9 However, in Lobo D’s study, significant differences have not been found between the patients treated with surgery and radiation and with surgery alone.7 In our case, the patient received surgery alone and the surgical margins were free. No local recurrence or distant metastases were detected during twenty-two months of follow-up. SCC has a more aggressive behavior and a worse prognosis than other tumors of the auditory canal and middle ear. In conclusion, we have presented an extremely rare case of multiple primary malignancies involving the tympanic membrane. Prior radiation therapy may play a role in inducing the SCC of the tympanic membrane. Our recent experience in treating such a patient seems important because it provided us with an opportunity to record this unusual case of multiple primary malignancies.


Acute epidural hematoma is a critical emergency all around the world, and its aggressive diagnosis and treatment are of vital importance. Emergent surgical evacuation of the hematoma is known as standard management; however, conservative procedures are also used for small ones. Spontaneous rapid resolution of these hematomas has also been reported in eight pediatric cases. Various theories have been proposed to explain the underlying pathophysiology of this resolution. Herein, we are reporting a new pediatric case with spontaneously resolving acute epidural hematoma 12 hours after admission to the emergency room.  Acute epidural hematoma is a serious complication of head injury; rapid diagnosis and early surgical evacuation of it are considered as the standard management , 2. HoweHerein, we are presenting an acute epidural hematoma case which resolved 12 hours after admitting to emergency department in a 4-year-old child and we are discussing the underlying mechanisms for our case in the light of the literature.Four-year-old boy was evaluated in the emergency department about one hour after a fall from a height of about two meters. His history was unremarkable except for mild headache. In the physical examination, there was edema, abrasion, and erythema on the right temporoparietal region. Neurological examination was normal. Computed tomography of the patient in the emergency department revealed acute epidural hematoma ; maximalTwelve hours after hospitalization, we took computed tomography for controlling the size of the hematoma. We saw complete resolution of it . PatientAlthough surgical evacuation is a settled opinion in the management of epidural hematomas, the opinion of conservative followup has made its ground in bonds in the literature. The main reasons of this may be summarized as increased presence of intensive care units more than before so the easy close followup of the patients with head injury, presence of neurosurgeon staff in many health centers during 24 hours, and easy access to computed tomography which is essential in the diagnosis and followup of such patients. Additionally, increased report of rapid spontaneous resolution of acute epidural hematoma cases after the first report in 1981 may also support the opinion of conservative followup .Rapid spontaneous resolution of epidural hematoma has been reported in children as well as in adults. The literature research reveals 8 pediatric cases excluding our case . HoweverResolution without fracture may be due to open cranial sutures. We consider that open sutures also cause a pressure gradient in a similar way as with fracture.The second probable mechanism in spontaneous resolution is the pressure-induced redistribution secondary to brain swelling; however dissipation of hematoma seems harder due to tenacious adhesions between dura matter and skull , 11, 12.3 with less than 15 mm thickness and with less than 5 mm midline shift in patients with a Glasgow Coma Score greater than 8 without focal deficit could be managed nonoperatively with serial computed tomographic scanning and close observation in a neurosurgical center [3, midline shift was less than 5 mm, and GCS was 15. We decided to manage the patient conservatively with serial CT. In our case, we detected the resolution after 12 hours. If we took the computed tomography sooner, perhaps we would see the resolution earlier. Önal et al. saw the resolution just after one hour, because of the deterioration of the general status of their patient. They retook CT and saw the resolution of acute epidural hematoma. Deterioration was due to hepatic artery injury in their patient [When we look at the resolution times in pediatric cases, the longest time was 72 hours , and thel center . In our  patient . We did The underlying pathophysiology of rapid spontaneous resolution of acute epidural hematomas has not yet been fully clarified, despite being more frequently reported cases, especially in children. We think that whether the skull contents fully fill the skull may be important in the pathophysiology of spontaneous rapid resolution. We believe that future experimental studies will shed light on our way.