Simple 46, XY gonadal dysgenesis syndrome, also called Swyer syndrome, is known as pure gonadal dysgenesis. Individuals with the syndrome are characterized by 46, XY karyotype and phenotypically female with female genital appearance, normal Müllerian structures and absent testicular tissue. The condition usually first becomes apparent in adolescence with delayed puberty and primary amenorrhea due to the gonads have no hormonal or reproductive potential. Herein, we report a case of dysgerminoma diagnosed in a dysgenetic gonad of a 21-year-old patient with Swyer syndrome. Doctor Swyer described two women whom had a 46, XY karyotype, tall stature, primary amenorrhea, female external genitalia, normal vagina  and cervix in 1955 [A 21 years old patient with a history of primary amenorrhea, 46, XY karyotype, was admitted to the hospital complainging of a low abdominal mass. The patient is 164 cm tall. She has breast development (tanner IV). The pubic hair distribution is adult in type but decrease in total quantity. Her menstruation had not started until she took estradiol valerate and medroxyprogesterone for 15 months, which was prescribed by doctor in her 17 years old. In 19 years old, menstruation started, 6 days at intervals of 23-40 days, blood is ruby-red, sometimes blood clot. However, menstruation disappeared at the age of 20 because she stopped the pills. Chromosome analysis with fluorescence in situ hybridization revealed 46, XY. Color doppler imaging (CDI) showed the uterus was 3.22 × 5.19 × 2.80 cm, thickness of the endometrium was 0.19 cm thick. There was a heterogenetic mixed echo (5.50 × 3.77 cm) behind the cervix. Laboratory investigations showed CA125 was 11.53 U/ml , testosterone free (F-TEST) was 34.5 pmol/L , FSH was 56.70 mIU/ml, LH was 21.50 mIU/ml, sex hormone binding globulin (SHBG) was 16.50 nmol/L  and carcino-embryonic antigen (CEA) was 0.55 ng/ml . Exploratory laparotomy was performed. Surgical findings: The uterus was small (3.0 × 5.0 × 2.0 cm). A 5.0 × 5.0 × 4.0 cm firm mass involved the right gonad with a smooth surface and good mobility. The left gonad was aplasia, like streak and without follicle. The bilateral fallopian tubes were slender. The serosa of the liver, the spleen and the stomach were smooth. There was no obvious nodule in the omentum. The result of the frozen section is malignant tumor of the ovary. Based on the agreement with the patient and her family bilateral salpingoopherectomy and hysterectomy (BSOH) and lymph node dissection were performed. Due to tumor was classified as stageIA, adjuvant therapy was not commented. Written consent for publication was obtained from the patient.A 5.0 × 5.0 × 4.0 cm firm mass involved the right gonad, while the left gonad appeared as streak gonad measuring 3.0 × 0.6 × 0.6 cm. The tumor was encapsulated with a smooth surface. The cut surface of the tumor was solid, fleshy, lobulated and gray-yellow Figure . The bilThe tumor and the other abscised tissues were fixed in 10% formalin and embedded in paraffin. Several 4-μm sections were cut from each paraffin block, and one was stained with HE (hematoxylin and eosin), the others were stained with IHC (immunohistochemistry). Immunohistochemical staining was performed using the streptavidin-peroxidase system  according to the manufacturer's instruction. Commercially available prediluted monoclonal antibodies against the following antigens were employed: CK(cell keratin), CD30, PLAP , CD117 .Microscopically, the tumor in the right dysgentic gonad showed the typical features of dysgerminoma. The tumor cells grouped themselves in well-defined nests separated by fibrous strands infiltrated by lymphocytes. The tumor cell membrane was prominent. The uniform, rounded tumor cells have clear to eosinophilic cytoplasm, and a central, large, rounded or flattened nucleus that contains one or a few prominent nucleoli. Calcification was seen Figure . No metaThe squamous cells of the cervix was normal, and lymphocytes infiltrated was seen in the cervix tissue. Figure . The endThe Swyer syndrome, 46, XY gonadal dysgenesis, belongs to the category of sexual abnormality . The synSwyer syndrome should be differentiated from the following two female phenotype (karyotype XY) syndrome. One is the familial syndrome of testicular feminization, the most common type of male pseudohermaphroditism. It occurs in individuals with a normal male chromosome constitution with an end-organ defect (androgen insensibility). It characterized by the presence of several well-developed female secondary sex characteristics. Individuals are found to have a vagina , bilateral cryptorchid testes but no uterus ,5. The oIn patients with gonadal dysgenesis, either "pure" or associated with the somatic features of Turner's syndrome , both gonads are represented by a streak of fibrous tissue that vaguely resembles ovarian stroma ,9. PreviIn this report, we provide a case of dysgerminoma diagnosed in a dysgenetic gonad of a 21-year-old patient with Swyer syndrome, who presented with primary amenorrhea and infertility of five years duration. Karyotype was consistent with 46, XY (pure). In our case, testosterone free was 34.5 pmol/L, sex hormone binding globulin was 16.50 nmol/L, and menstrual function was associated with estradiol valerate and medroxyprogesterone prescribed by doctor. Estrogen and progestin sequential therapy supports female secondary sex development in patients with gonadal dysgenesis. The patient did not have a bilateral gonadectomy after knowing she has a 46, XY caryotype because she wanted to be pregnant and did not believe she had the risk of suffering from malignent of ovary. It is necessary to take the familial screening of Swyer syndrome cases. As a malignant germ cell tumor of the ovary, dysgerminoma can be found either in a pure form or mixed with other germinal elements. Therefore in premenarchal patients with a pelvic mass, the karyotype should be determined.Differential diagnosis for dysgerminoma include diffuse large B cell lymphoma, poorly differentiated carcinoma, embryonal carcinoma and gonadoblastoma. Coexistence of dysgerminoma and gonadoblastoma is seen in about 50% of cases . HistoloAbout 65% of dysgerminomas are stage I at diagnosis. About 85 ~ 90% of stage I tumors are confined to one ovary; 10 ~ 15% are bilateral. Dysgerminoma is the only germ cell malignancy that has this significant rate of bilaterality, other germ cell tumors being rarely bilateral. The treatment of patient with early disgerminoma is resection of the primary lesion and proper surgical staging. Chemotherapy and/or radiotherapy are administered to patients with metastases. In patients whose contralateral ovary has been preserved, some disease can develop in 5 ~ 10% of the retained gonads over the next 2 years [The authors declare that they have no competing interests.YH analyzed the data and wrote the manuscript as a major contributor. YW, QL and SD contributed to management of the patient. AH and EW carried out the histopathological evaluation and helped to write manuscript. All authors have read and approved the final manuscript.


Cystic meningioma is an uncommon meningioma variant that is often difficult to distinguish from other intra-axial tumors, including necrotic gliomas. Cystic meningiomas located in the ventricle are particularly rare and may be misdiagnosed with other brain tumors, including ependymoma, choroid plexus papilloma and neurocytoma, due to its location. The present study discusses two cases of lateral ventricular meningiomas, which exhibited intratumoral or peritumoral cystic changes on magnetic resonance imaging scans. The two patients underwent surgical treatment and histological examination confirmed one case of metaplastic meningioma and the other case of psammomatous meningioma. The two patients were middle-aged females and had been misdiagnosed prior to surgery. Although this clinical entity is rare, the diagnosis of meningioma should be considered, particularly in middle-aged female patients. Intraventricular meningiomas are rare tumors that account for 0.5–3% of all intracranial meningiomas , howeverA 48-year-old female presented with a chronic headache and vertigo that had persisted over a period of 2 months, but had progressively worsened in the last 10 days. Neurological examination, laboratory tests and other systemic evaluations  revealed no abnormalities, with the exception of hypertension.MRI scans revealed a 4.5×5.0×4.0 cm heterogeneously irregular mass located in the trigone of the right lateral ventricle. An intratumoral cystic change was also observed. The tumoral parenchyma was hyperintense on T2-weighted images (WIs) and fluid attenuated inversion-recovery images, while appearing iso- to hypointense on T1WIs . Patchy Surgery via temporal-occipital craniotomy revealed that the tumor was an ivory-colored hard mass which was well-circumscribed in the right trigone of the lateral ventricle. Evident calcification was observed without a visible blood supply. Pathological examination confirmed the mass to be a metaplastic meningioma [World Health Organization (WHO) grade I; A 43-year-old female presented with a persistent headache and blurred vision which has lasted for longer than one month. Neurological examination revealed papilla edematous and increased intracranial pressure.MRI scans revealed a heterogeneously cystic-solid mass of 4.8×5.0×5.5 cm in the right lateral ventricle. The solid area of the tumor exhibited an iso- to hypointense signal on T2WIs and an isointense signal on T1WIs and diffusion weighted imaging. On an apparent diffusion coefficient map, the parenchyma of the tumor exhibited an isointense signal. Overt enhancement was observed on T1-weighted post-gadolinium contrast images in the solid area instead of the cystic component . The tumSurgery via a right temporoparietal craniotomy revealed that the tumor was a well-defined, yellowish in color, intact and encapsulated mass that adhered slightly to the walls of the lateral ventricle and the septum pellucidum. The marginal area of the tumor was cystic and the root originated from the choroid plexus. Pathological examination confirmed the mass to be a psammomatous meningioma  arise within the brain parenchyma of the cerebral hemispheres rather than from the ependymal cells lining the ventricular surfaces . In addiNotably, the current two cases had histologically different subtypes, which may be associated with their various MRI manifestations. Case 1 is a metaplastic meningioma, characterized by mesenchymal elements, including osseous, cartilaginous and myxoid tissue, which may lead to calcification or ossification and cystic formation as exhibited in the MRI scans. A previous case report  regardinIn conclusion, to the best of our knowledge, this is the first case report of cystic meningioma localized in the lateral ventricle in adult females. Although cystic meningioma located in the lateral ventricle is rare, this clinical entity should be included in the differential diagnosis of neoplasms in the lateral ventricle, particularly in adult female patients.


Osteosarcoma is the most common primary malignant bone tumor. The distal radius is a relatively common skeletal site for primary bone tumors, but not for osteosarcoma. It is difficult to treat Osteosarcoma of the distal radius; however, skeletal reconstruction and functional restoration following en bloc resection may be a promising technique. This report presents a 17-year-old male with osteosarcoma of the distal radius that was treated by en bloc resection and reconstruction using a fibular shaft to preserve the radiocarpal joint. After six months, radiographs revealed that the grafted fibular bone had healed well with the host bone. Physical examination demonstrated that active dorsiflexion of the affected wrist was to 90° and palmer flexion was to 45°. Fourteen months after surgery there was no evidence of wrist deformity, instability, metastasis or local recurrence. Therefore, this technique preserved the important structures and the joint surface for wrist stability and effective function. Osteosarcoma is the most common primary malignant bone tumor of obscure origin, which generally presents in the second decade of life . The dis2 skin), adriamycin (90 mg/m2 of skin) and ifosfamide (10 g/m2 of skin), pain diminished, the local mass decreased and became rigid, and the range of motion of the affected wrist returned to normal. The sclerotic changes and a good margin of the lesion were observed on plain radiographs  revealed diffuse swelling, mild tenderness and local heat at the distal end of the right radius. A hard mass with an irregular surface  was palpated in the dorsal aspect of the distal radius. Wrist motion was slightly restricted . Radiographs showed diffuse osteosclerosis and focal osteolysis with periosteal reaction in the distal radius metaphysis. Magnetic resonance imaging (MRI) revealed a hypointense lesion on T1-weighted images and a hyperintense lesion on T2-weighted images with soft tissue extension. No epiphyseal involvement was identified on the MRI . Technetiographs , and MRIs (et al , the tumThe patient underwent en bloc resection of the tumor and reconstruction with a free fibular shaft to preserve the radiocarpal joint. A longitudinal dorsal incision at the radiocarpal joint was used to approach the distal radius and an elliptical excision was made at the needle biopsy site. The extensor tendons were removed and preserved, and the flexor tendons were preserved. A 13×3-cm osteotomy was performed proximal to the radial styloid followed by en bloc resection of the distal radial osteosarcoma . The ulnVarious reconstructive procedures following the excision of malignant tumors in long bones have been reported, including prosthetic replacement, allografts, vascularized fibular grafts, autoclaved bone grafts and reimplantation of autologous inactivated bone –10. Geneet al (et al (The distal radius is a relatively common skeletal site for primary bone tumors, however, not for osteosarcomas; it has been reported that <1% of osteosarcomas arise in the distal radius . Previouet al  reportedl (et al  investigFor tumors located in the metaphysis or in contact with the epiphyseal line, limb salvage surgery, to preserve epiphysis or the native joint, is required . When joIn conclusion, the present report demonstrated that en bloc resection of a tumor and reconstruction with a free fibular shaft to preserve the radiocarpal articulation appears to be a promising procedure to treat osteosarcoma of the distal radius, with no evidence of epiphyseal invasion following effective preoperative chemotherapy. This technique preserved the important structures and joint surfaces to maintain wrist stability and effective function. However, the long-term outcomes of this technique require further investigation.


The authors report the case of 46-year-old man with recurrent hemoptysis. Bronchoscopy revealed a submucosal tumor protruding into the tracheal lumen. Transbronchial biopsy failed to obtain a conclusive diagnosis; only surgery allowed complete resection of the tumor and confirmed the diagnosis of tracheal mucoepidermoid carcinoma. We discuss the unusual submucosal presentation of this tumor, and the contribution of surgery for diagnosis and therapy. Tracheal mucoepidermoid carcinoma (MEC) is a rare airway tumor and represents, along with the adenoid cystic carcinoma (ACC), one of the two most common types of primary salivary-type tracheal tumors. These tumors are indistinguishable histologically from their salivary gland counterparts, and it is believed that they originate from the submucosal glands of the tracheobronchial tree and probably are related to structural homology between exocrine glands. MEC can be divided into low grade and high grade on the basis of histological criteria. The most important factors in the prognosis include histological grading and the ability to achieve a complete surgical resection .We report a case of a 46-year-old, previously healthy man, with a submucosal tracheal tumor, in whom transbronchial biopsies were inconclusive; only surgery allowed removal of the tumor and confirmed the diagnosis of tracheal MEC. To our knowledge, such a presentation of this tumor has not been previously reported.A 46-year-old male smoker presented with a 2-year history of hemoptysis. On admission, routine laboratory studies including pulmonary function tests were normal.The chest radiograph was normal. Computed tomography (CT) revealed a 4 × 4 × 3 cm circular mass in the lower third of the trachea, including the carina and repulsing the arch of azygous vein Figure . There wA flexible bronchoscopy showed a normal tracheal lumen, with bulging and erythematous mucosa, on the right posterolateral wall above the carina Figure . TransbrThrough a right posterolateral thoracotomy Figure , the azyThe postoperative course was uncomplicated; chest drain was removed on the fourth day following surgery. The final pathologic examination confirmed the diagnosis of primary low-grade MEC originating broadly from the trachea, without the involvement of the surgical margins. No evidence of metastatic MEC was identified in the lymph nodes that were excised. Follow-up 1 year after surgery is normal and the patient is today tumor free.Tracheal MEC arises from the submucosal glands of the tracheobronchial tree and probably is related to structure homology between exocrine glands. It is a rare airway tumor, representing 0.2% of all respiratory tumors and 1 to 5% of ‘bronchial adenomas’ , a term that was used to describe a group of slow-growing neoplasms thought to arise from the bronchial glands, and included ACC, MEC, mixed tumors and carcinoid tumors [Histologically, MEC of the tracheobronchial tree consists of variable proportions of mucus secreting cells, squamous cells, and so-called intermediate cells that show no particular differentiating characteristics . The tumGenerally, these tumors produce symptoms of upper respiratory tract obstruction, such as cough, dyspnea, hemoptysis, wheezing, atelectasis or postobstructive pneumonia, and it frequently is mistaken for asthma or chronic pulmonary obstructive disease, with no or partial response to treatment. When the symptoms are not resolved, a more scrupulous workup is usually performed and intratracheal tumors diagnosed.Imaging studies usually precede bronchoscopy, unless the patient arrives in respiratory distress. Improvements in technology now allow CT scanning to provide various images for airway visualization. As far as diseases involving the central airways are concerned, the transverse extent of disease and its relationship to adjacent structures are better shown on the usual transverse CT sections, but the longitudinal extent of the tumor is better demonstrated on the multiplanar reconstruction and three-dimensional images . In the Every patient suspected of having a tracheal tumor should undergo bronchoscopy with biopsy, which remains the main diagnostic modality in nonemergency cases. MEC often presents as a sessile tracheal tumor.In the present case, the MEC had an unusual submucosal presentation, explaining the negative bronchial biopsies and hence the need for surgery for therapeutic and diagnostic purposes. This surgery requires an accurate preoperative assessment, including a bronchoscopy and chest CT with multiplanar tracheal reconstructions. The usual recommendations of tracheal surgery must be respected, from the induction of anesthesia until the resection-anastomosis, without omitting the intraoperative ventilation technique. A biopsy with frozen section examination must be performed because the operative strategy would be affected by the results.The clinical course of these tumors correlates with the histological grade of the tumor. Patients with high-grade lesions have been reported to have a poor prognosis; the 5-year survival rate is 25 to 31% . Low-graComplete resection of the tumor on-bloc with the tracheal rings combined with reconstruction of the trachea is the mainstay of treatment. The role of radiotherapy and chemotherapy, before or after surgery, has yet to be well established .In summary, a tracheal MEC may be submucosal, and the transbronchial biopsy may remain negative. In this case, surgical excision for diagnostic and therapeutic purposes is possible, after performing CT with multiplanar reconstructions.Written informed consent was obtained from the patient for the study and publication of this case report and accompanying images. A copy of written consent is available for review from the Editor-in-Chief.ACC: Adenoid cystic carcinoma; CT: Computed tomography; MEC: Mucoepidermoid carcinoma.The authors declare that they have no competing interests.SM collected information and prepared the original draft. LM and GJ researched the relevant literature and revised the draft. OY helped with the literature research and preparing the manuscript. SM helped prepare the manuscript. All authors read and approved the final manuscript.


Synovial sarcoma is a malignant mesenchymal neoplasm that is frequently misdiagnosed as a benign condition because of its small size, slow growth, and well-delineated appearance. Rapid spread and early death occur rarely. Here we report a case of synovial sarcoma of the buttocks presenting with a non-healing wound and rapid progression after local resection in a 23-year-old woman. She initially found a slightly painful subcutaneous mass in the left buttock and underwent local excision. Postoperatively, she developed a non-healing wound that did not respond to conventional antibiotic therapy and local wound care, and pitting edema of the lower extremities. A magnetic resonance imaging scan revealed a large heterogeneous, irregular mass in the buttocks with regional lymph node involvement. Histological and immunohistochemical analyses suggested the diagnosis of a poorly differentiated synovial sarcoma. Her condition deteriorated dramatically shortly thereafter; she developed systemic edema and died of respiratory failure. This case suggests that synovial sarcoma may be fatal within months of recognition if improperly managed and stresses the importance of adequate pre-surgical evaluation and postoperative pathological analysis in the management of a subcutaneous mass. Synovial sarcoma is a malignant mesenchymal neoplasm that constitutes 8 to 10% of all sarcomas and has an annual incidence of 2.5 per 100,000. It mainly affects adults in the third to fifth decades of life . Rapid sth, 2011, the patient was referred to another hospital, where she underwent surgical debridement. Postoperatively, the wound deteriorated rapidly and developed a cauliflower-like growth. Meanwhile, she developed pitting edema of the lower extremities. Her general condition worsened.A 23-year-old woman was admitted to our hospital due to the development of a non-healing postoperative wound for approximately 4 months. In January 2011, she found a slightly painful subcutaneous mass that measured 1 × 1.5 cm in the left buttock and underwent local excision at a local hospital. On postoperative day 2, she developed bleeding from the wound and received hemostatic interventions. However, the wound remained unhealed and continued to produce purulent exudates, with no obvious improvement after repeated dressing changes. On March 26st, 2011, she was referred to our hospital. At the time of presentation, her general condition was poor and she had severe pallor. Her body posture was constrained and she could only sleep on the right side. There was obvious pitting edema of the bilateral lower extremities  and the lower abdomen. A T-shaped incision was noted on her left buttock. The cauliflower-like lesion measured 15 × 20 cm and was sufficiently deep to reach the ischial tuberosity, with a large amount of purulent discharge  scan was performed, which revealed a large heterogeneous, irregular mass in the buttocks, buttock ulceration, and multiple enlarged pelvic and inguinal lymph nodes that were fused together Figure . An initAs the patient developed severe anemia, hypoproteinemia and severe edema, she was treated with a blood transfusion, fluid infusion, supplementation with albumin and plasma, and regular dressing changes to maintain stable vital signs. However, her condition deteriorated dramatically and she rapidly developed systemic edema. As a result, she developed dyspnea, anorexia and anuria (<100 mL/d). The patient died of respiratory failure within a week after admission.Synovial sarcoma is the third most common histological type of soft tissue sarcoma of the extremities, which mainly affects adolescents and young adults . It is sSynovial sarcoma often has no specific clinical presentations that distinguish it from other sarcomas. Typically, it presents as a slowly enlarging, deep-seated mass, which is sometimes slightly painful. It is often misdiagnosed as a benign lesion, and simple excision is performed without adequate pre-surgical evaluation. In particular, as the majority of subcutaneous soft-tissue lesions are benign or inflammatory, simple excision is often performed without a biopsy by physicians and surgeons who do not have specific training in oncology . As a reAlthough synovial sarcomas are a high-grade soft tissue malignancy, most are characterized by slow growth. The mass may be present for an extended period before presentation and rapid spread and early deaths are seen only occasionally. In the present case, the disease progressed rapidly and the patient died within 4 months of symptom recognition. Given the small tumor size at presentation and the subsequent rapid disease progression, we surmise that local invasion and lymph node metastasis was likely to have occurred before the initial local excision of the mass. Therefore, a wide excision plus adjuvant radiotherapy and/or chemotherapy could have been performed. In addition, for the local excision of a subcutaneous tumor, violation of the underlying fascia may facilitate tumor local invasion, as the fascia provides an excellent deep barrier to tumor spread . OverallThe development of a non-healing wound that does not respond to conventional antibiotic therapy and local wound care is another interesting point of our case. Although cancer has been identified to be a common cause of non-healing wounds , there hMRI is the preferred imaging modality for the evaluation of soft tissue sarcomas because of its excellent tissue contrast and ability to depict the lesion in multiple planes. It is not only useful for evaluating the extent of the tumor and its involvement of adjacent soft-tissue structures, but also for differentiating tumor from muscle tissue and depicting the involvement of neurovascular structures, tendons, fascial/fat planes and bone marrow. On MR images, lesion margins of synovial sarcomas are well defined in 53 to 91% of cases and poorly defined in 9 to 47% . In our Synovial sarcoma has three major histological subtypes: the biphasic, monophasic and poorly differentiated types . The co-Immunohistochemically, most synovial sarcomas are positive for vimentin, cytokeratin and EMA, but have a lower immunoreactivity for S-100 and CD34 . AlthougAlthough curative resection and adjuvant irradiation can improve the local control of synovial sarcoma, metastases eventually develop in most patients. The lung and skeleton are the most commonly affected sites. Regional lymph nodes are involved in 10 to 23% of patients. In our case, although lung metastasis was not detected, pelvic and inguinal lymph node involvement was observed. The pitting edema could be caused by lymphedema due to the presence of lymph node metastases . PatientIn conclusion, we report a case of synovial sarcoma of the buttocks that presented with a non-healing wound and rapid progression after local resection. Despite slow growth in most cases, synovial sarcoma can be fatal within months of recognition if improperly managed. Our case stresses the importance of adequate preoperative evaluation and postoperative pathological analysis in the management of a subcutaneous mass.Written informed consent was obtained from the patient for publication of this case report and any accompanying images.EMA, Epithelial membrane antigen; MRI, Magnetic resonance imaging.The authors declare that they have no competing interests.ZHY collected the clinical data and performed immunohistochemistry. FY and ZZ collected the clinical data. GG carried out the pathological analysis. ZJS drafted the manuscript. All authors read and approved the final manuscript.


Primary meningeal rhabdomyosarcoma is a rare primary brain malignancy, with scant case reports. While most reports of primary intracranial rhabdomyosarcoma occur in pediatric patients, a handful of cases in adult patients have been reported in the medical literature. We report the case of a 44-year-old male who developed primary meningeal rhabdomyosarcoma. After developing episodes of right lower extremity weakness, word finding difficulty, and headaches, a brain magnetic resonance imaging (MRI) demonstrated a vertex lesion with radiographic appearance of a meningeal-derived tumor. Subtotal surgical resection was performed due to sagittal sinus invasion and initial pathology was interpreted as an anaplastic meningioma. Re-review of pathology demonstrated rhabdomyosarcoma negative for alveolar translocation t. Staging studies revealed no evidence of disseminated disease. He was treated with stereotactic radiotherapy with concurrent temozolamide to be followed by vincristine, actinomycin-D, and cyclophosphamide (VAC) systemic therapy. The patient was a 44-year-old Caucasian male with no significant past medical history who initially experienced episodes of right lower extremity weakness lasting 2-3 minutes that first occurred in June 2010. He had 2 such episodes in early June and within a few weeks was experiencing 5-6 episodes weekly. Shortly thereafter he noticed impairment in fine motor skills of the right leg with inability to put on shoes, word-finding difficulty, and severe headaches that awoke him from sleep. He was seen by his primary care physician, and MRI demonstrated a 2.7 × 2.5 × 3.1 cm brain lesion arising from the meninges . He was He was evaluated in the emergency room and admitted to the hospital for further evaluation and management. Surgical resection was performed July 2010 with pathology initially interpreted as an anaplastic meningioma. Complete resection was not possible given extensive invasion into the sagittal sinus. Postoperatively, the patient reported right-sided motor impairment that improved dramatically with physical therapy. His headaches and word-finding difficulty resolved completely postoperatively, and he was discharged from the hospital on postoperative day no.3. A brain MRI obtained 10 days following surgery showed a left frontoparietal vertex resection cavity measuring 2 × 1.5 cm with enhancement anteriorly and posteriorly. in situ hybridization was performed, and the typical gene rearrangement (t2:13) seen in alveolar rhabdomyosarcoma was not visualized. Additional staging studies, including PET imaging, spine MRI, bone marrow biopsy, and lumbar puncture, were obtained, showing no evidence of metastatic disease.He presented to our institution for an opinion regarding further therapy. On exam, he was noted to have right upper and lower extremity weakness and wide-based gait with right foot drop. Pending review of pathology, we tentatively recommended highly conformal radiotherapy. Pathologic review confirmed a high-grade sarcoma consistent with rhabdomyosarcoma . Tumor cAfter generating a variety of radiotherapy plans, a volumetric modulated arc therapy (VMAT) plan utilizing a Novalis Tx linear accelerator system  was ultimately selected in order to maximize target coverage while minimizing normal tissue toxicity. In addition, we offered concurrent temozolomide as a radiosensitizer . The priThe patient tolerated chemoradiation well. Aside from mild fatigue, he developed headaches during treatment which improved with a low dose of steroids that was gradually tapered. Blood counts remained stable throughout radiotherapy. At the conclusion of chemoradiotherapy, the patient began systemic therapy, consisting of vincristine, actinomycin-D, and cyclophosphamide (VAC) per standard treatment for rhabdomyosarcoma. Rhabdomyosarcoma, the most common pediatric soft tissue sarcoma, has a predilection for the head and neck, genitourinary organs, and extremities . The meaStaging with lumbar puncture and imaging is important for identification of all sites of disease. Lumbar puncture or imaging of the craniospinal axis can identify leptomeningeal or multifocal disease which can have implications on radiotherapy fields and total dose. Few cases have reported diffuse meningeal involvement, but it is associated with a poorer prognosis , 8. In aOnce histological diagnosis and staging is preformed, there is still much debate regarding appropriate further treatment. A review of published literature identified 38 previously reported cases of intracranial rhabdomyosarcoma with a mean survival of 9.1 months . In thisThe inherent difficulty in discerning the cell of origin in rhabdomyosarcoma makes the selection of appropriate chemotherapy difficult and usefulness debatable. In the review by Celli et al., chemotherapy was administered in 11 of 38 cases reported but given heterogeneity of treatments little could be determined regarding relative effectiveness. Recent case reports have implemented the use of VAC chemotherapy which is standard in the treatment of systemic rhabdomyosarcoma [Our patient represents one of the few adults with primary meningeal rhabdomyosarcoma. Given the rarity of these tumors, a multidisciplinary approach is imperative. While maximal safe surgical resection followed by adjuvant radiotherapy and chemotherapy is a logical approach to maximize long-term survival, further study is required to identify an optimal therapeutic strategy.


Thoracic splenosis is a rare entity resulting from splenic and diaphragmatic injury. Patients remain asymptomatic, and surgical intervention is not indicated in the majority of cases. We report a case of a 50-year-old male with a history of splenectomy due to a gunshot wound 30 years previously who presented with vague, progressively worsening chest pain. He was found to have a large intrathoracic splenosis. Unique features of our patient include the presence of symptoms, the significant interval growth of the splenic tissue, and the unprecedented size of the mass, which we believe to be the largest among those previously described. Thoracic splenosis is a rare, benign condition involving autotransplantation of splenic tissue into the pleural cavity via trauma or surgery. It is usually incidentally detected, asymptomatic, and therapy is not indicated. Due to the resemblance to malignancy on diagnostic imaging studies, the majority of patients have undergone extensive workups or invasive procedures in the past . It has 99mTc) sulfur colloid scintigraphy was performed based on a high index of suspicion for splenosis given the patient’s trauma history and subsequent splenectomy. Thick linear uptake was present in the medial left hemithorax, corresponding to the mass on the chest CT, and was consistent with splenic tissue (Figure A 50-year-old male presented with a history of left-sided chest pain for a few years that had been progressively worsening over the last 3 months. The pain was characterized as vague and intermittent, although there were some fluctuations in intensity. He had a gunshot wound to the left upper abdomen resulting in diaphragmatic and splenic rupture 30 years ago. He stated that his spleen had been removed at that time. Other medical history included chronic hepatitis C and a 30 pack-year history of smoking. His physical examination revealed several abdominal scars, but was otherwise unremarkable. Routine blood work demonstrated no significant laboratory abnormalities. A peripheral smear was not obtained. On presentation, his chest X-ray showed a large, ill-defined, clearly marginated mass along the left heart border on the posteroanterior view. In comparison, the chest radiograph obtained 1 year previously was reviewed, and the lesion was noted, in retrospect, to have been present at that time; it had obviously grown over the 12-month period between the two examinations (Figures 99mTc sulfur colloid scintigraphy was first used to diagnose splenosis based on the ability of the radiolabeled colloid to localize in the reticuloendothelial system. However, scintigraphy using 99mTc heat-damaged erythrocytes or indium 111-labeled platelets is more sensitive and specific for splenic uptake, making these tests the current diagnostic tools of choice [Thoracic splenosis, first reported by Shaw and Shafi in 1937 , describf choice . We postWritten informed consent was obtained from the patient for publication of this case report. A copy of written consent is available for review by the Editors-in Chief of this journal.The authors declare that they have no competing interests.SF and ST wrote the manuscript. AR, JY and SF performed surgery. MK supervised manuscript and entire treatment. All Authors read and approved the final manuscript.


We report a case highlighting the multidisciplinary management of a giant pleomorphic adenoma of the parotid gland that showed rapid growth in the third trimester of pregnancy.A 43-year-old Caucasian woman presented in her 32nd week of gestation with a tumor of the parotid gland. Ultrasonography of her neck showed a parotid lesion of 40 × 30 × 27.5 mm. A follow-up magnetic resonance imaging scan of the neck four weeks later revealed that the tumor had grown to 70 × 60 × 60 mm, reaching the parapharyngeal space with marked obstruction of the oropharynx of about 50%. After discussing the case with our multidisciplinary tumor board and the gynecologists it was decided to deliver the baby by caesarean section in the 38th week of gestation, and then to perform a surgical resection of the tumor.Indications for early surgical intervention of similar cases should be discussed on an individual patient basis in a multidisciplinary setting. With an incidence of 65%, pleomorphic adenoma is the most common tumor of the salivary glands , and 80%A 43-year-old Caucasian woman presented in her 32nd week of gestation with a tumor of the parotid gland. She had noticed the swelling increasing over the past six months. She was otherwise asymptomatic, without any pain, mouth-opening difficulties or facial nerve dysfunction. Ultrasonography of her neck showed a parotid lesion 40 × 30 × 27.5 mm in size, with an heterogeneous appearance. Considering her advanced state of pregnancy, we advised conservative management in the form of watchful waiting and regular review until the birth.At our patient's next review, only five weeks later, we noticed rapid growth of the lesion with expansion into the parapharyngeal space. There was still no facial nerve involvement, although our patient's mouth opening was slightly limited. Magnetic resonance imaging (MRI) of the neck showed that the tumor had increased to 70 × 60 × 60 mm, reaching the parapharyngeal space with marked obstruction of the oropharynx of about 50% Figures , 2. FurtAfter discussing this case with the gynecologists at our interdisciplinary tumor board meeting, it was decided to deliver the baby by caesarean section in the 38th week of gestation with epidural anesthesia using mepivacain and sufentanil. The surgical resection of the tumor was performed four days later. It was possible to completely remove the tumor via a standard cervicofacial incision without the need to resect the mandible. Although the tumor had stretched the facial nerve to double its usual length, there was no postoperative nerve dysfunction (House Brackmann grade I).On histological analysis, the lesion was identified as a pleomorphic adenoma without evidence of malignancy Figure . ImmunohThe incidence of head and neck cancers during pregnancy is rising . PleomorWe recommend frequent monitoring of tumors of the salivary glands diagnosed during pregnancy. Indications for early surgical intervention should be discussed on a individual patient basis in a multidisciplinary setting.Written informed consent was obtained from the patient for publication of the manuscript 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.FP was the major contributor in writing the manuscript. ML helped to assemble the data and assisted with the manuscript writing. JV provided expert advice, performed the Caesarean section, and revised his part of the manuscript. TU helped to revise the manuscript, HS performed the gland surgery and performed the final reading of the manuscript. All authors have read and approved the final manuscript.


Intracranial chondromas are rare benign tumors with an incidence of 0.2% to 0.3% of all intracranial tumors. This is the first case of an intracranial chondroma reported from Pakistan.We report a case of a 23-year-old Asian man presenting with intracerebral chondroma of the left frontal lobe, which was eroding the dura matter. The intracranial chondroma was completely removed by surgery.Intracranial chondromas are rare benign cartilaginous tumors. Through this case presentation we have discussed the diagnostic procedures, radiological and pathological findings. The purpose of presenting such a rare case is to develop awareness among clinicians and medical students and to highlight the requirement of immediate actions to ensure proper management of such cases. Intracranial chondromas are exceedingly rare neoplasms, which grow slowly by expansion. They are cysts of chondroid tissue, and present at different regions within the cranial cavity especially the skull base . Sellar We present a case of an intracerebral cystic chondroma of the left frontal lobe in a 23-year-old man which was diagnosed by radiological findings and further confirmed through pathological reports. Frontal craniotomy was performed for total tumor resection. This is the first case of intracranial chondroma reported from Pakistan. We have discussed the clinical presentation, etiology, radiological features, histological features and treatment of the case with references to previously reported cases.A 23-year-old Asian man with a history of generalized tonic–clonic seizures for the past year was admitted to a government hospital in Karachi, Pakistan.T1-weighted magnetic resonance imaging (MRI) revealed a space occupying lesion in the patient’s left frontal lobe Figures . T2-weigA frontal craniotomy was performed. The tumor had eroded the dura mater. The tumor was hard and cystic, with the cavity containing greenish fluid. The whole of the cyst tumor was recovered. Hemostasis was secured. The dura was closed followed by closure of the pericranium.Surgical pathology confirmed the case as intracranial cystic chondroma. The gross pathological report showed multiple tan, firm tissue fragments measuring 10.0×7.5×1.4cm. The surface appeared lobulated with focal areas of hemorrhage. The microscopic pathological report showed multiple fragments of chondroid tissue surrounded by a cyst wall. The outer surface of the cyst wall showed a thin fibrous capsule whereas the inner surface showed areas of hemorrhage. The chondroid tissue was composed of lobules of chondrocytes set in spaces against a chondroid matrix. Cells showed moderate cytoplasm, and the nuclei appeared round to oval and vesicular. A significant increase in cellularity, nuclear pleomorphism or mitosis was not noted. A few fragments of fibro-collagenous tissue with hemorrhage were also noted.Post-operative computed tomography (CT) scan showed successful resection of the tumor Figure .Intracranial chondromas are benign cartilaginous tumors. They are extremely rare and account for only 0.2% to 0.3% of all intracranial tumors ,6. IntraThese tumors mostly originate from rests of cartilaginous cells at sphenoethmoidal and spheno-occipital synchondroses at the base of the skull . They arIntracranial chondroma has also been reported as a component of Ollier’s multiple chondromatosis . PontineIntracranial chondromas may develop in a person at any age but they have been most frequently observed in the third decade  which inThe presenting symptoms range from headaches  to lowerGeneralized tonic–clonic seizures are also usually the presenting complaints in intracranial chondromas , as in oIntracranial chondromas are thought to arise from mainly the skull base, dura, brain parenchyma or within the ventricles . VariousRadiological findings of intradural chondromas are distinctive. On X-ray, intracranial chondromas represent hyperostosis of the internal table of the skull , enhanceet al. reported two instances of skull base chondromas that exhibited delayed contrast enhancement on CT after a high-dose of the contrast medium was administered. They suggested this CT feature to be employed in differential diagnosis of intracranial chondromas from meningiomas and neurinomas [A study reported that intradural chondromas possess two different CT appearances. The usually found type 1 shows mixed density with minimal or moderate enhancements. The rare type 2 shows an innermost less dense area containing a cyst . Tanohaturinomas .et al. reported variable signal intensity at different levels of MRI in a case of intracranial chondroma. At T1 they reported less intensity whereas at T2 the signal appeared to be of middle to high intensity [MRI has become an important diagnostic tool for intracranial chondromas. Brownlee ntensity .The treatment of choice for such a benign space-occupying tumor is complete tumor excision, as in our case. The treatment recommended by Krayenbühl and Yasargil is total surgical removal of tumors because partial excision can improve the symptoms for a few years only . RecurreConcluding the above discussion, intracranial chondromas are masses of well-differentiated, cytologically benign cartilage having a small tendency for sarcomatous change and they do not invade the surrounding parenchyma. Treatment of choice is complete tumor resection with a favorable long-term prognosis.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.The patient was under the care of MMU. MMU and JA surgically operated on the patient. MMU and AAM analyzed and interpreted the data. AAM wrote the manuscript. JA made additions to the manuscript. All authors reviewed and approved the final manuscript.


Cellular angiofibroma is a benign vascular neoplasm that typically arises in the paratesticular region in men and is easily confused with inguinal or scrotal hernia.We present a case of a cellular angiofibroma arising from the spermatic cord of a 74-year-old Caucasian man. Initially, the lesion was confused with a scrotal hernia, but imaging revealed a subcutaneous, inhomogeneous, but well-circumscribed lesion to the surrounding tissues with rich vasculature. Surgical resection of the lesion was performed. Histology revealed a benign tumor of vascular origin rich in fibroblasts.Angiofibroma can easily be confused with an inguinal hernia and should be differentiated from Schwann cell tumors, perineuromas, spindle-cell lipomas, aggressive angiomyxomas, angiomyofibroblastomas, solitary fibrous tumors, spindle-cell liposarcomas, and leiomyomas. A safe initial diagnosis is difficult because of its location, nature, and correlation with other structures of the area. ClinicaA 74-year-old Caucasian Greek man was referred to our surgical clinic for repair of a left inguinal hernia. The patient had noticed a gradually enlarging mass 10 years prior to presentation. His physical examination revealed an elastic, hard, slightly mobile mass that was initially confused with a scrotal hernia, although reduction maneuvers produced no result, even after the intramuscular administration of pethidine. No abnormal dermal findings were observed.Ultrasonography showed the presence of a large (9 cm × 4 cm), rigid, inhomogeneous structure starting from the left inguinal space under the skin but not penetrating the corresponding hemiscrotum. Doppler sonography demonstrated prominent, rich vasculature. On computed tomography (CT), the lesion was observed to be round, with a diameter of 13 cm, inhomogeneous to the surrounding fat tissue of the anterior abdominal wall at the level of the left spermatic cord, and pushing away the left testis , inflammatory infiltration of lymphocytes, plasma cells, mast cells, and abundant capillaries, many of which with regenerating and degenerating forms Figures .et al. [et al. [et al. Finally, Iwasa and Fletcher [Embryologically, the mesoderm of the scrotum gives birth to various tissues; thus tumors arising from that area have high diversity, and confirming a safe diagnosis between a benign and malignant lesion is difficult. Cellular AF was first described in 1997 by Nucci  [et al.  describeFletcher  reportedHistologically, the tumors are typically well circumscribed, quite cellular with spindle-shaped cells evenly distributed, and with short bundles of collagen. Less cellular areas are often associated with stromal edema or hyalinization, but significant pleomorphism and abnormal mitoses are absent. The numerous vessels observed are round, thick-walled, and hyalinized .Immunohistochemical diagnostic procedures reveal that 60% of patients have slight expression of CD34 (vascular origin), 21% have spinal muscular atrophy , and 8% reveal desmin (muscular origin) [The diagnostic imaging workup includes a CT scan without specific findings for this entity , while oIt may be difficult to distinguish cellular AF from other tumors of the scrotum on the basis of radiological data only. The differential diagnosis includes tumors of Schwann cells, perineuromas, spindle cell lipomas , aggressSurgical resection of the tumor is the therapeutic method of choice. Unfortunately, follow-up clinical data for cellular AF is limited, although recurrences have been reported . A complCellular AF is a benign neoplasm of the scrotal and inguinal area, is rich in fibroblasts, and of vascular origin. A safe initial diagnosis is difficult because of its location, nature, and correlation with other structures of the area. It can easily be confused with a hernia, especially when the lesion slides toward the scrotum. Moreover, it is crucial to differentiate cellular AF from AAM and other spindle-cell neoplasms, since they exhibit malignant behavior with recurrences and metastases.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.PD and AM analyzed and interpreted the patient data and drafted the manuscript. AZ performed the histological examination of the tumor. AM and SR critically revised the manuscript. All authors read and approved the final manuscript.


Splenic torsion is rare and as a result the appropriate management is unclear. While there has been a shift towards splenectomy and laparoscopic splenopexy, we present a successful case of non-operative management of splenic torsion in a patient with a history of a giant omphalocele.A 3 year-old female presented with a three-day history of abdominal pain, fever and non-bloody emesis three and a half years after repair of her giant omphalocele. Abdominal radiographs and ultrasound demonstrated migration of the spleen and a subsequent computerized tomography scan confirmed splenic torsion and an infarcted spleen. Given her late presentation, she was successfully managed with observation, analgesia, immunization against capsulated organisms and daily penicillin prophylaxis with excellent outcome at 19 months follow-up. A review of the literature revealed that splenic torsion is rarely managed non-operatively. Rarer still is the occurrence of splenic torsion following a history of omphalocele.Although rare, splenic torsion should be considered in a child with a history of omphalocele presenting with abdominal pain. Non-operative management of an infarcted spleen can be a safe treatment option to avoid surgery in complex patients. Splenic torsion and wandering spleen are rare causes of abdominal pain. The splenic attachments, when absent or underdeveloped, permit the spleen to migrate from its typical position in the left upper quadrant of the abdomen to anywhere in the abdomen. The wanSplenic torsion is difficult to diagnose due to presentation with symptoms that are non-specific including abdominal pain, fever, nausea and vomiting. Symptoms may or may not be accompanied by palpation of an abdominal mass on physical exam. Thus, a high index of suspicion and imaging is required for accurate diagnosis. Sonography is the imaging study of choice for wandering spleen although CT scan may be needed in certain cases to confirm diagnoses-3. In de3 uL, the hematocrit was 31.2 and platelet count was 279 103 uL. A CT scan showed the spleen to be located in the left flank, vertically oriented with the hilum facing laterally (Figure A 41 month-old female with a history of prenatally diagnosed giant omphalocele that was repaired at nine months of age following epithelialization of the sac and gradual reduction of the omphalocele. She presented to the emergency room with a three-day history of left upper quadrant abdominal pain, fatigue, one episode of non-bloody, non-bilious emesis and fever to 102 F. Physical examination noted a well-healed abdominal midline scar but did not elicit abdominal tenderness or palpable mass. Abdominal plain film was significant for a mass within the left flank (Figure There are six cases of wandering spleen reported in patients with a history of congenital diaphragmatic hernia. The literature consists of two prior reports of wandering spleen in patients with omphalocele; the first in 1987 and the second in 2008 which was managed by de-torsion of the spleen and splenopexy[Management of splenic torsion has evolved from primarily a splenectomy approach towards increasing numbers of splenopexies via the laparoscopic approach. Non-opePrior to our case, reports in literature have not advocated non-operative treatment as a feasible plan of care complication rates as high as 65%, resulting in splenic rupture and infarction due to torsion of the splenic pedicle,3,5-7. OThe initial method to diagnosing splenic torsion is to image the abdomen using either ultrasound or CT imaging, both sufficient methods used to diagnose splenic torsion and evaluate the vascular supply to the spleen,7. SonogWhile surgical options are usually regarded as the most favorable course for treating splenic torsion, non-operative management can be a viable option. As demonstrated with our case, the patient had clearly developed an infarcted spleen at the time of CT scan but was exhibiting a stable clinical picture given the resolution of fever, stable vital signs and resolution of abdominal pain post emesis. The patient has since remained free of infection by maintaining a regimen of antibiotic prophylaxis and immunizations. The benefit is that she was able to avoid an unnecessary operation. Non-operative management allowed the course of splenic infarction to continue naturally and resemble autosplenectomy as seen in children with sickle cell anemia. Though not identical to splenic torsion, the same risks are present for both etiologies including infection and hemorrhage. While there has not been any literature focused on non-operative management of splenic torsion in over 10 years, there was a recent study that reviewed the management of 190 patients with acute splenic sequestration crisis from 2000 to 2009. The study demonstrated that the 103 that underwent watchful waiting management had a favorable outcome overall despite some patients having a recurrence of acute splenic sequestration crisis.A literature review published in 2003 revealed 17 open splenectomy, 21 open splenopexy, and 2 laparoscopic splenopexy approaches for the management of splenic torsion and wandering spleen. Since 2A case published in 2003 reported an incidence of splenic rupture secondary to splenic torsion in a 25 year-old female. She was hypotensive, underwent an exploratory laparotomy and splenectomy and was placed on standard post-splenectomy precautions. While tWe suggest that non-operative management for splenic torsion be considered a safe alternative approach to managing select patients that are in stable condition. While splenectomy and splenopexy have been the foundation for treatment and the increase in use of laparoscopy and mesh have offered innovative approaches with sound results, non-operative management may be considered as an additional option for patients that are high risk surgical candidates.Written informed consent was obtained from the patient’s parent for publication of this Case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.FS made substantial contributions to acquisition of data, drafting the manuscript, agreed to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. ME made substantial contributions to acquisition of data, drafting the manuscript, agreed to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. IZ has been involved in drafting the manuscript and agreed to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. OO has been involved in drafting the manuscript, revising it critically for important intellectual content, has given final approval of the version to be published, and agreed to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. All authors read and approved the final manuscript.FS is a surgical resident and is currently completing a pediatric surgery research fellowship at Baylor College of Medicine.MS has a master’s degree in anatomy, completed a research fellowship at Baylor College of Medicine and is a medical student at Midwestern University.IZ is a surgical resident, currently completing a pediatric surgery research fellowship at Baylor College of Medicine, and obtaining his Masters in Public Health.OO is an attending pediatric surgeon, has a PhD in anatomy and is a professor of surgery, pediatrics, and obstetrics and gynecology at Baylor College of Medicine.


Herein, we present the case of a patient with recurrent hepatocellular carcinoma (HCC) who had paracaval lymph node (LN) metastases with an inferior vena cava (IVC) tumor thrombus after a hepatectomy. A 65-year-old man with chronic hepatitis B virus infection received an extended anterior segmentectomy because of two hepatic tumors, located in segments 7 and 8. Histological examination of both resected specimens showed mostly moderately differentiated HCC with some poorly differentiated areas, and liver cirrhosis (A2/F4). Because the patient had an elevated α-fetoprotein serum level, abdominal computed tomography (CT) was performed. Abdominal CT revealed a 9-mm-diameter recurrent tumor in hepatic segment 3 and paracaval LN metastases with an IVC tumor thrombus at 8 months after the first operation. The patient received transcatheter arterial chemoembolization as treatment for the intrahepatic recurrence, following resection of the paracaval LN metastases and removal of the IVC tumor thrombus. In this case, the paracaval LN metastases had directly infiltrated the IVC via the lumbar veins, resulting in an IVC tumor thrombus, which usually develops from an intrahepatic tumor via the hepatic vein. The development of an IVC tumor thrombus with HCC recurrence, as in this case, is very rare, and based on a PubMed search, we believe this report may be the first to describe this condition. Hepatocellular carcinoma (HCC) is a highly malignant form of cancer, which recurs frequently after hepatectomy . The mos18 F-fluorodeoxyglucose (18 F-FDG) revealed no other areas of increased uptake, except for an intense area of increased uptake  in the region that corresponded to the enlarged LNs, as previously seen with CT imaging. On the basis of these findings, we preoperatively diagnosed this condition as recurrence of intrahepatic carcinoma and paracaval LN metastases, with IVC invasion and thrombus formation. For treating the intrahepatic recurrence, the patient received transcatheter arterial chemoembolization (TACE) at the time of the CT angiography. Finally, as there was no metastasis in any other organs and the HCC was well controlled, we decided to perform surgical treatment for the LN metastases as a curative treatment.A 65-year-old man with hepatitis B virus (HBV)-related cirrhosis was admitted to our institution for the treatment of recurrent intrahepatic HCC and paracaval LN metastases, with an IVC invasion and an IVC tumor thrombus. Eight months before this visit, he had undergone a curative operation for HCC at our institution. This operation involved an extended anterior segmentectomy, including resection of hepatic segment 7, for the treatment of two hepatic tumors, and the histological findings of both resected specimens showed mostly moderately differentiated HCC, with some poorly differentiated areas, and liver cirrhosis , and they concluded that LN metastasis from HCC was sensitive to RT. Also, Hou et al. evaluated the influence of PV vs. IVC tumor thrombosis sites on the effectiveness of RT in advanced HCC with macrovascular invasion. They concluded that HCC patients with IVC thrombus treated with RT had a better response rate and longer survival than those with PV thrombus [p < 0.01) and concluded that selective lymphadenectomy of LN metastasis was a safe and efficacious procedure [Livraghi et al. reported a cohort study of radiofrequency ablation (RFA) and demonstrated that complete ablation of lesions smaller than 2 cm is possible in more than 90% of cases, with a local recurrence rate of less than 1% . Also, Bthrombus . In controcedure , althougrocedure . After erocedure . As Bruirocedure . In our rocedure . Howeverrocedure , and it In conclusion, our patient had an uncommon pattern of progression and growth of HCC, with LN metastases that comprised HCC and infiltrated the IVC and produced an IVC tumor thrombus. Surgeons should consider the possibility of LN skip metastases and infiltrative growth patterns with tumor thrombi after hepatectomies.Written informed consent was obtained from the patient for the publication of this report and any accompanying images.18 F-fluorodeoxyglucose; HBV: Hepatitis B virus; HCC: Hepatocellular carcinoma; IVC: Inferior vena cava; LC: Liver cirrhosis; LNs: Lymph nodes; PET: Positron-emission tomography; PIVKA-II: Protein induced by vitamin K absence or antagonist II; PV: Portal venous; RFA: Radiofrequency ablation; RT: Radiotherapy; SBRT: Stereotactic body radiotherapy; TACE: Transcatheter arterial chemoembolization.AFP: α-fetoprotein; CT: Computed tomography; CTA: Computed tomography during arteriography; CTAP: Computed tomography during arterioportography; F-FDG: The authors declare that they have no competing interests.SI and KI drafted the manuscript and made revisions. HO, HT, HA, TK, KI, KI, SI performed the surgery. HA and KC participated in the medical treatment. KA carried out the pathological examination. All authors read and approved the final manuscript.


Leiomyomas are benign tumors of the soft tissue and may develop in any location where smooth muscle is present. Leiomyoma in the inferior vena cava is a rarely seen pathology, and symplastic leiomyoma is also a rare histological variant of leiomyoma. In this case, we present a rare histological variant of symplastic leiomyoma in the inferior vena cava (IVC). This is the first radiologically reported case of a symplastic leiomyoma of the IVC. Leiomyomas are benign tumors of the soft tissue and may develop in any location where smooth muscle is present. They were first described by Virchow in 1854. These tumors are more frequently seen in peripheral blood vessels compared to the central vessels and have been identified in the vena cava in only a few cases  staining and negative results with p53 and S-100. The Ki-67 proliferation index was up to 10%. Accordingly, the histopathological diagnosis was considered as symplastic leiomyoma. One month after operation, MR imaging was performed and no residual mass was determined around the IVC . IVC wasThe role of MRI in the evaluation of retroperitoneal soft tissue masses is becoming more important. Localization of the lesion, pattern of spread, specific components and vascularity of the tumor narrow the spectrum of the differential diagnosis , 3.In our case, the mass had a right sided retroperitoneal localization which seemed to originate from the IVC and pancreas, because it could not be distinctly separated from them. However, the chance of a potential diagnosis of a pancreatic mass lesion is decreasing due to absence of laboratory and clinical findings to support pre-diagnosis and imaging evidence, in addition to MR imaging findings. Primary smooth muscle tumors originating from vessels are quite rare and most of them are malignant. Leiomyosarcomas are the most common primary malignancy of IVC that generate more than 90% of all reported IVC sarcomas. There are intimal and mural subtypes of IVC sarcomas according to their origin. Intimal ones, which are also called luminal, are differentiated sarcomas, undifferentiated pleomorphic sarcoma and angiosarcoma. Mural sarcomas are usually thought to develop into leiomyosarcomas from the muscle of media. Leiomyomas also arise from the smooth muscles of media -6. SinceIn our case, the delayed pattern of enhancement was present. This pattern is often seen in neurogenic tumor subtypes of benign tumors and in a few malignant tumors, such as desmoid tumors, hemangioma, leiomyoma and also myxoid liposarcoma, leiomyosarcoma and malignant lymphoma , 3. An eLeiomyomas and leiomyosarcomas are round or lobulated masses with regular contours. MR images of the lesions depend on the amount of fibrous and smooth muscle cells in each lesion. Lesions are similar to skeletal muscle on T1-weighted sequences; however, most of them show heterogeneous signal intensity. Typical appearances are hypo- or isointense on T1-weighted images and hypointense on T2 weighted images. Solid components often show contrast enhancement following injection of contrast material intravenously. Non-degenerated uterine leiomyomas have a typical appearance on MR images. They are well-defined masses that show hypointense homogeneous signal intensity on T2-weighted images compared to the muscle tissue. Cellular leiomyomas show relatively higher signal intensity on T2-weighted series and contrast enhancement following injection of contrast material intravenously . SimilarHistopathologically, there are typical, epithelioid, cellular, hemorrhagic cellular, lipoleiomyoma and symplastic variants of leiomyoma . The terGrowth patterns of leiomyoma variants are able to mimic that of more aggressive cancers. Disseminated peritoneal carcinomatosis may resemble peritoneal leiomyomatosis; parasitic leiomyoma or intravenous leiomyomatosis may resemble leiomyosarcomas; intravenous leiomyomatosis may resemble renal cell carcinoma in the involvement of IVC; and benign metastatic leiomyoma may resemble pulmonary or hepatic metastatic disease .Pleomorphic tumor cells with distinct atypical nuclei and low mitotic rate are important characteristics of the variants of the symplastic leiomyoma. Most pleomorphic cells often have multinucleated or multilobule nucleus, but the large and dilated mononuclear cells can be observed. These multinucleated cells can be present as focal, multifocal or diffuse throughout the mass, and generate more than 25% of the tumor in most cases. Although these tumors usually have degeneration, edema and hyaline changes, there is no coagulative tumor cell necrosis. Symplastic cells are observed more frequently on the edges of degenerative areas . It may In this study, we attempted to present a rare histological variant of vascular leiomyoma. It is difficult to identify the origin of these large tumors with imaging studies. As with other defined vascular leiomyomas, MRI shows signal intensity depending on the histopathological changes and tissue content. This rare variant can easily be confused with leiomyosarcoma . Examina


The presence of chronic renal disease (CRD) is a predisposing factor for the occurrence of soft and hard tissue lesions in the oral cavity. The cemento-ossifying fibroma (COF) is an uncommon benign fibroosseous lesion composed of fibrocellular component and calcified materials like cementum and woven bone. A 37-year-old female patient undergoing chronic haemodialysis reported to our institution with a complaint of slow growing, nontender swelling of mandible of 6-month duration. Computed tomography disclosed an ill-defined lesion showing thinning and expansion of buccal as well as lingual cortical plate with flecks of radiopacity in centre. Incision biopsy revealed histological characteristics consistent with cemento-ossifying fibroma. The lesion was excised under local anesthesia. The histopathological examination revealed irregularly shaped bone and cementum-like hard tissue calcifications contained within hypercellular fibrous tissue stroma, leading to a confirmation of the diagnosis of cemento-ossifying fibroma. This paper aims to provide light to the fact that the soft and hard tissues of the oral region may become susceptible to the development of pathological growths in case of some particular systemic conditions. Chronic renal disease (CRD) can be defined as the progressive and irreversible decline in kidney functions and up to 90% of patients having renal insufficiency may present with oral signs and symptoms in soft and hard tissues either as a result of the disease itself or the treatment protocol . HalitosCemento-ossifying fibroma (COF) is a distinct form of benign fibroosseous lesions of the mandible and maxilla containing fibrous tissue and varying amounts of calcified tissue resembling bone, cementum, or both . The patA 37-year-old patient reported to outpatient department of our institution with a complaint of swelling of the lower jaw of six-month duration. There was neither a history of trauma or teeth extraction related to the swelling nor any similar swelling in other parts of the body. Patient's history revealed that she had been suffering from chronic renal Disease for the past six years. She had also been undergoing hemodialysis once a week for the past two years. On further questioning, patient revealed that she was suffering from pain in all joints. Patient was having hypertension secondary to the declining kidney function, for which she was under constant medication. The patient was manual labourer in farms for the past 20 years. Because of her declining physical condition she had stopped her work for the past four years.Extra oral findings were nonremarkable. Intraoral examination revealed a nontender bony hard swelling of size 1.5 × 2 cm on the right mandibular body extending from central incisor to the first premolar with buccal cortical plate expansion. The overlying mucosa appeared to be normal. Pulp vitality of the teeth adjacent to the lesion proved positive. Orthopantomogram revealed an ill-defined radiolucency with radio-opaque specks located in the mandibular body. Root resorption was noted for the right canine and first premolar. Axial imaging showed the lesion to cause thinning and expansion of buccal as well as lingual cortical plate . LikewisThe concept of “fibroosseous lesions” of bone evolved over the last several decades to include two major entities: fibrous dysplasia and ossifying fibroma as well as the other less common. It is a slow-growing lesion most often seen in women between the third and fourth decades of life. Both jawbones can be affected, although 62% to 89% of the lesions arise in the mandible, involving premolar and molar regions in 77% of cases. While one-half of all cases are asymptomatic, the growth of the tumor over time may lead to facial asymmetry, with the appearance of a mass causing discomfort or mandibular expansion and the possible displacement of dental roots.Cemento-ossifying fibroma is a thought to have its origin from arises from the periodontal membrane . The perCemento-ossifying fibroma is a slow growing lesion composed of cellular fibroblastic tissue containing basophilic masses of cementum-like tissue. In addition, a varying amount of bony trabeculae is interspersed within the lesion, giving it its characteristic appearance . Reed usRadiological findings suggests that the lesion is usually well circumscribed and demarcated from surrounding bone, in contrast to true fibrous dysplasia. The central cementifying fibroma and the central ossifying fibroma, have a centrifugal growth pattern rather than a linear one. Therefore, lesions grow by expansion equally in all direction and present as round tumor masses.α-hydroxylase activity [The association of ossifying fibromas with renal diseases can be observed in hyperparathyroidism-jaw tumor syndrome, in which the patients suffer from familial parathyroid adenomas, ossifying fibroma of the jaws, renal cysts, and Wilms tumors , 14. Theactivity . Both FGactivity . Calcitractivity . Studiesactivity . In addiactivity . Thus, aactivity . This spDue to the good delimitation of the COF, surgical removal and curettage is the treatment of choice. In the case of very large lesions with important tissue ablation, the challenge is to replace the affected tissue. The prognosis is usually good, since recurrences are not frequent.Like any other systemic disease, CRD can also present with oral manifestations. Bony alterations in CRD are usually related to renal osteodystrophy. Further studies are needed to elucidate the pathogenesis of ossifying fibroma in patients with CRD.


Seventeen cases of peritonitis due to rupture of a pyonephrosis have been reported. The majority of these cases occur secondary to renal stones. Only two cases of ruptured pyonephrosis with concurrent kidney neoplasm have been described and only one of these presented as an acute peritonitis. In this presentation we discuss an unusual case of a 68 year old man with a chronic history of bilateral nephrolithiasis and recent pyonephrosis. He presented acutely with peritonitis and was later found to have a carcinosarcoma of the kidney. The case highlights the importance of recognizing the possibility of underling renal carcinoma in patients presenting with a ruptured pyonephrosis and discuss steps to avoid this serious complication. Peritoneal fistulization of a pyonephrosis is an extremely rare event which invariably leads to generalized peritonitis . RuptureThe aim of this paper is to present an unusual case of a 68 years old man, with a previous history of gallstone pyonephrosis, presenting with an acute abdomen and having a final diagnosis of renal carcinosarcoma.A 68 years old man with a chronic history of bilateral nephrolithiasis was admitted to our department with high grade fever, rigors, lower back and diffuse abdominal pain. His past medical history included insertion of a left urethral stent six months earlier for pyonephrosis. The double J stent had not resolved the hydronephrosis and was due to be changed in the coming weeks. There was no history of diabetes mellitus. On examination, temperature was 39.5°C, heart rate 103 beats per minute (b.p.m), respiratory rate 28 breaths per minute, blood pressure 178/88 mmHg and there was generalized abdominal guarding and rigidity. White Blood Cells (WBC) were 24.600/cu mm, C-reactive protein (CRP) >160 mg/L, Haemoglobin (Hb) 9.1 gr/dl and Lactate Dehydrogenase (LDH) 220.000 Ul/l. No pre-operative urine or blood cultures were performed.An abdominal computed tomography (CT) scan was performed which revealed massive distension of the left kidney contained within the Gerota capsule. There was severe distension of the ascending/transverse colon and the left sided intestinal loops with a minor fluid collection in the pelvis but no free air Figure  &2. The Analysis of the kidney specimen revealed a 20 cm × 11 cm mass with a gelatinous centre and pus filled cysts in the renal pelvis Figure  &4. The et al remarked that spontaneous pelvic rupture practically always occurs in a kidney which is seat of chronic pylonephritis or where there is dilatation secondary to obstruction [et al included the additional conditions; stone formation, chronic nephritis, tuberculosis, abscess formation, infarct, aneurysm and tumor [Rupture of the kidneys more commonly occurs at the site of the renal parenchyma over the renal pelvis. In these instances, hemorrhage is at the forefront. This may be limited to the subcapsular space, confined in the renal fossa by the circumrenal fascia, or so massive to involve one side of the abdomen, engulfing the kidney in a shell of hemorrhagic tissue. In contrast urinary extravasation without hemorrhage is characteristic of a ruptured pelvis, although secondary hemorrhage can often occur. If rupture was preceded by a pyonephrosis the extravasated material may be infected. Dispersion may be limited by Gerota's capsule or involve the retroperitoneum and this may lead to fistulization into the abdominal cavity . Abeshoutruction . Mathe end tumor . Miller nd tumor . These ind tumor .Sarcomatoid renal cell carcinoma (SRCC), first described by Farrow et al. in 1968, is defined pathologically by highly pleomorphic spindle cells and/or giant cells resembling sarcoma, with varying degrees of clear or granular epithelial cells that characterize SRCC . These tet al discuss the case of a 44 year old man presenting with an acute abdomen. Laparotomy revealed a stercoraceous peritonitis from diastatic colic rupture with a T4 kidney tumor infiltrating the right colon [In the literature, only two cases of kidney neoplasm with a background of recurrent pyonephrosis have been reported and only one of these presented as an acute peritonitis. There have been seventeen cases of peritonitis due to rupture of a pyonephrosis: seven derived from spontaneous rupture of pyonephrosis in patients with urolithiasis or hydronephrosis and one in a patient with renal tuberculosis -23. Two ht colon .Intraperitoneal rupture of a pyonephrosys is a rare event which needs immediate intervention. The clinical presentation is of an acute abdomen with increased inflammatory markers and occasionally an associated pleural effusion. The predominant abdominal symptoms mask the underlying renal cause, and the peritonitis is attributed to intestinal perforation or appendicitis. X-rays fail to demonstrate free air in the diaphragm, although in rare cases they can highlight radiopaque stones. CT scanning is certainly the most sensitive at demonstrating the presence of underlying renal disease and subsequent laparotomy is inevitable. Establishing the fistulous site may not be possible due to inflammation with adhesions between parietal peritoneum and omentum. A careful exploration of the peritoneal cavity and all intestinal tracts is therefore necessary.In conclusion, this report highlights the importance of recognizing the possibility of underlying renal carcinoma in patients presenting with peritonitis and a history of pyonephrosis and stresses the significance in early and full urological investigation to avoid severe complication.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.SQ, AM, PR, ER and GP made up the surgical and pathological team invovled in the case. AP and NB wrote and edited the manuscript. All authors read and approved the final manuscript.


Chondrosarcoma is the third most common primary malignant bone tumor after osteosarcoma and Ewing's sarcoma. Clear cell chondrosarcoma is a rare subtype variant of chondrosarcoma, most commonly encountered in the proximal part of the femur or humerus. Vertebral involvement is exceedingly rare and shows a predilection for the thoracic spine. We report the case of a woman with clear cell chondrosarcoma of the thoracic spine, which has been surgically excised, and review the pertinent literature (PubMed). Although it has a reasonably benign biological behavior, clear cell chondrosarcoma needs to be treated as a malignancy. The best treatment for spinal chondrosarcoma is surgery. It should be promptly and adequately resected. Gross-total resection should be the ultimate surgical goal. Radiation therapy should also be considered, especially in the case of subtotal resection or inoperable lesions. In conclusion, it is important to keep in mind this entity in the differential diagnosis of spinal tumors, in order to optimize treatment planning. With adequate treatment, local recurrence rates as low as 20% can be achieved. Clear cell chondrosarcoma is a rare subtype variant of chondrosarcoma, most commonly encountered in the proximal part of the femur or humerus. Vertebral involvement is exceedingly rare. Surgical treatment is of paramount importance, since it is considered as a low-grade malignancy with high survival rates but may be challenging due to its proximity to the spinal cord and nerve roots. We report the case of a woman with clear cell chondrosarcoma of the thoracic spine and review the pertinent literature.A female patient, 61 years old, presented to our emergency department complaining of aggravating weakness of the lower limbs and back pain of one month duration. Upon admittance she was unable to stand and walk. She had visited a physician twice in this period, but she had only been administered with NSAIDs and analgesics. A careful physical evaluation revealed muscle weakness and hypoesthesia in both lower limbs and a 5th thoracic sensory level.Plain radiographs of the thoracic spine did not show anything other than a cuneiform degeneration of the 4th thoracic vertebra. A computed tomography scan revealed a spinal mass, with osteolytic erosion of the posterior elements of the 3rd and 4th thoracic vertebrae, and paravertebral extension . MagnetiThe patient underwent 3rd and 4th thoracic laminectomy, with consecutive gross-total excision of the mass with the aid of the operating microscope. Infiltrated surrounding tissue was also surgically debrided until margins free of tumor were obtained. Pathology was compatible with clear cell chondrosarcoma, mixed with conventional grade I chondrosarcoma .She showed immediate relief of her symptoms, being able to walk on the second postoperative day. A mild paresis of the left lower limb persisted in the immediate postoperative period. The oncological evaluation suggested adjuvant radiotherapy to minimize the risk of potential residual tumor. Hence, the patient was referred for and received postoperative three-dimensional conformal radiation therapy (50 Gy) two months after surgery. At one year from the operation, the patient is doing physically well and is free of local recurrence  or seconChondrosarcoma is the third most common primary malignant bone tumor after osteosarcoma and Ewing's sarcoma. However, the incidence of spinal chondrosarcomas is estimated to be from 2% to 12% in various series [Clear cell chondrosarcoma is a rare subtype of chondrosarcoma first described by Unni et al. in 1976 . It accoHistologically clear cell chondrosarcoma is characterized by benign giant cells alongside tumor cells with clear or granular cytoplasm, mixed with conventional chondrosarcoma in 50% of the cases. The cells present with rounded and centrally located nuclei. In cases in which the spine is involved, bone formation and cartilaginous components are minimal or even absent . VasculaThe most common presenting symptom in chondrosarcoma is pain in the area of the lesion. The pain is often insidious in nature and can be present for weeks to years. Other complaints include a palpable mass and neurologic deficit in half of the patients . The neuAlthough clear cell chondrosarcomas are considered low-grade malignant tumors, dedifferentiation into a more aggressive neoplasm can occur. Distant metastases have been reported . Even thChondrosarcomas are poorly responsive to chemotherapy and radiation . HoweverFinally, appropriate staging is essential for diagnosis and treatment and includes radiologic and histological information. The appropriate imaging studies allow for better understanding of the extent of the lesion as well as differential diagnosis, which includes metastatic lesions, angioblastic meningioma, osteosarcoma, Ewing's sarcoma, and hemangiopericytoma . ImagingPrimary clear cell chondrosarcoma of the spine is a rare encounter in clinical practice; being a rather low-grade malignancy, it benefits greatly from prompt and adequate treatment. Thus, it is important to keep in mind this entity in the differential diagnosis of spinal tumors, in order to optimize treatment planning. Clear cell chondrosarcoma is a surgical disease. Gross-total resection should be the ultimate surgical goal, although not always is it feasible due to the nobility of nearby structures. Adjuvant radiotherapy should also be taken into consideration in the management of this tumor.


Inflammatory pseudotumor of the liver represents a fairly uncommon pathology. Although it is a benign tumor, the correct diagnosis can be missed.We report the case of a 55-year-old Caucasian man, who presented with a one-month history of abdominal pain and weight loss. He was diagnosed with a primary liver tumor by computed tomography and magnetic resonance imaging. Alpha-fetoprotein levels ranged within normal limits. A right posterior sectorectomy was performed. Histopathology revealed an inflammatory pseudotumor of the liver. Our patient remains in good condition one year later.Although inflammatory pseudotumor of the liver is usually a benign process, controversy regarding its management still exists. With this case report we review the existing literature and consider hepatectomy as a safe treatment approach. Inflammatory pseudotumors (IPTs) are reactive conditions that occur in many organs. They have been reported in the lung, which is the most common site of occurrence, central nervous system, major salivary glands, kidney, liver, omentum, ovary, larynx, urinary bladder, breast, pancreas, spleen, lymph nodes, skin, soft tissues, orbit etc. . The firThe literature survey to date indicates 130 publications with 289 cases of hepatic IPTs -6. The pWe report on a 55-year-old Caucasian man, who presented with a three-day history of right upper quadrant abdominal pain. He also complained of anorexia and weight loss (5 kg) in one month. He had no fever or night sweats and he presented without jaundice. Our patient was submitted to a chest X-ray, ultrasonography (USG) of the abdomen, triple-phase contrast computed tomography (CT) and magnetic resonance imaging (MRI) of the abdomen. As part of the metastatic work-up he was also submitted to a chest CT, esophagogastroscopy and colonoscopy. A laboratory blood test for serum albumin, serum total bilirubin, alanine aminotransferase, aspartate aminotransferase, prothrombin time, hepatitis B surface antigen, and antibodies to hepatitis C, serum alpha-fetoprotein (AFP), carcinoembryonic antigen and carbohydrate antigen 19-9 (CA 19-9) were obtained. An abdominal CT and MRI showed a well-defined heterogeneous mass situated in his right hepatic lobe measuring 4.0 cm × 4.0 cm  cases a percutaneous tumor biopsy provided the correct diagnosis. These patients were treated with antibiotics and/or corticosteroids with complete resolution of the lesion; however, some of these lesions have recurred . In contA further problem suggested in the literature, besides recurrence, is the potential malignant transformation of IPTs some years after hepatectomy. It has even been reported that some patients died of this disease. In particular, the authors report that these patients developed non-Hodgkin lymphoma, hepatic sarcoma and recurrence of IPTs with multiple hepatic masses and extension to the thorax some years later ,12.Regarding the current case, liver biopsy was not used as a diagnostic tool due to the strong suspicion of the lesion's malignant character, provided by the imaging findings. While liver biopsy has an undoubted role in the investigation and management of liver metastases of unknown origin, it plays a doubtful and probably dangerous role in patients with a solitary hepatic mass with high probability of malignancy . Needle A fairly uncommon clinical entity was presented. IPTs are often difficult to distinguish from hepatic malignant tumors. The safety of hepatectomy of non-cirrhotic patients has increased over the past 20 years, with mortality converging to 0%, therefore surgical resection should be considered as treatment of choice in such cases . This apWritten 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.AN wrote the manuscript. DK reviewed the literature and corrected the manuscript. DM performed the pathology tests of the specimen. KT interpreted the CT and MRI. AD corrected the manuscript and DV conceived the idea and corrected the manuscript. All authors have read and approved the final manuscript.


Central pontine myelinolysis is a demyelinating disorder characterized by the loss of myelin in the center of the basis pontis usually caused by rapid correction of chronic hyponatremia. The clinical features vary depending on the extent of involvement. Demyelination can occur outside the pons as well and diagnosis can be challenging if both pontine and extrapontine areas are involved. We herein report a case of myelinolysis involving pons, lateral geniculate bodies, subependymal region, and spinal cord. To the best of our knowledge, this case represents the second case of spinal cord involvement in osmotic demyelination syndrome and the first case of involvement of thoracic region of spinal cord. Central pontine myelinolysis (CPM) is a demyelinating disorder characterized by the loss of myelin in the center of the basis pontis, usually caused by rapid correction of chronic hyponatremia. On rare occasions, demyelination occurs outside the pons and is termed extrapontine myelinolysis (EPM). The term osmotic demyelination syndrome (ODS) refers to demyelination caused by changes in serum osmolality and may result in both pontine and extrapontine myelinolysis. Known risk factors for this condition include alcoholism, malnutrition, systemic medical disease, liver transplantation, and rarely, hemodialysis –3. We reA previously healthy 74-year-old Caucasian male presented to our facility for evaluation of fluctuating mentation, vision loss, and lower extremity weakness of three months duration. Four months prior to admission, he had frequent bouts of nausea and vomiting, resulting in weight loss. Following an episode of severe hematemesis, he received fluid resuscitation and blood transfusion at an outside facility. An esophagogastroduodenoscopy revealed peptic ulcer disease.A few days after his infusion, he became intermittently confused and started complaining of double vision. After a few days, he lost vision in his right eye and became profoundly weak on both legs. His left eye lost vision shortly after.His neurologic examination showed a confused, awake, and elderly man. His vision was limited to light perception in his right eye and finger counting in his left eye. Strength in both lower extremities was 1-2/5. A sensory level to pin prick was noted below the T10 dermatome and vibration and proprioception were intact. He was areflexic and had a right extensor plantar response.Magnetic resonance imaging (MRI) of his brain and entire spine without and with contrast was performed. This study revealed fluid attenuated inversion recovery (FLAIR) hyperintense lesion in the pons, along the walls of the lateral and third ventricles, and in the brainstem. There was restricted diffusion in the pons. On T1 postcontrast imaging, mild enhancement was noticed along the walls of the lateral ventricles. MRI of the spine revealed intramedullary T2 hyperintensity extending from T2 to T10 level . Vitamin B12, Thiamine, and serum NMO antibody titers were normal. CT scan of chest, abdomen, and pelvis was unremarkable. A whole body PET scan was performed, which did not reveal any abnormality.Neurosurgery was consulted for a biopsy, but the lesions were considered inaccessible. The patient was empirically started on 3 days of methylprednisolone but did not result in any clinical improvement. Due to continued deterioration, he was discharged to hospice where he passed away two weeks later.An autopsy was performed, revealing multiple areas of destructive lesions in the cerebrum, the lateral geniculate bodies, the caudate, both thalami, subependymal areas of the temporal horn of lateral, third, and fourth ventricles, the brain stem, and the spinal cord. The largest grossly identifiable lesion in the pons showed a roughly triangular shaped area of paleness, well demarcated from the surrounding tissue. Luxol fast blue combined with periodic acid schiff stain revealed pale staining, indicating a lack of myelin in the lesion. Immunohistochemical stain for neurofilament only demonstrated a mild to moderate loss of axonal fibers. The examination of the spinal cord revealed similar appearing lesions, mainly involving central regions of the cord, including gray matter. The lesion in the lateral geniculate bodies and subependymal regions also shared similar morphologic characteristics to the pons Figures  and 4. TAdams and colleagues first described CPM as an entity mostly affecting alcoholics and malnourished in 1959 . In theiOsmotic demyelination may occur in certain conditions such as alcoholism, malnutrition, after prolonged diuretic use, prolonged vomiting, burns, chronic psychogenic polydipsia, after liver transplant, and rarely, after pituitary surgery and after urological surgery/gynecological surgery, especially those requiring glycine infusions . ODS hasSymptoms of CPM are typically biphasic. Initial hyponatremia causes encephalopathy or seizures with symptoms of CPM being delayed for two to six days after correction of hyponatremia , 7. ClinDetection of ODS with the help of imaging is superior with an MRI of the brain than with a computed tomography scan. Findings on the MRI can develop as late as 4 weeks after the onset of symptoms, and imaging findings can even be present in asymptomatic individuals . MRI finThere were some unusual features in our case; namely, the patient did not have a history of alcoholism, liver disease, severe electrolyte disturbances, or any other major medical co-morbidities prior to his neurological deterioration. The most likely cause of osmotic demyelination was the aggressive correction with fluids when he had a bout of hematemesis. In our literature review, we found one case report of cervical cord involvement in ODS in a patient with pancreatitis  and anotThis case demonstrates the need to consider EPM as a differential diagnosis in cases where patients are at risk to develop ODS and their imaging suggests neurological damage affecting different parts of the brain and spinal cord, since ODS damage may not necessarily just be limited to the pons.