Juvenile angiofibroma is a rare benign lesion originating from the pterygopalatine fossa with distinctive epidemiologic features and growth patterns. The typical patient is an adolescent male with a clinical history of recurrent epistaxis and nasal obstruction. Although the use of nonsurgical therapies is described in the literature, surgery is currently considered the ideal treatment for juvenile angiofibroma. Refinement in preoperative embolization has provided significant reduction of complications and intraoperative bleeding with minimal risk of residual disease. During the last decade, an endoscopic technique has been extensively adopted as a valid alternative to external approaches in the management of small-intermediate size juvenile angiofibromas. Herein, we review the evolution in the management of juvenile angiofibroma with particular reference to recent advances in diagnosis and treatment. Juvenile angiofibroma (JA) is a benign vascular neoplasm which affects young males between 9 and 19 years of age and accounts for 0.05% of all head and neck tumors . Glad anHistologically, JA is a pseudocapsulated lesion characterized by an irregular vascular component composed of numerous blood vessels of different calibers embedded in a fibrous stroma, rich in collagen and fibroblasts. Vessels are slit or dilated, organized in clusters, without elastic fibers in their wall, and the muscular lining is incomplete in large vessels, and totally absent in the smaller ones. Mitotic figures are rare , 6 Figu. Since the 19th century, there is considerable debate concerning the fibrous or vascular origin of JA. Because of its extensive vascularization, several authors have considered the hypothesis of vasoproliferative malformation: Sternberg  and HubbThe observation that JA typically arises in adolescent males and that the lesion frequently regresses only after full development of secondary sex characteristics provided the evidence of hormonal influence on JA growth , 15. In Many studies have demonstrated numerous chromosomal alterations in patients affected by JA. Gains at chromosomes 4, 6, 8, and X and losses on chromosomes 17, 22, and Y are the most frequent chromosomal abnormalities detected , 20–23. β-catenin gene alterations are frequently detected in JA [β-catenin, which play a role in cell-cell adhesion and in the Wnt signaling pathway as a transcriptional activator. Nuclear accumulation of mutated β-catenin suggested that APC/β-catenin pathway might be involved in JA pathogenesis. Moreover, β-catenin can act as coactivator of androgen receptors and consequently increase tumor androgen sensitivity, which might explain why JA develops in adolescent males [The observation of an increased prevalence of JA in patients with familial adenomatous polyposis (FAP) suggested a possible association between these two pathologies . Althouged in JA . The APCnt males .Schick et al.  and NagaRenkonen and colleagues  investigThe oncogenes Ki-ras, Ha-ras, and Her-2/neu have been investigated with no detection of mutations , 30.JA is considered to arise in the area of the sphenopalatine foramen; based on the results on CT or MR imaging, some authors consider that the lesion originates in the pterygopalatine fossa at the level of vidian canal aperture . The groTypical symptoms for JA are progressive unilateral nasal obstruction (80–90%) with rhinorrhea and recurrent unilateral epistaxis (45–60%), and thus these complaints in a male adolescent should immediately generate suspicion. Headache (25%) and facial pain may arise secondarily to the blockage of paranasal sinuses, or impairment of Eustachian tube function with unilateral secretory otitis media, respectively. Tumor extension into the sinonasal cavity can cause chronic rhinosinusitis. Proptosis and alteration of the vision clearly indicate an involvement of the orbit. Swelling of the cheek, neurologic deficits, alteration in olfaction, rhinolalia clausa, and otalgia are also possible .Given the presenting symptoms, the patient should be examined by nasal endoscopy which usually shows a large, lobulated mass behind the middle turbinate filling the choana with a smooth surface and clear signs of hypervascularization . Since epidemiologic and endoscopic findings are typical, biopsy is absolutely contraindicated because of a considerable and undue risk of massive hemorrhage .Imaging techniques after contrast enhancement (MSCT and/or MR) are crucial to confirm the clinical suspicion pattern of vascularization and to assess the extension of the lesion. The diagnosis on imaging is based on three factors: the site of origin, hypervascularization after contrast enhancement, and patterns of growth , 32. ThePreoperative identification of blood supply is a crucial finding to select the most appropriate surgical strategy. Although angio-MR may help in the vascular assessment, the complete map of all feeders requires digital subtraction angiography. Typically the JA receives vascular supply via the external carotid system and particularly from internal maxillary, ascending pharyngeal, and vidian arteries . VasculaPreoperative embolization is recommended by most authors –40 as a Different staging systems based on tumor extension have been proposed to stratify patients with the intent of easing comparison between different series. Over the years, several authors have modified and adapted staging systems based on advances in diagnostic and treatment techniques. Since 1981, when the first staging system was introduced by Sessions et al. , many otAlthough several nonsurgical methods have been proposed, surgery is unanimously considered the treatment of choice for JA. In the last two decades, the surgical approach to the lesion has considerably evolved mainly in relation to the indication of endoscopic techniques. Transpalatal, transpharyngeal, transfacial through lateral rhinotomy, midfacial degloving, and Le Fort I osteotomy, other than infratemporal and subtemporal lateral approaches , 57, 58 In the 1990s, several authors reported their first experience of transnasal endoscopic resections for early stage JA, demonstrating the feasibility of this procedure and recurrence rates similar to that observed with external approaches, in addition to lower risk and morbidity , 60–66. The first surgical step when the surgeon approaches a JA endoscopically is to expose the tumor as extensively as possible through a middle turbinectomy, ethmoidectomy, wide antrostomy and sphenoidotomy, and resection of the posterior third of the nasal septum, which enhances the exposure of the nasopharyngeal portion of the lesion. The posterior wall of the maxillary sinus has to be resected as far lateral as dictated by the lateral extension of the lesion into the pterygopalatine and/or infratemporal fossae. For JA largely involving the infratemporal fossa, the surgeon can improve lateral exposure through a so-called Sturmann-Canfield maxillectomy, which provides resection of the anteromedial corner of the maxillary sinus . An endoBecause of its high degree of vascularization, bleeding during surgery is a crucial topic. Some studies compared the blood loss between endoscopic and external approaches, showing a lower loss in endoscopic surgery , 79. HowTo better control bleeding during the procedure, several authors have proposed the use of diode laser, KTP laser, or ultrasonic scalpel , 80–82.Based on the experience by Kania et al. , Lund etAlthough comparison between external approaches and endoscopic techniques is biased by the different staging systems and follow-up strategy adopted, recently Wang et al.  observedCurrently, the results of the two major series  of JA reIn a recent study on 95 JA, Sun et al.  identifiThe management of residual disease, especially when located intracranially or with a relationship with critical structures such the ICA, cavernous sinus, and the optic nerve, remains a source of discussion. Certainly, close survey with enhanced MR or MSCT is strongly indicated to evaluate the growth rate, and consequently, the need for surgical revision. Önerci et al.  prefer aThe use of radiation therapy (RT) in JA is still debated for the reported risk of sarcomatoid transformation  or radioThe use of chemotherapy in treatment of JA is supported by only a few reports –104. In Several studies on hormone pathogenesis have extensively demonstrated the hormonal dependence of this tumor, suggesting a promising role of estrogen or androgen receptor blockers in its treatment , 15–19. Juvenile angiofibroma is a pathology that should be included in the differential diagnosis of unilateral nasal obstruction, associated or not with epistaxis, especially in young adolescent males. The finding at nasal endoscopy, which is the first step in the diagnostic algorithm, of a hypervascularized lesion occupying the posterior half of the nasal fossa should immediately raise suspicion. Morphologic imaging confirms the diagnosis. Endoscopic surgery after embolization has been demonstrated to be a viable alternative to external techniques for the management of small-intermediate size JA. Resorting to external anterior or lateral approaches is still recommended in JAs encasing the ICA or with a massive feeder contribution from it, or in the rare instances of intradural spread.


Primary carcinoma of the vermiform appendix is a rare disease with few clinical symptoms. Accordingly, preoperative diagnosis of appendiceal cancer is challenging because of the lack of specific symptoms. We herein report a case of appendicular adenocarcinoma found unexpectedly during laparoscopic surgery in a 69-year-old Japanese female patient diagnosed with serous papillary adenocarcinoma, in order to determine whether optimal cytoreduction could successfully be achieved at the time of primary surgery. We performed diagnostic laparoscopic surgery in order to make a correct diagnosis based on the histological tissue. The vermiform appendix was found to contain a tumor measuring 1.5 cm wide and 4.5 cm long. Laparoscopic appendectomy, partial omentectomy, and partial resection of the lesion in the peritoneum were performed. The histological diagnosis was mucinous adenocarcinoma of the vermiform appendix, and the stage was T4NxM1. The patient received adjuvant chemotherapy with mFOLFOX 6 . She achieved stable disease and was alive with disease eleven months after surgery. We therefore recommend that gynecologists should not rule out the possibility of appendiceal cancer, even in cases with preoperative findings similar to those of serous papillary adenocarcinoma of the peritoneum with peritoneal disseminated tumors. Primary carcinoma of the vermiform appendix is a rare disease with few clinical symptoms . ConsequThe patient was a 69-year-old gravida 3 and para 3 Japanese female. She presented to another hospital with lower abdominal distension. She had undergone total abdominal hysterectomy with left salpingo-oophorectomy due to leiomyoma of the uteri at another hospital at 42 years of age, and her menstrual history included menopause after surgery. Colonoscopy and an upper gastrointestinal endoscopic examination revealed no evident primary lesions. On transvaginal ultrasonography, massive ascites was observed above the bladder and pouch of Douglas. The right ovary could not be detected. Magnetic resonance imaging (MRI) showed massive ascites and nodular/irregular thickening of the mesentery and peritoneum ; howeverComputed tomography showed massive ascites and a small mass suspected in the right normal ovary. A PET-CT scan demonstrated elevated FDG uptake in the areas of nodular/irregular thickening in the mesentery and peritoneum, although elevated FDG uptake was not observed in the appendix. The following tumor markers were measured: CA125: 116.3 U/mL , CA19-9: 380.1 U/mL , and CEA: 12.1 ng/mL . Based on these results, we suspected a diagnosis of serous papillary adenocarcinoma of the peritoneum as a preoperative diagnosis and planned to perform diagnostic laparoscopic surgery with a biopsy in order to make a correct diagnosis based on the histological tissue and determine whether optimal cytoreduction was likely to be achieved at the time of primary surgery.Diagnostic laparoscopic surgery was performed using a 4-port technique in the Trendelenburg position. Briefly, a 12 mm balloon trocar  used with a 0-degree, 10 mm laparoscope was inserted under direct visualization (open laparoscopy) through an intraumbilical incision of 1.5 cm. Three lateral 5 mm trocars were inserted  for the ancillary instruments. A massive amount of yellowish serous fluid was detected in the peritoneal cavity . After cThe specimen was resected without breaking the capsule. Subsequently, laparoscopic partial omentectomy and partial resection of the lesion in the peritoneum were performed to evaluate the site of the primary lesion. To prevent port-site metastasis, the specimens were placed in a Memobag  before being removed from the patient . As the The specimen of the vermiform appendix was found to be infiltrated by macroscopically solid areas and columnar and mucin-producing atypical cells that formed a ductal structure Figures . The omeThe patient received adjuvant chemotherapy with mFOLFOX 6  for 12 cycles. She achieved stable disease and was alive with disease 11 months after surgery.Primary adenocarcinoma of the appendix is a rare neoplasm of the gastrointestinal tract that constitutes approximately 0.5% of all gastrointestinal tract tumors . There aPeritoneal carcinomatosis observes approximately 35% of ovarian carcinomas. In patients with disease thought not to be optimally resectable, the use of neoadjuvant chemotherapy to reduce the tumor load can allow for interval surgical debulking . In caseAccordingly, laparoscopy has benefits in diagnosing serous surface adenocarcinoma, advanced ovarian carcinoma, and adenocarcinoma of unknown origin involving massive ascites and peritoneal disseminated tumors, such as those observed in the present case. In addition, in advanced cases with clear preoperative examination findings, performing laparoscopic surgery is not necessarily contraindicated.We therefore recommend that gynecologists should not rule out the possibility of appendiceal cancer, even in cases with preoperative findings similar to those of serous papillary adenocarcinoma of the peritoneum with peritoneal disseminated tumors.


Renal cell carcinoma (RCC) is a tumor known for its unusual presentations and high rate of metastasis. Metastasis to lung, liver, bone and brain are common, but to skeletal muscle(SM) is very rare. Because only 11% of the RCC metastases to SM present after 10 years of initial presentation, there is no general consensus for its annual surveillance.We report a case of a 58 year old male with a history of RCC, initially diagnosed 11 years ago, who presented with a large SM mass. A large mass measuring more than 25 cm was located in left posterior thigh and was present for more than a year. It initially was diagnosed as a large blood clot and was treated with warfarin for more than 6 months. Clinical work up including FDG-PET/CT and MRI raised the possibility of a tumor, but a negative biopsy made the diagnosis uncertain. Because of high suspicion for a tumor, patient underwent a complete resection of the mass.The resected mass measuring 28 × 18 × 7 cm was detailed as the largest skeletal muscle metastasis from RCC ever reported.This case emphasizes the importance of maintaining a high suspicion for metastasis even in less common metastatic sites mainly in patients with a history of RCC. It also highlights the importance of annual surveillance for metastasis in patients with RCC even after 10 years of initial presentation using FDG-PET/CT. Skeletal muscle is a rare site of metastasis accounting for <1% of metastasis.(Pompo et al. We describe an unusual presentation of skeletal muscle metastasis from RCC and emphasize on the annual surveillance for metastatic RCC even after curative nephrectomy.We present a 58 year old male with an unusual posterior thigh mass for more than a year. Patient had a past medical history significant for RCC, initially diagnosed at stage II, 11 years ago followed by left nephrectomy. Patient also had metastasis to tail of the pancreas and tip of spleen 6 years ago which was followed by total resection of pancreas and spleen. Patient was followed up for RCC and was last seen 2 years ago when his PET/CT showed slightly increased hypermetabolic area in the biceps femoris muscle which was interpreted as a muscle injury secondary to the rarity of the metastasis to the skeletal muscle from RCC. Patient during the current follow up visit developed a large mass in the posterior side of the thigh which was present for more than a year. As per patient, the mass initially was diagnosed as a blood clot on venous doppler, for which he was treated by his primary care physician with warfarin for more than 6 months. However, the mass progressively increased in size. Patient did not mention of any other constitutional symptoms. On physical examination, a painless, tense mass along the length of biceps femoris muscle measuring more than 25 cm was found in the posterior aspect of the left thigh. The mass was hypervascular and numerous varicosities of different sizes were noticeable on the surface. Patient was imaged using FDG-PET/CT which showed hypermetabolic activity with an uptake value of 3.8 to 4.1 in biceps femoris muscle with multiple serpiginous vessels throughout the tumor, in accordance with a large cavernous hemangioma or an angiosarcoma. This was followed by MRI to better understand the morphology of the tumor, demonstrating a mesenchymal component in an encapsulated mass, raising the possibility of a liposarcoma or an angiosarcoma. A core tissue biopsy was done which demonstrated well defined adipose tissue but because of high suspicion for malignancy, patient underwent preembolization followed by surgical resection. A 28 x 17 x 7 cm resected mass was determined to be metastasis from his primary RCC (Figures Patient on his follow up with a whole body FDG-PET/CT was also found to have metastasis to his lateral ventricle in the brain from the RCC.Renal cell carcinoma accounts for approximately 3% of all adult tumors (Sountoulides et al. It has been reported that approximately 0.4% of RCC metastasizes to skeletal muscle (Ali et al. RCC commonly metastasizes to soft tissues as a solitary soft tissue deposit developing any time between 6 months to 19 years, with the greatest risk in the first 5 years after initial presentation (Ali et al. Differentiating the unusual skeletal metastasis from other tumors still remains a diagnostic challenge and is pivotal because of the great differences in their treatment and prognosis (Pompo et al. Location, size, and lack of pain in our patient were suspicious for diagnosis of soft tissue sarcomas (Pompo et al. FDG-PET/CT is an effective tool in surveillance of RCC, known for its distant and unusual sites of metastasis (Picchio et al. We recommend maintaining a high level of suspicion for metastasis from RCC in a musculo-skeletal mass in a patient with RCC, even after curative nephrectomy. Because of advancement in treatment modalities, resulting in better survival rates, late presentation of the metastasis in RCC is increasing. We therefore recommend continuing intensive follow up for surveillance of the metastasis in RCC mainly in high grade tumors even after 10 years of initial presentation of the tumor or long disease free interval.


Synovial sarcoma is a rare malignant neoplasm of soft tissue that typically arising near large joints of the upper and lower extremities in young adult males. Only 3% of these neoplasms have been found to arise in the head and neck region. To our knowledge, there are limited reports in the literature of this neoplasm in the cervical spine.A case of biphasic synovial sarcoma of the cervical spine is reviewed. A 29 year-old male presented with pain on the left side of the cervical spine. Physical examination revealed a global loss of cervical motion and large, palpable mass in the left paravertebral area. The long-delayed Magnetic Resonance (MR) scan revealed a soft tissue mass measuring 8.3 centimeters (cm) × 5.7 cm that was surgically removed. A malignant biphasic synovial sarcoma was diagnosed on pathologic examination.The clinical and imaging findings of an atypically located synovial sarcoma are reviewed. This case report emphasizes the consequences of a limited differential diagnosis, prolonged treatment and the failure to perform timely diagnostic imaging in the presence of a paraspinal mass. Synovial sarcoma is a seldom encountered, aggressive malignant neoplasm of soft tissue that typically arises near large joints of the upper and lower extremities in young adults. Synovial sarcomas account for 7-10% of all soft-tissue sarcomas  and justThis paper describes the clinical, radiological and pathological findings of a synovial sarcoma that was located in the lower cervical paravertebral space. Although the radiological and clinical features of a typically located synovial sarcoma are documented in the literature, our review of the literature reveals limited reports of synovial sarcoma arising from the cervical spine .A 29 year-old male presented with muscle discomfort and pain in the posterior left cervical spine, especially after weight lifting. There was no history of recent trauma. Five years prior to presentation, the patient had sustained cervical injuries from a motor vehicle accident. The patient's motor vehicle related cervical spine complaints resolved with manipulation and physical therapy shortly after the accident. The patient recently returned to care and a regional orthopedic and neurological examination was performed with findings of "myofascial trigger points on the left levator scapulae muscle". The patient was diagnosed with "myofascial pain syndrome of the left levator scapulae" and was placed on a course of care that consisted of manipulation, post-isometric relaxation, stretching and "post-facilitated stretching once the trigger points are resolved." No initial imaging studies were performed. The presenting size of the paravertebral "trigger point" was not documented in the record.The patient underwent a course of chiropractic care, which totaled 24 treatments over a 13-month period. The clinical record noted both positive and negative subjective responses to the conservative care. Multiple comments in the chart notes indicated the "trigger point" and "swelling" were worsening during the course of care, but these observations of an enlarging mass were not accompanied by any change in treatment, re-evaluation or additional investigation with any form of diagnostic imaging. The patient eventually discontinued care as the paravertebral mass had steadily grown and was now clearly visible on visual inspection of the area. The subsequent treating clinician ordered a MR scan.MR images with pre and post gadolinium axial and coronal T1 weighted images revealed a complicated mass extending from the C3-C4 level to the T1-T2 level. The coronal MR revealed a septated kidney bean shaped mass with a large portion demonstrating elevated signal intensity on T1 weighted images . One cell type was spindle and the other was epithelial. The spindle cells were similar to fibrosarcoma. The epithelial cells presented as focal glandular formations with clusters and trebeculae. Calcification and ossification, often seen on CT imaging of these tumors, were noted in the pathological study but were never visualized in the imaging studies. The final pathological diagnosis was "synovial sarcoma, biphasic type, intracystic."The prevailing therapeutic approach to high-grade soft tissue sarcomas is wide surgical resection followed by radiation, chemotherapy or both . The patSynovial sarcoma is a malignant neoplasm of soft tissue that typically arises near large joints of the upper and lower extremities in the young adult male, particularly the knee; however, they do not arise from synovial tissue ,8,9 but Presenting clinical symptoms vary according to the size and location of the tumor. Those tumors arising in an extremity may present initially with swelling, pain or tenderness. Limitation in motion may be noted if the tumor is located near a joint. The non-specific nature of the symptoms may initially be interpreted as more commonly encountered soft tissue entities such as bursitis and myositis. The increasing size of the tumor also has the ability to compress nerves and result in the gradual onset of neurological deficits. A high degree of clinical suspicion, along with the observation of gradually developing mass should prompt the use of diagnostic imaging even in the absence of a history of trauma.Synovial sarcoma of the spine is quite uncommon and early diagnosis may be difficult without advanced imaging. The tumor may cause a variety of symptoms, again depending on the size and location of the mass. Neurological compromise is also possible with tumors located near the spine. A case of paravertebral synovial sarcoma in the lumbar spine was noted to produce a grade III weakness in dorsiflexion in the right great toe and decreased sensation of the L4-5 dermatome . A palpaSynovial sarcoma occurs in 2 histological subtypes: the biphasic form contains elements of both epithelial and spindle cells and the monophasic type contain only spindle cells .Synovial sarcomas may aggressively grow and imaging studies have shown they vary in size between 2 and 9 cm ,12. DeteSynovial sarcomas are usually treated aggressively with wide excision with negative margins, often including removal of adjacent muscle groups and even total amputation ,13. LimiThis case demonstrates the ever-present potential for an uncommon condition to present in an atypical location in the ambulatory outpatient setting. This patient would have benefitted from earlier diagnostic imaging and consultation with other practitioners when the patient began to develop a paraspinal mass. Although rare, synovial sarcomas and other forms of soft tissue tumor should be included in the differential diagnosis of paraspinal masses in patients, irrespective of their response to conservative care.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 they have no competing interests. SMF was involved in this case as a consultant for the patient after the tumor had been resected.SMF conducted the initial review of the case and prepared the first draft of the manuscript. MJS participated in the conception of the report, the revision and coordination of the final manuscript. Both authors read and approved of the final manuscript.


To present rare benign orbital tumors with bone destruction in children who could not be diagnosed pre-surgically and may simulate malignant ones.A retrospective review of cases. Clinical, operative and pathological records in all children with a diagnosis of benign orbital tumors who showed remarkable bone destruction at a tertiary Ophthalmic Center in China between Jan 1, 2000 and Dec 31, 2009 were reviewed. All patients had definitive histopathologic diagnosis.Eight patients with benign orbital tumors showed obvious bone destruction, including six cases of eosinophilic granuloma, one case of leiomyoma and one case of primary orbital intraosseous hemangioma. Among them, three patients were females and five patients were males. Tumors were unilateral in all cases, with both the right and left side affected equally. Age ranged from 3 to 7 years (mean 4.1 years). Symptom duration ranged from 1 to 5 weeks (mean 4.8 weeks). Eyelid swelling and palpable mass were the most common complaint. There was no evidence for multifocal involvement in cases with eosinophilic granuloma. Among six patients with eosinophilic granuloma, two were treated with low dose radiation (10 Gy), three received systemic corticosteroid and one was periodically observed only after incisional biopsy or subtotal curettage. There was no postoperative therapeutic intervention in the two patients with leiomyoma and intraosseous hemangioma. All eight patients regained normal vision without local recurrence after a mean follow-up time of 32.8 months.Benign orbital tumors such as isolated eosinophilic granuloma, leiomyoma and primary orbital intraosseous hemangioma may show remarkable bone destruction. In childhood, a large number of lesions can potentially cause bone destruction of the bony orbit with or without a solid soft tissue component. The most common benign tumor is the dermoid inclusion cyst, others such as fibrous dysplasia, juvenile ossifying fibroma are relatively common too Authors performed a retrospective review of patients with a diagnosis of benign orbital tumors which showed remarkable bone destruction in computed tomography scan and were treated at Zhongshan Ophthalmic Center, Sun Yat-sen University, Guangzhou, China between Jan 1, 2000 and Dec 31, 2009. The ethics committee of Zhongshan Ophthalmic Center approved this retrospective study and our paper has been conducted according to the principles expressed in the Declaration of Helsinki. The committee specifically waived the need for consent. The persons concerned  have seen this manuscript and figures and have provided written consent for publication. The clinical, operative and pathological records were reviewed, and orbital CT scans were examined. All patients had definitive histopathologic diagnosis, with all histologic patterns reexamined by two observers without knowledge of the previous diagnosis and clinical outcome. Among the inclusion criteria were that all patients had treatment by a single surgeon (JH Yan); systemic evaluation by a pediatric oncologist, including a complete medical history and physical examination, laboratory studies, and a bone scan; and minimum follow-up of 12 months.The data collected in this study included the general data such as the patient's age, sex, the duration of orbital lesion at presentation. The ocular data included the affected orbit, laterality, symptoms , signs . Tumor data included orbital location , configuration , size, margin , quality , tenderness , tissues or spaces involved, imaging findings, histopathologic examination.All patients had a presumed diagnosis of orbital malignancy before surgery. Usually, initial management involved incisional biopsy or subtotal curettage. This generally was accomplished through anterior orbitotomy. Tissue was submitted for pathological evaluation. After pathological diagnosis, the postoperative treatment included close follow-up observation, low-dose irradiation, or systemic corticosteroid.Eight patients met the inclusion criteria for this study . Among tThe histopathological examinations of specimens were suggestive of eosinophilic granuloma in 6 cases, and leiomyoma and primary orbital intraosseous hemangioma in 1 case respectively. Light microscopic examination of formalin-fixed specimens showed fairly uniform findings in patients with eosinophilic granuloma. The tumor tissues were comprised of pathologic Langerhans cells, eosinophils, scattered lymphocytes, plasma cells, and multinucleated giant cells. Immunohistochemical stainings for CD68, vimentin and S-100 were strongly positive in all 6 cases. In patient with leiomyoma, the histopathologic examination showed that the tumor was composed of spindle-shaped, benign-appearing cells organized in fascicles or loosely arranged in a myxoid stroma. In immunohistochemical staining, the specimen was positive for vimentin and alpha-smooth muscle actin, negative for S-100. The histopathologic finding of primary orbital intraosseous hemangioma was straightforward, consisting of the thin-walled blood vessels which are closely clustered and separated by normal bony tissue –3. AfteFollowing definitive diagnosis, in six patients with eosinophilic granuloma, two patient were treated with fractionated external beam radiation to total doses of 10 Gy, three patients received systemic corticosteroid and one patients was periodically observed only. Authors usually choose the course of systemic corticosteroids as follows: Starting drug was dexamethasone 0.2 mg/kg per day for 3 days. Then they used oral prednisone 1 mg/kg per day for two weeks, and tapered slowly for two to three months. They did not use local intralesional corticosteroids for their patients. There was no postoperative therapeutic intervention in the two patients with leiomyoma and primary orbital intraosseous hemangioma. To authors' surprise, all eight patients were doing quite well after treatment. None of the 6 patients with eosinophilic granuloma had local recurrence, other foci of LCH, or other serious illness. The 2 patients with leiomyoma and primary orbital intraosseous hemangioma had no recurrence too. All 8 patients regained normal vision after a mean follow-up time of 32.8 months (range 18 months to 60 months).Both benign and malignant masses of the orbit can have bone destruction Eosinophilic granuloma of the orbit is a subtype of Langerhans cell histiocytosis (LCH), an idiopathic reticuloendothelial proliferative disorder with clonal proliferative Langerhans cells. LCH embraces three main clinical subtypes (or syndromes): Unifocal eosinophilic granuloma disease, multifocal unisystem disease (Hand–Schüller–Christian syndrome) and multifocal multisystem disease (Letterer–Siwe syndrome). The isolated eosinophilic granuloma of the orbit is uncommon, with onset in the first or second decade, but predominantly in the 2–5 years age group. Males are affected twice as frequently as females Leiomyoma is a benign smooth muscle tumor that is most commonly encountered in the uterus, skin, and gastrointestinal tracts Primary intraosseous hemangiomas are rare, usually solitary, benign, slow-growing neoplasms, with more than 50% being found in the vertebra or skull. Primary orbital intraosseous hemangiomas are extremely rare, only 45 cases were reported in the English literature to date The differential diagnosis of benign orbital tumors with prominent bone destruction in children that simulate malignant tumors includes myofibroma, solitary fibrous tumors, haemangiopericytoma and so on. Orbit myofibroma typically occurs in childhood and presents as a firm, painless, well-circumscribed mass. Intraosseous occurance has been reported In summary, some benign orbital tumors, including isolated eosinophilic granuloma, leiomyoma and primary orbital intraosseous hemangioma may show remarkable bone destruction in children. Therefore, we should pay attention to these rare orbital benign tumors in the differentiation of tumors with bone destruction. The differential diagnosis includes myofibroma, solitary fibrous tumors, haemangiopericytoma and so on.


Department of Oncology, The Third Affiliated Hospital of Soochow University, Changzhou 213003, ChinaA patient with a seven-year history of recurrent metastatic hemangiopericytoma (HPC) was admitted. During his treatment, he received surgical resection, radiotherapy, radiofrequency hyperthermia and chemotherapy using combined doxorubicin, dacarbazin, vincristine, ginsenoside Rg3, and recombinant human endostatin. This synergistic method provides an encouraging model for treating HPC. HPC is rarely found in the liver in these reported cases. A case of HPC in the liver with recurrence and metastasis was reviewed in this report.Hemangiopericytoma (HPC) is a rare tumor of uncertain malignant potential. Approximately 1000 cases of HPC have been reported to date. This disease often occurs in the lower extremities (34.4%), retroperitoneum (24.5%), and meninges as well as in the head and neck 0TxN0M0).The patient was a 42-year-old man with a 7-year history of recurrent metastatic HPC. A CT scan during a physical examination in 2002 revealed a mass in the patient’s liver. He received a complete surgical resection of the mass that was pathologically and immunohistochemically confirmed as HPC , dacarbazine (DTIC), vincristine (VCR), ginsenoside Rg3, and recombinant human endostatin (Rh endostatin) for four 21-day cycles. ADM and VCR were administered on day 1, DTIC on days 2 to 6, Rh endostatin on days 0 to 6, and ginsenoside Rg3 on days 7 to 20. The patient also received synchronous radiofrequency hyperthermia in both lung lobes during the course of treatment.A CT scan after 2 cycles showed reduction of masses in the left kidney as well as stable lesions in the right kidney, both lungs, and liver. A CT scan after 4 cycles revealed a partial response in the left kidney . The excessive production of glucose of the tumor during the latter phase of the disease is related to the production of the insulin-like growth factor II by the lesion .A few characteristics of malignant HPC have been ignored. For instance, the tumor has typically been reported as a solitary lesion, and only a very few instances with multiple nodules have been observed This case indicated that multiple metastases in the lungs and the presence of NICTH may help diagnose HPC., providing the rationale for using anti-angiogenesis drugs, such as Rh endostatin and ginsenoside Rg3. HPC also expresses both the platelet-derived growth factor  and the vascular endothelial growth factor receptors .The most distinctive and consistent characteristic of HPC among sarcomas is its hypervascularity in vitro and can induce apoptosis . Previous studies have indicated that endostatin affects a network of potentially intersecting pathways that are important in the angiogenic phenotype. And ginsenoside Rg3 is a chemical trace component (MW 784.30 Da) of Panax ginseng . Many studies have shown that ginsenoside Rg3 can inhibit proliferation, infiltration, and metastasis of tumor cells . The chemical also presents anti-angiogenesis capable of controlling some tumors . Ginsenoside Rg3 has been proven useful in treating lung cancer in the Chinese version of National Comprehensive Cancer Network (NCCN) guidelines.Human endostatin specifically inhibits the proliferation and migration of capillary endothelial cells This case presented an encouraging synergistic treatment model with combination of several disciplines: surgery, radiation, intervention, anti-angiogenesis, and chemotherapy. The regimen of VCR/ADM/DTIC combined with the two anti-angiogenic drugs produced a marked response in terms of cancer cell inhibition. However, the present case cannot prove that the combined use of ginsenoside Rg3 and endostatin is necessarily effective.The present case is the first report wherein anti-angiogenic drugs are utilized for HPC treatment. This synergistic method actually provides an encouraging model for treating HPC although the effectiveness of single therapeutic modality remains uncertain.


Stafne bone defects (SBDs) are generally located in the lingual cortex, close to the mandibular angle. We report the occurrence of multiple SBDs in an asymptomatic patient, a 60-year-old man, referred to a radiology clinic to undergo examination for the purpose of implant planning. The case of multiple SBD presented here, probably the first reported in the literature, reinforces the hypothesis that some cases of SBD may be the result of a focal failure during the ossification of the mandible. Stafne bone defect (SBD), first described in 1942 .In addition to occurring more commonly in the posterior region of the mandible (posterior variant), SBD may also appear in the anterior region (anterior variant) and in the ascending ramus of the mandible (mandibular ramus variant) . NormallIt has been suggested that in a large number of cases of SBD the causal factor is mechanical pressure caused by the glandular tissue surrounding the lingual cortex of the mandible. This theory has been accepted because the radiolucent areas found on radiographs are associated with the submandibular and sublingual glands .Normal salivary gland tissue is the most common histological finding, suggesting a developmental origin in which a part of the submandibular gland was trapped in the lingual mandibular aspect . HoweverThis paper presents a rare case of multiple bone defects, one posterior and two anterior variants, confirmed by computed tomography.The asymptomatic patient, a 60-year-old man, was referred to a radiology clinic to undergo examination for the purpose of implant planning. The panoramic radiograph revealed no important alterations in the mandible . The patThe tomographic images clearly showed multiple cavitary defects, two in the anterior region (anterior variant) and one in the posterior region (posterior variant) Figures , and 4. Stafne  initiallPhilipsen et al.  affirmedMinowa et al.  considerBone erosion caused by lipoma is another causative factor hypothesized by Minowa et al. . This hyFor the case under discussion, we believe that the filling of the cavities with glandular and fat tissue shows that the neighboring tissues tend to occupy the bone defect, despite the fact that in most cases, the relationship between SBD and the salivary gland is unquestionable. In our opinion, the multiple occurrence of SBD described here, characterized by no evidence of cortex erosion and small-depth depression, confirms that in some cases, SBD may be the result of a focal failure of mandible ossification.


Primary sarcoma tumours of the prostate are rare and are classified, according to their histology, as stromal tumours of uncertain malignant potential (STUMP) and stromal prostatic sarcoma . We describe a case of a 71-year-old man that developed progressive urinary obstruction symptoms and was subjected to a transurethral prostatic resection (TURP). Histologically, there is a diffuse proliferation of epithelioid and spindle cells that showed rare atypical mitotic figures. Immunohistochemically, the neoplastic cells express diffusely CD34 and focally progesterone whereas no immunoreactivity was seen for cytocheratin, desmin, S-100, Bcl-2, chromogranin, CD117, and actin smooth muscle. A final diagnosis of low-grade prostatic stromal sarcoma (LG-PS) was made. This is a really rare neoplasm; in the literature, in fact, to our knowledge, only 6 cases are described and all of these were alive and free of disease at followup. Our patient too is free of disease at 15 months from the diagnosis. Malignant tumours of the prostate other than carcinomas are rare , among tPrimary sarcoma tumors of the prostate arise from specialized hormone-dependent mesenchymal cells  and are We describe a rare case of low-grade stromal sarcoma (LG-PS) of the specialised prostatic stroma, and we also review the literature concerning these tumours.A 71-year-old man developed progressive urinary obstruction symptoms and presented at our Hospital. Clinical examination suggested benign prostatic hyperplasia and a transurethral prostatic resection (TURP) was performed.Macroscopically, the excised tissue consist of many fragments of prostatic tissue weighing 30 grams.Microscopically, the histological specimens taken after TURP showed a diffuse proliferation of epithelioid and spindle cells, with a storiform and infiltrative growth pattern. There are not prostatic glands in the proliferation, no foci of necrosis, no vascular invasion. The neoplastic cells showed rare atypical mitotic figures in addition to moderate hypercellularity and moderate nuclear atypia with pleomorphism .Immunohistochemically the neoplastic cells characteristically express diffusely CD34 and focally progesterone whereas no immunoreactivity was seen for cytocheratin (clone AE1–AE3 and Cam 5.2), desmin, S-100, Bcl-2, chromogranin, CD117, and actin smooth muscle .We evaluated additional immunohistochemical markers for their potential diagnostic and therapeutic utility. Although p53 and Ki-67 showed a very low expression.On the basis of the morphological and immunohistochemical features, a final diagnosis of LG-PS was made.Spindle cell lesions of the prostate encompass a broad spectrum of benign and malignant processes. Some are characteristic of this organ and may be benign or malignant, including leiomyoma and leiomyosarcoma, rhabdomyosarcoma, inflammatory myofibroblastic tumour, solitary fibrous tumours, phyllodes tumour, and synovial sarcoma –7.Prostatic stromal tumors, arising from the specialized prostatic stroma, are rare and distinct tumours with diverse histologic pattern. In the past, these tumors have been reported under a variety of terms including atypical stromal hyperplasia, phyllodes type of atypical stromal hyperplasia, and cystic epithelial-stromal tumors . Now theBecause of their rarity and the nonspecific nature of clinical symptoms, their diagnosis is rarely suspected clinically.Histologically the diagnosis of HG-PS is based on observation of a neoplasm with a proliferation of epithelioid and spindle cells that showed different growth patterns . The neoplastic cells showed marked nuclear atypia, mitosis, and necrosis . LG-PS, In contrast to stromal sarcoma, the neoplastic nature of STUMPs is controversial. STUMPs may be considered neoplastic based on the observation that they may diffusely infiltrate the prostate gland and extend into adjacent tissues , 9.In most cases STUMP is not of an aggressive nature, occasional cases have been documented with an extension into adjacent tissues or recurrence after resection .The predominant pattern of STUMP consists of either normocellular or mildly hypercellular stroma with cytologically degenerative atypical cells associated with benign glands. The currently used classification of STUMP was first proposed in 1998 by Gaudin et al. , in whicThe epithelial component proliferations occurring within the STUMP, only recently described , is histIn the past, the histologically pattern of STUMP was often reported as benign prostatic hyperplasia (BPH) with atypia and in rare cases may be difficult to distinguish form stromal sarcoma . AlthougSometimes high- and low-grade prostatic stromal sarcoma was associated with STUMP. Herawi and Epstein, in fact, found that seven (14%) of the 50 stromal tumours of the prostate that they analyzed were STUMPs associated with sarcoma  [There are several difficulties with the histologic diagnosis of STUMP, LG-PS, and HG-PS; it may be difficult to differentiated LG-PS from STUMP, sometimes the tumour seemed histologically benign and only the presence of local infiltration or the presence of atypical mitotic figure was diagnostic of LG sarcoma.Prognosis and treatment of STUMP and sarcoma are also controversial. Factors to consider in deciding if to proceed with definitive resection for STUMP diagnoses on biopsy include patient's age, presence and size of the lesion, and extent of the lesion, on tissue sampling.Immunohistochemically, STUMP and stromal sarcomas characteristically express progesterone receptors (PR), and this finding is consistent with their arising from hormonally responsive mesenchymal cells of the prostate , 13.  Most of these tumors are positive for CD34 . Rarely,The primary prostatic sarcoma are really rare neoplasms. In the literature, to our knowledge, only 6 cases are described of LG-PS (age: 42 to 76, mean 59; see In the current literature only 16 cases are also reported of HG-PS –15.The HG-PS are more aggressive tumour, in fact only 6 patients of 16 cases, reported in the literature, were free of disease, 3 were alive with pulmonary metastases, 2 were alive with multiple metastases, and 3 died of disease and 2 were lost to followup –15.Our case of LG-PS is the seventh that was described in the literature, and our patient was free of disease at 15 months of followup.


Meningioangiomatosis (MA) is a rare hamartomatous lesion. Only six cases of cystic MA have been reported in the literature.We present a case of multicystic MA. A 21-year-old woman without any stigmata of neurofibromatosis type 2 presented with intractable seizures since 10 years. Brain magnetic resonance imaging revealed a well-defined, multicystic mass with heterogeneous signal intensity in the right temporal lobe. The patient underwent resection of the lesion and of the epileptogenic cortex under intraoperative electrocorticography (ECoG) assistance. Histopathological examination showed proliferation of perivascular cells that were arranged in a cuff pattern and were positive for vimentin, D2-40 and smooth muscle actin. Mutiple microcysts and enlarged perivascular spaces were present, which was similar to the structure of the arachnoid cavity. Hyalinized collagen fibers with round concentric acellular eosinophilic lamellae within areas of reactive gliosis were noted for the first time in MA. The patient was followed up without any clinical symptoms or recurrence for 2 years.MA may originate from arachnoid and vascular tissue trapped in the cortical parenchyma during brain development, and the cysts may have resulted from the gradual accumulation of cerebrospinal fluid in the perivascular spaces of the trapped tissue. Resection of the lesion and of the epileptogenic cortex is important not only for pathological diagnosis but also for seizure control, and intraoperative ECoG assistance is recommended. Meningioangiomatosis (MA) is a rare and benign congenital disorder that involves the cortex and the overlying leptomeninges and primarily affects children and young adults . It may A 21-year-old woman presented with a history of intractable focal evolving to bilateral convulsive seizures since the age of 10 years and complained of an increase in the frequency of the seizures in the last 5 months. Eleven years ago, she experienced a complicated febrile seizure with left limb convulsion and unconsciousness, which continued for 6 h; her mental status fully recovered after 24 h. Thereafter, generalized tonic-clonic seizures occurred almost every month, in spite of medical treatment with carbamazepine, phenobarbital and sodium valproate. She had no history of adverse perinatal events, developmental problems or head trauma. General physical and neurological examinations were unremarkable; she had no stigmata or family history of NF. Inpatient continuous video electroencephalography (EEG) revealed right temporal seizure foci with significant sharp wave activity and spike-and-slow waves complex during drowsiness and sleep. Magnetic resonance imaging (MRI) of the brain demonstrated a multicystic mass with low signal intensity on T1-weighted images and high signal intensity on T2-weighted images in the right temporal lobe  and perivascular cells, which resembled meningothelial or fibroblast cells, arranged in whorls and cuffs Figure A. MicrocTo determine the origin of the proliferative or lesional cells, immunohistochemical examination was performed using the Ventana BenchMark XT system . Antibodies against the following antigens were used: vimentin , smooth muscle actin , epithelial membrane antigen , cytokeratin , D2-40 , factor VIII (Maixin), CD31 , CD34 , H-cald , glial fibrillary acidic protein , neurofilament  and Ki-67 protein . The perivascular cells were mainly immunopositive for vimentin and D2-40, focally or weakly positive for SMA, EMA and CD34, and negative for CK, CD31, H-cald, GFAP and neurofilament (Figure MA is a rare and benign meningovascular hamartomatous condition that usually involves the cerebral cortex in the frontal or temporal regions, with varying involvement of the overlying leptomeninges. The lesions are often associated with NF2, although the majority of cases are sporadic . Sporadiet al. described the histopathological spectrum of MA, which can be broadly classified into predominantly cellular and predominantly vascular types [All MA lesions share common pathological features, namely, cortical vascular proliferation and perivascular cuffs of proliferative, spindle-shaped, meningothelial-like cells. Although approximately 120 cases of MA have been reported to date worldwide, only six cases of cysts within the MA lesion have been described in the literature (Table ar types . In our ar types ,6, patieThe mechanisms involved in the development of MA have not yet been elucidated. MA was considered a hamartomatous proliferation of meningothelial cells, blood vessels and fibroblasts in variable proportions . Howeveret al. reported that after surgical removal of the tumor, long-term seizures disappeared in 43% of patients, improvement occurred in 30% of patients and antiepileptic drug administration was required in more than 70% of patients [Although many cases of MA have been described, presurgical diagnosis remains difficult because the wide spectrum of clinical, imaging and electrophysiological features often impedes the clinical diagnosis ,18,19. Spatients . In patipatients . In our patients , which oThe present case demonstrates that MA may present as a hamartomatous lesion containing multiple microcysts in a patient with medically refractory seizures, and should be considered in the differential diagnosis of intracortical lesions, especially in young adults. It is possible that MA originates from arachnoid and vascular tissue trapped in the cortical parenchyma during brain development, and that the cysts result from gradual accumulation of CSF in the perivascular spaces that develop from the trapped tissue. Our data do not support a meningeal origin of MA, but rather suggest that pluripotent arachnoid cap cells differentiate into the various cell types found in MA. Resection of the lesion and of the epileptogenic cortex is important not only for pathological diagnosis but also for seizure control, and intraoperative ECoG assistance is recommended.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 of this journal.MA: Meningioangiomatosis; ECoG: Electrocorticography; NF: Neurofibromatosis; EEG: Electroencephalography; MRI: Magnetic resonance imaging; CSF: Cerebrospinal fluid; SMA: Smooth muscle actin; EMA: Epithelial membrane antigen; GFAP: Glial fibrillary acidic protein; HE: Hematoxylin-eosin.The authors declare that they have no competing of interests.Acquisition of data: PL, MG and ZW. Analysis and interpretation of data: YW and GC. Drafting of the manuscript: PL, GC and YW. Critical revision of the manuscript for important intellectual content: MG and ZW. All authors read and approved the final manuscript.The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1471-2377/14/32/prepub


Mesenteric cysts are not uncommon in pediatric age group but giant lymphatic cysts of mesentery are reported infrequently. This is a report of six years old female who had vague abdominal pain with distension for two years. Investigations revealed a large cystic mass in abdomen. On exploration a giant lymphatic cyst in the mesentery of transverse colon found. More than 1500 ml of milky fluid was drained. The cyst was unilocular and appeared to be the collection of lymph (chyle) between two leaves of the mesentery of transverse colon. It is postulated that trauma to or malformation of lymphatics at the root of mesentery might have lead to this pathology. Mesenteric cyst is a term applied to any cyst found in mesentery. Lymphatic cyst is a type of mesenteric cyst which is of lymphatic origin. It is usually a benign lesion. A closely related pathology is cystic hygroma which is also of lymphatic origin. This is usually found in the retroperitoneum. These are more common in small bowel mesentery but rarely reported in colonic mesentery  , 2.Lymphatic cysts usually contain pale yellow fluid. Chylous cysts are infrequently seen. Chylous cysts are classified as rare variants of mesenteric cysts. It constitutes 7.3% to 9.5% of all abdominal cysts .We report a case of giant mesenteric cyst at unusual location and with unique features.A six years old female child weighing 12 kg, admitted with history of gradual abdominal distension that was noticed by the mother for the last 2 years but did not seek any medical advice as there were no other associated complaints. With an increase of abdominal girth and vague abdominal pain, parents took the child to a nearby general practitioner who advised ultrasound and referred the child to our facility. On examination child appeared comfortable with abdominal fullness. Abdominal examination did not reveal a distinct mass though vague fullness was present oriented more in vertical direction. There was a definite feel of fluid thrill on palpation.Ultrasound done previously showed cystic mass in abdomen. With female gender a differential diagnosis included ovarian cyst in addition to mesenteric and omental cyst. CT scan abdomen was advised. This showed a large cystic mass occupying almost whole of the abdomen and located under anterior abdominal wall with viscera pushed posteriorly . A diagnosis of omental cyst was made at this stage.Laparotomy was performed. On opening peritoneum a huge lymphatic cyst was found . It was delivered out with difficulty and that too after partially evacuating it. More than 1500 ml of milky fluid was drained out. The anatomy was then identified more clearly. It was found between leaves of transverse colon mesentery and extending into its root . The redundant leaves of mesentery were excised without jeopardizing blood supply of colon and left open so as to prevent re-accumulation of fluid. Drain was kept and wound closed.Post-operative course was uneventful and patient discharged on 4th day. Biopsy was reported as consistent with mesenteric cyst. Parents were counseled again as to possible recurrence of the lesion and a regular follow up was planned. At three months follow up child is well. On ultrasound a small hypoechoic area of 4cm x 4cm was noted in mid abdomen. A repeat ultrasound after a month is advised to note the progress of the lesion.Cyst of lymphatic origin can be found within peritoneum  as well as in the retroperitoneal area. One of the theories proposed that these cysts represent benign proliferations of ectopic lymphatics. These channels lack communication with the main lymphatic system. Other theory suggested that due to the failure of joining venous system embryonic lymph channels gradually dilate. Still one of the theories suggested that as a result of non-fusion of leaves of mesentery, lymphatic fluid accumulates within this, supposedly a dead space. They can also result from trauma to the lymphatic channels . UsuallyThese cysts mostly remain asymptomatic and only come into attention when they cause abdominal fullness / distension or vague discomfort. In this patient it caused fullness and mild abdominal pain because of which parents seek advice. Though chronic symptoms predominate acute presentation is not uncommon which includes intestinal obstruction, volvulus, hemorrhage into cyst, rupture etc.  , 5. CliAbdominal ultrasonography usually provides working diagnosis in these patients and other investigations may be avoided though a more detailed picture can be provided by CT scan abdomen which may help in planning surgery. In reported patient CT scan revealed excellent details of the lesion. Thus where possible this modality may be used for diagnostic purposes.vSurgery is usually a straight forward affair. At times lesion can be removed completely without sacrificing any adjacent organ though resection of involved segment of small bowel, if limited area is involved, is more appropriate. The lesions of the mesentery can be excised as complete as possible, though in difficult cases and if cysts are at critical location de-roofing of the cysts is another approach, a type of marsupialization. In our patient the leaves of mesentery were extremely stretched out. Redundant part was excised easily. No well defined cyst found in this case. The histopathology reported the walls of lesion as of mesenteric origin with mesothelial lining. Considering lack of well defined limits of the lesion there are chances of recurrence of cysts in such cases. A regular follow up is thus advised. In our patient same is being followed. This case had many unusual features, namely being unilocular, of huge size containing milky fluid, in the mesentery of transverse colon and with apparently no distinct cystic structure. It appeared that mesenteric leaves simply stretched out due to accumulation of fluid. Presence of milky fluid within transverse colon mesentery may be explained on the basis of either malformed ruptured lymphatic channels of some trauma that might have caused leak of lymphatic fluid at the base of mesentery. This case may add to host of postulations related to lymphatic mesenteric cysts.Source of Support: NilConflict of Interest: None declared


Numb chin syndrome (NCS) is the presence of hypoaesthesia or paraethesia of the lip and chin over the distribution of the mental nerve. It is often caused by the presence of a metastatic tumour in the mandible or the base of skull and represents advanced malignancy.This paper presents an unusual case of NCS associated with metastatic adenocarcinoma of the lung, for which no obvious lesion was found in the mandible or base of the skull.NCS can oftentimes present itself in the absence of mandibular or base of skull metastatic lesions.NCS can be a sign of underlying advanced metastatic malignancy and therefore cannot be ignored and must be investigated fully. She also complained of increased sensation on the left half of her tongue and an alteration in taste to the left anterior two-thirds of the tongue along the distribution of the left lingual nerve.Her co-morbidities included spinal stenosis and neuralgia paraesthetica  that was being managed by analgesics and gabapentin. She quit smoking 30 years ago. She smoked a total of less than 5 pack years of smoking.On examination there were no signs of local dental pathology. An orthopantomogram X-ray showed no significant findings and due to ongoing concern an urgent MR scan of the head and neck area was requested.Nine days after this presentation the patient attended the Emergency Department and was admitted complaining of a sudden onset of left lateral pleuritic chest pain associated with shortness of breath. A working diagnosis of pulmonary embolism was made while awaiting the results of a CT Pulmonary Angiogram. This revealed a mass in the apical segment of the left lower lobe with a pleural effusion, most likely to be malignant, and multiple lesions in the liver.Pleural aspirate cytology confirmed the presence of cells consistent with an adenocarcinoma. The results of the MR scan of the head were now available showing a 3 cm wide left occipital mass with oedema of the white matter and another small lesion in the left frontal lobe see . This waHer symptoms of numb lip and chin were managed with gabapentin which was already prescribed and she was referred for further management to a chest physician and clinical oncologist. Palliative radio- and chemotherapy was offered, however, the patient refused treatment. There was a general deterioration of the patient's condition and the patient died 4 and a half months after her initial diagnosis of numb chin syndrome.3In the literature the reported aetiology of NCS is widely ranging. Local pathological processes such as trauma, infections, tumours and cysts are amongst common causes.Immune mediated systemic conditions such as temporal arteritis, vasculitis, multiple sclerosis Sjogren's syndrome have all been linked with NCS.Malignancy is one of the most prevalent causes of NCSPathogenic mechanisms behind NCS are thought to be either infiltration or compression of the inferior alveolar nerve sheath by tumour tissue. This can take place either at the level of the mandible or more proximally along the mandibular nerve near the base of the skull.A study of 42 patients in 1992 showed that the most common malignant cause of NCS was breast cancer or lymphoproliferative neoplasms. They comprised 78% of malignancy associated NCS cases with 50% of NCS cases as a result of mandibular metastasis, 22% as a result of leptomeningeal seeding and 14% as a result of base-of-skull lesions.Prognosis of patients with NCS as a result of malignancy is very poor with survival being measured in months.NCS is purely a sensory neuropathy usually presenting as unilateral numbness or paraesthesia over the distribution of the mental nerve.NCS cannot be taken lightly. It is important to recognise the potential for sinister underlying pathology associated with unilateral chin or lip numbness.Panoramic radiography of the jaw can provide valuable information. Any metastatic or local tumours can often be seen as well as any other causative local dental pathology. Further detailed views can be obtained through CT imaging. Imaging of the brain and skull base can also be very useful as it may reveal proximal lesions affecting the mandibular division of the trigeminal nerve. Some authors suggest the use of MRI if other forms of imaging are unrevealing.4In the case presented imaging did not show any evidence of mandibular metastasis or involvement of the base of skull. It was unlikely that compression by the occipital lobe metastasis was the cause of NCS, because one would expect this to cause other cranial nerve deficits.There have been several other case reports of NCS where there has been no evidence of a metastatic lesion in the mandible or base of skull on imaging.An important question is why are metastatic tumours selective for the mandible and mandibular nerve? Bodner et al. suggest that there may be a selective aspect in the site of seeding of metastatic tumours which may be responsible for the mandibular predilection in metastasis. Other authors have suggested that due to the presence of rich red bone marrow posterior mandible is rendered more susceptible to tumour embolisation.5NCS can be a sign of underlying advanced metastatic malignancy and therefore cannot be ignored. It must be investigated fully with the help of plain film and high resolution cross sectional imaging as appropriate for each case. It is possible that metastatic tumours infiltrating the inferior alveolar nerve may not be evident on imaging and can pose added difficulty in diagnosis.None declared.None.No ethical approval was required for this paper and I can confirm that I have consent from next of kin (son) as the patient has passed away.Faizan Zaheer (lead author): played a role in patient management and the lead author of writing up the case report.Khurrum Hussain: aided in writing up the case report and proof reading.Jeethendra Rao: clinical lead in patient management and aided in writing up the case report and proof reading.


Actinomyces species has been described as an opportunistic pathogen, particularly in the oral cavity; however, in rare cases these bacteria can cause actinomycosis which is characterized by formation of abscesses in the mouth, lungs, or gastrointestinal tract.Actinomycosis was commonly present in the pre-antibiotic era; however, it has a low prevalence now days. It has been recognized since 150 years ago, but because of its variable clinical presentation and indolent course, its recognition is difficult and patients are often misdiagnosed. Here we present a case of primary hepatic actinomycosis presenting as a metastatic liver tumor. This is the case of a 65-year-old male, who originally presented on August 18, 2008 with obstructive jaundice. His past medical history includes diabetes and hypertension. He underwent ERCP with stenting of the bile duct. The patient also had a spiral CT and an endoscopic ultrasound of the pancreas. These tests helped to determine resectability. He underwent a pancreaticoduodenectomy (Whipple procedure) on Sept. 8, 2008 for a pancreatic head adenocarcinoma. He had a standard whipple performed, with removal of the gallbladder and distal stomach, as well as the head of the pancreas and duodenum. There were no intraoperative events and no gallstones were spilled. He received adjuvant chemotherapy for 7 months. The chemotherapy was gemzar, oxaliplatinum and tarceva. The patient had routine follow-up and surveillance for recurrence every 4 months the first year and every 6 months for the second year. This included a CT scan and CA 19-9 level. Over the course of one year he was admitted to the hospital on several occasions for low-grade fever, for which the diagnosis was not established, and at times treated as an outpatient for multiple urinary tract infections. During these episodes the patient always had a normal white blood cell count and normal neutrophils/lymphocytes on differential. At no time did he complain of any abdominal pain or any difficulty tolerating food. All CT scans during this first year were negative for recurrent malignant disease.At approximately 18 months in follow-up, the repeated computed tomography (CT) scan showed a non-specific abnormality in the right lobe of the liver, suspicious for a mass. A magnetic resonance imaging (MRI) and positron emission tomography (PET) scan were then performed, which was positive for a mass, suspicious for an isolated tumor recurrence between segments 5 and 6 in the liver. Both the CT and MRI showed a large liver lesion present in the inferior aspect of segment 6 measuring 7 cm AP, 5 cm transverse, and 4.5 cm craniocaudal Figure .Laboratory examination of the patient's blood demonstrated: white blood cell count 6.1 (reference range 4.5-10.8 K/uL), alkaline phosphatase level 141 (reference range 38-126 U/L), and AST 36 (reference range 15-46 U/L), ALT 50 (reference range 13-69 U/L). The total and direct bilirubin levels were normal. The CA 19-9 was 84, which was increased from 39 on the previous sample from 3 months prior. Physical exam of the abdomen was unremarkable. No masses were palpated, the liver was not enlarged, and the abdomen was non-tender. There was no cervical, umbilical, or inguinal nodes appreciated.FNA biopsy was discussed at the GI tumor board, but in the setting of a rising CA19-9 and suspicious CT, MRI, and PET scan, the decision was made to proceed to surgery without a biopsy. The tumor board felt that this was recurrent pancreatic cancer and because of this decided the small risk of seeding the needle tract by doing an FNA was not warranted in this case.Patient underwent a resection of segment 5 and 6 of the liver without any complications. Final pathology of the specimen returned as inflammatory granulation tissue and granules consistent with hepatic actinomycosis. This was confirmed on tissue cultures. Special staining of this specimen showed gram positive filamentous bacteria.Infectious disease consult was called to review the case. They selected doxcycline iv, based on culture and sensitivity report, which would be changed to po doxcycline on discharge. The patient had an uneventful recovery and was discharged home on post-operative day 5. He was discharged home on oral doxycycline for a 6 month treatment course. 30-day follow-up shows no recurrence of this lesion and he continues to remain afebrile.Actinomyces israelii, which gives rise to chronic suppurative infection [The genus Actinomyces species are a slowing growing, gram-positive, non-spore-forming bacteria, that thrive in microareophilic and anaerobic conditions . There anfection ,3.They tend to be associated with infections of the cervicofacial and oral region. A rare cause of abdominal infection, the pathogenesis is presumed to be caused by hematogeneous spread via the portal vein from either a mucosal injury or other abdominal focus of injury and/or infection. The non-specific symptoms of fever, weight loss and abdominal pain make diagnosis of this condition a challenge. Despite advanced imaging techniques and difficulty in obtaining in positive cultures, the rate of preoperative diagnosis is less than 10% .Actinomyces infection is relatively rare, although strongly related to long-lasting intrauterine contraceptive device (IUD) application. An infective pathway has been postulated extending upward from the female perineum to the vagina and cervix. The traumatic effect of the device and a prior infection may contribute to the Actinomyces infection in the female genitalia.According to the literature, actinomycosis of the abdomen and pelvis accounts for 10-20% of reported cases. Typically, these patients have a history of recent or remote bowel surgery  or ingestion of foreign bodies , during which actinomycetes are introduced into the deep tissues. Diagnosis is usually established postoperatively, following exploratory laparotomy for a suspected malignancy. Pelvic actinomycosis most commonly ascends from the uterus in association with intrauterine contraceptive devices (IUCDs). Female genital Risk factors associated with this condition include abdominal wall trauma, gastrointestinal foreign body, previous abdominal/pelvic surgery, gastrointestinal tract lesions and immunosuppression . Since gLiver involvement has been reported in 15% of the cases, with majority of them resulting from translocation from either an intra-abdominal or intrathoracic site . PrimaryPrimary hepatic actinomycosis typically presents with an indolent course with symptoms typically present for greater than two weeks . ImmunocDifficult task for the management of actinomycosis is to reach the diagnosis before surgical approach is taken. In our patient the patient presents with isolated hepatic mass on MRI and PET scan strongly suspicious for recurrence of tumor after one year of tumor free survival. Because imaging studies frequently reveal single or multiple lesions, actinomycosis is often misdiagnosed as a primary or metastatic tumor. Radiographicallly, lesions frequently present as a single hypodense mass/abscess 68.4%) .For diagnosis, macroscopic, microscopic and histochemical examinations of surgical specimens are required, but the definitive diagnosis is based on tissue culture. The infection tends to lead a chronic and suppurative infection leading to fibrosis with draining sinuses that are pale yellow and often referred to as "sulfur granules" . The walTreatment of this infection consists of intravenous penicillin-G for four weeks and then oral penicillin-V for 12 months ,11. ProlOur conclusion from this case report is that primary hepatic actinomycosis can present in patients as a solitary hepatic mass after gastrointestinal malignancy. In our case it was mimicking a recurrence of the tumor. It is a rare and often overlooked etiology for a liver mass. An indolent course with unexplainable fevers, occasionally requiring hospital admissions, in patients with history of GI malignancies, should make physicians aware of this possible diagnosis. Nevertheless, the variable clinical presentation of this disease and its low incidence makes it difficult to recognize.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 journalThe authors declare that they have no competing interests.MW-main author and primary surgeon for patient; RN-assisted in writing case report; AC-assisted in gathering data; JS-assistant surgeon and helped edit the article; All authors read and approved the final manuscript.


Sclerosing peritonitis is a rare condition characterised by ascites, peritoneal and bowel wall thickening. Causes reported in the literature include luteal ovarian the comas, peritoneal dialysis, peritoneal chemotherapy and liver cirrhosis. We report an interesting case of a woman presenting with diarrhoea, abdominal distension, ascites and pleural effusion. She was subsequently diagnosed with Sclerosing Peritonitis caused by a degenerating fibroid which was successfully treated by Total Abdominal Hysterectomy and Bilateral Salpingoophrectomy. Sclerosing peritonitis is a rare condition characterised by ascites, peritoneal, and bowel wall thickening. Causes reported in the literature include luteal ovarian thecomas , peritonWe report an interesting case of a woman presenting with diarrhoea, abdominal distension, ascites, and pleural effusion. She was subsequently diagnosed with sclerosing peritonitis caused by a degenerating fibroid which was successfully treated by total abdominal hysterectomy and bilateral salpingo-ophrectomy. A 66-year-old lady presented to the general surgeons with a history of diarrhoea and abdominal distension. Examination revealed ascites and a CT scan was organised; this showed gross ascites with small pleural effusions, bulky ovaries, a fibroid uterus, and multiple peritoneal and mesenteric deposits. Tumour makers were within normal limits with Ca 125 17, CEA 2.1, and Ca19 9 6.2. A gynaecological oncology opinion was sought and an ascitic drain sited for symptom relief and to obtain possible cytological diagnosis. Histological analysis of ascitic fluid on two separate occasions revealed leukocytes, mesothelial cells, and scattered histiocytes with no malignant cells present. Further immunohistochemistry was performed confirming the cells to be lymphoid in origin.She continued to be symptomatic with abdominal distension and vomiting and therefore, a diagnostic laparoscopy was performed by the general surgeons. Laparoscopy confirmed gross ascites with what appeared to be widespread carcinomatosis with peritoneal and omental seedlings, bulky uterus and ovaries. Omental biopsies that were taken revealed an inflammatory process suggestive of possible sclerosing peritonitis. Histology showed omental fat with fibrous proliferation at the surface infiltrated by inflammatory cells and nodules of mesothelial proliferation, with no evidence of neoplastic infiltration. Immunohistochemical analysis was negative. Her clinical condition remained unchanged, and she required further ascitic drains for symptom relief. An MRI scan confirmed CT findings, and her case was discussed in detail at the Gynaecology and Colorectal MDT. As extensive investigation failed to establish a definitive diagnosis. In view of the small possibility of the fibroid uterus being responsible for her condition the decision was taken to perform a total abdominal hysterectomy, bilateral Salpingo-ophrectomy, omental biopsy, and serosal bowel biopsy. The gynaecological procedure was completed without difficulty with note only of an enlarged fibroid uterus. The peritoneal cavity was generally fibrosed extending throughout the bowel surface. Macroscopically the bowel was firm, solid with no obvious peristalsis due to the serosal fibrosis explaining the symptoms of obstruction. The bowel was inspected by the bowel surgeon and a number of biopsies were taken. Histological analysis from the peritoneal fluid showed mixed inflammatory cells and markedly reactive mesothelial cells. Interestingly, the ovaries were normal and the uterus contained an infracted ischaemic fibroid. Sections from the omentum and small bowel showed a fibrous process with spindle cells and inflammation. Overall this indicated diffuse sclerosing peritonitis. Her postoperative recovery was uneventful and at her six-week follow-up she had made an excellent recovery. She remained asymptomatic at her six-month follow-up. A CT that was performed at the time when compared to previous imaging showed complete resolution of her ascites and a normal appearance of the bowel. Sclerosing peritonitis is a rare inflammatory process which is often related to luteinised ovarian thecoma, peritoneal dialysis, chemotherapy, and liver cirrhosis , 2. Clinical presentation much like ovarian carcinoma is nonspecific including abdominal pain, weight loss, diarrhoea, and abdominal distension . SubsequThe process of peritonitis consists of fibroblast and myofibroblastic cell proliferation separated by collagen, fibrin, and inflammatory cells.  Ovarian spindle cells are commonly demonstrated . A treatment regime for sclerosing peritonitis has not been established but there are case reports in the literature that have indicated a role for total abdominal hysterectomy and BSO in removal of the thickened peritoneum and partial resection of the small bowel. Our literature search has failed to establish any previous reported cases of a fibroid uterus associated with sclerosing peritonitis. We reported a case of diffuse idiopathic sclerosing peritonitis. This is a rare benign pathological condition and in contrast to the majority of case reports where an ovarian mass was a feature, which was absent in our case. After a prolonged hospital admission the patient was treated with TAH and BSO which has subsequently led to a full and uneventful recovery.In unidentified causes of sclerosing peritonitis with a fibroid uterus this should be considered as a potential cause for the condition. Total abdominal hysterectomy and bilateral salpingo-ophrectomy should be considered as a possible treatment for the condition if their family is completed. In patients who have not completed their family a myomectomy could be considered.


Perinephric abscesses are life-threatening conditions, which are rarely associated with neoplasms of the kidney or upper tract collecting system. We report, to our knowledge, the first case of squamous carcinoma of the upper tract presenting as a perinephric abscess, diagnosed after radical nephrectomy. Staphylococcus aureus) [ Staphylococcus aureus, in which prompt diagnosis and treatment were key to therapeutic success [Renal and perinephric abscesses are life-threatening conditions, which may occur in different clinical scenarios , 2. Gene success .Malignant neoplasms are very rarely associated with renal and perirenal abscesses. We report an unusual case of perirenal abscess secondary to a squamous cell carcinoma and discuss the differential diagnosis and therapy.A 82-year-old man presented to the emergency room prostrated, with recent onset of fatigue and malaise. A few days earlier, he started feeling urinary urgency and had started treatment with oral fluoroquinolones for an alleged prostatitis.3; 1% myelocytes, 6% band neutrophils), and altered renal function tests . Urinalysis showed leukocyturia (10 leukocytes/field) and hematuria (10 blood cells/field). Urine culture was negative.Physical examination was unremarkable, there were no abdominal masses, and Giordano's sign was negative. Laboratory revealed mild anemia , leukocytosis  and hydration. After the initial treatment, the patient evolved with worsening of his clinical and laboratory findings. A noncontrasting abdominal and pelvic computerized tomography (CT) revealed a large heterogeneous mass in the right kidney, in contiguity with the lumbar musculature, the liver, and the vena cava, also dislodging the pancreatic head and the duodenum. In the collecting system of the right kidney, a large choraliform stone was found.In spite of the placement of a percutaneous drain in the perirenal space, the patient worsened, with progressive tachycardia, fever, and leukocytosis. Renal function, however, improved (serum Creatinine 1.4 mg/dL). An abdominal CT with intravenous (IV) contrast was then ordered and confirmed a large perirenal abscess , and surPathology revealed an 8.0 cm moderately differentiated (Grade 2) squamous carcinoma of the renal pelvis . NecrosiThe patient survived the perioperative period; however, his clinical condition has not evolved as expected. Roughly one month after the surgical procedure, the patient became septic and died of septic shock of an unknown source. Sepsis was presumed to be urinary, since chest X-rays were normal, and all cultures were negative at the time of death.Squamous cell cancers are very rare, comprising 0.7% to 7.0% of all urothelial tumors of the upper tract , 6. TheyThe association of renal tumors and perirenal abscesses is very rare. To our knowledge, this is the first published report associating an upper tract squamous cell carcinoma with a perirenal abscess. In our case, the extensive necrosis found in the surgical specimen was probably responsible for the genesis of the perirenal collection, which eventually brought the patient to clinical attention.The presence of a malignant neoplasm in our case was unsuspected until the time of the radical nephrectomy, and the initial treatment was concerned mostly with the resolution of the perinephric abscess.We hope that this case report may raise the awareness about the possible association of perirenal abscesses and renal tumors, which may facilitate the diagnosis and therapy of similar occurrences.


This is a case report of a 51-year-old female patient with benign mesenchymal tumors of paraurethral region which caused lower urinary tract symptoms. The pathological examination of the lesion was reported as angiomyxoma which is a distinct soft tissue tumor characterized by the presence of prominent myxoid matrix and numerous thin-walled blood vessels. This tumor has a predilection for the trunk, head and neck, extremities, and genitalia. It is a benign tumor, and total excision is curative. Recurrence is rare except for aggressive angiomyxomas. Benign mesenchymal tumors and tumor-like lesions of urethra are rare tumors and mostly arise from the soft tissue of the pelvic region in adult females, and various mesenchymal lesions may affect the because of their apparently shared origin from mesenchyme. We report a rare recurring mesenchymal neoplasia and represent a case by which it comes lower urinary tract symptoms because of an obstruction.We present case of a 51-year-old woman who admitted to our hospital with painless mass and progressively increasing voiding difficulties that involve initiating micturition, incomplete bladder emptying, intermittent stream, and massive hematuria. On physical examination, we observed a palpable mass near the urethral opening . MagnetiSuperficial angiomyxoma is a rare mesenchymal benign tumor arising from soft tissue of the pelvis and perineum. It is not thought of as cancer because it usually grows quite slowly and does not usually spread to other parts of the body. But there is an aggressive type that grows more quickly and involves the genital, perineal and pelvic regions of women, with great incidence occurring in the fourth decade . Among t


An aneurysmal bone cyst (ABC) is a benign tumor of the skeletal system, which most frequently occurs in long bones. An ABC arising from the rib is extremely rare and it is difficult to distinguish from other types of rib tumors. The present study describes an unusual case of a large ABC in the rib of a 17-year-old male. The entity is discussed with particular emphasis on the clinicopathological features, differential diagnosis and treatment. Due to difficulties in the pre-operative diagnosis, a possible diagnosis of ABC should be made aware when confronting an expansile rib mass. An en bloc resection of the mass and the affected portion of the rib is mandatory to obtain a satisfactory outcome. Aneurysmal bone cyst (ABC) was first reported by Jaffe and Lichtenstein in 1942 . ABC occA 17-year-old male was admitted to the Chinese People’s Liberation Army (PLA) General Hospital  with a chief complaint of progressive right-sided chest pain and chest distress for two months. There was no history of trauma, fever or respiratory embarrassment accompanying the pain and mass. On physical examination, the right posterior chest wall in the region of the seventh rib bulged to a minimal degree, but it was not tender to palpation. The respiratory sounds were slightly decreased on the right side.Plain and lateral chest radiographs revealed a posterior chest mass on the right side. Computed tomography (CT) of the chest revealed an irregular-shaped 12×10×10-cm tumor beside the 7th thoracic vertebra, which occupied almost half of the chest cavity. A mottled calcification was observed in the core of tumor. The tumor was mildly enhanced in the contrasted scan. The mass had expanded and destroyed the posterior part of 7th rib. Atelectasis of the right lower lobe and pleural effusion were also observed in the CT .CT-guided aspiration biopsy pre-operatively demonstrated that a neoplasm of spindle cell, solitary fibrous tumor of the pleura was possible. As the CT findings suggested the possibility of a malignant lesion, including sarcoma, surgery was performed on June 3, 2011.A right-sided thoracotomy revealed a 12×10-cm mass that originated from the posterior half of the 7th rib, which was medium-hard in texture. Numerous blood-filled cysts protruded from the surface of the mass, and an abundant blood supply was observed in the margin of the tumor . The maiThe microscopic examination demonstrated multiple blood-filled cavernous spaces that were separated by fibrous connective tissue, with hemosiderin pigment-laden macrophages, multinucleated osteoclastic giant cells and well-developed vascular spaces with hemorrhages in the septa lining the cystic spaces. There were focal areas of calcification and osteoid. The surgical margin was free. All these pathological findings were consistent with those of an ABC .The patient was discharged following an uneventful 12-day postoperative period, and has been followed up for two years with no evidence of recurrence.Primary rib neoplasms comprise 5–7% of all primary bone tumors , and accThe etiology of ABC is unclear. Certain studies have considered the condition to be secondary to an increased circulatory venous pressure or trauma, causing bone absorption and blood-filled cyst formation, thus explaining the expansile nature of ABC . AlternaThe most common symptoms in a patient with ABC of the rib are chest pain, swelling of the chest wall, dyspnea, paraplegia and pathological fractures . Some paAlthough the radiological appearance of ABC of the rib is not sufficiently specific to establish a definitive diagnosis, a CT scan is of great value to demonstrate the characteristic findings of the disease. CT may aid in showing a destructive pattern of rib lesions with a ‘soap bubble’ or ‘honeycomb’ appearance ,8. The dGrossly, the lesion is a cavity separated by septa. The spaces usually contain blood or serum. The macroscopic pathological findings tend to show a paper-thin cortex that is usually intact unless there has been a complicated pathological fracture. Under this shell, the bone is completely destroyed and replaced by various sizes of cysts containing blood or serum. In the present study, the cysts were separated by septa composed of loosely arranged spindle cells and benign giant cells. Cuboidal appearing cells line the cysts, and beneath this layer, osteoid tissue with osteoblasts are observed. Focal calcifications may also be evident ,11.Regarding the treatment of ABC, various methods have been described, including surgery, radiotherapy, embolization, cryotherapy, sclerotherapy and a wait-and-see strategy. To date, the ideal treatment option is total excision, as it has the lowest risk of local recurrence . An incoRadiotherapy should be considered only for inoperable cases. Extreme care should be taken with regard to the use of irradiation, as there is the possibility of a late development of sarcoma . Selectiet al recommended sclerotherapy for ABCs to avoid surgical risk and prolonged hospitalization, when the clinical presentation and radiological appearances are typical (Dubois  typical . The adv typical ,20.ABC arising from the rib is a rare, benign entity, which should be considered in the differential diagnosis of chest wall tumors. Complete surgical excision offers the best choice of treatment for curing ABC.


Since its introduction in the mid 1990s mechanical intra-abdominal morcellation of uterine tissue at the time of laparoscopic surgery is commonly performed to facilitate the removal of large uteri or leiomyomas through small incisions. However, with mechanical morcellation there are reported risks of metastasizing leiomyosarcoma, uterine cancer, introduction of complications from retained fragments of myomas, and endometriosis . Here weA perimenopausal 56 year old nulliparous woman underwent supracervical laparoscopic hysterectomy and bilateral salpingoophorectomy for pelvic pain, menorrhagia, and large uterine leiomyomas. She had no history of abnormal Pap smears and a preoperative Pap smear was normal. Her family history was negative for any known gynecologic, breast, or gastrointestinal malignancies. Preoperative chest radiograph and endometrial biopsy were not obtained. Due to menorrhagia, an endometrial curettage was performed at the initiation of the procedure. The specimen was sent for frozen section and was negative for malignancy. Intraoperative findings were significant for an 18–20 week size uterus weighing 1518 g. Prolonged mechanical morcellation with a ROTOCUT G1 morcellator  was required to remove the uterus and a small amount of superficial endometriosis in the pelvis was seen and cauterized. The fallopian tubes and ovaries were removed intact in an endoscopic bag. The uterus was not morcellated in a bag, and the fragments were subsequently collected from within the abdomen, and withdrawn from a laparoscopic port. Final pathology on six uterine fragments revealed weakly proliferative phase endometrium without atypia, adenomyosis and leiomyoma in the uterus. The ovaries showed fragments of benign ovarian parenchyma and patchy endosalpingiosis. No malignancy was identified in any specimen.The patient's initial recovery was unremarkable until 14 months post-operatively when she presented with left lower quadrant pain and persistent cough with shortness of breath. Examination revealed a firm palpable abdominal mass at the left lower quadrant and a fleshy lesion at the apex of the vagina. The cervix could not be identified. PET–CT revealed multiple hypermetabolic masses in the pelvis , abdomen2 every 21 days and had a partial response after two cycles. She completed six cycles of therapy, but unfortunately developed rapidly progressive disease. One month after her sixth cycle of chemotherapy, she presented with a complex fistula involving the pelvic tumor, abdominal wall, and sigmoid colon. She underwent a diverting loop colostomy. She strongly desired further therapy and received one cycle of doxorubicin 60 mg/m2. After admission for febrile neutropenia and pulmonary embolism, she elected to transition her care to hospice and died of her disease two months later.The patient was treated with intravenous carboplatin AUC 6 and paclitaxel 175 mg/mOur case illustrates the potential risk of metastatic adenocarcinoma diagnosed after hysterectomy with uterine morcellation. Definite causal relationship of metastatic disease as a result of morcellation cannot be proven; however, in the setting of an unknown cancer diagnosis at time of morcellation, subsequent widespread dissemination in the pelvis may result. Others have reported metastatic leiomyosarcoma, disseminated endometriosis , pelvic In our case the primary site of malignancy was uncertain. A plausible source could be that she had an unrecognized malignancy at the time of initial hysterectomy. Schneider et al. reported a case of undifferentiated adenocarcinoma five months after laparoscopic supracervical hysterectomy. The patient had undergone a macromorcellation supracervical hysterectomy and vaginal intrafascial cylindriform enucleation of the cervix and corpus uteri. In this procedure, the first core is performed vaginally to allow for a contiguous punched out specimen containing the cervix, endocervix, endometrium, and myometrium to optimize pathological evaluation. However, even with this additional step, initial pathology review showed no evidence of malignancy. When she was diagnosed with undifferentiated adenocarcinoma of a pelvic mass, retrospective review of the initial morcellated hysterectomy revealed clusters of malignant cells attached to the endocervical epithelium. However, the definite source of the malignant tissue could not be identified . AlthougAnother possible source of the malignancy is the malignant transformation of retained endometrial tissue or endometriosis after morcellation. Our patient had endometriosis involving the serosa of the uterus. Interestingly, Schuster et al. compared morcellated to non-morcellated hysterectomy cases and found no significant difference in outcomes in women that had endometriosis diagnosed at the time of hysterectomy. In this case–control study of 464 cases there was no difference in the incidence of new onset endometriosis between women undergoing a supracervical hysterectomy with uterine morcellation versus those women undergoing traditional vaginal or abdominal hysterectomy without morcellation . HoweverMinimally invasive surgery (MIS) has well documented benefits and uterine morcellation plays an important role in facilitating less invasive procedures for large uteri. However, the potential morbidity from mechanical uterine morcellation must be recognized. Appropriate pre-operative screening to rule out endometrial carcinoma in asymptomatic women prior to morcellation is challenging. Of the 708 women undergoing hysterectomy for pelvic organ prolapse, the incidence of endometrial cancer was only 0.6% . FurtherNone of the authors have any potential conflicts of interest.