Urachal carcinoma is a rare tumor that is usually associated with a poor prognosis, especially the pathological type, urachal mucinous adenocarcinoma. Surgery remains the primary treatment in prolonging the overall survival time of patients.We report on a 41-year-old woman with urachal mucinous adenocarcinoma who underwent three surgeries and several courses of chemotherapy over a 42-month period. The first surgery, involving en-bloc excision of the urachal mass, partial urinary bladder, urachal ligament, and umbilicus was performed in May 2007. It is well known that the correct surgical scheme plays a key role in preventing recurrence or metastasis. However, a second debulking surgery with only a single salpingo-oophorectomy may have contributed directly to the patient’s subsequent left ovarian metastasis. Therefore, we strongly recommend performing a bilateral salpingo-oophorectomy once ovarian metastasis has been detected, even if the metastasis is only present on one side. Although postoperative adjuvant chemotherapy regimens, first with Taxol, carboplatin, gemcitabine, and cisplatin, and then with IFO, EPI, and mesna were consecutively administered after the first and second surgeries, they seemed less effective, since recurrence and metastasis occurred shortly after each surgical treatment. After a third debulking surgery in June 2009, docetaxel, oxaliplatin, and capecitabine were administered. This chemotherapy regimen was chosen based on an immunohistochemical test that involved the multidrug resistance gene; this test indicated that the urachal mucinous adenocarcinoma was resistant to the two chemotherapy regimens used previously. Surprisingly, the patient exhibited a marker response to the new regimen and the metastatic foci entered into a stable disease stage. However, the patient still died of diffuse metastatic disease 1.5 years later. During the whole period of treatment, we found that serum tumor markers including CA724, CA125, CA19-9, and CEA were elevated in a linear pattern, with parallel increases in line with peritoneal carcinomatosis and parallel reductions in line with response to personalized chemotherapy.Personalized treatment can be given to those patients who experience a poor response to initial therapy. Moreover, an immunohistochemical test for the multidrug resistance gene and serum tumor markers may supply key information in the choice of reasonable chemotherapeutics. Urachal carcinoma is a rare form of tumor that usually originates in the bladder, and represents 0.01% of all cancers. Moreover, this lesion accounts for 0.34-0.7% of all bladder carcinomas . The mosIn June 2007, a 41-year-old woman was referred to our hospital with a palpable mass in her lower abdomen. Computed tomography showed a large mass located in the anterior pelvic cavity just superior to the bladder, but no other positive findings elsewhere. Because of the limited clinical evidence for diagnosis, laparotomy was performed for this patient. Laparotomy revealed a 12.0 × 12.0 × 9.0 cm solid lesion, which extended from the vertex of the bladder to the umbilicus. The lesion was removed en bloc together with the umbilicus, bladder dome, and adjacent peritoneum. Pathological diagnosis confirmed it to be a urachal mucinous adenocarcinoma Figure . MoreoveIt is sometimes difficult to differentiate between urachal and non-urachal carcinomas based only on symptoms. This is primarily because there is no specific symptom for urachal carcinoma. Furthermore, most of the tumors may develop in the submucosa or muscularis, and do not invade the mucosa of the bladder in the early stage, so symptoms are not prominent. According to the latest reports, several different imaging methods may be useful in aiding diagnosis. Ultrasound can demonstrate a tumor in the bladder dome associated with a mass containing calcification . ComputeTo date, the criteria for making a diagnosis of urachal carcinoma are not uniform, but clinical findings and histological evidence are both key factors. The typical criteria ,7-9 shouOur case was consistent with the typical criteria. However, for a few uncommon cases, this system is too restrictive to fulfill all the prognostic criteria. Two simpler criteria sets, which are more closely related to clinical practice, have been suggested by some scientists. The first set  is: (a) Surgical treatment plays a dominant role in the management of patients with urachal carcinoma. The achievement of a complete urachectomy including umbilectomy and negative surgical margins, and extended partial or total cystectomy, are crucial to long-term survival ,11. The Late presentation of symptoms and fast progression leading to advanced tumor stage at diagnosis have resulted in a poor prognosis for urachal carcinoma, which is consequently uniformly fatal. Moreover, urachal adenocarcinomas show a tendency to local recurrence and distant metastasis after surgical treatment, most often 81%) in the first 2 years % in the -14. HoweImmunohistochemistry is always one important method for differentiating between primary and metastatic ovarian mucinous tumors, although there is a considerable overlap in their immunohistochemical staining patterns. In line with the latest findings, Lee  has recoet al. [If suspected metastasis is encountered in the ovary, whether or not it occurs on one or both sides, it should be fully investigated and complete resection should be performed to prolong the patient’s overall survival time ,27. In met al.  reportedet al.  performeAlthough the role of chemotherapy and radiotherapy still remain unclear and the effects of other treatments have not been established, varied chemotherapy regimens have been reported to have some presumptive advantage -31 (TablDuring the whole period of treatment, serum tumor markers might be one of the most important prognostic factors that can reflect the efficacy of chemotherapy and tumor recurrence. Consistent with other enteric-type adenocarcinomas, urachal adenocarcinoma might express detectable serum levels of CEA, CA125, and CA19-9 . In our In conclusion, urachal carcinoma is not only a rare form of tumor, but also a difficult-to-treat disease. We support standard and radical resection to achieve a negative margin. However, even an enlarged or consecutive operation is encouraged when recurrence or peritoneal carcinomatosis occurs with a laparotomy. It is difficult to make an exact diagnosis preoperatively and a systematic review of all of the data is always essential in making a correct diagnosis based on the Mayo criteria. A standard chemotherapeutic strategy still needs to be explored in future studies, but personalized treatment can be given to those patients who experience a poor response to initial therapy. Moreover, an immunohistochemical test for multidrug resistance gene and serum tumor markers may supply key information in the choice of reasonable chemotherapeutics.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the editor-in-chief of this journal.Both authors declare that they have no conflicts of interest in the following areas: employment; consultancies; stock ownership; honoraria; paid expert testimony; patent applications or registrations; grants or other funding.The patient was examined and operated by PC and LZ, who are responsible for the postoperative care, follow-up, and clinical information. PC examined the patient and reviewed the patient’s files. LZ performed histopathological examination. The manuscript was drafted by LZ and critically reviewed by PC. Both authors read and approved the final manuscript.


A 30-year old woman was referred to our center because of suspicion of a primary lung tumor of the right upper lobe. Histological examination of the lung lesion revealed lung metastasis of a previously treated alveolar soft part sarcoma of the musculus vastus medialis of the right femur, which was resected 20 years ago. Alveolar soft-part sarcoma is a rare malignant tumor that occurs most often in the soft tissue of lower limbs. It is a slow-growing malignant soft tissue tumor arising in muscle tissue, usually in young adults. Due to pleural and extensive mediastinal infiltration with bilateral lung metastases, a systemic treatment with chemotherapy doxorubicin and ifosfamide was initiated. Late metastases from previously treated alveolar part sarcoma should be considered in patients with suspicious lung lesions even if surgical treatment was performed a long time ago. Alveolar soft-part sarcoma is a rare malignant tumor that occurs most often in the soft tissue of lower limbs. Alveolar soft-part sarcoma is a very uncommon soft tissue tumor known for late metastases to lung, bone, and brain. Lillehei and coworkers reported an interval of 21 years between primary presentation and development of lung metastasis and 33 years between primary presentation and development of brain and renal masses. Our case represents the second longest interval of 20 years between primary tumor presentation and development of a pulmonary recurrence.A 30-year old female patient was referred to our institution because of a large lung mass of the right upper lobe. The patient complained about increasing dyspnea for 2 months. Her initial physical examination was unremarkable and laboratory tests were all within normal limits. Spirometry revealed a mild restrictive pattern (TLC 88% soll) and reduced diffusion capacity (DLCO-SB 52% soll). CT thorax scan  revealedContrast-enhanced computed tomography scan  also revThe patient, interestingly, had a history of alveolar soft-part sarcoma of the musculus vastus medialis of the right femur successfully resected 20 years ago. The primary alveolar soft-part sarcoma had been initially staged as IA (T1 N0 M0) 20 years ago. All regular follow-up evaluations were unremarkable. However, the last chest X-ray evaluation was performed 8 years ago. Clinical workup revealed bilateral pulmonary metastases with ipsilateral pleural infiltration.Histological workup of material obtained during bronchoscopy showed a solid tumor composed of medium-sized to large cells arranged in compartmentalizing cords and nests, which were separated by delicate septa with capillaries. The cytoplasm of the tumor cells was broad and clear or eosinophilic. The nuclei were round and uniform, frequently with a single nucleolus .After immunohistochemistry demonstrating reactivity of the tumor cells for protein S100, but not for melanocytic markers or keratin markers, and comparison with the primary tumor, a diagnosis of metastatic alveolar soft-part sarcoma was rendered.Alveolar soft-part sarcoma was initially described in 1952 by Christopherson et al. . There iPulmonary metastases are encountered in 42–65% of patients, and the lung is most commonly involved. The brain and the skeleton are the two next most commonly involved metastatic sites . At firsChemotherapy is widely used in the treatment of nonresectable advanced disease, primarily with palliative intention. Initial standard chemotherapy for advanced or metastatic soft tissue sarcoma consists of single-agent anthracycline (mainly doxorubicin) or an anthracycline-based combination with, for example, ifosfamide and dacarbazine. The use of doxorubicin is limited, due to its cumulative cardiac toxicity . RegimenOur patient had been diagnosed with alveolar soft-part sarcoma of the lower extremity 20 years ago and was initially been successfully treated with complete resection of the tumor and adjuvant radiochemotherapy. Due to the extensive tumor manifestation at actual presentation with pleural infiltrations and bilateral lung metastases, a surgical option was not considered. Systemic chemotherapy with doxorubicin and ifosfamide was initiated.Alveolar soft-part sarcoma is an uncommon soft tissue tumor known for late metastases to lung, bone, and brain. Only Lillehei and coworkers reported an interval of 21 years between primary presentation and development of lung metastasis and 33 years between primary presentation and development of brain and renal masses .To the best of our knowledge of the current literature, our case represents the second documented patient with such an extremely late tumor recurrence of alveolar soft-part sarcoma.


Gossypiboma or a retained surgical sponge is a rare but avoidable surgical complication. It leads to considerable morbidity and at times even mortality. We report a case of a 24-year-old lady who presented one month after a Caesarean operation with complaints of fever, pain in abdomen, and vomiting. After the clinical examination a possibility of a retained surgical sponge was entertained. However a CT scan of abdomen revealed the complete diagnosis and helped in treating the patient surgically with a successful outcome. A review of the literature and all the relevant issues in the management of such a case have been discussed. Gossypiboma is a term used for a retained surgical sponge and is derived from gossypium (Latin cotton) and “boma” . Two usual responses lead to the detection of a retained sponge. The first type is an exudative inflammatory reaction with the formation of an abscess and usually leads to early detection and surgical removal. The second type is aseptic with a fibrotic reaction to the cotton material and development of a mass . A gossy A 24-year-old lady presented to our hospital one month after a Caesarean operation at another institute. This was her second Caesarean operation and was done as an emergency. Two days later the patient developed a pain in abdomen and fever; she was discharged on the eighth day. The symptoms exacerbated along with distension of abdomen, for which she consulted a private hospital and received cefotaxime and metronidazole. She was also referred back to the hospital where she was operated upon and she was treated symptomatically. However her condition worsened and she was referred to our hospital. She complained of fever, pain in abdomen, and vomiting. She also complained of loose stools and mucus discharge per rectum. On examination she was febrile and had tachycardia. Her abdomen was tender and lower abdomen had a vague tender mass.Her lab reports revealed neutrophilia and anaemia. A plain radiograph of abdomen was essentially normal. US of abdomen revealed a bulky subinvoluted uterus with a large collection within (pyometra), hypoperistaltic bowel loops, and mild hydroureteronephrosis on the right. However, the sonological impression of pyometra was not being correlated by gynaecological evaluation and hence a contrast-enhanced CT scan of abdomen was done . It reveEscherichia coli. There were multiple dense adhesions of the small bowel. On careful separation of the adhesions two ileal perforations were seen (Abdomen was accessed through a midline vertical incision. On exploration a retained surgical sponge was found along with one litre of pus lying in a walled off cavity in the lower abdomen. The retained surgical sponge was removed . The pusere seen . These pThe patient did well after the surgery. There was infection of the lower part of the abdominal wound, which was left open and a dressing was done regularly. A secondary suturing was done two weeks after the surgery and the patient was discharged.The hospital where the original surgery was done was informed to facilitate initiation of preventive steps. An inquiry into the details of the previous surgery revealed that the operation was done as an emergency and that it was difficult because of bowel adhesions due to previous Caesarean operation. The closure of abdomen was done by junior residents. A proper procedure for surgical counts was also found to be lacking.It is estimated that a gossypiboma may occur in 1 in 1,000–1,500 intra-abdominal operations . It was A pyoperitoneum or an organised hematoma may present in a similar manner. Thus, a high index of suspicion is required. A plain radiograph may help in a case of a retained sponge with a radio-opaque marker. In our case the sponge had no such marker. A US study helps in the diagnosis by revealing cystic masses with central echogenic wavy stripes with acoustic shadows. A contrast-enhanced CT shows air trapped nonenhancing mass . An uppe Sponge, sharp, and instrument counts have been used as protection against this problem. Four separate counts have been recommended: the first when the instruments are set up or sponges unpackaged, a second before surgery begins, a third as closure begins, and the final count performed during skin closure. This practice of sponge counts is heavily dependent on human performance practices and is thus subject to human error . SeveralMedico legal aspects of negligence and compensation also have to be addressed during the management.


Over the past decade, irinotecan has become one of the first-line chemotherapeutic agents used in the treatment of metastatic colorectal cancer. Recently, irinotecan has been administered transarterially in order to perform chemoembolization in the liver. In the limited number of reports available to date using this approach, serious adverse effects have not yet been reported. In this paper, we describe the formation of an intercostal artery pseudoaneurysm after transarterial chemoembolization with irinotecan-eluting beads in a patient with spinal metastasis from colorectal cancer. Approximately 20% of patients with colorectal cancer (CRC) present with metastatic disease at the time of diagnosis . CurrentIrinotecan (Camptosar) is a chemotherapeutic drug that has become more popular over the past decade due to its significant antitumoral activity against metastatic colorectal cancer, although major side effects have limited its systemic use . To miniInstitutional review board (IRB) approval was obtained for this case presentation. Two months prior to presentation at our institution, a 44-year-old Iranian male was diagnosed with adenocarcinoma of the colon. Initially, the patient received a low anterior resection and lymph node dissection which demonstrated extension into the peritoneal surface with lymph nodes positive for disease (stage T3N1). After being seen in January 2001, the patient underwent chemotherapy with 5-flurouracil, leucovorin, and irinotecan for six months. But in September 2002, his CT scan demonstrated a left lung nodule which was resected in October 2002 and diagnosed as metastatic adenocarcinoma of the colon. Postoperatively, the patient received a second regimen of chemotherapy with 5-flurouracil, leucovorin, and oxaliplatin for four months. Colonoscopy and serial CT scans in 2003 did not demonstrate any recurrent disease. But in February 2004, recurrent bilateral pulmonary nodules were detected and surgically resected which demonstrated again metastatic adenocarcinoma of the colon. A followup CT in November 2004 demonstrated a left pleural mass and bilateral lung nodules, and the patient was started on a third regimen of chemotherapy including 5-fluorouracil, leucovorin, irinotecan, and abciximab. In March 2006, these lesions demonstrated progression which led to a left chest wall resection with a portion of the lung. The lesions on the right were planned for radiofrequency ablation therapy, but in January 2007, the focus shifted to treating a large paravertebral mass involving the left T7, T8, and T9 vertebral bodies revealed on CT and MRI, which was invading the neural foramina with epidural extension . As the On the day of the chemoembolization, after written informed consent was obtained, the patient was prepped in a normal sterile fashion. A 5 Fr sheath was placed in the right femoral artery. An angled Glidewire , Mikaelsson catheter , Progreat catheter , and Transend wire  were used to sequentially select multiple intercostal arteries bilaterally. μm LC Beads  was injected in divided doses between the left T7, T8, and T9 vertebral arteries. Due to vasospasm, only partial embolization of the T9 intercostal artery was accomplished  no contrast opacification of the pseudoaneurysm lumen, (2) a coil mass measuring approximately 1.5 × 3.5 cm, and (3) a gentle regional concave deformity of the aorta, implying that the nonthrombosed pseudoaneurysm sac of the pseudoaneurysm was larger than what opacified originally . The patIrinotecan (Camptosar) is a chemotherapeutic drug that has become more popular over the past decade due to its significant anti-tumoral activity against metastatic colorectal cancer . It inhiTo minimize the side effects, transarterial chemoembolization with drug-eluting beads has been used. With chemoembolization, there is a significant reduction of these systemic side effects with only mild abdominal and shoulder pain, vomiting and mild asthenia, and some alopecia . No seriSeveral papers have demonstrated the palliative effects of chemoembolization in the treatment of spinal metastases from various primary cancers for patients who failed systemic chemotherapy, are not amenable to surgical resection or radiofrequency ablation of their spinal tumors, and have a dominant soft tissue component that precludes treatment with acrylic cement , 9. The The use of irinotecan in combination with 5-FU/folinic acid as a first- and second-line systemic treatment of advanced colorectal cancer is on the rise , 7, 10. As mentioned previously, the long-term outcome of irinotecan chemoembolization with drug-eluting beads is still pending. The preliminary trial of chemoembolization with irinotecan-eluting beads on colorectal cancer metastases to the liver by Aliberti et al. demonstrated a 50–90% decrease in CEA levels and a decrease in lesional contrast enhancement . Five moHowever, our patient experienced a side effect of a pseudoaneurysm formation after chemoembolization with irinotecan. This is a rare and interesting report of a pseudoaneurysm after chemoembolization with irinotecan. Our patient received approximately 20 mg of irinotecan-loaded beads in each of the three chemoembolized intercostal arteries (T7–T9). Two weeks after the procedure, a pseudoaneurysm was discovered in the proximal segment of the T8 intercostal artery. The overall incidence of intercostal artery pseudoaneurysms is rare, and only seven cases have been reported in the literature –17. FiveLaboratory evidence of vascular damage from chemotherapeutic drugs for metastatic CRC has been reported only for doxorubicin. At high doses, doxorubicin compromises the vascular wall by causing excoriation and apoptosis of endothelial and smooth muscle cells . AlthougThe standard treatment for patients with spinal metastases from colorectal cancer is surgical resection and/or RFA. In this case, the tumor had invaded the epidural space, limiting these options. The standard traditional option would have been radiation therapy with bland embolization. The addition of effective chemotherapeutic drugs with embolic material has gained favor for tumors not amenable to surgery or RFA. To date, there is only one study  in its pIn summary, this case demonstrates the formation of a pseudoaneurysm after transarterial chemoembolization with irinotecan-eluting beads. Further studies are warranted to evaluate the effects of irinotecan on the vascular wall and the possibility that this could affect the long-term clinical outcomes of irinotecan chemoembolization.


Cardiac myxomas are the most common type of benign cardiac tumors and most of them occur in the left atrium but the biatrial myxoma is uncommon. We present a rare case of giant biatrial myxoma nearly obstructing the orifice of the inferior vena cava. A 58-year old woman presented with exertional dyspnea and intermittent chest discomfort. The non-pedunculated tumor involved most of the interatrial septum and extended from the orifice of the inferior vena cava to the displaced mitral annulus and the lower left pulmonary vein. The resected specimen weighed 76 gram and measured 80 × 40 × 30 mm. She did not complain of dyspnea or show any sign of recurrence by echocardiography during the 2-year follow-up period. Cardiac myxoma is the most common type of primary cardiac neoplasm and accounts for 30% to 50% of all primary tumors of the heart with an annual incidence of 0.5 per million populations ,2. Over A 58-year old woman presented with exertional dyspnea and intermittent chest discomfort. She had no past medical or familial history, and physical and neurological examinations, chest radiography, and electrocardiography findings were normal, although ESR was high at 54 mm/hr. However, echocardiography revealed a huge, immobile mass without pedunculation occupying both atria Figure A. Due toCardiac myxoma is usually polypoid and pedunculated lesions, arising from endocardium . In cont.The Biatrial myxoma is uncommon and could be considered in the differential diagnosis of other cardiac tumors with several imaging modalities. Prognosis is excellent after surgical resectionWritten and informed consent obtained for publication of this case report and accompanying images. A copy of the written consent is available for review by editor-in- chief of this journal.The authors declare that they have no competing interests.CP and CC collected the data and wrote the manuscript. JK, YJ, JL, and KP participated in the design of the manuscript and they revised and critically reviewed the manuscript. All authors read and approved the final manuscript.


Cases with subcutaneous metastasis of differentiated hepatocellular carcinoma to the abdominal wall without prior seeding as a consequence of local interventions with a negative or normal alpha-fetoprotein level in the serum are extremely rare.This is the first report of a case with AFP-negative, differentiated hepatocellular carcinoma metastasis to the abdominal wall within a pre-existing subcutaneous lipoma since childhood after antiandrogen therapy with leuprorelin and buserelin acetate for prostate cancer without seeding.Clinical features including histology, immunohistochemistry, clinical course and surgical approach are presented.Histological examination revealed a hepatocellular carcinoma with a trabecular and pseudoglandular growth pattern with moderately atypical hepatocytes with multifocal bile formation within a lipoma. The postoperative course of abdominal wall reconstruction with a monocryl-prolene mesh and a local flap after potentially curative resection was uncomplicated.It may be that previous antiandrogen treatment for prostate carcinoma contributed to the fact that our patient developed alpha-fetoprotein-negative and androgen receptor-negative subcutaneous abdominal wall metastasis within a pre-existing lipoma since childhood. Hepatocellular carcinoma (HCC) remains one of the most common malignant diseases and the incidence is increasing worldwide -5. TodayCases with subcutaneous differentiated metastasis of hepatocellular carcinoma with a negative or normal alpha-fetoprotein (AFP) level in the serum are extremely rare , cytology, radiofrequency ablation or percutaneous ethanol injection (PEI). In the current literature during the last 10 years, only few cases have been reported with abdominal wall metastasis without prior seeding as a consequence of local interventions . He was in good general condition with an obese nutritional status (Body Mass Index 30.4 kg/m²) and an impression of fatty liver parenchyma in ultrasound and magnetic resonance imaging. He was reported to drink two glasses of whisky per day. The previous history included multiple subcutaneous lipomas within the abdominal wall since childhood and prostate carcinoma, which was diagnosed in 2005 and removed by prostatectomy  in the same year, followed by antiandrogen therapy with leuprorelin and buserelin acetate until August 2008. The multiple lipomas showed a marked progression in size under antiandrogen therapy from August 2007 until August 2008. Final diagnostic evaluation, including ultrasound-guided FNAB of the liver mass and magnetic resonance imaging, resulted in the diagnosis of a multilocular, well-differentiated and AFP-negative hepatocellular carcinoma in liver segments II/III, VI, and VIII in April 2010. AFP levels stayed within normal range at all times during further follow-up. Viral hepatitis or other risk factors for hepatocellular carcinoma like hemochromatosis, liver fibrosis or liver cirrhosis could be ruled out. After interdisciplinary tumor board consultation  transarterial chemoembolisation (TACE) was performed from July 2010 to January 2011 every two months leading to a complete remission.Consultation with our surgical service was initiated in November 2011 due to a hard palpable subcutaneous mass with a diameter of approximately 7 cm that had developed recently in the left upper abdominal wall, exactly where a soft subcutaneous lipoma manifestation has been, in the consciousness of the patient, since his childhood  subcutaneously with suction for a total of five days -related hepatocarcinogenesis and hepatocyte proliferation are increased by androgenic stimulation in TGF-alpha transgenic mice [It could be demonstrated that the majority of hepatocellular carcinoma cells express androgen receptors in higher levels as compared to normal hepatocytes -42. Takenic mice . Severalnic mice -47. Intenic mice .Androgen receptors have been demonstrated in HCC in 74 % of males and 37 % of females, and their presence is associated with a more rapid progression of disease . ArgumenMore recent studies with Cre-Lox conditional knockout mice model who lack androgen receptor (AR) expression demonstrated that the androgen receptor, but not androgens, may be a potential new and better therapeutic target for the treatment of HCC . A new aDue to the low incidence of subcutaneous abdominal wall metastasis of HCC, therapeutic algorithms are widely missing. In the current literature, surgical resection is the favored therapeutic option ,23,32. Het al. reported 21 patients with a subcutaneous HCC metastasis without seeding who were treated by radiotherapy , encompassing up-to-date interventional, chemotherapeutic, and surgical approaches, including liver resection and liver transplantation, in one of the largest academic treatment centres for HCC in Germany.


Castleman’s disease is a rare and poorly understood disease entity that may resemble more common conditions and represents a clinical challenge to the treating surgeon.In this report, we describe a case of a 61-year-old Caucasian woman with a symptomatic retroperitoneal mass. The specimen obtained from her resection contained a protuberant encapsulated mass, exhibiting microscopic features consistent with localized, unicentric Castleman’s disease. These characteristics included architectural features and immunohistochemical findings consistent with the hyaline vascular variant of Castleman’s disease.We report a very rare case of a retroperitoneal hyaline vascular type of Castleman’s disease. We discuss the diagnostic dilemma Castleman’s disease may present to the surgeon, with an emphasis on multidisciplinary management of these patients. We also review current data on pathogenesis, treatment and outcomes. Angiofollicular lymph node hyperplasia, or Castleman’s disease (CD), is a rare neoplastic disease first identified in 1956 by Castleman and colleagues -4. CliniThe classic histological subtype of CD, representing 76% to 91% of localized disease, is the hyaline-vascular type (HV-CD). The plasma cell variant (PC-CD) has been identified in 9% to 24% of CD cases, and a rare hyaline-vascular plasma cell (or mixed) subtype has also been reported ,9. CliniCD has an unclear etiology, although human herpes virus 8 (HHV-8) and HIV coinfection has been implicated in the etiology of multicentric disease (MCD). Although it has been hypothesized that treatment with highly active antiretroviral therapies might decrease the incidence of MCD, MCD has actually increased and further study elucidating the effects of viral seropositivity on dysregulation of the immune system is ongoing . AdditioHistorically, the treatment and prognosis of CD are based on the centricity of the disease rather than on the histopathology. Surgical resection of UCDs can be curative, with long-term, recurrence-free survival described in the majority of case reports and series. The tumor’s location and relationship with adjacent structures are important factors in predicting surgical morbidity ,14. MCD,In the present report, we describe a rare case of retroperitoneal UCD and discuss a multidisciplinary approach to diagnosis and treatment of patients with this rare clinical entity.A 61-year-old Caucasian woman was seen by her primary care physician for a palpable abdominal mass that had been present for 1½ years. She did not complain of systemic symptoms, general malaise, weight loss, flank pain or urinary symptoms, but described mild diarrhea and right periumbilical pain that radiated to the right lower quadrant. Her medical history included well-controlled hypertension and type 2 diabetes mellitus. Her previous colonoscopic and endoscopic evaluations were normal. During her physical examination, a nontender, immobile mass was palpable in the right lower quadrant without peripheral lymphadenopathy. Examination and laboratory studies were otherwise unremarkable. Computed tomography (CT) with intravenous and oral contrast of the abdomen and pelvis revealed a heterogeneously enhancing, 10.2 × 6.9 × 6.3–cm solid mass in the right retroperitoneal space immediately lateral to and displacing the inferior vena cava and right iliac vasculature  of small cluster of differentiation 4 (CD4)-positive T lymphocytes and plasma cells Figures  and 4. WOur patient presented to our institution with a rare, symptomatic, retroperitoneal, unicentric HV-CD tumor. Although etiologic factors continue to be elucidated , a majorPrimary retroperitoneal tumors are particularly difficult to diagnose because of their location and often are clinically detected only after the tumor reaches a size that is large enough to be found with a resultant mass effect and involvement of surrounding structures. Among primary retroperitoneal tumors, one-third are sarcomas . LiposarThe decision to biopsy retroperitoneal masses preoperatively remains controversial. At our institution, if a mass is suspected to be malignant and appears amenable to complete surgical resection, we frequently recommend surgical management without preoperative biopsy. Most surgeons agree that surgical resection in the appropriate clinical setting without biopsy is acceptable . Biopsy et al.[An important clinical consideration in CD tumors is the degree of vascularity, which is more prominent in the HV-CD variant than in the PC-CD type. The degree of vascularity is relevant to both the diagnosis and treatment of CD. When CD presents in a rare location , the increased vascularity may confound the diagnosis and cause suspicion for primary malignancy with associated angiogenesis . Zheng eet al. describeet al.-26.et al. [et al. [Once the diagnosis or real clinical suspicion of UCD has been established, complete surgical resection is the treatment of choice on the basis of its reported long-term, recurrence-free survival rate with possible cure ,14. Impoet al.  reported [et al.  describe [et al. -32. Our Although the decision-making regarding of UCD and its management is challenging, MCD presents an even greater problem for the treating physician and surgeon. The use of antiviral and antiretroviral therapies is considered necessary with the addition of chemotherapeutic agents, including regimens developed for treatment of lymphoma and rituximab alone, which have demonstrated variable responses . DespiteIn this case report, we describe a rare case of retroperitoneal unicentric HV-CD, a poorly understood disease that creates a diagnostic and therapeutic dilemma for surgeons. To date, the use of improved radiologic criteria for diagnosis, interventional techniques and operative, neoadjuvant and adjuvant strategies appear to play an increasingly important role in the diagnosis and management of patients with this rare disease. Because of the rarity of CD, the opportunity for clinical trials to produce uniform evidence-based approaches is unlikely. With regard to the treatment options for patients with these rare tumors, we encourage a collaborative, multidisciplinary approach that includes surgical specialists, radiologists, pathologists and oncologists to discuss treatment options to optimize patient outcomes.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.AW acquired data and drafted and revised the manuscript. AS and SH made the pathologic diagnosis and revised the manuscript. RR, MH, BB and MS were the pre- and postoperative treatment team and revised the manuscript. MW was responsible for surgical management and revised the manuscript. WB was the treating surgeon and drafted and revised the manuscript. All authors read and approved the final manuscript.


Magnetic resonance imaging, MRI has more advantages than ultrasound, computed tomography, CT, positron emission tomography, PET, or any other imaging modality in diagnosing focal hepatic masses. With a combination of basic T1 and T2 weighted sequences, diffusion weighted imaging, DWI, and hepatobiliary gadolinium contrast agents, that is gadobenate dimeglumine (Gd-BOPTA) and gadoxetic acid (Gd-EOB), most liver lesions can be adequately diagnosed. Benign lesions, as cyst, hemangioma, focal nodular hyperplasia, FNH or adenoma, can be distinguished from malignant lesions. In a non-cirrhotic liver, the most common malignant lesions are metastases which may be hypovascular or hypervascular. In the cirrhotic liver hepatocellular carcinoma, HCC, is of considerable importance. Besides, intrahepatic cholangiocarcinoma and other less common malignancies has to be assessed. In this review, the techniques and typical MRI features are presented as well as the new algorithm issued by American Association for the Study of the Liver Diseases (AASLD). In patients with liver tumors, it is crucial to detect and stage the tumors at an early stage . Therefore, an optimal preoperative evaluation of the liver is necessary, and a contrast-enhanced MRI is widely considered the state-of-the-art method . Liver MIn this review, a technical overview is followed by a presentation of the most common focal liver lesions.1) and focal fatty sparing of the liver. It is also valuable in diagnosis of iron storage diseases - the iron containing liver parenchyma is hypointense on in-phase and isointense on out-of-phase [The non enhanced T1 and T2 weighted sequences are decisive in the characterization of focal liver diseases in cirrhotic as well as non cirrhotic liver. High signal intensity, SI, on T1 can be caused by bleeding, fat or deposition of copper or glycogen and can be seen in dysplastic nodules and hepatocellular carcinoma, HCC [of-phase .2), with a sensitivity and specificity better than multidetector CT, 87% and 97% for DWI compared with 53% and 78% for CT [et al., it was shown that cysts have significantly higher values compared to hemangiomas which has significantly higher ADC than solid lesions - however, the solid lesions could not be distinguished from one another, using the ADC values [Diffusion weighted echo-planar imaging, DWI, has been shown to be a reliable method to detect liver metastases . Solid and vascularized lesions enhance whereas cystic and necrotic does not. ”Hypervascularized” lesions enhances intensely and early, in contrast to ”hypovascularized” tumors enhancing less and later (see below). 2), characterize liver lesions (with or without hepatocyte function/uptake) and to evaluate biliary excretion. There are two different hepatobiliary paramagnetic gadolinium chelates, gadobenate dimeglumine  and gadoxetic acid . Gd-BOPTA has higher relaxivity values and SI changes than the non liver specific Gd-based contrast agents but similar safety profile [Hepatobiliary contrast agents can be used to detect metastases  sequences should not be obtained after administration of Gd-EOB-DTPA, because the rapid biliary excretion of this contrast agent decreases SI of the biliary structures on T2 . HoweverDynamic contrast-enhanced MRI, DCE-MRI, has been analyzed to determine the effect of drugs on tumor angiogenesis and vascular disruption. Although early results show that this is beneficial, its practical application is far from straightforward. A review of the literature  has poinExperience in diffusion-weighted MRI in monitoring treatment response has shown promising results , 22. RecThe most common benign tumors have typical imaging features. Knowledge of these findings and the clinical history are essential in correctly diagnosing focal liver lesions. However, it is important to know that common liver tumors may manifest with atypical findings, posing a diagnostic dilemma when they do . 2). Congenital biliary cysts are common with incidence up to 14%. A typical hepatic cyst is unilocular with a nearly imperceptible wall and a homogeneous content with a remarkably low SI on T1 and extremely high SI on T2 weighted images  which increases on heavily T2 weighted images. Cysts do not enhance after contrast injection . The dia3). The contrast enhancement is peripheral and nodular in early phases, followed by progressive centripetal filling in late and delayed phases. The SI is similar to blood. Small hemangiomas, <2 cm, may have homogeneous enhancement in late arterial phase and resemble hepatocellular carcinoma and hypervascular metastasis. However, hemangiomas follow SI of blood [Cavernous hemangioma is the most common hepatic tumor, incidence 20%. A typical hemangioma is well delineated and hypointense as blood on T1w images and clearly hyperintense on T2w images . Accurate discrimination of FNH from hepatic adenoma is not possible on this imaging alone. However, if using hepatobiliary contrast enhancement an accurate differentiation is achievable on delayed T1, 1-3 hours after gadobenate dimeglumine enhancement: FNH appear hyper-isointense (97%), whereas adenomas are hypointense (100%) [5). A typical FNH is hyperintense in the arterial phase, and isointense before contrast and in the venous phase. Thin radiating septa divide the tumor, but there is no capsule. A majority of FNH (89%) is before contrast hyper- to isointense on T2 and iso- to hypointense on T1. After gadolinium administration, 98% of FNH show a rapid and intense enhancement during the arterial phase, followed by a hyper- to isointensity in portal venous and equilibrium phases   4, in contrast to fibrolamellar HCC with hypointense scar without contrast enhancement). A critical note is that, on Gd-EOB enhanced imaging, the central scar of focal nodular hyperplasia lesions does not typically demonstrate delayed enhancement [Half of FNH has a central scar, slightly hyperintense on T2 and with late Gd-contrast enhancement  and isointense in the hepatobiliary phase - need complementary studies, biopsy and/or follow up for correct diagnosis. Hepatocellular adenoma, HCA, is in the West a rare tumor of women on oral contraceptive (85-98%) or men on anabolic steroids. Risk of hemorrhage increases in adenomas larger than 4-5 cm and during pregnancy. However, evidence of minimal intratumoral bleeding is often present. Some adenomas are steatotic, and may be multifocal (60%) and even coexisting with FNH. HCAs are considered premalignant but rarely develops into HCC unless larger than 10 cm (10% risk). Those with male hormone administration, familial polyposis and mutated b–catenin has enhanced risk .5). A typical adenoma has heterogeneous SI on both T1 and T2 weighted sequences due to hemorrhage, necrosis and/or steatosis. The enhancement is after Gd-contrast heterogeneous in the arterial phase and hypointense in the hepatobiliary phase  DWI can’t be used reliably to differentiate adenoma from FNH or HCC , 7.It is considered that regenerative nodules, RN, can stepwise develop into hepatocellular carcinoma, HCC, via low-grade dysplastic nodules (DN), high-grade DN and well-differentiated HCC. In this development, from benign to malignant, the portal venous supply diminishes and the arterial supply increases. Regenerative nodules are hypointense on T1 and T2 weighted images. Dysplastic nodules (and ⅓ of HCC) are hyperintense on T1 weighted images . A focal lesion that is hyperintense on T1w/T2w imaging is a potential HCC and must be further characterized . In a ciLobar or segmental atrophy and confluent fibrosis are hypointense on T1 and hyperintense on T2 weighted imaging. After Gd, the enhancement is delayed and may finally be hyperintense to the liver parenchyma.Hepatocellular carcinoma (HCC) is a common primary tumor in the world and the third most common cause of cancer related death, after lung and stomach cancer. Identification of HCC at an early stage is crucial for prompt surgical resection or transplantation . Therefo6). According to the new AASLD recommendations, for lesions ≥1 cm, characteristic findings with arterial hypervascularity and venous washout, in one proper examination, including a dynamic multidetector CT or an MRI, with 4 phases , is diagnostic for HCC without biopsy. This apply to lesions in cirrhotic liver and to patients with chronic hepatitis B but not to nodules in otherwise normal liver. However, it is essential to point out that HCC cannot be ruled out by the absence of arterial hypervascularization or absence of delayed washout. The false-negative tumors should be diagnosed on follow-up imaging before the lesion reaches a size where the chance of cure is diminished [The standard of care for the radiological diagnosis of HCC in cirrhosis is dynamic contrast imaging exhibiting typical arterial enhancement and venous/late washout  minished .The accuracy of imaging diagnosis of HCC in liver cirrhosis depends largely on the degree of arterial hypervascularization, which increases with tumor size and grade of malignancy . Large HThe correlation of HCC size and sensitivity was readily shown in a Gd BOPTA study correlated to explanted livers and HCC. Overall there was an excellent sensitivity and specificity of 87% and 79% respectively and a positive predictive value of 65-66% in the detection of HCC. However, the size was crucial for the sensitivity: >2 cm 100%, 1-2 cm 83-89%, ≥1 cm 91-94%, <1 cm 29-43%. False positive lesions measuring 1-2 cm were dysplastic nodules (85-100%), and <1 cm were nonneoplastic arterial hypervascular lesions (80-86%). The delayed phase imaging had limited diagnostic value due to poor hepatocyte function and enhancement in this group of late cirrhosis .Large HCC are heterogeneous and regularly invades the portal vein, and this is a significant prognostic factor  .In a cirrhotic liver, a solid lesion hyperintense on T2 is suspected for HCC . A hypoiA main challenge now is to better characterize hypervascular nodules <2 cm, which often have nonspecific imaging characteristics . One optOn DWI, a typical HCC is hyperintense on images with high b-values and is dark (restricted diffusion) on ADC image. By adding DWI to CE-MRI the accuracy to differentiate HCC from dysplastic nodule increases from 75% to 93% . These alternate approaches may improve the diagnosis of small lesions. However, prospective studies, with accurate imaging-pathology correlations on explanted livers, are warranted before any alternate criterion is approved as the standard diagnostic technique for HCC. Fibrolamellar HCC is rare and seen without underlying cirrhosis. A typical fibrolamellar HCC is lobulated, large  with a calcified central scar, and has metastases in ⅔ of cases. It differs from FNH in several aspects: enhances heterogeneously and the scar is hypointense on T2w and does not enhance in arterial and venous phases and only partially on delayed imaging . Convente.g. primary sclerosing cholangitis) and cystic disease of the liver. A typical intrahepatic cholangiocarcinoma is hyperintense on T2w images and often obstruct vessels and bile ducts, with upstream ductal dilatation . In affected segment(s), there is a volume loss with capsular retraction. After contrast, the enhancement is delayed, starting in the periphery, resembling hemangioma. However, the enhancement of cholangiocarcinoma is not isointense to vessels. In addition, intrahepatic cholangiocarcinomas may also display homogeneous arterial contrast enhancement [Intrahepatic cholangiocarcinoma is quite rare and is less common than hilar/Klatskin tumors (90%). It is associated with repeated/chronic cholangitis .DWI is more sensitive than T2-weighted MRI for the detection of hepatic metastases. Hepatic metastases show higher SI on DWI than on T2-weighted fast SE images, whereas the signal from vessels and cysts are suppressed with DWI. Small lesions occasionally need further characterization , 64 . They are often multiple and do not follow the SI of vessels. On T2, the SI of hypervascular metastases is usually moderately elevated, and on T1 hypointense. Melanoma is an exception, where melanin accumulation may result in hyperintensity on T1. There are also common benign hypervascular liver lesions, hemangioma, focal nodular hyperplasia and some tumor-like liver conditions. On DWI, metastases with a marked hypervascularity can attain high ADC values [Liver metastases from renal cell carcinoma, neuroendocrine tumors, sarcoma, malignant melanoma, thyroid carcinoma, some breast and colorectal cancer are hyperintense in late arterial phase imaging  C values  and be fSecondary liver lymphoma is multiple, and primary lymphoma, which is rare, is solitary . LymphomIn echinococcosis, there is a large cystic mass with numerous peripheral daughter cysts. Calcifications, if present, are hard to see on MRI. Cyst wall and septa enhances after Gd-contrast injection. On DWI, hydatid cysts with restricted diffusion and low ADC value, differ from simple cysts, with free diffusion and high ADC value .9). Typically, cluster of small abscesses coalesce into a large cavity with air or fluid level and is surrounded by an enhancing capsule. Small abscesses (<1 cm) may enhance contrast and mimic hemangiomas. Metastases, necrotic after treatment, may be indistinguishable from abscesses. However, DWI studies have indicated that DWI could discriminate abscess and cystic or necrotic tumors  [Most liver abscesses are pyogenic and can be portal or biliary in origin  , 68.


Aluminum phosphide is a systemic lethal poison. Fistulous communication between esophagus and airway tract (esophagorespiratory fistula) has rarely been reported in the survivors of aluminum phosphide poisoning. We report a case of benign esophagobronchial fistula secondary to aluminum phosphide poisoning, which to best of our knowledge has not been reported in the medical literature. Aluminum phosphide is a solid fumigant used for the preservation of food grains. It is often used as a suicidal poison because of its easy availability. Aluminum phosphide poisoning, if ingested in toxic doses carries a mortality risk of approximately 80–90%. The toxic effects are due to release of highly poisonous phosphine gas.2 Those wA 17-year-old male presented to us with a history of acute dysphagia and severe cough following every swallow; both to liquids as well as solids. Careful history taking revealed a history of aluminum phosphide tablet ingestion 5–6 days back for which he was admitted in some other hospital, treated conservatively, and discharged uneventfully. Clinical examination of chest revealed fine crepts in lung bases while clinical examination of oral cavity and abdomen did not reveal any other significant finding. Laboratory examination, chest radiography, and ultrasonography of abdomen were unremarkable.Patient then underwent barium esophagogram that revealed irregular narrowing in upper esophagus  associated with fistulous communication with left main bronchus . HoweverPatient then underwent endoscopy at tertiary level institute that confirmed the radiological findings of a fistulous communication of esophagus with left main bronchus. The patient also underwent reconstructive surgery in the same institute with unremarkable postoperative period. Three-month postoperative follow-up of the patient at our hospital was unremarkable.Aluminum phosphide is a solid fumigant, available in tablet form for use as a food-grain preservative. It releases a toxic gas, phosphine, on exposure to moisture. It is often used as an oral suicidal poison due to its easy availability. On ingestion, it produces serious systemic effects within an hour. The toxic effects and prognosis is highly dependent on dose, freshness of tablets, time-elapsed following consumption, and promptness of resuscitative procedures.Phosphine acts by inhibiting cytochrome oxidase, a mitochondrial enzyme which is a part of a respiratory chain cycle. Acute cardiovascular collapse is the commonest mode of presentation followed by gastrointestinal, neurological, respiratory, and musculoskeletal manifestations. DisseminThere is no specific antidote of aluminum phosphide poisoning and hence the treatment is directed to reverse the signs of clinical deterioration especially cardiogenic shock, breathing difficulty, and CNS manifestations. Besides the usual resuscitative measures, intravenous magnesium sulfate has been found useful in reducing the mortality of patients, probably due to its membrane stabilizing and anti-peroxide effect. Intra-aoBenign strictures and tracheoesophageal fistula has been rarely reported in the medical literature in the survivors of aluminum phosphide poisoning.–6 Esopha‘Ono’s sign’. Barium esophagogram is diagnostic in majority of the cases. Esophagoscopy is needed for confirmation in few cases.The sequence of swallow and cough is a classical clinical presentation in a case of fistulous communication between esophagus and airway tract, also known as Direct repair of the fistulous openings with interposition of muscle flap is the procedure of choice. Recurrences are uncommon in benign esophagorespiratory fistula. If the esophagus is damaged beyond repair, esophageal bypass with transverse colon or stomach through retrosternal approach is the procedure of choice.To summarize, though fistulous communication between esophagus and airway tract  is a rare complication seen in survivors of aluminum phosphide poisoning yet it should be kept in mind especially in those patients presenting with dysphagia. If unrecognized, it can lead to severe morbidity and even death due to aspiration and secondary pulmonary infections. Also, it is worth following survivors of aluminum phosphide poisoning patients with barium esophagogram for early detection of fistulous complication.


Pelvic schwannoma typically forms a large, well-circumscribed mass in the retroperitoneum or presacral area and frequently undergoes cystic degeneration. It appears as a well-demarcated round or oval mass, often showing prominent cystic degeneration and calcification. Characteristics of these calcifications are punctate, mottled, or curvilinear and are seen along the walls of the mass. Herein, we describe a case of schwannoma presenting a huge pelvic mass with unique eggshell-like calcification. Schwannomas (neurilemmomas) are benign neurogenic tumors originating in Schwann cells of the nerve sheath. They generally occur in the head and neck and extremities but rarely in the pelvis or retroperitoneum . RetropeA 68-year-old man presented with anal pain for one month and underwent examination in a clinic. A giant pelvic mass was detected on imaging examinations, and he was admitted to our hospital for closer examination. He had no history of previous malignancy. On admission, he had only anal pain and no other symptoms. On rectal examination, a hard fixed mass was felt on the posterior wall of the rectum. No abnormal neurological signs were found. His blood test and tumor markers including CEA, CA19-9, and soluble IL-2 receptor were within normal limits.Contrast-enhanced computed tomography (CT) was performed using nonionic contrast material  and showed a well-defined hypoattenuation tumor in the pelvis measuring 87 × 65 mm in the transaxial diameter. The tumor was located close to the rectum, anterior to the sacrum. There were multiple, spotted, and oval eggshell-like calcifications within the tumor. Cystic change was not evident on CT. The tumor parenchyma revealed hypoattenuation in the early phase (40 s after administration of contrast agent) and was slightly enhanced in the delayed (240 s) phase . MagnetiLaparoscopic extirpation was carried out. The tumor was 110 mm on its major axis and was encapsulated. A cross section showed multiple spotted or oval calcifications. Histologically, sections showed the proliferation of spindle-shaped cells having wavy nuclei arranged in fascicular or palisading pattern. Myxoid degeneration, calcifications, and hyalinizations were also observed. Mitotic figures were inconspicuous. Immunohistochemically, tumor cells were positive for the S-100 protein. That feature indicated schwannoma, and there was no evidence of malignancy .The patient's postoperative course was uneventful, and there were no signs of recurrence on physical or imaging examinations 6 months after the operation.Pelvic or retroperitoneal schwannomas commonly locate in the presacral region or lower retroperitoneum at the pelvic brim . PreoperAt CT, a schwannoma typically appears as a well-demarcated round or oval mass, often showing prominent cystic degeneration and calcification. Calcifications are punctate, mottled, or curvilinear and are seen along the walls of the mass . HoweverIn our case, the tumor was a hypoattenuation pelvic mass with mottled or multiple oval, eggshell-like calcifications and was slightly enhanced at delayed phase on contrast-enhanced CT. The tumor demonstrated hypointensity and mild hyperintensity on T1-weighted image and T2-weighted image on MRI. Spotty areas of hyperintensity were also observed on T1-weighted image, which indicated hemorrhage. Typical cystic appearance was not evident on either CT or MRI. We speculated that the calcifications were a result of cystic degeneration or hemorrhage during their slow growth over a long period.It is difficult to confirm the diagnosis of retroperitoneal schwannomas using only imaging examinations such as CT or MRI, in particular when the tumors do not reveal a typical solid and cystic appearance. As a result, pelvic schwannomas are often diagnosed by surgically resected specimens. CT- or US-guided FNAB constitutes a useful option for the diagnosis of pelvic schwannomas.In conclusion, we describe a case of pelvic schwannoma with prominent calcifications. When diagnosing tumors, even those without typical solid and cystic appearances, the possibility of schwannoma should be considered. EUS-FNAB is also recommended in such difficult cases.


Cavernous hemangiomas are the most frequent neoplasms of the liver and in routine clinical practice they often need to be differentiated from malignant tumors and other benign focal lesions. The purpose of this study is to evaluate whether diagnostic accuracy of magnetic resonance imaging (MRI) of hepatic hemangiomas, showing atypical pattern on US, improves with the use of Gd-BOPTA in comparison with contrast-enhanced multi-phase computed tomography (CT).178 consecutive patients with ambiguous hepatic masses showing atypical hyperechoic pattern on grey-scale US, underwent unenhanced and contrast-enhanced multi-phase multi-detector CT and MR (1.5T) with the use of liver-specific contrast medium gadobenate dimeglumine (Gd-BOPTA). After intravenous contrast administration arterial (HAP), venous-portal (PVP), equilibrium phases (EP) both in CT and MR and additionally hepatobiliary phase (HBP) in MR were obtained. 398 lesions have been detected including 99 hemangiomas and 299 other lesions.In non-enhanced MDCT examination detection of hemangiomas was characterized by sensitivity of 76%, specificity of 90%, PPV of 71%, NPV of 92% and accuracy of 86%.Non-enhanced MR examination showed sensitivity of 98%, specificity of 99%, PPV of 99%, NPV of 99% and accuracy of 99%.After intravenous administration of contrast medium in MR the mentioned above parameters did not increase significantly.Gd-BOPTA-enhanced MR in comparison with unenhanced MRI does not improve diagnostic accuracy in discriminating hemangiomas that show non-specific appearance in ultrasound examination. Unenhanced MR as a method of choice should directly follow US in course of diagnostic algorithm in differentiation of hemangiomas from other liver tumors. Cavernous hemangiomas are the most frequent benign neoplasms of the liver, found in autopsy examinations within 0.4% to 20% of population ,2. Such Great technological development concerning both multi-detector computer tomography and hepatocyte-specific contrast agents in magnetic resonance imaging (MRI) has been observed in recent years.Computed tomography is a well established procedure of imaging of hepatic hemangiomas. Most hemangiomas show typical enhancement pattern: nodular or globular peripheral enhancement in arterial phase with a progressive fill-in in portal venous and equilibrium phases.In particular cases the use of magnetic resonance imaging leads to specification of diagnosis based on US and CT findings.Many studies have been dedicated to differential diagnosis of hemangiomas from malignant tumors -8. PreviThe purpose of this study is to evaluate whether assessment by means of MRI of hepatic hemangiomas, showing atypical appearance on gray-scale US, improves with the use of Gd-BOBTA in comparison with contrast-enhanced multiphase CT.178 consecutive patients with non-specific hepatic foci observed in US were included in this prospective study and spiral CT and MR examinations were performed in those patients within the period not exceeding 3 weeks.Inclusion criteria were as follows: presence of focal liver lesion that in grey-scale US did not present typical features of a simple cyst (anechoic focus with posterior acoustic enhancement) or a typical hemangioma  in axial and coronal plane with and without contrast enhancement in equilibrium and hepatobiliary phase, T2-weighted Express sequence  in axial plane, T2-weighted FSE  in coronal plane, performed also with fat saturation and T1-weighted out of phase sequence  without and after contrast administration in hepatic arterial dominant phase (HAP), portal venous dominant phase (PVP), equilibrium phase (EP) and hepatobiliary phase (HBP). Slice thickness was 5mm, intersection gap - 0.5, matrix 256 × 256. Dynamic MRI was obtained immediately after a bolus injection of Gd-BOPTA (dosage of 0.1 mmol per kilogram of body weight), followed by saline solution flush of 25mL through a 20G venous catheter positioned in the antecubital vein. Similarly to the multiphase CT study HAP, PVP, and EP were performed after respectively 25, 60 and 180sec. HBP was acquired 60min after contrast agent administration. The phase-encoding direction was anterior-posterior for all sequences. All images were acquired during breath-hold.Multi-phase multi-detector (6 and 32-row) CT examination of the liver has been conducted first unenhanced, then after application of a contrast agent in HAP, PVP and EP. 100ml of iodinated contrast agent at a rate of 4mL/sec through an 20G venous catheter positioned in an antecubital vein using a power injector was administrated. The CT section thickness was 2.5mm, images interval - 2.5mm and pitch - 1.0.Spiral CT and MRI were then interpreted by three independent radiologists with at least 10-years  and 5-years (AS) experience in abdominal imaging, who had no previous knowledge of patients' medical history. The interval between CT and MR examinations was from 3 to 5 weeks. Final assessment was based on the observers' consensus in case of disagreement.Radiological parameters assessed in CT and MR analysis were: size of detectable lesions, their density, signal intensity and type of enhancement.Signal intensity of each lesion in relation to adjacent liver parenchyma in T2-weighted TSE images was assessed. A 4-point scale has been used: 1-hypointense, 2-isointense, 3-hyperintense and 4-bright lesion  consisted only of liver hemangiomas, the second group (nonHH group) included focal liver lesions other than hemangiomas.All lesions were also classified as small or large according to their medians by the study coordinator (ES).P values less than 0.05 were considered as statistical significant.The interobserver agreement was measured with Cohen's kappa coefficient. Statistical analysis of size of detectable lesions, their density, signal intensity and type of enhancement of hemangiomas and other lesions in unenhanced, gadolinium-enhanced MR and CT studies has been performed with Statistica 8 software  and McNemar's test with Bonferroni correction for multiply comparison. The research was approved by Independent Bioethic Committee for Scientific Research of Medical University of Gdansk (NKEBN/649/2001-2002). All patients gave their written consent to participate.The study has been partially financed from the research grant ST-82 given by authorities of the Medical University of Gdansk to MS.178 patients were qualified for the study, amongst this group 161 underwent further analysis .In case of 17 patients we failed to establish the final diagnosis - 2 of the patients passed away, 15 did not show for the follow-up.High interobserver agreement  confirmed the methodology as reliable and reproducible.In 100 patients the final diagnosis was based upon histopathological examination results (66 cases by the means of surgery and 34 cases with biopsy) which disclosed: 41 cases of HCC, 21 FNHs, 28 cases of liver metastases, 4 cavernous hemangiomas, 4 solitary adenomas and 2 cases of peripheral cholangiocarcinoma.In the remaining 61 patients, the final diagnosis was based upon the clinical and diagnostic imaging follow-up. In this group we observed: 30 hepatic hemangiomas (confirmed in contrast-enhanced US examination and follow-up MR examination), 18 FNHs (confirmed in Gd-EOB-DTPA-enhanced MR), 13 cases of liver metastases (treated with chemotherapy and confirmed in follow-up CT examinations) and 2 inflammatory lesions (follow-up CT examinations showed no lesions).The HH group consisted of 99 liver hemangiomas, recognized in 34 patients. 21 patients with HH had isolated liver lesions and multifocal HHs were recognized in 13 people.The nonHH group included 127 patients with 299 focal liver lesions other than hemangioma: 144 metastases, 100 cases of hepatocellular carcinoma, 4 adenomas, 44 FNHs and 7 other lesions.The diameter of foci ranged from 6mm to 125mm (median 21mm). According to the mean foci diameter, all lesions were divided into two groups: small lesions (<2cm) - 199 foci and large lesions (≥2cm) - 199 foci. In the HH-group, 40 foci of cavernous hemangiomas were large lesions and the rest of 59 tumors were small lesions.Small hemangiomas more frequently (30/59) showed homogenous enhancement pattern in all three phases, while large hemangiomas (25/40) presented nodular or globular peripheral enhancement pattern in HAP and progressive fill-in in PVP and EP in dynamic MR study table . The typGd-BOPTA - enhanced MR and multi-phase spiral CT evaluation of hepatic hemangiomas was based upon typical enhancement pattern in three phases table :- homogenous in HAP and PVP or EP figure ,- nodular peripheral or globular in HAP with progressive fill-in enhancement in PVP and EP figure .In the hepatobiliary phase of MR examination all hemangiomas showed a lower signal in comparison to the adjacent liver tissue figure . 233 nonSpiral CT correctly characterized 75 hemangiomas.Signal intensity observed in moderately and heavily T2-weighted images in hemangiomas and other liver lesions is presented in table In unenhanced MR false-negative diagnosis was obtained in one case of hemangioma and one false-positive diagnosis of hemangioma was noted in case of cystic metastasis from ovarian carcinoma.Gd-BOPTA-enhanced MR study allowed to correctly diagnose cystic metastasis from ovarian carcinoma and therefore revealed 98 hemangiomas. Still one hemangioma was unrecognized figure .p < 0.0001 for both modalities), also in case of small  and large lesions assessed separately (p = 0.0055 for unenhanced MR and p = 0.0009 for Gd-BOPTA-enhanced MR).Unenhanced MR and Gd-BOPTA-enhanced MR show higher diagnostic accuracy in differentiation of hemangiomas than CT  and high signal intensity in T2-weighted images -14.In dynamic examination the enhancement pattern of hemangiomas does not differ in neither CT nor MR study.Distinction between metastases and cavernous hemangiomas on the basis of different patterns of enhancement is well described, but to our knowledge, only few reports focus on the type of enhancement, diagnosed with hepatocyte-specific contrast-enhanced MRI .In presented study the assessment of focal lesions in MR was performed with the use of moderately and heavily T2-weighted sequences and dynamic examination after Gd-BOPTA administration.Ultrasound was the initial examination qualifying patients into this study as it allowed to distinguish a group of patients with ambiguous focal liver lesions, some of suspected malignant nature.Patients presenting lesions of typical ultrasound appearance of hemangiomas and simple cysts were excluded from further studies as such lesions may be successfully monitored in grey-scale ultrasound examinations.To assess foci in moderately T2-weighted images qualitative criteria proposed by Fenlon were used . In modeA high diagnostic efficacy of marked hyperintensity in moderately and heavily T2-weighted images as a feature allowing to diagnose hepatic hemangiomas was proven with indexes of diagnostic efficacy reaching 99% table .Similar observation was made by Ito et al. , who gaiFenlon et al. compared qualitative and quantitative analysis of hepatic tumors using heavily T2-weighted SE technique and they noted that quantitative method with measurement of T2-relaxation times allowed significantly better differentiation between benign and malignant neoplasms with accuracy about 100% than that of the subjective visual assessment of focal liver lesions .In our series a false-negative diagnosis concerned one case of hemangioma with areas of fibrosis and hyalinization, which mimicked a solid part of malignant necrotic neoplasm. One false-positive diagnosis of hemangioma was reported in case of cystic metastasis from ovarian cancer. We did not observe characteristic strong hyperintensity typical for hemangiomas in other cases of malignant tumors especially in necrotic or hypervascular metastases as noted in literature ,28.The next radiological feature that allows recognition of HH is the type of enhancement observed in successive phases of dynamic CT or MR examination after extracellular iodine or gadolinium based contrast medium administration. Ros et al.  distingu- early homogenous enhancement seen in foci of less than 1.5 cm in diameter,- nodular peripheral enhancement in HAP with a progressive centripetal fill-in pattern, to homogenous enhancement in the late phase (foci from 1.5 to 5cm in diameter),- nodular peripheral enhancement in HAP with a hypointense center of the lesion in the late phase (foci larger than 5 cm in diameter).Essentially in our study two types of enhancement patterns in HAP were stated: nodular peripheral and homogenous table . At the In hemangiomas greater than 2cm in diameter a nodular peripheral enhancement pattern in HAP with progressive slow centripetal fill-in during next phases has been observed. This type of enhancement has been described as specific for hemangiomas since 1986 -37.The value of PPV and accuracy and sensitivity in differentiation of hemangiomas from other focal liver lesions based of enhancement pattern presented in multi-phase sCT study are 0.71, 0.86 and 0.76 and noticeably lower than all indexes of diagnostic efficacy obtained by using unenhanced-MRI (PPV-0.99). In the group of lesions smaller than 2cm, sensitivity of diagnostic methods based on contrast CT enhancement pattern is even lower, at the range of 0.66.Presented study in accordance to the other authors ,38 showsIn this study all hemangiomas presented weaker enhancement in hepatobiliary phase in comparison to the adjacent liver parenchyma, similarly to metastases and the majority of HCCs. This results from the characteristic of applied hepatocyte-specific contrast media - Gd-BOPTA. Gadobenate dimeglumine (Gd-BOPTA) is a gadolinium-based paramagnetic contrast with a vascular-interstitial distribution during first few minutes. The majority of this contrast agent is excreted by the kidneys in the urine, however, about 4% of the injected dose is taken up by the hepatocytes and eliminated via anionic transporter across the sinusoidal membrane into the bile .Due to contrast agent presence within hepatic cells, it is possible to observe the enhancement of the liver parenchyma that persists for 1-3 hours after its administration and this period is called hepatobiliary phase.A tumor that does not contain functioning hepatocytes in which hepatobiliary metabolism is active, is not able to uptake Gd-BOPTA.Hemangiomas consist of multiple vascular channels limited by single layer of endothelial cells within a thin fibrous stroma and the enhancement in hepatobiliary phase is not observed, what makes hemangiomas impossible to differentiate from other hepatocyte-devoided liver tumors ,40,41.Main restrictions of this study are a small number of cases of hemangiomas confirmed in histological examination, lack of quantitative analysis of focal liver lesions signal intensity as described by other authors  and no iOur results indicate that qualitative analysis of signal intensity of hepatic lesions in moderately and heavily T2-weighted images is the most sensitive method of differentiation between liver hemangiomas of non-specific appearance in ultrasound from malignant tumors. Assessment of the enhancement pattern provides information about vascularity of the lesion although the type of enhancement pattern itself is not as important in characterization of hemangiomas as the intensity of the lesion in T2-weighted images.Application of contrast-enhanced MR, especially with administration of liver-specific contrast agents in diagnosing liver hemangiomas is not advisable as it leads to elongation of examination time and unnecessary exposure to potential side effects of intravenous contrast agents.Gd-BOPTA-enhanced MR in comparison with unenhanced MRI does not improve diagnostic accuracy in discriminating hemangiomas that show non-specific appearance in ultrasound examination. Unenhanced MR as a method of choice should directly follow ultrasound examination in course of diagnostic algorithm in differentiation of hemangiomas from other liver tumors.CT: computer tomography; EP: equilibrium phase; FNH: focal nodular hyperplasia; FSE: fast spin echo; HAP: hepatic arterial dominant phase; HBP: hepatobiliary phase; HH: hepatic hemangioma; MDCT: multidetector computer tomography; MR: magnetic resonance; MRI: magnetic resonance imaging; nonHH: other lesion than hemangioma; NPV: negative predictive value; PPV: positive predictive value; PVP: portal venous dominant phase; ROI: region of interest; sCT: spiral computed tomography; SE: spin echo; TE: echo time; TR: repetition time; US: ultrasound.The authors declare that they have no competing interests.ES has had substantial contribution to study design, data collection and interpretation, statistic analysis and manuscript preparation. TN has contributed to statistic analysis and manuscript preparation. EIS has contributed to data collection and interpretation, as well as manuscript preparation. JW and ADJ has contributed to study design and manuscript preparation. AS has contributed to data collection and interpretation. KM has contributed to manuscript preparation. MS has contributed to study design and has coordinated research team. All authors have read and approved the final manuscript.The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1471-230X/11/43/prepub


Xanthogranulomatous inflammation is a rare clinico-pathological condition involving many organ systems. Breast involvement with this rare condition reported from a few cases of mastitis has been limited to only microscopic involvement on histology. We would like to report an unusual presentation of this inflammatory process presenting as a solid lump mimicking malignancy in latissimus dorsi donor site scar and implant-based breast reconstruction as a result of a ruptured silicone gel implant. To our knowledge there have been no previous reports on similar presentation published in the literature. This case highlights a rare complication of a leaked silicone gel implant triggering a xanthomatous response in the absence of the usual infective or obstructing etiologies. This condition is of benign nature with complete clearance on surgical excision and excellent clinical prognosis reported from other organ involvement. Xanthogranulomatous inflammation involves various organ systems such as the gall bladder, kidney, pancreas, appendix, eyes, female genital tracts, colon, and urachus -8; breasA 56-year-old lady presented with an abnormal mass arising from her latissimus dorsi (LD) donor site scar Figure  and hardOn examination she had a 10 cm mass arising from the donor site scar. The mass was well defined with an area of softness on the anterior surface. The left reconstructed breast had a marked capsular contracture Figure  and a suInitial investigation of the mass with ultrasound (US) showed solid tissue within the mass with a central fluid collection that on fine needle aspiration showed necrotic debris. US findings were confirmed by the contrast-enhanced computer tomography (CT) scan with no evidence of any distant metastasis. A magnetic resonance imaging (MRI) scan Figure  of the bExcision biopsy of the back mass lesion and revision of the reconstructed breast for definitive histological diagnosis was performed in two stages. In stage one, the mass was excised with a transverse ellipse and the wound closed with advancement flaps. The microscopic appearances of the excised lesion were similar to core biopsy findings with xanthoma-laden large macrophages interlaced with inflammatory cells and no malignancy Figure . FindingXanthogranulomatous inflammation is a benign pathological condition considered to be a primary disease process of unknown etiology. Various organ system can be involved to varying degrees; most commonly affected are the kidneys and gall bladder with a few rare cases of colonic involvement. Evidence of breast involvement comes from cases of inflammatory mastitis  showing The exact etiology of the disease is unknown; organ obstruction, suppurative infections and hemorrhage trigger tissue damage within the involved organs, usually eliciting a microscopic response of the disease process. The case study by Luc and colleagues  followinXanthogranulomatous inflammation disease presentation is variable, with both localized and diffuse responses. A diffuse inflammation is the more severe form of the disease, resulting in a complete obliteration of the native parenchymal tissue and marked fibrosis which clinically presents as solid lumps. Xanthogranulomatous inflammationcan also have a cutaneous involvement which can be primary or secondary . SecondaRadiological diagnosis of xanthogranulomatous inflammation using US and CT is challenging. Diagnostic accuracy can be increased using contrast-based imaging, as demonstrated using sonazoid contrast enhancing agent with US scan in the diagnosis of chronic inflamed and thickened gall bladders, and CT urography studies for renal xanthogranulomatous inflammatory disease ,14. The The microscopic appearances of xanthogranulomatous inflammation on histology show the characteristic multinucleate giant cells interspersed with lipid-laden macrophages which impart the characteristic yellow macroscopic appearance. This appearance, however, should be differentiated from a pseudo xanthomatous inflammation or malakoplakia characterized by Michaelis-Gutman bodies which stain positive with Von Kossa calcium and Prussian blue stains .In our case, an implant leak triggered the inflammatory process. Presentation of a well localized mass in the LD back scar was a result of an extensive tissue inflammation and fibrosis from posterior tracking of the leaked silicone. However, what surprisingly was absent in our case was the postulated triggers of obstruction or infection at surgery; gram staining of both implant and back tissue was negative too. The pre-operative imaging was mostly helpful in delineating the anatomy of the mass and excluding any systemic spread from previously treated breast primary; failure to accurately diagnose the condition confirms the limitations of both CT and MRI in these cases.The recommended treatment of solid lumps with diffuse involvement is usually surgical as this condition is not pre-malignant . A complThe silicone gel-filled breast implants are safe with a prolonged life span; however, implant-related problems increases with time  and, therefore, surgeons should be wary of these unusual presentations when dealing with the complications of long-standing implants.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.CT, computer tomography; LD, latissimus dorsi; MRI, magnetic resonance imaging; US, ultrasound..The authors report that they have no conflict of interests.TH did the literature search and has been a major contributor in writing the manuscript. PJK did the final draft proof reading and approval. EL provided the pathology images. All authors read and approved the final manuscript.


Tubercular spinal epidural abscess (SEA) is is a devastating infectious disease. Its presence without associated osseous involvement may be considered an extremely rare scenario. We present a rare case of tubercular SEA complicated by paraplegia in an immune-competent 58-year-old male patient. MRI shows a multisegmental posterior collection of epidural fluid extending from C7 to L2 vertebral level and displaces the ventrally located thecal sac, without any evidence of vertebral involvement. The patient made an uneventful recovery following surgical decompression and antitubercular chemotherapy. The diagnosis was confirmed by histopathological demonstration of Mycobacterium tuberculosis in drained pus. Such presentation of tubercular SEA has not been reported previously in the English language based medical literature to the best of our knowledge.


Introduction. Enchondroma protuberans is an extremely rare benign cartilaginous bone tumor. We report the first case report of enchondroma protuberans in the forearm. Presentation of Case. We report a case of enchondroma protuberans originating in the left ulnar bone of a young woman. A 20-year-old female referred to our hospital complaining of progressive sustained left forearm pain with a radiation to fourth and fifth finger. Conventional radiography revealed a well-defined eccentric osteolytic lesion in the distal diaphysis of ulna with expansion of overlying cortex . Magnetic resonance imaging showed a well-defined ovoid intramedullary lesion, which was exophytically protruding from medial surface of left ulnar bone. Histopathology confirmed the diagnosis. Discussion. Enchondroma protuberans typically present as a well-defined intramedullary osteolytic lesion that may be accompanied by a fine matricidal calcification. The connection between the intramedullary portion and the exophytic protrusion can be seen well by magnetic resonance imaging. Conclusion. Enchondroma protuberans should be considered in the differential diagnosis of osteochondroma, enchondroma, and periosteal chondroid tumors. Enchondroma protuberans is a rare benign cartilaginous bone tumor. It arises from the intramedullary cavity of long bones, which usually protrudes beyond the cortex. Based on our extensive medical database search, there are only handfuls of cases reported previously –10. MostWe present a 20-year-old female who referred to our hospital complaining of progressive sustained left forearm pain with radiation to fourth and fifth finger of one-year duration. There was also a bulging mass, progressively growing on the lower dorsal surface of her left forearm. On physical examination, there was a 1.5 by 1.5 cm firm tender mass on the lower dorsal surface of her left forearm. The skin overlying the lesion was normal and the elbow joint had a normal range of motion. She had no weakness in her hands. Strength was measured 5/5 at wrist and forearm bilaterally. Sensation was normal to light touch, temprature, and crude touch in all five finger. There was no paresthesia, numbness, or tingling. She was able to determine static two-point discrimination of 1 cm in the radial, median, and ulnar nerve distributions of her both hands. All other local physical examinations were normal.All laboratory studies including blood cell and erythrocyte sedimentation rate were within normal limit.Antero-posterior and lateral Figures  X-rays rMagnetic resonance imaging showed a well-defined ovoid intramedullary lesion measured 2 by 1 cm in the distal diaphysis of left ulnar bone that was exophytically protruding from the medial surface of left ulnar bone. The lesion was of low signal intensity on T1-weighted images  and highComplete resection of the epiphytic cartilage cap with intramedullary curettage was performed. Histopathology showed a cartilaginous tissue with uniformly sized chondrocytes in a myxoid matrix located in the round lacunae, compatible with enchondroma and with no evidence of cytological dysplastic cell . The posEnchondroma protuberans originates from the cartilage in the medullary cavity. It is defined as a protruded enchondroma that is beyond the cortex. There were 13 reported cases of the enchondroma protuberans in the literature and all were located in the humerus, ribs, and hand regions .Enchondroma almost always appears as a well-defined osteolytic lesion that is usually located within the metadiaphysis of long bone. Due to bone expansion the cortex may appear thin, but usually remains intact. Enchondroma rarely expands through the cortex, and if accompanied by a cortical defect, will result in enchondroma protuberans .Radiologically, enchondroma protuberans is presented as a well-defined intramedullary osteolytic lesion and may be accompanied by fine matricidal calcification, cortical expansion and cortical defect, and round well-defined soft-tissue expansion , 6, 10.The typical presentation of enchondroma protuberans in MR images is as a well-defined intramedullary lesion with low signal intensity in T1-W image and high signal intensity in T2-W and STIR sequences accompanied by cortical expansion and cortical defect , 10.Although this benign tumor is usually diagnosed by conventional radiography, but according to X-ray findings in our and two previous case reports and also by An et al., sometimes it is not possible to be diagnosed based on conventional X-ray alone . The conEnchondroma protuberans should be considered in the differential diagnosis of osteochondroma, Enchondroma, and periosteal chondroid tumors. Periosteal chondroma is benign cartilaginous tumor of periosteal origin, which occurs in young adults. Osteochondroma is continuous with the bone, which it originates and also contains a dense osteoid formation in the cortex and medulla.The absence of cartilaginous cap and underlying trabecular bone differentiates it from osteochondroma and delineation of contiguous intramedullary involvement can be a strong indicator to differentiate it from periosteal chondroma. Because it has a potential risk of transformation to chondrosarcomatous, it is important to accurately diagnose and plane for a surgical treatment. To surgically treat the osteochondroma, excision of the cap is sufficient. But in patients with enchondroma protuberans, resection of the exophytic cartilage mass should be accompanied by intramedullary curettage .Enchondroma protuberans should be considered in the differential diagnosis of osteochondroma, enchondroma, and periosteal chondroid tumors.


Laryngeal carcinoma and laryngeal papilloma are the most commonly encountered tumorous lesions in the larynx. Herein, we report a case of the mass arising from the left vocal cord in a 49-year-old Japanese man. Endoscopic examination suggested that the mass is a tumor such as carcinoma and papilloma. Pathological examination showed that the specimen demonstrated actinomycosis in the left vocal cord. Although vocal cord actinomycosis is extremely rare, the otolaryngologist should recognize this condition during the inspection of the larynx. Actinomyces israelii. Actinomyces israelii is an anaerobic, Gram-positive organism that is normally present in the oral cavity. Clinical manifestation mainly occurs in one of three forms: cervicofacial, abdominal-pelvic, or pulmonary actinomycosis. However, the actinomycosis of the larynx is very rare. Here, we report a case of primary vocal cord actinomycosis mimicking a laryngeal tumor.Laryngeal carcinoma and laryngeal papilloma are the most commonly encountered tumorous lesions in the larynx. Actinomycosis is a disease that is mainly caused by Approximately one year ago, a 49-year-old Japanese man came for treatment with the complaint of his hoarseness of voice for 2 years. He had no fever, odynophagia, weight loss, or dysphagia. There was no history of any operation in head and neck, and he had diabetes mellitus and dental caries. Laryngoscopy showed a mass of surface irregularity in the anterior one-third part of the left vocal cord . The resActinomyces israelii. Actinomyces israelii is a commensal saprophyte of the normal oral flora [Actinomycosis in human involves most commonly (60~70%) the cervicofacial region and is most commonly due to the al flora –4. TraumLaryngeal actinomycosis is quite rare. A review of the literature reveals that actinomycosis of the larynx is often associated with an underlying history of squamous cell carcinoma of the larynx and therapeutic radiation therapy . An undeThe pathogenesis of laryngeal actinomycosis is not clear. Why the microorganism, which is normally present in the mouth, should became pathogenic is poorly understood. Poor local hygiene, local tissue damage, diabetes mellitus, immune suppression, and malnutrition are suggested predisposing factors . TherefoIn summary, we describe an additional case of vocal cord actinomycosis in this paper.


We present a rare case of rhabdomyosarcoma of lip in a neonate with multiple lesions within the head and necksub site hitherto unreported in the medical literature. This case report also reviews the scant medical literature on neonatal rhabdomyosarcoma. Rhabdomyosarcoma (RMS) is a malignant soft tissue neoplasm of the skeletal muscle origin. The tumour accounts for 4–8% of all malignancies in children of less than 15 years of age . RMS alsAn 11-day-old neonate presented to the “Paediatric Otorhinolaryngology OPD” of our Kalawati Saran Paediatric Hospital, attached to Lady Hardinge Medical College, New Delhi, India, a tertiary care university teaching hospital with a swelling in the upper lip since birth and complaint of difficulty in suckling. In general the baby was irritable and always crying.A large ovoid swelling was seen in the upper lip and adjoining part of upper face. The lesion involved the anterior nares causing stenosis. On the buccal surface, the skin over the swelling was normal with no sinus, punctum, or ulceration. The alveolus was free of the swelling. The swelling measured 5 × 3 cm in maximum dimensions. The swelling was tender, firm in consistency and the skin over it was indurated in patches with engorgement of veins . A diffePatient underwent CT scan and MRI scan which revealed soft tissue intensity lesions in the lip, orbit and submandibular region  Figures  and 3. FIn view of multiple lesions, the said patient underwent chemotherapy with Vincristine, Actinomycin, Cyclophosphamide, and Dexamethasone. Subsequently, patient developed metastatic lesions in bone and lungs. Unfortunately, despite aggressive therapy, patient succumbed to his disseminated disease 3 months later.RMS is uncommon in neonates; thus, not much medical literature is available on the cited subject. Etiology of RMS is obscure. However, it is associated with genetic conditions like: neurofibromatosis and Li-Fraumeni syndrome , 4. The Histopathologically, the tumour is classified as embryonic, alveolar, and pleomorphic types . The embThese tumours have no specific clinical presentation . OrofaciMultidisciplinary approach in the management of RMS with surgery, radiotherapy, and chemotherapy has dramatically improved the prognosis for this potentially fatal tumour, with the current medical literature reporting a five-year survival rate of 74–77% , 2, 6, 8age less than 1 year;delayed diagnosis;less aggressive treatment attributed to the side effects of the chemo-radiation therapy;metastatsis; genetically: PAX3-FHR translocations.This case merits discussion on many accounts. This case is reported because of extreme rarity of RMS to occur in neonates and in the oral cavity too (upper lip). Moreover, the lesion within a short span of 10 days increased rapidly in size causing symptoms in the neonate. Further, the radiological examination revealed multiple subsite involvement within the head and neck region. Though it has been noted in congenital RMS that the disease may be metastatic at the time of birth , in a maThis unusual presentation of rhabdomyosarcoma of the lip in a neonate prompted us to share our clinical experience with other members of medical fraternity.


Solitary fibrous tumors of the central nervous system usually present as dura-based masses and clinically resemble meningiomas. There are very few reported cases of intra-ventricular solitary fibrous tumors, particularly in the fourth ventricle.Our patient was a 52-year-old African-American man, who presented to our facility with a two-month history of progressive weakness and numbness in all extremities. A computed tomography scan and brain magnetic resonance imaging scan revealed a homogeneous, avidly enhancing 4.5 × 3.7 × 2.7cm fourth ventricular mass, with compression of adjacent medulla and cerebellum and extension into the foramen of Luschka. Our patient underwent a suboccipital craniotomy and resection of the tumor. A histological examination showed a spindle cell neoplasm with prominent collagenized stroma. The neoplastic cells were strongly and diffusely positive for CD34, vimentin and Bcl-2, and negative for S-100, CD99 and epithelial membrane antigen. The molecular immunology Borstel-1 (MIB-1) proliferation index was low (1%). CD31 immunostain highlighted the endothelial cells but the spindle cells were negative. Reticulin stain demonstrated a moderate reticulin network but individual cells were not invested by reticulin fibers. The histological features and immunoprofile was consistent with a solitary fibrous tumor.In the central nervous system, solitary fibrous tumors are usually indolent tumors, with only rare examples showing hypercellularity and increased mitotic activity; features that were absent in our patient’s case. We present an uncommon central nervous system neoplasm in a rare location. Although uncommon, solitary fibrous tumors should be included in the differential diagnosis of intra-ventricular tumors in adults. Solitary fibrous tumors (SFTs) were originally described in pleura and subsequently in soft tissue and many other organs including the central nervous system. SFTs of the central nervous system are rare entities, usually presenting as dura-based masses predominantly in the posterior fossa and spinal region . SFTs arOur patient was a 52-year-old African-American man who presented to our facility with a two-month history of progressive weakness and numbness in all extremities. A computed tomography (CT) scan and a brain magnetic resonance imaging (MRI) scan revealed a 4.5 × 3.7 × 2.7cm, homogeneous, avidly enhancing mass located in the fourth ventricle, compressing adjacent medulla and cerebellum and extending into the foramen of Luschka Figure . AngiogrOur patient underwent a suboccipital craniotomy and resection of the tumor. During the operation, a cavitron ultrasonic surgical aspirator as well as bipolar cautery and suction were used to debulk the tumor, followed by a combination of RHOTON™ dissectors as well as microscissors to separate the tumor. The entire tumor was successfully removed.Histological examination showed a spindle cell neoplasm with prominent collagenized stroma Figure A. MitotiOur patient experienced no neurological deficit after surgery, and there were no signs of recurrence at one-year follow-up.Intraventricular tumors can have a wide spectrum of differential diagnosis based on radiological and histopathological features. A bland spindle cell tumor with collagenized stroma and avid contrast enhancement initially suggested a diagnosis of fibroblastic meningioma. However the immunoprofile of the neoplastic cells was typical of a solitary fibrous tumor . In contrast, meningiomas are EMA positive and CD34 negative. Hemangiopericytomas can have histological overlap with solitary fibrous tumor, but CD34 and Bcl-2 staining is weak and patchy .To the best of our knowledge, just 17 Intraventricular solitary fibrous tumors have been reported in the literature to date. Six of these tumors were located in the fourth ventricle -7, eightWe report a rare case of SFT in an unusual location. Though rare, SFTs should be considered in the histological differential diagnosis of intra-ventricular tumors particularly, spindle cell tumors with collagenized stroma, along with meningioma and schwannoma.Written informed consent was obtained from the patient for publication of this manuscript and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.CW performed the gross examination of the specimen, conceived the case report, searched the literature and drafted the manuscript. VM performed the histopathological evaluation of the slides and made substantial revisions to the manuscript. SF conducted the radiology examinations and made revisions to the manuscript. MW operated on our patient and made revisions to the manuscript. AM performed the histopathological evaluation of the slides and critical analyzed the manuscript. All authors read and approved the final manuscript.